Successful management of an extreme example of neonatal hyperprostaglandin-E syndrome (Bartter's syndrome) with the new cyclooxygenase-2 inhibitor rofecoxib.

Abstract

OBJECTIVE:

To describe the successful treatment of an unusual case of severe neonatal Bartter's syndrome refractory to treatment with indomethacin.

DESIGN:

Case report, clinical.

SETTING:

Tertiary care intensive care unit.

PATIENTS:

A patient with neonatal hyperprostaglandin-E syndrome and excessive requirements of intravenous (via central venous catheter) water and salt supplementation, failure to thrive, vomiting, and massive growth retardation, despite adequate treatment with indomethacin.

MAIN RESULT:

Four weeks after induction of the new cyclooxygenase-2 inhibitor rofecoxib, the patient was well, on full enteral feeds, thriving, and had gained 600 g in weight. A lower supplementary potassium, magnesium, and sodium intake was required. Reinstitution of indomethacin therapy resulted in severe deterioration, despite high indomethacin doses; symptoms improved again after rofecoxib administration. No side effects have been seen thus far.

CONCLUSION:

This report shows that in selected patients with a severe form of neonatal Bartter's syndrome, the new cyclooxygenase-2 inhibitor rofecoxib may control the clinical symptoms of hyperprostaglandin-E syndrome after ineffective indomethacin therapy.

• Authors: Nikolaus A Haas, Robert Nossal, Christoph H Schneider, Martin A G Lewin, Volker Ocker, Martin Holder, Frank Uhlemann
• Journal: Pediatric critical care medicine : a journal of the Society of Critical Care Medicine and the World Federation of Pediatric Intensive and Critical Care Societies (Pediatr Crit Care Med) Vol. 4 Issue 2 Pg. 249-51 (Apr 2003) ISSN: 1529-7535 [Print] United States
• PMID: 12749662 (Publication Type: Case Reports, Journal Article)

Chemical References

• Cyclooxygenase Inhibitors
• Lactones
• Sulfones
• rofecoxib

Topics

• Bartter Syndrome (drug therapy, physiopathology)
• Cyclooxygenase Inhibitors (therapeutic use)
• Humans
• Infant, Newborn
• Lactones (therapeutic use)
• Sulfones