Treatment of congenital nephrogenic diabetes insipidus by hydrochlorothiazide and cyclooxygenase-2 inhibitor.

Abstract	A 1-month-old male infant presented with failure to thrive, polyuria, and severe hypernatremic dehydration. Based on family history, lack of response to vasopressin, and normal sonography of the urinary system, the diagnosis of congenital nephrogenic diabetes insipidus (cNDI) was established. The infant responded well to indomethacin in combination with hydrochlorothiazide (HCTZ), but quickly developed gastrointestinal bleeding. The substitution of indomethacin by amiloride and later by tolmetin was found to be ineffective. Treatment with HCTZ (3 mg/kg per day) and rofecoxib (1 mg/kg per day, both divided into three doses) combined with a low-salt formula resulted in a dramatic decrease in urinary free water losses. No side effects of the combination were noted. At age 8.5 months, the infant demonstrated catch-up growth and normal neurodevelopmental milestones. We conclude that the combination HCTZ/cyclooxygenase-2 inhibitor could be successfully used to treat infantile cNDI.
Authors	Anirut Pattaragarn, Uri S Alon
Journal	Pediatric nephrology (Berlin, Germany) (Pediatr Nephrol) Vol. 18 Issue 10 Pg. 1073-6 (Oct 2003) ISSN: 0931-041X [Print] Germany
PMID	12883974 (Publication Type: Case Reports, Journal Article, Research Support, Non-U.S. Gov't)
Chemical References	Cyclooxygenase 2 Inhibitors Cyclooxygenase Inhibitors Diuretics Isoenzymes Lactones Membrane Proteins Sodium Chloride Symporter Inhibitors Sulfones Hydrochlorothiazide rofecoxib Cyclooxygenase 2 PTGS2 protein, human Prostaglandin-Endoperoxide Synthases
Topics	Cyclooxygenase 2 Cyclooxygenase 2 Inhibitors Cyclooxygenase Inhibitors (administration & dosage) Diabetes Insipidus, Nephrogenic (drug therapy) Diuretics Drug Therapy, Combination Humans Hydrochlorothiazide (administration & dosage) Infant, Newborn Isoenzymes (antagonists & inhibitors) Lactones (administration & dosage) Male Membrane Proteins Prostaglandin-Endoperoxide Synthases Sodium Chloride Symporter Inhibitors (administration & dosage) Sulfones

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