Failure of ursodeoxycholic acid to prevent a cholestatic episode in a patient with benign recurrent intrahepatic cholestasis: a study of bile acid metabolism.

[benign recurrent intrahepatic cholestasis]

Ursodeoxycholic acid was administered to a patient with benign recurrent intrahepatic cholestasis to prevent cholestatic episodes . A detailed study of bile acid metabolism in this patient was carried out in the anicteric and icteric phases before and after ursodeoxycholic acid (750 mg /day ) administration . Urinary , biliary and serum bile acids were measured by gas chromatography-mass spectrometry and by high -performance liquid chromatography techniques . During the anicteric phase the daily urinary excretion and serum concentrations of bile acids were within normal ranges , indicating normal hepatic uptake and secretion of bile acids during the cholestasis -free period . Only slight qualitative differences from normal individuals were observed ; the relative proportions of deoxycholic acid in the bile and serum were higher , and 12 -oxo-lithocholic acid was the predominant urinary bile acid . During the icteric phase a marked increase in the urinary excretion of primary bile acids and C-1 , C-2 , C-4 and C-6 hydroxylated metabolites was found . Serum bile acid concentrations increased before the rise in bilirubin , suggesting an acute disturbance in bile acid transport at the onset of the cholestatic attack . After ursodeoxycholic acid administration in the anicteric phase , bile became enriched with the exogenous bile acid , but little qualitative change was found in the other metabolites present in the urine , serum or bile during the anicteric or icteric phases . Prolonged administration of ursodeoxycholic acid failed to prevent recurrence of a cholestatic episode , suggesting that in benign recurrent intrahepatic cholestasis , oral ursodeoxycholic acid may be of little benefit in the treatment or prevention of cholestasis despite marked enrichment of the bile acid pool with this hydrophilic bile acid .

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Diseases presenting "marked increase in the urinary excretion" symptom

benign recurrent intrahepatic cholestasis

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