Background: Congenital entropion and atrichosis of the lower eyelids associated with tarsal hypoplasia is a rare condition, with isolated reports in the literature and without previous pathologic examination.
Methods: Case report. A father and son with mild entropion, lower eyelash atrichosis, and tarsal hypoplasia are described. A family pedigree demonstrates autosomal dominant inheritance.
Results: Histopathology from an entropion repair shows rudimentary tarsus and Meibomian glands consistent with hypoplasia of the tarsus.
Conclusions: Isolated congenital entropion may occur in association with atrichosis of the lower eyelashes and hypoplasia of the tarsus as an isolated anomaly. The condition may be inherited as an autosomal dominant trait and appears to be well tolerated.