Spinal epidural arteriovenous fistula with improved sphincter impairment detected by intraoperative neurophysiological monitoring

BACKGROUND: A spinal epidural arteriovenous fistula (SEAVF) is a rare type of arteriovenous shunt that occurs mainly in the thoracic or lumbar spine. Patients with SEAVF develop motor/sensory disturbances of the lower extremities and sphincter dysfunction. Among these symptoms, sphincter impairments show less improvement than others, and its relevance to neurophysiological monitoring has not been documented. CASE DESCRIPTION: A 77-year-old woman presented with progressive motor weakness and numbness in the lower extremities and urinary and fecal incontinence. Spinal magnetic resonance imaging showed spinal cord edema in Th5-Th11 and enlarged perimedullary veins. We performed spinal angiography and endovascular treatment under intraoperative neurophysiological monitoring (IOM), including sensory evoked potential (SEP), motor evoked potential (MEP), and bulbocavernosus reflex (BCR) monitoring. Diagnostic angiography revealed a SEAVF with perimedullary venous drainage fed by the left L2 segmental artery. The shunt was completely embolized using N-butyl-2-cyanoacrylate. Although SEP and MEP of the lower legs were recordable during treatment, anal MEP and BCR were not observed. The sphincter symptoms improved 1.5 years after the treatment. Follow-up angiography revealed no shunt recurrence and improved venous congestion. Anal MEP and BCR were detected during angiography, indicating neurophysiological improvement in sphincter function. The prolonged latency of the monitoring suggested a pudendal nerve injury. CONCLUSION: This case report first described improvement of the IOM correlated with the functional recovery of sphincters after embolization of a SEAVF. Follow-up neurophysiological monitoring is important to assess the functional recovery of the sphincter.