Objective: A case of dapsone hypersensitivity syndrome (DHS) in an AIDS patient receiving dapsone for Pneumocystis pneumonia (PCP) prophylaxis is discussed. Case Summary: A 50-year-old Caucasian male with a history of AIDS was admitted with suspicion of pneumonia. The clinical presentation consisted of shortness of breath, occasional hemoptysis, odynophagia, skin rash, transaminitis, anemia, malaise, and peripheral neuropathy. His medications included nystatin, fluconazole, valacyclovir, and pantoprazole. For PCP prophylaxis, the patient was initiated on dapsone 100 mg daily approximately 4 weeks prior to admission. The patient was also on a 3-drug regimen for the treatment of mycobacterium avium complex: clarithromycin, rifabutin, and ethambutol. After extensive work up, DHS was diagnosed. Dapsone was immediately discontinued and the patient was started on intravenous methylprednisolone, gradually tapered, and transitioned to oral prednisone. On discontinuation of dapsone, the patient improved, evidenced by decreased need for oxygen and reversal of peripheral neuropathy, anemia, and transaminitis. The patient's skin rash improved within a few days. An objective causality assessment, with the Naranjo scale, suggests that DHS was probably related to dapsone. Discussion: Our patient only exhibited some of the classic symptoms observed in DHS such as skin eruptions, malaise, hepatic failure, and hemolytic anemia. The patient, however, did not present with lymphadenopathy and fever commonly noted in DHS, which may be attributed to his immunosuppressed state, evidenced by a CD4 count of 7 cell/mm 3 . Conclusions: A patient on dapsone for PCP prophylaxis was diagnosed with DHS. Clinicians and patients should be educated on risk factors and clinical presentation of DHS so that when initial symptoms appear they can be treated promptly. Clinicians should also be aware that patients infected with HIV/AIDS may not present with all the classic symptoms of DHS due to their immunocompromised state. Therefore, practitioners should be vigilant when initiating dapsone in this patient population.