Abstract
Objective To report the case histories of two children in whom cholestatic liver disease preceded other manifestations of histiocytosis X. To discuss the possible relevance of the timing and nature of liver involvement in the immunologie pathogenesis of this disease. Outcome In both patients, the diagnosis of histiocytosis X was made only upon the appearance of extrahepatic manifestations of the disease. A review of the initial liver biopsy specimens failed to reveal any evidence of histiocytic involvement. Conclusion In children with cholestatic jaundice simulating sclerosing cholangitis, a diagnosis of histiocytosis X should be considered and extrahepatic histiocytic involvement actively sought.
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