Renal dysgenesis in a monozygotic twin: association with in utero exposure to indomethacin.

Abstract

We report oligohydramnios and renal dysgenesis in one of identical twins, which might have resulted from in utero exposure to early, prolonged high-dose indomethacin. The proposita was the second of twin girls born at 36 weeks of gestation. Pregnancy was complicated initially by polyhydramnios in both amniotic sacs and premature uterine contractions. After administration of indomethacin and terbutaline from 16 to 30 weeks' gestation, serial prenatal ultrasound examinations ultimately showed oligohydramnios in twin B and resolution of polyhydramnios in twin A. On day 5 twin B developed hematuria, hypertension, renal failure, hyponatremia, hyperkalemia, metabolic acidosis, sodium wasting and severe, transient inability to excrete potassium. Renal sonography showed enlarged, hyperechoic kidneys with almost no corticomedullary differentiation. Renal biopsy revealed immature glomeruli, dilated Bowman's spaces, dilated tubules, and interstitial fibrosis. The liver was histologically normal. Indomethacin may induce oligohydramnios and transient renal insufficiency in humans and renal dysgenesis in fetal monkeys; it might have induced the abnormalities in this patient.

• Authors: I Restaino, B S Kaplan, P Kaplan, H K Rosenberg, C Witzleben, N Roberts
• Journal: American journal of medical genetics (Am J Med Genet) Vol. 39 Issue 3 Pg. 252-7 (Jun 01 1991) ISSN: 0148-7299 [Print] United States
• PMID: 1867274 (Publication Type: Case Reports, Journal Article)

Chemical References

• Indomethacin

Topics

• Adult
• Diseases in Twins
• Female
• Humans
• Indomethacin (administration & dosage, adverse effects)
• Infant, Newborn
• Kidney (abnormalities, drug effects, physiopathology)
• Maternal-Fetal Exchange
• Obstetric Labor, Premature (drug therapy)
• Polyhydramnios (drug therapy)
• Pregnancy
• Twins, Monozygotic