Visual dysfunction developing in association with acquired immunodeficiency syndrome (AIDS) can be multifactorial. Two patients with this syndrome and cryptococcal meningitis had papilledema and visual loss. Both were treated by optic nerve sheath fenestration. One patient had bilateral nonsimultaneous optic nerve sheath fenestrations; visual function improved in one eye. The other patient had bilateral visual improvement after a unilateral optic nerve sheath fenestration. Cryptococcal organisms were present in the dural sheath specimens of both patients despite ongoing therapy with antifungal medication. Postoperative orbital infectious complications did not occur. Autopsy examination of one patient showed that the sites of fenestration were patent. Medical treatment of cryptococcal meningitis associated with AIDS has a guarded prognosis. Optic nerve sheath fenestration offers a treatment alternative for papilledema and visual loss that occur with cryptococcal meningitis.