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.
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: Cinzia Cortesi; Sebastiano A G Lava; Alberto Bettinelli; Fabiana Tammaro; Olivier Giannini; Maria Caiata-Zufferey; Mario G Bianchetti Journal: Pediatr Nephrol Date: 2010-06-13 Impact factor: 3.714
Authors: Jeffery T Fletcher; Nicole Graf; Anthony Scarman; Hamda Saleh; Stephen I Alexander Journal: Pediatr Nephrol Date: 2006-09-06 Impact factor: 3.714