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Abstract:  Isotretinoin for acne vulgaris in the setting of chronic granulomatous disease (CGD) is a controversial therapeutic option. In this inherited immunodeficiency, inefficient tissue responses to bacteria and fungi lead to chronic and recurrent infections. Isotretinoin is known to be associated with several mucocutaneous side effects that could worsen the immune response in individuals with CGD. We report the fourth published case of acne vulgaris treated with isotretinoin in an individual with CGD, with a safe and successful outcome.