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© 1994 British Society for Rheumatology

case-report

FATAL CHOLESTATIC HEPATITIS CAUSED BY D-PENICILLAMINE

J. W. G. JACOBS*, F. R. VAN DER WEIDE* and M. W. M. KRUIJSEN{dagger}

*Department of Rheumatology, F02.223, University Hospital Utrecht PO Box 85500,3508 GA Utrecht
{dagger}Hospita1 ‘Medisch Spectrum Twente’ Enschede, The Netherlands

A 37-yr-old woman with RF-positive RA developed cholestatic hepatitis after 10 days of D-penicillamine therapy. This was discontinued immediately. The cholestasis persisted for the remaining 14 months of her life. Severe hypercholesterolemia developed with xanthelasmata and eventually pancytopenia, which was caused by a massive infiltration of the bone marrow by lipid containing foam cells. The patient died of sepsis. Review of the literature shows intrahepatic cholestasis to be a rare and idiosyncratic complication of D-penicillamine therapy. To our knowledge, ours is the first documented case of persistent cholestatic icterus.

KEY WORDS: Cholestatic hepatitis, Jaundice, Adverse reactions, Drug-allergy, D-Penicillamine


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