High cervical spinal subdural hemorrhage as a harbinger of craniocervical arteriovenous fistula: an unusual clinical presentation

Abstract Background context Craniocervical dural arteriovenous fistula (dAVF) is rare as compared with the typical thoracolumbar dAVFs of the spine and usually presents with hemorrhagic manifestation, predominantly intracranial subarachnoid hemorrhage. Purpose We describe the first case of craniocervical dAVF with initial presentation as neck pain and spinal subdural hemorrhage. Study design Case report. Methods We present the case of a 59-year-old woman who presented with sudden onset of neck pain at an outside institution emergency department (ED) and was discharged after negative cervical spine radiographs. Magnetic resonance imaging of the cervical spine performed because of persistent pain demonstrated presence of high cervical spinal subdural hematoma and she was managed conservatively. She subsequently presented to our ED a week later with headache and was found to have an intraventricular hemorrhage on computed tomography scan of the head, which on subsequent workup with an angiography revealed the presence of a craniocervical dAVF. Results Surgical obliteration of the fistula was performed with use of intraoperative angiography as an adjunct to confirm complete fistula obliteration. She had an excellent clinical outcome with no deficits at her last follow-up at 9 months. Conclusions Even though hemorrhagic presentation is fairly common in craniocervical dAVFs, there is no report of a craniocervical dAVF presenting with spinal subdural hemorrhage. The present case further highlights the propensity of these vascular lesions to bleed and emphasizes the clinical importance of including these lesions in the differential diagnosis of hemorrhage in the vicinity of foramen magnum region, whether subarachnoid or subdural in location. Physicians treating spinal pathologies should be aware of this entity and clinical presentation, as an angiography needs to be considered in these cases to direct appropriate referral and treatment.