Clostridial Myonecrosis in an Adolescent Male
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Clostridial Myonecrosis in an Adolescent Male Melissa Langhan, MD, and Linda Arnold, MD ABSTRACT. Extremity pain is a common complaint in and he was admitted to the hospital with a presumptive diagnosis adolescents. Pain out of proportion to examination find- of viral myositis for pain control, continued hydration, and mon- ings should raise suspicion of deep tissue infection. Clos- itoring of renal function. Within 2 hours of admission, he devel- tridial myonecrosis is a rapidly progressive disease con- oped a violaceous rash over his legs and became hypotensive. On examination, he was noted to have subcutaneous emphysema in sisting of muscle necrosis and systemic toxicity. It is both lower extremities. His legs were noted to be markedly en- usually seen in elderly and immunocompromised pa- larged, and distally they were cool and pale. The symptoms soon tients. Here we report a case of clostridial myonecrosis in spread to involve his right arm. Surgery was consulted and an an adolescent male. Pediatrics 2005;116:e735–e737. URL: emergent computed tomography scan was obtained, which www.pediatrics.org/cgi/doi/10.1542/peds.2004-2876; infec- showed gas extending from his calves to his psoas muscles bilat- tion, pain. erally and also in his right arm (Fig 1). Based on these findings, a diagnosis of necrotizing fasciitis was made. He received penicillin, clindamycin, cefotaxime, and van- lostridial myonecrosis is a rare disease in the comycin. The patient was taken to the operating room for surgical United States, with an estimated incidence of debridement. He was then transferred to an outside institution for 900 to 1000 cases per year.1 However, its vir- hyperbaric oxygen therapy. On arrival after transport, he was C noted to have massive necrosis of his extremities and underwent ulent course and high mortality rate necessitate a bilateral disarticulations at the hips and removal of his right bicep high degree of suspicion among treating physicians. muscle. Blood and wound cultures grew Clostridium septicum.He Here we present a case in an otherwise healthy teen- was given a final diagnosis of clostridial myonecrosis. ager and a review of the recent literature. After amputation, he received antibiotic and hyperbaric oxygen therapy. His clinical course also necessitated intubation and vaso- CASE REPORT pressor treatment. Limited diagnostic testing for both immunode- ficiency and malignancies, including HIV testing, abdominal com- A previously healthy 17-year-old male presented to the emer- puted tomography scan, and immunoglobulin and complement gency department with a 2-day history of bilateral leg pain. The levels were negative at the outside institution. One month after his day before admission he was seen by his pediatrician and found to presentation, he was stable and transferred to a rehabilitation have fever, facial swelling, and oral ulcers. He was diagnosed with facility. He has had no additional infections, signs of immunode- a presumptive viral illness and treated with supportive measures. ficiency, or medical problems. He returned the next day with increasingly severe leg pain, and additional evaluation was undertaken. His past medical history consisted of a diagnosis of Epstein- DISCUSSION Barr virus 1 month before presentation from which he still com- plained of fatigue. He had done some light weight-lifting several Clostridial myonecrosis, also known as nontrau- days before the onset of pain but had no recent participation in matic or spontaneous gas gangrene, is an extremely organized sports activities. There was no history of trauma, and he rare occurrence in pediatric patients. The infection is denied drug or steroid use. characterized by muscle necrosis and systemic toxic- His initial vital signs were notable for a temperature of 39.1°C, respiratory rate of 32 breaths per minute, heart rate of 132 beats ity caused by several clostridial exotoxins, the most per minute, and a blood pressure of 141/62 mm Hg. He was pale, notable of which is ␣-toxin (phospholipase C).2,3 diaphoretic, and unable to stand secondary to his pain. Mild C septicum, an anaerobic Gram-positive rod, is the orbital edema was present as well as gingival swelling and a single causative organism in the majority of cases, followed oral ulceration. There was severe tenderness involving both of his 4,5 thighs and his right calf muscles. by Clostridium perfringens. Unlike typical cases of Urinalysis revealed hematuria and proteinuria and the absence gas gangrene, which follow trauma and are caused of red blood cells. Blood counts showed a white blood cell count by C perfringens, the majority of cases of spontaneous of 2700/L (57% bands, 5% segmented neutrophils, 13% lympho- gas gangrene have no obvious external portal of cytes, 19% monocytes), and his creatine phosphokinase was ele- entry. The presumed source is mucosal defects of the vated at 3594 IU/L. His electrolytes and coagulation profile were 6 within normal limits. The erythrocyte sedimentation rate was 42 intestinal tract. C septicum is substantially more ae- mm/hour. rotolerant than C perfringens, and the inoculum re- Despite intravenous hydration and multiple doses of morphine quired for infection is 300 times smaller, which may and ketorolac, there was no improvement in the patient’s pain, explain the absence of a portal of entry.1 In the adult population, gas gangrene is often as- sociated with trauma, underlying immunodeficiency From the Department of Pediatric Emergency Medicine, Yale-New Haven states, venous insufficiency, or malignancy.7 Simi- Children’s Hospital, New Haven, Connecticut. Accepted for publication May 19, 2005. larly, in children, it has been associated with malig- doi:10.1542/peds.2004-2876 nancies, leukemia, neutropenia, and immunosup- No conflict of interest declared. pression.8,9 Spontaneous gas gangrene has a Address correspondence to Melissa Langhan, MD, Department of Pediatric particular association with colon cancer.3,4,10 In 1 Emergency Medicine, Yale-New Haven Children’s Hospital, 20 York St, WP 143, New Haven, CT 06851. E-mail: [email protected] review of spontaneous C septicum infections, 34% of PEDIATRICS (ISSN 0031 4005). Copyright © 2005 by the American Acad- the patients were found to have colorectal cancer, emy of Pediatrics. and 40% were found to have a hematologic malig- www.pediatrics.org/cgi/doi/10.1542/peds.2004-2876Downloaded from www.aappublications.org/newsPEDIATRICS by guest on September Vol. 11625, 2021 No. 5 November 2005 e735 Fig 1. Scout film demonstrating the presence of gas diffusely throughout the bilateral legs and right pelvis. nancy.11 Those survivors of C septicum gas gangrene changes, pain, and systemic toxicity associated with who are found to be immunocompetent will produce this disease process.14 Aggressive surgical debride- antibodies to ␣-toxin.6 ment is the cornerstone of treatment for all of these The classic presentation of gas gangrene is that of soft tissue infections. severe pain and underlying crepitus. However, these The diagnosis of gas gangrene is based on clinical are frequently late symptoms; early findings are of- findings, demonstration of myonecrosis at surgery, ten nonspecific. Other symptoms include a sudden and supporting microbiological data. Bacteremia is onset of fever, abdominal pain, vomiting, diarrhea, documented in only 10% to 15% of patients with and rapid progression to shock. Up to 25% of pa- clostridial myonecrosis.6 Ultimately, gas gangrene is tients have myonecrosis at metastatic sites.11 a surgical diagnosis made when involved muscle is The sudden onset of severe pain is often the first visualized. The extent of myonecrosis is often greater symptom experienced by the patient. Typically, the than the skin changes indicate. pain is out of proportion to physical examination Treatment includes early operative debridement findings and is minimally relieved by medications. and antibiotic therapy. Because of the polymicrobial Initial physical examination of the site may be nor- nature of some necrotizing soft tissue infections, em- mal; within minutes to hours, localized tense edema, piric antibiotic coverage is implemented. Antibiotic pallor, and tenderness are seen.5–7 combinations used to treat gas gangrene usually in- Systemic findings include diaphoresis, tachycardia clude penicillin and clindamycin. Although high- disproportionate to temperature, and extreme anxi- dose penicillin was once the drug of choice for clos- ety on the part of the patient, who often remains tridial infections, it has been found to be less effective exceedingly alert until the very terminal stages. Late in severe disease. Clindamycin is beneficial because complications include intravascular hemolysis, he- it has the ability to reduce the production of exotox- moglobinuria, hypotension, renal failure, and meta- ins produced by Clostridium species.1,7,13 bolic acidosis. The disease progresses rapidly over In some instances, hyperbaric oxygen therapy is the course of hours and is frequently fatal.3,5,7 also useful. The rationale for the use of hyperbaric The severity of the pain and presence of systemic oxygen includes the observations that raising local toxicity help to differentiate Clostridial myonecrosis tissue oxygen concentrations arrests clostridial repli- from other soft tissue infections including necrotiz- cation and ␣-toxin production and that hyperbaric ing fasciitis and crepitant cellulitis. Necrotizing fas- oxygen treatment of experimental animals reduces ciitis is frequently caused by group A