AbstractWe present the case of a congenital, deep ulcer of the buttock in an otherwise healthy newborn girl. Histopathology excluded any specific cause of ulceration. The ulcer healed spontaneously at 4 weeks of age, leaving a scar. Because the patient also had symmetrical pits on her knees and because the ulcer was located in an area typically affected by dimples, we interpret this unique manifestation as a congenital defect in the spectrum of congenital dimples.