Abstract
Long-term copper balance studies were performed in fifteen patients with Wilson's disease, six of their parents and siblings, and one normal control subject. Patients with Wilson's disease required a daily dietary copper intake of 1.1 mg or less to attain negative copper balance whereas their family members were in balance at 1.4 mg/day. Penicillamine therapy elicited a markedly negative copper balance initially by increasing the urinary excretion of copper, but as therapy continued, both urinary and fecal copper excretion diminished. The effect of oral ehelators on fecal copper varied: DTPA was of no value; carbacrylamine resin and potassium sulfide increased the fecal copper content. Most patients with Wilson's disease could be maintained in negative copper balance with a daily dietary copper intake of 1.5 mg or less with these latter agents. The addition of these drugs offered no advantage over the use of penicillamine alone. The combination of a low copper diet and the oral chelating agent, carbacrylamine resin, should be considered for interim periods to treat presymptomatic Wilson's disease or in patients with clinical Wilson's disease whose tissue copper stores have been reduced by penicillamine therapy.
Published Version
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