We report a case of ductopenia associated with cholestatic hepatitis i
n a 59-year-old woman treated for 41 years for temporal epilepsy, The
patient developed jaundice, without any clinical or biochemical featur
es of hypersensitivity, 10 months after the beginning of treatment wit
h sulpiride, Liver biopsy showed ballooning and acidophilic degenerati
on of the hepatocytes, macrophages packed with lipofuscin, biliary pig
ment in Kupffer cells, some biliary plugs, confluent necrosis and abse
nce of biliary ducts in all the portal tracts. These features and the
presence of foci of cholangiolitis suggest a destructive cholangitis a
s the pathogenetic mechanism causing ductopenia. Other causes of ducto
penia were excluded, Sulpiride is known to produce severe cholestatic
jaundice, which we believe is due to ductopenia. The absence of hypers
ensitivity and the 10-month latency suggest that sulpiride may cause l
iver damage through a toxic mechanism in genetically susceptible subje
cts.