Anti-reflux surgery in children with congenital diaphragmatic hernia: A prospective cohort study on a controversial practice


      • What is currently known about this topic? Gastro-esophageal reflux disease affects 50% of children born with congenital diaphragmatic hernia (CDH). No guidelines exist on the use of anti-reflux surgery in these children, and performing preventive fundoplication when performing a diaphragmatic patch repair remains controversial.
      • What new information is contained in this article? Children with a CDH patch repair have a higher risk of undergoing curative fundoplication. However, performing a preventive fundoplication during the initial CDH repair does not decrease the need for curative fundoplication but increases the rates of failure to thrive, tube feed supplementation, and oral aversion.



      Gastro-esophageal reflux disease (GERD) is the most frequent long-term morbidity of congenital diaphragmatic hernia (CDH) survivors. Performing a preventive fundoplication during CDH repair remains controversial. This study aimed to: (1) Analyze the variability in practices regarding preventive fundoplication; (2) Identify predictive factors for fundoplication.
      (3) Evaluate the impact of preventive fundoplication on gastro-intestinal outcomes in children with a CDH patch repair;


      This prospective multi-institutional cohort study (French CDH Registry) included CDH neonates born in France between January 1st, 2010-December 31st, 2018. Patch CDH was defined as need for synthetic patch or muscle flap repair. Main outcome measures included need for curative fundoplication, tube feed supplementation, failure to thrive, and oral aversion.


      Of 762 CDH neonates included, 81 underwent fundoplication (10.6%), either preventive or curative. Median follow-up was 3.0 years (IQR: 1.0–5.0).
      (1) Preventive fundoplication is considered in only 31% of centers. The rates of both curative fundoplication (9% vs 3%, p = 0.01) and overall fundoplication (20% vs 3%, p < 0.0001) are higher in centers that perform preventive fundoplication compared to those that do not.
      (2) Predictive factors for preventive fundoplication were: prenatal diagnosis (p = 0.006), intra-thoracic liver (p = 0.005), fetal tracheal occlusion (p = 0.002), CDH-grade C-D (p < 0.0001), patch repair (p < 0.0001). After CDH repair, 8% (n = 51) required curative fundoplication (median age: 101 days), for which a patch repair was the only independent predictive factors identified upon multivariate analysis.
      (3) In neonates with patch CDH, preventive fundoplication did not decrease the need for curative fundoplication (15% vs 11%, p = 0.53), and was associated with higher rates of failure to thrive (discharge: 81% vs 51%, p = 0.03; 6-months: 81% vs 45%, p = 0.008), tube feeds (6-months: 50% vs 21%, p = 0.02; 2-years: 65% vs 26%, p = 0.004), and oral aversion (6-months: 67% vs 37%, p = 0.02; 1-year: 71% vs 40%, p = 0.03).


      Children undergoing a CDH patch repair are at high risk of requiring a curative fundoplication. However, preventive fundoplication during a patch repair does not decrease the need for curative fundoplication and is associated with worse gastro-intestinal outcomes in children.

      Level of evidence

      II – Prospective Study.


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


        • McGivern M.R.
        • Best K.E.
        • Rankin J.
        • et al.
        Epidemiology of congenital diaphragmatic hernia in Europe: a register-based study.
        Arch Dis Child Fetal Neonatal Ed. 2015; 100: F137-F144
        • Barrière F.
        • Michel F.
        • Loundou A.D.
        • et al.
        One-year outcome for congenital diaphragmatic hernia: results from the French national register.
        J Pediatr. 2018; 193: 204-210
        • Putnam L.R.
        • Harting M.T.
        • Tsao K.
        • et al.
        Congenital diaphragmatic hernia defect size and infant morbidity at discharge.
        Pediatrics. 2016; 138
        • Peetsold M.G.
        • Heij H.A.
        • Kneepkens C.M.F.
        • et al.
        The long-term follow-up of patients with a congenital diaphragmatic hernia: a broad spectrum of morbidity.
        Pediatr Surg Int. 2009; 25: 1-17
        • Jaillard S.M.
        • Pierrat V.
        • Dubois A.
        • et al.
        Outcome at 2 years of infants with congenital diaphragmatic hernia: a population-based study.
        Ann Thorac Surg. 2003; 75: 250-256
        • Rosen R.
        • Vandenplas Y.
        • Singendonk M.
        • et al.
        Pediatric gastroesophageal reflux clinical practice guidelines: joint recommendations of the North American society for pediatric gastroenterology, hepatology, and nutrition and the european society for pediatric gastroenterology, hepatology, and nutritio.
        J Pediatr Gastroenterol Nutr. 2018; 66: 516-554
        • Power B.
        • Shibuya S.
        • Lane B.
        • et al.
        Long-term feeding issue and its impact on the daily life of congenital diaphragmatic hernia survivors: results of the first patient-led survey.
        Pediatr Surg Int. 2020; 36: 63-68
        • Diamond I.R.
        • Mah K.
        • Kim P.C.W.
        • et al.
        Predicting the need for fundoplication at the time of congenital diaphragmatic hernia repair.
        J Pediatr Surg. 2007; 42: 1066-1070
        • Chamond C.
        • Morineau M.
        • Gouizi G.
        • et al.
        Preventive antireflux surgery in patients with congenital diaphragmatic hernia.
        World J Surg. 2008; 32: 2454-2458
        • Yokota K.
        • Uchida H.
        • Kaneko K.
        • et al.
        Surgical complications, especially gastroesophageal reflux disease, intestinal adhesion obstruction, and diaphragmatic hernia recurrence, are major sequelae in survivors of congenital diaphragmatic hernia.
        Pediatr Surg Int. 2014; 30: 895-899
        • Snoek K.G.
        • Reiss I.K.M.M.
        • Greenough A.
        • et al.
        Standardized postnatal management of infants with congenital diaphragmatic hernia in Europe: the CDH EURO consortium consensus - 2015 update.
        Neonatology. 2016; 110: 66-74
        • Puligandla P.S.
        • Grabowski J.
        • Austin M.
        • et al.
        Management of congenital diaphragmatic hernia: a systematic review from the APSA outcomes and evidence based practice committee.
        J Pediatr Surg. 2015; 50: 1958-1970
        • Storme L.
        • Boubnova J.
        • Mur S.
        • et al.
        Review shows that implementing a nationwide protocol for congenital diaphragmatic hernia was a key factor in reducing mortality and morbidity.
        Acta Paediatr Int J Paediatr. 2018; 107: 1131-1139
      1. Montalva L., Carricaburu E., Bonnard A. (2020) Diagnosis and management of gastroesophageal reflux: a national survey across 29 centres. Manuscr Prep

        • Reiss I.
        • Schaible T.
        • Van Den Hout L.
        • et al.
        Standardized postnatal management of infants with congenital diaphragmatic hernia in Europe: the CDH EURO consortium consensus.
        Neonatology. 2010; 98: 354-364
        • Jancelewicz T.
        • Brindle M.E.
        • Guner Y.S.
        • et al.
        Toward standardized management of congenital diaphragmatic hernia: an analysis of practice guidelines.
        J Surg Res. 2019; 243: 229-235
      2. World Health Organization.

      3. GraphPad Prism version 8.4.0 for MacOs, GraphPad Software, San Diego, California USA,

      4. Dariel A., Rozé J.C., Piloquet H., Podevin G. (2011) Impact of prophylactic fundoplication on survival without growth disorder in left congenital diaphragmatic hernias requiring a patch repair

        • Guner Y.S.
        • Elliott S.
        • Marr C.C.
        • Greenholz S.K.
        Anterior fundoplication at the time of congenital diaphragmatic hernia repair.
        Pediatr Surg Int. 2009; 25: 715-718
        • Marseglia L.
        • Manti S.
        • D'Angelo G.
        • et al.
        Gastroesophageal reflux and congenital gastrointestinal malformations.
        World J Gastroenterol. 2015; 21: 8508-8515
        • Montalva L.
        • Mariani A.
        • Schmitt F.
        • et al.
        Concomitant gastrostomy tube insertion during laparoscopic Nissen fundoplication for gastro-esophageal reflux disease: analysis of risk factors for fundoplication failure.
        Surg Endosc. 2020;
        • Lopez-Fernandez S.
        • Hernandez F.
        • Hernandez-Martin S.
        • et al.
        Failed nissen fundoplication in children: causes and management.
        Eur J Pediatr Surg. 2014; 24: 79-82
        • Ngerncham M.
        • Barnhart D.C.
        • Haricharan R.N.
        • et al.
        Risk factors for recurrent gastroesophageal reflux disease after fundoplication in pediatric patients: a case-control study.
        J Pediatr Surg. 2007; 42: 1478-1485
        • Rayyan M.
        • Omari T.
        • Debeer A.
        • et al.
        Characterization of esophageal motility in infants with congenital diaphragmatic hernia using high-resolution manometry.
        J Pediatr Gastroenterol Nutr. 2019; 69: 32-38
        • Di Pace M.
        • Caruso A.
        • Catalano P.
        • et al.
        Evaluation of esophageal motility using multichannel intraluminal impedance in healthy children and children with gastroesophageal reflux.
        J Pediatr Gastroenterol Nutr. 2011; 52: 26-30
        • Maier S.
        • Zahn K.
        • Wessel L.M.
        • et al.
        Preventive antireflux surgery in neonates with congenital diaphragmatic hernia: a single-blinded prospective study.
        J Pediatr Surg. 2011; 46: 1510-1515
        • Su W.
        • Berry M.
        • Puligandla P.S.
        • et al.
        Predictors of gastroesophageal reflux in neonates with congenital diaphragmatic hernia.
        J Pediatr Surg. 2007; 42: 1639-1643
        • Janssen S.
        • Heiwegen K.
        • van Rooij I.A.
        • et al.
        Factors related to long-term surgical morbidity in congenital diaphragmatic hernia survivors.
        J Pediatr Surg. 2018; 53: 508-512
        • Muratore C.S.
        • Utter S.
        • Jaksic T.
        • et al.
        Nutritional morbidity in survivors of congenital diaphragmatic hernia.
        J Pediatr Surg. 2001; 36: 1171-1176
        • Terui K.
        • Taguchi T.
        • Goishi K.
        • et al.
        Prognostic factors of gastroesophageal reflux disease in congenital diaphragmatic hernia: a multicenter study.
        Pediatr Surg Int. 2014; 30: 1129-1134
        • Vanamo K.
        • Rintala R.J.
        • Lindahl H.
        • Louhimo I.
        Long-term gastrointestinal morbidity in patients with congenital diaphragmatic defects.
        J Pediatr Surg. 1996; 31: 551-554
        • Zani A.
        • Zani-Ruttenstock E.
        • Pierro A.
        Advances in the surgical approach to congenital diaphragmatic hernia.
        Semin Fetal Neonatal Med. 2014; 19: 364-369
        • IJsselstijn H.
        • Breatnach C.
        • Hoskote A.
        • et al.
        Defining outcomes following congenital diaphragmatic hernia using standardised clinical assessment and management plan (SCAMP) methodology within the CDH EURO consortium.
        Pediatr Res. 2018; 84: 181-189
        • Gischler S.J.
        • van der Cammen-van Zijp M.H.M.
        • Mazer P.
        • et al.
        A prospective comparative evaluation of persistent respiratory morbidity in esophageal atresia and congenital diaphragmatic hernia survivors.
        J Pediatr Surg. 2009; 44: 1683-1690
        • Schneider A.
        • Blanc S.
        • Bonnard A.
        • et al.
        Results from the French national esophageal atresia register: one-year outcome.
        Orphanet J Rare Dis. 2014; 9: 206
        • Krishnan U.
        • Mousa H.
        • Dall'Oglio L.
        • et al.
        ESPGHAN-NASPGHAN guidelines for the evaluation and treatment of gastrointestinal and nutritional complications in children with esophageal atresia-tracheoesophageal fistula.
        J Pediatr Gastroenterol Nutr. 2016; 63: 550-570
        • Montalva L.
        • Raffler G.
        • Riccio A.
        • et al.
        Neurodevelopmental impairment in children with congenital diaphragmatic hernia: not an uncommon complication for survivors.
        J Pediatr Surg. 2020; 55: 625-634
        • John C.C.
        • Black M.M.
        • Nelson C.A.
        Neurodevelopment: the impact of nutrition and inflammation during early to middle childhood in low-resource settings.
        Pediatrics. 2017; 139: S59-S71
        • Morandi A.
        • Macchini F.
        • Zanini A.
        • et al.
        Endoscopic surveillance for congenital diaphragmatic hernia: unexpected prevalence of silent esophagitis.
        Eur J Pediatr Surg. 2016; 26: 291-295
        • Steven M.J.
        • Fyfe A.H.B.
        • Raine P.A.M.
        • Watt I.
        Esophageal adenocarcinoma: a long-term complication of congenital diaphragmatic hernia?.
        J Pediatr Surg. 2007; 42: e1-e3