Sickle cell disease is a common hematologic disorder with some uncommon clinical manifestations. While there are an abundance of described disease manifestations of the head and neck, extramedullary hematopoesis within the paranasal sinuses is exceedingly rare. Here, we present a case of a young boy with known sickle cell disease who presented to hospital with headaches of new onset. Imaging demonstrated an expansile mass lesion occupying the sphenoid sinus and on biopsy was found to be active hematopoetic bone marrow. A description of the case and review of subtle but characteristic radiographic findings follow.
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