Abstract
A case of primary sclerosing cholangitis (PSC) following extirpation of chronic sclerosing sialadenitis of the submandibular gland (so-called Kuttner's tumor) was reported. To our knowledge no case of PSC has been reported in association with Kuttner's tumor. The pathogenesis of both disorder is still unknown, although vari ous hypotheses have been disathsed in the literature, e. g. autoimmunity. The patient, a 62-year-old male, was admitted to our hospital with liver dysfunction and jaundice. He was experienced extirpation of bi-lateral submandibular glands 1 year 8 months ago, and the pathological diagnosis of specimens was so-called Kuttner's tumor. The examination of ultrasound revealed thickening and layered structure of the common hepatic duct, and cholangiographical evaluation revealed marked stricture of that portion. Cytological examination showed no malignancy, thus diagnosed as PSC, cholecysto-choledochotomy and hepatico-jejunostomy were carried out. Histopathological examination of the resected bile duct showed inflammatory reaction with increased collagen fiber and chronic inflammatory cell-infiltration of lymphocytes. The patient has been in good health for 3 years and 6 months after resection.
