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Spontaneous spinal epidural hematoma (SSEH) is a rare condition, often causing significant neurologic morbidity owing to its insidious nature and difficulty in diagnosis. Initial nonspecific clinical findings make the timely diagnosis challenging. A variety of underlying etiologies predispose patients to SSEH such as anticoagulation therapy, bleeding diatheses, vascular malformations, tumors, as well as spontaneous and idiopathic cases. Early diagnosis and intervention are critical for optimal patient outcomes. Here, we present a case of SSEH where coagulopathy was originating from underlying cholestasis. This, to our knowledge, represents the first case reported in the literature where a primary cholestatic disease is the underlying etiology.