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Abstract Spontaneous extradural hematoma is rare in patients with sickle cell disease. We report a clinical case of a 19-year-old young man with sickle cell anemia who presented a sickle cell crisis complicated by the development of multiple acute extradural and subgaleal hematomas that had not been treated surgically. We discuss the physiopathology of this event. Although it is rare, clinicians should be aware of this phenomenon as part of a spectrum of neurologic complications in these patients.