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Case report.To report an unusual case of spontaneous spinal subdural hematoma and to review relevant literature and discuss the etiology, pathogenesis, clinical features, imaging, and prognosis.Spontaneous spinal subdural hematoma with no underline pathology is a very rare condition. Only 19 cases have been previously reported.The case of a 44-year-old man is presented. Pubmed (Medline) was used to search publications.Our patient presented with sudden severe low back pain following a minimal effort, with rapid onset of complete paraplegia. MRI revealed an anterior subdural hematoma from T2–T6 with cord compression. An urgent laminectomy was performed. MRI scan, surgery, and CT angiogram did not reveal any underlying pathology to account for the subdural hematoma. The patient demonstrated substantial clinical improvement after 6 weeks of bed rest and intense rehabilitation program.Spinal subdural hematoma (SSDH) is uncommon and can be caused by abnormalities of coagulation, blood dyscrasias, or trauma, underlying neoplasm, and arteriovenous malformation. SSDH is very rare in the absence of these underlying conditions. It occurs most commonly in the thoracic spine and presents with sudden back pain radiating to the arms, legs or trunk, varying degrees of motor, sensory, and autonomic disturbances. On MRI, SSDH is seen as a space-occupying lesion, usually ventrally, contained within the dura matter, and can demonstrate variable T1 and T2 signal depending on the age of the hematoma. The prognosis is variable. The majority of cases in the literature had surgical decompression, although cases that were managed conservatively have been reported to be successful as well. The indications of surgery need to be clarified.