Spinal arteriovenous fistulae (AVFs) are rare vascular malformations that can mimic acute ischemic stroke, leading to inappropriate thrombolysis and severe complications.

A 74-year-old woman with chronic headaches, thyroid disease, hyperlipidemia, and diabetes presented with sudden occipital headache radiating to the neck and shoulders. During imaging evaluation, she developed acute right-sided paresthesias and weakness (NIHSS 5). Examination showed flaccid right arm weakness and hyperesthesia without facial asymmetry or cranial nerve involvement. Head CT and CTA were negative, and intravenous tenecteplase was administered for presumed ischemic stroke.

Subsequent cervical spine MRI demonstrated C3 cord signal change consistent with infarction and a dorsal epidural hematoma from C2–C5. Spinal angiography confirmed an epidural AVF from the right vertebral artery at C1–C2 with venous congestion. The patient underwent C1–C2 laminectomy with epidural vein coagulation, followed by embolization with coils and Onyx. With rehabilitation, she regained partial upper extremity strength.

This case illustrates how cervical AVFs can masquerade as ischemic stroke. Thrombolysis, though appropriate for suspected cerebral infarction, is ineffective in spinal cord ischemia and carries risk of hemorrhage in unrecognized vascular malformations. Our patient achieved meaningful neurological recovery following surgical and endovascular treatment, underscoring that favorable outcomes are possible when the diagnosis is promptly recognized. Nonetheless, clinicians should maintain vigilance for spinal pathology in atypical stroke presentations—particularly when there is face-sparing weakness, prominent neck pain, or discordance between negative cerebral imaging and clinical deficits—to avoid delays in care and prevent potential catastrophic outcomes.