ABSTRACT
Allopurinol-induced hypersensitivity syndrome is characterized by an idiosyncratic reaction involving multiple-organs, which usually begins 2 to 6 weeks after starting allopurinol. In rare cases, the adverse reactions to allopurinol are accompanied by a variety of liver injury, such as reactive hepatitis, granulomatous hepatitis, vanishing bile duct syndrome, or fulminant hepatic failure. Here we report a case with granulomatous hepatitis and ductopenia. A 69-year-old man with chronic renal failure, hyperuricemia, and previously normal liver function presented with jaundice, skin rash, and fever 2 weeks after taking allopurinol (200 mg/day). In histopathology, a liver biopsy specimen showed mild spotty necrosis of hepatocytes, marked cholestasis in parenchyma, and some granulomas in the portal area. There were vacuolar degeneration in the interlobular bile ducts and ductopenia in the portal tracts. Pathologic criteria strongly suggested the presence of allopurinol-induced granulomatous hepatitis with ductopenia and cholestasis. The patient fully recovered following the early administration of systemic corticosteroid therapy.
Subject(s)
Allopurinol/adverse effects , Antimetabolites/adverse effects , Bile Duct Diseases/chemically induced , Bile Ducts, Intrahepatic/drug effects , Chemical and Drug Induced Liver Injury/pathology , Cholestasis/chemically induced , Granuloma/chemically induced , Aged , Allopurinol/therapeutic use , Antimetabolites/therapeutic use , Bile Duct Diseases/diagnosis , Bile Duct Diseases/pathology , Bile Ducts, Intrahepatic/pathology , Cholestasis/diagnosis , Cholestasis/pathology , Drug Eruptions/pathology , Granuloma/pathology , Humans , Kidney Failure, Chronic/complications , Kidney Failure, Chronic/drug therapy , MaleABSTRACT
Pneumatosis cystoides intestinalis is an uncommon condition of unknown etiology, characterized by the presence of multiple gas filled cysts in the gastrointestinal tract. Many different causes of pneumatosis cystoides intestinalis have been proposed, including mechanical, pulmonary, and bacterial causes. Approximately 85% of cases are thought to be secondary to coexisting disorders of the gastrointestinal tract or the respiratory system. The condition has been associated with the therapeutic uses of lactulose, steroids, and various cancer chemotherapeutic regimens. Lactitol is a disaccharide analogue of lactulose which is available as a pure crystalline powder. There are three previous case reports suggestive of lactulose causing pneumatosis intestinalis. We report a case of recurrent pneumatosis cystoides intestinalis associated with benign recurrent pneumoperitoneum developed probably secondary to lactitol therapy.