Dual intestinal anomalies in dizygotic twins

Abstract

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.

Prenatal imaging features and perinatal outcomes of foetal volvulus–A literature review

“…Three (3.4%) infants 19,27,57 developed sepsis in the perinatal period, one of which also developed an intraventricular haemorrhage. Prelabour rupture of membranes occurred in four (4.5%) cases, 6,12,21,47 although none of these cases had recorded sepsis.…”

“…In cases where gestation was recorded at diagnosis ( n = 80), ultrasound features of the underlying pathology were visualised at a median gestational age of 31.9 weeks (IQR 27–34) with 65.6% of signs first manifesting between 30 and 36 weeks. Twenty‐three (26.1%) cases 3,5,6,9,12,14,17,19,24,29,32,34,42–48 documented progression of ultrasound findings as gestation advanced (Table ). Twenty (22.7%) women 3,4,11,15,24,25,29–35,49 noted a decrease in foetal movements.…”

“…Overall, 61/77 (79.2%) infants 3–6,8,9,11,12,14,15,18–21,24,26,27,29–31,33–35,42–52,54–59,63,64 were delivered before 37 completed weeks gestation. Gestation at diagnosis was reported in 80 (90.9%) cases 2–7,9,11–22,24–27,29,30,32–35,42–63,65 with a median gestation of 31.9 weeks (IQR 27–34)—in 32 of these cases 2,4,5,11,14,16,17,24–27,30,31,34,35,42,43,45–49,51,52,56–58 ultrasound findings precipitated iatrogenic delivery at a mean gestation of 34.2 weeks (SD 2.3). In 72 (81.8%) cases, 2–6,9,11,12,14–20,22,24–27,29–35,42,43,45–49,51–59,63–68 the mode of birth was reported.…”

“…In all cases of multiple pregnancy only one twin was affected. Birth weight was reported for 61 infants 2,3,5–9,11,12,14,16,18–22,24–27,29–35,42,43,45–49,51–54,57–59,63–68 —the mean birthweight was 2400.2 g (SD 628.4). Of these, 11/61 (18.0%) 16,19,24,46–48,51,53,57 were classified as SGA.…”

“…Three (3.4%) foetuses 29,43,52 underwent prenatal paracentesis for foetal ascites, and 1 (1.1%) 27 underwent diagnostic cordocentesis. Following delivery, 71 (80.7%) cases 2–4,6–9,12–15,17–22,24–27,29–31,33–35,42–49,51–54,56–61,63–66 had recorded timeframes for treatment of volvulus. Fifty‐three (53/71, 74.6%) of these infants 2,4–8,14–17,19–21,24–26,30,31,33–35,42–47,49,51,52,54–59,63 were treated immediately after birth or within 24 h of age.…”