Abstract
Indomethacin improves the polyuria associated with Fanconi syndrome in patients with cystinosis. However, its use may be vitiated by its deleterious effect on GFR.
J.B. presented at 4 y with cystinosis. After correction of the acid base and serum electrolyte abnormalities with supplemental therapy, indomethacin, 2 mg/kg/d was started. Urine output decreased from 1.9 to 0.9 1/d and she gained weight. 3 y later, serum creatinine suddenly increased from 2.3 to 3.8 mg/dl. After stopping indomethacin, the creatinine decreased to 2.5 mg/dl but polyuria reappeared.
H.T. was diagnosed at 14 m. Indomethacin, 2 mg/kg/d was instituted; urine output decreased from 2.3 to 1.3 1/d and she gained weight. 2 y later, serum creatinine rose from 1.4 to 2.6 mg/dl and the drug was stopped. 2 w later, creatinine was 1.7 mg/dl. Because of rapid reappearance of polyuria associated with irritability, indomethacin was re-started at 1 mg/kg/d. Urine output decreased and 2 m later, creatinine stabilized at 1.4 mg/dl.
Indomethacin is definitely effective in reducing polyuria in cystinotic patients, but can also produce a reversible increase in the serum creatinine concentration.
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Lemire, J., Kaplan, B. 1517 PROLONGED USE OF INDOMETHACIN IN CYSTINOSIS. Pediatr Res 15 (Suppl 4), 696 (1981). https://doi.org/10.1203/00006450-198104001-01540
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DOI: https://doi.org/10.1203/00006450-198104001-01540