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A rare cause of radiculomyelitis: dural arteriovenous fistula. IMPORTANCE: Dural arteriovenous fistula is a very rare cause of myelitis that can only be treated interventionally or surgically. OBSERVATIONS: A man in his 30s with paraparesis and urinary incontinence had a long-segment thoracic lesion on spinal magnetic resonance imaging. Transverse myelitis was the initial diagnosis. Although a pulse steroid and intravenous immunoglobulin treatment regimen was given, no definite clinical response was seen. A spinal angiogram was performed in our center demonstrating right T7 to T8 spinal dural arteriovenous fistula. The fistula was occluded with embolization and the patient showed recovery following the endovascular treatment. CONCLUSIONS AND RELEVANCE: Myelitis usually is known to respond well to immunosuppressive treatments. Despite adequate medical treatment, if slow progression is seen in the follow-up clinically and radiologically, dural arteriovenous fistulas should be kept in mind in the etiopathogenesis.