Compartment syndrome of the forearm or leg has been well documented in the literature. However, there have been few published reports of hand compartment syndrome. We hereby present the first reported case in the literature of hand compartment syndrome secondary to an epileptic seizure. A 50-year old gentleman with known epilepsy presented to the Emergency Department following a witnessed tonic-clonic seizure. The patient’s chief complaints were a grossly swollen and excruciatingly painful dominant right hand. Examination revealed severely reduced range of motion and neurovascular compromise. An emergency decompression fasciotomy was performed in the operating theatre, where severe oedema was noted with viable muscle throughout. Compartment syndrome can occur in any muscle compartment of the body, including in the hand. Any crush injury to the hand should trigger a high index of suspicion by the clinician to enable prompt recognition of this surgical emergency and initiate timely management.