Successful management of an extreme example of neonatal hyperprostaglandin-E syndrome (Bartter's syndrome) with the new cyclooxygenase-2 inhibitor rofecoxib.
OBJECTIVE
To describe the successful treatment of an unusual case of severe neonatal Bartter's syndrome refractory to treatment with indomethacin.


METHODS
Case report, clinical.


METHODS
Tertiary care intensive care unit.


METHODS
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.


RESULTS
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.


CONCLUSIONS
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.
Department of Pediatric Cardiology and Pediatric Intensive Care, Olgahospital Stuttgart, Stuttgart, Germany.
Nikolaus A
NA
Haas
Robert
R
Nossal
Christoph H
CH
Schneider
Martin A G
MA
Lewin
Volker
V
Ocker
Martin
M
Holder
Frank
F
Uhlemann
D001477:Bartter Syndrome / Q000188:drug therapy* / Q000503:physiopathology; D016861:Cyclooxygenase Inhibitors / Q000627:therapeutic use*; D006801:Humans; D007231:Infant, Newborn; D007783:Lactones / Q000627:therapeutic use*; D013450:Sulfones
D016861:Cyclooxygenase Inhibitors; D007783:Lactones; D013450:Sulfones; C116926:rofecoxib