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Unusual association of diseases/symptoms
CASE REPORT
Dual intestinal anomalies in dizygotic twins
  1. Ashley Ridley1,
  2. Yann Chaussy2,3,
  3. Nicolas Mottet3,4,
  4. Frédéric Auber2,3
  1. 1Réanimation pédiatrique, Centre Hospitalier Universitaire de Besancon, Besancon, France
  2. 2chirurgie pédiatrique, Centre Hospitalier Regional Universitaire de Besançon, Besançon, France
  3. 3Laboratoire Nanomédecine, Imagerie et Thérapeutique (NIT) - EA 4662, Université de Franche-Comté, Besançon, France
  4. 4Gynécologie-Obstétrique, Centre Hospitalier Regional Universitaire de Besançon, Besançon, France
  1. Correspondence to Dr Yann Chaussy, yannchaussy{at}hotmail.com

Summary

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.

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

https://doi.org/10.1136/bcr-2018-225176

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    Footnotes

    • Contributors AR: conception and design, drafting the article. YC: acquisition of data (surgical and radiological data), interpretation of data, revising manuscript and final approval. NM: acquisition of data (antenatal data), interpretation of antenatal data, revising manuscript and final approval. FA: conception and design, acquisition of data (surgical data), revising manuscript and final approval.

    • Funding The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors.

    • Competing interests None declared.

    • Patient consent Parental/guardian consent obtained.

    • Provenance and peer review Not commissioned; externally peer reviewed.