Case Reports

THE CASE

A 35-year-old African-American man, who was an active duty service member, presented to the Troop Medical Clinic with a 4-hour history of priapism. He had been taking sertraline 100 mg and prazosin 10 mg nightly for 4 months to treat his posttraumatic stress disorder (PTSD) with no reported adverse effects. These doses were titrated 2 months prior to presentation. The patient reported that he took his usual medication doses before bed and awoke at 3 am with a penile erection. At 7 am, he presented to the clinic because of pain from the continued erection.

THE DIAGNOSIS

A penile erection was present on physical exam. All medications were reviewed for adverse effects. A work-up for anemia, sickle cell disease, thalassemia, and platelet abnormalities was negative. A blood gas analysis performed on blood aspirated from the corpus cavernosum showed hypoxemia, hypercarbia, and acidosis, confirming a diagnosis of ischemic priapism.

DISCUSSION

Priapism is a prolonged erection of the penis that is usually not associated with sexual activity or stimulation. It is considered a urologic emergency and requires prompt treatment to prevent long-term complications, such as permanent erectile dysfunction.

Priapism is classified as one of 2 types: ischemic (“low flow”) or nonischemic (“high flow”).

Ischemic priapism is the most common type. It is caused by dysfunctional cavernosal smooth muscle, which creates a compartment-like syndrome in the cavernous tissue that leads to hypoxia and acidosis.¹ Nonischemic priapism is often caused by a fistula between the cavernosal artery and corpus cavernosum and is common with traumatic injuries. Nonischemic priapism has a lower risk for long-term complications (due to the blood being well-oxygenated) and often resolves spontaneously without treatment.^2,3

Certain medications can cause priapism

Our patient’s ischemic priapism was most likely caused by the combined antagonistic properties of prazosin and sertraline on alpha-1 adrenergic receptors.^3,4 Adrenergic alpha-blockers block the sympathetic system, which can in turn inhibit penile detumescence and cause priapism.⁴

An increasingly common Tx combination. Selective serotonin reuptake inhibitors (SSRIs) such as sertraline are considered first-line treatment for the symptoms of PTSD, and prazosin has been found to be effective in the treatment of nightmares associated with PTSD. (Treatment of PTSD-related nightmares with prazosin is an off-label but frequent use of the medication.) This combination of medications is becoming increasingly common for the treatment of PTSD and its associated symptoms.^5-7

Continue to: Cases to date provide interesting insight into this adverse effect

 

 

Cases to date provide interesting insight into this adverse effect

In our literature review, no documented cases of priapism were attributed to prazosin when it was used for the treatment of nightmares, but there are multiple case reports of priapism linked to the drug’s use for hypertension.

In the majority of these case reports, the dosage exceeded 10 mg/d and was much higher than the dosage typically used to treat nightmares.⁷ Many of the affected patients also had associated comorbidities such as diabetes or chronic kidney disease.⁴

Sertraline has been associated with priapism when used as monotherapy and in combination therapy with antipsychotics. All SSRIs have antagonistic properties to alpha-1 adrenergic receptors, but sertraline appears to have more than a 10-fold increase in affinity when compared to other SSRIs.³

Treatment: An injection and aspiration

Our patient was treated with phenylephrine injection and aspiration, which resolved the priapism. Prazosin was stopped, and the patient was weaned off of sertraline. He continued to follow up closely with Behavioral Health for further management of his PTSD and associated symptoms.

Continue to: THE TAKEAWAY

 

 

THE TAKEAWAY

PTSD is being diagnosed more frequently, especially in active duty soldiers, veterans, members of the National Guard, and reservists.⁸ Because nightmares are a common symptom of PTSD and SSRIs are first-line treatment for PTSD, the combination of prazosin and an SSRI for the treatment of PTSD is frequently encountered.^5-7 Providers who prescribe and/or care for patients treated with these medications need to counsel patients on the risk of priapism and the risks associated with a delay in seeking medical care.

All SSRIs have antagonistic properties to alpha-1adrenergic receptors, but sertraline appears to have more than a 10-fold increase in affinity when compared to other SSRIs.

If a patient who is taking these medications presents with priapism, contact Urology immediately for acute management. Both medications must be stopped to prevent future episodes; prazosin can be stopped immediately, but patients must be weaned off of sertraline to avoid experiencing withdrawal symptoms. Patients will need to follow up with a behavioral health team for continued management of their PTSD symptoms.

CORRESPONDENCE
Caleb Dickison, DO, Fort Belvoir Community Hospital, 9300 Dewitt Loop, Fort Belvoir, VA 22060; [email protected].

THE CASE

A 35-year-old African-American man, who was an active duty service member, presented to the Troop Medical Clinic with a 4-hour history of priapism. He had been taking sertraline 100 mg and prazosin 10 mg nightly for 4 months to treat his posttraumatic stress disorder (PTSD) with no reported adverse effects. These doses were titrated 2 months prior to presentation. The patient reported that he took his usual medication doses before bed and awoke at 3 am with a penile erection. At 7 am, he presented to the clinic because of pain from the continued erection.

THE DIAGNOSIS

A penile erection was present on physical exam. All medications were reviewed for adverse effects. A work-up for anemia, sickle cell disease, thalassemia, and platelet abnormalities was negative. A blood gas analysis performed on blood aspirated from the corpus cavernosum showed hypoxemia, hypercarbia, and acidosis, confirming a diagnosis of ischemic priapism.

DISCUSSION

Priapism is a prolonged erection of the penis that is usually not associated with sexual activity or stimulation. It is considered a urologic emergency and requires prompt treatment to prevent long-term complications, such as permanent erectile dysfunction.

Priapism is classified as one of 2 types: ischemic (“low flow”) or nonischemic (“high flow”).

Ischemic priapism is the most common type. It is caused by dysfunctional cavernosal smooth muscle, which creates a compartment-like syndrome in the cavernous tissue that leads to hypoxia and acidosis.¹ Nonischemic priapism is often caused by a fistula between the cavernosal artery and corpus cavernosum and is common with traumatic injuries. Nonischemic priapism has a lower risk for long-term complications (due to the blood being well-oxygenated) and often resolves spontaneously without treatment.^2,3

Certain medications can cause priapism

Our patient’s ischemic priapism was most likely caused by the combined antagonistic properties of prazosin and sertraline on alpha-1 adrenergic receptors.^3,4 Adrenergic alpha-blockers block the sympathetic system, which can in turn inhibit penile detumescence and cause priapism.⁴

An increasingly common Tx combination. Selective serotonin reuptake inhibitors (SSRIs) such as sertraline are considered first-line treatment for the symptoms of PTSD, and prazosin has been found to be effective in the treatment of nightmares associated with PTSD. (Treatment of PTSD-related nightmares with prazosin is an off-label but frequent use of the medication.) This combination of medications is becoming increasingly common for the treatment of PTSD and its associated symptoms.^5-7

Continue to: Cases to date provide interesting insight into this adverse effect

 

 

Cases to date provide interesting insight into this adverse effect

In our literature review, no documented cases of priapism were attributed to prazosin when it was used for the treatment of nightmares, but there are multiple case reports of priapism linked to the drug’s use for hypertension.

In the majority of these case reports, the dosage exceeded 10 mg/d and was much higher than the dosage typically used to treat nightmares.⁷ Many of the affected patients also had associated comorbidities such as diabetes or chronic kidney disease.⁴

Sertraline has been associated with priapism when used as monotherapy and in combination therapy with antipsychotics. All SSRIs have antagonistic properties to alpha-1 adrenergic receptors, but sertraline appears to have more than a 10-fold increase in affinity when compared to other SSRIs.³

Treatment: An injection and aspiration

Our patient was treated with phenylephrine injection and aspiration, which resolved the priapism. Prazosin was stopped, and the patient was weaned off of sertraline. He continued to follow up closely with Behavioral Health for further management of his PTSD and associated symptoms.

Continue to: THE TAKEAWAY

 

 

THE TAKEAWAY

PTSD is being diagnosed more frequently, especially in active duty soldiers, veterans, members of the National Guard, and reservists.⁸ Because nightmares are a common symptom of PTSD and SSRIs are first-line treatment for PTSD, the combination of prazosin and an SSRI for the treatment of PTSD is frequently encountered.^5-7 Providers who prescribe and/or care for patients treated with these medications need to counsel patients on the risk of priapism and the risks associated with a delay in seeking medical care.

All SSRIs have antagonistic properties to alpha-1adrenergic receptors, but sertraline appears to have more than a 10-fold increase in affinity when compared to other SSRIs.

If a patient who is taking these medications presents with priapism, contact Urology immediately for acute management. Both medications must be stopped to prevent future episodes; prazosin can be stopped immediately, but patients must be weaned off of sertraline to avoid experiencing withdrawal symptoms. Patients will need to follow up with a behavioral health team for continued management of their PTSD symptoms.

CORRESPONDENCE
Caleb Dickison, DO, Fort Belvoir Community Hospital, 9300 Dewitt Loop, Fort Belvoir, VA 22060; [email protected].

THE CASE

A 35-year-old African-American man, who was an active duty service member, presented to the Troop Medical Clinic with a 4-hour history of priapism. He had been taking sertraline 100 mg and prazosin 10 mg nightly for 4 months to treat his posttraumatic stress disorder (PTSD) with no reported adverse effects. These doses were titrated 2 months prior to presentation. The patient reported that he took his usual medication doses before bed and awoke at 3 am with a penile erection. At 7 am, he presented to the clinic because of pain from the continued erection.

THE DIAGNOSIS

A penile erection was present on physical exam. All medications were reviewed for adverse effects. A work-up for anemia, sickle cell disease, thalassemia, and platelet abnormalities was negative. A blood gas analysis performed on blood aspirated from the corpus cavernosum showed hypoxemia, hypercarbia, and acidosis, confirming a diagnosis of ischemic priapism.

DISCUSSION

Priapism is a prolonged erection of the penis that is usually not associated with sexual activity or stimulation. It is considered a urologic emergency and requires prompt treatment to prevent long-term complications, such as permanent erectile dysfunction.

Priapism is classified as one of 2 types: ischemic (“low flow”) or nonischemic (“high flow”).

Ischemic priapism is the most common type. It is caused by dysfunctional cavernosal smooth muscle, which creates a compartment-like syndrome in the cavernous tissue that leads to hypoxia and acidosis.¹ Nonischemic priapism is often caused by a fistula between the cavernosal artery and corpus cavernosum and is common with traumatic injuries. Nonischemic priapism has a lower risk for long-term complications (due to the blood being well-oxygenated) and often resolves spontaneously without treatment.^2,3

Certain medications can cause priapism

Our patient’s ischemic priapism was most likely caused by the combined antagonistic properties of prazosin and sertraline on alpha-1 adrenergic receptors.^3,4 Adrenergic alpha-blockers block the sympathetic system, which can in turn inhibit penile detumescence and cause priapism.⁴

An increasingly common Tx combination. Selective serotonin reuptake inhibitors (SSRIs) such as sertraline are considered first-line treatment for the symptoms of PTSD, and prazosin has been found to be effective in the treatment of nightmares associated with PTSD. (Treatment of PTSD-related nightmares with prazosin is an off-label but frequent use of the medication.) This combination of medications is becoming increasingly common for the treatment of PTSD and its associated symptoms.^5-7

Continue to: Cases to date provide interesting insight into this adverse effect

 

 

Cases to date provide interesting insight into this adverse effect

In our literature review, no documented cases of priapism were attributed to prazosin when it was used for the treatment of nightmares, but there are multiple case reports of priapism linked to the drug’s use for hypertension.

In the majority of these case reports, the dosage exceeded 10 mg/d and was much higher than the dosage typically used to treat nightmares.⁷ Many of the affected patients also had associated comorbidities such as diabetes or chronic kidney disease.⁴

Sertraline has been associated with priapism when used as monotherapy and in combination therapy with antipsychotics. All SSRIs have antagonistic properties to alpha-1 adrenergic receptors, but sertraline appears to have more than a 10-fold increase in affinity when compared to other SSRIs.³

Treatment: An injection and aspiration

Our patient was treated with phenylephrine injection and aspiration, which resolved the priapism. Prazosin was stopped, and the patient was weaned off of sertraline. He continued to follow up closely with Behavioral Health for further management of his PTSD and associated symptoms.

Continue to: THE TAKEAWAY

 

 

THE TAKEAWAY

PTSD is being diagnosed more frequently, especially in active duty soldiers, veterans, members of the National Guard, and reservists.⁸ Because nightmares are a common symptom of PTSD and SSRIs are first-line treatment for PTSD, the combination of prazosin and an SSRI for the treatment of PTSD is frequently encountered.^5-7 Providers who prescribe and/or care for patients treated with these medications need to counsel patients on the risk of priapism and the risks associated with a delay in seeking medical care.

All SSRIs have antagonistic properties to alpha-1adrenergic receptors, but sertraline appears to have more than a 10-fold increase in affinity when compared to other SSRIs.

If a patient who is taking these medications presents with priapism, contact Urology immediately for acute management. Both medications must be stopped to prevent future episodes; prazosin can be stopped immediately, but patients must be weaned off of sertraline to avoid experiencing withdrawal symptoms. Patients will need to follow up with a behavioral health team for continued management of their PTSD symptoms.

CORRESPONDENCE
Caleb Dickison, DO, Fort Belvoir Community Hospital, 9300 Dewitt Loop, Fort Belvoir, VA 22060; [email protected].

ABSTRACT

Instability of the proximal tibiofibular joint (PTFJ) is a rare clinical condition that presents unique challenges to treatment. We present the case of an active 26-year-old woman with a 4-year history of recurrent PTFJ subluxations, treated surgically at our institution using a split biceps femoris tendon graft for PTFJ reconstruction. She underwent several attempts at nonoperative management until we decided to proceed with surgical intervention. A split biceps femoris graft was used to restore stability of the PTFJ. Approximately 5 years postoperatively, she achieved full range of motion as well as functional and clinical Knee Society Scores of 94 and 90 points, respectively. To the best of our knowledge, this is the first case report of PTFJ instability treated surgically with long-term follow-up. Future studies should focus on the long-term satisfactory outcomes of soft tissue stabilization of a chronically unstable PTFJ.

The instability of the proximal tibiofibular joint (PTFJ) is a rare clinical condition that commonly occurs secondary to an initial pivoting or twisting event of a flexed knee. Although acute PTFJ dislocations respond well to closed reduction and casting, the treatment of chronic PTFJ instability presents a unique challenge.¹ Surgical fixation methods include tibiofibular joint recreation using either a split semitendinosus or biceps femoris graft, as well as a Tightrope device.^2-6 Older surgical options for chronic PTFJ instability include fibular head resection or PTFJ arthrodesis.⁷ However, these older techniques have fallen out of favor, and the optimal surgical technique for the treatment of this injury remains a point of contention.

We present the case of an active 26-year-old woman with a 4-year history of recurrent PTFJ subluxations. The patient was surgically treated at our institution by using a split biceps femoris tendon graft for PTFJ reconstruction. This article specifically details the surgical technique used, provides data obtained at the 5-year clinical follow-up, and reviews prior publications on this injury. The patient provided written informed consent for print and electronic publication of this case report.

CASE

A 26-year-old woman presented with a 4-year history of lateral right knee pain with any physical activity. She stated that her pain began immediately following a fall, which was initially treated with casting and immobilization for approximately 6 weeks. After treatment, she began to develop symptoms of “popping on the outside of the knee.” In the 8 months prior to her presentation to our practice, these symptoms had intensified in pain severity and frequency. She reported that the popping events occurred most often with deep squatting.

No gross deformity was observed upon physical examination, and both knees were visibly symmetric. Evidence of effusion was absent. The patient felt no pain with the passive motion of her knee, and she presented the full range of motion (ROM) from 0° to 120°. Anterior drawer, McMurray, Lachman, and pivot shift tests were all negative. Upon the application of manual pressure, the fibular head could be dislocated anteriorly (Video 1). This dislocation recreated the patient’s symptoms. The fibular head could not be subluxed or dislocated posteriorly. Flexing the knee to 90° facilitated reproducing manual anterior dislocation. The contralateral knee was examined and demonstrated no appreciable PTFJ instability. The patient exhibited no other signs of generalized ligamentous laxity. Her sensation in the lower leg was intact, and she reported no tingling or numbness in the peroneal nerve distribution. Tinel’s test of the peroneal nerve was negative.

Continue to: X-ray imaging revealed...

When the patient presented to our office, she reported having undergone several failed efforts of nonoperative treatment, including bracing and activity modification. On the basis of the chronicity of the reported symptoms, level of pain, and the desire of the patient to return to full activity, we recommended the surgical reconstruction of the PTFJ by using a split biceps femoris tendon graft.

OPERATIVE TECHNIQUE

The patient was positioned supine on a Jackson table. General anesthesia was utilized. Biplanar fluoroscopic imaging of the fibula was obtained with the fibular head manually dislocated and reduced. A bump was placed beneath the right thigh to create resting knee flexion. The patient was prepped and draped in sterile fashion, and a tourniquet was applied.

A 10-cm curvilinear surgical incision was made centered over the fibular neck and extending proximally within the interval between the iliotibial band and the biceps femoris tendon. Dissection was performed. The peroneal nerve was identified, carefully dissected out, and then isolated with a vessel loop. The biceps femoris tendon insertion on the fibular head was dissected while ensuring that the nerve was isolated, and the anterior half of the tendon was marked approximately 14 cm proximally using a surgical marker. A 15-blade was then used to split the tendon proximally along the marked path while taking care to preserve the tendinous insertion on the fibular head. The split portion of the tendon was freed from all underlying tissue, and the most distal 2 cm was tubularized using a running baseball stitch and No. 2 Ethibond.

The anterior and posterior aspects of the fibular head were then débrided of tissue, and a guidewire was placed anteriorly-to-posterior. After the position of the guidewire was confirmed with fluoroscopy, a 5-0 cannulated reamer was used to drill through the fibular head. Next, the interval between the biceps femoris and iliotibial band was found, and the lateral head of the gastrocnemius was retracted posteriorly within this interval. A portion of the soleus muscle was also elevated off of the posterior capsule and posterior tibia. The iliotibial band insertion at Gerdy’s tubercle was then identified, and a guidewire was placed from anterior-to-posterior within the tibia, with the starting point just posterior to Gerdy’s tubercle. The wire was advanced under direct visualization with an ACL tibial guide and confirmed fluoroscopically. A 5-mm cannulated reamer was then used to drill over the guidewire through the anterior and posterior cortex of the tibia. A suture passer was passed anterior-to-posterior through this tunnel to retrieve the tubularized portion of the biceps femoris graft, which was then shuttled through the tibial tunnel. This same tubularized graft segment was then shuttled anteriorly-to-posteriorly through the fibular tunnel. At this point, approximately 3 cm of the graft protruded from the posterior aspect of the fibular tunnel.

Continue to: The remaining graft was held...

POSTOPERATIVE COURSE

The patient was initially kept in a knee immobilizer following surgery and instructed to use touch-down weight-bearing for 3 weeks. She was switched to a hinged brace at 1 week postoperatively. Physical therapy began with range of motion exercises, and an active flexion was withheld until 6 weeks postoperatively. After 6 weeks, the patient was allowed to progress to an active ROM and increase to weight-bearing as tolerated.  Strengthening was started at 12 weeks.

MRI was performed at 4 months postoperatively because the patient reported pain with running. The MRI demonstrated no evidence of stress reaction or fracture in the area of reconstruction. She was advised to continue with physical therapy and stop running. At 5-month post-reconstruction, the patient reported that her pain had resolved and that she had no complaints of any peroneal nerve neuropraxia. At 6 months she had returned to normal activity without complaints. At this point, she was instructed to follow-up as needed.

The patient was seen in office 5.5 years after the initial surgery for an unrelated orthopedic issue. At this time, follow-up data were obtained for her PTFJ reconstruction. She stated that she was very satisfied with the results of her surgery. She claimed to be pain free and had been performing normal activities without any difficulty. Upon physical examination, she achieved full range of motion.  She had no extension lag or flexion contracture. She achieved functional and clinical Knee Society Scores of 94 and 90 points, respectively.

DISCUSSION

This article details a soft tissue PTFJ reconstruction using a split biceps femoris graft with over 5 years of clinical follow-up. Chronic PTFJ instability is a rare clinical entity, and unless gross instability is evident upon physical examination, its diagnosis may be confused with the diagnosis of more common complaints, such as meniscal tears or iliotibial band syndrome.

Continue to: Ogden first described...

Past instances of PTFJ instability have been managed with closed reduction and protected weight-bearing, as well as with various open reduction techniques.^2-7 Surgical reconstruction is commonly considered in chronic cases or if nonoperative modalities have failed. Although PTFJ arthrodesis or fibular head resection has been used as a prior treatment option, the postoperative complications associated with each of these techniques have since caused them to fall out of favor.

The split biceps femoris graft has been successfully used in the soft tissue reconstruction of PTFJ.^3,5-7 The soft tissue reconstruction of the PTFJ provides advantages over arthrodesis or fibular head resection because it preserves normal anatomy and avoids secondary stresses to the ankle encountered in the latter procedure. Fibular head resection also presents secondary complications, such as the loss of the biceps femoris and posterolateral corner ligament insertion points.⁹ Similar to this study, prior works have reported returns to functionality. However, this study represents the longest clinical postoperative follow-up of PTFJ ligament reconstruction. By using a split biceps graft, the insertion point of the biceps on the fibular head is preserved, thus maintaining normal function while still allowing for an easily tubularized graft for anatomic PTFJ ligament reconstruction.

CONSLUSION

We present data for over 5 years of follow-up for our surgical approach to this rare pathology. To the best of our knowledge, this is the first case report of PTFJ instability that was treated surgically and with a long-term follow-up. The patient did not demonstrate loss of knee motion, pain, or peroneal nerve symptoms. Moreover, she was very satisfied with the procedure at the most recent follow-up and had returned to unrestricted activity. The soft tissue stabilization of a chronically unstable PTFJ is a viable treatment modality that provides good results, and future studies should confirm these satisfactory outcomes in the long-term.

This paper will be judged for the Resident Writer’s Award.

ABSTRACT

Instability of the proximal tibiofibular joint (PTFJ) is a rare clinical condition that presents unique challenges to treatment. We present the case of an active 26-year-old woman with a 4-year history of recurrent PTFJ subluxations, treated surgically at our institution using a split biceps femoris tendon graft for PTFJ reconstruction. She underwent several attempts at nonoperative management until we decided to proceed with surgical intervention. A split biceps femoris graft was used to restore stability of the PTFJ. Approximately 5 years postoperatively, she achieved full range of motion as well as functional and clinical Knee Society Scores of 94 and 90 points, respectively. To the best of our knowledge, this is the first case report of PTFJ instability treated surgically with long-term follow-up. Future studies should focus on the long-term satisfactory outcomes of soft tissue stabilization of a chronically unstable PTFJ.

The instability of the proximal tibiofibular joint (PTFJ) is a rare clinical condition that commonly occurs secondary to an initial pivoting or twisting event of a flexed knee. Although acute PTFJ dislocations respond well to closed reduction and casting, the treatment of chronic PTFJ instability presents a unique challenge.¹ Surgical fixation methods include tibiofibular joint recreation using either a split semitendinosus or biceps femoris graft, as well as a Tightrope device.^2-6 Older surgical options for chronic PTFJ instability include fibular head resection or PTFJ arthrodesis.⁷ However, these older techniques have fallen out of favor, and the optimal surgical technique for the treatment of this injury remains a point of contention.

We present the case of an active 26-year-old woman with a 4-year history of recurrent PTFJ subluxations. The patient was surgically treated at our institution by using a split biceps femoris tendon graft for PTFJ reconstruction. This article specifically details the surgical technique used, provides data obtained at the 5-year clinical follow-up, and reviews prior publications on this injury. The patient provided written informed consent for print and electronic publication of this case report.

CASE

A 26-year-old woman presented with a 4-year history of lateral right knee pain with any physical activity. She stated that her pain began immediately following a fall, which was initially treated with casting and immobilization for approximately 6 weeks. After treatment, she began to develop symptoms of “popping on the outside of the knee.” In the 8 months prior to her presentation to our practice, these symptoms had intensified in pain severity and frequency. She reported that the popping events occurred most often with deep squatting.

No gross deformity was observed upon physical examination, and both knees were visibly symmetric. Evidence of effusion was absent. The patient felt no pain with the passive motion of her knee, and she presented the full range of motion (ROM) from 0° to 120°. Anterior drawer, McMurray, Lachman, and pivot shift tests were all negative. Upon the application of manual pressure, the fibular head could be dislocated anteriorly (Video 1). This dislocation recreated the patient’s symptoms. The fibular head could not be subluxed or dislocated posteriorly. Flexing the knee to 90° facilitated reproducing manual anterior dislocation. The contralateral knee was examined and demonstrated no appreciable PTFJ instability. The patient exhibited no other signs of generalized ligamentous laxity. Her sensation in the lower leg was intact, and she reported no tingling or numbness in the peroneal nerve distribution. Tinel’s test of the peroneal nerve was negative.

Continue to: X-ray imaging revealed...

When the patient presented to our office, she reported having undergone several failed efforts of nonoperative treatment, including bracing and activity modification. On the basis of the chronicity of the reported symptoms, level of pain, and the desire of the patient to return to full activity, we recommended the surgical reconstruction of the PTFJ by using a split biceps femoris tendon graft.

OPERATIVE TECHNIQUE

The patient was positioned supine on a Jackson table. General anesthesia was utilized. Biplanar fluoroscopic imaging of the fibula was obtained with the fibular head manually dislocated and reduced. A bump was placed beneath the right thigh to create resting knee flexion. The patient was prepped and draped in sterile fashion, and a tourniquet was applied.

A 10-cm curvilinear surgical incision was made centered over the fibular neck and extending proximally within the interval between the iliotibial band and the biceps femoris tendon. Dissection was performed. The peroneal nerve was identified, carefully dissected out, and then isolated with a vessel loop. The biceps femoris tendon insertion on the fibular head was dissected while ensuring that the nerve was isolated, and the anterior half of the tendon was marked approximately 14 cm proximally using a surgical marker. A 15-blade was then used to split the tendon proximally along the marked path while taking care to preserve the tendinous insertion on the fibular head. The split portion of the tendon was freed from all underlying tissue, and the most distal 2 cm was tubularized using a running baseball stitch and No. 2 Ethibond.

The anterior and posterior aspects of the fibular head were then débrided of tissue, and a guidewire was placed anteriorly-to-posterior. After the position of the guidewire was confirmed with fluoroscopy, a 5-0 cannulated reamer was used to drill through the fibular head. Next, the interval between the biceps femoris and iliotibial band was found, and the lateral head of the gastrocnemius was retracted posteriorly within this interval. A portion of the soleus muscle was also elevated off of the posterior capsule and posterior tibia. The iliotibial band insertion at Gerdy’s tubercle was then identified, and a guidewire was placed from anterior-to-posterior within the tibia, with the starting point just posterior to Gerdy’s tubercle. The wire was advanced under direct visualization with an ACL tibial guide and confirmed fluoroscopically. A 5-mm cannulated reamer was then used to drill over the guidewire through the anterior and posterior cortex of the tibia. A suture passer was passed anterior-to-posterior through this tunnel to retrieve the tubularized portion of the biceps femoris graft, which was then shuttled through the tibial tunnel. This same tubularized graft segment was then shuttled anteriorly-to-posteriorly through the fibular tunnel. At this point, approximately 3 cm of the graft protruded from the posterior aspect of the fibular tunnel.

Continue to: The remaining graft was held...

POSTOPERATIVE COURSE

The patient was initially kept in a knee immobilizer following surgery and instructed to use touch-down weight-bearing for 3 weeks. She was switched to a hinged brace at 1 week postoperatively. Physical therapy began with range of motion exercises, and an active flexion was withheld until 6 weeks postoperatively. After 6 weeks, the patient was allowed to progress to an active ROM and increase to weight-bearing as tolerated.  Strengthening was started at 12 weeks.

MRI was performed at 4 months postoperatively because the patient reported pain with running. The MRI demonstrated no evidence of stress reaction or fracture in the area of reconstruction. She was advised to continue with physical therapy and stop running. At 5-month post-reconstruction, the patient reported that her pain had resolved and that she had no complaints of any peroneal nerve neuropraxia. At 6 months she had returned to normal activity without complaints. At this point, she was instructed to follow-up as needed.

The patient was seen in office 5.5 years after the initial surgery for an unrelated orthopedic issue. At this time, follow-up data were obtained for her PTFJ reconstruction. She stated that she was very satisfied with the results of her surgery. She claimed to be pain free and had been performing normal activities without any difficulty. Upon physical examination, she achieved full range of motion.  She had no extension lag or flexion contracture. She achieved functional and clinical Knee Society Scores of 94 and 90 points, respectively.

DISCUSSION

This article details a soft tissue PTFJ reconstruction using a split biceps femoris graft with over 5 years of clinical follow-up. Chronic PTFJ instability is a rare clinical entity, and unless gross instability is evident upon physical examination, its diagnosis may be confused with the diagnosis of more common complaints, such as meniscal tears or iliotibial band syndrome.

Continue to: Ogden first described...

Past instances of PTFJ instability have been managed with closed reduction and protected weight-bearing, as well as with various open reduction techniques.^2-7 Surgical reconstruction is commonly considered in chronic cases or if nonoperative modalities have failed. Although PTFJ arthrodesis or fibular head resection has been used as a prior treatment option, the postoperative complications associated with each of these techniques have since caused them to fall out of favor.

The split biceps femoris graft has been successfully used in the soft tissue reconstruction of PTFJ.^3,5-7 The soft tissue reconstruction of the PTFJ provides advantages over arthrodesis or fibular head resection because it preserves normal anatomy and avoids secondary stresses to the ankle encountered in the latter procedure. Fibular head resection also presents secondary complications, such as the loss of the biceps femoris and posterolateral corner ligament insertion points.⁹ Similar to this study, prior works have reported returns to functionality. However, this study represents the longest clinical postoperative follow-up of PTFJ ligament reconstruction. By using a split biceps graft, the insertion point of the biceps on the fibular head is preserved, thus maintaining normal function while still allowing for an easily tubularized graft for anatomic PTFJ ligament reconstruction.

CONSLUSION

We present data for over 5 years of follow-up for our surgical approach to this rare pathology. To the best of our knowledge, this is the first case report of PTFJ instability that was treated surgically and with a long-term follow-up. The patient did not demonstrate loss of knee motion, pain, or peroneal nerve symptoms. Moreover, she was very satisfied with the procedure at the most recent follow-up and had returned to unrestricted activity. The soft tissue stabilization of a chronically unstable PTFJ is a viable treatment modality that provides good results, and future studies should confirm these satisfactory outcomes in the long-term.

This paper will be judged for the Resident Writer’s Award.

ABSTRACT

Instability of the proximal tibiofibular joint (PTFJ) is a rare clinical condition that presents unique challenges to treatment. We present the case of an active 26-year-old woman with a 4-year history of recurrent PTFJ subluxations, treated surgically at our institution using a split biceps femoris tendon graft for PTFJ reconstruction. She underwent several attempts at nonoperative management until we decided to proceed with surgical intervention. A split biceps femoris graft was used to restore stability of the PTFJ. Approximately 5 years postoperatively, she achieved full range of motion as well as functional and clinical Knee Society Scores of 94 and 90 points, respectively. To the best of our knowledge, this is the first case report of PTFJ instability treated surgically with long-term follow-up. Future studies should focus on the long-term satisfactory outcomes of soft tissue stabilization of a chronically unstable PTFJ.

The instability of the proximal tibiofibular joint (PTFJ) is a rare clinical condition that commonly occurs secondary to an initial pivoting or twisting event of a flexed knee. Although acute PTFJ dislocations respond well to closed reduction and casting, the treatment of chronic PTFJ instability presents a unique challenge.¹ Surgical fixation methods include tibiofibular joint recreation using either a split semitendinosus or biceps femoris graft, as well as a Tightrope device.^2-6 Older surgical options for chronic PTFJ instability include fibular head resection or PTFJ arthrodesis.⁷ However, these older techniques have fallen out of favor, and the optimal surgical technique for the treatment of this injury remains a point of contention.

We present the case of an active 26-year-old woman with a 4-year history of recurrent PTFJ subluxations. The patient was surgically treated at our institution by using a split biceps femoris tendon graft for PTFJ reconstruction. This article specifically details the surgical technique used, provides data obtained at the 5-year clinical follow-up, and reviews prior publications on this injury. The patient provided written informed consent for print and electronic publication of this case report.

CASE

A 26-year-old woman presented with a 4-year history of lateral right knee pain with any physical activity. She stated that her pain began immediately following a fall, which was initially treated with casting and immobilization for approximately 6 weeks. After treatment, she began to develop symptoms of “popping on the outside of the knee.” In the 8 months prior to her presentation to our practice, these symptoms had intensified in pain severity and frequency. She reported that the popping events occurred most often with deep squatting.

No gross deformity was observed upon physical examination, and both knees were visibly symmetric. Evidence of effusion was absent. The patient felt no pain with the passive motion of her knee, and she presented the full range of motion (ROM) from 0° to 120°. Anterior drawer, McMurray, Lachman, and pivot shift tests were all negative. Upon the application of manual pressure, the fibular head could be dislocated anteriorly (Video 1). This dislocation recreated the patient’s symptoms. The fibular head could not be subluxed or dislocated posteriorly. Flexing the knee to 90° facilitated reproducing manual anterior dislocation. The contralateral knee was examined and demonstrated no appreciable PTFJ instability. The patient exhibited no other signs of generalized ligamentous laxity. Her sensation in the lower leg was intact, and she reported no tingling or numbness in the peroneal nerve distribution. Tinel’s test of the peroneal nerve was negative.

Continue to: X-ray imaging revealed...

When the patient presented to our office, she reported having undergone several failed efforts of nonoperative treatment, including bracing and activity modification. On the basis of the chronicity of the reported symptoms, level of pain, and the desire of the patient to return to full activity, we recommended the surgical reconstruction of the PTFJ by using a split biceps femoris tendon graft.

OPERATIVE TECHNIQUE

The patient was positioned supine on a Jackson table. General anesthesia was utilized. Biplanar fluoroscopic imaging of the fibula was obtained with the fibular head manually dislocated and reduced. A bump was placed beneath the right thigh to create resting knee flexion. The patient was prepped and draped in sterile fashion, and a tourniquet was applied.

A 10-cm curvilinear surgical incision was made centered over the fibular neck and extending proximally within the interval between the iliotibial band and the biceps femoris tendon. Dissection was performed. The peroneal nerve was identified, carefully dissected out, and then isolated with a vessel loop. The biceps femoris tendon insertion on the fibular head was dissected while ensuring that the nerve was isolated, and the anterior half of the tendon was marked approximately 14 cm proximally using a surgical marker. A 15-blade was then used to split the tendon proximally along the marked path while taking care to preserve the tendinous insertion on the fibular head. The split portion of the tendon was freed from all underlying tissue, and the most distal 2 cm was tubularized using a running baseball stitch and No. 2 Ethibond.

The anterior and posterior aspects of the fibular head were then débrided of tissue, and a guidewire was placed anteriorly-to-posterior. After the position of the guidewire was confirmed with fluoroscopy, a 5-0 cannulated reamer was used to drill through the fibular head. Next, the interval between the biceps femoris and iliotibial band was found, and the lateral head of the gastrocnemius was retracted posteriorly within this interval. A portion of the soleus muscle was also elevated off of the posterior capsule and posterior tibia. The iliotibial band insertion at Gerdy’s tubercle was then identified, and a guidewire was placed from anterior-to-posterior within the tibia, with the starting point just posterior to Gerdy’s tubercle. The wire was advanced under direct visualization with an ACL tibial guide and confirmed fluoroscopically. A 5-mm cannulated reamer was then used to drill over the guidewire through the anterior and posterior cortex of the tibia. A suture passer was passed anterior-to-posterior through this tunnel to retrieve the tubularized portion of the biceps femoris graft, which was then shuttled through the tibial tunnel. This same tubularized graft segment was then shuttled anteriorly-to-posteriorly through the fibular tunnel. At this point, approximately 3 cm of the graft protruded from the posterior aspect of the fibular tunnel.

Continue to: The remaining graft was held...

POSTOPERATIVE COURSE

The patient was initially kept in a knee immobilizer following surgery and instructed to use touch-down weight-bearing for 3 weeks. She was switched to a hinged brace at 1 week postoperatively. Physical therapy began with range of motion exercises, and an active flexion was withheld until 6 weeks postoperatively. After 6 weeks, the patient was allowed to progress to an active ROM and increase to weight-bearing as tolerated.  Strengthening was started at 12 weeks.

MRI was performed at 4 months postoperatively because the patient reported pain with running. The MRI demonstrated no evidence of stress reaction or fracture in the area of reconstruction. She was advised to continue with physical therapy and stop running. At 5-month post-reconstruction, the patient reported that her pain had resolved and that she had no complaints of any peroneal nerve neuropraxia. At 6 months she had returned to normal activity without complaints. At this point, she was instructed to follow-up as needed.

The patient was seen in office 5.5 years after the initial surgery for an unrelated orthopedic issue. At this time, follow-up data were obtained for her PTFJ reconstruction. She stated that she was very satisfied with the results of her surgery. She claimed to be pain free and had been performing normal activities without any difficulty. Upon physical examination, she achieved full range of motion.  She had no extension lag or flexion contracture. She achieved functional and clinical Knee Society Scores of 94 and 90 points, respectively.

DISCUSSION

This article details a soft tissue PTFJ reconstruction using a split biceps femoris graft with over 5 years of clinical follow-up. Chronic PTFJ instability is a rare clinical entity, and unless gross instability is evident upon physical examination, its diagnosis may be confused with the diagnosis of more common complaints, such as meniscal tears or iliotibial band syndrome.

Continue to: Ogden first described...

Past instances of PTFJ instability have been managed with closed reduction and protected weight-bearing, as well as with various open reduction techniques.^2-7 Surgical reconstruction is commonly considered in chronic cases or if nonoperative modalities have failed. Although PTFJ arthrodesis or fibular head resection has been used as a prior treatment option, the postoperative complications associated with each of these techniques have since caused them to fall out of favor.

The split biceps femoris graft has been successfully used in the soft tissue reconstruction of PTFJ.^3,5-7 The soft tissue reconstruction of the PTFJ provides advantages over arthrodesis or fibular head resection because it preserves normal anatomy and avoids secondary stresses to the ankle encountered in the latter procedure. Fibular head resection also presents secondary complications, such as the loss of the biceps femoris and posterolateral corner ligament insertion points.⁹ Similar to this study, prior works have reported returns to functionality. However, this study represents the longest clinical postoperative follow-up of PTFJ ligament reconstruction. By using a split biceps graft, the insertion point of the biceps on the fibular head is preserved, thus maintaining normal function while still allowing for an easily tubularized graft for anatomic PTFJ ligament reconstruction.

CONSLUSION

We present data for over 5 years of follow-up for our surgical approach to this rare pathology. To the best of our knowledge, this is the first case report of PTFJ instability that was treated surgically and with a long-term follow-up. The patient did not demonstrate loss of knee motion, pain, or peroneal nerve symptoms. Moreover, she was very satisfied with the procedure at the most recent follow-up and had returned to unrestricted activity. The soft tissue stabilization of a chronically unstable PTFJ is a viable treatment modality that provides good results, and future studies should confirm these satisfactory outcomes in the long-term.

This paper will be judged for the Resident Writer’s Award.

Patients presenting to the ED with injuries due to accidental self-injection with an epinephrine pen typically receive treatment to alleviate symptoms and reduce the potential of digital ischemia leading to gangrene and loss of tissue and function. Although there is no consensus or set guidelines in the literature regarding the management protocol of such cases, many reports support pharmacological intervention. There are, however, other reports that advocate conservative, nonpharmaceutical management (eg, immersing the affected digit in warm water) or an observation-only approach.

We present the first case report in Saudi Arabia of digital ischemia due to accidental injection of an epinephrine autoinjector, along with a review of the literature and management recommendations.

Case

A 28-year-old woman presented to the ED in significant pain and discomfort 20 minutes after she accidentally injected the entire contents of her aunt’s epinephrine autoinjector (0.3 mg of 1:1000) into her right thumb. The patient, who was in significant pain and discomfort, stated that she was unable to remove the injector needle, which was firmly embedded in the bone of the palmer aspect of the distal phalanx in a manner similar to that of an intraosseous injection (Figure 1).

The patient’s vital signs and oxygen saturation on presentation were within normal limits. The emergency physician successfully removed the embedded needle through moderate countertraction. On examination, the patient’s right thumb was pale and cold, and had poor capillary refill (Figure 2). Due to concerns of the potential for digital tissue ischemia leading to tissue loss and gangrene, warm, moist compresses were applied to the affected thumb, followed by 2% topical nitroglycerin paste, after which the thumb was covered with an occlusive dressing. Since there was no improvement in circulation after 20 minutes, an infiltrate of 5 mg (0.5 mL of 10 mg/mL) of phentolamine (α-agonist) mixed with 2.5 mL of 2% lidocaine was injected at the puncture site and base of the right thumb.¹ Hyperemia developed immediately at both injection sites, and the patient’s right thumb returned to a normal color and sensation 1 hour later, with a return to normal capillary refill. She remained in stable condition and was discharged home. Prior to discharge, the patient was educated on the proper handling and administration of an epinephrine autoinjector.

Discussion

Epinephrine is an ὰ- and β-adrenergic agonist that binds to the ὰ-adrenergic receptors of blood vessels, causing an increase in vascular resistance and vasoconstriction. Although the plasma half-life of epinephrine is approximately 2 to 3 minutes, subcutaneous or intramuscular injection resulting in local vasoconstriction may delay absorption; therefore, the effects of epinephrine may last much longer than its half-life.

The incidence of accidental injection from an epinephrine autoinjector is estimated to be 1 per 50,000 units dispensed.² To date, there are no established treatment guidelines on managing cases of digital injection. An online PubMed and Google Scholar search of the literature found one systematic review,³ four observational studies,^4-7 seven case series,^8-14 and several case reports^1,15-33 on the subject. Most of the patients in the published retrospective studies (71%) were treated conservatively with warming of the affected hand and observation, and the majority of patients in the case reports (87%) were treated pharmacologically, most commonly with topical nitroglycerin and phentolamine.^1,3-34 All of the patients in both the retrospective studies and case reports had restoration of perfusion without necrosis, irrespective of treatment modality. However, patients who were managed conservatively or who were treated with topical nitroglycerin required a longer duration of stay in the ED, suffered from severe reperfusion pain, and in some cases, had a longer time to complete recovery (≥10 weeks).⁸

Pharmaceutical and Nonpharmaceutical Management

Phentolamine. Phentolamine is a nonselective ὰ-adrenergic antagonist that binds to ὰ1 and ὰ2 receptors of blood vessels, resulting in a decrease in peripheral vascular resistance and vasodilation. Phentolamine directly antagonizes the effect of epinephrine by blocking the ὰ-adrenergic receptors, which in our patient resulted in immediate return of digital circulation and full resolution of symptoms.

Topical Nitroglycerin. Nitroglycerin is a nitrate vasodilator that when metabolically converted to nitric oxide, results in smooth muscle relaxation, venodilation, and arteriodilation. Patients suffering from digital ischemia and vasoconstriction may be treated with topical nitroglycerin paste to reverse ischemia by causing smooth muscle relaxation of digital blood vessels. Conservative Management. As previously noted, not all cases of digital epinephrine injection are treated pharmacologically. Some patients are not treated, but kept in observation until the ischemic effects of epinephrine have resolved. Likewise, some patients are treated conservatively with warm water compresses or by fully immersing the affected digit in warm water to facilitate reversal of vasoconstriction and ischemia.^3,8

Treatment Efficacy

In 2007, Fitzcharles-Bowe et al⁸ published a review of 59 cases of digital injection with high-dose epinephrine from 1989 to 2005. In this review, 32 of the 59 patients received no treatment, 25 patients received pharmacological treatment and in two patients, the treatment was unknown. Phentolamine was the most commonly used pharmacological agent (15 of 25 cases or 60%). Although none of the patients experienced digital necrosis, those treated with a local infiltration of phentolamine experienced a faster resolution of symptoms and normalization of perfusion. In 2004, Turner¹ reported a case of a 10-year-old boy who was treated with phentolamine following an accidental injection of epinephrine into his left hand. While circulation returned to the affected digit within 5 minutes of receiving the phentolamine injection, the patient continued to experience reduced sensation in the digit 6 weeks later.⁸

Interestingly, one of the coauthors of the Fitzcharles-Bowe et al⁸ report intentionally injected three of the digits of his left hand (middle, ring, and small fingers) at the same time with high-dose epinephrine to carefully observe and document the outcomes. All three of the digits became very pale and cool, with decreased sensation. The author treated himself conservatively (observation-only). He experienced spontaneous return of circulation in two of the digits within 6 to 10 hours. Although there was some spontaneous return of circulation to the third digit after 13 hours, the author noted prolonged, intense reperfusion pain 4 hours after return of circulation. He also suffered from neuropraxia in the third digit, which did not fully resolve until 10 weeks after the injury.⁸

A review of the literature shows phentolamine to be a safe and effective treatment for patients presenting with digital ischemia, with no long-term adverse effects or complications. Moreover, phentolamine appears to be safe and effective for use in both adult and pediatric patients.^3,8,35-38

Accidental Injection Prevention

Some of the cases of accidental epinephrine injection are due to user error. For example, a novice user may be holding the incorrect end of the injector in his or her hand when attempting to administer/deploy the device, resulting in premature dislodgement of the needle.³⁹

Although, most of the autoinjector devices available today are user-friendly, we believe the addition of a safety feature such as a trigger or safety-lock may further help to reduce accidents. The European Medicines Agency recommends that all patients and caregivers receive training on the proper handling and administration of epinephrine autoinjectors, citing this as the most important factor to ensure successful use of an epinephrine autoinjector and reduce accidental injury.⁴⁰ The patient in this case had not received any formal education or training regarding autoinjector use prior to this incident.

Safety of Lidocaine-Containing Epinephrine in Digital Anesthesia

Aside from cases of accidental digital epinephrine injection, clinicians have traditionally been taught to avoid using lidocaine with epinephrine for digital anesthesia. However, since the introduction of commercial lidocaine with epinephrine in 1948, there are no case reports of digital gangrene from commercially available lidocaine-epinephrine formulations.^41,42 In a multicenter prospective study by Lalonde et al⁴³ of 3,110 consecutive cases of elective injection of low-dose epinephrine in the hand, the authors concluded the likelihood of finger infarction is remote, particularly with possible phentolamine rescue therapy. Moreover, lidocaine-containing epinephrine (1%-2%) has a much lower concentration of epinephrine per mL of solution (5-10 mcg/mL) and appears to be safe for digital use.

Conclusion

This case describes the presentation and treatment of accidental digital injection of epinephrine, highlighting and supporting the benefits of local infiltration with phentolamine and observation until full recovery of perfusion. Local treatment with phentolamine not only facilitates recovery and return of capillary refill, but also shortens the duration of symptoms and alleviates vasoconstriction. In less severe cases, watchful waiting and observation may be appropriate and effective.

This case also underscores the importance of patient and caregiver education on the proper handling and administration of epinephrine autoinjectors to decrease the incidence of accidental injection.

Patients presenting to the ED with injuries due to accidental self-injection with an epinephrine pen typically receive treatment to alleviate symptoms and reduce the potential of digital ischemia leading to gangrene and loss of tissue and function. Although there is no consensus or set guidelines in the literature regarding the management protocol of such cases, many reports support pharmacological intervention. There are, however, other reports that advocate conservative, nonpharmaceutical management (eg, immersing the affected digit in warm water) or an observation-only approach.

We present the first case report in Saudi Arabia of digital ischemia due to accidental injection of an epinephrine autoinjector, along with a review of the literature and management recommendations.

Case

A 28-year-old woman presented to the ED in significant pain and discomfort 20 minutes after she accidentally injected the entire contents of her aunt’s epinephrine autoinjector (0.3 mg of 1:1000) into her right thumb. The patient, who was in significant pain and discomfort, stated that she was unable to remove the injector needle, which was firmly embedded in the bone of the palmer aspect of the distal phalanx in a manner similar to that of an intraosseous injection (Figure 1).

The patient’s vital signs and oxygen saturation on presentation were within normal limits. The emergency physician successfully removed the embedded needle through moderate countertraction. On examination, the patient’s right thumb was pale and cold, and had poor capillary refill (Figure 2). Due to concerns of the potential for digital tissue ischemia leading to tissue loss and gangrene, warm, moist compresses were applied to the affected thumb, followed by 2% topical nitroglycerin paste, after which the thumb was covered with an occlusive dressing. Since there was no improvement in circulation after 20 minutes, an infiltrate of 5 mg (0.5 mL of 10 mg/mL) of phentolamine (α-agonist) mixed with 2.5 mL of 2% lidocaine was injected at the puncture site and base of the right thumb.¹ Hyperemia developed immediately at both injection sites, and the patient’s right thumb returned to a normal color and sensation 1 hour later, with a return to normal capillary refill. She remained in stable condition and was discharged home. Prior to discharge, the patient was educated on the proper handling and administration of an epinephrine autoinjector.

Discussion

Epinephrine is an ὰ- and β-adrenergic agonist that binds to the ὰ-adrenergic receptors of blood vessels, causing an increase in vascular resistance and vasoconstriction. Although the plasma half-life of epinephrine is approximately 2 to 3 minutes, subcutaneous or intramuscular injection resulting in local vasoconstriction may delay absorption; therefore, the effects of epinephrine may last much longer than its half-life.

The incidence of accidental injection from an epinephrine autoinjector is estimated to be 1 per 50,000 units dispensed.² To date, there are no established treatment guidelines on managing cases of digital injection. An online PubMed and Google Scholar search of the literature found one systematic review,³ four observational studies,^4-7 seven case series,^8-14 and several case reports^1,15-33 on the subject. Most of the patients in the published retrospective studies (71%) were treated conservatively with warming of the affected hand and observation, and the majority of patients in the case reports (87%) were treated pharmacologically, most commonly with topical nitroglycerin and phentolamine.^1,3-34 All of the patients in both the retrospective studies and case reports had restoration of perfusion without necrosis, irrespective of treatment modality. However, patients who were managed conservatively or who were treated with topical nitroglycerin required a longer duration of stay in the ED, suffered from severe reperfusion pain, and in some cases, had a longer time to complete recovery (≥10 weeks).⁸

Pharmaceutical and Nonpharmaceutical Management

Phentolamine. Phentolamine is a nonselective ὰ-adrenergic antagonist that binds to ὰ1 and ὰ2 receptors of blood vessels, resulting in a decrease in peripheral vascular resistance and vasodilation. Phentolamine directly antagonizes the effect of epinephrine by blocking the ὰ-adrenergic receptors, which in our patient resulted in immediate return of digital circulation and full resolution of symptoms.

Topical Nitroglycerin. Nitroglycerin is a nitrate vasodilator that when metabolically converted to nitric oxide, results in smooth muscle relaxation, venodilation, and arteriodilation. Patients suffering from digital ischemia and vasoconstriction may be treated with topical nitroglycerin paste to reverse ischemia by causing smooth muscle relaxation of digital blood vessels. Conservative Management. As previously noted, not all cases of digital epinephrine injection are treated pharmacologically. Some patients are not treated, but kept in observation until the ischemic effects of epinephrine have resolved. Likewise, some patients are treated conservatively with warm water compresses or by fully immersing the affected digit in warm water to facilitate reversal of vasoconstriction and ischemia.^3,8

Treatment Efficacy

In 2007, Fitzcharles-Bowe et al⁸ published a review of 59 cases of digital injection with high-dose epinephrine from 1989 to 2005. In this review, 32 of the 59 patients received no treatment, 25 patients received pharmacological treatment and in two patients, the treatment was unknown. Phentolamine was the most commonly used pharmacological agent (15 of 25 cases or 60%). Although none of the patients experienced digital necrosis, those treated with a local infiltration of phentolamine experienced a faster resolution of symptoms and normalization of perfusion. In 2004, Turner¹ reported a case of a 10-year-old boy who was treated with phentolamine following an accidental injection of epinephrine into his left hand. While circulation returned to the affected digit within 5 minutes of receiving the phentolamine injection, the patient continued to experience reduced sensation in the digit 6 weeks later.⁸

Interestingly, one of the coauthors of the Fitzcharles-Bowe et al⁸ report intentionally injected three of the digits of his left hand (middle, ring, and small fingers) at the same time with high-dose epinephrine to carefully observe and document the outcomes. All three of the digits became very pale and cool, with decreased sensation. The author treated himself conservatively (observation-only). He experienced spontaneous return of circulation in two of the digits within 6 to 10 hours. Although there was some spontaneous return of circulation to the third digit after 13 hours, the author noted prolonged, intense reperfusion pain 4 hours after return of circulation. He also suffered from neuropraxia in the third digit, which did not fully resolve until 10 weeks after the injury.⁸

A review of the literature shows phentolamine to be a safe and effective treatment for patients presenting with digital ischemia, with no long-term adverse effects or complications. Moreover, phentolamine appears to be safe and effective for use in both adult and pediatric patients.^3,8,35-38

Accidental Injection Prevention

Some of the cases of accidental epinephrine injection are due to user error. For example, a novice user may be holding the incorrect end of the injector in his or her hand when attempting to administer/deploy the device, resulting in premature dislodgement of the needle.³⁹

Although, most of the autoinjector devices available today are user-friendly, we believe the addition of a safety feature such as a trigger or safety-lock may further help to reduce accidents. The European Medicines Agency recommends that all patients and caregivers receive training on the proper handling and administration of epinephrine autoinjectors, citing this as the most important factor to ensure successful use of an epinephrine autoinjector and reduce accidental injury.⁴⁰ The patient in this case had not received any formal education or training regarding autoinjector use prior to this incident.

Safety of Lidocaine-Containing Epinephrine in Digital Anesthesia

Aside from cases of accidental digital epinephrine injection, clinicians have traditionally been taught to avoid using lidocaine with epinephrine for digital anesthesia. However, since the introduction of commercial lidocaine with epinephrine in 1948, there are no case reports of digital gangrene from commercially available lidocaine-epinephrine formulations.^41,42 In a multicenter prospective study by Lalonde et al⁴³ of 3,110 consecutive cases of elective injection of low-dose epinephrine in the hand, the authors concluded the likelihood of finger infarction is remote, particularly with possible phentolamine rescue therapy. Moreover, lidocaine-containing epinephrine (1%-2%) has a much lower concentration of epinephrine per mL of solution (5-10 mcg/mL) and appears to be safe for digital use.

Conclusion

This case describes the presentation and treatment of accidental digital injection of epinephrine, highlighting and supporting the benefits of local infiltration with phentolamine and observation until full recovery of perfusion. Local treatment with phentolamine not only facilitates recovery and return of capillary refill, but also shortens the duration of symptoms and alleviates vasoconstriction. In less severe cases, watchful waiting and observation may be appropriate and effective.

This case also underscores the importance of patient and caregiver education on the proper handling and administration of epinephrine autoinjectors to decrease the incidence of accidental injection.

Patients presenting to the ED with injuries due to accidental self-injection with an epinephrine pen typically receive treatment to alleviate symptoms and reduce the potential of digital ischemia leading to gangrene and loss of tissue and function. Although there is no consensus or set guidelines in the literature regarding the management protocol of such cases, many reports support pharmacological intervention. There are, however, other reports that advocate conservative, nonpharmaceutical management (eg, immersing the affected digit in warm water) or an observation-only approach.

We present the first case report in Saudi Arabia of digital ischemia due to accidental injection of an epinephrine autoinjector, along with a review of the literature and management recommendations.

Case

A 28-year-old woman presented to the ED in significant pain and discomfort 20 minutes after she accidentally injected the entire contents of her aunt’s epinephrine autoinjector (0.3 mg of 1:1000) into her right thumb. The patient, who was in significant pain and discomfort, stated that she was unable to remove the injector needle, which was firmly embedded in the bone of the palmer aspect of the distal phalanx in a manner similar to that of an intraosseous injection (Figure 1).

The patient’s vital signs and oxygen saturation on presentation were within normal limits. The emergency physician successfully removed the embedded needle through moderate countertraction. On examination, the patient’s right thumb was pale and cold, and had poor capillary refill (Figure 2). Due to concerns of the potential for digital tissue ischemia leading to tissue loss and gangrene, warm, moist compresses were applied to the affected thumb, followed by 2% topical nitroglycerin paste, after which the thumb was covered with an occlusive dressing. Since there was no improvement in circulation after 20 minutes, an infiltrate of 5 mg (0.5 mL of 10 mg/mL) of phentolamine (α-agonist) mixed with 2.5 mL of 2% lidocaine was injected at the puncture site and base of the right thumb.¹ Hyperemia developed immediately at both injection sites, and the patient’s right thumb returned to a normal color and sensation 1 hour later, with a return to normal capillary refill. She remained in stable condition and was discharged home. Prior to discharge, the patient was educated on the proper handling and administration of an epinephrine autoinjector.

Discussion

Epinephrine is an ὰ- and β-adrenergic agonist that binds to the ὰ-adrenergic receptors of blood vessels, causing an increase in vascular resistance and vasoconstriction. Although the plasma half-life of epinephrine is approximately 2 to 3 minutes, subcutaneous or intramuscular injection resulting in local vasoconstriction may delay absorption; therefore, the effects of epinephrine may last much longer than its half-life.

The incidence of accidental injection from an epinephrine autoinjector is estimated to be 1 per 50,000 units dispensed.² To date, there are no established treatment guidelines on managing cases of digital injection. An online PubMed and Google Scholar search of the literature found one systematic review,³ four observational studies,^4-7 seven case series,^8-14 and several case reports^1,15-33 on the subject. Most of the patients in the published retrospective studies (71%) were treated conservatively with warming of the affected hand and observation, and the majority of patients in the case reports (87%) were treated pharmacologically, most commonly with topical nitroglycerin and phentolamine.^1,3-34 All of the patients in both the retrospective studies and case reports had restoration of perfusion without necrosis, irrespective of treatment modality. However, patients who were managed conservatively or who were treated with topical nitroglycerin required a longer duration of stay in the ED, suffered from severe reperfusion pain, and in some cases, had a longer time to complete recovery (≥10 weeks).⁸

Pharmaceutical and Nonpharmaceutical Management

Phentolamine. Phentolamine is a nonselective ὰ-adrenergic antagonist that binds to ὰ1 and ὰ2 receptors of blood vessels, resulting in a decrease in peripheral vascular resistance and vasodilation. Phentolamine directly antagonizes the effect of epinephrine by blocking the ὰ-adrenergic receptors, which in our patient resulted in immediate return of digital circulation and full resolution of symptoms.

Topical Nitroglycerin. Nitroglycerin is a nitrate vasodilator that when metabolically converted to nitric oxide, results in smooth muscle relaxation, venodilation, and arteriodilation. Patients suffering from digital ischemia and vasoconstriction may be treated with topical nitroglycerin paste to reverse ischemia by causing smooth muscle relaxation of digital blood vessels. Conservative Management. As previously noted, not all cases of digital epinephrine injection are treated pharmacologically. Some patients are not treated, but kept in observation until the ischemic effects of epinephrine have resolved. Likewise, some patients are treated conservatively with warm water compresses or by fully immersing the affected digit in warm water to facilitate reversal of vasoconstriction and ischemia.^3,8

Treatment Efficacy

In 2007, Fitzcharles-Bowe et al⁸ published a review of 59 cases of digital injection with high-dose epinephrine from 1989 to 2005. In this review, 32 of the 59 patients received no treatment, 25 patients received pharmacological treatment and in two patients, the treatment was unknown. Phentolamine was the most commonly used pharmacological agent (15 of 25 cases or 60%). Although none of the patients experienced digital necrosis, those treated with a local infiltration of phentolamine experienced a faster resolution of symptoms and normalization of perfusion. In 2004, Turner¹ reported a case of a 10-year-old boy who was treated with phentolamine following an accidental injection of epinephrine into his left hand. While circulation returned to the affected digit within 5 minutes of receiving the phentolamine injection, the patient continued to experience reduced sensation in the digit 6 weeks later.⁸

Interestingly, one of the coauthors of the Fitzcharles-Bowe et al⁸ report intentionally injected three of the digits of his left hand (middle, ring, and small fingers) at the same time with high-dose epinephrine to carefully observe and document the outcomes. All three of the digits became very pale and cool, with decreased sensation. The author treated himself conservatively (observation-only). He experienced spontaneous return of circulation in two of the digits within 6 to 10 hours. Although there was some spontaneous return of circulation to the third digit after 13 hours, the author noted prolonged, intense reperfusion pain 4 hours after return of circulation. He also suffered from neuropraxia in the third digit, which did not fully resolve until 10 weeks after the injury.⁸

A review of the literature shows phentolamine to be a safe and effective treatment for patients presenting with digital ischemia, with no long-term adverse effects or complications. Moreover, phentolamine appears to be safe and effective for use in both adult and pediatric patients.^3,8,35-38

Accidental Injection Prevention

Some of the cases of accidental epinephrine injection are due to user error. For example, a novice user may be holding the incorrect end of the injector in his or her hand when attempting to administer/deploy the device, resulting in premature dislodgement of the needle.³⁹

Although, most of the autoinjector devices available today are user-friendly, we believe the addition of a safety feature such as a trigger or safety-lock may further help to reduce accidents. The European Medicines Agency recommends that all patients and caregivers receive training on the proper handling and administration of epinephrine autoinjectors, citing this as the most important factor to ensure successful use of an epinephrine autoinjector and reduce accidental injury.⁴⁰ The patient in this case had not received any formal education or training regarding autoinjector use prior to this incident.

Safety of Lidocaine-Containing Epinephrine in Digital Anesthesia

Aside from cases of accidental digital epinephrine injection, clinicians have traditionally been taught to avoid using lidocaine with epinephrine for digital anesthesia. However, since the introduction of commercial lidocaine with epinephrine in 1948, there are no case reports of digital gangrene from commercially available lidocaine-epinephrine formulations.^41,42 In a multicenter prospective study by Lalonde et al⁴³ of 3,110 consecutive cases of elective injection of low-dose epinephrine in the hand, the authors concluded the likelihood of finger infarction is remote, particularly with possible phentolamine rescue therapy. Moreover, lidocaine-containing epinephrine (1%-2%) has a much lower concentration of epinephrine per mL of solution (5-10 mcg/mL) and appears to be safe for digital use.

Conclusion

This case describes the presentation and treatment of accidental digital injection of epinephrine, highlighting and supporting the benefits of local infiltration with phentolamine and observation until full recovery of perfusion. Local treatment with phentolamine not only facilitates recovery and return of capillary refill, but also shortens the duration of symptoms and alleviates vasoconstriction. In less severe cases, watchful waiting and observation may be appropriate and effective.

This case also underscores the importance of patient and caregiver education on the proper handling and administration of epinephrine autoinjectors to decrease the incidence of accidental injection.

Purulent pericarditis is an infection within the pericardial space rarely seen in the modern antibiotic era. Most cases are secondary to another infectious process of bacterial, viral, fungal, or parasitic origin.^1,2 Predisposing factors include malignancy, chronic kidney disease, immunosuppression, diabetes mellitus, and alcohol misuse disorder.¹ Although purulent pericarditis has been described extensively in the literature, it is a challenging diagnosis if it is not initially considered within the differential diagnosis repertoire.^1-4 Most authors agree that this may be because it has become an infrequent diagnosis.^1,2 In addition, purulent pericarditis may have an atypical presentation when compared with a classic case of pericarditis.^2,3 The authors believe that this forgotten entity will be revisited through this case.

Case Presentation

A 66-year-old-man was transferred to Veterans Affairs Caribbean Healthcare System (VACHS) from a community hospital with a diagnosis of community-acquired pneumonia (CAP) and bilateral pleural effusions. Four days prior to arrival at the community hospital, the patient had developed diffuse, watery diarrhea, which resolved in 3 days. After resolution of diarrhea, he began experiencing shortness of breath on exertion that progressed to onset at rest. The patient reported no fever, chills, nausea, vomiting, cough, or contact with others who were not healthy. He had a history of alcohol misuse without liver cirrhosis and reported no chronic diseases or use of medications. The patient had no history of tuberculosis exposure or pneumococcal vaccination, and had a negative interferon gamma release assay.

On admission to the community hospital, the patient was treated for CAP with ceftriaxone and azithromycin. On hospital day 3, the patient developed hypoxemia and an altered mental status. He was started on supplemental oxygen and transferred to the intensive care unit (ICU). Antibiotic therapy consequently was changed to levofloxacin and meropenem. However, no clinical improvement was noted on the following days.

On hospital day 7, the patient developed acute respiratory failure that required mechanical ventilation while being transferred to VACHS via air ambulance. His vital signs on arrival were the following: temperature, 97° F; heart rate, 86 beats/min; blood pressure, 103/61 mm Hg; respiratory rate, 14 breaths/min and SaO₂ of 97%, measured while he breathed supplemental oxygen at an FiO₂ of 0.4. 

Hours after arrival, the patient developed sinus tachycardia and hypotension. A bedside 2D echocardiogram demonstrated a large pericardial effusion with diastolic collapse of the right atrium (Figure 2). 

The patient’s clinical condition improved following drainage of pericardial fluid, with no further need for inotropic support. Antibiotic therapy was changed to vancomycin and meropenem. Initial microbiologic samples from pericardial fluid demonstrated Gram-positive diplococci, suggestive of Streptococcus pneumoniae (S pneumoniae) (Figure 4). Other diagnostic pericardial fluid test results included: WBC count 25,330 cmm, with 99% neutrophils and 1% lymphocytes; total protein, 3.8 mg/dL; glucose, < 2.0 mg/dL,LDH, > 2,500 U/L, potassium hydroxide preparation. The tests found no fungus, and the acid fast bacilli smear revealed no Bacillus. However, the pericardial fluid culture failed to demonstrate growth of any organism. Blood cultures also were negative.

The patient underwent anterior thoracotomy with partial pericardiectomy, and a pericardial tube was left in place connected to drainage. During the procedure, an abundant amount of fibrinous tissue was evacuated from the pericardial space (Figure 5). 

The patient was extubated, pericardial and pleural tubes were removed, and he was transferred to the internal medicine ward 24 days after admission to the ICU. He received in-patient physical rehabilitation while completing a 6-week course of IV antibiotics (vancomycin and meropenem). After completion of therapy, the patient received the pneumococcal polysaccharide vaccination, and an echocardiography was repeated. No significant re-accumulation of pericardial effusion or constrictive pattern was evidenced. The patient was discharged to his out-of-state home, and follow-up was consequently lost.

Discussion

Purulent pericarditis is an infection localized within the pericardial space. Most cases are secondary to an infectious process elsewhere, which could be of bacterial, viral, fungal, or parasitic etiology.¹ Five mechanisms could lead the infecting organism to infect the pericardial space; contiguous spread from intrathoracic site, hematogenous spread, extension from myocardial site, perforating injury or surgery, and extension from a subdiaphragmatic site.¹ Predisposing factors for the development of this condition include malignancy, chronic kidney disease, immunosuppression, diabetes mellitus, and alcohol misuse. Pericarditis is an infection localized within the pericardial space.

Purulent pericarditis has become a rare entity in the antibiotic era.² Prior to the development of antibiotics, most cases were secondary to S pneumoniae.^1,2,5,6 As per Cilloniz and colleagues, about 40% to 50% of all cases of purulent pericarditis are caused by Gram-positive bacteria, mostly S pneumoniae.⁵ In this case study, bacterial culture did not reveal growth of an organism—most likely because the patient had received antibiotics elsewhere. However, Gram-positive cocci were seen within the initial pericardial aspirate. This organism was suspected to have spread contiguously from a pulmonary focus, which also led to pleural effusions.

Since the patient in this case study had no history of thoracic surgery, malignancy, or other immunosuppression, the patient’s history of alcohol misuse was the only predisposing factor for development of purulent pericarditis. Contrary to the common presentation of pericarditis, purulent pericarditis may not have the common clinical findings, such as chest pain, pericardial friction rub, and distended neck veins.^2,3 Furthermore, according to Parikh and colleagues, about 35% of affected patients may have a normal electrocardiogram.² Hence, the diagnosis of purulent pericarditis often is missed because the classic signs of pericarditis are often absent, and other nonspecific symptoms are attributed to initial underlying infection.⁷

A high index of suspicion is needed to diagnose purulent pericarditis. Once a diagnosis is made, initial treatment should consist of prompt drainage of pericardial fluid combined with systemic antibiotic therapy. Vancomycin and a third-generation cephalosporin may be started empirically until results of pericardial fluid cultures become available.³ Drainage can be achieved by pericardiocentesis, pericardiotomy, or pericardiectomy (partial or total).¹ In cases of hemodynamic instability due to cardiac tamponade, sonographically guided pericardiocentesis should be undertaken and an indwelling pericardial catheter left in place.¹ Although this is the simplest and fastest method of evacuation, it may not be effective when dealing with thick, fibrinous fluid. In such cases, intrapericardial fibrinolysis may be considered. This approach may be undertaken early in the process, after drainage insertion, or as salvage therapy, when there has been incomplete evacuation of purulent material or open surgical drainage is not available.

Streptokinase, urokinase, and tissue plasminogen activator have been used for intrapericadial fibrinolysis.¹ However, there is no definite data on dosage or frequency at which these medications should be administered. No matter the therapeutic approach, effective drainage of the pericardial fluid is crucial to avoid the development of pericardial constriction. Constrictive pericarditis occurs when fibrosis and adhesions create a dense pericardium that encases the heart. This causes impaired ventricular filling that can lead eventually to heart failure.⁴ Pericardiectomy is the definitive treatment for constrictive pericarditis.

Conclusion

Although purulent pericarditis has become a rare diagnosis since the development of antibiotics, knowledge of how to identify it is essential since mortality reaches 100% if the diagnosis is missed.⁴ Even when the condition is promptly diagnosed and treated, mortality is 40%, mainly due to cardiac tamponade, septic shock, or constriction.¹ The case presented here illustrates the clinical features associated with this condition. Knowing these features can translate in a successful patient outcome.

Purulent pericarditis is an infection within the pericardial space rarely seen in the modern antibiotic era. Most cases are secondary to another infectious process of bacterial, viral, fungal, or parasitic origin.^1,2 Predisposing factors include malignancy, chronic kidney disease, immunosuppression, diabetes mellitus, and alcohol misuse disorder.¹ Although purulent pericarditis has been described extensively in the literature, it is a challenging diagnosis if it is not initially considered within the differential diagnosis repertoire.^1-4 Most authors agree that this may be because it has become an infrequent diagnosis.^1,2 In addition, purulent pericarditis may have an atypical presentation when compared with a classic case of pericarditis.^2,3 The authors believe that this forgotten entity will be revisited through this case.

Case Presentation

A 66-year-old-man was transferred to Veterans Affairs Caribbean Healthcare System (VACHS) from a community hospital with a diagnosis of community-acquired pneumonia (CAP) and bilateral pleural effusions. Four days prior to arrival at the community hospital, the patient had developed diffuse, watery diarrhea, which resolved in 3 days. After resolution of diarrhea, he began experiencing shortness of breath on exertion that progressed to onset at rest. The patient reported no fever, chills, nausea, vomiting, cough, or contact with others who were not healthy. He had a history of alcohol misuse without liver cirrhosis and reported no chronic diseases or use of medications. The patient had no history of tuberculosis exposure or pneumococcal vaccination, and had a negative interferon gamma release assay.

On admission to the community hospital, the patient was treated for CAP with ceftriaxone and azithromycin. On hospital day 3, the patient developed hypoxemia and an altered mental status. He was started on supplemental oxygen and transferred to the intensive care unit (ICU). Antibiotic therapy consequently was changed to levofloxacin and meropenem. However, no clinical improvement was noted on the following days.

On hospital day 7, the patient developed acute respiratory failure that required mechanical ventilation while being transferred to VACHS via air ambulance. His vital signs on arrival were the following: temperature, 97° F; heart rate, 86 beats/min; blood pressure, 103/61 mm Hg; respiratory rate, 14 breaths/min and SaO₂ of 97%, measured while he breathed supplemental oxygen at an FiO₂ of 0.4. 

Hours after arrival, the patient developed sinus tachycardia and hypotension. A bedside 2D echocardiogram demonstrated a large pericardial effusion with diastolic collapse of the right atrium (Figure 2). 

The patient’s clinical condition improved following drainage of pericardial fluid, with no further need for inotropic support. Antibiotic therapy was changed to vancomycin and meropenem. Initial microbiologic samples from pericardial fluid demonstrated Gram-positive diplococci, suggestive of Streptococcus pneumoniae (S pneumoniae) (Figure 4). Other diagnostic pericardial fluid test results included: WBC count 25,330 cmm, with 99% neutrophils and 1% lymphocytes; total protein, 3.8 mg/dL; glucose, < 2.0 mg/dL,LDH, > 2,500 U/L, potassium hydroxide preparation. The tests found no fungus, and the acid fast bacilli smear revealed no Bacillus. However, the pericardial fluid culture failed to demonstrate growth of any organism. Blood cultures also were negative.

The patient underwent anterior thoracotomy with partial pericardiectomy, and a pericardial tube was left in place connected to drainage. During the procedure, an abundant amount of fibrinous tissue was evacuated from the pericardial space (Figure 5). 

The patient was extubated, pericardial and pleural tubes were removed, and he was transferred to the internal medicine ward 24 days after admission to the ICU. He received in-patient physical rehabilitation while completing a 6-week course of IV antibiotics (vancomycin and meropenem). After completion of therapy, the patient received the pneumococcal polysaccharide vaccination, and an echocardiography was repeated. No significant re-accumulation of pericardial effusion or constrictive pattern was evidenced. The patient was discharged to his out-of-state home, and follow-up was consequently lost.

Discussion

Purulent pericarditis is an infection localized within the pericardial space. Most cases are secondary to an infectious process elsewhere, which could be of bacterial, viral, fungal, or parasitic etiology.¹ Five mechanisms could lead the infecting organism to infect the pericardial space; contiguous spread from intrathoracic site, hematogenous spread, extension from myocardial site, perforating injury or surgery, and extension from a subdiaphragmatic site.¹ Predisposing factors for the development of this condition include malignancy, chronic kidney disease, immunosuppression, diabetes mellitus, and alcohol misuse. Pericarditis is an infection localized within the pericardial space.

Purulent pericarditis has become a rare entity in the antibiotic era.² Prior to the development of antibiotics, most cases were secondary to S pneumoniae.^1,2,5,6 As per Cilloniz and colleagues, about 40% to 50% of all cases of purulent pericarditis are caused by Gram-positive bacteria, mostly S pneumoniae.⁵ In this case study, bacterial culture did not reveal growth of an organism—most likely because the patient had received antibiotics elsewhere. However, Gram-positive cocci were seen within the initial pericardial aspirate. This organism was suspected to have spread contiguously from a pulmonary focus, which also led to pleural effusions.

Since the patient in this case study had no history of thoracic surgery, malignancy, or other immunosuppression, the patient’s history of alcohol misuse was the only predisposing factor for development of purulent pericarditis. Contrary to the common presentation of pericarditis, purulent pericarditis may not have the common clinical findings, such as chest pain, pericardial friction rub, and distended neck veins.^2,3 Furthermore, according to Parikh and colleagues, about 35% of affected patients may have a normal electrocardiogram.² Hence, the diagnosis of purulent pericarditis often is missed because the classic signs of pericarditis are often absent, and other nonspecific symptoms are attributed to initial underlying infection.⁷

A high index of suspicion is needed to diagnose purulent pericarditis. Once a diagnosis is made, initial treatment should consist of prompt drainage of pericardial fluid combined with systemic antibiotic therapy. Vancomycin and a third-generation cephalosporin may be started empirically until results of pericardial fluid cultures become available.³ Drainage can be achieved by pericardiocentesis, pericardiotomy, or pericardiectomy (partial or total).¹ In cases of hemodynamic instability due to cardiac tamponade, sonographically guided pericardiocentesis should be undertaken and an indwelling pericardial catheter left in place.¹ Although this is the simplest and fastest method of evacuation, it may not be effective when dealing with thick, fibrinous fluid. In such cases, intrapericardial fibrinolysis may be considered. This approach may be undertaken early in the process, after drainage insertion, or as salvage therapy, when there has been incomplete evacuation of purulent material or open surgical drainage is not available.

Streptokinase, urokinase, and tissue plasminogen activator have been used for intrapericadial fibrinolysis.¹ However, there is no definite data on dosage or frequency at which these medications should be administered. No matter the therapeutic approach, effective drainage of the pericardial fluid is crucial to avoid the development of pericardial constriction. Constrictive pericarditis occurs when fibrosis and adhesions create a dense pericardium that encases the heart. This causes impaired ventricular filling that can lead eventually to heart failure.⁴ Pericardiectomy is the definitive treatment for constrictive pericarditis.

Conclusion

Although purulent pericarditis has become a rare diagnosis since the development of antibiotics, knowledge of how to identify it is essential since mortality reaches 100% if the diagnosis is missed.⁴ Even when the condition is promptly diagnosed and treated, mortality is 40%, mainly due to cardiac tamponade, septic shock, or constriction.¹ The case presented here illustrates the clinical features associated with this condition. Knowing these features can translate in a successful patient outcome.

Purulent pericarditis is an infection within the pericardial space rarely seen in the modern antibiotic era. Most cases are secondary to another infectious process of bacterial, viral, fungal, or parasitic origin.^1,2 Predisposing factors include malignancy, chronic kidney disease, immunosuppression, diabetes mellitus, and alcohol misuse disorder.¹ Although purulent pericarditis has been described extensively in the literature, it is a challenging diagnosis if it is not initially considered within the differential diagnosis repertoire.^1-4 Most authors agree that this may be because it has become an infrequent diagnosis.^1,2 In addition, purulent pericarditis may have an atypical presentation when compared with a classic case of pericarditis.^2,3 The authors believe that this forgotten entity will be revisited through this case.

Case Presentation

A 66-year-old-man was transferred to Veterans Affairs Caribbean Healthcare System (VACHS) from a community hospital with a diagnosis of community-acquired pneumonia (CAP) and bilateral pleural effusions. Four days prior to arrival at the community hospital, the patient had developed diffuse, watery diarrhea, which resolved in 3 days. After resolution of diarrhea, he began experiencing shortness of breath on exertion that progressed to onset at rest. The patient reported no fever, chills, nausea, vomiting, cough, or contact with others who were not healthy. He had a history of alcohol misuse without liver cirrhosis and reported no chronic diseases or use of medications. The patient had no history of tuberculosis exposure or pneumococcal vaccination, and had a negative interferon gamma release assay.

On admission to the community hospital, the patient was treated for CAP with ceftriaxone and azithromycin. On hospital day 3, the patient developed hypoxemia and an altered mental status. He was started on supplemental oxygen and transferred to the intensive care unit (ICU). Antibiotic therapy consequently was changed to levofloxacin and meropenem. However, no clinical improvement was noted on the following days.

On hospital day 7, the patient developed acute respiratory failure that required mechanical ventilation while being transferred to VACHS via air ambulance. His vital signs on arrival were the following: temperature, 97° F; heart rate, 86 beats/min; blood pressure, 103/61 mm Hg; respiratory rate, 14 breaths/min and SaO₂ of 97%, measured while he breathed supplemental oxygen at an FiO₂ of 0.4. 

Hours after arrival, the patient developed sinus tachycardia and hypotension. A bedside 2D echocardiogram demonstrated a large pericardial effusion with diastolic collapse of the right atrium (Figure 2). 

The patient’s clinical condition improved following drainage of pericardial fluid, with no further need for inotropic support. Antibiotic therapy was changed to vancomycin and meropenem. Initial microbiologic samples from pericardial fluid demonstrated Gram-positive diplococci, suggestive of Streptococcus pneumoniae (S pneumoniae) (Figure 4). Other diagnostic pericardial fluid test results included: WBC count 25,330 cmm, with 99% neutrophils and 1% lymphocytes; total protein, 3.8 mg/dL; glucose, < 2.0 mg/dL,LDH, > 2,500 U/L, potassium hydroxide preparation. The tests found no fungus, and the acid fast bacilli smear revealed no Bacillus. However, the pericardial fluid culture failed to demonstrate growth of any organism. Blood cultures also were negative.

The patient underwent anterior thoracotomy with partial pericardiectomy, and a pericardial tube was left in place connected to drainage. During the procedure, an abundant amount of fibrinous tissue was evacuated from the pericardial space (Figure 5). 

The patient was extubated, pericardial and pleural tubes were removed, and he was transferred to the internal medicine ward 24 days after admission to the ICU. He received in-patient physical rehabilitation while completing a 6-week course of IV antibiotics (vancomycin and meropenem). After completion of therapy, the patient received the pneumococcal polysaccharide vaccination, and an echocardiography was repeated. No significant re-accumulation of pericardial effusion or constrictive pattern was evidenced. The patient was discharged to his out-of-state home, and follow-up was consequently lost.

Discussion

Purulent pericarditis is an infection localized within the pericardial space. Most cases are secondary to an infectious process elsewhere, which could be of bacterial, viral, fungal, or parasitic etiology.¹ Five mechanisms could lead the infecting organism to infect the pericardial space; contiguous spread from intrathoracic site, hematogenous spread, extension from myocardial site, perforating injury or surgery, and extension from a subdiaphragmatic site.¹ Predisposing factors for the development of this condition include malignancy, chronic kidney disease, immunosuppression, diabetes mellitus, and alcohol misuse. Pericarditis is an infection localized within the pericardial space.

Purulent pericarditis has become a rare entity in the antibiotic era.² Prior to the development of antibiotics, most cases were secondary to S pneumoniae.^1,2,5,6 As per Cilloniz and colleagues, about 40% to 50% of all cases of purulent pericarditis are caused by Gram-positive bacteria, mostly S pneumoniae.⁵ In this case study, bacterial culture did not reveal growth of an organism—most likely because the patient had received antibiotics elsewhere. However, Gram-positive cocci were seen within the initial pericardial aspirate. This organism was suspected to have spread contiguously from a pulmonary focus, which also led to pleural effusions.

Since the patient in this case study had no history of thoracic surgery, malignancy, or other immunosuppression, the patient’s history of alcohol misuse was the only predisposing factor for development of purulent pericarditis. Contrary to the common presentation of pericarditis, purulent pericarditis may not have the common clinical findings, such as chest pain, pericardial friction rub, and distended neck veins.^2,3 Furthermore, according to Parikh and colleagues, about 35% of affected patients may have a normal electrocardiogram.² Hence, the diagnosis of purulent pericarditis often is missed because the classic signs of pericarditis are often absent, and other nonspecific symptoms are attributed to initial underlying infection.⁷

A high index of suspicion is needed to diagnose purulent pericarditis. Once a diagnosis is made, initial treatment should consist of prompt drainage of pericardial fluid combined with systemic antibiotic therapy. Vancomycin and a third-generation cephalosporin may be started empirically until results of pericardial fluid cultures become available.³ Drainage can be achieved by pericardiocentesis, pericardiotomy, or pericardiectomy (partial or total).¹ In cases of hemodynamic instability due to cardiac tamponade, sonographically guided pericardiocentesis should be undertaken and an indwelling pericardial catheter left in place.¹ Although this is the simplest and fastest method of evacuation, it may not be effective when dealing with thick, fibrinous fluid. In such cases, intrapericardial fibrinolysis may be considered. This approach may be undertaken early in the process, after drainage insertion, or as salvage therapy, when there has been incomplete evacuation of purulent material or open surgical drainage is not available.

Streptokinase, urokinase, and tissue plasminogen activator have been used for intrapericadial fibrinolysis.¹ However, there is no definite data on dosage or frequency at which these medications should be administered. No matter the therapeutic approach, effective drainage of the pericardial fluid is crucial to avoid the development of pericardial constriction. Constrictive pericarditis occurs when fibrosis and adhesions create a dense pericardium that encases the heart. This causes impaired ventricular filling that can lead eventually to heart failure.⁴ Pericardiectomy is the definitive treatment for constrictive pericarditis.

Conclusion

Although purulent pericarditis has become a rare diagnosis since the development of antibiotics, knowledge of how to identify it is essential since mortality reaches 100% if the diagnosis is missed.⁴ Even when the condition is promptly diagnosed and treated, mortality is 40%, mainly due to cardiac tamponade, septic shock, or constriction.¹ The case presented here illustrates the clinical features associated with this condition. Knowing these features can translate in a successful patient outcome.

Case

An 84-year-old man, who was a resident at a local nursing home, presented for evaluation after the nursing staff noticed an increasingly swollen mass on the patient’s left groin. The patient’s medical history was significant for bilateral aortofemoral graft surgery, dementia, hypertension, and severe peripheral artery disease (PAD). He was not on any anticoagulation or antiplatelet agents. Due to the patient’s dementia, he was unable to provide a history regarding the onset of the swelling or any other signs or symptoms.

On examination, the patient did not appear in distress. His son, who was the patient’s durable power of attorney, was likewise unable to provide a clear timeframe regarding onset of the mass. The patient had no recent history of trauma and had not undergone any recent medical procedures. Vital signs at presentation were: blood pressure, 110/70 mm Hg; heart rate, 84 beats/min; respiratory rate, 13 breaths/min; and temperature, 98.6°F. Oxygen saturation was 94% on room air.

Clinical examination revealed a pulsatile, purple left groin mass and bruit. The mass was located around the left inguinal ligament and extended down the proximal, inner thigh (Figure 1). There was no drainage or lesions from the mass. Inspection of the patient’s hip demonstrated decreased adduction, limited by the mass; otherwise, there was normal range of motion. The dorsalis pedis and posterior tibial pulses were equal and intact, and the rest of the physical examination was unremarkable.

Discussion

A true aneurysm differs from a PSA in that true aneurysms involve all three layers of the vessel wall. A PSA consists partly of the vessel wall and partly of encapsulating fibrous tissue or surrounding tissue.

Etiology

Femoral artery PSAs can be iatrogenic, for example, develop following cardiac catheterization or at the anastomotic site of previous surgery.¹ The incidence of diagnostic postcatheterization PSA ranges from 0.05% to 2%, whereas interventional postcatheterization PSA ranges from 2% to 6%.²

With the increasing number of peripheral coronary diagnostics and interventions, emergency physicians should include PSA in the differential diagnosis of patients with a recent or remote history of catheterization or bypass grafts. Less commonly, femoral PSAs are caused by non-surgical trauma or infection (ie, mycotic PSA). Patient risk factors for development of PSA include obesity, hypertension, PAD, and anticoagulation.³ Patients with femoral artery PSAs may present with a painful or painless pulsatile mass. Mass effect of the PSA can compress nearby neurovascular structures, leading to femoral neuropathies or limb edema secondary to venous obstruction.⁴ Complications of embolization or thrombosis can cause limb ischemia, neuropathy, and claudication, while rupture may present with a rapidly expanding groin hematoma. Additionally, sizeable PSAs can cause overlying skin necrosis.⁵

Imaging Studies

Diagnosis of a PSA can be made through Doppler ultrasound, which is the preferred imaging modality due to its accuracy, noninvasive nature, and low cost. Doppler ultrasound has been found to have a sensitivity of 94% and specificity of 97% in detecting PSAs. Additional imaging with CTA can provide further definition of vasculopathy.⁶ Treatment should be considered for patients with a symptomatic femoral PSA, a PSA measuring more than 3 cm, or patients who are on anticoagulation therapy. Studies have shown that observation-only and follow-up may be appropriate for patients with a PSA measuring less than 3 cm. A study by Toursarkissian et al⁷ found that the majority of PSAs smaller than 3 cm spontaneously resolved in a mean of 23 days without limb-threatening complications.

Treatment

Traditionally, open surgical repair techniques were the only treatment option for PSAs. However, in the early 1990s, the advent of new techniques such as stenting, coil insertion, ultrasound-guided compression, and ultrasound-guided thrombin injection, have developed as alternatives to open surgical repair; there has been variable success to these minimally invasive approaches.^5,8

Ultrasound-Guided Compression. A conservative approach to treating PSAs, ultrasound-guided compression requires sustained compression by a skilled physician. This technique is associated with significant discomfort to the patient.⁵ Ultrasound-Guided Thrombin Injection. This technique is the treatment of choice for postcatheterization PSA. However, this intervention is contraindicated in patients who have concerning features such as an infected PSA, rapid expansion, skin necrosis, or signs of limb ischemia. Additionally, ultrasound-guided thrombin injection is not appropriate for use in patients with a PSA occurring at anastomosis of a synthetic graft and native artery.⁵

Conclusion

Based on our patient’s clinical presentation and history of aortofemoral bypass surgery, we suspected a femoral PSA. While the PSA noted in our patient was sizeable, imaging studies and clinical examination showed no sign of limb ischemia or rupture.

Femoral PSAs are usually iatrogenic in nature, typically developing shortly after catheterization or a previous bypass surgery. The most serious complication of a PSA is rupture, but a thorough examination of the distal extremity is warranted to assess for limb ischemia as well. Ultrasound imaging is considered the modality of choice based on its high sensitivity and sensitivity for detecting PSAs.

Small PSAs (<3 cm) can be managed medically, but larger PSAs (>3 cm) require treatment. Newer techniques, including stenting, coil insertion, ultrasound-guided compression, and ultrasound-guided thrombin injection are alternatives to open surgical repair of larger, uncomplicated PSAs. However, urgent open surgical repair is the only option when there is evidence of a ruptured PSA, ischemia, or skin necrosis.

Case

An 84-year-old man, who was a resident at a local nursing home, presented for evaluation after the nursing staff noticed an increasingly swollen mass on the patient’s left groin. The patient’s medical history was significant for bilateral aortofemoral graft surgery, dementia, hypertension, and severe peripheral artery disease (PAD). He was not on any anticoagulation or antiplatelet agents. Due to the patient’s dementia, he was unable to provide a history regarding the onset of the swelling or any other signs or symptoms.

On examination, the patient did not appear in distress. His son, who was the patient’s durable power of attorney, was likewise unable to provide a clear timeframe regarding onset of the mass. The patient had no recent history of trauma and had not undergone any recent medical procedures. Vital signs at presentation were: blood pressure, 110/70 mm Hg; heart rate, 84 beats/min; respiratory rate, 13 breaths/min; and temperature, 98.6°F. Oxygen saturation was 94% on room air.

Clinical examination revealed a pulsatile, purple left groin mass and bruit. The mass was located around the left inguinal ligament and extended down the proximal, inner thigh (Figure 1). There was no drainage or lesions from the mass. Inspection of the patient’s hip demonstrated decreased adduction, limited by the mass; otherwise, there was normal range of motion. The dorsalis pedis and posterior tibial pulses were equal and intact, and the rest of the physical examination was unremarkable.

Discussion

A true aneurysm differs from a PSA in that true aneurysms involve all three layers of the vessel wall. A PSA consists partly of the vessel wall and partly of encapsulating fibrous tissue or surrounding tissue.

Etiology

Femoral artery PSAs can be iatrogenic, for example, develop following cardiac catheterization or at the anastomotic site of previous surgery.¹ The incidence of diagnostic postcatheterization PSA ranges from 0.05% to 2%, whereas interventional postcatheterization PSA ranges from 2% to 6%.²

With the increasing number of peripheral coronary diagnostics and interventions, emergency physicians should include PSA in the differential diagnosis of patients with a recent or remote history of catheterization or bypass grafts. Less commonly, femoral PSAs are caused by non-surgical trauma or infection (ie, mycotic PSA). Patient risk factors for development of PSA include obesity, hypertension, PAD, and anticoagulation.³ Patients with femoral artery PSAs may present with a painful or painless pulsatile mass. Mass effect of the PSA can compress nearby neurovascular structures, leading to femoral neuropathies or limb edema secondary to venous obstruction.⁴ Complications of embolization or thrombosis can cause limb ischemia, neuropathy, and claudication, while rupture may present with a rapidly expanding groin hematoma. Additionally, sizeable PSAs can cause overlying skin necrosis.⁵

Imaging Studies

Diagnosis of a PSA can be made through Doppler ultrasound, which is the preferred imaging modality due to its accuracy, noninvasive nature, and low cost. Doppler ultrasound has been found to have a sensitivity of 94% and specificity of 97% in detecting PSAs. Additional imaging with CTA can provide further definition of vasculopathy.⁶ Treatment should be considered for patients with a symptomatic femoral PSA, a PSA measuring more than 3 cm, or patients who are on anticoagulation therapy. Studies have shown that observation-only and follow-up may be appropriate for patients with a PSA measuring less than 3 cm. A study by Toursarkissian et al⁷ found that the majority of PSAs smaller than 3 cm spontaneously resolved in a mean of 23 days without limb-threatening complications.

Treatment

Traditionally, open surgical repair techniques were the only treatment option for PSAs. However, in the early 1990s, the advent of new techniques such as stenting, coil insertion, ultrasound-guided compression, and ultrasound-guided thrombin injection, have developed as alternatives to open surgical repair; there has been variable success to these minimally invasive approaches.^5,8

Ultrasound-Guided Compression. A conservative approach to treating PSAs, ultrasound-guided compression requires sustained compression by a skilled physician. This technique is associated with significant discomfort to the patient.⁵ Ultrasound-Guided Thrombin Injection. This technique is the treatment of choice for postcatheterization PSA. However, this intervention is contraindicated in patients who have concerning features such as an infected PSA, rapid expansion, skin necrosis, or signs of limb ischemia. Additionally, ultrasound-guided thrombin injection is not appropriate for use in patients with a PSA occurring at anastomosis of a synthetic graft and native artery.⁵

Conclusion

Based on our patient’s clinical presentation and history of aortofemoral bypass surgery, we suspected a femoral PSA. While the PSA noted in our patient was sizeable, imaging studies and clinical examination showed no sign of limb ischemia or rupture.

Femoral PSAs are usually iatrogenic in nature, typically developing shortly after catheterization or a previous bypass surgery. The most serious complication of a PSA is rupture, but a thorough examination of the distal extremity is warranted to assess for limb ischemia as well. Ultrasound imaging is considered the modality of choice based on its high sensitivity and sensitivity for detecting PSAs.

Small PSAs (<3 cm) can be managed medically, but larger PSAs (>3 cm) require treatment. Newer techniques, including stenting, coil insertion, ultrasound-guided compression, and ultrasound-guided thrombin injection are alternatives to open surgical repair of larger, uncomplicated PSAs. However, urgent open surgical repair is the only option when there is evidence of a ruptured PSA, ischemia, or skin necrosis.

Case

An 84-year-old man, who was a resident at a local nursing home, presented for evaluation after the nursing staff noticed an increasingly swollen mass on the patient’s left groin. The patient’s medical history was significant for bilateral aortofemoral graft surgery, dementia, hypertension, and severe peripheral artery disease (PAD). He was not on any anticoagulation or antiplatelet agents. Due to the patient’s dementia, he was unable to provide a history regarding the onset of the swelling or any other signs or symptoms.

On examination, the patient did not appear in distress. His son, who was the patient’s durable power of attorney, was likewise unable to provide a clear timeframe regarding onset of the mass. The patient had no recent history of trauma and had not undergone any recent medical procedures. Vital signs at presentation were: blood pressure, 110/70 mm Hg; heart rate, 84 beats/min; respiratory rate, 13 breaths/min; and temperature, 98.6°F. Oxygen saturation was 94% on room air.

Clinical examination revealed a pulsatile, purple left groin mass and bruit. The mass was located around the left inguinal ligament and extended down the proximal, inner thigh (Figure 1). There was no drainage or lesions from the mass. Inspection of the patient’s hip demonstrated decreased adduction, limited by the mass; otherwise, there was normal range of motion. The dorsalis pedis and posterior tibial pulses were equal and intact, and the rest of the physical examination was unremarkable.

Discussion

A true aneurysm differs from a PSA in that true aneurysms involve all three layers of the vessel wall. A PSA consists partly of the vessel wall and partly of encapsulating fibrous tissue or surrounding tissue.

Etiology

Femoral artery PSAs can be iatrogenic, for example, develop following cardiac catheterization or at the anastomotic site of previous surgery.¹ The incidence of diagnostic postcatheterization PSA ranges from 0.05% to 2%, whereas interventional postcatheterization PSA ranges from 2% to 6%.²

With the increasing number of peripheral coronary diagnostics and interventions, emergency physicians should include PSA in the differential diagnosis of patients with a recent or remote history of catheterization or bypass grafts. Less commonly, femoral PSAs are caused by non-surgical trauma or infection (ie, mycotic PSA). Patient risk factors for development of PSA include obesity, hypertension, PAD, and anticoagulation.³ Patients with femoral artery PSAs may present with a painful or painless pulsatile mass. Mass effect of the PSA can compress nearby neurovascular structures, leading to femoral neuropathies or limb edema secondary to venous obstruction.⁴ Complications of embolization or thrombosis can cause limb ischemia, neuropathy, and claudication, while rupture may present with a rapidly expanding groin hematoma. Additionally, sizeable PSAs can cause overlying skin necrosis.⁵

Imaging Studies

Diagnosis of a PSA can be made through Doppler ultrasound, which is the preferred imaging modality due to its accuracy, noninvasive nature, and low cost. Doppler ultrasound has been found to have a sensitivity of 94% and specificity of 97% in detecting PSAs. Additional imaging with CTA can provide further definition of vasculopathy.⁶ Treatment should be considered for patients with a symptomatic femoral PSA, a PSA measuring more than 3 cm, or patients who are on anticoagulation therapy. Studies have shown that observation-only and follow-up may be appropriate for patients with a PSA measuring less than 3 cm. A study by Toursarkissian et al⁷ found that the majority of PSAs smaller than 3 cm spontaneously resolved in a mean of 23 days without limb-threatening complications.

Treatment

Traditionally, open surgical repair techniques were the only treatment option for PSAs. However, in the early 1990s, the advent of new techniques such as stenting, coil insertion, ultrasound-guided compression, and ultrasound-guided thrombin injection, have developed as alternatives to open surgical repair; there has been variable success to these minimally invasive approaches.^5,8

Ultrasound-Guided Compression. A conservative approach to treating PSAs, ultrasound-guided compression requires sustained compression by a skilled physician. This technique is associated with significant discomfort to the patient.⁵ Ultrasound-Guided Thrombin Injection. This technique is the treatment of choice for postcatheterization PSA. However, this intervention is contraindicated in patients who have concerning features such as an infected PSA, rapid expansion, skin necrosis, or signs of limb ischemia. Additionally, ultrasound-guided thrombin injection is not appropriate for use in patients with a PSA occurring at anastomosis of a synthetic graft and native artery.⁵

Conclusion

Based on our patient’s clinical presentation and history of aortofemoral bypass surgery, we suspected a femoral PSA. While the PSA noted in our patient was sizeable, imaging studies and clinical examination showed no sign of limb ischemia or rupture.

Femoral PSAs are usually iatrogenic in nature, typically developing shortly after catheterization or a previous bypass surgery. The most serious complication of a PSA is rupture, but a thorough examination of the distal extremity is warranted to assess for limb ischemia as well. Ultrasound imaging is considered the modality of choice based on its high sensitivity and sensitivity for detecting PSAs.

Small PSAs (<3 cm) can be managed medically, but larger PSAs (>3 cm) require treatment. Newer techniques, including stenting, coil insertion, ultrasound-guided compression, and ultrasound-guided thrombin injection are alternatives to open surgical repair of larger, uncomplicated PSAs. However, urgent open surgical repair is the only option when there is evidence of a ruptured PSA, ischemia, or skin necrosis.

Phlegmasia cerulea dolens (PCD), a life-threatening complication of deep venous thrombosis (DVT), is characterized by massive iliofemoral thrombus that extends to the collateral veins, leading to fluid sequestration and elevated compartment pressures that ultimately compromise arterial flow. Phlegmasia cerulea dolens can rapidly progress to compartment syndrome and gangrene.^1,2 The affected limbs of patients with PCD can be hypoxic and appear purple in color due to substantial lack of blood flow, with diminished or absent pulses. Risk factors for PCD include malignancy, hypercoagulable states, venous stasis, contraceptive agents, inferior vena cava (IVC) filter, aneurysm, history of DVT, trauma, heparin-induced thrombocytopenia, femoral vein catheterization, antiphospholipid syndrome, or pregnancy.^3-6 Failure to treat PCD early and aggressively carries an amputation rate of up to 50% and a mortality rate of up to 40%.⁴

We present the case of a patient with PCD, whose condition rapidly deteriorated despite prompt diagnosis and treatment.

Case

A 58-year-old woman presented to the ED with a 1-day history of back and leg pain and difficulty walking. When asked about the severity of her pain, she rated her leg pain at 10 on a scale of 0 to 10. The patient’s history was significant for DVT and pulmonary embolism (PE), for which a Greenfield IVC had been placed and for which she was on prophylactic warfarin therapy. The patient stated that she had been taken off warfarin several weeks prior to presentation in preparation for an elective colonoscopy and dental procedure, but had restarted the warfarin therapy 2 days prior to presentation. She had no history of diabetes mellitus or renal disease.

Initial vital signs at presentation were: blood pressure, 120/91 mm Hg; heart rate, 110 beats/min; respiratory rate, 24 breaths/min; and temperature, 96.6°F. Oxygen saturation was 100% on a nonrebreather mask.

On examination, the patient was alert and oriented to person, time, and place, but appeared dyspneic. An electrocardiogram revealed sinus tachycardia. On physical examination, lung sounds were clear to auscultation bilaterally with good air movement, and the abdomen was soft and nontender with normal bowel sounds. The dorsalis pedis and posterior tibial pulses were absent bilaterally, lower extremity capillary refill was 3 seconds, and the legs appeared mildly erythematous and cool to touch. No speech or neurological deficits were present.

Laboratory evaluation was remarkable for metabolic acidosis, venous pH, 7.11; bicarbonate, 11.7; partial pressure of carbon dioxide, 37.6; lactic acid, 8.8 mEq/L leukocytosis, 24,900 u/L; glucose, 296 mg/dL; creatinine, 2.41 mg/dL; and international normalized ratio, 1.36.

Before additional laboratory studies and imaging could be obtained, the patient developed altered mental status, hypotension, and paralysis of the lower extremities. She was orally intubated for airway protection and was given a total of 4 L of normal saline intravenously (IV) for hypotension and acidosis; sodium bicarbonate for metabolic acidosis; norepinephrine for hypotension; fentanyl for pain; and ondansetron for nausea. A central line and arterial line were placed for administering medication and hemodynamic monitoring.

Discussion

This case illustrates the severity and complications of PCD and the rapidity with which this condition can deteriorate. At the time of ED presentation, the patient had already developed bilateral PCD, metabolic acidosis, and bilateral PE. Unfortunately, due to decreased venous return, decreased cardiac output, and severe shock, she quickly became unstable and progressed rapidly to multisystem organ failure leading to death.

Risk Factors

A prior patient history DVT and an IVC filter are both significant risk factors for the progression of DVT to PCD;^3,6 however, in this case, IVC filter failed to prevent emboli from reaching the lungs. Extensive thrombi led to severely decreased venous return and cardiac output, causing life-threatening shock, ischemia, and metabolic acidosis. A lactic acid level taken on hospital day 2 was elevated at 19 mEq/L, demonstrating the severity, morbidity, and progression of PCD.

Signs and Symptoms

The three cardinal signs that lead to a clinical diagnosis of PCD are edema, pain, and violaceous discoloration or skin mottling.³ Although most commonly found in the lower extremity, PCD can occur in any limb due to occlusion of venous outflow.⁷ Unfortunately, a clinical diagnosis of PCD is not often made until the venous occlusion becomes severe enough to impair arterial flow and cause venous gangrene, tissue ischemia, shock, and death.⁸

Although IVC filters are designed to prevent life-threatening PE, there are risk factors associated with their use. Whether placed recently or decades prior, urgent investigation, such as immediate CT scan, should be undertaken in patients presenting with DVT-like symptoms who have a history of an IVC filter, to ensure the filter has not shifted from its original placement and is not occluding the IVC.

Conclusion

Phlegmasia cerulea dolens is an uncommon vascular emergency, but one that has a high-morbidity and high-mortality rate. This case demonstrates the importance of early diagnosis, aggressive treatment, and the severe complications that can develop in PCD.

There are cases in the literature where patients diagnosed with PCD had a successful outcome with pharmacological or surgical intervention such as thrombectomy. Treatment for PCD is most effective when instituted early in onset. As seen in our patient, the tendency for rapid deterioration in PCD can limit potentially lifesaving therapeutic options, decreasing the chances of a successful outcome. Emergency physicians, therefore, must be aware of the high-mortality rate associated with this disorder and the possibility of rapid progression from stable to critical condition.

Phlegmasia cerulea dolens (PCD), a life-threatening complication of deep venous thrombosis (DVT), is characterized by massive iliofemoral thrombus that extends to the collateral veins, leading to fluid sequestration and elevated compartment pressures that ultimately compromise arterial flow. Phlegmasia cerulea dolens can rapidly progress to compartment syndrome and gangrene.^1,2 The affected limbs of patients with PCD can be hypoxic and appear purple in color due to substantial lack of blood flow, with diminished or absent pulses. Risk factors for PCD include malignancy, hypercoagulable states, venous stasis, contraceptive agents, inferior vena cava (IVC) filter, aneurysm, history of DVT, trauma, heparin-induced thrombocytopenia, femoral vein catheterization, antiphospholipid syndrome, or pregnancy.^3-6 Failure to treat PCD early and aggressively carries an amputation rate of up to 50% and a mortality rate of up to 40%.⁴

We present the case of a patient with PCD, whose condition rapidly deteriorated despite prompt diagnosis and treatment.

Case

A 58-year-old woman presented to the ED with a 1-day history of back and leg pain and difficulty walking. When asked about the severity of her pain, she rated her leg pain at 10 on a scale of 0 to 10. The patient’s history was significant for DVT and pulmonary embolism (PE), for which a Greenfield IVC had been placed and for which she was on prophylactic warfarin therapy. The patient stated that she had been taken off warfarin several weeks prior to presentation in preparation for an elective colonoscopy and dental procedure, but had restarted the warfarin therapy 2 days prior to presentation. She had no history of diabetes mellitus or renal disease.

Initial vital signs at presentation were: blood pressure, 120/91 mm Hg; heart rate, 110 beats/min; respiratory rate, 24 breaths/min; and temperature, 96.6°F. Oxygen saturation was 100% on a nonrebreather mask.

On examination, the patient was alert and oriented to person, time, and place, but appeared dyspneic. An electrocardiogram revealed sinus tachycardia. On physical examination, lung sounds were clear to auscultation bilaterally with good air movement, and the abdomen was soft and nontender with normal bowel sounds. The dorsalis pedis and posterior tibial pulses were absent bilaterally, lower extremity capillary refill was 3 seconds, and the legs appeared mildly erythematous and cool to touch. No speech or neurological deficits were present.

Laboratory evaluation was remarkable for metabolic acidosis, venous pH, 7.11; bicarbonate, 11.7; partial pressure of carbon dioxide, 37.6; lactic acid, 8.8 mEq/L leukocytosis, 24,900 u/L; glucose, 296 mg/dL; creatinine, 2.41 mg/dL; and international normalized ratio, 1.36.

Before additional laboratory studies and imaging could be obtained, the patient developed altered mental status, hypotension, and paralysis of the lower extremities. She was orally intubated for airway protection and was given a total of 4 L of normal saline intravenously (IV) for hypotension and acidosis; sodium bicarbonate for metabolic acidosis; norepinephrine for hypotension; fentanyl for pain; and ondansetron for nausea. A central line and arterial line were placed for administering medication and hemodynamic monitoring.

Discussion

This case illustrates the severity and complications of PCD and the rapidity with which this condition can deteriorate. At the time of ED presentation, the patient had already developed bilateral PCD, metabolic acidosis, and bilateral PE. Unfortunately, due to decreased venous return, decreased cardiac output, and severe shock, she quickly became unstable and progressed rapidly to multisystem organ failure leading to death.

Risk Factors

A prior patient history DVT and an IVC filter are both significant risk factors for the progression of DVT to PCD;^3,6 however, in this case, IVC filter failed to prevent emboli from reaching the lungs. Extensive thrombi led to severely decreased venous return and cardiac output, causing life-threatening shock, ischemia, and metabolic acidosis. A lactic acid level taken on hospital day 2 was elevated at 19 mEq/L, demonstrating the severity, morbidity, and progression of PCD.

Signs and Symptoms

The three cardinal signs that lead to a clinical diagnosis of PCD are edema, pain, and violaceous discoloration or skin mottling.³ Although most commonly found in the lower extremity, PCD can occur in any limb due to occlusion of venous outflow.⁷ Unfortunately, a clinical diagnosis of PCD is not often made until the venous occlusion becomes severe enough to impair arterial flow and cause venous gangrene, tissue ischemia, shock, and death.⁸

Although IVC filters are designed to prevent life-threatening PE, there are risk factors associated with their use. Whether placed recently or decades prior, urgent investigation, such as immediate CT scan, should be undertaken in patients presenting with DVT-like symptoms who have a history of an IVC filter, to ensure the filter has not shifted from its original placement and is not occluding the IVC.

Conclusion

Phlegmasia cerulea dolens is an uncommon vascular emergency, but one that has a high-morbidity and high-mortality rate. This case demonstrates the importance of early diagnosis, aggressive treatment, and the severe complications that can develop in PCD.

There are cases in the literature where patients diagnosed with PCD had a successful outcome with pharmacological or surgical intervention such as thrombectomy. Treatment for PCD is most effective when instituted early in onset. As seen in our patient, the tendency for rapid deterioration in PCD can limit potentially lifesaving therapeutic options, decreasing the chances of a successful outcome. Emergency physicians, therefore, must be aware of the high-mortality rate associated with this disorder and the possibility of rapid progression from stable to critical condition.

Phlegmasia cerulea dolens (PCD), a life-threatening complication of deep venous thrombosis (DVT), is characterized by massive iliofemoral thrombus that extends to the collateral veins, leading to fluid sequestration and elevated compartment pressures that ultimately compromise arterial flow. Phlegmasia cerulea dolens can rapidly progress to compartment syndrome and gangrene.^1,2 The affected limbs of patients with PCD can be hypoxic and appear purple in color due to substantial lack of blood flow, with diminished or absent pulses. Risk factors for PCD include malignancy, hypercoagulable states, venous stasis, contraceptive agents, inferior vena cava (IVC) filter, aneurysm, history of DVT, trauma, heparin-induced thrombocytopenia, femoral vein catheterization, antiphospholipid syndrome, or pregnancy.^3-6 Failure to treat PCD early and aggressively carries an amputation rate of up to 50% and a mortality rate of up to 40%.⁴

We present the case of a patient with PCD, whose condition rapidly deteriorated despite prompt diagnosis and treatment.

Case

A 58-year-old woman presented to the ED with a 1-day history of back and leg pain and difficulty walking. When asked about the severity of her pain, she rated her leg pain at 10 on a scale of 0 to 10. The patient’s history was significant for DVT and pulmonary embolism (PE), for which a Greenfield IVC had been placed and for which she was on prophylactic warfarin therapy. The patient stated that she had been taken off warfarin several weeks prior to presentation in preparation for an elective colonoscopy and dental procedure, but had restarted the warfarin therapy 2 days prior to presentation. She had no history of diabetes mellitus or renal disease.

Initial vital signs at presentation were: blood pressure, 120/91 mm Hg; heart rate, 110 beats/min; respiratory rate, 24 breaths/min; and temperature, 96.6°F. Oxygen saturation was 100% on a nonrebreather mask.

On examination, the patient was alert and oriented to person, time, and place, but appeared dyspneic. An electrocardiogram revealed sinus tachycardia. On physical examination, lung sounds were clear to auscultation bilaterally with good air movement, and the abdomen was soft and nontender with normal bowel sounds. The dorsalis pedis and posterior tibial pulses were absent bilaterally, lower extremity capillary refill was 3 seconds, and the legs appeared mildly erythematous and cool to touch. No speech or neurological deficits were present.

Laboratory evaluation was remarkable for metabolic acidosis, venous pH, 7.11; bicarbonate, 11.7; partial pressure of carbon dioxide, 37.6; lactic acid, 8.8 mEq/L leukocytosis, 24,900 u/L; glucose, 296 mg/dL; creatinine, 2.41 mg/dL; and international normalized ratio, 1.36.

Before additional laboratory studies and imaging could be obtained, the patient developed altered mental status, hypotension, and paralysis of the lower extremities. She was orally intubated for airway protection and was given a total of 4 L of normal saline intravenously (IV) for hypotension and acidosis; sodium bicarbonate for metabolic acidosis; norepinephrine for hypotension; fentanyl for pain; and ondansetron for nausea. A central line and arterial line were placed for administering medication and hemodynamic monitoring.

Discussion

This case illustrates the severity and complications of PCD and the rapidity with which this condition can deteriorate. At the time of ED presentation, the patient had already developed bilateral PCD, metabolic acidosis, and bilateral PE. Unfortunately, due to decreased venous return, decreased cardiac output, and severe shock, she quickly became unstable and progressed rapidly to multisystem organ failure leading to death.

Risk Factors

A prior patient history DVT and an IVC filter are both significant risk factors for the progression of DVT to PCD;^3,6 however, in this case, IVC filter failed to prevent emboli from reaching the lungs. Extensive thrombi led to severely decreased venous return and cardiac output, causing life-threatening shock, ischemia, and metabolic acidosis. A lactic acid level taken on hospital day 2 was elevated at 19 mEq/L, demonstrating the severity, morbidity, and progression of PCD.

Signs and Symptoms

The three cardinal signs that lead to a clinical diagnosis of PCD are edema, pain, and violaceous discoloration or skin mottling.³ Although most commonly found in the lower extremity, PCD can occur in any limb due to occlusion of venous outflow.⁷ Unfortunately, a clinical diagnosis of PCD is not often made until the venous occlusion becomes severe enough to impair arterial flow and cause venous gangrene, tissue ischemia, shock, and death.⁸

Although IVC filters are designed to prevent life-threatening PE, there are risk factors associated with their use. Whether placed recently or decades prior, urgent investigation, such as immediate CT scan, should be undertaken in patients presenting with DVT-like symptoms who have a history of an IVC filter, to ensure the filter has not shifted from its original placement and is not occluding the IVC.

Conclusion

Phlegmasia cerulea dolens is an uncommon vascular emergency, but one that has a high-morbidity and high-mortality rate. This case demonstrates the importance of early diagnosis, aggressive treatment, and the severe complications that can develop in PCD.

There are cases in the literature where patients diagnosed with PCD had a successful outcome with pharmacological or surgical intervention such as thrombectomy. Treatment for PCD is most effective when instituted early in onset. As seen in our patient, the tendency for rapid deterioration in PCD can limit potentially lifesaving therapeutic options, decreasing the chances of a successful outcome. Emergency physicians, therefore, must be aware of the high-mortality rate associated with this disorder and the possibility of rapid progression from stable to critical condition.

Facial paralysis is a common medical complaint—one that has fascinated ancient and contemporary physicians alike.¹ An idiopathic facial nerve paresis involving the lower motor neuron was described in 1821 by Sir Charles Bell. This entity became known as a Bell’s palsy, the hallmark of which was weakness or complete paralysis of the muscles of one side of the face, with no sparing of the muscles of the forehead. However, not all facial paralysis is due to Bell’s palsy.

We present a case of a patient with a Bell’s palsy mimic to facilitate and guide the differential diagnosis and distinguish conditions from the classical presentation that Bell first described to the more concerning symptoms that may not be immediately obvious. Our case further underscores the importance of performing a thorough assessment to determine the presence of other neurological findings.

Case

A 61-year-old woman presented to the ED for evaluation of right facial droop and sensation of “room spinning.” The patient stated both symptoms began approximately 36 hours prior to presentation, upon awakening. She further noted that the right side of her face felt “funny” and numb.

The patient denied any headache, neck or chest pain, extremity numbness, or weakness, but stated that she felt like she was going to fall toward her right side whenever she attempted to walk. The patient’s medical history was significant for hypertension, for which she was taking losartan. Her surgical history was notable for a left oophorectomy secondary to an ovarian cyst. Regarding the social history, the patient admitted to smoking 90 packs of cigarettes per year, but denied alcohol or illicit drug use.

Upon arrival at the ED, the patient’s vital signs were: blood pressure, 164/86 mm Hg: pulse, 89 beats/min; respiratory rate, 18 breaths/min; and temperature, 98.6°F. Oxygen saturation was 98% on room air.

Physical examination revealed the patient had a right facial droop consistent with right facial palsy. She was unable to wrinkle her right forehead or fully close her right eye. There were no field cuts on confrontation. The patient’s speech was noticeable for a mild dysarthria. The motor examination revealed mild weakness of the left upper extremity and impaired right facial sensation. There were no rashes noted on the face, head, or ears. The patient had slightly impaired hearing in the right ear, which was new in onset. The remainder of the physical examination was unremarkable.

Although the patient exhibited the classic signs of Bell’s palsy, including complete paralysis of the muscles of one side of the face, inability to wrinkle the muscle of the right forehead, and inability to fully close the right eye, she also had concerning symptoms of vertigo, dysarthria, and contralateral upper extremity weakness.

A computed tomography (CT) scan of the head was ordered, which revealed a large mass lesion centered in the right petrous apex, with an associated large component extending medially into the right cerebellopontine angle (CPA) that caused a mass effect on the adjacent brainstem (Figures 1a and 1b).

Discussion

Accounts of facial paralysis and facial nerve disorders have been noted throughout history and include accounts of the condition by Hippocrates.¹ Bell’s palsy was named after surgeon Sir Charles Bell, who described a peripheral-nerve paralysis of the facial nerve in 1821. Bell’s work helped to elucidate the anatomy and functional role of the facial nerve.^1,2

Signs and Symptoms

The classic presentation of Bell’s palsy is weakness or complete paralysis of the muscles of one side of the face, with no sparing of the muscles of the forehead. The eyelid on the affected side generally does not close, which can result in ocular irritation due to ineffective lubrication.

A scoring system has been developed by House and Brackmann which grades the degree impairment based on such characteristics as facial muscle function and eye closure.^3,4 Approximately 96% of patients with a Bell’s palsy will improve to a House-Brackmann score of 2 or better within 1 year from diagnosis,⁵ and 85% of patients with Bell’s palsy will show at least some improvement within 3 weeks of onset (Table).² Although the classic description of Bell’s palsy notes the condition as idiopathic, there is an increasing body of evidence in the literature showing a link to herpes simplex virus 1.^5-7

Ramsey-Hunt Syndrome

The relationship between Bell’s palsy and Ramsey-Hunt syndrome is complex and controversial. Ramsey-Hunt syndrome is a constellation of possible complications from varicella-virus infection. Symptoms of Ramsey-Hunt syndrome include facial paralysis, tinnitus, hearing loss, vertigo, hyperacusis (increased sensitivity to certain frequencies and volume ranges of sound), and decreased ocular tearing.⁸ Due to the nature of symptoms associated with Ramsey-Hunt syndrome, it is apparent that the condition involves more than the seventh cranial nerve. In fact, studies have shown that Ramsey-Hunt syndrome can affect the fifth, sixth, eighth, and ninth cranial nerves.⁸

Ramsey-Hunt syndrome, which can present in the absence of cutaneous rash (referred to as zoster sine herpete), is estimated to occur in 8% to 20% of unilateral facial nerve palsies in adult patients.^8,9 Regardless of the etiology of Bell’s palsy, a review of the literature makes it clear that facial nerve paralysis is not synonymous with Bell’s palsy.¹⁰ In one example, Yetter et al¹⁰ describe the case of a patient who, though initially diagnosed with Bell’s palsy, ultimately was found to have a facial palsy due to a parotid gland malignancy.

Likewise, Stomeo¹¹ describes a case of a patient with facial paralysis and profound ipsilateral hearing loss who ultimately was found to have a mucoepithelial carcinoma of the parotid gland. In their report, the authors note that approximately 80% of facial nerve paralysis is due to Bell’s palsy, while 5% is due to malignancy.

In another report, Clemis¹² describes a case in which a patient who initially was diagnosed with Bell’s palsy eventually was found to have an adenoid cystic carcinoma of the parotid. Thus, the authors appropriately emphasize in their report that “all that palsies is not Bell’s.”

Differential Diagnosis

Historical factors, including timing and duration of symptom onset, help to distinguish a Bell’s palsy from other disorders that can mimic this condition. In their study, Brach VanSwewaringen¹³ highlight the fact that “not all facial paralysis is Bell’s palsy.” In their review, the authors describe clues to help distinguish conditions that mimic Bell’s palsy. For example, maximal weakness from Bell’s Palsy typically occurs within 3 to 7 days from symptom onset, and that a more gradual onset of symptoms, with slow or negligible improvement over 6 to 12 months, is more indicative of a space-occupying lesion than Bell’s palsy.¹³It is, however, important to note that although the patient in our case had a central lesion, she experienced an acute onset of symptoms.

The presence of additional symptoms may also suggest an alternative diagnosis. Brach and VanSwearingen¹³ further noted that symptoms associated with the eighth nerve, such as vertigo, tinnitus, and hearing loss may be found in patients with a CPA tumor. In patients with larger tumors, ninth and 10th nerve symptoms, including the impaired hearing noted in our patient, may be present. Some patients with ninth and 10th nerve symptoms may perceive a sense of facial numbness, but actual sensory changes in the facial nerve distribution are unlikely in Bell’s palsy. Gustatory changes, however, are consistent with Bell’s palsy.

Ear pain is consistent with Bell’s palsy and is a signal to be vigilant for the possible emergence of an ear rash, which would suggest the diagnosis of herpes zoster oticus along the trajectory of Ramsey-Hunt syndrome. Facial pain in the area of the facial nerve is inconsistent with Bell’s palsy, while hyperacusis is consistent with Bell’s palsy. Hearing loss is an eighth nerve symptom that is inconsistent with Bell’s palsy.

Similarly, there are physical examination findings that can help distinguish a true Bell’s palsy from a mimic. Changes in tear production are consistent with Bell’s palsy, but imbalance and disequilibrium are not.¹⁴

As previously noted, the patient in this case had difficulty walking and felt as if she was falling toward her right side.

One way to organize the causes of facial paralysis has been proposed by Adour et al.¹⁵ In this system, etiologies are listed as either acute paralysis or chronic, progressive paralysis. Acute paralysis (ie, the sudden onset of symptoms with maximal severity within 2 weeks), of which Bell’s palsy is the most common, can be seen in cases of polyneuritis.

A new case of Bell’s palsy has been estimated to occur in the United States every 10 minutes.⁸ Guillain-Barré syndrome and Lyme disease are also in this category, as is Ramsey-Hunt syndrome. Patients with Lyme disease may have a history of a tick bite or rash.¹⁴

Trauma can also cause acute facial nerve paralysis (eg, blunt trauma-associated facial fracture, penetrating trauma, birth trauma). Unilateral central facial weakness can have a neurological cause, such as a lesion to the contralateral cortex, subcortical white matter, or internal capsule.^2,15 Otitis media can sometimes cause facial paralysis.¹⁶ A cholesteatoma can cause acute facial paralysis.² Malignancies cause 5% of all cases of facial paralysis. Primary parotid tumors of various types are in this category. Metastatic disease from breast, lung, skin, colon, and kidney may cause facial paralysis. As our case illustrates, CPA tumors can cause facial paralysis.¹⁵ It is important to also note that a patient can have both a Bell’s palsy and a concurrent disease. There are a number of case reports in the literature that describe acute onset of facial paralysis as a presenting symptom of malignancy.¹⁷ In addition, there are cases wherein a neurological finding on imaging, such as an acoustic neuroma, was presumed to be the cause of facial paralysis, yet the patient’s symptoms resolved in a manner consistent with Bell’s palsy.¹⁸

For example, Lagman et al¹⁹ described a patient in which a CPA lipoma was presumed to be the cause of the facial paralysis, but the eventual outcome showed the lipoma to have been an incidentaloma.

Conclusion

This case demonstrates a presenting symptom of facial palsy and the presence of a CPA tumor. The presence of vertigo along with other historical and physical examination findings inconsistent with Bell’s palsy prompted the CT scan of the head. A review of the literature suggests a number of important findings in patients with facial palsy to assist the clinician in distinguishing true Bell’s palsy from other diseases that can mimic this condition. This case serves as a reminder of the need to perform a thorough and diligent workup to determine the presence or absence of other neurologic findings prior to closing on the diagnosis of Bell’s palsy.

Facial paralysis is a common medical complaint—one that has fascinated ancient and contemporary physicians alike.¹ An idiopathic facial nerve paresis involving the lower motor neuron was described in 1821 by Sir Charles Bell. This entity became known as a Bell’s palsy, the hallmark of which was weakness or complete paralysis of the muscles of one side of the face, with no sparing of the muscles of the forehead. However, not all facial paralysis is due to Bell’s palsy.

We present a case of a patient with a Bell’s palsy mimic to facilitate and guide the differential diagnosis and distinguish conditions from the classical presentation that Bell first described to the more concerning symptoms that may not be immediately obvious. Our case further underscores the importance of performing a thorough assessment to determine the presence of other neurological findings.

Case

A 61-year-old woman presented to the ED for evaluation of right facial droop and sensation of “room spinning.” The patient stated both symptoms began approximately 36 hours prior to presentation, upon awakening. She further noted that the right side of her face felt “funny” and numb.

The patient denied any headache, neck or chest pain, extremity numbness, or weakness, but stated that she felt like she was going to fall toward her right side whenever she attempted to walk. The patient’s medical history was significant for hypertension, for which she was taking losartan. Her surgical history was notable for a left oophorectomy secondary to an ovarian cyst. Regarding the social history, the patient admitted to smoking 90 packs of cigarettes per year, but denied alcohol or illicit drug use.

Upon arrival at the ED, the patient’s vital signs were: blood pressure, 164/86 mm Hg: pulse, 89 beats/min; respiratory rate, 18 breaths/min; and temperature, 98.6°F. Oxygen saturation was 98% on room air.

Physical examination revealed the patient had a right facial droop consistent with right facial palsy. She was unable to wrinkle her right forehead or fully close her right eye. There were no field cuts on confrontation. The patient’s speech was noticeable for a mild dysarthria. The motor examination revealed mild weakness of the left upper extremity and impaired right facial sensation. There were no rashes noted on the face, head, or ears. The patient had slightly impaired hearing in the right ear, which was new in onset. The remainder of the physical examination was unremarkable.

Although the patient exhibited the classic signs of Bell’s palsy, including complete paralysis of the muscles of one side of the face, inability to wrinkle the muscle of the right forehead, and inability to fully close the right eye, she also had concerning symptoms of vertigo, dysarthria, and contralateral upper extremity weakness.

A computed tomography (CT) scan of the head was ordered, which revealed a large mass lesion centered in the right petrous apex, with an associated large component extending medially into the right cerebellopontine angle (CPA) that caused a mass effect on the adjacent brainstem (Figures 1a and 1b).

Discussion

Accounts of facial paralysis and facial nerve disorders have been noted throughout history and include accounts of the condition by Hippocrates.¹ Bell’s palsy was named after surgeon Sir Charles Bell, who described a peripheral-nerve paralysis of the facial nerve in 1821. Bell’s work helped to elucidate the anatomy and functional role of the facial nerve.^1,2

Signs and Symptoms

The classic presentation of Bell’s palsy is weakness or complete paralysis of the muscles of one side of the face, with no sparing of the muscles of the forehead. The eyelid on the affected side generally does not close, which can result in ocular irritation due to ineffective lubrication.

A scoring system has been developed by House and Brackmann which grades the degree impairment based on such characteristics as facial muscle function and eye closure.^3,4 Approximately 96% of patients with a Bell’s palsy will improve to a House-Brackmann score of 2 or better within 1 year from diagnosis,⁵ and 85% of patients with Bell’s palsy will show at least some improvement within 3 weeks of onset (Table).² Although the classic description of Bell’s palsy notes the condition as idiopathic, there is an increasing body of evidence in the literature showing a link to herpes simplex virus 1.^5-7

Ramsey-Hunt Syndrome

The relationship between Bell’s palsy and Ramsey-Hunt syndrome is complex and controversial. Ramsey-Hunt syndrome is a constellation of possible complications from varicella-virus infection. Symptoms of Ramsey-Hunt syndrome include facial paralysis, tinnitus, hearing loss, vertigo, hyperacusis (increased sensitivity to certain frequencies and volume ranges of sound), and decreased ocular tearing.⁸ Due to the nature of symptoms associated with Ramsey-Hunt syndrome, it is apparent that the condition involves more than the seventh cranial nerve. In fact, studies have shown that Ramsey-Hunt syndrome can affect the fifth, sixth, eighth, and ninth cranial nerves.⁸

Ramsey-Hunt syndrome, which can present in the absence of cutaneous rash (referred to as zoster sine herpete), is estimated to occur in 8% to 20% of unilateral facial nerve palsies in adult patients.^8,9 Regardless of the etiology of Bell’s palsy, a review of the literature makes it clear that facial nerve paralysis is not synonymous with Bell’s palsy.¹⁰ In one example, Yetter et al¹⁰ describe the case of a patient who, though initially diagnosed with Bell’s palsy, ultimately was found to have a facial palsy due to a parotid gland malignancy.

Likewise, Stomeo¹¹ describes a case of a patient with facial paralysis and profound ipsilateral hearing loss who ultimately was found to have a mucoepithelial carcinoma of the parotid gland. In their report, the authors note that approximately 80% of facial nerve paralysis is due to Bell’s palsy, while 5% is due to malignancy.

In another report, Clemis¹² describes a case in which a patient who initially was diagnosed with Bell’s palsy eventually was found to have an adenoid cystic carcinoma of the parotid. Thus, the authors appropriately emphasize in their report that “all that palsies is not Bell’s.”

Differential Diagnosis

Historical factors, including timing and duration of symptom onset, help to distinguish a Bell’s palsy from other disorders that can mimic this condition. In their study, Brach VanSwewaringen¹³ highlight the fact that “not all facial paralysis is Bell’s palsy.” In their review, the authors describe clues to help distinguish conditions that mimic Bell’s palsy. For example, maximal weakness from Bell’s Palsy typically occurs within 3 to 7 days from symptom onset, and that a more gradual onset of symptoms, with slow or negligible improvement over 6 to 12 months, is more indicative of a space-occupying lesion than Bell’s palsy.¹³It is, however, important to note that although the patient in our case had a central lesion, she experienced an acute onset of symptoms.

The presence of additional symptoms may also suggest an alternative diagnosis. Brach and VanSwearingen¹³ further noted that symptoms associated with the eighth nerve, such as vertigo, tinnitus, and hearing loss may be found in patients with a CPA tumor. In patients with larger tumors, ninth and 10th nerve symptoms, including the impaired hearing noted in our patient, may be present. Some patients with ninth and 10th nerve symptoms may perceive a sense of facial numbness, but actual sensory changes in the facial nerve distribution are unlikely in Bell’s palsy. Gustatory changes, however, are consistent with Bell’s palsy.

Ear pain is consistent with Bell’s palsy and is a signal to be vigilant for the possible emergence of an ear rash, which would suggest the diagnosis of herpes zoster oticus along the trajectory of Ramsey-Hunt syndrome. Facial pain in the area of the facial nerve is inconsistent with Bell’s palsy, while hyperacusis is consistent with Bell’s palsy. Hearing loss is an eighth nerve symptom that is inconsistent with Bell’s palsy.

Similarly, there are physical examination findings that can help distinguish a true Bell’s palsy from a mimic. Changes in tear production are consistent with Bell’s palsy, but imbalance and disequilibrium are not.¹⁴

As previously noted, the patient in this case had difficulty walking and felt as if she was falling toward her right side.

One way to organize the causes of facial paralysis has been proposed by Adour et al.¹⁵ In this system, etiologies are listed as either acute paralysis or chronic, progressive paralysis. Acute paralysis (ie, the sudden onset of symptoms with maximal severity within 2 weeks), of which Bell’s palsy is the most common, can be seen in cases of polyneuritis.

A new case of Bell’s palsy has been estimated to occur in the United States every 10 minutes.⁸ Guillain-Barré syndrome and Lyme disease are also in this category, as is Ramsey-Hunt syndrome. Patients with Lyme disease may have a history of a tick bite or rash.¹⁴

Trauma can also cause acute facial nerve paralysis (eg, blunt trauma-associated facial fracture, penetrating trauma, birth trauma). Unilateral central facial weakness can have a neurological cause, such as a lesion to the contralateral cortex, subcortical white matter, or internal capsule.^2,15 Otitis media can sometimes cause facial paralysis.¹⁶ A cholesteatoma can cause acute facial paralysis.² Malignancies cause 5% of all cases of facial paralysis. Primary parotid tumors of various types are in this category. Metastatic disease from breast, lung, skin, colon, and kidney may cause facial paralysis. As our case illustrates, CPA tumors can cause facial paralysis.¹⁵ It is important to also note that a patient can have both a Bell’s palsy and a concurrent disease. There are a number of case reports in the literature that describe acute onset of facial paralysis as a presenting symptom of malignancy.¹⁷ In addition, there are cases wherein a neurological finding on imaging, such as an acoustic neuroma, was presumed to be the cause of facial paralysis, yet the patient’s symptoms resolved in a manner consistent with Bell’s palsy.¹⁸

For example, Lagman et al¹⁹ described a patient in which a CPA lipoma was presumed to be the cause of the facial paralysis, but the eventual outcome showed the lipoma to have been an incidentaloma.

Conclusion

This case demonstrates a presenting symptom of facial palsy and the presence of a CPA tumor. The presence of vertigo along with other historical and physical examination findings inconsistent with Bell’s palsy prompted the CT scan of the head. A review of the literature suggests a number of important findings in patients with facial palsy to assist the clinician in distinguishing true Bell’s palsy from other diseases that can mimic this condition. This case serves as a reminder of the need to perform a thorough and diligent workup to determine the presence or absence of other neurologic findings prior to closing on the diagnosis of Bell’s palsy.

Facial paralysis is a common medical complaint—one that has fascinated ancient and contemporary physicians alike.¹ An idiopathic facial nerve paresis involving the lower motor neuron was described in 1821 by Sir Charles Bell. This entity became known as a Bell’s palsy, the hallmark of which was weakness or complete paralysis of the muscles of one side of the face, with no sparing of the muscles of the forehead. However, not all facial paralysis is due to Bell’s palsy.

We present a case of a patient with a Bell’s palsy mimic to facilitate and guide the differential diagnosis and distinguish conditions from the classical presentation that Bell first described to the more concerning symptoms that may not be immediately obvious. Our case further underscores the importance of performing a thorough assessment to determine the presence of other neurological findings.

Case

A 61-year-old woman presented to the ED for evaluation of right facial droop and sensation of “room spinning.” The patient stated both symptoms began approximately 36 hours prior to presentation, upon awakening. She further noted that the right side of her face felt “funny” and numb.

The patient denied any headache, neck or chest pain, extremity numbness, or weakness, but stated that she felt like she was going to fall toward her right side whenever she attempted to walk. The patient’s medical history was significant for hypertension, for which she was taking losartan. Her surgical history was notable for a left oophorectomy secondary to an ovarian cyst. Regarding the social history, the patient admitted to smoking 90 packs of cigarettes per year, but denied alcohol or illicit drug use.

Upon arrival at the ED, the patient’s vital signs were: blood pressure, 164/86 mm Hg: pulse, 89 beats/min; respiratory rate, 18 breaths/min; and temperature, 98.6°F. Oxygen saturation was 98% on room air.

Physical examination revealed the patient had a right facial droop consistent with right facial palsy. She was unable to wrinkle her right forehead or fully close her right eye. There were no field cuts on confrontation. The patient’s speech was noticeable for a mild dysarthria. The motor examination revealed mild weakness of the left upper extremity and impaired right facial sensation. There were no rashes noted on the face, head, or ears. The patient had slightly impaired hearing in the right ear, which was new in onset. The remainder of the physical examination was unremarkable.

Although the patient exhibited the classic signs of Bell’s palsy, including complete paralysis of the muscles of one side of the face, inability to wrinkle the muscle of the right forehead, and inability to fully close the right eye, she also had concerning symptoms of vertigo, dysarthria, and contralateral upper extremity weakness.

A computed tomography (CT) scan of the head was ordered, which revealed a large mass lesion centered in the right petrous apex, with an associated large component extending medially into the right cerebellopontine angle (CPA) that caused a mass effect on the adjacent brainstem (Figures 1a and 1b).

Discussion

Accounts of facial paralysis and facial nerve disorders have been noted throughout history and include accounts of the condition by Hippocrates.¹ Bell’s palsy was named after surgeon Sir Charles Bell, who described a peripheral-nerve paralysis of the facial nerve in 1821. Bell’s work helped to elucidate the anatomy and functional role of the facial nerve.^1,2

Signs and Symptoms

The classic presentation of Bell’s palsy is weakness or complete paralysis of the muscles of one side of the face, with no sparing of the muscles of the forehead. The eyelid on the affected side generally does not close, which can result in ocular irritation due to ineffective lubrication.

A scoring system has been developed by House and Brackmann which grades the degree impairment based on such characteristics as facial muscle function and eye closure.^3,4 Approximately 96% of patients with a Bell’s palsy will improve to a House-Brackmann score of 2 or better within 1 year from diagnosis,⁵ and 85% of patients with Bell’s palsy will show at least some improvement within 3 weeks of onset (Table).² Although the classic description of Bell’s palsy notes the condition as idiopathic, there is an increasing body of evidence in the literature showing a link to herpes simplex virus 1.^5-7

Ramsey-Hunt Syndrome

The relationship between Bell’s palsy and Ramsey-Hunt syndrome is complex and controversial. Ramsey-Hunt syndrome is a constellation of possible complications from varicella-virus infection. Symptoms of Ramsey-Hunt syndrome include facial paralysis, tinnitus, hearing loss, vertigo, hyperacusis (increased sensitivity to certain frequencies and volume ranges of sound), and decreased ocular tearing.⁸ Due to the nature of symptoms associated with Ramsey-Hunt syndrome, it is apparent that the condition involves more than the seventh cranial nerve. In fact, studies have shown that Ramsey-Hunt syndrome can affect the fifth, sixth, eighth, and ninth cranial nerves.⁸

Ramsey-Hunt syndrome, which can present in the absence of cutaneous rash (referred to as zoster sine herpete), is estimated to occur in 8% to 20% of unilateral facial nerve palsies in adult patients.^8,9 Regardless of the etiology of Bell’s palsy, a review of the literature makes it clear that facial nerve paralysis is not synonymous with Bell’s palsy.¹⁰ In one example, Yetter et al¹⁰ describe the case of a patient who, though initially diagnosed with Bell’s palsy, ultimately was found to have a facial palsy due to a parotid gland malignancy.

Likewise, Stomeo¹¹ describes a case of a patient with facial paralysis and profound ipsilateral hearing loss who ultimately was found to have a mucoepithelial carcinoma of the parotid gland. In their report, the authors note that approximately 80% of facial nerve paralysis is due to Bell’s palsy, while 5% is due to malignancy.

In another report, Clemis¹² describes a case in which a patient who initially was diagnosed with Bell’s palsy eventually was found to have an adenoid cystic carcinoma of the parotid. Thus, the authors appropriately emphasize in their report that “all that palsies is not Bell’s.”

Differential Diagnosis

Historical factors, including timing and duration of symptom onset, help to distinguish a Bell’s palsy from other disorders that can mimic this condition. In their study, Brach VanSwewaringen¹³ highlight the fact that “not all facial paralysis is Bell’s palsy.” In their review, the authors describe clues to help distinguish conditions that mimic Bell’s palsy. For example, maximal weakness from Bell’s Palsy typically occurs within 3 to 7 days from symptom onset, and that a more gradual onset of symptoms, with slow or negligible improvement over 6 to 12 months, is more indicative of a space-occupying lesion than Bell’s palsy.¹³It is, however, important to note that although the patient in our case had a central lesion, she experienced an acute onset of symptoms.

The presence of additional symptoms may also suggest an alternative diagnosis. Brach and VanSwearingen¹³ further noted that symptoms associated with the eighth nerve, such as vertigo, tinnitus, and hearing loss may be found in patients with a CPA tumor. In patients with larger tumors, ninth and 10th nerve symptoms, including the impaired hearing noted in our patient, may be present. Some patients with ninth and 10th nerve symptoms may perceive a sense of facial numbness, but actual sensory changes in the facial nerve distribution are unlikely in Bell’s palsy. Gustatory changes, however, are consistent with Bell’s palsy.

Ear pain is consistent with Bell’s palsy and is a signal to be vigilant for the possible emergence of an ear rash, which would suggest the diagnosis of herpes zoster oticus along the trajectory of Ramsey-Hunt syndrome. Facial pain in the area of the facial nerve is inconsistent with Bell’s palsy, while hyperacusis is consistent with Bell’s palsy. Hearing loss is an eighth nerve symptom that is inconsistent with Bell’s palsy.

Similarly, there are physical examination findings that can help distinguish a true Bell’s palsy from a mimic. Changes in tear production are consistent with Bell’s palsy, but imbalance and disequilibrium are not.¹⁴

As previously noted, the patient in this case had difficulty walking and felt as if she was falling toward her right side.

One way to organize the causes of facial paralysis has been proposed by Adour et al.¹⁵ In this system, etiologies are listed as either acute paralysis or chronic, progressive paralysis. Acute paralysis (ie, the sudden onset of symptoms with maximal severity within 2 weeks), of which Bell’s palsy is the most common, can be seen in cases of polyneuritis.

A new case of Bell’s palsy has been estimated to occur in the United States every 10 minutes.⁸ Guillain-Barré syndrome and Lyme disease are also in this category, as is Ramsey-Hunt syndrome. Patients with Lyme disease may have a history of a tick bite or rash.¹⁴

Trauma can also cause acute facial nerve paralysis (eg, blunt trauma-associated facial fracture, penetrating trauma, birth trauma). Unilateral central facial weakness can have a neurological cause, such as a lesion to the contralateral cortex, subcortical white matter, or internal capsule.^2,15 Otitis media can sometimes cause facial paralysis.¹⁶ A cholesteatoma can cause acute facial paralysis.² Malignancies cause 5% of all cases of facial paralysis. Primary parotid tumors of various types are in this category. Metastatic disease from breast, lung, skin, colon, and kidney may cause facial paralysis. As our case illustrates, CPA tumors can cause facial paralysis.¹⁵ It is important to also note that a patient can have both a Bell’s palsy and a concurrent disease. There are a number of case reports in the literature that describe acute onset of facial paralysis as a presenting symptom of malignancy.¹⁷ In addition, there are cases wherein a neurological finding on imaging, such as an acoustic neuroma, was presumed to be the cause of facial paralysis, yet the patient’s symptoms resolved in a manner consistent with Bell’s palsy.¹⁸

For example, Lagman et al¹⁹ described a patient in which a CPA lipoma was presumed to be the cause of the facial paralysis, but the eventual outcome showed the lipoma to have been an incidentaloma.

Conclusion

This case demonstrates a presenting symptom of facial palsy and the presence of a CPA tumor. The presence of vertigo along with other historical and physical examination findings inconsistent with Bell’s palsy prompted the CT scan of the head. A review of the literature suggests a number of important findings in patients with facial palsy to assist the clinician in distinguishing true Bell’s palsy from other diseases that can mimic this condition. This case serves as a reminder of the need to perform a thorough and diligent workup to determine the presence or absence of other neurologic findings prior to closing on the diagnosis of Bell’s palsy.

ABSTRACT

Soft tissue defects associated with exposed tendon pose difficult reconstructive problems because of tendon adhesions, poor range of motion, poor cosmetic appearance, and donor site morbidity. Dermal regeneration template is a skin substitute widely used in reconstructive surgery, including the occasional coverage of tendons. However, postoperative functionality of the tendons has not been well documented. We report a case of using dermal regeneration template for soft tissue reconstruction overlying tendons with loss of paratenon in a patient with Dupuytren’s contracture. Dermal regeneration template may offer an alternative option for immediate tendon coverage in the hand.

Soft tissue defects overlying exposed tendon with loss of paratenon often precipitate poor clinical outcomes because of the dichotomous demands of both closing the overlying soft-tissue defect and providing a gliding surface for the underlying tendons.¹ Although avoidance of adhesions and restoration of function are the primary goals of the procedure, satisfactory appearance is also desirable. Likewise, any form of coverage should ideally provide good vasculature required for complete healing and an early form of closure following débridement.² Simple skin grafts do not adequately meet these demands because they result in a high rate of tendon adhesions,³ and also are limited in patients with limited donor skin availability or questionable underlying wound bed viability, such as in scleroderma.

In order to reduce the frequency of tendon adhesions by creating a gliding surface, the use of interpositional materials, both artificial and biologic, has been employed with varying degrees of success, including cellophane, chitosan membrane, fibrin sealant, autogenous fascial flaps, and autogenous venous grafts.^4-7 Many of the autogenous flaps and grafts have been employed with good success.⁸ However, complications and donor site morbidity encourage alternative procedures, including the use of artificial substances.^2,8-10

We present our clinical experience with a patient who underwent successful placement of Integra (Integra LifeSciences) Dermal Regeneration Template (DRT) directly over exposed tendons with a subsequent full-thickness skin graft several weeks later. The procedures were performed per the manufacturer’s specifications, resulting in 2 stages of reconstruction. In our experience, DRT can offer immediate coverage unrestricted by wound size, and provides shorter operative time and decreased donor site and surgical morbidity compared with flap coverage, while demonstrating good cosmetic results. The patient provided written informed consent for print and electronic publication of this case report.

CASE

A 74-year-old right-handed man with Dupuytren’s contracture was evaluated for recurrent symptomatic contracture causing difficulty with daily activities. He reported palpable cords and contractures in the ring and small fingers of the right hand. He had 2 prior open surgical procedures, including palmar and digital fasciectomy of both hands. On the right hand, the ring and small fingers demonstrated 90° proximal interphalangeal (PIP) and 60° metacarpophalangeal (MCP) flexion contractures. Palpable central cords were present on the flexor surfaces of both the ring and small fingers. A well-healed surgical incision, performed 22 years earlier, was present over the palmar aspect of the ring finger.

Continue to: With consideration given...

With consideration given to the patient’s recurrent contracture after a prior surgical procedure, we discussed surgical excision of the diseased cords in order to eliminate the possibility of a second recurrence and maximize the gain of motion. Following discussion with the patient, we performed palmar and digital fasciectomy of the ring and small finger contractures. Postoperatively, the patient was followed closely for wound complications and vascular status. On his return to our clinic 11 days later, the patient was noted to have dehiscence of the digital wounds in the ring and small fingers (Figure 1). 

STAGE 1

During the first stage, completed 14 days following the index procedure, débridement of the wounds was performed, followed by provisional DRT coverage of the tendons, secured with 5-0 nylon sutures (Figure 2). 

STAGE 2

At approximately 2 weeks after application of the DRT, a full-thickness skin graft was applied. The thickness of the graft was chosen to allow for durable coverage of the palmar skin defects. Upon successful completion of the second stage, the patient was followed and evaluated for complete wound healing. On performing an examination 14 days after surgery, the ring and small fingers demonstrated only partially healed skin graft but significantly improved range of motion (ROM), with 40° to 90° arc of motion in the PIP joint and 25° to 90° arc of motion in the MCP joint (Figure 4). Owing to their limited size, the wounds were treated with dressing changes until successful healing (Figure 5).

Hand therapy was instituted to achieve maximum mobility for covered soft tissue and tendons and to maximize tendon gliding. At 1-year follow-up, the skin was fully healed and the patient’s active PIP motion was 30° to 90°, active MCP motion was 0° to 90°, and grip strength was 90 lb on both sides. The tendons glided under a well-vascularized tissue at the DRT placement site, and no secondary tenolysis procedure was deemed necessary.

DISCUSSION

Soft tissue defects with exposed tendons may offer a number of challenges for coverage. The primary concern is the creation of a gliding surface and the restoration of a functional tendon without adhesions.² However, surgeons must use their own clinical judgment when choosing the method of coverage so as to minimize the effects of donor site morbidity and maximize the overall functional and cosmetic outcomes. All options must be considered while selecting a material or flap that is likely to survive in the relatively avascular tendon plane.^2,8,11 When considering the reconstructive ladder, skin grafts may not represent a viable option in the presence of a nonvascularized wound bed, such as exposed tendon or bone, where paratenon or periosteum have been damaged. That leaves the surgeon with local flaps, regional flaps, free flaps, and skin substitutes.

Continue to : Before planning closure...

As Levin¹² noted in 1993, decisions regarding repair of any soft tissue defect may follow a well-delineated ladder beginning with the primary choice of split-thickness skin grafts and ending with free flaps. When treating tissue defects in the hand complex, flaps are an excellent option as they replace like with like, allow minimal scarring and early rehabilitation. ^13,14 Nevertheless, a few general disadvantages are inherent in flap procedure: increase in operating time, risk of flap loss, and in case of free flaps, knowledge, experience, and microsurgical ability.² In reference to complications, the rate of flap loss found by Khouri and colleagues¹⁵ was 4.1% with a 12.1% chance of incurring some measured complication, including wound dehiscence, arterial insufficiency, and flap necrosis.

Likewise, some of the conventional local and free flaps, including cutaneous and muscular flaps, prove ineffective in preventing tendon adhesions, create unsightly postoperative contours, or increase the area of trauma on the wounded hand, encouraging the use of free fascial flaps.¹¹ Among the wide array of potential free fascial flaps, the temporoparietal, scapular, lateral arm, radial forearm, and free serratus fascial flaps are some of the most popular for hand defects.^8,9 However, these procedures require an additional surgical site, meticulous dissection, microsurgical technique at times, and increased operating cost and time.^2,8-10 Furthermore, free fascial flaps have demonstrated occasional partial flap loss and a decreased survival of the overlying skin graft, leading some to advocate delayed skin graft placement.^10,16,17

On the basis of these complications, Bray and colleagues¹¹ noted that the utility of free flaps may be limited in smaller clinical settings. The primary disadvantage of using DRTs is the necessity for a second operative procedure to harvest and place the skin graft. Traditionally, this is performed 2 to 3 weeks after the initial DRT application. Nevertheless, a 1-stage procedure can be performed in an outpatient setup, minimizing the burden to the patient and the medical costs, followed by secondary intention healing.

In response to critics of the 2-stage technique, Sanger and colleagues¹⁸ described single-stage use of DRT with split-thickness skin grafts with placement of an overlying wound vacuum-assisted closure to help speed incorporation of the DRT and improve survival of the immediately grafted skin. Another viable alternative is the McCash open-palm technique.¹⁹ In the open-palm technique, a Brunner zigzag incision is made in the affected digit. A transverse incision is made in the palm. A partial fasciectomy is performed in the palm and digit. After release, the digital incision is closed, and the palmar incision is left open. Although this well-studied and well-reported technique is known to reduce the risk of flap necrosis due to tension and hematoma,²⁰ its main application is in the palm, as the name implies. Because in our patient the defect was palmar-digital with exposed “white structures,” we elected to use DRT.

Continue to: Although there is still...

ABSTRACT

Soft tissue defects associated with exposed tendon pose difficult reconstructive problems because of tendon adhesions, poor range of motion, poor cosmetic appearance, and donor site morbidity. Dermal regeneration template is a skin substitute widely used in reconstructive surgery, including the occasional coverage of tendons. However, postoperative functionality of the tendons has not been well documented. We report a case of using dermal regeneration template for soft tissue reconstruction overlying tendons with loss of paratenon in a patient with Dupuytren’s contracture. Dermal regeneration template may offer an alternative option for immediate tendon coverage in the hand.

Soft tissue defects overlying exposed tendon with loss of paratenon often precipitate poor clinical outcomes because of the dichotomous demands of both closing the overlying soft-tissue defect and providing a gliding surface for the underlying tendons.¹ Although avoidance of adhesions and restoration of function are the primary goals of the procedure, satisfactory appearance is also desirable. Likewise, any form of coverage should ideally provide good vasculature required for complete healing and an early form of closure following débridement.² Simple skin grafts do not adequately meet these demands because they result in a high rate of tendon adhesions,³ and also are limited in patients with limited donor skin availability or questionable underlying wound bed viability, such as in scleroderma.

In order to reduce the frequency of tendon adhesions by creating a gliding surface, the use of interpositional materials, both artificial and biologic, has been employed with varying degrees of success, including cellophane, chitosan membrane, fibrin sealant, autogenous fascial flaps, and autogenous venous grafts.^4-7 Many of the autogenous flaps and grafts have been employed with good success.⁸ However, complications and donor site morbidity encourage alternative procedures, including the use of artificial substances.^2,8-10

We present our clinical experience with a patient who underwent successful placement of Integra (Integra LifeSciences) Dermal Regeneration Template (DRT) directly over exposed tendons with a subsequent full-thickness skin graft several weeks later. The procedures were performed per the manufacturer’s specifications, resulting in 2 stages of reconstruction. In our experience, DRT can offer immediate coverage unrestricted by wound size, and provides shorter operative time and decreased donor site and surgical morbidity compared with flap coverage, while demonstrating good cosmetic results. The patient provided written informed consent for print and electronic publication of this case report.

CASE

A 74-year-old right-handed man with Dupuytren’s contracture was evaluated for recurrent symptomatic contracture causing difficulty with daily activities. He reported palpable cords and contractures in the ring and small fingers of the right hand. He had 2 prior open surgical procedures, including palmar and digital fasciectomy of both hands. On the right hand, the ring and small fingers demonstrated 90° proximal interphalangeal (PIP) and 60° metacarpophalangeal (MCP) flexion contractures. Palpable central cords were present on the flexor surfaces of both the ring and small fingers. A well-healed surgical incision, performed 22 years earlier, was present over the palmar aspect of the ring finger.

Continue to: With consideration given...

With consideration given to the patient’s recurrent contracture after a prior surgical procedure, we discussed surgical excision of the diseased cords in order to eliminate the possibility of a second recurrence and maximize the gain of motion. Following discussion with the patient, we performed palmar and digital fasciectomy of the ring and small finger contractures. Postoperatively, the patient was followed closely for wound complications and vascular status. On his return to our clinic 11 days later, the patient was noted to have dehiscence of the digital wounds in the ring and small fingers (Figure 1). 

STAGE 1

During the first stage, completed 14 days following the index procedure, débridement of the wounds was performed, followed by provisional DRT coverage of the tendons, secured with 5-0 nylon sutures (Figure 2). 

STAGE 2

At approximately 2 weeks after application of the DRT, a full-thickness skin graft was applied. The thickness of the graft was chosen to allow for durable coverage of the palmar skin defects. Upon successful completion of the second stage, the patient was followed and evaluated for complete wound healing. On performing an examination 14 days after surgery, the ring and small fingers demonstrated only partially healed skin graft but significantly improved range of motion (ROM), with 40° to 90° arc of motion in the PIP joint and 25° to 90° arc of motion in the MCP joint (Figure 4). Owing to their limited size, the wounds were treated with dressing changes until successful healing (Figure 5).

Hand therapy was instituted to achieve maximum mobility for covered soft tissue and tendons and to maximize tendon gliding. At 1-year follow-up, the skin was fully healed and the patient’s active PIP motion was 30° to 90°, active MCP motion was 0° to 90°, and grip strength was 90 lb on both sides. The tendons glided under a well-vascularized tissue at the DRT placement site, and no secondary tenolysis procedure was deemed necessary.

DISCUSSION

Soft tissue defects with exposed tendons may offer a number of challenges for coverage. The primary concern is the creation of a gliding surface and the restoration of a functional tendon without adhesions.² However, surgeons must use their own clinical judgment when choosing the method of coverage so as to minimize the effects of donor site morbidity and maximize the overall functional and cosmetic outcomes. All options must be considered while selecting a material or flap that is likely to survive in the relatively avascular tendon plane.^2,8,11 When considering the reconstructive ladder, skin grafts may not represent a viable option in the presence of a nonvascularized wound bed, such as exposed tendon or bone, where paratenon or periosteum have been damaged. That leaves the surgeon with local flaps, regional flaps, free flaps, and skin substitutes.

Continue to : Before planning closure...

As Levin¹² noted in 1993, decisions regarding repair of any soft tissue defect may follow a well-delineated ladder beginning with the primary choice of split-thickness skin grafts and ending with free flaps. When treating tissue defects in the hand complex, flaps are an excellent option as they replace like with like, allow minimal scarring and early rehabilitation. ^13,14 Nevertheless, a few general disadvantages are inherent in flap procedure: increase in operating time, risk of flap loss, and in case of free flaps, knowledge, experience, and microsurgical ability.² In reference to complications, the rate of flap loss found by Khouri and colleagues¹⁵ was 4.1% with a 12.1% chance of incurring some measured complication, including wound dehiscence, arterial insufficiency, and flap necrosis.

Likewise, some of the conventional local and free flaps, including cutaneous and muscular flaps, prove ineffective in preventing tendon adhesions, create unsightly postoperative contours, or increase the area of trauma on the wounded hand, encouraging the use of free fascial flaps.¹¹ Among the wide array of potential free fascial flaps, the temporoparietal, scapular, lateral arm, radial forearm, and free serratus fascial flaps are some of the most popular for hand defects.^8,9 However, these procedures require an additional surgical site, meticulous dissection, microsurgical technique at times, and increased operating cost and time.^2,8-10 Furthermore, free fascial flaps have demonstrated occasional partial flap loss and a decreased survival of the overlying skin graft, leading some to advocate delayed skin graft placement.^10,16,17

On the basis of these complications, Bray and colleagues¹¹ noted that the utility of free flaps may be limited in smaller clinical settings. The primary disadvantage of using DRTs is the necessity for a second operative procedure to harvest and place the skin graft. Traditionally, this is performed 2 to 3 weeks after the initial DRT application. Nevertheless, a 1-stage procedure can be performed in an outpatient setup, minimizing the burden to the patient and the medical costs, followed by secondary intention healing.

In response to critics of the 2-stage technique, Sanger and colleagues¹⁸ described single-stage use of DRT with split-thickness skin grafts with placement of an overlying wound vacuum-assisted closure to help speed incorporation of the DRT and improve survival of the immediately grafted skin. Another viable alternative is the McCash open-palm technique.¹⁹ In the open-palm technique, a Brunner zigzag incision is made in the affected digit. A transverse incision is made in the palm. A partial fasciectomy is performed in the palm and digit. After release, the digital incision is closed, and the palmar incision is left open. Although this well-studied and well-reported technique is known to reduce the risk of flap necrosis due to tension and hematoma,²⁰ its main application is in the palm, as the name implies. Because in our patient the defect was palmar-digital with exposed “white structures,” we elected to use DRT.

Continue to: Although there is still...

ABSTRACT

Soft tissue defects associated with exposed tendon pose difficult reconstructive problems because of tendon adhesions, poor range of motion, poor cosmetic appearance, and donor site morbidity. Dermal regeneration template is a skin substitute widely used in reconstructive surgery, including the occasional coverage of tendons. However, postoperative functionality of the tendons has not been well documented. We report a case of using dermal regeneration template for soft tissue reconstruction overlying tendons with loss of paratenon in a patient with Dupuytren’s contracture. Dermal regeneration template may offer an alternative option for immediate tendon coverage in the hand.

Soft tissue defects overlying exposed tendon with loss of paratenon often precipitate poor clinical outcomes because of the dichotomous demands of both closing the overlying soft-tissue defect and providing a gliding surface for the underlying tendons.¹ Although avoidance of adhesions and restoration of function are the primary goals of the procedure, satisfactory appearance is also desirable. Likewise, any form of coverage should ideally provide good vasculature required for complete healing and an early form of closure following débridement.² Simple skin grafts do not adequately meet these demands because they result in a high rate of tendon adhesions,³ and also are limited in patients with limited donor skin availability or questionable underlying wound bed viability, such as in scleroderma.

In order to reduce the frequency of tendon adhesions by creating a gliding surface, the use of interpositional materials, both artificial and biologic, has been employed with varying degrees of success, including cellophane, chitosan membrane, fibrin sealant, autogenous fascial flaps, and autogenous venous grafts.^4-7 Many of the autogenous flaps and grafts have been employed with good success.⁸ However, complications and donor site morbidity encourage alternative procedures, including the use of artificial substances.^2,8-10

We present our clinical experience with a patient who underwent successful placement of Integra (Integra LifeSciences) Dermal Regeneration Template (DRT) directly over exposed tendons with a subsequent full-thickness skin graft several weeks later. The procedures were performed per the manufacturer’s specifications, resulting in 2 stages of reconstruction. In our experience, DRT can offer immediate coverage unrestricted by wound size, and provides shorter operative time and decreased donor site and surgical morbidity compared with flap coverage, while demonstrating good cosmetic results. The patient provided written informed consent for print and electronic publication of this case report.

CASE

A 74-year-old right-handed man with Dupuytren’s contracture was evaluated for recurrent symptomatic contracture causing difficulty with daily activities. He reported palpable cords and contractures in the ring and small fingers of the right hand. He had 2 prior open surgical procedures, including palmar and digital fasciectomy of both hands. On the right hand, the ring and small fingers demonstrated 90° proximal interphalangeal (PIP) and 60° metacarpophalangeal (MCP) flexion contractures. Palpable central cords were present on the flexor surfaces of both the ring and small fingers. A well-healed surgical incision, performed 22 years earlier, was present over the palmar aspect of the ring finger.

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With consideration given to the patient’s recurrent contracture after a prior surgical procedure, we discussed surgical excision of the diseased cords in order to eliminate the possibility of a second recurrence and maximize the gain of motion. Following discussion with the patient, we performed palmar and digital fasciectomy of the ring and small finger contractures. Postoperatively, the patient was followed closely for wound complications and vascular status. On his return to our clinic 11 days later, the patient was noted to have dehiscence of the digital wounds in the ring and small fingers (Figure 1). 

STAGE 1

During the first stage, completed 14 days following the index procedure, débridement of the wounds was performed, followed by provisional DRT coverage of the tendons, secured with 5-0 nylon sutures (Figure 2). 

STAGE 2

At approximately 2 weeks after application of the DRT, a full-thickness skin graft was applied. The thickness of the graft was chosen to allow for durable coverage of the palmar skin defects. Upon successful completion of the second stage, the patient was followed and evaluated for complete wound healing. On performing an examination 14 days after surgery, the ring and small fingers demonstrated only partially healed skin graft but significantly improved range of motion (ROM), with 40° to 90° arc of motion in the PIP joint and 25° to 90° arc of motion in the MCP joint (Figure 4). Owing to their limited size, the wounds were treated with dressing changes until successful healing (Figure 5).

Hand therapy was instituted to achieve maximum mobility for covered soft tissue and tendons and to maximize tendon gliding. At 1-year follow-up, the skin was fully healed and the patient’s active PIP motion was 30° to 90°, active MCP motion was 0° to 90°, and grip strength was 90 lb on both sides. The tendons glided under a well-vascularized tissue at the DRT placement site, and no secondary tenolysis procedure was deemed necessary.

DISCUSSION

Soft tissue defects with exposed tendons may offer a number of challenges for coverage. The primary concern is the creation of a gliding surface and the restoration of a functional tendon without adhesions.² However, surgeons must use their own clinical judgment when choosing the method of coverage so as to minimize the effects of donor site morbidity and maximize the overall functional and cosmetic outcomes. All options must be considered while selecting a material or flap that is likely to survive in the relatively avascular tendon plane.^2,8,11 When considering the reconstructive ladder, skin grafts may not represent a viable option in the presence of a nonvascularized wound bed, such as exposed tendon or bone, where paratenon or periosteum have been damaged. That leaves the surgeon with local flaps, regional flaps, free flaps, and skin substitutes.

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As Levin¹² noted in 1993, decisions regarding repair of any soft tissue defect may follow a well-delineated ladder beginning with the primary choice of split-thickness skin grafts and ending with free flaps. When treating tissue defects in the hand complex, flaps are an excellent option as they replace like with like, allow minimal scarring and early rehabilitation. ^13,14 Nevertheless, a few general disadvantages are inherent in flap procedure: increase in operating time, risk of flap loss, and in case of free flaps, knowledge, experience, and microsurgical ability.² In reference to complications, the rate of flap loss found by Khouri and colleagues¹⁵ was 4.1% with a 12.1% chance of incurring some measured complication, including wound dehiscence, arterial insufficiency, and flap necrosis.

Likewise, some of the conventional local and free flaps, including cutaneous and muscular flaps, prove ineffective in preventing tendon adhesions, create unsightly postoperative contours, or increase the area of trauma on the wounded hand, encouraging the use of free fascial flaps.¹¹ Among the wide array of potential free fascial flaps, the temporoparietal, scapular, lateral arm, radial forearm, and free serratus fascial flaps are some of the most popular for hand defects.^8,9 However, these procedures require an additional surgical site, meticulous dissection, microsurgical technique at times, and increased operating cost and time.^2,8-10 Furthermore, free fascial flaps have demonstrated occasional partial flap loss and a decreased survival of the overlying skin graft, leading some to advocate delayed skin graft placement.^10,16,17

On the basis of these complications, Bray and colleagues¹¹ noted that the utility of free flaps may be limited in smaller clinical settings. The primary disadvantage of using DRTs is the necessity for a second operative procedure to harvest and place the skin graft. Traditionally, this is performed 2 to 3 weeks after the initial DRT application. Nevertheless, a 1-stage procedure can be performed in an outpatient setup, minimizing the burden to the patient and the medical costs, followed by secondary intention healing.

In response to critics of the 2-stage technique, Sanger and colleagues¹⁸ described single-stage use of DRT with split-thickness skin grafts with placement of an overlying wound vacuum-assisted closure to help speed incorporation of the DRT and improve survival of the immediately grafted skin. Another viable alternative is the McCash open-palm technique.¹⁹ In the open-palm technique, a Brunner zigzag incision is made in the affected digit. A transverse incision is made in the palm. A partial fasciectomy is performed in the palm and digit. After release, the digital incision is closed, and the palmar incision is left open. Although this well-studied and well-reported technique is known to reduce the risk of flap necrosis due to tension and hematoma,²⁰ its main application is in the palm, as the name implies. Because in our patient the defect was palmar-digital with exposed “white structures,” we elected to use DRT.

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