Advertisement

Relationship between volume and outcome for gastroschisis: A systematic review

  • Johannes Morche
    Correspondence
    Corresponding Author.
    Affiliations
    Institute for Research in Operative Medicine, Faculty of Health, School of Medicine, Witten/Herdecke University, Ostmerheimer Str. 200, Building, 38, 51109, Cologne, Germany

    Medical Consultancy Department, Federal Joint Committee, Gutenbergstraße 13, 10587, Berlin, Germany
    Search for articles by this author
  • Tim Mathes
    Affiliations
    Institute for Research in Operative Medicine, Faculty of Health, School of Medicine, Witten/Herdecke University, Ostmerheimer Str. 200, Building 38, 51109, Cologne, Germany

    Institute for Medical Statistics, University Medical Center Goettingen, Humboldtallee 32, 37073, Göttingen, Germany
    Search for articles by this author
  • Anja Jacobs
    Affiliations
    Medical Consultancy Department, Federal Joint Committee, Gutenbergstraße 13, 10587, Berlin, Germany
    Search for articles by this author
  • Lucas Wessel
    Affiliations
    Department of Pediatric Surgery, Medical Faculty Mannheim (UMM), University of Heidelberg, Theodor-Kutzer-Ufer 1-3, 68167, Mannheim, Germany
    Search for articles by this author
  • Edmund A.M. Neugebauer
    Affiliations
    Center for Health Services Research Brandenburg, Brandenburg Medical School Theodor Fontane, Campus Rüdersdorf, Seebad 82/83, 15562, Rüdersdorf, Germany
    Search for articles by this author
  • Dawid Pieper
    Affiliations
    Institute for Research in Operative Medicine, Faculty of Health, School of Medicine, Witten/Herdecke University, Ostmerheimer Str. 200, Building 38, 51109, Cologne, Germany

    Center for Health Services Research Brandenburg, Brandenburg Medical School Theodor Fontane, Campus Rüdersdorf, Seebad 82/83, 15562, Rüdersdorf, Germany

    Faculty of Health Sciences Brandenburg, Brandenburg Medical School Theodor Fontane, Institute for Health Services and Health System Research, Campus Rüdersdorf, Seebad 82/83, 15562, Rüdersdorf, Germany
    Search for articles by this author

      Highlights

      • What is currently known about this topic?
        Previously, there was no systematic analysis of the evidence on the relationship between hospital as well as surgeon volume and outcomes for neonates with gastroschisis.
      • What new information is contained in this article?
        Findings from this systematic review suggest that higher hospital volume may reduce in-hospital mortality for gastroschisis. There is no evidence on the surgeon volume-outcome relationship.

      Abstract

      Background

      Newborns with gastroschisis need surgery to reduce intestines into the abdominal cavity and to close the abdominal wall. Due to an existing volume-outcome relationship for other high-risk, low-volume procedures, we aimed at examining the relationship between hospital or surgeon volume and outcomes for gastroschisis.

      Methods

      We conducted a systematic literature search in Medline, Embase, CENTRAL, CINAHL and Biosis Previews in June 2021 and searched for additional literature. We included (cluster-) randomized controlled trials (RCTs) and prospective or retrospective cohort studies analyzing the relationship between hospital or surgeon volume and mortality, morbidity or quality of life. We assessed risk of bias of included studies using ROBINS-I and performed a systematic synthesis without meta-analysis and used GRADE for assessing the certainty of the evidence.

      Results

      We included 12 cohort studies on hospital volume. Higher hospital volume may reduce in-hospital mortality of neonates with gastroschisis, while the evidence is very uncertain for other outcomes. Findings are based on a low certainty of the evidence for in-hospital mortality and a very low certainty of the evidence for all other analyzed outcomes, mainly due to risk of bias and imprecision. We did not identify any study on surgeon volume.

      Conclusion

      The evidence suggests that higher hospital volume reduces in-hospital mortality of newborns with gastroschisis. However, the magnitude of this effect seems to be heterogeneous and results should be interpreted with caution. There is no evidence on the relationship between surgeon volume and outcomes.

      Keywords

      List of abbreviations:

      GRADE (The Grading of Recommendations Assessment Development and Evaluation), LOS (Length of Stay), RoB (Risk of Bias), ROBINS-I (Risk of bias in non-randomized studies of intervention)
      To read this article in full you will need to make a payment

      Purchase one-time access:

      Academic & Personal: 24 hour online accessCorporate R&D Professionals: 24 hour online access
      One-time access price info
      • For academic or personal research use, select 'Academic and Personal'
      • For corporate R&D use, select 'Corporate R&D Professionals'

      Subscribe:

      Subscribe to Journal of Pediatric Surgery
      Already a print subscriber? Claim online access
      Already an online subscriber? Sign in
      Institutional Access: Sign in to ScienceDirect

      References

        • Anderson J.E.
        • Galganski L.A.
        • Cheng Y.
        • et al.
        Epidemiology of gastroschisis: a population-based study in California from 1995 to 2012.
        J Pediatr Surg. 2018; 53: 2399-2403https://doi.org/10.1016/j.jpedsurg.2018.08.035
        • Bhatt P.
        • Lekshminarayanan A.
        • Donda K.
        • et al.
        Trends in incidence and outcomes of gastroschisis in the United States: analysis of the national inpatient sample 2010-2014.
        Pediatr Surg Int. 2018; 34: 919-929https://doi.org/10.1007/s00383-018-4308-3
        • Friedman A.M.
        • Ananth C.V.
        • Siddiq Z.
        • et al.
        Gastroschisis: epidemiology and mode of delivery, 2005-2013.
        Am J Obstet Gynecol. 2016; 215 (348 e1-9)https://doi.org/10.1016/j.ajog.2016.03.039
        • Stallings E.B.
        • Isenburg J.L.
        • Short T.D.
        • et al.
        Population-based birth defects data in the United States, 2012-2016: a focus on abdominal wall defects.
        Birth Defects Res. 2019; 111: 1436-1447https://doi.org/10.1002/bdr2.1607
        • Gamba P.
        • Midrio P.
        Abdominal wall defects: prenatal diagnosis, newborn management, and long-term outcomes.
        Semin Pediatr Surg. 2014; 23: 283-290https://doi.org/10.1053/j.sempedsurg.2014.09.009
        • Skarsgard E.D.
        Management of gastroschisis.
        Curr Opin Pediatr. 2016; 28: 363-369https://doi.org/10.1097/MOP.0000000000000336
        • Bergholz R.
        • Boettcher M.
        • Reinshagen K.
        • et al.
        Complex gastroschisis is a different entity to simple gastroschisis affecting morbidity and mortality-a systematic review and meta-analysis.
        J Pediatr Surg. 2014; 49: 1527-1532https://doi.org/10.1016/j.jpedsurg.2014.08.001
        • Molik K.A.
        • Gingalewski C.A.
        • West K.W.
        • et al.
        Gastroschisis: a plea for risk categorization.
        J Pediatr Surg. 2001; 36: 51-55https://doi.org/10.1053/jpsu.2001.20004
        • Dommergues M.
        • Ansker Y.
        • Aubry M.C.
        • et al.
        Serial transabdominal amnioinfusion in the management of gastroschisis with severe oligohydramnios.
        J Pediatr Surg. 1996; 31: 1297-1299https://doi.org/10.1016/s0022-3468(96)90257-2
        • Luton D.
        • Mitanchez D.
        • Winer N.
        • et al.
        A randomised controlled trial of amnioexchange for fetal gastroschisis.
        BJOG. 2019; 126: 1233-1241https://doi.org/10.1111/1471-0528.15804
        • Sapin E.
        • Mahieu D.
        • Borgnon J.
        • et al.
        Transabdominal amnioinfusion to avoid fetal demise and intestinal damage in fetuses with gastroschisis and severe oligohydramnios.
        J Pediatr Surg. 2000; 35: 598-600https://doi.org/10.1053/jpsu.2000.0350598
        • Kirollos D.W.
        • ME Abdel-Latif
        Mode of delivery and outcomes of infants with gastroschisis: a meta-analysis of observational studies.
        Arch Dis Child Fetal Neonatal. 2018; 103: F355-FF63https://doi.org/10.1136/archdischild-2016-312394
        • Lopez A.
        • Benjamin R.H.
        • Raut J.R.
        • et al.
        Mode of delivery and mortality among neonates with gastroschisis: a population-based cohort in Texas.
        Paediatr Perinat Epidemiol. 2019; 33: 204-212https://doi.org/10.1111/ppe.12554
        • Grant N.H.
        • Dorling J.
        • Thornton J.G.
        Elective preterm birth for fetal gastroschisis.
        Cochrane Database Syst Rev. 2013; CD009394https://doi.org/10.1002/14651858.CD009394.pub2
        • Landisch R.M.
        • Yin Z.
        • Christensen M.
        • et al.
        Outcomes of gastroschisis early delivery: a systematic review and meta-analysis.
        J Pediatr Surg. 2017; 52: 1962-1971https://doi.org/10.1016/j.jpedsurg.2017.08.068
        • Carnaghan H.
        • Pereira S.
        • James C.P.
        • et al.
        Is early delivery beneficial in gastroschisis?.
        J Pediatr Surg. 2014; 49 (discussion 33): 928-933https://doi.org/10.1016/j.jpedsurg.2014.01.027
        • Haddock C.
        • Skarsgard E.D.
        Understanding gastroschisis and its clinical management: where are we?.
        Expert Rev Gastroenterol Hepatol. 2018; 12: 405-415https://doi.org/10.1080/17474124.2018.1438890
        • Prefumo F.
        • Izzi C.
        Fetal abdominal wall defects.
        Best Pract Res Clin Obstet Gynaecol. 2014; 28: 391-402https://doi.org/10.1016/j.bpobgyn.2013.10.003
        • Chesley P.M.
        • Ledbetter D.J.
        • Meehan J.J.
        • et al.
        Contemporary trends in the use of primary repair for gastroschisis in surgical infants.
        Am J Surg. 2015; 209 (discussion 5-6): 901-905https://doi.org/10.1016/j.amjsurg.2015.01.012
        • Archampong D.
        • Borowski D.
        • Wille-Jørgensen P.
        • et al.
        Workload and surgeon´s specialty for outcome after colorectal cancer surgery.
        Cochrane Database Syst Rev. 2012; https://doi.org/10.1002/14651858.CD005391.pub3
        • Gooiker G.A.
        • van Gijn W.
        • Post P.N.
        • et al.
        A systematic review and meta-analysis of the volume-outcome relationship in the surgical treatment of breast cancer. Are breast cancer patients better of with a high volume provider?.
        Eur J Surg Oncol. 2010; 36: S27-S35https://doi.org/10.1016/j.ejso.2010.06.024
        • Gooiker G.A.
        • Van Gijn W.
        • Wouters M.W.J.M.
        • et al.
        Systematic review and meta-analysis of the volume-outcome relationship in pancreatic surgery.
        Br J Surg. 2011; 98: 485-494
        • Wouters M.W.J.M.
        • Gooiker G.A.
        • Van Sandick J.W.
        • et al.
        The volume-outcome relation in the surgical treatment of esophageal cancer: a systematic review and meta-analysis.
        Cancer. 2012; 118: 1754-1763
        • Birkmeyer J.D.
        • Siewers A.E.
        • Finlayson E.V.
        • et al.
        Hospital volume and surgical mortality in the United States.
        N Engl J Med. 2002; 346: 1128-1137https://doi.org/10.1056/NEJMsa012337
        • Birkmeyer J.D.
        • Stukel T.A.
        • Siewers A.E.
        • et al.
        Surgeon volume and operative mortality in the United States.
        N Engl J Med. 2003; 349: 2117-2127https://doi.org/10.1056/NEJMsa035205
        • Morche J.
        • Mathes T.
        • Pieper D.
        Relationship between surgeon volume and outcomes: a systematic review of systematic reviews.
        Syst Rev. 2016; 5: 204https://doi.org/10.1186/s13643-016-0376-4
        • Pieper D.
        • Mathes T.
        • Neugebauer E.
        • et al.
        State of evidence on the relationship between high-volume hospitals and outcomes in surgery: a systematic review of systematic reviews.
        J Am Coll Surg. 2013; 216 (1015–25 e18)https://doi.org/10.1016/j.jamcollsurg.2012.12.049
        • Taylor J.S.
        • Shew S.B.
        Impact of societal factors and health care delivery systems on gastroschisis outcomes.
        Semin Pediatr Surg. 2018; 27: 316-320https://doi.org/10.1053/j.sempedsurg.2018.08.010
        • Baird R.
        • Eeson G.
        • Safavi A.
        • et al.
        Institutional practice and outcome variation in the management of congenital diaphragmatic hernia and gastroschisis in Canada: a report from the Canadian Pediatric Surgery Network.
        J Pediatr Surg. 2011; 46: 801-807https://doi.org/10.1016/j.jpedsurg.2011.02.008
        • Dubrovsky G.
        • Sacks G.D.
        • Friedlander S.
        • et al.
        Understanding the relationship between hospital volume and patient outcomes for infants with gastroschisis.
        J Pediatr Surg. 2017; 52: 1977-1980https://doi.org/10.1016/j.jpedsurg.2017.08.065
        • Sacks G.D.
        • Ulloa J.G.
        • Shew S.B.
        Is there a relationship between hospital volume and patient outcomes in gastroschisis repair?.
        J Pediatr Surg. 2016; 51: 1650-1654https://doi.org/10.1016/j.jpedsurg.2016.04.009
        • Youssef F.
        • Laberge J.M.
        • Puligandla P.
        • et al.
        Determinants of outcomes in patients with simple gastroschisis.
        J Pediatr Surg. 2017; 52: 710-714https://doi.org/10.1016/j.jpedsurg.2017.01.019
        • Page M.J.
        • McKenzie J.E.
        • Bossuyt P.M.
        • et al.
        The PRISMA 2020 statement: an updated guideline for reporting systematic reviews.
        Bmj. 2021; 372: n71https://doi.org/10.1136/bmj.n71
        • Morche J.
        • Mathes T.
        • Jacobs A.
        • et al.
        Relationship between volume and outcome for gastroschisis: a systematic review protocol.
        Syst Rev. 2020; 9: 203https://doi.org/10.1186/s13643-020-01462-y
        • Morche J.
        • Mathes T.
        • Jacobs A.
        • et al.
        Relationship between volume and outcome for congenital diaphragmatic hernia: a systematic review protocol.
        Syst Rev. 2018; 7: 185https://doi.org/10.1186/s13643-018-0872-9
        • Morche J.
        • Mathes T.
        • Jacobs A.
        • et al.
        Relationship between volume and outcome for surgery on congenital diaphragmatic hernia: a systematic review.
        J Pediatr Surg. 2020; 55: 2255-2265https://doi.org/10.1016/j.jpedsurg.2020.03.025
        • Allin B.S.R.
        • Hall N.J.
        • Ross A.R.
        • et al.
        Development of a gastroschisis core outcome set.
        Arch Dis Child Fetal Neonatal. 2019; 104: F76-F82https://doi.org/10.1136/archdischild-2017-314560
        • Allin B.S.
        • Irvine A.
        • Patni N.
        • et al.
        Variability of outcome reporting in Hirschsprung's Disease and gastroschisis: a systematic review.
        Sci Rep. 2016; 6: 38969https://doi.org/10.1038/srep38969
        • Gorra A.S.
        • Needelman H.
        • Azarow K.S.
        • et al.
        Long-term neurodevelopmental outcomes in children born with gastroschisis: the tiebreaker.
        J Pediatr Surg. 2012; 47: 125-129https://doi.org/10.1016/j.jpedsurg.2011.10.028
        • Lao O.B.
        • Larison C.
        • Garrison M.M.
        • et al.
        Outcomes in neonates with gastroschisis in U.S. children's hospitals.
        Am J Perinatol. 2010; 27: 97-101https://doi.org/10.1055/s-0029-1241729
        • Suominen J.
        • Rintala R.
        Medium and long-term outcomes of gastroschisis.
        Semin Pediatr Surg. 2018; 27: 327-329https://doi.org/10.1053/j.sempedsurg.2018.08.008
        • Sterne J.A.
        • Hernan M.A.
        • Reeves B.C.
        • et al.
        ROBINS-I: a tool for assessing risk of bias in non-randomised studies of interventions.
        Bmj. 2016; 355: i4919https://doi.org/10.1136/bmj.i4919
        • Campbell M.
        • McKenzie J.E.
        • Sowden A.
        • et al.
        Synthesis without meta-analysis (SWiM) in systematic reviews: reporting guideline.
        Bmj. 2020; 368: l6890https://doi.org/10.1136/bmj.l6890
        • Guyatt G.H.
        • Oxman A.D.
        • Montori V.
        • et al.
        GRADE guidelines: 5. Rating the quality of evidence–publication bias.
        J Clin Epidemiol. 2011; 64: 1277-1282https://doi.org/10.1016/j.jclinepi.2011.01.011
        • Balshem H.
        • Helfand M.
        • Schunemann H.J.
        • et al.
        GRADE guidelines: 3. Rating the quality of evidence.
        J Clin Epidemiol. 2011; 64: 401-406https://doi.org/10.1016/j.jclinepi.2010.07.015
        • Schunemann H.J.
        • Cuello C.
        • Akl E.A.
        • et al.
        GRADE guidelines: 18. How ROBINS-I and other tools to assess risk of bias in nonrandomized studies should be used to rate the certainty of a body of evidence.
        J Clin Epidemiol. 2019; 111: 105-114https://doi.org/10.1016/j.jclinepi.2018.01.012
        • Apfeld J.C.
        • Kastenberg Z.J.
        • Sylvester K.G.
        • et al.
        The Effect of Level of Care on Gastroschisis Outcomes.
        J Pediatr. 2017; 190 (79–84.e1)https://doi.org/10.1016/j.jpeds.2017.07.008
        • Gonzalez D.O.
        • Cooper J.N.
        • St Peter S.D.
        • et al.
        Variability in outcomes after gastroschisis closure across U.S. children's hospitals.
        J Pediatr Surg. 2018; 53: 513-520https://doi.org/10.1016/j.jpedsurg.2017.04.012
        • Hong C.R.
        • Fullerton B.S.
        • Han S.M.
        • et al.
        Impact of disease-specific volume and hospital transfer on outcomes in gastroschisis.
        J Pediatr Surg. 2019; 54: 65-69https://doi.org/10.1016/j.jpedsurg.2018.10.034
        • Somme S.
        • Shahi N.
        • McLeod L.
        • et al.
        Neonatal surgery in low- vs. high-volume institutions: a KID inpatient database outcomes and cost study after repair of congenital diaphragmatic hernia, esophageal atresia, and gastroschisis.
        Pediatr Surg Int. 2019; 35: 1293-1300https://doi.org/10.1007/s00383-019-04525-x
        • Stanger J.
        • Mohajerani N.
        • Skarsgard E.D.
        Practice variation in gastroschisis: factors influencing closure technique.
        J Pediatr Surg. 2014; 49: 720-723https://doi.org/10.1016/j.jpedsurg.2014.02.066
        • Towner D.
        • Greve L.
        Outcomes of gastroschisis based on volume of cases at the treating center.
        Am J Obstet Gynecol. 2002; 187: S189
        • Youssef F.
        • Cheong L.H.
        • Emil S.
        Gastroschisis outcomes in North America: a comparison of Canada and the United States.
        J Pediatr Surg. 2016; 51: 891-895https://doi.org/10.1016/j.jpedsurg.2016.02.046
        • Glinianaia S.V.
        • Morris J.K.
        • Best K.E.
        • et al.
        Long-term survival of children born with congenital anomalies: a systematic review and meta-analysis of population-based studies.
        PLoS Med. 2020; 17e1003356https://doi.org/10.1371/journal.pmed.1003356
        • Giudici L.
        • Bokser V.S.
        • Maricic M.A.
        • et al.
        Babies born with gastroschisis and followed up to the age of six years faced long-term morbidity and impairments.
        Acta Paediatr. 2016; 105: e275-e280https://doi.org/10.1111/apa.13374
        • Levaillant M.
        • Marcilly R.
        • Levaillant L.
        • et al.
        Assessing the hospital volume-outcome relationship in surgery: a scoping review.
        BMC Med Res Methodol. 2021; 21: 204https://doi.org/10.1186/s12874-021-01396-6
        • Payet C.
        • Polazzi S.
        • Lifante J.C.
        • et al.
        Influence of trends in hospital volume over time on patient outcomes for high-risk surgery.
        BMC Health Serv Res. 2020; 20: 274https://doi.org/10.1186/s12913-020-05126-4
        • Morche J.
        • Renner D.
        • Pietsch B.
        • et al.
        International comparison of minimum volume standards for hospitals.
        Health Policy. 2018; 122: 1165-1176https://doi.org/10.1016/j.healthpol.2018.08.016
        • Nasr A.
        • Langer J.C.
        Canadian Paediatric Surgery N. Influence of location of delivery on outcome in neonates with gastroschisis.
        J Pediatr Surg. 2012; 47: 2022-2025https://doi.org/10.1016/j.jpedsurg.2012.07.037
        • Savoie K.B.
        • Huang E.Y.
        • Aziz S.K.
        • et al.
        Improving gastroschisis outcomes: does birth place matter?.
        J Pediatr Surg. 2014; 49: 1771-1775https://doi.org/10.1016/j.jpedsurg.2014.09.019
        • Buhn S.
        • Holstiege J.
        • Pieper D.
        Are patients willing to accept longer travel times to decrease their risk associated with surgical procedures? A systematic review.
        BMC Public Health. 2020; 20: 253https://doi.org/10.1186/s12889-020-8333-5
        • Finlayson S.R.
        The volume-outcome debate revisited.
        Am Surg. 2006; 72 (discussion 61-9, 133-48): 1038-1042
      1. Hewitt M for the Committee on Quality of Health Care in America and the National Cancer Policy Board. Interpreting the volume-outcome relationship in the context of cancer care Washington DC: National Academic Press; 2001 [07.08.2019]. Available from: http://www.ncbi.nlm.nih.gov/books/NBK223578/pdf/Bookshelf_NBK223578.pdf.

        • Aldrink J.H.
        • Caniano D.A.
        • Nwomeh B.C.
        Variability in gastroschisis management: a survey of North American pediatric surgery training programs.
        J Surg Res. 2012; 176: 159-163https://doi.org/10.1016/j.jss.2011.05.012
        • DeUgarte D.A.
        • Calkins K.L.
        • Guner Y.
        • et al.
        Adherence to and outcomes of a University-Consortium gastroschisis pathway.
        J Pediatr Surg. 2020; 55: 45-48https://doi.org/10.1016/j.jpedsurg.2019.09.048
        • Haddock C.
        • Al Maawali A.G.
        • Ting J.
        • et al.
        Impact of multidisciplinary standardization of care for gastroschisis: treatment, outcomes, and cost.
        J Pediatr Surg. 2018; 53: 892-897https://doi.org/10.1016/j.jpedsurg.2018.02.013
        • Lusk L.A.
        • Brown E.G.
        • Overcash R.T.
        • et al.
        Multi-institutional practice patterns and outcomes in uncomplicated gastroschisis: a report from the University of California Fetal Consortium (UCfC).
        J Pediatr Surg. 2014; 49: 1782-1786https://doi.org/10.1016/j.jpedsurg.2014.09.018