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Efficacy of COX-2 inhibitors in a case of congenital nephrogenic diabetes insipidus

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Abstract

A 17-month-old boy presented with failure to thrive, polyuria, and vomiting. He had been diagnosed clinically with nephrogenic diabetes insipidus and treated by amiloride and hydrochlorothiazide combination without a satisfactory outcome at another center since 1 year of age. The diagnosis was confirmed by genetic analysis (AVPR2mutation), and the treatment was modified to include rofecoxib (a selective cyclooxygenase-2 inhibitor) in addition to hydrochlorothiazide and amiloride. This combination along with a low-salt diet resulted in a dramatic decrease in urinary free-water loss, while no side effect was noted. Because of prohibition of rofecoxib, it had to be substituted first by indomethacin and then by ibuprofen. However, both drugs were ineffective in controlling water diuresis. Thus, we had to replace these drugs by celecoxib (another selective cyclooxygenase-2 inhibitor). We conclude that the combination hydrochlorothiazide/amiloride/cyclooxygenase-2 inhibitor could be successfully used to treat congenital nephrogenic diabetes insipidus.

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Acknowledgement

We thank Professor Nine Knoers, University Medical Centre Nijmegen, Department of Human Genetics, the Netherlands, for the genetic analyses.

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Authors and Affiliations

  1. Dokuz Eylül University Medical Faculty, Department of Pediatrics, Izmir, Turkey

    Alper Soylu, Belde Kasap, Nilüfer Öğün, Yeşim Őztürk, Mehmet Türkmen & Salih Kavukçu

  2. University Medical Centre Nijmegen, Department of Human Genetics, Nijmegen, The Netherlands

    Lies Hoefsloot

  3. Mithatpaşa Cad. No:665/4, Küçükyalı, 35280, Izmir, Turkey

    Salih Kavukçu

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  1. Alper Soylu
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  2. Belde Kasap
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  3. Nilüfer Öğün
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  4. Yeşim Őztürk
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  5. Mehmet Türkmen
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  6. Lies Hoefsloot
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  7. Salih Kavukçu
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Correspondence to Salih Kavukçu.

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Soylu, A., Kasap, B., Öğün, N. et al. Efficacy of COX-2 inhibitors in a case of congenital nephrogenic diabetes insipidus. Pediatr Nephrol 20, 1814–1817 (2005). https://doi.org/10.1007/s00467-005-2057-8

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  • DOI: https://doi.org/10.1007/s00467-005-2057-8

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