The occurrence of a spontaneous spinal epidural hematoma is uncommon. Cases where it may mimic an ischaemic stroke were seldom reported and misdiagnosis may have important therapeutic and prognostic implications. We report a case of a 58-year-old woman with dyslipidemia who was admitted to the emergency department with right-sided hemiparesis, doubtful right facial paresis, and cervicalgia, with a total NIHSS of 8. There was no history of trauma or cervical manipulation. Brain and neck CT/CTA imaging disclosed no acute lesions nor large vessel changes. The patient received intravenous thrombolysis without complications. After a brain MRI without ischaemic lesions, cervical imaging identified a spinal epidural hematoma (SEH) at C3–C6 levels with associated oedema (Fig. 1). Retrospectively, a discrete intracanalar hyperdensity was visible on admission CT/CTA. No aetiology was found after blood workup and cerebral and cervical digital subtraction angiography. Patient was discharged without any neurological deficits. 3-month follow-up MRI showed partial hematoma reabsorption. SEH is a rare cause of spinal cord compression and can lead to permanent neurological deficits. Most hematomas are spontaneous, and some may be associated with vascular malformations, coagulopathy, physical exertion, and possibly uncontrolled hypertension [1]. Sudden onset of radicular/neck/back pain associated with sensory/motor deficits configure the typical presentation of a SHE [1]. Hemibody neurological deficits are present in a minority of patients [2]. MRI is the imaging modality of choice when suspecting this entity. However, many centres do not have access to MRI when managing a stroke code patient at the emergency department, and misdiagnosis may occur. In a recent study, 5 out of 2866 patients (0.2%) admitted through stroke code had a final diagnosis of SHE [2]. The administration of thrombolysis can have significant prognostic implications in this situation. In patients with spontaneous SEH, while a conservative approach may also lead to complete recovery, emergent surgical evacuation and decompression may be needed, increasing the probability of a favourable outcome [3]. In comparison with previous case-reports [2–5], ours was the first without clinical deterioration and need of subsequent surgery after thrombolysis. In all cases, the presence of pain followed by hemiparesis was the main clinical presentation. In our case, the presence of a right central facial paresis was misinterpreted probably due to unknown constitutional facial asymmetry, and the presence of cervicalgia evoked a potential dissection. In conclusion, our case-report describes a patient with a SEH mimicking an acute ischaemic stroke, where a careful clinical and neuroradiological assessment would have prevented intravenous thrombolysis treatment, potentially deleterious in this situation. In patients with acute neurological deficits and cervicalgia, SEH should be considered as a differential diagnosis.