Caustic sclerosing cholangitis treated with orthotopic liver transplantation.

Caustic sclerosing cholangitis, so named by Belghiti et al. in 1986 [1], is a rare entity caused by injection of a scolicidal solution into hydatid cysts during surgery. Until now, only 40 cases have been reported in the world medical literature [2], and the histologjc features are not well described. We report a case of a teenage girl with formaldehyde-induced sclerosing cholangitis who was treated by orthotopic liver transplantation. This enabled us to study in detail the macroscopic and microscopic features of this entity. To the best of our knowledge, this is the first reported case of caustic sclerosing cholangitis in a patient who had previously undergone liver transplantation. We describe the unique macroscopic features, characterized by severe scarring with a calcified mass in the area of previous injection of formaldehyde.