OBJECTIVETo describe the effective treatment of a ruptured lateral sacral artery aneurysm presenting as a spinal epidural hematoma.DESIGNCase report and literature review.SETTINGUniversity teaching hospital, neurovascular center.PARTICIPANTA man with a history of renal allograft who presented with sudden onset low back pain.INTERVENTIONThe patient underwent emergent decompressive surgery, diagnostic angiogram and endovascular embolization.MAIN OUTCOME MEASUREObliteration of aneurysm and restoration of neurological function.RESULTSSurgery resulted in decompression of the cauda equina. The patient's aneurysm was endovascularly obliterated. There was complete restoration of neurological function.CONCLUSIONLateral sacral artery aneurysm formation and rupture is a rare cause of epidural hemorrhage and one described only in patients who have undergone renal transplantation. It may be caused by high flow from transpelvic collaterals between both internal iliac arteries. Treatment includes endovascular obliteration and may necessitate operative decompression in the setting of neurological compromise.