STUDY DESIGN: A case report and review of the literature. OBJECTIVE: To report a rare case of paraspinal compartment syndrome that presented a diagnostic challenge. SUMMARY OF BACKGROUND DATA: Paraspinal compartment syndrome is a rare condition defined as increased pressure within a closed fibro-osseous space, resulting in reduced blood flow and tissue perfusion in that space. The reduced perfusion causes ischemic pain and irreversible damage to the tissues of the compartment if unrecognized or left untreated. METHODS: A 20-year-old African-American man presented with 2 days of new-onset progressive back pain after repetitive lifting of 235 lbs in a deadlift exercise. The patient had significantly tender lumbar paraspinal muscles, was unable rise from a supine position, and had severe pain with attempted active rolling or sitting. Findings of supine radiographs were normal. Findings of initial laboratory investigations were consistent with rhabdomyolysis and acute kidney injury. Despite aggressive hydration and narcotic analgesia, the patient's creatinine kinase and myoglobin continued to rise and his pain continued to worsen. Computed tomography of the spine revealed enlarged paraspinal musculature and decreased enhancement bilaterally. Gadolinium-enhanced magnetic resonance imaging of the spine showed increased T2 signal and paraspinal muscle edema with areas of decreased contrast uptake, consistent with ischemia and necrosis. Compartment pressures measured 78 mm Hg on the left and 26 mm Hg on the right. RESULTS: Because hydration and analgesia had failed, the patient was taken urgently to the operating room for bilateral paraspinal fasciotomies with delayed closure. His symptoms and rhabdomyolysis then resolved during the next 2 days. CONCLUSION: The diagnostic challenge presented by this case, especially considering the rarity of paraspinal compartment syndrome, indicates the need for a high index of suspicion in the appropriate setting.