Dual intestinal anomalies in dizygotic twins.

Fiche publication


Date publication

août 2018

Journal

BMJ case reports

Auteurs

Membres identifiés du Cancéropôle Est :
Dr CHAUSSY Yann, Pr AUBER Frédéric


Tous les auteurs :
Ridley A, Chaussy Y, Mottet N, Auber F

Résumé

We report on the case of two digestive malformations in dizygotic/dichorionic/diamniotic twins born at 31 weeks of gestation. The mother (gravida 1 para 0) was treated by hydroxychloroquine for systemic lupus erythematosus during pregnancy. Twin A presented an arch-like dilatation on antenatal ultrasounds, characteristic of segmental volvulus. After birth, twin B presented repeated vomiting on feeding, leading us to diagnose ileal atresia despite normal antenatal ultrasounds. Both twins underwent surgery and the postoperative period was uneventful. After 1 year of follow-up, the twins are in excellent health without digestive sequelae. Genetic testing for cystic fibrosis was negative. The placenta showed diffuse signs of hypoxia and ischaemia, indicating that the root cause was vascular. The pathophysiology of intestinal atresia is hypothesised to derive from a vascular incident during fetal development. We are therefore led to believe that an intrauterine vascular event is the most likely cause of the dual malformation.

Mots clés

congenital disorders, gastrointestinal surgery, materno-fetal medicine, neonatal and paediatric intensive care, paediatric surgery

Référence

BMJ Case Rep. 2018 Aug 4;2018: