Rationale:Spontaneous spinal epidural hematoma (SSEH) is a rare but highly disabling neurological emergency. The initial presentations are variable. Most patients of SSEH present with paraplegia or tetraplegia clinically, but recurrent hemiparesis with complete spontaneous recovery, mimicking transient ischemic attack (TIA), is a very rare initial presentation of SSEH.Patient concerns:A 71-year-old female presented to the emergency department with 2 episodes of transient right hemiparesis in 5 hours. Two days later, above symptom reappeared and progressed to quadriplegia, dyspnea, and uroschesis quickly. The neurological examination showed tetraplegia and hypalgesia below the C2 level, but neither facial palsy nor aphasia was found.Diagnosis:The patient was initially misdiagnosed as TIA and treated with antiplatelet therapy. But during the hospital day, the cervical magnetic resonance imaging showed a dorsal epidural hematoma extending from C2 to C6 level and she was diagnosed as SSEH.Interventions:She underwent surgical decompression and hematoma removal 1 week later.Outcomes:One week after operation, the sensory deficit above C6 level improved, but there was no improvement in her muscle strength and dyspnea. Unfortunately, she died 1 month later.Lessons:Our case highlights recurrent hemiparesis with complete spontaneous recovery mimicking TIA is a rare initial presentation of SSEH. It is important to perform careful clinical assessments and neuroimaging investigations for correct diagnosis. Neck pain and hemiparesis sparing cranial nerve are important signs for distinction of SSEH from acute ischemic cerebrovascular diseases.