Fetal repair of surgically-created gastroschisis in a lamb model may improve postnatal feeding tolerance and intestinal motility: A pilot study

Anderson, Jamie E.; Galganski, Laura A.; Pivetti, Christopher D.; Vanover, Melissa; Stephenson, Jacob T.; Brownlee, Rachel D; Ma, Betty; Farmer, Diana L.; Hirose, Shinjiro

doi:10.1016/j.jpedsurg.2018.11.021

Cited by 5 publications

(12 citation statements)

References 35 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…First simulated in fetal lambs by Langer et al 48 , the ischemic injury is believed to cause the intestinal volvulus, atresia, stenosis, necrosis and perforation observed in complex GS. This was recently confirmed by a study of Anderson et al 49 , in which a model of complex GS with bowel atresia or volvulus at birth (term, 145 days) was induced surgically. At mid-gestation (75 days' gestation), the authors made a 1.5-cm horizontal incision of the abdominal wall, eviscerated the small bowel, placed a silicon ring of 1.5 cm in diameter around the bowel and secured the ring to the abdominal wall.…”

Section: Established Natural Historymentioning

confidence: 62%

Complex gastroschisis: a new indication for fetal surgery?

Joyeux

Belfort

Coppi

et al. 2021

Ultrasound in Obstet & Gyne

View full text Add to dashboard Cite

Gastroschisis (GS) is a congenital abdominal wall defect, in which the bowel eviscerates from the abdominal cavity. It is a non‐lethal isolated anomaly and its pathogenesis is hypothesized to occur as a result of two hits: primary rupture of the ‘physiological’ umbilical hernia (congenital anomaly) followed by progressive damage of the eviscerated bowel (secondary injury). The second hit is thought to be caused by a combination of mesenteric ischemia from constriction in the abdominal wall defect and prolonged amniotic fluid exposure with resultant inflammatory damage, which eventually leads to bowel dysfunction and complications. GS can be classified as either simple or complex, with the latter being complicated by a combination of intestinal atresia, stenosis, perforation, volvulus and/or necrosis. Complex GS requires multiple neonatal surgeries and is associated with significantly greater postnatal morbidity and mortality than is simple GS. The intrauterine reduction of the eviscerated bowel before irreversible damage occurs and subsequent defect closure may diminish or potentially prevent the bowel damage and other fetal and neonatal complications associated with this condition. Serial prenatal amnioexchange has been studied in cases with GS as a potential intervention but never adopted because of its unproven benefit in terms of survival and bowel and lung function. We believe that recent advances in prenatal diagnosis and fetoscopic surgery justify reconsideration of the antenatal management of complex GS under the rubric of the criteria for fetal surgery established by the International Fetal Medicine and Surgery Society (IFMSS). Herein, we discuss how conditions for fetoscopic repair of complex GS might be favorable according to the IFMSS criteria, including an established natural history, an accurate prenatal diagnosis, absence of fully effective perinatal treatment due to prolonged need for neonatal intensive care, experimental evidence for fetoscopic repair and maternal and fetal safety of fetoscopy in expert fetal centers. Finally, we propose a research agenda that will help overcome barriers to progress and provide a pathway toward clinical implementation. © 2021 International Society of Ultrasound in Obstetrics and Gynecology.

show abstract

Section: Established Natural Historymentioning

confidence: 62%

Complex gastroschisis: a new indication for fetal surgery?

Joyeux

Belfort

Coppi

et al. 2021

Ultrasound in Obstet & Gyne

View full text Add to dashboard Cite

show abstract

“…From August 2010 to May 2018, all ewes and select lambs ( ovis aries) that were part of multiple research protocols at the UCD were tested for C. burnetii . 18–31 No additional animals were obtained specifically for this study. Sheep were of various breeds, including Dorper, Merino, and Polypay, and were all female due to use of parturient animal models for all of the protocols.…”

Section: Methodsmentioning

confidence: 99%

Minimizing the risk of occupational Q fever exposure: A protocol for ensuring Coxiella burnetii–negative pregnant ewes are used for medical research

et al. 2020

Self Cite

View full text Add to dashboard Cite

Q fever is a worldwide zoonosis caused by Coxiella burnetii that can lead to abortion, endocarditis, and death in humans. Researchers utilizing parturient domestic ruminants, including sheep, have an increased risk of occupational exposure. This study evaluated the effectiveness of our screening protocol in eliminating C. burnetii–positive sheep from our facility. From August 2010 to May 2018, all ewes ( N = 306) and select lambs ( N = 272; ovis aries) were screened twice for C. burnetii utilizing a serum Phase I and Phase II antibody immunofluorescence assay (IFA). The first screen was performed by the vendor prior to breeding, and the second screen was performed on arrival to the research facility. Ewes that were positive on arrival screening were quarantined and retested using repeat IFA serology, enzyme-linked immunosorbent assay, buffy coat polymerase chain reaction (PCR), and amniotic fluid PCR. The overall individual seroprevalence of C. burnetii in the flocks tested by the vendor was 14.2%. Ewes with negative Phase I and Phase II IFA results were selected for transport to the research facility. Upon arrival to the facility, two (0.7%) ewes had positive Phase I IFA results. Repeat testing demonstrated seropositivity in one of these two ewes, though amniotic fluid PCR was negative in both. The repeat seropositive ewe was euthanized prior to use in a research protocol. No Q fever was reported among husbandry, laboratory or veterinary staff during the study period. Serologic testing for C. burnetii with IFA prior to transport and following arrival to a research facility limits potential exposure to research staff.

show abstract

“…Open in utero repair of gastroschisis has been demonstrated in rabbit 66 and sheep animal models. 52,[67][68][69]72 The primary defect is surgically created midgestation (between 75 and 115 days gestation in the sheep model) via maternal laparotomy and hysterotomy. Subsequent fetal gastroschisis closure is then performed in the study group and compared to controls with no gastroschisis repair.…”

Section: Open In Utero Repairmentioning

confidence: 99%

“…Open in utero repair of gastroschisis has been demonstrated in rabbit 66 and sheep animal models 52,67‐69,72 . The primary defect is surgically created midgestation (between 75 and 115 days gestation in the sheep model) via maternal laparotomy and hysterotomy.…”

Section: Fetal Interventionsmentioning

confidence: 99%

“…Subsequent fetal gastroschisis closure is then performed in the study group and compared to controls with no gastroschisis repair. Stephenson et al 69 and Anderson et al 67 have both demonstrated the feasibility of fetal intestinal reduction and abdominal wall closure using the fetal lamb model, but abdominal compartment syndrome is a potential drawback of their technique. Bowel inflammation is often accompanied by significant edema and swelling, which when reduced into the fetal abdomen may result in marked elevations of intra‐abdominal compartment pressures.…”

Section: Fetal Interventionsmentioning

confidence: 99%

See 1 more Smart Citation

Is there a role for fetal interventions in gastroschisis management? – An updated comprehensive review

et al. 2020

View full text Add to dashboard Cite

We conducted a comprehensive evidence‐based review on the epidemiology and current standard of care of gastroschisis management as well as the pathophysiology, rationale and feasibility of fetal therapy as a viable alternative. Gastroschisis is a periumbilical abdominal wall defect characterized by abdominal viscera herniation in utero. It affects 4 in 10 000 live births, but the prevalence has steadily increased in recent years. Gastroschisis is typically diagnosed on routine second‐trimester ultrasound. The overall prognosis is favorable, but complex gastroschisis, which accounts for about 10% to 15% of cases, is associated with a higher mortality, significant disease burden and higher healthcare costs due to long‐ and short‐term complications. The current standard of care has yet to be established but generally involves continued fetal surveillance and multidisciplinary perinatal care. Postnatal surgical repair is achieved with primary closure, staged silo closure or sutureless repair. Experimental animal studies have demonstrated the feasibility of in utero closure, antiinflammatory therapy and prenatal regenerative therapy. However, reports of early preterm delivery and amnioinfusion trials have failed to show any benefit in humans. Further experimental studies and human trials are necessary to demonstrate the potential benefit of fetal therapy in gastroschisis.

show abstract

Fetal repair of surgically-created gastroschisis in a lamb model may improve postnatal feeding tolerance and intestinal motility: A pilot study

Cited by 5 publications

References 35 publications

Complex gastroschisis: a new indication for fetal surgery?

Complex gastroschisis: a new indication for fetal surgery?

Minimizing the risk of occupational Q fever exposure: A protocol for ensuring Coxiella burnetii–negative pregnant ewes are used for medical research

Is there a role for fetal interventions in gastroschisis management? – An updated comprehensive review

Contact Info

Product

Resources

About