Vanishing gut in infants with gastroschisis

Kimble, Roy M.; Blakelock, R. T.; Cass, Danny

doi:10.1007/s003830050644

Cited by 31 publications

(23 citation statements)

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“…In the majority of vanishing gastroschisis cases reported in the literature, an external stump of bowel adjacent to the umbilicus is evident at birth even when the underlying defect has closed [9,10,11,12,13,14,15]. In cases 1-3 the external abdominal appearances were entirely normal.…”

Section: Discussionmentioning

confidence: 88%

Gastroschisis and the Risk of Short Bowel Syndrome: Outcomes and Counselling

et al. 2013

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Background: Gastroschisis can be associated with short bowel syndrome (SBS). We present 4 cases with significant bowel shortening with very different ante- and postnatal presentations. Cases: Three of the cases demonstrated ‘vanishing' gastroschisis with progressively worsening antenatal intra-abdominal bowel dilatation with no defect present at birth. The fourth case followed an uneventful course antenatally but developed SBS considered secondary to gastroschisis-related atresia and necrosis. All 4 cases were classified as having SBS and were enrolled within the paediatric Intestinal Rehabilitation Programme. This involves autologous gastrointestinal reconstruction following a period of bowel expansion with concomitant parenteral and enteral nutrition. Discussion: These cases demonstrate that the serious complication of significant bowel loss cannot always be anticipated. Early multidisciplinary discussion with the parents enables a shared understanding of potential outcomes. It can aid the recognition of ‘vanishing' gastroschisis enabling expedient investigations and early intervention, minimising morbidity and maximising bowel length.

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Section: Discussionmentioning

confidence: 88%

Gastroschisis and the Risk of Short Bowel Syndrome: Outcomes and Counselling

et al. 2013

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“…A review of the literature shows that these cases are rare, since only 7 cases were found [1,3,[5][6][7][8][9] (table 1). The case presented demonstrates part of the natural history of this unusual complication of gastroschisis.…”

Section: Discussionmentioning

confidence: 99%

“…Kimble et al [3] reviewed 8 cases of reported extensive gut atresia with no childhood survivor. Table 1 shows that none of the previous reported cases of extensive resorption of extraabdominal bowels in fetuses with gastroschisis survived.…”

Section: Discussionmentioning

confidence: 99%

“…Intestinal atresia, which has been reported to occur in 5-30% of cases [1,2], is associated with a worse prognosis. In cases with extensive midgut atresia, mortality secondary to short gut syndrome, hyperalimenation and cholestatic liver insufficiency is invariable [3]. This is a presentation of a case of gastroschisis associated with extensive midgut atresia with an unusual presentation that survived after intestinal-liver transplantation.…”

Section: Introductionmentioning

confidence: 99%

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Gastroschisis Complicated by Midgut Atresia, Absorption of Bowel, and Closure of the Abdominal Wall Defect

Ogunyemi¹

2001

Fetal Diagn Ther

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We present a case of gastroschisis that was associated with progressive resorption of the extra-abdominal bowel loops and dilation of intra-abdominal bowel loops. After preterm delivery at 32 weeks, a small paraumbilical remnant was present. There was complete atresia of most of the jejunum, ileum, cecum, and the proximal half of the transverse colon. At laparotomy, the jejunum was anastomosed to the transverse colon. The neonate developed short gut syndrome and eventually received a liver and intestinal transplant.

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“…Another theory is that a volvulus occurs, which in turn leads to infarction and reabsorption of small bowel. 4 The baby will be born with either a small (<1 cm) or no abdominal wall defect remaining. The extrinsic bowel is then either reabsorbed or mummified.…”

Section: Introductionmentioning

confidence: 99%

Closed gastroschisis, vanishing midgut and extreme short bowel syndrome: Case report and review of the literature

Dennison

2016

Ultrasound

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Gastroschisis alone has excellent survival rates. Occasionally reported is closed gastroschisis, leading to vanishing small bowel and extreme short bowel syndrome. It is believed that the abdominal wall defect can contract or close in utero, which leads to strangulation of the eviscerated bowel and the rare ''vanishing gut syndrome.'' This has a very poor prognosis with mortality as high as 70%. An 18-year-old primigravid patient's 13 week scan diagnosed a large gastroschisis affecting the fetus. After counselling, she decided to continue with the pregnancy. Between 20 and 22 weeks, the gastroschisis disappeared, and the bowel within the abdomen became markedly dilated. Spontaneous labour occurred at 33 þ 3 weeks gestation. There was no abdominal wall defect seen at delivery. Imaging and an exploratory laparotomy demonstrated absence of most of the midgut. Because available options for treatment would be very aggressive and risky, palliative care was thought to be the most feasible and practical option. He died at home on day 29 after birth. Extreme short gut syndrome (less than 25 cm of remaining small bowel) is rare. There are 13 reported cases in the literature from year 2000 to 2013. Treatment is aggressive and involves a bowel lengthening procedure or small bowel transplant. All require total parenteral nutrition and liver failure, and liver transplant is a common complication. Of these cases, 12 were born alive and 7 had aggressive treatment. Only two cases were confirmed to still be alive in infancy. If gastroschisis is seen to be reducing and ''disappearing'' antenatally, parents should be made aware of this rare complication so that they might be prepared if a poor outcome is anticipated.

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Vanishing gut in infants with gastroschisis

Cited by 31 publications

References 0 publications

Gastroschisis and the Risk of Short Bowel Syndrome: Outcomes and Counselling

Gastroschisis and the Risk of Short Bowel Syndrome: Outcomes and Counselling

Gastroschisis Complicated by Midgut Atresia, Absorption of Bowel, and Closure of the Abdominal Wall Defect

Closed gastroschisis, vanishing midgut and extreme short bowel syndrome: Case report and review of the literature

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