Dura mater involvement in ochronosis (alkaptonuria)

Clinical manifestations of alkaptonuria have been well described and are mo st commonly characterized by ochronosis or pigmentation of connective tissu e. Sites most commonly involved in ochronosis include joints, heart, skin, and kidney. We describe a 66-year-old woman with a history of alkaptonuria who had widespread ochronosis. The dura mater showed extensive pigment depo sition, which was evident both grossly and microscopically at autopsy. To o ur knowledge, description of dura mater involvement by ochronosis has not b een previously reported in the literature.