Gastroschisis: one year outcomes from national cohort study

Timothy J Bradnock, Sean Marven, Anthony Owen, Paul Johnson, Jennifer J Kurinczuk, Patsy Spark, Elizabeth S Draper, Marian Knight, BAPS-CASS, Adil Aslam, Christopher Parsons, Marcin Kamierski, Anthony Lander, Andrew Robb, Humza Malik, Eleri Cusick, Evelyn Ong, Nicholas Madden, Clare Skerritt, Anindya Niyogi, Gordon MacKinlay, Boma Adikibi, Atul Sabharwal, Joe Curry, Sanja Besarovic, Paul Johnson, Javaid Sadiq, Mark Davenport, Ian Sugarman, Anthony Owen, Shawqui Nour, Thomas Tsang, Brian Davies, Richard Stewart, Prem Puri, Farhan Tareen, Robin Abel, Christopher Driver, Anies Mahomed, Rebecca Lisle, William A McCallion, Suzanne Lawther, Paul Losty, David Wilkinson, Catherine Cord-Udy, Simon Phelps, Antonino Morabito, Anju Goyal, Gareth Hosie, Sean Marven, Karen Lloyd, David Burge, Melanie Drewett, Bruce Okoye, Catherine Richards, Simon Huddart, Daniela Vieten, Ram Shrestha, Timothy J Bradnock, Sean Marven, Anthony Owen, Paul Johnson, Jennifer J Kurinczuk, Patsy Spark, Elizabeth S Draper, Marian Knight, BAPS-CASS, Adil Aslam, Christopher Parsons, Marcin Kamierski, Anthony Lander, Andrew Robb, Humza Malik, Eleri Cusick, Evelyn Ong, Nicholas Madden, Clare Skerritt, Anindya Niyogi, Gordon MacKinlay, Boma Adikibi, Atul Sabharwal, Joe Curry, Sanja Besarovic, Paul Johnson, Javaid Sadiq, Mark Davenport, Ian Sugarman, Anthony Owen, Shawqui Nour, Thomas Tsang, Brian Davies, Richard Stewart, Prem Puri, Farhan Tareen, Robin Abel, Christopher Driver, Anies Mahomed, Rebecca Lisle, William A McCallion, Suzanne Lawther, Paul Losty, David Wilkinson, Catherine Cord-Udy, Simon Phelps, Antonino Morabito, Anju Goyal, Gareth Hosie, Sean Marven, Karen Lloyd, David Burge, Melanie Drewett, Bruce Okoye, Catherine Richards, Simon Huddart, Daniela Vieten, Ram Shrestha

Abstract

Objective: To describe one year outcomes for a national cohort of infants with gastroschisis.

Design: Population based cohort study of all liveborn infants with gastroschisis born in the United Kingdom and Ireland from October 2006 to March 2008.

Setting: All 28 paediatric surgical centres in the UK and Ireland.

Participants: 301 infants (77%) from an original cohort of 393.

Main outcome measures: Duration of parenteral nutrition and stay in hospital; time to establish full enteral feeding; rates of intestinal failure, liver disease associated with intestinal failure, unplanned reoperation; case fatality.

Results: Compared with infants with simple gastroschisis (intact, uncompromised, continuous bowel), those with complex gastroschisis (bowel perforation, necrosis, or atresia) took longer to reach full enteral feeding (median difference 21 days, 95% confidence interval 9 to 39 days); required a longer duration of parenteral nutrition (median difference 25 days, 9 to 46 days) and a longer stay in hospital (median difference 57 days, 29 to 95 days); were more likely to develop intestinal failure (81% (25 infants) v 41% (102); relative risk 1.96, 1.56 to 2.46) and liver disease associated with intestinal failure (23% (7) v 4% (11); 5.13, 2.15 to 12.3); and were more likely to require unplanned reoperation (42% (13) v 10% (24); 4.39, 2.50 to 7.70). Compared with infants managed with primary fascial closure, those managed with preformed silos took longer to reach full enteral feeding (median difference 5 days, 1 to 9) and had an increased risk of intestinal failure (52% (50) v 32% (38); 1.61, 1.17 to 2.24). Event rates for the other outcomes were low, and there were no other significant differences between these management groups. Twelve infants died (4%).

Conclusions: This nationally representative study provides a benchmark against which individual centres can measure outcome and performance. Stratifying neonates with gastroschisis into simple and complex groups reliably predicts outcome at one year. There is sufficient clinical equipoise concerning the initial management strategy to embark on a multicentre randomised controlled trial comparing primary fascial closure with preformed silos in infants suitable at presentation for either treatment to determine the optimal initial management strategy and define algorithms of care.

Conflict of interest statement

Competing interests: All authors have completed the ICMJE uniform disclosure form at www.icmje.org/coi_disclosure.pdf (available on request from the corresponding author) and declare: no support from any organisation for the submitted work; no financial relationships with any organisations that might have an interest in the submitted work in the previous three years; no other relationships or activities that could appear to have influenced the submitted work.

Figures

https://www.ncbi.nlm.nih.gov/pmc/articles/instance/4788183/bin/brat879460.f1_default.jpg
Case reporting and completeness of 1 year follow-up in infants with gastroschisis

References

    1. Keys C, Drewett M, Burge DM. Gastroschisis: the cost of an epidemic. J Pediatr Surg 2008;43:654-7.
    1. Kilby MD. The incidence of gastroschisis. BMJ 2006;332:250-1.
    1. Alvarez SM, Burd RS. Increasing prevalence of gastroschisis repairs in the United States: 1996-2003. J Pediatr Surg 2007;42:943-6.
    1. European Surveillance of Congenital Anomalies (EUROCAT). EUROCAT report 8: Surveillance of congenital anomalies 1980-1999. EUROCAT Central Registry, University of Ulster, 2001.
    1. Laughon M, Meyer R, Bose C, Wall A, Otero E, Heerens A, et al. Rising birth prevalence of gastroschisis. J Perinatol 2003;23:291-3.
    1. Centers for Disease Control and Prevention. Hospital stays, hospital charges, and in-hospital deaths among infants with selected birth defects—United States, 2003. MMWR Morb Mortal Wkly Rep 2007;56:25-9.
    1. Knutrud O, Bjordal RI, Ro J, Bo G. Gastroschisis and omphalocele. Prog Pediatr Surg 1979;13:51-61.
    1. Chang DC, Salazar-Osuna JH, Choo SS, Arnold MA, Colombani PM, Abdullah F. Benchmarking the quality of care of infants with low-risk gastroschisis using a novel risk stratification index. Surgery 2010;147:766-71.
    1. Nembhard WN, Waller DK, Sever LE, Canfield MA. Patterns of first-year survival among infants with selected congenital anomalies in Texas, 1995-1997. Teratology 2001;64:267-75.
    1. Weinsheimer RL, Yanchar NL, Bouchard SB, Kim PK, Laberge JM, Skarsgard ED, et al. Gastroschisis closure—does method really matter? J Pediatr Surg 2008;43:874-8.
    1. Arnold MA, Chang DC, Nabaweesi R, Colombani PM, Bathurst MA, Mon KS, et al. Risk stratification of 4344 patients with gastroschisis into simple and complex categories. J Pediatr Surg 2007;42:1520-5.
    1. Arnold MA, Chang DC, Nabaweesi R, Colombani PM, Fischer AC, Lau HT, et al. Development and validation of a risk stratification index to predict death in gastroschisis. J Pediatr Surg 2007;42:950-6.
    1. Caniano DA, Brokaw B, Ginn-Pease ME. An individualized approach to the management of gastroschisis. J Pediatr Surg 1990;25:297-300.
    1. Molik KA, Gingalewski CA, West KW, Rescorla FJ, Scherer LR, Engum SA, et al. Gastroschisis: a plea for risk categorization. J Pediatr Surg 2001;36:51-5.
    1. Allotey J, Davenport M, Njere I, Charlesworth P, Greenough A, Ade-Ajayi N, et al. Benefit of preformed silos in the management of gastroschisis. Pediatr Surg Int 2007;23:1065-9.
    1. Bianchi A, Dickson AP, Alizai NK. Elective delayed midgut reduction-no anesthesia for gastroschisis: selection and conversion criteria. J Pediatr Surg 2002;37:1334-6.
    1. Allen RG, Wrenn EL Jr. Silon as a sac in the treatment of omphalocele and gastroschisis. J Pediatr Surg 1969;4:3-8.
    1. Fonkalsrud EW, Smith MD, Shaw KS, Borick JM, Shaw A. Selective management of gastroschisis according to the degree of visceroabdominal disproportion. Ann Surg 1993;218:742-7.
    1. Fischer JD, Chun K, Moores DC, Andrews HG. Gastroschisis: a simple technique for staged silo closure. J Pediatr Surg 1995;30:1169-71.
    1. Owen A, Marven S, Jackson L, Antao B, Roberts J, Walker J, et al. Experience of bedside preformed silo staged reduction and closure for gastroschisis. J Pediatr Surg 2006;41:1830-5.
    1. Owen A, Marven S, Johnson P, Kurinczuk J, Spark P, Draper ES, et al. Gastroschisis: a national cohort study to describe contemporary surgical strategies and outcomes. J Pediatr Surg 2010;45:1808-16.
    1. Minkes RK, Langer JC, Mazziotti MV, Skinner MA, Foglia RP. Routine insertion of a silastic spring-loaded silo for infants with gastroschisis. J Pediatr Surg 2000;35:843-6.
    1. Schlatter M, Norris K, Uitvlugt N, DeCou J, Connors R. Improved outcomes in the treatment of gastroschisis using a preformed silo and delayed repair approach. J Pediatr Surg 2003;38:459-64.
    1. Banyard D, Ramones T, Phillips SE, Leys CM, Rauth T, Yang EY. Method to our madness: an 18-year retrospective analysis on gastroschisis closure. J Pediatr Surg 2010;45:579-84.
    1. Eggink BH, Richardson CJ, Malloy MH, Angel CA. Outcome of gastroschisis: a 20-year case review of infants with gastroschisis born in Galveston, Texas. J Pediatr Surg 2006;41:1103-8.
    1. Jager LC, Heij HA. Factors determining outcome in gastroschisis: clinical experience over 18 years. Pediatr Surg Int 2007;23:731-6.
    1. Skarsgard ED, Claydon J, Bouchard S, Kim PC, Lee SK, Laberge JM, et al. Canadian Pediatric Surgical Network: a population-based pediatric surgery network and database for analyzing surgical birth defects. The first 100 cases of gastroschisis. J Pediatr Surg 2008;43:30-4.
    1. BINOCAR. British Isles network of congenital anomaly registers. .
    1. Knight M, Kurinczuk JJ, Tuffnell D, Brocklehurst P. The UK Obstetric Surveillance System for rare disorders of pregnancy. BJOG 2005;112:263-5.
    1. Intestinal Failure Working Group British Society of Paediatric Gastroenterology Hepatology and Nutrition and the British Association of Paediatric Surgeons. Intestinal failure: recommendations for tertiary management of infants and children 2007. .
    1. Lao OB, Larison C, Garrison MM, Waldhausen JH, Goldin AB. Outcomes in neonates with gastroschisis in US children’s hospitals. Am J Perinatol 2010;27:97-101.
    1. Abdullah F, Arnold MA, Nabaweesi R, Fischer AC, Colombani PM, Anderson KD, et al. Gastroschisis in the United States 1988-2003: analysis and risk categorization of 4344 patients. J Perinatol 2007;27:50-5.
    1. Mills JA, Lin Y, Macnab YC, Skarsgard ED. Perinatal predictors of outcome in gastroschisis. J Perinatol 2010;30:809-13.
    1. Islam S. Clinical care outcomes in abdominal wall defects. Curr Opin Pediatr 2008;20:305-10.
    1. Pastor AC, Phillips JD, Fenton SJ, Meyers RL, Lamm AW, Raval MV, et al. Routine use of a SILASTIC spring-loaded silo for infants with gastroschisis: a multicenter randomized controlled trial. J Pediatr Surg 2008;43:1807-12.

Source: PubMed

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