Acute idiopathic thenar and hypothenar compartment syndrome

Compartment syndrome (CS) is a condition associated with intra-compartment pressures exceeding the perfusion pressure of the tissue. This condition can occur in any body compartment surrounded by fascia, however CS of some regions like the thenar and hypothenar is extremely rare. Acute CS is often caused by major trauma and idiopathic acute CS is sparsely reported in literature. We present a case report of a 39-year-old male, with a medical history of rheumatoid arthritis (RA) for which he uses adalimumab and low-dose prednisone, with idiopathic acute CS of thenar and hypothenar compartments. Unexplained tremendous pain, paresthesia and swelling of his hand were the only symptoms observed. Due to direct recognition of the condition there was no delay in time to treatment. Treatment involved a fasciotomy of the thenar and hypothenar compartments as well as a release of the carpal tunnel. This direct surgical approach resulted in retaining excellent function and appearance of the hand. We hypothesized several causes for the etiology of the condition (e.g. rhabdomyolysis, rheumatologic disease-related condition, other systemic diseases, trauma, infection), but all could be rejected based on laboratory or clinical findings. Remarkable is that the patient experienced a comparable episode in his other hand two years earlier. Although the acute CS is of unknown origin in the presented case, the development of acute CS in both hands over time suggests a predisposing factor in idiopathic acute CS that is far from being elucidated.