Successful treatment of cystinuria with captopril

Cystinuria is an autosomal recessive hereditary disorder associated with nephrolithiasis and its attendant complications. Traditional management aimed at increasing urinary cystine solubility using oral alkali, D-penicillamine, or mercaptopropionyglycine is often unsuccessful due to intolerable side effects. Two cystinuric patients intolerant of traditional therapy were treated with captopril for 1 year, resulting in a marked decline in urinary cystine excretion. Neither patient experienced recurrent nephrolithiasis or adverse drug effects. Captopril should be considered an alternative to traditional drug management of cystinuria.