A patient with steatocystoma multiplex was treated with 0.75 mg per kg per day of oral isotretinoin. After one month of minimal improvement the patient discontinued therapy. Over the next two months he noted some shrinkage of his pre-existing lesions, which persisted for a follow-up period of six months. Isotretinoin may show a beneficial response in some cases of steatocystoma multiplex even after use of the drug is discontinued. Isotretinoin is known to have effects beyond its treatment period that are related to its long serum half-life. A small number of patients with steatocystoma multiplex have been treated with isotretinoin, with varied responses. This is the first case of a patient who discontinued therapy before a response occurred and then showed a response later.