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Ebbeson, Regan Lorraine MD*; De Kock, Matthys Johannes MD; Penny, Norgrove MD, FRCS(C); Kollman, Tobias R. MD, PhD§

The Pediatric Infectious Disease Journal: September 2009 - Volume 28 - Issue 9 - p 850-852
doi: 10.1097/INF.0b013e3181a33522
Brief Reports

We present a case of para-influenza type 1 induced rhabdomyolysis, acute renal failure, and extensive compartment syndrome in a 6-year-old previously well child. Fasciotomies and subsequent skin grafting of both lower extremities and a prolonged course of hemodialysis led to a complete clinical and biochemical recovery.

From the *Department of Pediatrics, §Division of Infectious Diseases and Immunology, British Columbia's Children's Hospital, British Columbia, Vancouver, Canada; †Vancouver Island Health Authority, British Columbia, Canada; and ‡Department of Orthopedics, Faculty of Medicine, University of British Columbia, Vancouver, Canada.

Accepted for publication February 27, 2009.

No sources of support or financial funding consent was obtained for publication of this case report from the child's mother.

Address for correspondence: Regan Ebbeson, MD, 3838 Hoskins Road, North Vancouver, BC, V7K 2N8. E-mail:

Complications of rhabdomyolysis include acute renal failure and compartment syndrome. Viral infections account for one-third of the cases of acute pediatric rhabdomyolysis.1 Most of these cases are attributed to influenza infection. We are aware of only 4 cases of para-influenza virus induced rhabdomyolysis infection described in the literature.2–5 This is the second report of acute renal failure after rhabdomyolysis5; no case of parainfluenza virus-induced rhabdomyolysis complicated by compartment syndrome has been reported.

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A previously healthy 6-year-old girl presented in December 2007 with a 7 day history of fever, malaise, and sore throat. She had no significant respiratory symptoms. Before admission, she had been seen by her family physician and treated with amoxicillin for a presumptive streptococcal throat infection, but the throat culture taken before initiation of antibiotics was negative. Two days before admission she developed vomiting, neck pain, and diffuse myalgia. The past medical history was unremarkable. She had received all her routine immunizations. There was no family history of importance. She had suffered a small abrasion of the anterior right shin 10 days before admission which was healing well.

She was presented to a local hospital with a decreased level of consciousness, severe dehydration, and severe bilateral thigh and calf pain and swelling. She was tachycardic with a heart rate of 155 beats per minute and peripherally cool. Blood pressure was 119/89 mm Hg. Oxygen saturation was 99%. She was afebrile although she had a documented temperature of 38.4°C axillary earlier that day and had been given acetominophen. Intravenous normal saline led to an improvement in her neurologic and cardiovascular status. Initial laboratory analysis revealed an white blood cell count of 11.3 × 109/L, neutrophils of 7.4 × 109/L, and lymphocytes of 3.5 × 109/L, hemoglobin 183 g/L, platelet count of 239 × 109/L, sodium of 125 mmol/L, potassium of 4.6 mmol/L, blood urea nitrogen (BUN) of 11 mmol/L, and a creatinine of 71 μmol/L. A blood culture was drawn before empiric treatment with intravenous cefotaxime The presumptive diagnosis was septic shock. She was transferred to the intensive care unit at the regional hospital for further care.

On arrival at the regional hospital, she was alert and responsive and in stable condition. She required no ongoing volume resuscitation beyond maintenance fluids. She remained afebrile. On admission, her heart rate was 120 beats per minute, blood pressure was 123/88 mm Hg, and respiratory rate and oxygen saturation were normal. Initial investigations revealed a normal complete blood count and differential, sodium 129 mmol/L, potassium 5.1 mmol/L, chloride 101 mmol/L, bicarbonate 19 mmol/L, anion gap 9, BUN 7.1 mmol/L, and creatinine 50 μmol/L. Aspartate aminotransferase was 156 IU/L and creatinine phosphokinase was 8014 IU/L. Alanine aminotransferase was not measured. Alkaline phosphatase and gamma-glutamyl transpeptidase were normal. Blood and urine cultures were repeated. Antibiotic coverage was broadened to include intravenous cloxacillin, clindamycin, and cefotaxime. She had exquisite pain and tenderness to palpation in her upper and lower legs bilaterally, both anteriorly and posteriorly, and was unable to move her legs. There were no associated skin changes except for the healing abrasion on the right anterior shin. All lower extremity compartments were tense to palpation. Findings were symmetric. She had good capillary circulation in her toes and good pedal pulses and she was able to move her toes spontaneously. With concern for significant bilateral compartment syndrome, she was taken to the operating room for measurement of compartment pressures and fasciotomies as necessary.

Compartment pressures of the anterior thigh were 41 mm Hg on the right and 44 mm Hg on the left. The anterior pretibial compartment pressures were 82 mm Hg on the right and 70 mm Hg on the left. The posterior superior calf compartment pressures were 60 mm Hg on the right and 60 mm Hg on the left. Mean arterial pressure at the time of measurements was 88 mm Hg. Normal compartment pressures in children are considered less than 10 to 12 mm Hg and at greater than 35 mm Hg, capillary circulation ceases.6 Open fasciotomies of the quadriceps, pretibial, peroneal, superficial posterior, and deep posterior compartments bilaterally was performed. The muscles appeared edematous and bulged significantly but appeared vascular and healthy with no evidence of necrotic tissue. Specimens were taken for bacterial and viral culture and histology.

Blood and urine cultures remained negative. A nasopharyngeal washing direct immuno-fluorescence assay, was positive for parainfluenza type 1. The presumptive diagnosis was severe viral myositis with compartment syndrome. She remained stable and returned to the operating room 48 hours later for reevaluation and possible wound closure. The muscle compartments were still bulging. The quadriceps bilaterally looked dusky with an area of significant hemorrhagic necrosis in the right lateral quadriceps. The peroneal and pretibial musculatures also appeared dusky. Specimens were again obtained for bacterial and viral culture as well as histology. The left peroneal compartment was closed and the rest of the wounds were left open given evidence of ongoing inflammation and possible necrosis.

On the third day of her admission, her urine output decreased and the BUN and creatinine rose to 10 mmol/L and 117 μmol/L, respectively. The creatinine phosphokinase rose to >50,000 IU/L. On the fourth day of admission, she developed significant respiratory distress. Chest roentgenogram revealed bilateral pulmonary edema. Blood gas showed a mixed respiratory and metabolic acidosis. She was intubated and ventilated. She was transferred to a tertiary children's hospital where she was found to be anuric, necessitating continuous renal replacement therapy. On day 11 of her admission, the fasciotomies were closed. She remained intubated for a total of 13 days. She remained anuric for 17 days and required a total of 20 days of hemodialysis. She was discharged home after 30 days.

Multiple nasopharyngeal washings for viral rapid diagnosis were positive for parainfluenza type 1 and negative for other respiratory viruses. Polymerase chain reaction of muscle tissue obtained from the operating room on her day or admission was negative for parainfluenza type 1 and viral culture from the muscle tissue was also negative. The muscle biopsies showed focal perimysial and interstitial collections of neutrophils without destruction of the myofibres. There was no evidence of vasculitis or myopathy. Stains for fungal and bacterial organisms were negative. A metabolic work-up including plasma amino acids, urine organic acids, lactate, ammonia, and glucose was unremarkable.

In follow-up, she has made a complete clinical and biochemical recovery. There has been no recurrence of rhabdomyolysis during 8 months of follow-up. Her renal function is normal. She has no evidence of muscle contractures and is ambulating well with no functional impairment of her gross motor skills.

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In a recent case series of acute pediatric rhabdomyolysis, viral myositis was the leading cause accounting for 38% of cases.1 The pathogenesis of pediatric rhabdomyolysis is quite different from that reported in the adult literature where trauma and ischemia account for the majority of cases and viral infection accounts for only 1% of reported cases.1 Complications of rhabdomyolysis include acute renal failure and compartment syndrome.

There are only 4 other reports of viral infection associated rhabdomyolysis leading to compartment syndrome. Ng et al7 reported a case in an 18-year-old man with extensive compartment syndrome of the right arm and both legs associated with bilateral femoral nerve palsy secondary to influenza A. Swaringen et al8 reported a 10-year-old girl with rhabdomyolysis complicated by extensive compartment syndrome requiring fasciotomies in all 4 extremities. She had multiorgan dysfunction with respiratory, cardiac, and renal failure. Her tracheal aspirate was positive for influenza A virus. Marinella9 reported a case of a 49-year-old woman who had acute rhabdomyolysis complicated by compartment syndrome of both legs associated with a rise in antibodies to coxsackieviruses B4 and B5. Paletta et al10 reported a 6-year-old boy who developed rhabdomyolysis and bilateral lower extremity compartment syndrome requiring fasciotomies due to infection with influenza B virus. To date, there have been no reports of parainfluenza virus causing a compartment syndrome.

Four cases of parainfluenza virus induced rhabdomyolysis infection have been described in the literature. Ueda et al2 reported a 4-year-old boy who developed an acute generalized necrotizing rhabdomyopathy following a parainfluenza type 3 respiratory tract infection, rapidly progressing to respiratory failure and death. O'Connor et al3 reported a 38-year-old man who developed acute myoglobinuria. Parainfluenza type 2 infection was documented. Renal function remained normal. Zvolanek4 reported an 8-year-old boy who developed benign acute myositis associated with parainfluenza type 2 infection. Vsralovic et al5 reported a 5-year-old boy with spastic quadriplegia who developed rhabdomyolysis complicated by acute renal failure in the setting of parainfluenza type 1 infection.

The pathophysiologic sequence of viral infection leading to rhabdomyolysis is not clear. We were not able to identify viral particles within the tissue in this case. Most cases of virally induced rhabdomyolysis do not have a positive identification of the viral pathogen from the affected muscle tissue suggesting an indirect mechanism, such as an inflammatory mediated process.11 Type 1 interferon is a known cause of rhabdomyolysis,12,13 and high titers of type I interferon are produced during infection with paramyxoviruses, such as parainfluenza 1.14 A potential role of specific host-virus interactions (eg, parainfluenza virus induces interferon regulatory factor-3, which drives type 1 interferon production15–17) may be involved in cases of virally induced rhabdomyolysis, if they occur in a genetically susceptible host (eg, genetic polymorphisms in interferon regulatory factor-3 control total type 1 interferon production).18

In conclusion, this is only the second report of acute renal failure following rhabdomyolysis caused by parainfluenza type 1 infection in a child and the first report of compartment syndrome resulting from parainfluenza type 1 induced rhabdomyolysis. Rhabdomyolysis in children is most commonly caused by viral infections. Influenza A and B are the most common pathogens involved, but parainfluenza virus should be added to the list of possible culprits.

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rhabdomyolysis; parainfluenza type 1 virus; Acute renal failure; compartment syndrome; children

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