Abstract
A case of hepatolenticular degeneration with several unusual features is reported. Clinically, the course was marked by insidious onset with mental symptoms, later by major seizures with focal electroencephalographic abnormality, and terminally by fever and an allergic skin rash. Biochemical studies revealed the usual abnormalities found in this disorder with a marked diminution of ceruloplasmin, hypercupriuria, increased copper in tissue and aminoaciduria. Pathologic study revealed disseminated brain lesions which correlated well with the clinical picture of multiple system involvement. Of special interest was the finding of acute and subacute hepatic necrotic lesions. These lesions were chronologically related to the period of treatment with dimercaprol (BAL) and calcium disodium ethylenediaminetetraacetic acid (versenate). The use of these drugs also coincided with the acute febrile terminal course. Both the adverse clinical response and the toxic hepatic lesions appeared to have been aggravated or precipitated by the drug therapy.
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