Two rare cases of distinctive thymic remnants occurring in the skin are described. The lesions were present at birth, and involved the side of the neck. One child had a complete cleft lip and palate. The other had multiple congenital anomalies consistent with a rare syndrome entitled branchio-oculo-facial syndrome. Microscopically, lobular foci were present in the dermis and consistent with both prethymic and thymic remnants. Immunohistochemical studies using antibodies to T cells and B cells showed a distribution similar to that seen in normal thymus. Dermal thymus appears to be a distinct entity and may be associated with other faciobranchial defects.