Purpose We report the case of a 10-year old boy who had been diagnosed with megalencephalic leukoencephalopathy several years earlier. Because of the patient's oral dystonic activity, a traumatic, nonhealing, chronic ulcer had developed on his lower lip. Materials and Methods Botox-A was injected into the mentalis, orbicularis oris, and bilateral masseter muscles. Results The patient showed decreased dystonia and gradual complete healing of the traumatic ulcer of the lower lip. Conclusions The treatment of patients with self-mutilation to the lips will often be difficult. Traditionally, patients have been treated with various medications, oral appliances, and even tooth extraction. The results of the present case report suggest that Botox should be considered as a possible first-line strategy, along with oral appliances.