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Genital herpes is mostly caused by the herpes simplex virus Type 2 and is frequent in patients infected with the human immunodeficiency virus. In this setting, the disease may have atypical clinical presentations, including an unusual pseudotumoral hypertrophic form that may cause diagnostic and therapeutic problems, which usually shows an unsatisfactory response to first-line systemic antiretrovirals. The authors report a case of hypertrophic genital herpes refractory to oral acyclovir treatment and effectively treated with surgical excision, with no recurrence after the procedure.