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Fatal cholestatic hepatitis caused by D-penicillamine
J W Jacobs1, F R Van der Weide, M W Kruijsen
1Department of Rheumatology, University Hospital Utrecht, The Netherlands.
British Journal of Rheumatology
|August 1, 1994
Summary
D-penicillamine therapy can cause rare cholestatic hepatitis in rheumatoid arthritis patients. This case highlights a persistent and severe form, emphasizing the need for vigilant monitoring of D-penicillamine adverse effects.
Area of Science:
- Hepatology
- Rheumatology
- Pharmacology
Background:
- Rheumatoid arthritis (RA) is an autoimmune disease often treated with disease-modifying antirheumatic drugs (DMARDs).
- D-penicillamine is a chelating agent used as a DMARD, known for potential adverse effects.
- Intrahepatic cholestasis is a liver condition characterized by impaired bile flow.
Observation:
- A 37-year-old female with rheumatoid factor-positive RA developed cholestatic hepatitis 10 days after initiating D-penicillamine.
- The D-penicillamine was immediately discontinued, but cholestasis persisted for 14 months.
- The patient subsequently developed severe hypercholesterolemia, xanthelasmata, and pancytopenia due to bone marrow infiltration by lipid-laden foam cells.
Findings:
- This case represents a rare and severe idiosyncratic reaction to D-penicillamine.
- The persistent cholestatic icterus and subsequent complications are documented here for the first time.
- Literature review confirms intrahepatic cholestasis as a rare complication of D-penicillamine.
Implications:
- Clinicians should be aware of the potential for severe, persistent cholestatic hepatitis with D-penicillamine use.
- Early recognition and discontinuation of the drug may not always reverse the cholestatic process.
- This case underscores the importance of monitoring liver function and hematologic parameters in patients receiving D-penicillamine.