Case Reports

According to the Centers for Disease Control and Prevention, abdominal pain is the leading reason for ED visits in the United States, with approximately 10 million visits per year.¹ Though a large number of presentations are due to nontraumatic causes of abdominal pain, one etiology is among the most time-sensitive and critical diagnoses: acute colonic perforation.

Colonic perforations can be caused by diverticulitis, trauma, malignancy, ulcerative colitis, and other etiologies.² A rare, yet life-threatening cause of colonic perforation, of which only a few cases have been documented in the literature, is stercoral colonic perforation.²

Regardless of the etiology, the critical actions for any colonic perforation are quick recognition, medical stabilization, and surgical evaluation. This case report highlights the diagnosis and treatment of acute stercoral colonic perforation with peritonitis secondary to hypothyroidism.

Case

A 49-year-old woman with a medical history significant for hypothyroidism presented to the ED for evaluation of diffuse abdominal pain, nausea, and nonbilious, nonbloody vomiting that started in the early evening of presentation. The patient denied any previous pain or associated symptoms, and said she had a small, hard bowel movement 1 day prior to arrival. She began experiencing mild abdominal pain on the morning of presentation. Her symptoms acutely worsened at approximately 5:00 pm. She denied having any diarrhea, melena, or hematochezia. Her surgical history was significant for a cesarean delivery. She denied any history of small bowel obstruction. The patient admitted that she had stopped taking levothyroxine (150 mcg daily) prescribed for hypothyroidism approximately 1 month earlier, but had been restarted on it approximately 2 to 3 days prior to her presentation.

On physical examination, her vital signs were: heart rate, 156 beats/min; blood pressure, 134/84 mm Hg; respiratory rate, 20 breaths/min, and temperature, 97.4°F. The patient appeared ill and diaphoretic, writhing on the stretcher. Abdominal examination was significant for diminished bowel sounds, diffuse abdominal distension, rigidity, and tenderness with light palpation.

Laboratory evaluation showed an elevated lactic acid level of 7.7 mmol/L, a white blood cell count of 7,200 cells/mm³ (segment form, 69.5%), and the following abnormal blood chemistry results: creatinine, 2.08 mg/dL; aspartate aminotransferase, 176 U/L; alanine aminotransferase, 138 U/L; and thyroid-stimulating hormone (TSH), 225.3 mcIU/mL. Other laboratory results were within normal range. Her electrocardiogram showed sinus tachycardia with a rate of 154 beats/min, a QTc within normal limits, and no ST elevations or depressions.

An abdominopelvic computed tomography (CT) scan revealed free air, free fluid, and possibly stool within the abdomen and pelvis. The findings were consistent with a ruptured hollow viscus, possibly a sigmoid colonic perforation. The radiologist also noted hepatomegaly and significant hepatic steatosis. A surgeon was immediately notified and evaluated the patient in the ED. The working diagnosis was stercoral colonic perforation secondary to severe hypothyroidism, and the patient was taken emergently to the operating room for repair.

Intraoperatively, the patient underwent exploratory laparotomy, which revealed gross fecal contamination of the abdomen. The surgeon noted that there was fecal staining along the serosal surface of the small bowel and throughout the pelvis. There were also large, hard stool balls outside of the colon. The perforation was along the mesenteric surface of the sigmoid just above the rectosigmoid junction.

The abdomen was copiously irrigated, the perforated segment was resected, and a Hartmann colostomy was created. The diagnosis was stercoral sigmoid perforation with peritonitis, and the patient was transferred to the intensive care unit for antibiotic treatment and further medical care, including intravenous (IV) levothyroxine.

She was extubated uneventfully on postoperative day 2, and the acute renal failure improved with supportive care only. Her bowel function slowly returned without complication. She was switched to oral levothyroxine on postoperative day 3. On day 13, she was given strict instructions for continuation of her thyroid medication and close monitoring for postsurgical complications, and was discharged home with appropriate follow-up.

Discussion

Multiple contributing factors can lead to bowel perforation. In this case, severe hypothyroidism with constipation caused a colonic perforation. Our patient had severe constipation that increased intraluminal pressure, causing the bowel wall to become ischemic and subsequently perforate.³ Any disease that causes significant constipation or obstruction of transit could lead to the same catastrophic result.

According to Huang et al,⁴ as of 2002, fewer than 90 cases of general stercoral bowel perforation had been reported, with no clear age range. However, patients in their mid-50s to mid-60s appear to be the most commonly affected age group.⁴ Our patient was younger than this age group, making identification of the problem by age alone difficult.

Hypothyroidism

The incidence of hypothyroidism in iodine-replete communities varies between 1% to 2% of the general population.⁵ The condition is more common in older women, affecting approximately 10% of those over age 65 years. In the United States, the prevalence of biochemical hypothyroidism is 4.6%; however, clinically evident hypothyroidism is present in only 0.3%.⁶ Common causes for hypothyroidism are listed in the Table.^7,8

Myxedema Coma

Untreated, hypothyroidism can lead to potentially fatal conditions, such as myxedema coma, which is characterized by hypothermia, hypotension, bradycardia, respiratory depression, and altered mental status.⁷ Severe myxedema coma can result in cardiovascular collapse, and eventual death. Electrocardiography findings of severe hypothyroidism include bradycardia, low-voltage QRS, and widespread T-wave inversions.⁷ Our patient was tachycardic and did not have any acute findings to suggest myxedema coma.

Treatment for myxedema coma includes supportive care with ventilatory support and pressor support if necessary. Patients should be given IV hydrocortisone, 100 mg, to treat possible adrenal insufficiency and T₄, 4 mcg/kg by slow IV infusion.⁷ Caution should be taken if giving a patient T₃ due to the risk of dysrhythmias and myocardial infarction (MI).⁷ As our patient was not displaying myxedema coma, the surgeon elected not to start IV thyroid replacement to avoid exacerbating the patient’s tachycardia and possibly precipitating an MI intraoperatively.

Conclusion

Our case underscores the importance of promptly recognizing the signs and symptoms of stercoral colonic perforation in patients who present with nontraumatic abdominal pain accompanied by nausea and nonbilious, nonbloody vomiting. Although stercoral colonic perforation is a rare cause of nontraumatic abdominal pain, as with any type of colonic perforation, it constitutes a life-threatening medical emergency. As our case illustrates, prompt diagnosis through a thorough history taking, physical examination, and laboratory and imaging studies is critical to ensure medical stabilization and surgical management to reduce morbidity and mortality.

According to the Centers for Disease Control and Prevention, abdominal pain is the leading reason for ED visits in the United States, with approximately 10 million visits per year.¹ Though a large number of presentations are due to nontraumatic causes of abdominal pain, one etiology is among the most time-sensitive and critical diagnoses: acute colonic perforation.

Colonic perforations can be caused by diverticulitis, trauma, malignancy, ulcerative colitis, and other etiologies.² A rare, yet life-threatening cause of colonic perforation, of which only a few cases have been documented in the literature, is stercoral colonic perforation.²

Regardless of the etiology, the critical actions for any colonic perforation are quick recognition, medical stabilization, and surgical evaluation. This case report highlights the diagnosis and treatment of acute stercoral colonic perforation with peritonitis secondary to hypothyroidism.

Case

A 49-year-old woman with a medical history significant for hypothyroidism presented to the ED for evaluation of diffuse abdominal pain, nausea, and nonbilious, nonbloody vomiting that started in the early evening of presentation. The patient denied any previous pain or associated symptoms, and said she had a small, hard bowel movement 1 day prior to arrival. She began experiencing mild abdominal pain on the morning of presentation. Her symptoms acutely worsened at approximately 5:00 pm. She denied having any diarrhea, melena, or hematochezia. Her surgical history was significant for a cesarean delivery. She denied any history of small bowel obstruction. The patient admitted that she had stopped taking levothyroxine (150 mcg daily) prescribed for hypothyroidism approximately 1 month earlier, but had been restarted on it approximately 2 to 3 days prior to her presentation.

On physical examination, her vital signs were: heart rate, 156 beats/min; blood pressure, 134/84 mm Hg; respiratory rate, 20 breaths/min, and temperature, 97.4°F. The patient appeared ill and diaphoretic, writhing on the stretcher. Abdominal examination was significant for diminished bowel sounds, diffuse abdominal distension, rigidity, and tenderness with light palpation.

Laboratory evaluation showed an elevated lactic acid level of 7.7 mmol/L, a white blood cell count of 7,200 cells/mm³ (segment form, 69.5%), and the following abnormal blood chemistry results: creatinine, 2.08 mg/dL; aspartate aminotransferase, 176 U/L; alanine aminotransferase, 138 U/L; and thyroid-stimulating hormone (TSH), 225.3 mcIU/mL. Other laboratory results were within normal range. Her electrocardiogram showed sinus tachycardia with a rate of 154 beats/min, a QTc within normal limits, and no ST elevations or depressions.

An abdominopelvic computed tomography (CT) scan revealed free air, free fluid, and possibly stool within the abdomen and pelvis. The findings were consistent with a ruptured hollow viscus, possibly a sigmoid colonic perforation. The radiologist also noted hepatomegaly and significant hepatic steatosis. A surgeon was immediately notified and evaluated the patient in the ED. The working diagnosis was stercoral colonic perforation secondary to severe hypothyroidism, and the patient was taken emergently to the operating room for repair.

Intraoperatively, the patient underwent exploratory laparotomy, which revealed gross fecal contamination of the abdomen. The surgeon noted that there was fecal staining along the serosal surface of the small bowel and throughout the pelvis. There were also large, hard stool balls outside of the colon. The perforation was along the mesenteric surface of the sigmoid just above the rectosigmoid junction.

The abdomen was copiously irrigated, the perforated segment was resected, and a Hartmann colostomy was created. The diagnosis was stercoral sigmoid perforation with peritonitis, and the patient was transferred to the intensive care unit for antibiotic treatment and further medical care, including intravenous (IV) levothyroxine.

She was extubated uneventfully on postoperative day 2, and the acute renal failure improved with supportive care only. Her bowel function slowly returned without complication. She was switched to oral levothyroxine on postoperative day 3. On day 13, she was given strict instructions for continuation of her thyroid medication and close monitoring for postsurgical complications, and was discharged home with appropriate follow-up.

Discussion

Multiple contributing factors can lead to bowel perforation. In this case, severe hypothyroidism with constipation caused a colonic perforation. Our patient had severe constipation that increased intraluminal pressure, causing the bowel wall to become ischemic and subsequently perforate.³ Any disease that causes significant constipation or obstruction of transit could lead to the same catastrophic result.

According to Huang et al,⁴ as of 2002, fewer than 90 cases of general stercoral bowel perforation had been reported, with no clear age range. However, patients in their mid-50s to mid-60s appear to be the most commonly affected age group.⁴ Our patient was younger than this age group, making identification of the problem by age alone difficult.

Hypothyroidism

The incidence of hypothyroidism in iodine-replete communities varies between 1% to 2% of the general population.⁵ The condition is more common in older women, affecting approximately 10% of those over age 65 years. In the United States, the prevalence of biochemical hypothyroidism is 4.6%; however, clinically evident hypothyroidism is present in only 0.3%.⁶ Common causes for hypothyroidism are listed in the Table.^7,8

Myxedema Coma

Untreated, hypothyroidism can lead to potentially fatal conditions, such as myxedema coma, which is characterized by hypothermia, hypotension, bradycardia, respiratory depression, and altered mental status.⁷ Severe myxedema coma can result in cardiovascular collapse, and eventual death. Electrocardiography findings of severe hypothyroidism include bradycardia, low-voltage QRS, and widespread T-wave inversions.⁷ Our patient was tachycardic and did not have any acute findings to suggest myxedema coma.

Treatment for myxedema coma includes supportive care with ventilatory support and pressor support if necessary. Patients should be given IV hydrocortisone, 100 mg, to treat possible adrenal insufficiency and T₄, 4 mcg/kg by slow IV infusion.⁷ Caution should be taken if giving a patient T₃ due to the risk of dysrhythmias and myocardial infarction (MI).⁷ As our patient was not displaying myxedema coma, the surgeon elected not to start IV thyroid replacement to avoid exacerbating the patient’s tachycardia and possibly precipitating an MI intraoperatively.

Conclusion

Our case underscores the importance of promptly recognizing the signs and symptoms of stercoral colonic perforation in patients who present with nontraumatic abdominal pain accompanied by nausea and nonbilious, nonbloody vomiting. Although stercoral colonic perforation is a rare cause of nontraumatic abdominal pain, as with any type of colonic perforation, it constitutes a life-threatening medical emergency. As our case illustrates, prompt diagnosis through a thorough history taking, physical examination, and laboratory and imaging studies is critical to ensure medical stabilization and surgical management to reduce morbidity and mortality.

According to the Centers for Disease Control and Prevention, abdominal pain is the leading reason for ED visits in the United States, with approximately 10 million visits per year.¹ Though a large number of presentations are due to nontraumatic causes of abdominal pain, one etiology is among the most time-sensitive and critical diagnoses: acute colonic perforation.

Colonic perforations can be caused by diverticulitis, trauma, malignancy, ulcerative colitis, and other etiologies.² A rare, yet life-threatening cause of colonic perforation, of which only a few cases have been documented in the literature, is stercoral colonic perforation.²

Regardless of the etiology, the critical actions for any colonic perforation are quick recognition, medical stabilization, and surgical evaluation. This case report highlights the diagnosis and treatment of acute stercoral colonic perforation with peritonitis secondary to hypothyroidism.

Case

A 49-year-old woman with a medical history significant for hypothyroidism presented to the ED for evaluation of diffuse abdominal pain, nausea, and nonbilious, nonbloody vomiting that started in the early evening of presentation. The patient denied any previous pain or associated symptoms, and said she had a small, hard bowel movement 1 day prior to arrival. She began experiencing mild abdominal pain on the morning of presentation. Her symptoms acutely worsened at approximately 5:00 pm. She denied having any diarrhea, melena, or hematochezia. Her surgical history was significant for a cesarean delivery. She denied any history of small bowel obstruction. The patient admitted that she had stopped taking levothyroxine (150 mcg daily) prescribed for hypothyroidism approximately 1 month earlier, but had been restarted on it approximately 2 to 3 days prior to her presentation.

On physical examination, her vital signs were: heart rate, 156 beats/min; blood pressure, 134/84 mm Hg; respiratory rate, 20 breaths/min, and temperature, 97.4°F. The patient appeared ill and diaphoretic, writhing on the stretcher. Abdominal examination was significant for diminished bowel sounds, diffuse abdominal distension, rigidity, and tenderness with light palpation.

Laboratory evaluation showed an elevated lactic acid level of 7.7 mmol/L, a white blood cell count of 7,200 cells/mm³ (segment form, 69.5%), and the following abnormal blood chemistry results: creatinine, 2.08 mg/dL; aspartate aminotransferase, 176 U/L; alanine aminotransferase, 138 U/L; and thyroid-stimulating hormone (TSH), 225.3 mcIU/mL. Other laboratory results were within normal range. Her electrocardiogram showed sinus tachycardia with a rate of 154 beats/min, a QTc within normal limits, and no ST elevations or depressions.

An abdominopelvic computed tomography (CT) scan revealed free air, free fluid, and possibly stool within the abdomen and pelvis. The findings were consistent with a ruptured hollow viscus, possibly a sigmoid colonic perforation. The radiologist also noted hepatomegaly and significant hepatic steatosis. A surgeon was immediately notified and evaluated the patient in the ED. The working diagnosis was stercoral colonic perforation secondary to severe hypothyroidism, and the patient was taken emergently to the operating room for repair.

Intraoperatively, the patient underwent exploratory laparotomy, which revealed gross fecal contamination of the abdomen. The surgeon noted that there was fecal staining along the serosal surface of the small bowel and throughout the pelvis. There were also large, hard stool balls outside of the colon. The perforation was along the mesenteric surface of the sigmoid just above the rectosigmoid junction.

The abdomen was copiously irrigated, the perforated segment was resected, and a Hartmann colostomy was created. The diagnosis was stercoral sigmoid perforation with peritonitis, and the patient was transferred to the intensive care unit for antibiotic treatment and further medical care, including intravenous (IV) levothyroxine.

She was extubated uneventfully on postoperative day 2, and the acute renal failure improved with supportive care only. Her bowel function slowly returned without complication. She was switched to oral levothyroxine on postoperative day 3. On day 13, she was given strict instructions for continuation of her thyroid medication and close monitoring for postsurgical complications, and was discharged home with appropriate follow-up.

Discussion

Multiple contributing factors can lead to bowel perforation. In this case, severe hypothyroidism with constipation caused a colonic perforation. Our patient had severe constipation that increased intraluminal pressure, causing the bowel wall to become ischemic and subsequently perforate.³ Any disease that causes significant constipation or obstruction of transit could lead to the same catastrophic result.

According to Huang et al,⁴ as of 2002, fewer than 90 cases of general stercoral bowel perforation had been reported, with no clear age range. However, patients in their mid-50s to mid-60s appear to be the most commonly affected age group.⁴ Our patient was younger than this age group, making identification of the problem by age alone difficult.

Hypothyroidism

The incidence of hypothyroidism in iodine-replete communities varies between 1% to 2% of the general population.⁵ The condition is more common in older women, affecting approximately 10% of those over age 65 years. In the United States, the prevalence of biochemical hypothyroidism is 4.6%; however, clinically evident hypothyroidism is present in only 0.3%.⁶ Common causes for hypothyroidism are listed in the Table.^7,8

Myxedema Coma

Untreated, hypothyroidism can lead to potentially fatal conditions, such as myxedema coma, which is characterized by hypothermia, hypotension, bradycardia, respiratory depression, and altered mental status.⁷ Severe myxedema coma can result in cardiovascular collapse, and eventual death. Electrocardiography findings of severe hypothyroidism include bradycardia, low-voltage QRS, and widespread T-wave inversions.⁷ Our patient was tachycardic and did not have any acute findings to suggest myxedema coma.

Treatment for myxedema coma includes supportive care with ventilatory support and pressor support if necessary. Patients should be given IV hydrocortisone, 100 mg, to treat possible adrenal insufficiency and T₄, 4 mcg/kg by slow IV infusion.⁷ Caution should be taken if giving a patient T₃ due to the risk of dysrhythmias and myocardial infarction (MI).⁷ As our patient was not displaying myxedema coma, the surgeon elected not to start IV thyroid replacement to avoid exacerbating the patient’s tachycardia and possibly precipitating an MI intraoperatively.

Conclusion

Our case underscores the importance of promptly recognizing the signs and symptoms of stercoral colonic perforation in patients who present with nontraumatic abdominal pain accompanied by nausea and nonbilious, nonbloody vomiting. Although stercoral colonic perforation is a rare cause of nontraumatic abdominal pain, as with any type of colonic perforation, it constitutes a life-threatening medical emergency. As our case illustrates, prompt diagnosis through a thorough history taking, physical examination, and laboratory and imaging studies is critical to ensure medical stabilization and surgical management to reduce morbidity and mortality.

Laboratory studies revealed leukocytosis with a left shift. Chest radiographs were negative for pneumonia. A magnetic resonance image (MRI) of the lumbar spine was obtained to evaluate for diskitis osteomyelitis. A radiograph of the pelvis was also obtained to evaluate the patient’s THAs, and a computed tomography scan (CT) of the abdomen and pelvis with contrast was obtained for further evaluation. Representative CT, radiographic, and MRI images are shown at left (Figures 1-3).

What is the suspected diagnosis?

Answer

The MRI of the lumbar spine demonstrated no evidence of diskitis osteomyelitis. However, T₂-weighted axial images showed enlarged heterogeneous bilateral psoas muscles with bright signal, indicating the presence of fluid (white arrows, Figure 4).

On the pelvic radiographs, both femoral heads appeared off-center within the acetabular cups (red arrows, Figure 5). This eccentric positioning indicated wear of the polyethylene in the THAs that normally occupies the space between the acetabular cup and the femoral head. In addition, focal lucency in the right acetabulum indicated breakdown of the bone, a condition referred to as osteolysis (white asterisk, Figure 5).

An abdominopelvic CT scan with contrast was performed and confirmed the findings of polyethylene wear and osteolysis. The CT scan also demonstrated large bilateral hip joint effusions (white arrows, Figure 6), decompressed along distended bilateral iliopsoas bursae (red asterisks, Figure 6), and communicating with the bilateral psoas muscle collections (red arrows, Figure 6).

Osteolysis With Iliopsoas Bursitis

Bursae are fluid-filled sacs lined by synovial tissue located throughout the body to reduce friction at sites of movement between muscles, bones, and tendons. Bursitis develops when these sacs become inflamed and/or infected and fill with fluid. The iliopsoas bursa lies between the anterior capsule of the hip and the psoas tendon, iliacus tendon, and muscle fibers.^1,2 This bursa frequently communicates with the hip joint.^3,4 Iliopsoas bursal distension has been reported following THA in the setting of polyethylene wear,⁵ and aseptic bursitis is a commonly seen incidental finding at the time of revision surgery.⁶

In this patient, long-standing polyethylene-induced synovitis had markedly expanded the hip joints and iliopsoas bursae, eventually resulting in superinfection, which accounted for the patient’s symptoms.

Treatment

Based on the imaging findings, interventional radiology services were contacted. The interventional radiologist drained the bilateral psoas abscesses. Cultures of the fluid were positive for both methicillin-resistant Staphylococcus aureus (MRSA) and methicillin-susceptible S aureus (MSSA). The patient was admitted to the hospital for treatment of MRSA and MSSA with intravenous antibiotic therapy. He recovered from the infection and was discharged on hospital day 2, with instructions to follow up with an orthopedic surgeon to discuss eventual revision of the bilateral THAs.

Laboratory studies revealed leukocytosis with a left shift. Chest radiographs were negative for pneumonia. A magnetic resonance image (MRI) of the lumbar spine was obtained to evaluate for diskitis osteomyelitis. A radiograph of the pelvis was also obtained to evaluate the patient’s THAs, and a computed tomography scan (CT) of the abdomen and pelvis with contrast was obtained for further evaluation. Representative CT, radiographic, and MRI images are shown at left (Figures 1-3).

What is the suspected diagnosis?

Answer

The MRI of the lumbar spine demonstrated no evidence of diskitis osteomyelitis. However, T₂-weighted axial images showed enlarged heterogeneous bilateral psoas muscles with bright signal, indicating the presence of fluid (white arrows, Figure 4).

On the pelvic radiographs, both femoral heads appeared off-center within the acetabular cups (red arrows, Figure 5). This eccentric positioning indicated wear of the polyethylene in the THAs that normally occupies the space between the acetabular cup and the femoral head. In addition, focal lucency in the right acetabulum indicated breakdown of the bone, a condition referred to as osteolysis (white asterisk, Figure 5).

An abdominopelvic CT scan with contrast was performed and confirmed the findings of polyethylene wear and osteolysis. The CT scan also demonstrated large bilateral hip joint effusions (white arrows, Figure 6), decompressed along distended bilateral iliopsoas bursae (red asterisks, Figure 6), and communicating with the bilateral psoas muscle collections (red arrows, Figure 6).

Osteolysis With Iliopsoas Bursitis

Bursae are fluid-filled sacs lined by synovial tissue located throughout the body to reduce friction at sites of movement between muscles, bones, and tendons. Bursitis develops when these sacs become inflamed and/or infected and fill with fluid. The iliopsoas bursa lies between the anterior capsule of the hip and the psoas tendon, iliacus tendon, and muscle fibers.^1,2 This bursa frequently communicates with the hip joint.^3,4 Iliopsoas bursal distension has been reported following THA in the setting of polyethylene wear,⁵ and aseptic bursitis is a commonly seen incidental finding at the time of revision surgery.⁶

In this patient, long-standing polyethylene-induced synovitis had markedly expanded the hip joints and iliopsoas bursae, eventually resulting in superinfection, which accounted for the patient’s symptoms.

Treatment

Based on the imaging findings, interventional radiology services were contacted. The interventional radiologist drained the bilateral psoas abscesses. Cultures of the fluid were positive for both methicillin-resistant Staphylococcus aureus (MRSA) and methicillin-susceptible S aureus (MSSA). The patient was admitted to the hospital for treatment of MRSA and MSSA with intravenous antibiotic therapy. He recovered from the infection and was discharged on hospital day 2, with instructions to follow up with an orthopedic surgeon to discuss eventual revision of the bilateral THAs.

Laboratory studies revealed leukocytosis with a left shift. Chest radiographs were negative for pneumonia. A magnetic resonance image (MRI) of the lumbar spine was obtained to evaluate for diskitis osteomyelitis. A radiograph of the pelvis was also obtained to evaluate the patient’s THAs, and a computed tomography scan (CT) of the abdomen and pelvis with contrast was obtained for further evaluation. Representative CT, radiographic, and MRI images are shown at left (Figures 1-3).

What is the suspected diagnosis?

Answer

The MRI of the lumbar spine demonstrated no evidence of diskitis osteomyelitis. However, T₂-weighted axial images showed enlarged heterogeneous bilateral psoas muscles with bright signal, indicating the presence of fluid (white arrows, Figure 4).

On the pelvic radiographs, both femoral heads appeared off-center within the acetabular cups (red arrows, Figure 5). This eccentric positioning indicated wear of the polyethylene in the THAs that normally occupies the space between the acetabular cup and the femoral head. In addition, focal lucency in the right acetabulum indicated breakdown of the bone, a condition referred to as osteolysis (white asterisk, Figure 5).

An abdominopelvic CT scan with contrast was performed and confirmed the findings of polyethylene wear and osteolysis. The CT scan also demonstrated large bilateral hip joint effusions (white arrows, Figure 6), decompressed along distended bilateral iliopsoas bursae (red asterisks, Figure 6), and communicating with the bilateral psoas muscle collections (red arrows, Figure 6).

Osteolysis With Iliopsoas Bursitis

Bursae are fluid-filled sacs lined by synovial tissue located throughout the body to reduce friction at sites of movement between muscles, bones, and tendons. Bursitis develops when these sacs become inflamed and/or infected and fill with fluid. The iliopsoas bursa lies between the anterior capsule of the hip and the psoas tendon, iliacus tendon, and muscle fibers.^1,2 This bursa frequently communicates with the hip joint.^3,4 Iliopsoas bursal distension has been reported following THA in the setting of polyethylene wear,⁵ and aseptic bursitis is a commonly seen incidental finding at the time of revision surgery.⁶

In this patient, long-standing polyethylene-induced synovitis had markedly expanded the hip joints and iliopsoas bursae, eventually resulting in superinfection, which accounted for the patient’s symptoms.

Treatment

Based on the imaging findings, interventional radiology services were contacted. The interventional radiologist drained the bilateral psoas abscesses. Cultures of the fluid were positive for both methicillin-resistant Staphylococcus aureus (MRSA) and methicillin-susceptible S aureus (MSSA). The patient was admitted to the hospital for treatment of MRSA and MSSA with intravenous antibiotic therapy. He recovered from the infection and was discharged on hospital day 2, with instructions to follow up with an orthopedic surgeon to discuss eventual revision of the bilateral THAs.

Take-Home Points

• Adolescent growth spurt, height-to-weight ratio, and perioperative weight loss are risk factors associated with SMA syndrome following pediatric spine surgery.
• Must recognize nonspecific symptoms such as abdominal pain, tenderness, distention, bilious or projectile vomiting, hypoactive bowel sounds, and anorexia postoperatively.
• Complications of SMA syndrome can potentially lead to aspiration pneumonia, acute gastric rupture, or cardiovascular collapse and death.

Superior mesenteric artery (SMA) syndrome resulting from surgical treatment of scoliosis has been recognized in the medical literature since 1752.¹ Throughout the literature, SMA syndrome variably has been referred to as cast syndrome, Wilkie syndrome, arteriomesenteric duodenal obstruction, and chronic duodenal ileus.² We now recognize numerous etiologies of SMA syndrome, as several sources can externally compress the duodenum. Classic acute symptoms of bowel obstruction include bilious vomiting, nausea, and epigastric pain. Chronic manifestations of SMA syndrome may include weight loss and decreased appetite. Our literature review revealed that adolescent growth spurt, height-to-weight ratio, and perioperative weight loss are risk factors associated with SMA syndrome after pediatric spine surgery.

We report the case of a 14-year-old boy who developed SMA syndrome after undergoing scoliosis surgery. The patient and his mother provided written informed consent for print and electronic publication of this case report.

Case Report

A 14-year-old boy with a history of idiopathic scoliosis presented to Cohen Children’s Hospital (Long Island Jewish Medical Center) with bilious vomiting that had persisted for 7 days after posterior T9–L4 fusion with instrumentation.

Discussion

SMA syndrome is attributed to the anatomical orientation of the third part of the duodenum, which passes between the aorta and the SMA (Figure 4).

Adolescents are particularly vulnerable to this condition. Faster adolescent bone growth relative to visceral growth is accompanied by a decrease in SMA angle.³ Occasionally, body casts are used after surgery to immobilize the vertebrae and augment healing. Cast syndrome occurs when pressure from a body cast causes a bowel obstruction secondary to spinal hyperextension and amplified spinal lordosis.² This finding, dating to the 19th century, was reported by Willet⁴ when a patient died 48 hours after application of a body cast. In 1950, the term cast syndrome was coined after a motorcyclist’s injuries were treated with a hip spica cast and the patient died of cardiovascular collapse secondary to persistent vomiting.⁵

Table 1 summarizes various evaluation, diagnosis, and treatment algorithms designed to optimize nutrition and weight in patients developing signs and symptoms of SMA syndrome after posterior spinal instrumentation and fusion for adolescent idiopathic scoliosis (AIS).

Take-Home Points

• Adolescent growth spurt, height-to-weight ratio, and perioperative weight loss are risk factors associated with SMA syndrome following pediatric spine surgery.
• Must recognize nonspecific symptoms such as abdominal pain, tenderness, distention, bilious or projectile vomiting, hypoactive bowel sounds, and anorexia postoperatively.
• Complications of SMA syndrome can potentially lead to aspiration pneumonia, acute gastric rupture, or cardiovascular collapse and death.

Superior mesenteric artery (SMA) syndrome resulting from surgical treatment of scoliosis has been recognized in the medical literature since 1752.¹ Throughout the literature, SMA syndrome variably has been referred to as cast syndrome, Wilkie syndrome, arteriomesenteric duodenal obstruction, and chronic duodenal ileus.² We now recognize numerous etiologies of SMA syndrome, as several sources can externally compress the duodenum. Classic acute symptoms of bowel obstruction include bilious vomiting, nausea, and epigastric pain. Chronic manifestations of SMA syndrome may include weight loss and decreased appetite. Our literature review revealed that adolescent growth spurt, height-to-weight ratio, and perioperative weight loss are risk factors associated with SMA syndrome after pediatric spine surgery.

We report the case of a 14-year-old boy who developed SMA syndrome after undergoing scoliosis surgery. The patient and his mother provided written informed consent for print and electronic publication of this case report.

Case Report

A 14-year-old boy with a history of idiopathic scoliosis presented to Cohen Children’s Hospital (Long Island Jewish Medical Center) with bilious vomiting that had persisted for 7 days after posterior T9–L4 fusion with instrumentation.

Discussion

SMA syndrome is attributed to the anatomical orientation of the third part of the duodenum, which passes between the aorta and the SMA (Figure 4).

Adolescents are particularly vulnerable to this condition. Faster adolescent bone growth relative to visceral growth is accompanied by a decrease in SMA angle.³ Occasionally, body casts are used after surgery to immobilize the vertebrae and augment healing. Cast syndrome occurs when pressure from a body cast causes a bowel obstruction secondary to spinal hyperextension and amplified spinal lordosis.² This finding, dating to the 19th century, was reported by Willet⁴ when a patient died 48 hours after application of a body cast. In 1950, the term cast syndrome was coined after a motorcyclist’s injuries were treated with a hip spica cast and the patient died of cardiovascular collapse secondary to persistent vomiting.⁵

Table 1 summarizes various evaluation, diagnosis, and treatment algorithms designed to optimize nutrition and weight in patients developing signs and symptoms of SMA syndrome after posterior spinal instrumentation and fusion for adolescent idiopathic scoliosis (AIS).

Take-Home Points

• Adolescent growth spurt, height-to-weight ratio, and perioperative weight loss are risk factors associated with SMA syndrome following pediatric spine surgery.
• Must recognize nonspecific symptoms such as abdominal pain, tenderness, distention, bilious or projectile vomiting, hypoactive bowel sounds, and anorexia postoperatively.
• Complications of SMA syndrome can potentially lead to aspiration pneumonia, acute gastric rupture, or cardiovascular collapse and death.

Superior mesenteric artery (SMA) syndrome resulting from surgical treatment of scoliosis has been recognized in the medical literature since 1752.¹ Throughout the literature, SMA syndrome variably has been referred to as cast syndrome, Wilkie syndrome, arteriomesenteric duodenal obstruction, and chronic duodenal ileus.² We now recognize numerous etiologies of SMA syndrome, as several sources can externally compress the duodenum. Classic acute symptoms of bowel obstruction include bilious vomiting, nausea, and epigastric pain. Chronic manifestations of SMA syndrome may include weight loss and decreased appetite. Our literature review revealed that adolescent growth spurt, height-to-weight ratio, and perioperative weight loss are risk factors associated with SMA syndrome after pediatric spine surgery.

We report the case of a 14-year-old boy who developed SMA syndrome after undergoing scoliosis surgery. The patient and his mother provided written informed consent for print and electronic publication of this case report.

Case Report

A 14-year-old boy with a history of idiopathic scoliosis presented to Cohen Children’s Hospital (Long Island Jewish Medical Center) with bilious vomiting that had persisted for 7 days after posterior T9–L4 fusion with instrumentation.

Discussion

SMA syndrome is attributed to the anatomical orientation of the third part of the duodenum, which passes between the aorta and the SMA (Figure 4).

Adolescents are particularly vulnerable to this condition. Faster adolescent bone growth relative to visceral growth is accompanied by a decrease in SMA angle.³ Occasionally, body casts are used after surgery to immobilize the vertebrae and augment healing. Cast syndrome occurs when pressure from a body cast causes a bowel obstruction secondary to spinal hyperextension and amplified spinal lordosis.² This finding, dating to the 19th century, was reported by Willet⁴ when a patient died 48 hours after application of a body cast. In 1950, the term cast syndrome was coined after a motorcyclist’s injuries were treated with a hip spica cast and the patient died of cardiovascular collapse secondary to persistent vomiting.⁵

Table 1 summarizes various evaluation, diagnosis, and treatment algorithms designed to optimize nutrition and weight in patients developing signs and symptoms of SMA syndrome after posterior spinal instrumentation and fusion for adolescent idiopathic scoliosis (AIS).

Paraneoplastic palmoplantar keratoderma (PPK) is an acquired dermatosis that presents with hyperkeratosis of the palms and soles in association with visceral malignancies, such as esophageal, gastric, pulmonary, and bladder carcinomas. This condition may either be acquired or inherited.¹

Case Report

A 72-year-old woman was referred to our dermatology clinic for evaluation of a nonpruritic hyperkeratotic eruption predominantly on the palms and soles of 2 to 3 months’ duration (Figure 1A). Review of systems was remarkable for chronic anxiety, unintentional weight loss of 10 lb over the last 6 months, and a mild cough of 10 days’ duration. The differential diagnosis included eczematous dermatitis, tinea manuum, new-onset palmoplantar psoriasis, and PPK.

A punch biopsy of the medial hypothenar eminence of the left hand was performed, revealing notable lichenified hyperkeratosis with vascular ectasia (Figure 2). Periodic acid–Schiff staining was negative for fungal elements. Given the suspicion of PPK, multiple carcinoma markers were ordered. Cancer antigen 125 measured at 68 U/mL (reference range upper limit, 21 U/mL). Cancer antigen 27-29 was 50 U/mL (reference range, <38 U/mL) and cancer antigen 19-9 was 24 U/mL (reference range, <37 U/mL). Computed tomography of the chest revealed a large mass in the left lower lung associated with hilar lymphadenopathy. The patient was referred to oncology for further evaluation. Computed tomography–guided biopsy revealed metastatic uterine adenocarcinoma, which prompted subsequent chemotherapy. The combination of visceral malignancy with PPK led to the diagnosis of acquired PPK secondary to uterine cancer. After the completion of chemotherapy, the palmar dermatosis notably decreased (Figure 1B).

Comment

Paraneoplastic PPK is not uncommon. Ninety percent of acquired diffuse PPK is secondary to cancer,² which occurs more frequently in male patients. Associated visceral malignancies include localized esophageal,³ myeloma,⁴ pulmonary, urinary/bladder,⁵ and gastric carcinoma.⁶ Paraneoplastic PPK in women is rare but has been linked to ovarian and breast carcinoma.⁷

The findings under light microscopy include thickening of any or all of the cell layers of the epidermis, which can include hyperkeratosis, acanthosis, and papillomatosis (Figure 2). A moderate amount of mononuclear cell infiltrates also can be visualized.

Palmoplantar keratoderma associated with uterine malignancy is rare. However, many other paraneoplastic dermatoses resulting from uterine cancer have been described as well as nonuterine gynecological malignancies (Table).^8-17

The first step in managing acquired PPK is to determine its etiology via a complete history and a total-body skin examination. If findings are consistent with a hereditary PPK, then genetic workup is advised. Other suspected etiologies should be investigated via imaging and laboratory analysis.¹⁸

The first approach in managing acquired PPK is to treat the underlying cause. In prior cases, complete resolution of skin findings resulted once the malignancy or associated dermatosis had been treated.^8-17 Adjunctive medication includes topical keratolytics (eg, urea, salicylic acid, lactic acid), topical retinoids, topical psoralen plus UVA, and topical corticosteroids.¹⁸ Vitamin A analogues have been found to be an effective treatment of many hyperkeratotic dermatoses.¹⁹ Isotretinoin and etretinate have been used to treat the cutaneous findings and prevent the onset and progression of esophageal malignancy of the inherited forms of PPK. The oral retinoid acitretin has been shown to rapidly resolve lesions, have persistent effects after 5 months of cessation, and have minimal side effects. Thus, it has been suggested as the first-line treatment of chronic PPK.¹⁹ One study found no response to topical keratolytics (urea cream and salicylic acid ointment) and a 2-week course of oral prednisone; however, low-dose oral acitretin 10 mg once daily resulted in notable improvement over several weeks.⁷ Physical debridement also may be necessary.¹⁸

Conclusion

Palmoplantar keratoderma is a condition that presents with hyperkeratosis of the palms and soles. Acquired PPK often occurs as a paraneoplastic response as well as a stigma of other dermatoses. It occurs more frequently in male patients. Reports of PPK secondary to uterine cancer are not common in the literature. Management of PPK includes a complete history and total-body skin examination. After appropriate imaging and laboratory analysis, treatment of the underlying cause is the best approach. Adjunctive medications include topical keratolytics, topical retinoids, topical psoralen plus UVA, and topical corticosteroids. Oral isotretinoin and etretinate have demonstrated promising results.

Paraneoplastic palmoplantar keratoderma (PPK) is an acquired dermatosis that presents with hyperkeratosis of the palms and soles in association with visceral malignancies, such as esophageal, gastric, pulmonary, and bladder carcinomas. This condition may either be acquired or inherited.¹

Case Report

A 72-year-old woman was referred to our dermatology clinic for evaluation of a nonpruritic hyperkeratotic eruption predominantly on the palms and soles of 2 to 3 months’ duration (Figure 1A). Review of systems was remarkable for chronic anxiety, unintentional weight loss of 10 lb over the last 6 months, and a mild cough of 10 days’ duration. The differential diagnosis included eczematous dermatitis, tinea manuum, new-onset palmoplantar psoriasis, and PPK.

A punch biopsy of the medial hypothenar eminence of the left hand was performed, revealing notable lichenified hyperkeratosis with vascular ectasia (Figure 2). Periodic acid–Schiff staining was negative for fungal elements. Given the suspicion of PPK, multiple carcinoma markers were ordered. Cancer antigen 125 measured at 68 U/mL (reference range upper limit, 21 U/mL). Cancer antigen 27-29 was 50 U/mL (reference range, <38 U/mL) and cancer antigen 19-9 was 24 U/mL (reference range, <37 U/mL). Computed tomography of the chest revealed a large mass in the left lower lung associated with hilar lymphadenopathy. The patient was referred to oncology for further evaluation. Computed tomography–guided biopsy revealed metastatic uterine adenocarcinoma, which prompted subsequent chemotherapy. The combination of visceral malignancy with PPK led to the diagnosis of acquired PPK secondary to uterine cancer. After the completion of chemotherapy, the palmar dermatosis notably decreased (Figure 1B).

Comment

Paraneoplastic PPK is not uncommon. Ninety percent of acquired diffuse PPK is secondary to cancer,² which occurs more frequently in male patients. Associated visceral malignancies include localized esophageal,³ myeloma,⁴ pulmonary, urinary/bladder,⁵ and gastric carcinoma.⁶ Paraneoplastic PPK in women is rare but has been linked to ovarian and breast carcinoma.⁷

The findings under light microscopy include thickening of any or all of the cell layers of the epidermis, which can include hyperkeratosis, acanthosis, and papillomatosis (Figure 2). A moderate amount of mononuclear cell infiltrates also can be visualized.

Palmoplantar keratoderma associated with uterine malignancy is rare. However, many other paraneoplastic dermatoses resulting from uterine cancer have been described as well as nonuterine gynecological malignancies (Table).^8-17

The first step in managing acquired PPK is to determine its etiology via a complete history and a total-body skin examination. If findings are consistent with a hereditary PPK, then genetic workup is advised. Other suspected etiologies should be investigated via imaging and laboratory analysis.¹⁸

The first approach in managing acquired PPK is to treat the underlying cause. In prior cases, complete resolution of skin findings resulted once the malignancy or associated dermatosis had been treated.^8-17 Adjunctive medication includes topical keratolytics (eg, urea, salicylic acid, lactic acid), topical retinoids, topical psoralen plus UVA, and topical corticosteroids.¹⁸ Vitamin A analogues have been found to be an effective treatment of many hyperkeratotic dermatoses.¹⁹ Isotretinoin and etretinate have been used to treat the cutaneous findings and prevent the onset and progression of esophageal malignancy of the inherited forms of PPK. The oral retinoid acitretin has been shown to rapidly resolve lesions, have persistent effects after 5 months of cessation, and have minimal side effects. Thus, it has been suggested as the first-line treatment of chronic PPK.¹⁹ One study found no response to topical keratolytics (urea cream and salicylic acid ointment) and a 2-week course of oral prednisone; however, low-dose oral acitretin 10 mg once daily resulted in notable improvement over several weeks.⁷ Physical debridement also may be necessary.¹⁸

Conclusion

Palmoplantar keratoderma is a condition that presents with hyperkeratosis of the palms and soles. Acquired PPK often occurs as a paraneoplastic response as well as a stigma of other dermatoses. It occurs more frequently in male patients. Reports of PPK secondary to uterine cancer are not common in the literature. Management of PPK includes a complete history and total-body skin examination. After appropriate imaging and laboratory analysis, treatment of the underlying cause is the best approach. Adjunctive medications include topical keratolytics, topical retinoids, topical psoralen plus UVA, and topical corticosteroids. Oral isotretinoin and etretinate have demonstrated promising results.

Paraneoplastic palmoplantar keratoderma (PPK) is an acquired dermatosis that presents with hyperkeratosis of the palms and soles in association with visceral malignancies, such as esophageal, gastric, pulmonary, and bladder carcinomas. This condition may either be acquired or inherited.¹

Case Report

A 72-year-old woman was referred to our dermatology clinic for evaluation of a nonpruritic hyperkeratotic eruption predominantly on the palms and soles of 2 to 3 months’ duration (Figure 1A). Review of systems was remarkable for chronic anxiety, unintentional weight loss of 10 lb over the last 6 months, and a mild cough of 10 days’ duration. The differential diagnosis included eczematous dermatitis, tinea manuum, new-onset palmoplantar psoriasis, and PPK.

A punch biopsy of the medial hypothenar eminence of the left hand was performed, revealing notable lichenified hyperkeratosis with vascular ectasia (Figure 2). Periodic acid–Schiff staining was negative for fungal elements. Given the suspicion of PPK, multiple carcinoma markers were ordered. Cancer antigen 125 measured at 68 U/mL (reference range upper limit, 21 U/mL). Cancer antigen 27-29 was 50 U/mL (reference range, <38 U/mL) and cancer antigen 19-9 was 24 U/mL (reference range, <37 U/mL). Computed tomography of the chest revealed a large mass in the left lower lung associated with hilar lymphadenopathy. The patient was referred to oncology for further evaluation. Computed tomography–guided biopsy revealed metastatic uterine adenocarcinoma, which prompted subsequent chemotherapy. The combination of visceral malignancy with PPK led to the diagnosis of acquired PPK secondary to uterine cancer. After the completion of chemotherapy, the palmar dermatosis notably decreased (Figure 1B).

Comment

Paraneoplastic PPK is not uncommon. Ninety percent of acquired diffuse PPK is secondary to cancer,² which occurs more frequently in male patients. Associated visceral malignancies include localized esophageal,³ myeloma,⁴ pulmonary, urinary/bladder,⁵ and gastric carcinoma.⁶ Paraneoplastic PPK in women is rare but has been linked to ovarian and breast carcinoma.⁷

The findings under light microscopy include thickening of any or all of the cell layers of the epidermis, which can include hyperkeratosis, acanthosis, and papillomatosis (Figure 2). A moderate amount of mononuclear cell infiltrates also can be visualized.

Palmoplantar keratoderma associated with uterine malignancy is rare. However, many other paraneoplastic dermatoses resulting from uterine cancer have been described as well as nonuterine gynecological malignancies (Table).^8-17

The first step in managing acquired PPK is to determine its etiology via a complete history and a total-body skin examination. If findings are consistent with a hereditary PPK, then genetic workup is advised. Other suspected etiologies should be investigated via imaging and laboratory analysis.¹⁸

The first approach in managing acquired PPK is to treat the underlying cause. In prior cases, complete resolution of skin findings resulted once the malignancy or associated dermatosis had been treated.^8-17 Adjunctive medication includes topical keratolytics (eg, urea, salicylic acid, lactic acid), topical retinoids, topical psoralen plus UVA, and topical corticosteroids.¹⁸ Vitamin A analogues have been found to be an effective treatment of many hyperkeratotic dermatoses.¹⁹ Isotretinoin and etretinate have been used to treat the cutaneous findings and prevent the onset and progression of esophageal malignancy of the inherited forms of PPK. The oral retinoid acitretin has been shown to rapidly resolve lesions, have persistent effects after 5 months of cessation, and have minimal side effects. Thus, it has been suggested as the first-line treatment of chronic PPK.¹⁹ One study found no response to topical keratolytics (urea cream and salicylic acid ointment) and a 2-week course of oral prednisone; however, low-dose oral acitretin 10 mg once daily resulted in notable improvement over several weeks.⁷ Physical debridement also may be necessary.¹⁸

Conclusion

Palmoplantar keratoderma is a condition that presents with hyperkeratosis of the palms and soles. Acquired PPK often occurs as a paraneoplastic response as well as a stigma of other dermatoses. It occurs more frequently in male patients. Reports of PPK secondary to uterine cancer are not common in the literature. Management of PPK includes a complete history and total-body skin examination. After appropriate imaging and laboratory analysis, treatment of the underlying cause is the best approach. Adjunctive medications include topical keratolytics, topical retinoids, topical psoralen plus UVA, and topical corticosteroids. Oral isotretinoin and etretinate have demonstrated promising results.

Take-Home Points

• Nail breakage is a known complication of intramedullary nail (IMN) fixation of tibial fractures.
• Several techniques have been described for broken IMN extraction.

Intramedullary nail (IMN) fixation is reliably used to manage tibial fractures and has become very popular for managing fractures of varying complexity.^1-4 An occasional complication of intramedullary nailing is nail breakage,^5-7 which can result from a fatigue fracture (from excessive fracture site instability caused by inadequate nail diameter, delayed fracture healing, or fracture nonunion) and direct traumatic impact.^5-7 Several case reports have described unique methods used to facilitate removal of broken hollow and solid IMNs from tibias and femurs.^4,8-16 In this article, we describe an efficient technique for extracting broken tibial IMNs—a technique that can be used before attempting more invasive extraction methods. The patient provided written informed consent for print and electronic publication of this case report.

Case Report and Surgical Technique

A 34-year-old male logger presented to our facility (Department of Orthopaedics, Warren Alpert School of Medicine, Brown University) with a new fracture of the left tibia and fibula with an associated broken IMN after a tree fell on his leg at work (Figures 1A, 1B).

The original IMN had been placed through a paramedian incision, with lateral to medial distal locking screws. The tibial shaft fracture and broken nail were displaced in the coronal plane (Figures 1A, 1B). For restoration of the central canal of the nail, closed reduction was performed in the operating room (Figure 2A). Once the fracture was reduced, the more proximal of the 2 distal interlocking screws was partially backed out so the extraction hook could be passed antegrade into the distal segment of the nail (Figure 2A).

Discussion

IMN fixation of tibial fractures is reliable.^1-4 An occasional complication of intramedullary nailing is nail breakage. Several case reports have described unique methods used to facilitate removal of broken hollow and solid IMNs from knees and femurs.^4,8-16

Our patient’s case involved a cannulated tibial IMN that broke secondary to an acute traumatic event. Several techniques have been used to remove the distal segment of broken cannulated tibial IMNs.^8,9,14,17 Abdelgawad and Kanlic⁸ described a technique in which a small distractor hook was introduced past the distal end of the broken distal piece, and a small (~2 in) piece of flexible nail was introduced into the slot of the distal interlocking screw hole. The hook was pulled back and became incarcerated in the nail by the flexible nail piece, allowing the hook to extract the distal segment of the nail.

Charnley and Farrington⁹ used Petelin laparoscopic grasping forceps to extract the distal segment of a broken cannulated tibial IMN under fluoroscopic guidance. This tibial canal was initially reamed before inserting the instrument and removing the distal segment of the nail.

Levine and Georgiadis¹⁴ used a 4.5-mm bit to drill a hole in the distal aspect of the medial malleolus. A smooth Steinmann pin was used to engage the tip of the IMN. The nail was hammered several centimeters up the medullary canal of the tibia. A 3.0-mm ball-tipped guide wire was inserted in the hole in the medial malleolus and advanced through the distal aspect of the nail under fluoroscopic guidance. The guide wire was advanced through the extent of the nail proximally until it emerged through the knee incision. The distal segment of the broken nail was extracted with the guide wire; the end of the guide wire with the ball engaged the distal aspect of the nail.

Our technique allowed us to use a nail extraction device to extract the distal segment of a broken tibial IMN. This device is usually on hand for routine nail extraction. We used the more distal of the 2 distal interlocking screws to push the extraction hook over the distal lip of the nail, allowing for extraction without additional incisions or additional drill holes in bone. Our technique was efficient in this particular situation and avoided more time-consuming extraction methods. In cases in which the extraction hook does not engage the distal aspect of the nail secondary to bone ingrowth, our technique should be used before attempting other extraction methods.

Am J Orthop. 2017;46(2):E112-E115. Copyright Frontline Medical Communications Inc. 2017. All rights reserved.

Take-Home Points

• Nail breakage is a known complication of intramedullary nail (IMN) fixation of tibial fractures.
• Several techniques have been described for broken IMN extraction.

Intramedullary nail (IMN) fixation is reliably used to manage tibial fractures and has become very popular for managing fractures of varying complexity.^1-4 An occasional complication of intramedullary nailing is nail breakage,^5-7 which can result from a fatigue fracture (from excessive fracture site instability caused by inadequate nail diameter, delayed fracture healing, or fracture nonunion) and direct traumatic impact.^5-7 Several case reports have described unique methods used to facilitate removal of broken hollow and solid IMNs from tibias and femurs.^4,8-16 In this article, we describe an efficient technique for extracting broken tibial IMNs—a technique that can be used before attempting more invasive extraction methods. The patient provided written informed consent for print and electronic publication of this case report.

Case Report and Surgical Technique

A 34-year-old male logger presented to our facility (Department of Orthopaedics, Warren Alpert School of Medicine, Brown University) with a new fracture of the left tibia and fibula with an associated broken IMN after a tree fell on his leg at work (Figures 1A, 1B).

The original IMN had been placed through a paramedian incision, with lateral to medial distal locking screws. The tibial shaft fracture and broken nail were displaced in the coronal plane (Figures 1A, 1B). For restoration of the central canal of the nail, closed reduction was performed in the operating room (Figure 2A). Once the fracture was reduced, the more proximal of the 2 distal interlocking screws was partially backed out so the extraction hook could be passed antegrade into the distal segment of the nail (Figure 2A).

Discussion

IMN fixation of tibial fractures is reliable.^1-4 An occasional complication of intramedullary nailing is nail breakage. Several case reports have described unique methods used to facilitate removal of broken hollow and solid IMNs from knees and femurs.^4,8-16

Our patient’s case involved a cannulated tibial IMN that broke secondary to an acute traumatic event. Several techniques have been used to remove the distal segment of broken cannulated tibial IMNs.^8,9,14,17 Abdelgawad and Kanlic⁸ described a technique in which a small distractor hook was introduced past the distal end of the broken distal piece, and a small (~2 in) piece of flexible nail was introduced into the slot of the distal interlocking screw hole. The hook was pulled back and became incarcerated in the nail by the flexible nail piece, allowing the hook to extract the distal segment of the nail.

Charnley and Farrington⁹ used Petelin laparoscopic grasping forceps to extract the distal segment of a broken cannulated tibial IMN under fluoroscopic guidance. This tibial canal was initially reamed before inserting the instrument and removing the distal segment of the nail.

Levine and Georgiadis¹⁴ used a 4.5-mm bit to drill a hole in the distal aspect of the medial malleolus. A smooth Steinmann pin was used to engage the tip of the IMN. The nail was hammered several centimeters up the medullary canal of the tibia. A 3.0-mm ball-tipped guide wire was inserted in the hole in the medial malleolus and advanced through the distal aspect of the nail under fluoroscopic guidance. The guide wire was advanced through the extent of the nail proximally until it emerged through the knee incision. The distal segment of the broken nail was extracted with the guide wire; the end of the guide wire with the ball engaged the distal aspect of the nail.

Our technique allowed us to use a nail extraction device to extract the distal segment of a broken tibial IMN. This device is usually on hand for routine nail extraction. We used the more distal of the 2 distal interlocking screws to push the extraction hook over the distal lip of the nail, allowing for extraction without additional incisions or additional drill holes in bone. Our technique was efficient in this particular situation and avoided more time-consuming extraction methods. In cases in which the extraction hook does not engage the distal aspect of the nail secondary to bone ingrowth, our technique should be used before attempting other extraction methods.

Am J Orthop. 2017;46(2):E112-E115. Copyright Frontline Medical Communications Inc. 2017. All rights reserved.

Take-Home Points

• Nail breakage is a known complication of intramedullary nail (IMN) fixation of tibial fractures.
• Several techniques have been described for broken IMN extraction.

Intramedullary nail (IMN) fixation is reliably used to manage tibial fractures and has become very popular for managing fractures of varying complexity.^1-4 An occasional complication of intramedullary nailing is nail breakage,^5-7 which can result from a fatigue fracture (from excessive fracture site instability caused by inadequate nail diameter, delayed fracture healing, or fracture nonunion) and direct traumatic impact.^5-7 Several case reports have described unique methods used to facilitate removal of broken hollow and solid IMNs from tibias and femurs.^4,8-16 In this article, we describe an efficient technique for extracting broken tibial IMNs—a technique that can be used before attempting more invasive extraction methods. The patient provided written informed consent for print and electronic publication of this case report.

Case Report and Surgical Technique

A 34-year-old male logger presented to our facility (Department of Orthopaedics, Warren Alpert School of Medicine, Brown University) with a new fracture of the left tibia and fibula with an associated broken IMN after a tree fell on his leg at work (Figures 1A, 1B).

The original IMN had been placed through a paramedian incision, with lateral to medial distal locking screws. The tibial shaft fracture and broken nail were displaced in the coronal plane (Figures 1A, 1B). For restoration of the central canal of the nail, closed reduction was performed in the operating room (Figure 2A). Once the fracture was reduced, the more proximal of the 2 distal interlocking screws was partially backed out so the extraction hook could be passed antegrade into the distal segment of the nail (Figure 2A).

Discussion

IMN fixation of tibial fractures is reliable.^1-4 An occasional complication of intramedullary nailing is nail breakage. Several case reports have described unique methods used to facilitate removal of broken hollow and solid IMNs from knees and femurs.^4,8-16

Our patient’s case involved a cannulated tibial IMN that broke secondary to an acute traumatic event. Several techniques have been used to remove the distal segment of broken cannulated tibial IMNs.^8,9,14,17 Abdelgawad and Kanlic⁸ described a technique in which a small distractor hook was introduced past the distal end of the broken distal piece, and a small (~2 in) piece of flexible nail was introduced into the slot of the distal interlocking screw hole. The hook was pulled back and became incarcerated in the nail by the flexible nail piece, allowing the hook to extract the distal segment of the nail.

Charnley and Farrington⁹ used Petelin laparoscopic grasping forceps to extract the distal segment of a broken cannulated tibial IMN under fluoroscopic guidance. This tibial canal was initially reamed before inserting the instrument and removing the distal segment of the nail.

Levine and Georgiadis¹⁴ used a 4.5-mm bit to drill a hole in the distal aspect of the medial malleolus. A smooth Steinmann pin was used to engage the tip of the IMN. The nail was hammered several centimeters up the medullary canal of the tibia. A 3.0-mm ball-tipped guide wire was inserted in the hole in the medial malleolus and advanced through the distal aspect of the nail under fluoroscopic guidance. The guide wire was advanced through the extent of the nail proximally until it emerged through the knee incision. The distal segment of the broken nail was extracted with the guide wire; the end of the guide wire with the ball engaged the distal aspect of the nail.

Our technique allowed us to use a nail extraction device to extract the distal segment of a broken tibial IMN. This device is usually on hand for routine nail extraction. We used the more distal of the 2 distal interlocking screws to push the extraction hook over the distal lip of the nail, allowing for extraction without additional incisions or additional drill holes in bone. Our technique was efficient in this particular situation and avoided more time-consuming extraction methods. In cases in which the extraction hook does not engage the distal aspect of the nail secondary to bone ingrowth, our technique should be used before attempting other extraction methods.

Am J Orthop. 2017;46(2):E112-E115. Copyright Frontline Medical Communications Inc. 2017. All rights reserved.

THE CASE

A 30-year-old woman presented to our emergency department with severe polyarthralgia, a high-grade fever (102.6º F), and a diffuse maculopapular rash on her trunk and extremities. She had returned from her honeymoon in Jamaica 6 days earlier. During her time there, she ate local cuisine, hiked in the jungle, and was bitten by many mosquitoes. The patient was nauseous, and had been experiencing headaches, generalized weakness, and fatigue. Her physical exam revealed a maculopapular rash on her trunk and upper extremities. She had tenderness and pain, as well as decreased range of motion in her ankles, knees, and wrists. The patient had no erythema, swelling, petechiae, or bruising. She had a past medical history of Graves’ disease and had received all of her childhood immunizations.

THE DIAGNOSIS

Our lab work-up included a complete blood count, liver function tests, blood pathogens for malaria, and serologic tests for dengue fever, measles, mumps, rubella, Lyme disease, human immunodeficiency virus (HIV), and parvovirus. We ruled out dengue fever because the patient had no evidence of hemorrhage, thrombocytopenia, rising hematocrit, or neutropenia. HIV antibody screening tests were performed, although, in retrospect, confirmatory HIV quantitative RNA testing should’ve been obtained because of the acute nature of the patient’s symptoms. Regardless, the work-up was negative, with the exception of a positive parvovirus immunoglobulin G (IgG).

Given our patient’s travel history, unremarkable lab results, and physical exam (notably the rash and joint pain), we suspected that she was infected with the chikungunya virus and tested for it. The results of chikungunya serum titers returned 13 days later and were positive for both immunoglobulin M (IgM) and IgG, confirming our suspicion.

DISCUSSION

Chikungunya is a viral infection that is most commonly transmitted to humans via mosquitoes. The infection was first identified in West Africa in the mid-1900s and predominantly occurs in tropical and subtropical regions due to the numbers of mosquitoes in those areas. Since 2000, outbreaks have been most common in Africa and Asia, with the largest outbreaks occurring from 2005 to 2006.^1,2 Chikungunya has since become more widespread in the Indian Ocean islands, and in 2013, it was first identified in the Caribbean islands. As of March 2017, over one million cases have been reported in the Americas, according to the Pan American Health Organization.³ In 2014, 12 locally transmitted cases were reported in Florida.⁴

The most common mosquito vectors for chikungunya are the Aedes albopictus (Asian tiger mosquito) and Aedes aegypti mosquitoes. These mosquitoes also transmit dengue fever, yellow fever, and Zika virus. Both of these mosquitoes are well-adapted to urban areas and can breed in standing water. A aegypti mosquitoes are found only in the southern United States, while A albopictus mosquitoes can be found in more temperate climates—areas like New York, New Jersey, and Pennsylvania.⁴ Both species are daytime biters, so their activity peaks during the dawn and dusk periods. Because local mosquito vectors exist as far north as New York, local transmission and outbreaks are possible in many parts of the United States.

When to suspect chikungunya, and what to look for

Suspect chikungunya in patients returning from endemic areas. After a 3- to 7-day incubation period, the clinical presentation of chikungunya typically begins with fever and malaise, followed by polyarthralgia that starts 2 to 5 days after the onset of fever. Headache, nausea, and conjunctivitis may also occur. Polyarthralgia and arthritis usually present in symmetrical distal joints and are accompanied by face and trunk flushing that is followed by a maculopapular rash. The rash predominantly appears on the trunk and limbs, but can also occur on the face, palms, and soles. Tendons and ligaments—especially the Achilles tendon—may become inflamed, as well. Symptoms typically resolve within 2 to 3 weeks, although polyarthralgia may last for months or even years.

The clinical presentation of chikungunya typically begins with fever and malaise, followed by polyarthralgia that starts 2 to 5 days after the onset of fever.

There is no known risk of transmission through breast milk or in utero, although vertical transmission through vaginal or cesarean birth is common in viremic women. Blood-borne transmission can occur in the laboratory, nosocomially, or through the transfusion of blood products if exposure occurs during the early viremic phase.^1,2 Dual infections are possible (typically with yellow fever, malaria, Zika virus, or dengue fever) and should be considered based on the patient’s travel history.⁵ Abnormal laboratory findings are less common, but may include lymphopenia (most common), thrombocytopenia, elevations in blood urea nitrogen and creatinine (indicating an acute kidney injury), and elevated liver transaminases.

Chikungunya is generally considered a self-limiting disease, but severe atypical manifestations can lead to meningoencephalitis, respiratory failure, and even death. Severe disease is more commonly seen in infants, patients over age 65 years, and in those with chronic medical conditions.

The differential diagnosis for chikungunya virus includes dengue fever, Zika virus, malaria, measles, rubella, parvovirus, primary HIV infection, Lyme disease, and other inflammatory joint conditions. The differential depends on where a patient lives, their travel history, and exposures.

Dengue and chikungunya have similar features, which often make them difficult to distinguish. However, patients with dengue fever present more often with neutropenia, thrombocytopenia, and signs or symptoms of shock or hemorrhage.⁶ The chikungunya rash is typically a maculopapular rash on the trunk, hands, and feet that appears within the first 2 days of illness, as opposed to dengue, which has a similar rash, but appears later in the disease (Days 2-5). Avoid nonsteroidal anti-inflammatory drugs (NSAIDs) if dengue fever is suspected, as they can worsen hemorrhaging.⁷

Chikungunya is becoming more common among travelers returning to the United States from the Caribbean islands.

Zika virus typically presents with milder symptoms compared to chikungunya. Patients may have a skin rash and occasionally, conjunctivitis, but limited high fevers or joint pain. Zika rash is maculopapular, but typically starts on the face on the first day of the illness. Zika has been associated with neurological complications such as Guillain-Barré syndrome and microcephaly in fetuses of infected pregnant woman.⁸

Choice of testing modality depends on when symptoms began

Laboratory diagnosis of chikungunya can be accomplished 3 ways: viral culture, reverse transcriptase-polymerase chain reaction (RT-PCR) viral RNA, and serology.¹ The choice of which modality to use depends on the time between the onset of symptoms and the date on which a serum sample is drawn.

• If the patient presents within the first 3 days of illness, viral culture can detect chikungunya. Chikungunya virus testing is available through the Centers for Disease Control and Prevention (CDC), some state laboratories, and one commercial lab.⁸ Viral cultures are considered the gold standard for diagnosis, but a requirement for an elevated biosafety level, as well as a longer incubation time, make them less useful in the clinical setting.^1,9
• If symptoms started less than 5 to 8 days prior, serum should be sent for RT-PCR for viral RNA.
• If symptoms occurred more than 8 days prior, serum should be sent for IgM and IgG serologic testing.

If the acute sample was negative (and chikungunya is still suspected), a second serum sample should be drawn 2 to 3 weeks later during the convalescent phase and sent for IgG serologic testing.

There are no specific treatments or vaccines available for chikungunya, but both live and inactivated vaccines are being tested. To prevent chikungunya virus infection, advise patients traveling to endemic areas to reduce mosquito exposure by avoiding outdoor activities during dusk and dawn, wearing long-sleeved shirts and long pants, and using an insect repellent that contains DEET. Recommend that patients who will be sleeping in a high-risk area use bed netting treated with permethrin. The CDC Web site has excellent additional information, available at http://www.cdc.gov/chikungunya.¹⁰

Our patient was briefly hospitalized due to intractable pain. She was discharged after 2 days and given prescriptions for motrin 800 mg and percocet 5/325 mg for breakthrough pain. Her wrist and ankle pain persisted after discharge, but slowly resolved after 2 to 3 months.

THE TAKEAWAY

Chikungunya is becoming more common among travelers returning to the United States from the Caribbean islands, and mosquito vectors found in parts of the United States are enhancing the possibility of local outbreaks. Travel to endemic regions—and the classic symptoms of fever, polyarthralgia, and a maculopapular rash—should lead you to put chikungunya high on the differential. Chikungunya is considered a reportable disease and treatment is largely supportive. Co-infections are possible and should be considered based on the patient’s travel history.

THE CASE

A 30-year-old woman presented to our emergency department with severe polyarthralgia, a high-grade fever (102.6º F), and a diffuse maculopapular rash on her trunk and extremities. She had returned from her honeymoon in Jamaica 6 days earlier. During her time there, she ate local cuisine, hiked in the jungle, and was bitten by many mosquitoes. The patient was nauseous, and had been experiencing headaches, generalized weakness, and fatigue. Her physical exam revealed a maculopapular rash on her trunk and upper extremities. She had tenderness and pain, as well as decreased range of motion in her ankles, knees, and wrists. The patient had no erythema, swelling, petechiae, or bruising. She had a past medical history of Graves’ disease and had received all of her childhood immunizations.

THE DIAGNOSIS

Our lab work-up included a complete blood count, liver function tests, blood pathogens for malaria, and serologic tests for dengue fever, measles, mumps, rubella, Lyme disease, human immunodeficiency virus (HIV), and parvovirus. We ruled out dengue fever because the patient had no evidence of hemorrhage, thrombocytopenia, rising hematocrit, or neutropenia. HIV antibody screening tests were performed, although, in retrospect, confirmatory HIV quantitative RNA testing should’ve been obtained because of the acute nature of the patient’s symptoms. Regardless, the work-up was negative, with the exception of a positive parvovirus immunoglobulin G (IgG).

Given our patient’s travel history, unremarkable lab results, and physical exam (notably the rash and joint pain), we suspected that she was infected with the chikungunya virus and tested for it. The results of chikungunya serum titers returned 13 days later and were positive for both immunoglobulin M (IgM) and IgG, confirming our suspicion.

DISCUSSION

Chikungunya is a viral infection that is most commonly transmitted to humans via mosquitoes. The infection was first identified in West Africa in the mid-1900s and predominantly occurs in tropical and subtropical regions due to the numbers of mosquitoes in those areas. Since 2000, outbreaks have been most common in Africa and Asia, with the largest outbreaks occurring from 2005 to 2006.^1,2 Chikungunya has since become more widespread in the Indian Ocean islands, and in 2013, it was first identified in the Caribbean islands. As of March 2017, over one million cases have been reported in the Americas, according to the Pan American Health Organization.³ In 2014, 12 locally transmitted cases were reported in Florida.⁴

The most common mosquito vectors for chikungunya are the Aedes albopictus (Asian tiger mosquito) and Aedes aegypti mosquitoes. These mosquitoes also transmit dengue fever, yellow fever, and Zika virus. Both of these mosquitoes are well-adapted to urban areas and can breed in standing water. A aegypti mosquitoes are found only in the southern United States, while A albopictus mosquitoes can be found in more temperate climates—areas like New York, New Jersey, and Pennsylvania.⁴ Both species are daytime biters, so their activity peaks during the dawn and dusk periods. Because local mosquito vectors exist as far north as New York, local transmission and outbreaks are possible in many parts of the United States.

When to suspect chikungunya, and what to look for

Suspect chikungunya in patients returning from endemic areas. After a 3- to 7-day incubation period, the clinical presentation of chikungunya typically begins with fever and malaise, followed by polyarthralgia that starts 2 to 5 days after the onset of fever. Headache, nausea, and conjunctivitis may also occur. Polyarthralgia and arthritis usually present in symmetrical distal joints and are accompanied by face and trunk flushing that is followed by a maculopapular rash. The rash predominantly appears on the trunk and limbs, but can also occur on the face, palms, and soles. Tendons and ligaments—especially the Achilles tendon—may become inflamed, as well. Symptoms typically resolve within 2 to 3 weeks, although polyarthralgia may last for months or even years.

The clinical presentation of chikungunya typically begins with fever and malaise, followed by polyarthralgia that starts 2 to 5 days after the onset of fever.

There is no known risk of transmission through breast milk or in utero, although vertical transmission through vaginal or cesarean birth is common in viremic women. Blood-borne transmission can occur in the laboratory, nosocomially, or through the transfusion of blood products if exposure occurs during the early viremic phase.^1,2 Dual infections are possible (typically with yellow fever, malaria, Zika virus, or dengue fever) and should be considered based on the patient’s travel history.⁵ Abnormal laboratory findings are less common, but may include lymphopenia (most common), thrombocytopenia, elevations in blood urea nitrogen and creatinine (indicating an acute kidney injury), and elevated liver transaminases.

Chikungunya is generally considered a self-limiting disease, but severe atypical manifestations can lead to meningoencephalitis, respiratory failure, and even death. Severe disease is more commonly seen in infants, patients over age 65 years, and in those with chronic medical conditions.

The differential diagnosis for chikungunya virus includes dengue fever, Zika virus, malaria, measles, rubella, parvovirus, primary HIV infection, Lyme disease, and other inflammatory joint conditions. The differential depends on where a patient lives, their travel history, and exposures.

Dengue and chikungunya have similar features, which often make them difficult to distinguish. However, patients with dengue fever present more often with neutropenia, thrombocytopenia, and signs or symptoms of shock or hemorrhage.⁶ The chikungunya rash is typically a maculopapular rash on the trunk, hands, and feet that appears within the first 2 days of illness, as opposed to dengue, which has a similar rash, but appears later in the disease (Days 2-5). Avoid nonsteroidal anti-inflammatory drugs (NSAIDs) if dengue fever is suspected, as they can worsen hemorrhaging.⁷

Chikungunya is becoming more common among travelers returning to the United States from the Caribbean islands.

Zika virus typically presents with milder symptoms compared to chikungunya. Patients may have a skin rash and occasionally, conjunctivitis, but limited high fevers or joint pain. Zika rash is maculopapular, but typically starts on the face on the first day of the illness. Zika has been associated with neurological complications such as Guillain-Barré syndrome and microcephaly in fetuses of infected pregnant woman.⁸

Choice of testing modality depends on when symptoms began

Laboratory diagnosis of chikungunya can be accomplished 3 ways: viral culture, reverse transcriptase-polymerase chain reaction (RT-PCR) viral RNA, and serology.¹ The choice of which modality to use depends on the time between the onset of symptoms and the date on which a serum sample is drawn.

• If the patient presents within the first 3 days of illness, viral culture can detect chikungunya. Chikungunya virus testing is available through the Centers for Disease Control and Prevention (CDC), some state laboratories, and one commercial lab.⁸ Viral cultures are considered the gold standard for diagnosis, but a requirement for an elevated biosafety level, as well as a longer incubation time, make them less useful in the clinical setting.^1,9
• If symptoms started less than 5 to 8 days prior, serum should be sent for RT-PCR for viral RNA.
• If symptoms occurred more than 8 days prior, serum should be sent for IgM and IgG serologic testing.

If the acute sample was negative (and chikungunya is still suspected), a second serum sample should be drawn 2 to 3 weeks later during the convalescent phase and sent for IgG serologic testing.

There are no specific treatments or vaccines available for chikungunya, but both live and inactivated vaccines are being tested. To prevent chikungunya virus infection, advise patients traveling to endemic areas to reduce mosquito exposure by avoiding outdoor activities during dusk and dawn, wearing long-sleeved shirts and long pants, and using an insect repellent that contains DEET. Recommend that patients who will be sleeping in a high-risk area use bed netting treated with permethrin. The CDC Web site has excellent additional information, available at http://www.cdc.gov/chikungunya.¹⁰

Our patient was briefly hospitalized due to intractable pain. She was discharged after 2 days and given prescriptions for motrin 800 mg and percocet 5/325 mg for breakthrough pain. Her wrist and ankle pain persisted after discharge, but slowly resolved after 2 to 3 months.

THE TAKEAWAY

Chikungunya is becoming more common among travelers returning to the United States from the Caribbean islands, and mosquito vectors found in parts of the United States are enhancing the possibility of local outbreaks. Travel to endemic regions—and the classic symptoms of fever, polyarthralgia, and a maculopapular rash—should lead you to put chikungunya high on the differential. Chikungunya is considered a reportable disease and treatment is largely supportive. Co-infections are possible and should be considered based on the patient’s travel history.

THE CASE

A 30-year-old woman presented to our emergency department with severe polyarthralgia, a high-grade fever (102.6º F), and a diffuse maculopapular rash on her trunk and extremities. She had returned from her honeymoon in Jamaica 6 days earlier. During her time there, she ate local cuisine, hiked in the jungle, and was bitten by many mosquitoes. The patient was nauseous, and had been experiencing headaches, generalized weakness, and fatigue. Her physical exam revealed a maculopapular rash on her trunk and upper extremities. She had tenderness and pain, as well as decreased range of motion in her ankles, knees, and wrists. The patient had no erythema, swelling, petechiae, or bruising. She had a past medical history of Graves’ disease and had received all of her childhood immunizations.

THE DIAGNOSIS

Our lab work-up included a complete blood count, liver function tests, blood pathogens for malaria, and serologic tests for dengue fever, measles, mumps, rubella, Lyme disease, human immunodeficiency virus (HIV), and parvovirus. We ruled out dengue fever because the patient had no evidence of hemorrhage, thrombocytopenia, rising hematocrit, or neutropenia. HIV antibody screening tests were performed, although, in retrospect, confirmatory HIV quantitative RNA testing should’ve been obtained because of the acute nature of the patient’s symptoms. Regardless, the work-up was negative, with the exception of a positive parvovirus immunoglobulin G (IgG).

Given our patient’s travel history, unremarkable lab results, and physical exam (notably the rash and joint pain), we suspected that she was infected with the chikungunya virus and tested for it. The results of chikungunya serum titers returned 13 days later and were positive for both immunoglobulin M (IgM) and IgG, confirming our suspicion.

DISCUSSION

Chikungunya is a viral infection that is most commonly transmitted to humans via mosquitoes. The infection was first identified in West Africa in the mid-1900s and predominantly occurs in tropical and subtropical regions due to the numbers of mosquitoes in those areas. Since 2000, outbreaks have been most common in Africa and Asia, with the largest outbreaks occurring from 2005 to 2006.^1,2 Chikungunya has since become more widespread in the Indian Ocean islands, and in 2013, it was first identified in the Caribbean islands. As of March 2017, over one million cases have been reported in the Americas, according to the Pan American Health Organization.³ In 2014, 12 locally transmitted cases were reported in Florida.⁴

The most common mosquito vectors for chikungunya are the Aedes albopictus (Asian tiger mosquito) and Aedes aegypti mosquitoes. These mosquitoes also transmit dengue fever, yellow fever, and Zika virus. Both of these mosquitoes are well-adapted to urban areas and can breed in standing water. A aegypti mosquitoes are found only in the southern United States, while A albopictus mosquitoes can be found in more temperate climates—areas like New York, New Jersey, and Pennsylvania.⁴ Both species are daytime biters, so their activity peaks during the dawn and dusk periods. Because local mosquito vectors exist as far north as New York, local transmission and outbreaks are possible in many parts of the United States.

When to suspect chikungunya, and what to look for

Suspect chikungunya in patients returning from endemic areas. After a 3- to 7-day incubation period, the clinical presentation of chikungunya typically begins with fever and malaise, followed by polyarthralgia that starts 2 to 5 days after the onset of fever. Headache, nausea, and conjunctivitis may also occur. Polyarthralgia and arthritis usually present in symmetrical distal joints and are accompanied by face and trunk flushing that is followed by a maculopapular rash. The rash predominantly appears on the trunk and limbs, but can also occur on the face, palms, and soles. Tendons and ligaments—especially the Achilles tendon—may become inflamed, as well. Symptoms typically resolve within 2 to 3 weeks, although polyarthralgia may last for months or even years.

The clinical presentation of chikungunya typically begins with fever and malaise, followed by polyarthralgia that starts 2 to 5 days after the onset of fever.

There is no known risk of transmission through breast milk or in utero, although vertical transmission through vaginal or cesarean birth is common in viremic women. Blood-borne transmission can occur in the laboratory, nosocomially, or through the transfusion of blood products if exposure occurs during the early viremic phase.^1,2 Dual infections are possible (typically with yellow fever, malaria, Zika virus, or dengue fever) and should be considered based on the patient’s travel history.⁵ Abnormal laboratory findings are less common, but may include lymphopenia (most common), thrombocytopenia, elevations in blood urea nitrogen and creatinine (indicating an acute kidney injury), and elevated liver transaminases.

Chikungunya is generally considered a self-limiting disease, but severe atypical manifestations can lead to meningoencephalitis, respiratory failure, and even death. Severe disease is more commonly seen in infants, patients over age 65 years, and in those with chronic medical conditions.

The differential diagnosis for chikungunya virus includes dengue fever, Zika virus, malaria, measles, rubella, parvovirus, primary HIV infection, Lyme disease, and other inflammatory joint conditions. The differential depends on where a patient lives, their travel history, and exposures.

Dengue and chikungunya have similar features, which often make them difficult to distinguish. However, patients with dengue fever present more often with neutropenia, thrombocytopenia, and signs or symptoms of shock or hemorrhage.⁶ The chikungunya rash is typically a maculopapular rash on the trunk, hands, and feet that appears within the first 2 days of illness, as opposed to dengue, which has a similar rash, but appears later in the disease (Days 2-5). Avoid nonsteroidal anti-inflammatory drugs (NSAIDs) if dengue fever is suspected, as they can worsen hemorrhaging.⁷

Chikungunya is becoming more common among travelers returning to the United States from the Caribbean islands.

Zika virus typically presents with milder symptoms compared to chikungunya. Patients may have a skin rash and occasionally, conjunctivitis, but limited high fevers or joint pain. Zika rash is maculopapular, but typically starts on the face on the first day of the illness. Zika has been associated with neurological complications such as Guillain-Barré syndrome and microcephaly in fetuses of infected pregnant woman.⁸

Choice of testing modality depends on when symptoms began

Laboratory diagnosis of chikungunya can be accomplished 3 ways: viral culture, reverse transcriptase-polymerase chain reaction (RT-PCR) viral RNA, and serology.¹ The choice of which modality to use depends on the time between the onset of symptoms and the date on which a serum sample is drawn.

• If the patient presents within the first 3 days of illness, viral culture can detect chikungunya. Chikungunya virus testing is available through the Centers for Disease Control and Prevention (CDC), some state laboratories, and one commercial lab.⁸ Viral cultures are considered the gold standard for diagnosis, but a requirement for an elevated biosafety level, as well as a longer incubation time, make them less useful in the clinical setting.^1,9
• If symptoms started less than 5 to 8 days prior, serum should be sent for RT-PCR for viral RNA.
• If symptoms occurred more than 8 days prior, serum should be sent for IgM and IgG serologic testing.

If the acute sample was negative (and chikungunya is still suspected), a second serum sample should be drawn 2 to 3 weeks later during the convalescent phase and sent for IgG serologic testing.

There are no specific treatments or vaccines available for chikungunya, but both live and inactivated vaccines are being tested. To prevent chikungunya virus infection, advise patients traveling to endemic areas to reduce mosquito exposure by avoiding outdoor activities during dusk and dawn, wearing long-sleeved shirts and long pants, and using an insect repellent that contains DEET. Recommend that patients who will be sleeping in a high-risk area use bed netting treated with permethrin. The CDC Web site has excellent additional information, available at http://www.cdc.gov/chikungunya.¹⁰

Our patient was briefly hospitalized due to intractable pain. She was discharged after 2 days and given prescriptions for motrin 800 mg and percocet 5/325 mg for breakthrough pain. Her wrist and ankle pain persisted after discharge, but slowly resolved after 2 to 3 months.

THE TAKEAWAY

Chikungunya is becoming more common among travelers returning to the United States from the Caribbean islands, and mosquito vectors found in parts of the United States are enhancing the possibility of local outbreaks. Travel to endemic regions—and the classic symptoms of fever, polyarthralgia, and a maculopapular rash—should lead you to put chikungunya high on the differential. Chikungunya is considered a reportable disease and treatment is largely supportive. Co-infections are possible and should be considered based on the patient’s travel history.

Take-Home Points

• Post-infectious or post-inflammatory pathological knee arthrodesis is one of the most challenging complications in orthopedics.
• It can result in significant patient distress with some struggling to maintain any range of motion for functionality.
• TKA for the correction of knee ankylosis is an option, but not without significant morbidity and failure rates.

Spontaneous knee fusion is an unusual and rarely reported phenomenon. Progressive stiffness is commonly experienced by patients with arthritis. However, most patients maintain some range of knee motion, which may be enhanced with medical treatment, rehabilitation with physiotherapy, and ambulation devices. To our knowledge, this article is the first report of a case of spontaneous and progressive bony fusion of a knee joint without a prior diagnosis of inflammatory or septic arthritis or surgical arthrodesis. The patient provided written informed consent for print and electronic publication of this case report.

Case Report

In 2015, a 51-year-old woman presented to the orthopedics department with a 13-year history of complete loss of left knee flexion. She denied a history of trauma to or surgical intervention for the knee and denied a medical history of inflammatory or septic arthritis.

On initial referral to the department, in 2002, the patient, age 38 years at the time, had a 1-year history of progressive left knee stiffness and reduced range of motion (ROM). At the time, she recalled injuring the knee during an aerobics class 2 months prior. A physiotherapy trial (ROM actively and passively assessed 10°-90°) failed. All movement was painful, and 2 crutches were needed for ambulation. The patient was treated nonoperatively with analgesia and was advised to return to physiotherapy. Plain radiographs showed a small effusion but no bony abnormalities or fractures (Figures 1A, 1B).

Discussion

We have reported a rare case of spontaneous knee fusion in a middle-aged patient with no significant predisposing factors and no clear diagnosis. Serologic results were normal and not significant, but imaging was highly suggestive of an inflammatory process and provided a probable diagnosis of an underlying inflammatory condition and/or infection.

In the literature, there are no other reports of similar cases of spontaneous knee joint fusion, though there are some rare cases of the phenomenon in other joints. In 2005, Budoff and Lichtman¹ reported a case of spontaneous wrist fusion in an 18-year-old patient with a background of Kienböck disease, which may have predisposed the patient to an underlying synovitis progressing to autofusion of the joint. In 2014, Lui² described the case of a 64-year-old woman with spontaneous subtalar fusion complicating a subtalar arthroereisis. Although an extensive literature review on the topic is difficult owing to the rarity of the condition, these few cases, unlike our case, appear to describe a predisposing factor or inciting event.

The reversibility of knee arthrodesis remains an issue in our patient’s case and in other cases, and total knee arthroplasty (TKA) may be the most obvious operative intervention. Cameron and Hu³ reported 17 cases of knee fusion take-down with conversion to TKA, and Kim and colleagues⁴ reported 16 TKAs performed after spontaneous osseous ankylosis and 14 performed after formal knee fusion take-down. Although functional improvements were found in both studies, complication rates were relatively high, at least 53%. Other authors have used TKAs in cases of knee ankylosis after infectious or inflammatory arthritis, but results were suboptimal and unpredictable, and complication rates were 27% and 53.3%.^5,6In this difficult scenario, our middle-aged patient’s fixed extension deformity of the knee, likely the result of an idiopathic process, led to severe debilitation and poor quality of life. To perform a TKA in a 51-year-old patient is far from ideal. The reversibility of formally fused and spontaneously fused knees is still in question, and, though there are reports of relatively satisfactory results, most operative options are fraught with complications.

Am J Orthop. 2017;46(2):E83-E85. Copyright Frontline Medical Communications Inc. 2017. All rights reserved.

Take-Home Points

• Post-infectious or post-inflammatory pathological knee arthrodesis is one of the most challenging complications in orthopedics.
• It can result in significant patient distress with some struggling to maintain any range of motion for functionality.
• TKA for the correction of knee ankylosis is an option, but not without significant morbidity and failure rates.

Spontaneous knee fusion is an unusual and rarely reported phenomenon. Progressive stiffness is commonly experienced by patients with arthritis. However, most patients maintain some range of knee motion, which may be enhanced with medical treatment, rehabilitation with physiotherapy, and ambulation devices. To our knowledge, this article is the first report of a case of spontaneous and progressive bony fusion of a knee joint without a prior diagnosis of inflammatory or septic arthritis or surgical arthrodesis. The patient provided written informed consent for print and electronic publication of this case report.

Case Report

In 2015, a 51-year-old woman presented to the orthopedics department with a 13-year history of complete loss of left knee flexion. She denied a history of trauma to or surgical intervention for the knee and denied a medical history of inflammatory or septic arthritis.

On initial referral to the department, in 2002, the patient, age 38 years at the time, had a 1-year history of progressive left knee stiffness and reduced range of motion (ROM). At the time, she recalled injuring the knee during an aerobics class 2 months prior. A physiotherapy trial (ROM actively and passively assessed 10°-90°) failed. All movement was painful, and 2 crutches were needed for ambulation. The patient was treated nonoperatively with analgesia and was advised to return to physiotherapy. Plain radiographs showed a small effusion but no bony abnormalities or fractures (Figures 1A, 1B).

Discussion

We have reported a rare case of spontaneous knee fusion in a middle-aged patient with no significant predisposing factors and no clear diagnosis. Serologic results were normal and not significant, but imaging was highly suggestive of an inflammatory process and provided a probable diagnosis of an underlying inflammatory condition and/or infection.

In the literature, there are no other reports of similar cases of spontaneous knee joint fusion, though there are some rare cases of the phenomenon in other joints. In 2005, Budoff and Lichtman¹ reported a case of spontaneous wrist fusion in an 18-year-old patient with a background of Kienböck disease, which may have predisposed the patient to an underlying synovitis progressing to autofusion of the joint. In 2014, Lui² described the case of a 64-year-old woman with spontaneous subtalar fusion complicating a subtalar arthroereisis. Although an extensive literature review on the topic is difficult owing to the rarity of the condition, these few cases, unlike our case, appear to describe a predisposing factor or inciting event.

The reversibility of knee arthrodesis remains an issue in our patient’s case and in other cases, and total knee arthroplasty (TKA) may be the most obvious operative intervention. Cameron and Hu³ reported 17 cases of knee fusion take-down with conversion to TKA, and Kim and colleagues⁴ reported 16 TKAs performed after spontaneous osseous ankylosis and 14 performed after formal knee fusion take-down. Although functional improvements were found in both studies, complication rates were relatively high, at least 53%. Other authors have used TKAs in cases of knee ankylosis after infectious or inflammatory arthritis, but results were suboptimal and unpredictable, and complication rates were 27% and 53.3%.^5,6In this difficult scenario, our middle-aged patient’s fixed extension deformity of the knee, likely the result of an idiopathic process, led to severe debilitation and poor quality of life. To perform a TKA in a 51-year-old patient is far from ideal. The reversibility of formally fused and spontaneously fused knees is still in question, and, though there are reports of relatively satisfactory results, most operative options are fraught with complications.

Am J Orthop. 2017;46(2):E83-E85. Copyright Frontline Medical Communications Inc. 2017. All rights reserved.

Take-Home Points

• Post-infectious or post-inflammatory pathological knee arthrodesis is one of the most challenging complications in orthopedics.
• It can result in significant patient distress with some struggling to maintain any range of motion for functionality.
• TKA for the correction of knee ankylosis is an option, but not without significant morbidity and failure rates.

Spontaneous knee fusion is an unusual and rarely reported phenomenon. Progressive stiffness is commonly experienced by patients with arthritis. However, most patients maintain some range of knee motion, which may be enhanced with medical treatment, rehabilitation with physiotherapy, and ambulation devices. To our knowledge, this article is the first report of a case of spontaneous and progressive bony fusion of a knee joint without a prior diagnosis of inflammatory or septic arthritis or surgical arthrodesis. The patient provided written informed consent for print and electronic publication of this case report.

Case Report

In 2015, a 51-year-old woman presented to the orthopedics department with a 13-year history of complete loss of left knee flexion. She denied a history of trauma to or surgical intervention for the knee and denied a medical history of inflammatory or septic arthritis.

On initial referral to the department, in 2002, the patient, age 38 years at the time, had a 1-year history of progressive left knee stiffness and reduced range of motion (ROM). At the time, she recalled injuring the knee during an aerobics class 2 months prior. A physiotherapy trial (ROM actively and passively assessed 10°-90°) failed. All movement was painful, and 2 crutches were needed for ambulation. The patient was treated nonoperatively with analgesia and was advised to return to physiotherapy. Plain radiographs showed a small effusion but no bony abnormalities or fractures (Figures 1A, 1B).

Discussion

We have reported a rare case of spontaneous knee fusion in a middle-aged patient with no significant predisposing factors and no clear diagnosis. Serologic results were normal and not significant, but imaging was highly suggestive of an inflammatory process and provided a probable diagnosis of an underlying inflammatory condition and/or infection.

In the literature, there are no other reports of similar cases of spontaneous knee joint fusion, though there are some rare cases of the phenomenon in other joints. In 2005, Budoff and Lichtman¹ reported a case of spontaneous wrist fusion in an 18-year-old patient with a background of Kienböck disease, which may have predisposed the patient to an underlying synovitis progressing to autofusion of the joint. In 2014, Lui² described the case of a 64-year-old woman with spontaneous subtalar fusion complicating a subtalar arthroereisis. Although an extensive literature review on the topic is difficult owing to the rarity of the condition, these few cases, unlike our case, appear to describe a predisposing factor or inciting event.

The reversibility of knee arthrodesis remains an issue in our patient’s case and in other cases, and total knee arthroplasty (TKA) may be the most obvious operative intervention. Cameron and Hu³ reported 17 cases of knee fusion take-down with conversion to TKA, and Kim and colleagues⁴ reported 16 TKAs performed after spontaneous osseous ankylosis and 14 performed after formal knee fusion take-down. Although functional improvements were found in both studies, complication rates were relatively high, at least 53%. Other authors have used TKAs in cases of knee ankylosis after infectious or inflammatory arthritis, but results were suboptimal and unpredictable, and complication rates were 27% and 53.3%.^5,6In this difficult scenario, our middle-aged patient’s fixed extension deformity of the knee, likely the result of an idiopathic process, led to severe debilitation and poor quality of life. To perform a TKA in a 51-year-old patient is far from ideal. The reversibility of formally fused and spontaneously fused knees is still in question, and, though there are reports of relatively satisfactory results, most operative options are fraught with complications.

Am J Orthop. 2017;46(2):E83-E85. Copyright Frontline Medical Communications Inc. 2017. All rights reserved.

Case Report

A 39-year-old woman with a 20-year history of rheumatoid arthritis (RA) presented to a university-affiliated tertiary care hospital with painful ulcerations on the bilateral dorsal feet that started as bullae 16 weeks prior to presentation. Initial skin biopsy performed by an outside dermatologist 8 weeks prior to presentation showed vasculitis and culture was positive for methicillin-sensitive Staphylococcus aureus. She was started on a prednisone taper and cephalexin, which did not improve the lower extremity ulcerations and the pain became progressively worse. At the time of presentation to our dermatology department, the patient was taking prednisone, hydroxychloroquine, hydrocodone-acetaminophen, and gabapentin. Prior therapy with sulfasalazine failed; etanercept and methotrexate were discontinued years prior due to side effects. The patient had no history of deep vein thrombosis, pulmonary embolism, or miscarriage.

At presentation, the patient was afebrile and her vital signs were stable. Physical examination showed multiple ulcers and erosions on the bilateral dorsal feet with a few scattered retiform red-purple patches (Figure). One bulla was present on the right dorsal foot. All lesions were tender to the touch and edema was present on the bilateral feet. No oral ulcerations were present and no focal neuropathies or palpable cords were appreciated in the lower extremities. There were no other cutaneous abnormalities.

Laboratory studies showed a white blood cell count of 9.54×10³/µL (reference range, 4.16-9.95×10³/µL), hemoglobin count of 12.4 g/dL (reference range, 11.6-15.2 g/dL), and a platelet count of 175×10³/µL (reference range, 143-398×10³/µL). A basic metabolic panel was normal except for an elevated glucose level of 185 mg/dL (reference range, 65-100 mg/dL). Urinalysis was normal. Erythrocyte sedimentation rate and C-reactive protein level were not elevated. Antinuclear antibodies and double-stranded DNA antibodies were normal. Prothrombin time was 10.4 seconds (reference range, 9.2-11.5 seconds) and dilute viper's venom time was negative. Rheumatoid factor level was elevated at 76 IU/mL (reference range, <25 IU/mL) and anti-citrullinated peptide antibody was moderately elevated at 42 U/mL (negative, <20 U/mL; weak positive, 20-39 U/mL; moderate positive, 40-59 U/mL; strong positive, >59 U/mL). The cardiolipin antibodies IgG, IgM, and IgA were within reference range. Results of β₂-glycoprotein I IgG and IgM antibody tests were normal, but IgA was elevated at 34 µg/mL (reference range, <20 µg/mL). Wound cultures grew moderate Enterobacter cloacae and Staphylococcus lugdunensis.

Slides from 2 prior punch biopsies obtained by an outside hospital approximately 8 weeks prior from the right and left dorsal foot lesions were reviewed. Both biopsies were histologically similar. Postcapillary venules showed extensive vasculitis with numerous fibrin thrombi in the lumens in both biopsy specimens. The biopsy from the right foot showed prominent ulceration of the epidermis, with a few of the affected vessels showing minimal accompanying nuclear dust; however, the predominant pattern was not that of leukocytoclastic vasculitis. Biopsy from the left foot showed prominent epidermal necrosis with focal reepithelialization and scattered eosinophils. The pathologist felt that a vasculitis secondary to coagulopathy was most likely but that a drug reaction and rheumatoid vasculitis would be other entities to consider in the differential. A review of the laboratory findings from the outside hospital from approximately 12 weeks prior to presentation showed IgM was normal but IgG was elevated at 28 U/mL (reference range, 0-15 U/mL) and IgA was elevated at 8 U/mL (reference range, 0-7 U/mL); β₂-glycoprotein I IgG antibodies were elevated at 37 mg/dL (reference range, 0-25.0 mg/dL) and β₂-glycoprotein I IgA antibodies were elevated at 5 mg/dL (reference range, 0-4.0 mg/dL).

The clinical suspicion of a thrombotic event on the dorsal feet, which was confirmed histologically, and the persistently positive antiphospholipid (aPL) antibody titers helped to establish the diagnosis of antiphospholipid syndrome (APS) in the setting of RA. The dose of prednisone was increased from 10 mg daily on admission to 40 mg daily. The patient was started on enoxaparin 60 mg subcutaneously twice daily at initial presentation and was bridged to oral warfarin 2 mg daily after the diagnosis of APS was established. Oral doxycycline 100 mg twice daily was started for wound infection. The ulcerations gradually improved over the course of her 7-day hospitalization. She was continued on prednisone, hydroxychloroquine, and warfarin as an outpatient and has had no recurrence of lesions after 3 years of follow-up on this regimen.

Comment

Antiphospholipid syndrome is an autoimmune condition defined by a venous and/or arterial thrombotic event and/or pregnancy morbidity in the presence of persistently elevated aPL antibody titers. The most frequently detected subgroups of aPL are anticardiolipin (aCL) antibodies, anti-β₂-glycoprotein I antibodies, and lupus anticoagulants.¹ Primary APS occurs as an isolated entity, whereas secondary APS occurs in the setting of a preexisting autoimmune disease, infection, malignancy, or medication.² The diagnostic criteria for APS requires positive aPL titers at least 12 weeks apart and a clinically confirmed thrombotic event or pregnancy morbidity.³

About one-third to half of patients with APS exhibit cutaneous manifestations.^4,5 Livedo reticularis is most commonly observed and represents the first clinical sign of APS in 17.5% of cases.6 Cutaneous findings of APS also include anetoderma, cutaneous ulceration and necrosis, necrotizing vasculitis, livedoid vasculitis, thrombophlebitis, purpura, ecchymoses, painful skin nodules, and subungual hemorrhages.⁷ The various cutaneous manifestations of APS are associated with a range of histopathologic findings, but noninflammatory thrombosis in small arteries and/or veins in the dermis and subcutaneous fat tissue is the most common histologic feature.⁴ Our patient exhibited cutaneous ulceration and necrosis, and biopsy clearly showed the presence of vasculitis and fibrin thrombi within postcapillary venules. These findings along with the persistently elevated β₂-glycoprotein I IgA solidified the diagnosis of APS.

The most common cutaneous manifestations of RA are nodules (32%), Raynaud phenomenon (10%), and vasculitis (3%).⁸ The mean prevalence of aPL antibodies in patients with RA is 28%, though reports range from 5% to 75%.¹ The presence of aPL or aCL does not predict the development of thrombosis and/or thrombocytopenia in RA patients^9,10; however, aCL antibodies in RA patients are associated with a higher risk for developing rheumatoid nodules. It is hypothesized that the majority of aCL antibodies identified in RA patients have different specificities than those identified in other diseases that are associated with thrombotic events.¹

Anticoagulation has been proven to decrease the risk for recurrent thrombotic events in patients with APS.¹¹ Patients should discontinue the use of estrogen-containing oral contraceptives; avoid smoking cigarettes; and treat hypertension, hyperlipidemia, and diabetes mellitus, if present. The type and duration of anticoagulation therapy, especially for the treatment of the cutaneous manifestations of APS, is less well defined. Antiplatelet therapies such as low-dose aspirin or dipyridamole often are used for less severe cutaneous manifestations such as livedoid vasculopathy. Warfarin with a target international normalized ratio of 2.0 to 3.0 is most commonly used following major thrombotic events, including cutaneous necrosis and digital gangrene. The role of corticosteroids and immunosuppressants is unclear; one study showed that these therapies did not prevent further thrombotic events in patients with systemic lupus erythematosus.⁴

Conclusion

Although aPL antibodies are most prevalent in patients with systemic lupus erythematosus, an estimated 28% of patients with RA have elevated aPL titers. The aPL antibodies recognized in RA patients are thought to have a different specificity than those recognized in other APS-associated diseases because elevated aPL antibody titers are not associated with an increased incidence of thrombotic events in RA patients; however, larger studies are needed to clarify this phenomenon. It remains to be determined if this case of APS and RA represents a coincidence or a true disease association, but the recognition of the cutaneous and histological features of APS is crucial for establishing a diagnosis and initiating anticoagulation therapy to prevent further morbidity and mortality.

Case Report

A 39-year-old woman with a 20-year history of rheumatoid arthritis (RA) presented to a university-affiliated tertiary care hospital with painful ulcerations on the bilateral dorsal feet that started as bullae 16 weeks prior to presentation. Initial skin biopsy performed by an outside dermatologist 8 weeks prior to presentation showed vasculitis and culture was positive for methicillin-sensitive Staphylococcus aureus. She was started on a prednisone taper and cephalexin, which did not improve the lower extremity ulcerations and the pain became progressively worse. At the time of presentation to our dermatology department, the patient was taking prednisone, hydroxychloroquine, hydrocodone-acetaminophen, and gabapentin. Prior therapy with sulfasalazine failed; etanercept and methotrexate were discontinued years prior due to side effects. The patient had no history of deep vein thrombosis, pulmonary embolism, or miscarriage.

At presentation, the patient was afebrile and her vital signs were stable. Physical examination showed multiple ulcers and erosions on the bilateral dorsal feet with a few scattered retiform red-purple patches (Figure). One bulla was present on the right dorsal foot. All lesions were tender to the touch and edema was present on the bilateral feet. No oral ulcerations were present and no focal neuropathies or palpable cords were appreciated in the lower extremities. There were no other cutaneous abnormalities.

Laboratory studies showed a white blood cell count of 9.54×10³/µL (reference range, 4.16-9.95×10³/µL), hemoglobin count of 12.4 g/dL (reference range, 11.6-15.2 g/dL), and a platelet count of 175×10³/µL (reference range, 143-398×10³/µL). A basic metabolic panel was normal except for an elevated glucose level of 185 mg/dL (reference range, 65-100 mg/dL). Urinalysis was normal. Erythrocyte sedimentation rate and C-reactive protein level were not elevated. Antinuclear antibodies and double-stranded DNA antibodies were normal. Prothrombin time was 10.4 seconds (reference range, 9.2-11.5 seconds) and dilute viper's venom time was negative. Rheumatoid factor level was elevated at 76 IU/mL (reference range, <25 IU/mL) and anti-citrullinated peptide antibody was moderately elevated at 42 U/mL (negative, <20 U/mL; weak positive, 20-39 U/mL; moderate positive, 40-59 U/mL; strong positive, >59 U/mL). The cardiolipin antibodies IgG, IgM, and IgA were within reference range. Results of β₂-glycoprotein I IgG and IgM antibody tests were normal, but IgA was elevated at 34 µg/mL (reference range, <20 µg/mL). Wound cultures grew moderate Enterobacter cloacae and Staphylococcus lugdunensis.

Slides from 2 prior punch biopsies obtained by an outside hospital approximately 8 weeks prior from the right and left dorsal foot lesions were reviewed. Both biopsies were histologically similar. Postcapillary venules showed extensive vasculitis with numerous fibrin thrombi in the lumens in both biopsy specimens. The biopsy from the right foot showed prominent ulceration of the epidermis, with a few of the affected vessels showing minimal accompanying nuclear dust; however, the predominant pattern was not that of leukocytoclastic vasculitis. Biopsy from the left foot showed prominent epidermal necrosis with focal reepithelialization and scattered eosinophils. The pathologist felt that a vasculitis secondary to coagulopathy was most likely but that a drug reaction and rheumatoid vasculitis would be other entities to consider in the differential. A review of the laboratory findings from the outside hospital from approximately 12 weeks prior to presentation showed IgM was normal but IgG was elevated at 28 U/mL (reference range, 0-15 U/mL) and IgA was elevated at 8 U/mL (reference range, 0-7 U/mL); β₂-glycoprotein I IgG antibodies were elevated at 37 mg/dL (reference range, 0-25.0 mg/dL) and β₂-glycoprotein I IgA antibodies were elevated at 5 mg/dL (reference range, 0-4.0 mg/dL).

The clinical suspicion of a thrombotic event on the dorsal feet, which was confirmed histologically, and the persistently positive antiphospholipid (aPL) antibody titers helped to establish the diagnosis of antiphospholipid syndrome (APS) in the setting of RA. The dose of prednisone was increased from 10 mg daily on admission to 40 mg daily. The patient was started on enoxaparin 60 mg subcutaneously twice daily at initial presentation and was bridged to oral warfarin 2 mg daily after the diagnosis of APS was established. Oral doxycycline 100 mg twice daily was started for wound infection. The ulcerations gradually improved over the course of her 7-day hospitalization. She was continued on prednisone, hydroxychloroquine, and warfarin as an outpatient and has had no recurrence of lesions after 3 years of follow-up on this regimen.

Comment

Antiphospholipid syndrome is an autoimmune condition defined by a venous and/or arterial thrombotic event and/or pregnancy morbidity in the presence of persistently elevated aPL antibody titers. The most frequently detected subgroups of aPL are anticardiolipin (aCL) antibodies, anti-β₂-glycoprotein I antibodies, and lupus anticoagulants.¹ Primary APS occurs as an isolated entity, whereas secondary APS occurs in the setting of a preexisting autoimmune disease, infection, malignancy, or medication.² The diagnostic criteria for APS requires positive aPL titers at least 12 weeks apart and a clinically confirmed thrombotic event or pregnancy morbidity.³

About one-third to half of patients with APS exhibit cutaneous manifestations.^4,5 Livedo reticularis is most commonly observed and represents the first clinical sign of APS in 17.5% of cases.6 Cutaneous findings of APS also include anetoderma, cutaneous ulceration and necrosis, necrotizing vasculitis, livedoid vasculitis, thrombophlebitis, purpura, ecchymoses, painful skin nodules, and subungual hemorrhages.⁷ The various cutaneous manifestations of APS are associated with a range of histopathologic findings, but noninflammatory thrombosis in small arteries and/or veins in the dermis and subcutaneous fat tissue is the most common histologic feature.⁴ Our patient exhibited cutaneous ulceration and necrosis, and biopsy clearly showed the presence of vasculitis and fibrin thrombi within postcapillary venules. These findings along with the persistently elevated β₂-glycoprotein I IgA solidified the diagnosis of APS.

The most common cutaneous manifestations of RA are nodules (32%), Raynaud phenomenon (10%), and vasculitis (3%).⁸ The mean prevalence of aPL antibodies in patients with RA is 28%, though reports range from 5% to 75%.¹ The presence of aPL or aCL does not predict the development of thrombosis and/or thrombocytopenia in RA patients^9,10; however, aCL antibodies in RA patients are associated with a higher risk for developing rheumatoid nodules. It is hypothesized that the majority of aCL antibodies identified in RA patients have different specificities than those identified in other diseases that are associated with thrombotic events.¹

Anticoagulation has been proven to decrease the risk for recurrent thrombotic events in patients with APS.¹¹ Patients should discontinue the use of estrogen-containing oral contraceptives; avoid smoking cigarettes; and treat hypertension, hyperlipidemia, and diabetes mellitus, if present. The type and duration of anticoagulation therapy, especially for the treatment of the cutaneous manifestations of APS, is less well defined. Antiplatelet therapies such as low-dose aspirin or dipyridamole often are used for less severe cutaneous manifestations such as livedoid vasculopathy. Warfarin with a target international normalized ratio of 2.0 to 3.0 is most commonly used following major thrombotic events, including cutaneous necrosis and digital gangrene. The role of corticosteroids and immunosuppressants is unclear; one study showed that these therapies did not prevent further thrombotic events in patients with systemic lupus erythematosus.⁴

Conclusion

Although aPL antibodies are most prevalent in patients with systemic lupus erythematosus, an estimated 28% of patients with RA have elevated aPL titers. The aPL antibodies recognized in RA patients are thought to have a different specificity than those recognized in other APS-associated diseases because elevated aPL antibody titers are not associated with an increased incidence of thrombotic events in RA patients; however, larger studies are needed to clarify this phenomenon. It remains to be determined if this case of APS and RA represents a coincidence or a true disease association, but the recognition of the cutaneous and histological features of APS is crucial for establishing a diagnosis and initiating anticoagulation therapy to prevent further morbidity and mortality.

Case Report

A 39-year-old woman with a 20-year history of rheumatoid arthritis (RA) presented to a university-affiliated tertiary care hospital with painful ulcerations on the bilateral dorsal feet that started as bullae 16 weeks prior to presentation. Initial skin biopsy performed by an outside dermatologist 8 weeks prior to presentation showed vasculitis and culture was positive for methicillin-sensitive Staphylococcus aureus. She was started on a prednisone taper and cephalexin, which did not improve the lower extremity ulcerations and the pain became progressively worse. At the time of presentation to our dermatology department, the patient was taking prednisone, hydroxychloroquine, hydrocodone-acetaminophen, and gabapentin. Prior therapy with sulfasalazine failed; etanercept and methotrexate were discontinued years prior due to side effects. The patient had no history of deep vein thrombosis, pulmonary embolism, or miscarriage.

At presentation, the patient was afebrile and her vital signs were stable. Physical examination showed multiple ulcers and erosions on the bilateral dorsal feet with a few scattered retiform red-purple patches (Figure). One bulla was present on the right dorsal foot. All lesions were tender to the touch and edema was present on the bilateral feet. No oral ulcerations were present and no focal neuropathies or palpable cords were appreciated in the lower extremities. There were no other cutaneous abnormalities.

Laboratory studies showed a white blood cell count of 9.54×10³/µL (reference range, 4.16-9.95×10³/µL), hemoglobin count of 12.4 g/dL (reference range, 11.6-15.2 g/dL), and a platelet count of 175×10³/µL (reference range, 143-398×10³/µL). A basic metabolic panel was normal except for an elevated glucose level of 185 mg/dL (reference range, 65-100 mg/dL). Urinalysis was normal. Erythrocyte sedimentation rate and C-reactive protein level were not elevated. Antinuclear antibodies and double-stranded DNA antibodies were normal. Prothrombin time was 10.4 seconds (reference range, 9.2-11.5 seconds) and dilute viper's venom time was negative. Rheumatoid factor level was elevated at 76 IU/mL (reference range, <25 IU/mL) and anti-citrullinated peptide antibody was moderately elevated at 42 U/mL (negative, <20 U/mL; weak positive, 20-39 U/mL; moderate positive, 40-59 U/mL; strong positive, >59 U/mL). The cardiolipin antibodies IgG, IgM, and IgA were within reference range. Results of β₂-glycoprotein I IgG and IgM antibody tests were normal, but IgA was elevated at 34 µg/mL (reference range, <20 µg/mL). Wound cultures grew moderate Enterobacter cloacae and Staphylococcus lugdunensis.

Slides from 2 prior punch biopsies obtained by an outside hospital approximately 8 weeks prior from the right and left dorsal foot lesions were reviewed. Both biopsies were histologically similar. Postcapillary venules showed extensive vasculitis with numerous fibrin thrombi in the lumens in both biopsy specimens. The biopsy from the right foot showed prominent ulceration of the epidermis, with a few of the affected vessels showing minimal accompanying nuclear dust; however, the predominant pattern was not that of leukocytoclastic vasculitis. Biopsy from the left foot showed prominent epidermal necrosis with focal reepithelialization and scattered eosinophils. The pathologist felt that a vasculitis secondary to coagulopathy was most likely but that a drug reaction and rheumatoid vasculitis would be other entities to consider in the differential. A review of the laboratory findings from the outside hospital from approximately 12 weeks prior to presentation showed IgM was normal but IgG was elevated at 28 U/mL (reference range, 0-15 U/mL) and IgA was elevated at 8 U/mL (reference range, 0-7 U/mL); β₂-glycoprotein I IgG antibodies were elevated at 37 mg/dL (reference range, 0-25.0 mg/dL) and β₂-glycoprotein I IgA antibodies were elevated at 5 mg/dL (reference range, 0-4.0 mg/dL).

The clinical suspicion of a thrombotic event on the dorsal feet, which was confirmed histologically, and the persistently positive antiphospholipid (aPL) antibody titers helped to establish the diagnosis of antiphospholipid syndrome (APS) in the setting of RA. The dose of prednisone was increased from 10 mg daily on admission to 40 mg daily. The patient was started on enoxaparin 60 mg subcutaneously twice daily at initial presentation and was bridged to oral warfarin 2 mg daily after the diagnosis of APS was established. Oral doxycycline 100 mg twice daily was started for wound infection. The ulcerations gradually improved over the course of her 7-day hospitalization. She was continued on prednisone, hydroxychloroquine, and warfarin as an outpatient and has had no recurrence of lesions after 3 years of follow-up on this regimen.

Comment

Antiphospholipid syndrome is an autoimmune condition defined by a venous and/or arterial thrombotic event and/or pregnancy morbidity in the presence of persistently elevated aPL antibody titers. The most frequently detected subgroups of aPL are anticardiolipin (aCL) antibodies, anti-β₂-glycoprotein I antibodies, and lupus anticoagulants.¹ Primary APS occurs as an isolated entity, whereas secondary APS occurs in the setting of a preexisting autoimmune disease, infection, malignancy, or medication.² The diagnostic criteria for APS requires positive aPL titers at least 12 weeks apart and a clinically confirmed thrombotic event or pregnancy morbidity.³

About one-third to half of patients with APS exhibit cutaneous manifestations.^4,5 Livedo reticularis is most commonly observed and represents the first clinical sign of APS in 17.5% of cases.6 Cutaneous findings of APS also include anetoderma, cutaneous ulceration and necrosis, necrotizing vasculitis, livedoid vasculitis, thrombophlebitis, purpura, ecchymoses, painful skin nodules, and subungual hemorrhages.⁷ The various cutaneous manifestations of APS are associated with a range of histopathologic findings, but noninflammatory thrombosis in small arteries and/or veins in the dermis and subcutaneous fat tissue is the most common histologic feature.⁴ Our patient exhibited cutaneous ulceration and necrosis, and biopsy clearly showed the presence of vasculitis and fibrin thrombi within postcapillary venules. These findings along with the persistently elevated β₂-glycoprotein I IgA solidified the diagnosis of APS.

The most common cutaneous manifestations of RA are nodules (32%), Raynaud phenomenon (10%), and vasculitis (3%).⁸ The mean prevalence of aPL antibodies in patients with RA is 28%, though reports range from 5% to 75%.¹ The presence of aPL or aCL does not predict the development of thrombosis and/or thrombocytopenia in RA patients^9,10; however, aCL antibodies in RA patients are associated with a higher risk for developing rheumatoid nodules. It is hypothesized that the majority of aCL antibodies identified in RA patients have different specificities than those identified in other diseases that are associated with thrombotic events.¹

Anticoagulation has been proven to decrease the risk for recurrent thrombotic events in patients with APS.¹¹ Patients should discontinue the use of estrogen-containing oral contraceptives; avoid smoking cigarettes; and treat hypertension, hyperlipidemia, and diabetes mellitus, if present. The type and duration of anticoagulation therapy, especially for the treatment of the cutaneous manifestations of APS, is less well defined. Antiplatelet therapies such as low-dose aspirin or dipyridamole often are used for less severe cutaneous manifestations such as livedoid vasculopathy. Warfarin with a target international normalized ratio of 2.0 to 3.0 is most commonly used following major thrombotic events, including cutaneous necrosis and digital gangrene. The role of corticosteroids and immunosuppressants is unclear; one study showed that these therapies did not prevent further thrombotic events in patients with systemic lupus erythematosus.⁴

Conclusion

Although aPL antibodies are most prevalent in patients with systemic lupus erythematosus, an estimated 28% of patients with RA have elevated aPL titers. The aPL antibodies recognized in RA patients are thought to have a different specificity than those recognized in other APS-associated diseases because elevated aPL antibody titers are not associated with an increased incidence of thrombotic events in RA patients; however, larger studies are needed to clarify this phenomenon. It remains to be determined if this case of APS and RA represents a coincidence or a true disease association, but the recognition of the cutaneous and histological features of APS is crucial for establishing a diagnosis and initiating anticoagulation therapy to prevent further morbidity and mortality.

Take-Home Points

• Pronator teres muscle injuries are rare.
• Injury can be mistaken for MUCL injury in athletes.
• Tenderness and weak/painful forearm pronation are common findings.
• MRI confirms the diagnosis and helps grade the muscle strain injury.
• Conservative treatment is recommended and prognosis is excellent even for high-grade strains.

Pronator teres muscle strain is a rare sporting injury reported only in cricket players, and now in a golfer whose forearm experienced an eccentric force during resisted elbow flexion and pronation.^1,2 The injury occurs when the sporting club or racket strikes the ground during a swing, impeding forward progress and subjecting the pronator teres muscle to eccentric forces in excess of what it can withstand. The pronator teres, one of several muscles that comprise the flexor wad of the forearm, consists of 2 heads, originating proximally from the medical epicondyle and attaching distally to the shaft of the radius on its lateral surface and just distal to the supinator. The oblique orientation of the muscle belly allows it to serve in its primary rotatory role as the main pronator of the forearm. Injuries to the soft tissue of the medial forearm are common in both elite and recreational athletes, especially in racket and club sports.³ Often, these injuries are related to overuse and chronic fatigue of the surrounding soft tissue—caused by repetitive flexing, gripping, or swinging. Even when identified early, these injuries can result in a significant loss of training time.⁴ In this article, we report a case of pronator teres muscle tear at the myotendinous junction. The patient provided written informed consent for print and electronic publication of this case report.

Case Report

A right-hand–dominant 36-year-old man presented to the clinic with pain on the medial side of his right elbow after sustaining an injury to the elbow while playing golf several days earlier. The patient, an advertising executive, was playing recreational golf several times a month and had no significant medical history or previous symptoms related to the elbow. Initial pain symptoms began during a second round of play, immediately after the patient miss-hit an iron shot, making contact mostly with the ground and causing the club to forcefully stop. The pain was on the medial side of the elbow and forearm. The patient noted progressive swelling and bruising at the pain site and development of forearm weakness. Physical examination during the clinic presentation revealed ecchymosis on the anterior medial forearm, medial elbow, and medial triceps (Figure 1).

Noncontrast magnetic resonance imaging (MRI) showed a high-grade partial tear of the pronator teres myotendinous junction (Figures 2A-2C).

Discussion

A tear of the pronator teres is an exceedingly rare injury. Our results with conservative treatment and a full return to previous activity level are consistent with the only other case reported in the literature.⁵ In contrast to our patient, the previous patient sustained a tear of the pronator teres after a prolonged period of batting during a recreational cricket match.

Our patient’s pronator teres injury occurred at the myotendinous junction, a muscle-tendon transition zone often susceptible to injury. What is unusual for this athletic medial elbow injury is that the patient reported no previous symptoms, and it appears that, though the surrounding muscle may have been fatigued by overuse from the round of golf earlier that day, the pathology was caused by an acute eccentric force. During a golf swing, tremendous forces are put on the entire body, from the lower extremities to the forearm and the fingers. Successful completion of the transfer of energy from the golf club to the ball requires both proper technique and proper functioning of key muscles. Specifically, parameters such as ball positioning, club angle, and wrist control play a major role.⁶ Altered forearm positioning or swing arc can significantly affect club head velocity and energy transfer without putting more stress on the golfer.⁷ Therefore, it is easy to understand how prolonged or extended play may fatigue the surrounding elbow muscles, leading to altered technique and increased susceptibility to acute injury. Biomechanical analysis of shoulder motion can provide a helpful baseline for assessing injury-related changes in golf swing and developing specific exercise and rehabilitation programs.^8,9Although injury to the pronator teres is rare, sport physicians should be aware that, after a valgus stress or force, bruising and swelling along the medial elbow do not always indicate a medial ulnar collateral ligament (MUCL) tear or medial epicondylitis. The key examination findings that differentiate this injury from a MUCL injury are the exact location of pain, the milking maneuver for MUCL incompetence, and the extensive bruising over the muscle course of the pronator teres. MRI plays a pivotal role in proper diagnosis.⁴ In addition, MRI allows for evaluation of any concomitant injuries that may be obscuring the clinical presentation.

Successful treatment of such injuries is important for both elite and recreational athletes. With rest and physical therapy, our patient recovered from this rare isolated injury to the pronator teres with complete resolution of symptoms and full ROM. In the literature, we found no other reports of isolated full-thickness myotendinous rupture of the pronator teres or avulsion from the medial epicondyle. Therefore, it is unclear whether the same outcome can be expected with conservative therapy. However, because of the good outcomes for partial-thickness injuries treated conservatively and the lack of robust tendinous tissue to repair at the myotendinous junction, we recommend an initial course of conservative treatment. Sports physicians should be aware of this exceedingly rare injury to the elbow and understand the large forces experienced by the soft tissues of the forearm during the golf swing.^9,10

Conclusion

Pronator teres muscle strain is a rare sporting injury reported in cricket and golf players. The elbow experiences a large eccentric force during resisted elbow flexion and pronation. The injury appears to occur when the sporting club or racket strikes the ground during a forceful swing impeding forward progress of the arm. The injury can be confused with a MUCL injury, or exacerbation of medial epicondylitis. Physical examination reveals bruising and tenderness over the course of the pronator teres, often distal to the elbow. Advanced imaging confirms the diagnosis and helps grade the severity of muscle strain. Treatment is often conservative, with return to function and sport after 4 to 6 weeks of rest and restricted activities. The patient in this case report had complete return to sporting function, with no residual weakness or pain.

Am J Orthop. 2017;46(2):E105-E107. Copyright Frontline Medical Communications Inc. 2017. All rights reserved.

Take-Home Points

• Pronator teres muscle injuries are rare.
• Injury can be mistaken for MUCL injury in athletes.
• Tenderness and weak/painful forearm pronation are common findings.
• MRI confirms the diagnosis and helps grade the muscle strain injury.
• Conservative treatment is recommended and prognosis is excellent even for high-grade strains.

Pronator teres muscle strain is a rare sporting injury reported only in cricket players, and now in a golfer whose forearm experienced an eccentric force during resisted elbow flexion and pronation.^1,2 The injury occurs when the sporting club or racket strikes the ground during a swing, impeding forward progress and subjecting the pronator teres muscle to eccentric forces in excess of what it can withstand. The pronator teres, one of several muscles that comprise the flexor wad of the forearm, consists of 2 heads, originating proximally from the medical epicondyle and attaching distally to the shaft of the radius on its lateral surface and just distal to the supinator. The oblique orientation of the muscle belly allows it to serve in its primary rotatory role as the main pronator of the forearm. Injuries to the soft tissue of the medial forearm are common in both elite and recreational athletes, especially in racket and club sports.³ Often, these injuries are related to overuse and chronic fatigue of the surrounding soft tissue—caused by repetitive flexing, gripping, or swinging. Even when identified early, these injuries can result in a significant loss of training time.⁴ In this article, we report a case of pronator teres muscle tear at the myotendinous junction. The patient provided written informed consent for print and electronic publication of this case report.

Case Report

A right-hand–dominant 36-year-old man presented to the clinic with pain on the medial side of his right elbow after sustaining an injury to the elbow while playing golf several days earlier. The patient, an advertising executive, was playing recreational golf several times a month and had no significant medical history or previous symptoms related to the elbow. Initial pain symptoms began during a second round of play, immediately after the patient miss-hit an iron shot, making contact mostly with the ground and causing the club to forcefully stop. The pain was on the medial side of the elbow and forearm. The patient noted progressive swelling and bruising at the pain site and development of forearm weakness. Physical examination during the clinic presentation revealed ecchymosis on the anterior medial forearm, medial elbow, and medial triceps (Figure 1).

Noncontrast magnetic resonance imaging (MRI) showed a high-grade partial tear of the pronator teres myotendinous junction (Figures 2A-2C).

Discussion

A tear of the pronator teres is an exceedingly rare injury. Our results with conservative treatment and a full return to previous activity level are consistent with the only other case reported in the literature.⁵ In contrast to our patient, the previous patient sustained a tear of the pronator teres after a prolonged period of batting during a recreational cricket match.

Our patient’s pronator teres injury occurred at the myotendinous junction, a muscle-tendon transition zone often susceptible to injury. What is unusual for this athletic medial elbow injury is that the patient reported no previous symptoms, and it appears that, though the surrounding muscle may have been fatigued by overuse from the round of golf earlier that day, the pathology was caused by an acute eccentric force. During a golf swing, tremendous forces are put on the entire body, from the lower extremities to the forearm and the fingers. Successful completion of the transfer of energy from the golf club to the ball requires both proper technique and proper functioning of key muscles. Specifically, parameters such as ball positioning, club angle, and wrist control play a major role.⁶ Altered forearm positioning or swing arc can significantly affect club head velocity and energy transfer without putting more stress on the golfer.⁷ Therefore, it is easy to understand how prolonged or extended play may fatigue the surrounding elbow muscles, leading to altered technique and increased susceptibility to acute injury. Biomechanical analysis of shoulder motion can provide a helpful baseline for assessing injury-related changes in golf swing and developing specific exercise and rehabilitation programs.^8,9Although injury to the pronator teres is rare, sport physicians should be aware that, after a valgus stress or force, bruising and swelling along the medial elbow do not always indicate a medial ulnar collateral ligament (MUCL) tear or medial epicondylitis. The key examination findings that differentiate this injury from a MUCL injury are the exact location of pain, the milking maneuver for MUCL incompetence, and the extensive bruising over the muscle course of the pronator teres. MRI plays a pivotal role in proper diagnosis.⁴ In addition, MRI allows for evaluation of any concomitant injuries that may be obscuring the clinical presentation.

Successful treatment of such injuries is important for both elite and recreational athletes. With rest and physical therapy, our patient recovered from this rare isolated injury to the pronator teres with complete resolution of symptoms and full ROM. In the literature, we found no other reports of isolated full-thickness myotendinous rupture of the pronator teres or avulsion from the medial epicondyle. Therefore, it is unclear whether the same outcome can be expected with conservative therapy. However, because of the good outcomes for partial-thickness injuries treated conservatively and the lack of robust tendinous tissue to repair at the myotendinous junction, we recommend an initial course of conservative treatment. Sports physicians should be aware of this exceedingly rare injury to the elbow and understand the large forces experienced by the soft tissues of the forearm during the golf swing.^9,10

Conclusion

Pronator teres muscle strain is a rare sporting injury reported in cricket and golf players. The elbow experiences a large eccentric force during resisted elbow flexion and pronation. The injury appears to occur when the sporting club or racket strikes the ground during a forceful swing impeding forward progress of the arm. The injury can be confused with a MUCL injury, or exacerbation of medial epicondylitis. Physical examination reveals bruising and tenderness over the course of the pronator teres, often distal to the elbow. Advanced imaging confirms the diagnosis and helps grade the severity of muscle strain. Treatment is often conservative, with return to function and sport after 4 to 6 weeks of rest and restricted activities. The patient in this case report had complete return to sporting function, with no residual weakness or pain.

Am J Orthop. 2017;46(2):E105-E107. Copyright Frontline Medical Communications Inc. 2017. All rights reserved.

Take-Home Points

• Pronator teres muscle injuries are rare.
• Injury can be mistaken for MUCL injury in athletes.
• Tenderness and weak/painful forearm pronation are common findings.
• MRI confirms the diagnosis and helps grade the muscle strain injury.
• Conservative treatment is recommended and prognosis is excellent even for high-grade strains.

Pronator teres muscle strain is a rare sporting injury reported only in cricket players, and now in a golfer whose forearm experienced an eccentric force during resisted elbow flexion and pronation.^1,2 The injury occurs when the sporting club or racket strikes the ground during a swing, impeding forward progress and subjecting the pronator teres muscle to eccentric forces in excess of what it can withstand. The pronator teres, one of several muscles that comprise the flexor wad of the forearm, consists of 2 heads, originating proximally from the medical epicondyle and attaching distally to the shaft of the radius on its lateral surface and just distal to the supinator. The oblique orientation of the muscle belly allows it to serve in its primary rotatory role as the main pronator of the forearm. Injuries to the soft tissue of the medial forearm are common in both elite and recreational athletes, especially in racket and club sports.³ Often, these injuries are related to overuse and chronic fatigue of the surrounding soft tissue—caused by repetitive flexing, gripping, or swinging. Even when identified early, these injuries can result in a significant loss of training time.⁴ In this article, we report a case of pronator teres muscle tear at the myotendinous junction. The patient provided written informed consent for print and electronic publication of this case report.

Case Report

A right-hand–dominant 36-year-old man presented to the clinic with pain on the medial side of his right elbow after sustaining an injury to the elbow while playing golf several days earlier. The patient, an advertising executive, was playing recreational golf several times a month and had no significant medical history or previous symptoms related to the elbow. Initial pain symptoms began during a second round of play, immediately after the patient miss-hit an iron shot, making contact mostly with the ground and causing the club to forcefully stop. The pain was on the medial side of the elbow and forearm. The patient noted progressive swelling and bruising at the pain site and development of forearm weakness. Physical examination during the clinic presentation revealed ecchymosis on the anterior medial forearm, medial elbow, and medial triceps (Figure 1).

Noncontrast magnetic resonance imaging (MRI) showed a high-grade partial tear of the pronator teres myotendinous junction (Figures 2A-2C).

Discussion

A tear of the pronator teres is an exceedingly rare injury. Our results with conservative treatment and a full return to previous activity level are consistent with the only other case reported in the literature.⁵ In contrast to our patient, the previous patient sustained a tear of the pronator teres after a prolonged period of batting during a recreational cricket match.

Our patient’s pronator teres injury occurred at the myotendinous junction, a muscle-tendon transition zone often susceptible to injury. What is unusual for this athletic medial elbow injury is that the patient reported no previous symptoms, and it appears that, though the surrounding muscle may have been fatigued by overuse from the round of golf earlier that day, the pathology was caused by an acute eccentric force. During a golf swing, tremendous forces are put on the entire body, from the lower extremities to the forearm and the fingers. Successful completion of the transfer of energy from the golf club to the ball requires both proper technique and proper functioning of key muscles. Specifically, parameters such as ball positioning, club angle, and wrist control play a major role.⁶ Altered forearm positioning or swing arc can significantly affect club head velocity and energy transfer without putting more stress on the golfer.⁷ Therefore, it is easy to understand how prolonged or extended play may fatigue the surrounding elbow muscles, leading to altered technique and increased susceptibility to acute injury. Biomechanical analysis of shoulder motion can provide a helpful baseline for assessing injury-related changes in golf swing and developing specific exercise and rehabilitation programs.^8,9Although injury to the pronator teres is rare, sport physicians should be aware that, after a valgus stress or force, bruising and swelling along the medial elbow do not always indicate a medial ulnar collateral ligament (MUCL) tear or medial epicondylitis. The key examination findings that differentiate this injury from a MUCL injury are the exact location of pain, the milking maneuver for MUCL incompetence, and the extensive bruising over the muscle course of the pronator teres. MRI plays a pivotal role in proper diagnosis.⁴ In addition, MRI allows for evaluation of any concomitant injuries that may be obscuring the clinical presentation.

Successful treatment of such injuries is important for both elite and recreational athletes. With rest and physical therapy, our patient recovered from this rare isolated injury to the pronator teres with complete resolution of symptoms and full ROM. In the literature, we found no other reports of isolated full-thickness myotendinous rupture of the pronator teres or avulsion from the medial epicondyle. Therefore, it is unclear whether the same outcome can be expected with conservative therapy. However, because of the good outcomes for partial-thickness injuries treated conservatively and the lack of robust tendinous tissue to repair at the myotendinous junction, we recommend an initial course of conservative treatment. Sports physicians should be aware of this exceedingly rare injury to the elbow and understand the large forces experienced by the soft tissues of the forearm during the golf swing.^9,10

Conclusion

Pronator teres muscle strain is a rare sporting injury reported in cricket and golf players. The elbow experiences a large eccentric force during resisted elbow flexion and pronation. The injury appears to occur when the sporting club or racket strikes the ground during a forceful swing impeding forward progress of the arm. The injury can be confused with a MUCL injury, or exacerbation of medial epicondylitis. Physical examination reveals bruising and tenderness over the course of the pronator teres, often distal to the elbow. Advanced imaging confirms the diagnosis and helps grade the severity of muscle strain. Treatment is often conservative, with return to function and sport after 4 to 6 weeks of rest and restricted activities. The patient in this case report had complete return to sporting function, with no residual weakness or pain.

Am J Orthop. 2017;46(2):E105-E107. Copyright Frontline Medical Communications Inc. 2017. All rights reserved.