Twin reversed arterial perfusion (TRAP) sequence in association with VACTERL association: a case report

Abstract

IntroductionTwin reversed arterial perfusion (TRAP) sequence is a rare complication of multiple pregnancy caused by defects in early embryogenesis. The pump twin supplies the acardiac recipient twin with blood, and although the pump twin is usually structurally normal, congenital anomalies have sometimes been reported. We report a unique case of twin reversed arterial perfusion sequence with a prenatal diagnosis of VACTERL association in the surviving pump twin.Case presentationA 24-year-old Caucasian woman presented at 11 weeks' gestation with a monochorionic, monoamniotic twin pregnancy. A reversed arterial flow was noted on a Doppler imaging study coming from the larger, apparently normal twin to the smaller, grossly abnormal twin, and a diagnosis of twin reversed arterial perfusion sequence was made. Cardiac activity was undetectable in the recipient twin by 16 weeks' gestation. Further detailed assessment at 18 weeks' gestation revealed multiple congenital anomalies of the surviving pump twin, in keeping with a diagnosis of VACTERL association. A live infant girl was delivered at 39 weeks by elective cesarean section. She underwent extensive surgery with subsequent normal development at the age of two years.ConclusionThe co-existence of two rare and complex conditions in this unique case raises interesting questions about the role of early defects in embryogenesis and their subsequent effects on fetal development. This case also highlights the importance of prenatal diagnosis of major congenital anomalies to the plan treatment, reduce morbidity and aid the survival of affected children.

Prenatal diagnosis of the VACTERL association using routine ultrasound examination

“…Because severe cardiac malformations may not be compatible with extrauterine life, they may be more common in fetal populations (Khoshnood et al, ). However, their prenatal detection rate was low in our study when compared with the detection rate reported in the literature (Gembruch and Hansmann, ; Tongsong et al, ; Miller and Kolon, ; Chen et al, ; Athwal et al, ). In our study, most of the prenatally detected cases survived presumably because they were mainly nonsevere isolated septal defects.…”

“…The literature reports on the prenatal detection of vertebral anomalies in 16 cases of VACTERL association (Claiborne et al, ; Gembruch and Hansmann, ; McGahan et al, ; Baarsma et al, ; Tongsong et al, ; Miller and Kolon, ; Ardiet et al, ; Gedikbasi et al, ; Athwal et al, ; Chen et al, ). In our study, however, prenatal detection of vertebral malformations was low (12.5%), because they did not include segmentation defects and abnormal curvatures of the spinal cord (Wax et al, ).…”

Conservative management of single fetal death in twin pregnancy at a tertiary health institution in southern Nigeria: a case report

“…Here, we present the management of a case of single fetal death of a twin which was discovered during pregnancy and subsequently managed conservatively for 33 more days to improve the chance of fetal survival and then delivered vaginally at term following a successful induction of labour. Similar cases had been reported in other parts of the world which were also managed conservatively until delivery [7][8][9][10].…”

“…However, situations may arise sometimes in which caesarean section may be indicated and the rate varies considerably ranging from 19 -92% [12]. In a number of cases reported, delivery was by caesarean section, the indication in most cases being fetal heart rate irregularities [8,9,10]. Cord abnormality (commonly torsion of the umbilical cord) was observed in some of these fetuses and was thought to have caused intrauterine hypoperfusion resulting in the death of a twin.…”

Acardiac Twin: A Rare Case Report

“…The early pressure flow in one twin exceeds that of other, leading to the reversal of flow in the umbilical artery of the co-twin. 12,16 ii. The alternative hypothesis is that the acardia is a primary defect in embryogenesis in one twin leads to failure of cardiac development.…”