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Eur J Pediatr Surg 2015; 25(04): 326-332
DOI: 10.1055/s-0035-1559818
Review Article
Georg Thieme Verlag KG Stuttgart · New York

Dysphagia in Children with Esophageal Atresia: Current Diagnostic Options

Maissa Rayyan
1   Neonatal Intensive Care Unit, University Hospital Leuven, Leuven, Belgium
,
Karel Allegaert
1   Neonatal Intensive Care Unit, University Hospital Leuven, Leuven, Belgium
,
Taher Omari
2   Translational Research Center for Gastrointestinal Disorders, KU Leuven, Leuven, Belgium
3   School of Medicine, Flinders University, Adelaide, Australia
,
Nathalie Rommel
2   Translational Research Center for Gastrointestinal Disorders, KU Leuven, Leuven, Belgium
4   Neurosciences ExpORL, KU Leuven, Leuven, Belgium
› Author Affiliations
Further Information

Publication History

24 June 2015

26 June 2015

Publication Date:
26 August 2015 (online)

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Abstract

Dysphagia or swallowing disorder is very common (range, 15–52%) in patients with esophageal atresia. Children present with a wide range of symptoms. The most common diagnostic tools to evaluate esophageal dysphagia, such as upper barium study and manometry, aim to characterize anatomy and function of the esophageal body and the esophagogastric junction (EGJ). Using these technologies, a variety of pathological motor patterns have been identified in children with esophageal atresia. However, the most challenging part of diagnosing patients with esophageal dysphagia lies in the fact that these methods fail to link functional symptoms such as dysphagia with the esophageal motor disorders observed. A recent method, called pressure-flow analysis (PFA), uses simultaneously acquired impedance and manometry measurements, and applies an integrated analysis of these recordings to derive quantitative pressure-flow metrics. These pressure-flow metrics allow detection of the interplay between bolus flow, motor patterns, and symptomatology by combining data on bolus transit and bolus flow resistance. Based on a dichotomous categorization, flow resistance at the EGJ and ineffective esophageal bolus transit can be determined. This method has the potential to guide therapeutic decisions for esophageal dysmotility in pediatric patients with esophageal atresia.

Keywords

swallowing - esophageal atresia - children - manometry - impedance - measurement
 

  • References

  • 1 Pedersen RN, Calzolari E, Husby S, Garne E ; EUROCAT Working group. Oesophageal atresia: prevalence, prenatal diagnosis and associated anomalies in 23 European regions. Arch Dis Child 2012; 97 (3) 227-232
    CrossrefPubMedGoogle Scholar
  • 2 Deurloo JA, Ekkelkamp S, Schoorl M, Heij HA, Aronson DC. Esophageal atresia: historical evolution of management and results in 371 patients. Ann Thorac Surg 2002; 73 (1) 267-272
    CrossrefPubMedGoogle Scholar
  • 3 Lilja HE, Wester T. Outcome in neonates with esophageal atresia treated over the last 20 years. Pediatr Surg Int 2008; 24 (5) 531-536
    CrossrefPubMedGoogle Scholar
  • 4 Lopez PJ, Keys C, Pierro A , et al. Oesophageal atresia: improved outcome in high-risk groups?. J Pediatr Surg 2006; 41 (2) 331-334
    CrossrefPubMedGoogle Scholar
  • 5 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
    CrossrefPubMedGoogle Scholar
  • 6 Deurloo JA, Ekkelkamp S, Hartman EE, Sprangers MA, Aronson DC. Quality of life in adult survivors of correction of esophageal atresia. Arch Surg 2005; 140 (10) 976-980
    CrossrefPubMedGoogle Scholar
  • 7 Taylor AC, Breen KJ, Auldist A , et al. Gastroesophageal reflux and related pathology in adults who were born with esophageal atresia: a long-term follow-up study. Clin Gastroenterol Hepatol 2007; 5 (6) 702-706
    CrossrefPubMedGoogle Scholar
  • 8 Liu XM, Aras-Lopez R, Martinez L, Tovar JA. Abnormal development of lung innervation in experimental esophageal atresia. Eur J Pediatr Surg 2012; 22 (1) 67-73
    Article in Thieme ConnectPubMedGoogle Scholar
  • 9 Little DC, Rescorla FJ, Grosfeld JL, West KW, Scherer LR, Engum SA. Long-term analysis of children with esophageal atresia and tracheoesophageal fistula. J Pediatr Surg 2003; 38 (6) 852-856
    CrossrefPubMedGoogle Scholar
  • 10 Chetcuti P, Myers NA, Phelan PD, Beasley SW. Adults who survived repair of congenital oesophageal atresia and tracheo-oesophageal fistula. BMJ 1988; 297 (6644) 344-346
    CrossrefPubMedGoogle Scholar
  • 11 Lemoine C, Aspirot A, Le Henaff G, Piloquet H, Lévesque D, Faure C. Characterization of esophageal motility following esophageal atresia repair using high-resolution esophageal manometry. J Pediatr Gastroenterol Nutr 2013; 56 (6) 609-614
    CrossrefPubMedGoogle Scholar
  • 12 Lopes MF, Botelho MF. Midterm follow-up of esophageal anastomosis for esophageal atresia repair: long-gap versus non-long-gap. Dis Esophagus 2007; 20 (5) 428-435
    CrossrefPubMedGoogle Scholar
  • 13 Omari T, Tack J, Rommel N. Impedance as an adjunct to manometric testing to investigate symptoms of dysphagia: What it has failed to do and what it may tell us in the future. United European Gastroenterol J 2014; 2 (5) 355-366
    PubMedGoogle Scholar
  • 14 Aspirot A, Faure C. Esophageal dysmotility: characterization and pathophysiology. Dis Esophagus 2013; 26 (4) 405-409
    CrossrefPubMedGoogle Scholar
  • 15 Romeo G, Zuccarello B, Proietto F, Romeo C. Disorders of the esophageal motor activity in atresia of the esophagus. J Pediatr Surg 1987; 22 (2) 120-124
    CrossrefPubMedGoogle Scholar
  • 16 Romeo C, Bonanno N, Baldari S , et al. Gastric motility disorders in patients operated on for esophageal atresia and tracheoesophageal fistula: long-term evaluation. J Pediatr Surg 2000; 35 (5) 740-744
    CrossrefPubMedGoogle Scholar
  • 17 Lemoine C, Aspirot A, Morris M, Faure C. Esophageal dysmotility is present before surgery in isolated tracheoesophageal fistula. J Pediatr Gastroenterol Nutr 2015; 60 (5) 642-644
    CrossrefPubMedGoogle Scholar
  • 18 Boleken M, Demirbilek S, Kirimiloglu H , et al. Reduced neuronal innervation in the distal end of the proximal esophageal atretic segment in cases of esophageal atresia with distal tracheoesophageal fistula. World J Surg 2007; 31 (7) 1512-1517
    CrossrefPubMedGoogle Scholar
  • 19 Nakazato Y, Landing BH, Wells TR. Abnormal Auerbach plexus in the esophagus and stomach of patients with esophageal atresia and tracheoesophageal fistula. J Pediatr Surg 1986; 21 (10) 831-837
    CrossrefPubMedGoogle Scholar
  • 20 Qi BQ, Merei J, Farmer P , et al. The vagus and recurrent laryngeal nerves in the rodent experimental model of esophageal atresia. J Pediatr Surg 1997; 32 (11) 1580-1586
    CrossrefPubMedGoogle Scholar
  • 21 Midrio P, Alaggio R, Strojna A , et al. Reduction of interstitial cells of Cajal in esophageal atresia. J Pediatr Gastroenterol Nutr 2010; 51 (5) 610-617
    CrossrefPubMedGoogle Scholar
  • 22 Cucchiara S, Staiano A, Di Lorenzo C , et al. Esophageal motor abnormalities in children with gastroesophageal reflux and peptic esophagitis. J Pediatr 1986; 108 (6) 907-910
    CrossrefPubMedGoogle Scholar
  • 23 Davies MR. Anatomy of the extrinsic motor nerve supply to mobilized segments of the oesophagus disrupted by dissection during repair of oesophageal atresia with distal fistula. Br J Surg 1996; 83 (9) 1268-1270
    CrossrefPubMedGoogle Scholar
  • 24 Shono T, Suita S, Arima T , et al. Motility function of the esophagus before primary anastomosis in esophageal atresia. J Pediatr Surg 1993; 28 (5) 673-676
    CrossrefPubMedGoogle Scholar
  • 25 Baird R, Laberge JM, Lévesque D. Anastomotic stricture after esophageal atresia repair: a critical review of recent literature. Eur J Pediatr Surg 2013; 23 (3) 204-213
    Article in Thieme ConnectPubMedGoogle Scholar
  • 26 McCann F, Michaud L, Aspirot A, Levesque D, Gottrand F, Faure C. Congenital esophageal stenosis associated with esophageal atresia. Dis Esophagus 2015; 28 (3) 211-215
    CrossrefPubMedGoogle Scholar
  • 27 Sun LY, Laberge JM, Yousef Y, Baird R. The Esophageal Anastomotic Stricture Index (EASI) for the management of esophageal atresia. J Pediatr Surg 2015; 50 (1) 107-110
    CrossrefPubMedGoogle Scholar
  • 28 Gyawali CP, Bredenoord AJ, Conklin JL , et al. Evaluation of esophageal motor function in clinical practice. Neurogastroenterol Motil 2013; 25 (2) 99-133
    CrossrefPubMedGoogle Scholar
  • 29 Bredenoord AJ, Fox M, Kahrilas PJ, Pandolfino JE, Schwizer W, Smout AJ ; International High Resolution Manometry Working Group. Chicago classification criteria of esophageal motility disorders defined in high resolution esophageal pressure topography. Neurogastroenterol Motil 2012; 24 (Suppl. 01) 57-65
    CrossrefPubMedGoogle Scholar
  • 30 Kahrilas PJ, Bredenoord AJ, Fox M , et al; International High Resolution Manometry Working Group. The Chicago Classification of esophageal motility disorders, v3.0. Neurogastroenterol Motil 2015; 27 (2) 160-174
    CrossrefPubMedGoogle Scholar
  • 31 van Wijk M, Knüppe F, Omari T, de Jong J, Benninga M. Evaluation of gastroesophageal function and mechanisms underlying gastroesophageal reflux in infants and adults born with esophageal atresia. J Pediatr Surg 2013; 48 (12) 2496-2505
    CrossrefPubMedGoogle Scholar
  • 32 Pedersen RN, Markøw S, Kruse-Andersen S , et al. Esophageal atresia: gastroesophageal functional follow-up in 5-15 year old children. J Pediatr Surg 2013; 48 (12) 2487-2495
    CrossrefPubMedGoogle Scholar
  • 33 Di Pace MR, Caruso AM, Catalano P, Casuccio A, Cimador M, De Grazia E. Evaluation of esophageal motility and reflux in children treated for esophageal atresia with the use of combined multichannel intraluminal impedance and pH monitoring. J Pediatr Surg 2011; 46 (3) 443-451
    CrossrefPubMedGoogle Scholar
  • 34 Fröhlich T, Otto S, Weber P , et al. Combined esophageal multichannel intraluminal impedance and pH monitoring after repair of esophageal atresia. J Pediatr Gastroenterol Nutr 2008; 47 (4) 443-449
    CrossrefPubMedGoogle Scholar
  • 35 Catalano P, Di Pace MR, Caruso AM, Casuccio A, De Grazia E. Gastroesophageal reflux in young children treated for esophageal atresia: evaluation with pH-multichannel intraluminal impedance. J Pediatr Gastroenterol Nutr 2011; 52 (6) 686-690
    CrossrefPubMedGoogle Scholar
  • 36 Omari TI, Papathanasopoulos A, Dejaeger E , et al. Reproducibility and agreement of pharyngeal automated impedance manometry with videofluoroscopy. Clin Gastroenterol Hepatol 2011; 9 (10) 862-867
    CrossrefPubMedGoogle Scholar
  • 37 Omari TI, Dejaeger E, Van Beckevoort D , et al. A novel method for the nonradiological assessment of ineffective swallowing. Am J Gastroenterol 2011; 106 (10) 1796-1802
    CrossrefPubMedGoogle Scholar
  • 38 Rommel N, Van Oudenhove L, Tack J, Omari TI. Automated impedance manometry analysis as a method to assess esophageal function. Neurogastroenterol Motil 2014; 26 (5) 636-645
    CrossrefPubMedGoogle Scholar
  • 39 Omari TI, Dejaeger E, van Beckevoort D , et al. A method to objectively assess swallow function in adults with suspected aspiration. Gastroenterology 2011; 140 (5) 1454-1463
    CrossrefPubMedGoogle Scholar
  • 40 Rommel N, Omari TI, Selleslagh M , et al. High-resolution manometry combined with impedance measurements discriminates the cause of dysphagia in children (e-pub ahead of print). Eur J Pediatr 2015; ; doi: 10.1007/s00431-015-2582-9
    PubMedGoogle Scholar
  • 41 Myers JC, Nguyen NQ, Jamieson GG , et al. Susceptibility to dysphagia after fundoplication revealed by novel automated impedance manometry analysis. Neurogastroenterol Motil 2012; 24 (9) 812-e393
    CrossrefPubMedGoogle Scholar
  • 42 Nguyen NQ, Holloway RH, Smout AJ, Omari TI. Automated impedance-manometry analysis detects esophageal motor dysfunction in patients who have non-obstructive dysphagia with normal manometry. Neurogastroenterol Motil 2013; 25 (3) 238-245 , e164
    CrossrefPubMedGoogle Scholar
  • 43 Chen CL, Yi CH, Liu TT, Hsu CS, Omari TI. Characterization of esophageal pressure-flow abnormalities in patients with non-obstructive dysphagia and normal manometry findings. J Gastroenterol Hepatol 2013; 28 (6) 946-953
    CrossrefPubMedGoogle Scholar
  • 44 Wheatley MJ, Coran AG, Wesley JR. Efficacy of the Nissen fundoplication in the management of gastroesophageal reflux following esophageal atresia repair. J Pediatr Surg 1993; 28 (1) 53-55
    CrossrefPubMedGoogle Scholar