Mini-symposium: Oesophageal Atresia and Tracheo-oesophageal Fistula
Feeding Difficulties in Children with Esophageal Atresia

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Summary

The current available literature evaluating feeding difficulties in children with esophageal atresia was reviewed. The published literature was searched through PubMed using a pre-defined search strategy. Feeding difficulties are commonly encountered in children and adults with repaired esophageal atresia [EA]. The mechanism for abnormal feeding includes both esophageal and oropharyngeal dysphagia. Esophageal dysphagia is commonly reported in patients with EA and causes include dysmotility, anatomic lesions, esophageal outlet obstruction and esophageal inflammation. Endoscopic evaluation, esophageal manometry and esophograms can be useful studies to evaluate for causes of esophageal dysphagia. Oropharyngeal dysfunction and aspiration are also important mechanisms for feeding difficulties in patients with EA. These patients often present with respiratory symptoms. Videofluoroscopic swallow study, salivagram, fiberoptic endoscopic evaluation of swallowing and high-resolution manometry can all be helpful tools to identify aspiration. Once diagnosed, management goals include reduction of aspiration during swallowing, reducing full column reflux into the oropharynx and continuation of oral feeding to maintain skills. We review specific strategies which can be used to reduce aspiration of gastric contents, including thickening feeds, changing feeding schedule, switching formula, trialing transpyloric feeds and fundoplication.

Introduction

Esophageal atresia, with or without tracheoesophageal fistula (TEF), is a congenital anomaly of the esophagus affecting one in every 2500-3000 live births [1]. Infant survival of this condition is high, with reported survival rates of over 90% [2]. However, gastrointestinal and respiratory complications are well documented in children, adolescents and adults with repaired EA [3], [4], [5], [6], [7], [8], [9]. Feeding disorders in children with esophageal atresia are common in clinical practice but the literature supporting these observations is limited. Between 6% and 52% of patients have some abnormalities of feeding [7], [10], [11]. The majority of studies focus on esophageal abnormalities as source of feeding difficulties. There are no prospective studies on oropharyngeal dysfunction or aerodigestive abnormalities in patients with esophageal atresia.

Section snippets

Overview of Feeding Difficulties

Feeding difficulties have been described in patients with EA. Puntis et al., [11] first characterized feeding difficulties in 124 children with EA. Compared to healthy controls, children born with EA were significantly more likely to eat slowly, refuse meals, cough or choke during eating and vomit with meals. Chetcuti et al., [12] described similar feeding difficulties in childhood, but noted that overall, these difficulties lessen with age, with < 10% of patients age 15 or older reporting

Esophageal dysphagia as a cause for feeding difficulty

Dysphagia is a common complaint in patients with EA and causes include dysmotility, anatomic lesions, esophageal outlet obstruction and esophageal inflammation. The reported prevalence of dysphagia in patients with EA ranges from 38% to 85% [3], [8], [9], [12], [16], [17]. A recent systematic review and meta-analysis by Connor et al. found an overall pooled estimated prevalence of 50.3% (95% CI 35.7 – 64.8) [5]. The evaluation of dysphagia involves (1) an esophagram to assess for strictures or,

Diagnosis of Aspiration

There are many different methods of diagnosing oropharyngeal dysphagia and all of them are complementary with no clear gold standard test. Studies have suggested that the agreement between studies is poor. Baikie et al. for example, studied the agreement between three tests for aspiration – barium videofluoroscopy, salivagram and milk scan - in 63 children with cerebral palsy [35]. The authors found that overall agreement between the tests was poor, with a maximum kappa of 0.20 suggesting that

Management of oropharyngeal dysphagia with aspiration

Many causes of aspiration improve over time and thus management decisions regarding feeding must be made in the context of the likelihood of improvement in aspiration. Management goals include: reduction of aspiration during swallowing, reducing full column reflux into the oropharynx and continuation of oral feeding to maintain skills. While there are no studies on the management of aspiration in children with EA, conclusions can be drawn from the available literature in other populations.

Discussion

Feeding difficulties are common in patients with repaired EA. In this review, we investigate the mechanisms for abnormal feeding in these patients. Esophageal dysphagia is well-described in patients with EA and is often due to anatomic abnormalities such as strictures, esophageal dysmotility or mucosal inflammation. While respiratory symptoms are highly prevalent in patients with EA, there are very few studies evaluating oropharyngeal dysfunction in this population. Aspiration is an

Funding Source

This work was supported by the Translational Research Program at Children's Hospital Boston and NIH NIDDK R01 DK097112. It was funded by the NASPGHAN/ASTRA research award for disorders of the upper tract.

Financial Disclosures

None.

Educational Aims

  • To describe the mechanism of esophageal dysphagia and oropharyngeal dysphagia/aspiration as mechanisms for feeding difficulties in patients with esophageal atresia

  • To highlight the prevalence of respiratory symptoms in patients with esophageal atresia

  • To review methods for diagnosing aspiration

  • To discuss treatment strategies for management of aspiration

Directions for Future Research

  • To characterize oropharyngeal dysphagia and aspiration in patients with esophageal atresia.

  • To investigate the relationship between aspiration, respiratory symptoms and feeding difficulties in patients with esophageal atresia.

  • To determine the best method of diagnosing aspiration in patients with repaired esophageal atresia.

  • To optimize management of aspiration in patients with esophageal atresia.

References (69)

  • K.A. Weir et al.

    Oropharyngeal aspiration and silent aspiration in children

    Chest

    (2011)
  • A.P. da Silva et al.

    Comparison between videofluoroscopy and endoscopic evaluation of swallowing for the diagnosis of dysphagia in children

    Otolaryngol Head Neck Surg

    (2010)
  • M. Sitton et al.

    Fiberoptic Endoscopic Evaluation of Swallowing in children: feeding outcomes related to diagnostic groups and endoscopic findings

    Int J Pediatr Otorhinolaryngol

    (2011)
  • T.I. Omari et al.

    A method to objectively assess swallow function in adults with suspected aspiration

    Gastroenterology

    (2011)
  • J.S.W. Lee et al.

    Can continuous pump feeding reduce the incidence of pneumonia in nasogastric tube-fed patients? A randomized controlled trial

    Clin Nutr

    (2010)
  • T.E. Bowling et al.

    The effects of bolus and continuous nasogastric feeding on gastro-oesophageal reflux and gastric emptying in healthy volunteers: A randomised three-way crossover pilot study

    Clinical Nutrition

    (2008)
  • S.L. Lee et al.

    Hospital admissions for respiratory symptoms and failure to thrive before and after Nissen fundoplication

    Journal of Pediatric Surgery

    (2008)
  • L. Spitz

    Oesophageal atresia

    Orphanet J Rare Dis

    (2007)
  • H.E. Lilja et al.

    Outcome in neonates with esophageal atresia treated over the last 20 years

    Pediatr Surg Int

    (2008)
  • K. Malmström et al.

    Longitudinal Follow-up of Bronchial Inflammation, Respiratory Symptoms, and Pulmonary Function in Adolescents after Repair of Esophageal Atresia with Tracheoesophageal Fistula

    The Journal of Pediatrics

    (2008)
  • T. Kovesi

    Long-term respiratory complications of congenital esophageal atresia with or without tracheoesophageal fistula: an update

    Dis Esophagus

    (2013)
  • C. Legrand et al.

    Long-term outcome of children with oesophageal atresia type III

    Archives of Disease in Childhood

    (2012)
  • P. Chetcuti et al.

    Adults who survived repair of congenital oesophageal atresia and tracheo-oesophageal fistula

    BMJ

    (1988)
  • R. Baird et al.

    A pilot investigation of feeding problems in children with esophageal atresia

    Dis Esophagus

    (2015)
  • J.W. Puntis et al.

    Growth and feeding problems after repair of oesophageal atresia

    Archives of Disease in Childhood

    (1990)
  • P. Chetcuti et al.

    Gastrointestinal morbidity and growth after repair of oesophageal atresia and tracheo-oesophageal fistula

    Archives of Disease in Childhood

    (1993)
  • M. Ramsay et al.

    Feeding difficulties in children with esophageal atresia: treatment by a multidisciplinary team

    Dis Esophagus

    (2013)
  • J. Castilloux et al.

    Endoscopic assessment of children with esophageal atresia: Lack of relationship of esophagitis and esophageal metaplasia to symptomatology

    Can J Gastroenterol

    (2010)
  • S.J. Sistonen et al.

    Esophageal Morbidity and Function in Adults With Repaired Esophageal Atresia With Tracheoesophageal Fistula

    Annals of Surgery

    (2010)
  • J.A. Deurloo et al.

    Adults with corrected oesophageal atresia: is oesophageal function associated with complaints and/or quality of life?

    Pediatr Surg Int

    (2008)
  • M.F. Lopes et al.

    Midterm follow-up of esophageal anastomosis for esophageal atresia repair: long-gap versus non-long-gap

    Dis Esophagus

    (2007)
  • C. Lemoine et al.

    Characterization of esophageal motility following esophageal atresia repair using high-resolution esophageal manometry

    Journal of Pediatric Gastroenterology and Nutrition

    (2013)
  • T. Fröhlich et al.

    Combined esophageal multichannel intraluminal impedance and pH monitoring after repair of esophageal atresia

    Journal of Pediatric Gastroenterology and Nutrition

    (2008)
  • C. Lemoine et al.

    Esophageal dysmotility is present before surgery in isolated tracheoesophageal fistula

    Journal of Pediatric Gastroenterology and Nutrition

    (2015)
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