All-trans retinoic acid (ATRA) has been shown to improve the outcome in patients with acute promyelocytic leukemia compared with chemotherapy alone, but it is associated with adverse effects. We report the development of scrotal ulcer in a patient with acute promyleocytic leukemia (APL) within 10 days of treatment with ATRA at a dose of 40 mg orally twice daily. The ulcer did not respond to antibiotic treatment and healed shortly after withholding ATRA. The biopsy showed inflammation only, and other microbiological workup was negative.