Congenital infiltrating lipomatosis of the face is a rare entity that has not been reported in the ophthalmic literature. The authors describe two patients affected by this condition. Diagnosis and management of congenital infiltrating lipomatosis of the face involving the eye adnexa is challenging and the psychological impact on such patients must be considered. [J Pediatr Ophthalmol Strabismus. 2022;59(3):e46-e49.].