We report on a combined cutaneous smooth muscle hamartoma and reticulate port-wine stain on the thigh of a 26-year-old woman. The lesion measured 30 cm in longest diameter and presented clinically with a macular erythematous telangiectatic vascular appearance. It was focally ulcerated, which represented the patient's main complaint. Histopathological study demonstrated smooth muscle bundles closely intermingled with a vascular component comprised of capillaries and venules. Such a morphological presentation is, to the best of our knowledge, unique in the literature.