Case Presentation: A 34-year-old male with recently diagnosed Philadelphia chromosome-positive acute lymphoblastic leukemia developed neutropenic fever with right arm pain on day thirteen of induction chemotherapy. He was placed on empiric piperacillin-tazobactam. Blood cultures grew Streptococcus agalactiae. The following day, the patient acutely developed erythema, edema and pain in his right forearm, concerning for cellulitis. Vancomycin was added to his antimicrobial regimen. Computed tomography of the patient’s right upper extremity demonstrated diffuse subcutaneous edema without discernable abscess or fluid collection. The patient’s right forearm erythema, edema and parasthesias worsened, prompting orthopedic consult due to concern for compartment syndrome. An urgent right forearm palmar compartment fasciotomy was performed. Intraoperatively, the flexor digitorum superficialis and flexor carpi ulnaris muscles were dusky with associated bulging. A right carpal tunnel release was performed, with the muscle immediately becoming more viable and contractile. Surgical pathology of muscle tissue obtained intraoperatively showed ischemic and reactive changes consistent with compartment syndrome. There was no evidence of vasculitis, vasculopathy or a leukemic infiltrate. Tissue cultures were negative. The patient underwent two additional debridements of the right upper extremity prior to delayed primary closure with skin grafting. The patient had a full recovery of neuromuscular function; he continues to participate in occupational therapy while awaiting planned haploidentical stem cell transplantation.
Discussion: Non-traumatic acute compartment syndrome is an under-recognized entity for internists. While most cases of non-traumatic compartment syndrome are attributable to ischemia-reperfusion, burns, or prolonged compression of the involved limb (1), infectious etiologies must remain in the differential diagnosis, especially in immunocompromised hosts. While compartment syndrome due to infection is rare, it is important for providers to be aware of this critical complication of cellulitis to prevent ischemia and possible loss of limb.
Conclusions: Cellulitis progressing to infectious compartment syndrome is a very rare clinical entity, with group A streptococcus implicated in several reported cases (2). Although tissue cultures from this patient were negative in the setting of empiric antibiotic administration, it is posited that this patient’s Streptococcus agalactiae bacteremia seeded his right upper extremity, leading to the development of cellulitis and subsequent compartment syndrome. Delay in this diagnosis could have led to loss of the patient’s right arm. This case illustrates the importance of considering non-traumatic compartment syndrome in the differential diagnosis of febrile patients presenting with acute extremity pain and tense compartments, especially in the setting of immunosuppression.