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Gastrointestinal Dysmotility and the Implications for Respiratory Disease

Abstract

Purpose of the review

Gastroesophageal reflux disease (GERD) is frequently implicated as a cause for respiratory disease. However, there is growing evidence that upper gastrointestinal dysmotility may play a significantly larger role in genesis of respiratory symptoms and development of underlying pulmonary pathology. This paper will discuss the differential diagnosis for esophageal and gastric dysmotility in aerodigestive patients and will review the key diagnostic and therapeutic interventions for this dysmotility.

Recent findings

Previous studies have shown an association between GERD and pulmonary pathology in children with aerodigestive disorders. Recent publications have demonstrated the presence of esophageal and gastric dysfunction, using fluoroscopic and nuclear medicine studies, in aerodigestive patients who commonly present to pulmonary and otolaryngology clinics. High-resolution impedance manometry (HRIM) has revolutionized our understanding of esophageal dysmotility and its role in pathogenesis of aspiration and esophageal dysfunction and subsequent respiratory compromise.

Summary

Esophageal and gastric dysmotility have a profound effect on development of respiratory symptoms and pulmonary sequalae in aerodigestive patients. However, our understanding of the pathophysiology is in its infancy. Prospective studies are needed to address key clinical questions such as: What degree of dysmotility initiates respiratory compromise? What diagnostic tests and therapeutic options best predict aerodigestive outcomes?

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References and Recommended Reading

Papers of particular interest, published recently, have been highlighted as: • Of importance •• Of major importance

  1. Chumpitazi B, Nurko S. Pediatric gastrointestinal motility disorders: challenges and a clinical update. Gastroenterol Hepatol (N Y). 2008;4(2):140–8.

    Google Scholar 

  2. Scholes MA, McEvoy T, Mousa H, Wiet GJ. Cricopharyngeal achalasia in children: botulinum toxin injection as a tool for diagnosis and treatment. Laryngoscope. 2014;124(6):1475–80.

    Article  PubMed  Google Scholar 

  3. Huoh KC, Messner AH. Cricopharyngeal achalasia in children: indications for treatment and management options. Curr Opin Otolaryngol Head Neck Surg. 2013;21(6):576–80.

    PubMed  Google Scholar 

  4. Mohan S, Bowe SN, Hirner LM, Zar-Kessler C, Hartnick CJ. Modified approach for pediatric external cricopharyngeal myotomy. Int J Pediatr Otorhinolaryngol. 2018;105:111–4.

    Article  PubMed  Google Scholar 

  5. Kocdor P, Siegel ER, Tulunay-Ugur OE. Cricopharyngeal dysfunction: a systematic review comparing outcomes of dilatation, botulinum toxin injection, and myotomy. Laryngoscope. 2016;126(1):135–41.

    Article  CAS  PubMed  Google Scholar 

  6. Muraji T, Takamizawa S, Satoh S, Nishijima E, Tsugawa C, Tamura A, et al. Congenital cricopharyngeal achalasia: diagnosis and surgical management. J Pediatr Surg. 2002;37(5):E12.

    Article  PubMed  Google Scholar 

  7. Kahrilas PJ, Bredenoord AJ, Fox M, Gyawali CP, Roman S, Smout AJ, et al. The Chicago Classification of esophageal motility disorders, v3.0. Neurogastroenterol Motil. 2015;27(2):160–74.

    Article  CAS  PubMed  Google Scholar 

  8. • Rommel N, Selleslagh M, Hoffman I, Smet MH, Davidson G, Tack J, et al. Objective assessment of swallow function in children with suspected aspiration using pharyngeal automated impedance manometry. J Pediatr Gastroenterol Nutr. 2014;58(6):789–94 This study highlights the relationship between UES pressures and bolus flow dynamics in children by using automated impedance manometry to define swallow risk index in order to predict aspiration risk in children. Increased post swallow pharyngeal residue and UES pressures during maximum bolus flow were found in children with aspiration.

    PubMed  Google Scholar 

  9. • Rosen R, Garza JM, Tipnis N, Nurko S. An ANMS-NASPGHAN consensus document on esophageal and antroduodenal manometry in children. Neurogastroenterol Motil. 2018;30(3). This study provides an ANMS-NASGPHAN first consensus document on esophageal and antroduodenal manometry in children. It specifically describes the indications, performance, and interpretation of the tests.

  10. Sewell RK, Bauman NM. Congenital cricopharyngeal achalasia: management with botulinum toxin before myotomy. Arch Otolaryngol Head Neck Surg. 2005;131(5):451–3.

    Article  PubMed  Google Scholar 

  11. Messner A, Ho AS, Malhotra PS, Koltai PJ, Barnes MA. The use of botulinum toxin for pediatric cricopharyngeal achalasia. Int J Pediatr Otorhinolaryngol. 2011;75(6):830–4.

    Article  PubMed  Google Scholar 

  12. Erdeve O, Kologlu M, Saygili B, Atasay B, Arsan S. Primary cricopharyngeal achalasia in a newborn treated by balloon dilatation: a case report and review of the literature. Int J Pediatr Otorhinolaryngol. 2007;71(1):165–8.

    Article  PubMed  Google Scholar 

  13. Gollu G, Demir N, Ates U, Aslan SS, Ergun E, Kucuk G, et al. Effective management of cricopharyngeal achalasia in infants and children with dilatation alone. J Pediatr Surg. 2016;51(11):1751–4.

    Article  PubMed  Google Scholar 

  14. Vaezi MF, Pandolfino JE, Vela MF. ACG clinical guideline: diagnosis and management of achalasia. Am J Gastroenterol. 2013;108(8):1238–49 quiz 50.

  15. Krill JT, Naik RD, Vaezi MF. Clinical management of achalasia: current state of the art. Clin Exp Gastroenterol. 2016;9:71–82.

    CAS  PubMed  PubMed Central  Google Scholar 

  16. Marlais M, Fishman JR, Fell JM, Haddad MJ, Rawat DJ. UK incidence of achalasia: an 11-year national epidemiological study. Arch Dis Child. 2011;96(2):192–4.

    Article  CAS  PubMed  Google Scholar 

  17. Smits M, van Lennep M, Vrijlandt R, Benninga M, Oors J, Houwen R, et al. Pediatric achalasia in the Netherlands: incidence, clinical course, and quality of life. J Pediatr. 2016;169:110–5 e3.

    Article  PubMed  Google Scholar 

  18. Upadhyaya VD, Gangopadhyaya AN, Gupta DK, Sharma SP, Kumar V, Gopal SC. Esophageal achalasia of unknown etiology in infants. World J Pediatr. 2008;4(1):63–5.

    Article  PubMed  Google Scholar 

  19. Zilberstein B, de Cleva R, Gabriel AG, Neto SG, Gama-Rodrigues JJ. Congenital achalasia: facts and fantasies. Dis Esophagus. 2005;18(5):335–7.

    Article  CAS  PubMed  Google Scholar 

  20. Boeckxstaens GE, Jonge WD, van den Wijngaard RM, Benninga MA. Achalasia: from new insights in pathophysiology to treatment. J Pediatr Gastroenterol Nutr. 2005;41(Suppl 1):S36–7.

    Article  PubMed  Google Scholar 

  21. Iwanczak F, Smigiel R, Blitek A, Huebner A. The triple “a” syndrome confirmed by molecular analysis: a case report of 7-year-old boy. J Pediatr Gastroenterol Nutr. 2005;40(1):87–9.

    Article  PubMed  Google Scholar 

  22. Myers NA, Jolley SG, Taylor R. Achalasia of the cardia in children: a worldwide survey. J Pediatr Surg. 1994;29(10):1375–9.

    Article  CAS  PubMed  Google Scholar 

  23. Hussain SZ, Thomas R, Tolia V. A review of achalasia in 33 children. Dig Dis Sci. 2002;47(11):2538–43.

    Article  CAS  PubMed  Google Scholar 

  24. Zhang Y, Xu CD, Zaouche A, Cai W. Diagnosis and management of esophageal achalasia in children: analysis of 13 cases. World J Pediatr. 2009;5(1):56–9.

    Article  PubMed  Google Scholar 

  25. Fisichella PM, Raz D, Palazzo F, Niponmick I, Patti MG. Clinical, radiological, and manometric profile in 145 patients with untreated achalasia. World J Surg. 2008;32(9):1974–9.

    Article  PubMed  Google Scholar 

  26. Lee CW, Kays DW, Chen MK, Islam S. Outcomes of treatment of childhood achalasia. J Pediatr Surg. 2010;45(6):1173–7.

    Article  PubMed  PubMed Central  Google Scholar 

  27. Ambartsumyan L, Rodriguez L. Gastrointestinal motility disorders in children. Gastroenterol Hepatol (N Y). 2014;10(1):16–26.

    Google Scholar 

  28. Nurko S. Motility Disorders in Children. Pediatr Clin N Am. 2017;64(3):593–612.

    Article  Google Scholar 

  29. Kahrilas PJ, Bredenoord AJ, Fox M, Gyawali CP, Roman S, Smout A, et al. Expert consensus document: advances in the management of oesophageal motility disorders in the era of high-resolution manometry: a focus on achalasia syndromes. Nat Rev Gastroenterol Hepatol. 2017;14(11):677–88.

    Article  PubMed  Google Scholar 

  30. Stavropoulos SN, Friedel D, Modayil R, Parkman HP. Diagnosis and management of esophageal achalasia. BMJ. 2016;354:i2785.

    Article  PubMed  Google Scholar 

  31. •• Makharia GK, Seith A, Sharma SK, Sinha A, Goswami P, Aggarwal A, et al. Structural and functional abnormalities in lungs in patients with achalasia. Neurogastroenterol Motil. 2009;21(6):603–8, e20 This study illustrates the degree of lung abnormalities found in adults who present with achalasia. Greater than half of patients with achalasia were found to have restrictive and/or obstructive pattern on pulmonary function testing and structural changes on high resolution lung CT.

    Article  CAS  PubMed  Google Scholar 

  32. Wauters L, Van Oudenhove L, Selleslagh M, Vanuytsel T, Boeckxstaens G, Tack J, et al. Balloon dilation of the esophago-gastric junction affects lower and upper esophageal sphincter function in achalasia. Neurogastroenterol Motil. 2014;26(1):69–76.

    Article  CAS  PubMed  Google Scholar 

  33. Pandolfino JE, Kwiatek MA, Nealis T, Bulsiewicz W, Post J, Kahrilas PJ. Achalasia: a new clinically relevant classification by high-resolution manometry. Gastroenterology. 2008;135(5):1526–33.

    Article  PubMed  Google Scholar 

  34. Morera C, Nurko S. Heterogeneity of lower esophageal sphincter function in children with achalasia. J Pediatr Gastroenterol Nutr. 2012;54(1):34–40.

    Article  PubMed  Google Scholar 

  35. • Singendonk MMJ, Rosen R, Oors J, Rommel N, van Wijk MP, Benninga MA, et al. Intra- and interrater reliability of the Chicago Classification of achalasia subtypes in pediatric high-resolution esophageal manometry (HRM) recordings. Neurogastroenterol Motil 2017;29(11). This multicenter study examines the utility and applicability of the Chicago Classification in children with achalasia using high resolution esophageal manometry. The Chicago Classification can be used to differentiate childen with and without achalasia with excellent intra-rater variablity and moderate inter-rater variability.

  36. Babu R, Grier D, Cusick E, Spicer RD. Pneumatic dilatation for childhood achalasia. Pediatr Surg Int. 2001;17(7):505–7.

    Article  CAS  PubMed  Google Scholar 

  37. Saliakellis E, Thapar N, Roebuck D, Cristofori F, Cross K, Kiely E, et al. Long-term outcomes of Heller’s myotomy and balloon dilatation in childhood achalasia. Eur J Pediatr. 2017;176(7):899–907.

    Article  PubMed  Google Scholar 

  38. Mehra M, Bahar RJ, Ament ME, Waldhausen J, Gershman G, Georgeson K, et al. Laparoscopic and thoracoscopic esophagomyotomy for children with achalasia. J Pediatr Gastroenterol Nutr. 2001;33(4):466–71.

    Article  CAS  PubMed  Google Scholar 

  39. Tannuri AC, Tannuri U, Velhote MC, Romao RL. Laparoscopic extended cardiomyotomy in children: an effective procedure for the treatment of esophageal achalasia. J Pediatr Surg. 2010;45(7):1463–6.

    Article  PubMed  Google Scholar 

  40. Corda L, Pacilli M, Clarke S, Fell JM, Rawat D, Haddad M. Laparoscopic oesophageal cardiomyotomy without fundoplication in children with achalasia: a 10-year experience: a retrospective review of the results of laparoscopic oesophageal cardiomyotomy without an anti-reflux procedure in children with achalasia. Surg Endosc. 2010;24(1):40–4.

    Article  PubMed  Google Scholar 

  41. Mattioli G, Esposito C, Pini Prato A, Doldo P, Castagnetti M, Barabino A, et al. Results of the laparoscopic Heller-Dor procedure for pediatric esophageal achalasia. Surg Endosc. 2003;17(10):1650–2.

    Article  CAS  PubMed  Google Scholar 

  42. •• Pacilli M, Davenport M. Results of laparoscopic Heller’s myotomy for achalasia in children: a systematic review of the literature. J Laparoendosc Adv Surg Tech A. 2017;27(1):82–90 This study is a systamatic review of 21 studies of 331 children with laparoscopic Heller’s myotomy (LHM) for achalasia. LHM was found to be affective in 85%. Moreover, when comparing children with and without fundoplication, there was no difference in dysphagia or incidence of reflux.

    Article  PubMed  Google Scholar 

  43. Schlottmann F, Luckett DJ, Fine J, Shaheen NJ, Patti MG. Laparoscopic Heller myotomy versus peroral endoscopic myotomy (POEM) for achalasia: a systematic review and meta-analysis. Ann Surg. 2018;267(3):451–60.

    Article  PubMed  Google Scholar 

  44. Awaiz A, Yunus RM, Khan S, Memon B, Memon MA. Systematic review and meta-analysis of perioperative outcomes of peroral endoscopic myotomy (POEM) and laparoscopic Heller Myotomy (LHM) for Achalasia. Surg Laparosc Endosc Percutan Tech. 2017;27(3):123–31.

    Article  PubMed  Google Scholar 

  45. Li C, Tan Y, Wang X, Liu D. Peroral endoscopic myotomy for treatment of achalasia in children and adolescents. J Pediatr Surg. 2015;50(1):201–5.

    Article  PubMed  Google Scholar 

  46. Tan Y, Zhu H, Li C, Chu Y, Huo J, Liu D. Comparison of peroral endoscopic myotomy and endoscopic balloon dilation for primary treatment of pediatric achalasia. J Pediatr Surg. 2016;51(10):1613–8.

    Article  PubMed  Google Scholar 

  47. Kethman WC, Thorson CM, Sinclair TJ, Berquist WE, Chao SD, Wall JK. Initial experience with peroral endoscopic myotomy for treatment of achalasia in children. J Pediatr Surg. 2017;53(8):1532–6.

    Article  PubMed  Google Scholar 

  48. Parshad R, Devana SK, Panchanatheeswaran K, Saraya A, Makharia GK, Sharma SK, et al. Clinical, radiological and functional assessment of pulmonary status in patients with achalasia cardia before and after treatment. Eur J Cardiothorac Surg. 2012;42(5):e90–5.

    Article  PubMed  Google Scholar 

  49. Khandelwal S, Petersen R, Tatum R, Sinan H, Aaronson D, Mier F, et al. Improvement of respiratory symptoms following Heller myotomy for achalasia. J Gastrointest Surg. 2011;15(2):235–9.

    Article  PubMed  Google Scholar 

  50. • Andolfi C, Kavitt RT, Herbella FA, Patti MG. Achalasia and respiratory symptoms: effect of laparoscopic Heller myotomy. J Laparoendosc Adv Surg Tech A. 2016;26(9):675–9 Findings of this study highlight the predominance of respiratory symptoms in patients with achalasia. Moreover, it emphasizes the critical point that respiratory symptoms are alleviated in greater than 90 % of patients who undergo laparoscopic heller myotomy to correct the distal functional obstruction.

    Article  PubMed  Google Scholar 

  51. Gupta M, Ghoshal UC, Jindal S, Misra A, Nath A, Saraswat VA. Respiratory dysfunction is common in patients with achalasia and improves after pneumatic dilation. Dig Dis Sci. 2014;59(4):744–52.

    Article  CAS  PubMed  Google Scholar 

  52. Smith N. Oesophageal atresia and tracheo-oesophageal fistula. Early Hum Dev. 2014;90(12):947–50.

    Article  PubMed  Google Scholar 

  53. Shaw-Smith C. Oesophageal atresia, tracheo-oesophageal fistula, and the VACTERL association: review of genetics and epidemiology. J Med Genet. 2006;43(7):545–54.

    Article  CAS  PubMed  Google Scholar 

  54. Kovesi T, Rubin S. Long-term complications of congenital esophageal atresia and/or tracheoesophageal fistula. Chest. 2004;126(3):915–25.

    Article  PubMed  Google Scholar 

  55. Stoll C, Alembik Y, Dott B, Roth MP. Associated anomalies in cases with esophageal atresia. Am J Med Genet A. 2017;173(8):2139–57.

    Article  CAS  PubMed  Google Scholar 

  56. Roberts K, Karpelowsky J, Fitzgerald DA, Soundappan SS. Outcomes of oesophageal atresia and tracheo-oesophageal fistula repair. J Paediatr Child Health. 2016;52(7):694–8.

    Article  PubMed  Google Scholar 

  57. Teague WJ, Karpelowsky J. Surgical management of oesophageal atresia. Paediatr Respir Rev. 2016;19:10–5.

    PubMed  Google Scholar 

  58. Sadreameli SC, McGrath-Morrow SA. Respiratory care of infants and children with congenital tracheo-oesophageal fistula and oesophageal atresia. Paediatr Respir Rev. 2016;17:16–23.

    PubMed  Google Scholar 

  59. Kovesi T. Long-term respiratory complications of congenital esophageal atresia with or without tracheoesophageal fistula: an update. Dis Esophagus. 2013;26(4):413–6.

    Article  CAS  PubMed  Google Scholar 

  60. Gottrand M, Michaud L, Sfeir R, Gottrand F. Motility, digestive and nutritional problems in esophageal atresia. Paediatr Respir Rev. 2016;19:28–33.

    PubMed  Google Scholar 

  61. Lilja HE, Wester T. Outcome in neonates with esophageal atresia treated over the last 20 years. Pediatr Surg Int. 2008;24(5):531–6.

    Article  PubMed  Google Scholar 

  62. •• Krishnan U, Mousa H, Dall’Oglio L, Homaira N, Rosen R, Faure C, 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(5):550–70 This study highlights the ESPGHAN-NASPGHAN guidelines for the evaluation and treatment of symptomatic patients with EA/TEF. They present a stepwise algorithmic approach to the evaluation of children with EA/TEF who present with gastrointestinal and extra-intestinal symptoms specifically emphasizing the importance of ruling out anatomical and mucosal abnormalities followed by evaluation for esophageal and gastric dysmotility.

    Article  PubMed  Google Scholar 

  63. Chetcuti P, Phelan PD. Gastrointestinal morbidity and growth after repair of oesophageal atresia and tracheo-oesophageal fistula. Arch Dis Child. 1993;68(2):163–6.

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  64. Chetcuti P, Phelan PD, Greenwood R. Lung function abnormalities in repaired oesophageal atresia and tracheo-oesophageal fistula. Thorax. 1992;47(12):1030–4.

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  65. Banjar HH, Al-Nassar SI. Gastroesophageal reflux following repair of esophageal atresia and tracheoesophageal fistula. Saudi Med J. 2005;26(5):781–5.

    PubMed  Google Scholar 

  66. Castilloux J, Noble AJ, Faure C. Risk factors for short- and long-term morbidity in children with esophageal atresia. J Pediatr. 2010;156(5):755–60.

    Article  PubMed  Google Scholar 

  67. Bouguermouh D, Salem A. Esophageal atresia: a critical review of management at a single center in Algeria. Dis Esophagus. 2015;28(3):205–10.

    Article  CAS  PubMed  Google Scholar 

  68. 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):852–6.

    Article  CAS  PubMed  Google Scholar 

  69. Connor MJ, Springford LR, Kapetanakis VV, Giuliani S. Esophageal atresia and transitional care--step 1: a systematic review and meta-analysis of the literature to define the prevalence of chronic long-term problems. Am J Surg. 2015;209(4):747–59.

    Article  PubMed  Google Scholar 

  70. Beucher J, Wagnon J, Daniel V, Habonimana E, Fremond B, Lapostolle C, et al. Long-term evaluation of respiratory status after esophageal atresia repair. Pediatr Pulmonol. 2013;48(2):188–94.

    Article  CAS  PubMed  Google Scholar 

  71. •• Pedersen RN, Markow S, Kruse-Andersen S, Qvist N, Gerke O, Husby S, et al. Long-term pulmonary function in esophageal atresia—a case-control study. Pediatr Pulmonol. 2017;52(1):98–106 This study characterizes the long term respiratory symptoms and functional outcomes of children and adolescents. Greater than fifity percent had history of recurrent pneumonias. Significant obstructive and restrictive ventilatory abnormalities were found on pulmonary function testing when compared to healthy controls.

    Article  PubMed  Google Scholar 

  72. Legrand C, Michaud L, Salleron J, Neut D, Sfeir R, Thumerelle C, et al. Long-term outcome of children with oesophageal atresia type III. Arch Dis Child. 2012;97(9):808–11.

    Article  PubMed  Google Scholar 

  73. Dhaliwal J, Tobias V, Sugo E, Varjavandi V, Lemberg D, Day A, et al. Eosinophilic esophagitis in children with esophageal atresia. Dis Esophagus. 2014;27(4):340–7.

    Article  CAS  PubMed  Google Scholar 

  74. Mohammad AA, Wu SZ, Ibrahim O, Bena J, Rizk M, Piliang M, et al. Prevalence of atopic comorbidities in eosinophilic esophagitis: a case-control study of 449 patients. J Am Acad Dermatol. 2017;76(3):559–60.

    Article  PubMed  Google Scholar 

  75. Gomez Torrijos E, Mur Gimeno P, Martin Iglesias A, Garcia Rodriguez R, Galindo Bonilla P, El Kaddioui S, et al. Non-asthmatic eosinophilic bronchitis and idiopathic eosinophilic esophagitis. J Investig Allergol Clin Immunol. 2016;26(3):190–1.

    Article  CAS  PubMed  Google Scholar 

  76. Krupp NL, Sehra S, Slaven JE, Kaplan MH, Gupta S, Tepper RS. Increased prevalence of airway reactivity in children with eosinophilic esophagitis. Pediatr Pulmonol. 2016;51(5):478–83.

    Article  PubMed  Google Scholar 

  77. Kubik M, Thottam P, Shaffer A, Choi S. The role of the otolaryngologist in the evaluation and diagnosis of eosinophilic esophagitis. Laryngoscope. 2017;127(6):1459–64.

    Article  PubMed  Google Scholar 

  78. Hill CA, Ramakrishna J, Fracchia MS, Sternberg D, Ojha S, Infusino S, et al. Prevalence of eosinophilic esophagitis in children with refractory aerodigestive symptoms. JAMA Otolaryngol Head Neck Surg. 2013;139(9):903–6.

    Article  PubMed  Google Scholar 

  79. • Chan LJ, Tan L, Dhaliwal J, Briglia F, Clarkson C, Krishnan U. Treatment outcomes for eosinophilic esophagitis in children with esophageal atresiaFindings from this study suggest that mucosal abnormalities such as Eosinophilic Esophagitis (EoE) should be suspected in children with EA/TEF who are symptomatic. Affective treatment of EoE will decrease symptoms of reflux and dysphagia and complications such as strictures. Dis Esophagus. 2016;29(6):563–71.

    Article  CAS  PubMed  Google Scholar 

  80. •• van Wijk M, Knuppe 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):2496–505 Findings of this study suggest that abnormalities in esophageal and gastric motility play a critical role in symptomatic patients with EA/TEF and suspected gastroesophageal reflux. Abnormal gastric emptying was demonstrated in 71.4% of infants and those with abnormall esophageal motility were found to have poor esophageal clearance approximately 70.3% of the time.

    Article  PubMed  Google Scholar 

  81. Tong S, Mallitt KA, Krishnan U. Evaluation of gastroesophageal reflux by combined multichannel intraluminal impedance and pH monitoring and esophageal motility patterns in children with esophageal atresia. Eur J Pediatr Surg. 2016;26(4):322–31.

    Article  PubMed  Google Scholar 

  82. Pedersen RN, Markow S, Kruse-Andersen S, Qvist N, Hansen TP, Gerke O, et al. Esophageal atresia: gastroesophageal functional follow-up in 5–15 year old children. J Pediatr Surg. 2013;48(12):2487–95.

    Article  PubMed  Google Scholar 

  83. Lemoine C, Aspirot A, Le Henaff G, Piloquet H, Levesque D, Faure C. Characterization of esophageal motility following esophageal atresia repair using high-resolution esophageal manometry. J Pediatr Gastroenterol Nutr. 2013;56(6):609–14.

    Article  PubMed  Google Scholar 

  84. Faure C, Righini Grunder F. Dysmotility in esophageal atresia: pathophysiology, characterization, and treatment. Front Pediatr. 2017;5:130.

    Article  PubMed  PubMed Central  Google Scholar 

  85. •• Duncan DR, Amirault J, Johnston N, Mitchell P, Larson K, Rosen RL. Gastroesophageal reflux burden, even in children that aspirate, does not increase pediatric hospitalization. J Pediatr Gastroenterol Nutr. 2016;63(2):210–7 This study suggests that there is a lack of clear association between the results of reflux testing and pulmonary outcomes. The degree of reflux burden, as demonstrated using pH-impedance, did not change the rate of hospitalization of children regardless of their aspiration risk.

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  86. Rintala RJ. Fundoplication in patients with esophageal atresia: patient selection, indications, and outcomes. Front Pediatr. 2017;5:109.

    Article  PubMed  PubMed Central  Google Scholar 

  87. Mahoney L, Rosen R. Feeding problems and their underlying mechanisms in the esophageal atresia-tracheoesophageal fistula patient. Front Pediatr. 2017;5:127.

    Article  PubMed  PubMed Central  Google Scholar 

  88. •• Duncan DR, Mitchell PD, Larson K, Rosen RL. Presenting signs and symptoms do not predict aspiration risk in children. J Pediatr. 2018;201:141–6 Findings of this study demonstrate the poor correlation between presenting symptoms of aspiration in children being evaluated for oropharyngeal dysphagia and the findings of aspiratio on videofluoroscopic study (VFSS). The authors also demonstrate that the lcinical feeding evaluation is inferior to VFSS in detecting aspiration and if suspected VFSS is warranted.

    Article  PubMed  PubMed Central  Google Scholar 

  89. Mortellaro VE, Pettiford JN, St Peter SD, Fraser JD, Ho B, Wei J. Incidence, diagnosis, and outcomes of vocal fold immobility after esophageal atresia (EA) and/or tracheoesophageal fistula (TEF) repair. Eur J Pediatr Surg. 2011;21(6):386–8.

    Article  CAS  PubMed  Google Scholar 

  90. Hseu A, Recko T, Jennings R, Nuss R. Upper airway anomalies in congenital tracheoesophageal fistula and esophageal atresia patients. Ann Otol Rhinol Laryngol. 2015;124(10):808–13.

    Article  PubMed  Google Scholar 

  91. Hathorn KE, Chan WW, Lo WK. Role of gastroesophageal reflux disease in lung transplantation. World J Transplant. 2017;7(2):103–16.

    Article  PubMed  PubMed Central  Google Scholar 

  92. •• Jamie Dy F, Freiberger D, Liu E, Boyer D, Visner G, Rosen R. Impact of gastroesophageal reflux and delayed gastric emptying on pediatric lung transplant outcomes. J Heart Lung Transplant. 2017;36(8):854–61 This study demonstrates the role of gastric dysmotility, specifically delayed gastric emptying, in children with allograft rejection following lung trasplantation. Despite half of the children having pathological reflux on pH-impedance, there was no association between reflux burden and allograft rejection.

    Article  PubMed  Google Scholar 

  93. Patti MG, Albanese CT, Holcomb GW 3rd, Molena D, Fisichella PM, Perretta S, et al. Laparoscopic Heller myotomy and Dor fundoplication for esophageal achalasia in children. J Pediatr Surg. 2001;36(8):1248–51.

    Article  CAS  PubMed  Google Scholar 

  94. Benden C, Aurora P, Curry J, Whitmore P, Priestley L, Elliott MJ. High prevalence of gastroesophageal reflux in children after lung transplantation. Pediatr Pulmonol. 2005;40(1):68–71.

    Article  PubMed  Google Scholar 

  95. Hooft N, Smith M, Huang J, Bremner R, Walia R. Gastroparesis is common after lung transplantation and may be ameliorated by botulinum toxin-A injection of the pylorus. J Heart Lung Transplant. 2014;33(12):1314–6.

    Article  PubMed  Google Scholar 

  96. Raviv Y, D’Ovidio F, Pierre A, Chaparro C, Freeman M, Keshavjee S, et al. Prevalence of gastroparesis before and after lung transplantation and its association with lung allograft outcomes. Clin Transpl. 2012;26(1):133–42.

    Article  Google Scholar 

  97. Ciriza de Los Rios C, Canga Rodriguez-Valcarcel F, de Pablo Gafas A, Castel de Lucas I, Lora Pablos D, Castellano Tortajada G. Esophageal motor disorders are frequent during pre and post lung transplantation. Can they influence lung rejection? Rev Esp Enferm Dig. 2018;110(6):344–51.

    CAS  PubMed  Google Scholar 

  98. •• Tangaroonsanti A, Lee AS, Crowell MD, Vela MF, Jones DR, Erasmus D, et al. Impaired esophageal motility and clearance post-lung transplant: risk for chronic allograft failure. Clin Transl Gastroenterol. 2017;8(6):e102 This study highlights the role of esophageal dysmotility in development and progression of chronic lung allograft dysfunction (CLAD). HRM with impedance demonstrated risk factors for CLAD which included esophagogastric junction outflow obstruction, incomplete bolus clearance, and proximal reflux.

    Article  PubMed  PubMed Central  Google Scholar 

  99. Tangaroonsanti A, Vela MF, Crowell MD, DeVault KR, Houghton LA. Esophageal dysmotility according to Chicago classification v3.0 vs v2.0: implications for association with reflux, bolus clearance, and allograft failure post-lung transplantation. Neurogastroenterol Motil. 2018;30(6):e13296.

    Article  CAS  PubMed  Google Scholar 

  100. Mertens V, Blondeau K, Pauwels A, Farre R, Vanaudenaerde B, Vos R, et al. Azithromycin reduces gastroesophageal reflux and aspiration in lung transplant recipients. Dig Dis Sci. 2009;54(5):972–9.

    Article  CAS  PubMed  Google Scholar 

  101. Martini G, Foeldvari I, Russo R, Cuttica R, Eberhard A, Ravelli A, et al. Systemic sclerosis in childhood: clinical and immunologic features of 153 patients in an international database. Arthritis Rheum. 2006;54(12):3971–8.

    Article  CAS  PubMed  Google Scholar 

  102. Guariso G, Conte S, Galeazzi F, Vettorato MG, Martini G, Zulian F. Esophageal involvement in juvenile localized scleroderma: a pilot study. Clin Exp Rheumatol. 2007;25(5):786–9.

    CAS  PubMed  Google Scholar 

  103. Solomon JJ, Olson AL, Fischer A, Bull T, Brown KK, Raghu G. Scleroderma lung disease. Eur Respir Rev. 2013;22(127):6–19.

    Article  PubMed  PubMed Central  Google Scholar 

  104. Christmann RB, Wells AU, Capelozzi VL, Silver RM. Gastroesophageal reflux incites interstitial lung disease in systemic sclerosis: clinical, radiologic, histopathologic, and treatment evidence. Semin Arthritis Rheum. 2010;40(3):241–9.

    Article  PubMed  Google Scholar 

  105. Savarino E, Bazzica M, Zentilin P, Pohl D, Parodi A, Cittadini G, et al. Gastroesophageal reflux and pulmonary fibrosis in scleroderma: a study using pH-impedance monitoring. Am J Respir Crit Care Med. 2009;179(5):408–13.

    Article  PubMed  Google Scholar 

  106. Raja J, Ng CT, Sujau I, Chin KF, Sockalingam S. High-resolution oesophageal manometry and 24-h impedance-pH study in systemic sclerosis patients: association with clinical features, symptoms and severity. Clin Exp Rheumatol. 2016;34(100 Suppl 5):115–21.

    PubMed  Google Scholar 

  107. Kimmel JN, Carlson DA, Hinchcliff M, Carns MA, Aren KA, Lee J, et al. The association between systemic sclerosis disease manifestations and esophageal high-resolution manometry parameters. Neurogastroenterol Motil. 2016;28(8):1157–65.

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  108. Marie I, Dominique S, Levesque H, Ducrotte P, Denis P, Hellot MF, et al. Esophageal involvement and pulmonary manifestations in systemic sclerosis. Arthritis Rheum. 2001;45(4):346–54.

    Article  CAS  PubMed  Google Scholar 

  109. Myers JC, Nguyen NQ, Jamieson GG, Van’t Hek JE, Ching K, Holloway RH, et al. Susceptibility to dysphagia after fundoplication revealed by novel automated impedance manometry analysis. Neurogastroenterol Motil. 2012;24(9):812–e393.

    Article  CAS  PubMed  Google Scholar 

  110. Loots C, van Herwaarden MY, Benninga MA, VanderZee DC, van Wijk MP, Omari TI. Gastroesophageal reflux, esophageal function, gastric emptying, and the relationship to dysphagia before and after anti-reflux surgery in children. J Pediatr. 2013;162(3):566–73 e2.

    Article  PubMed  Google Scholar 

  111. Hoshino M, Srinivasan A, Mittal SK. High-resolution manometry patterns of lower esophageal sphincter complex in symptomatic post-fundoplication patients. J Gastrointest Surg. 2012;16(4):705–14.

    Article  PubMed  Google Scholar 

  112. Rosen R, Hart K, Warlaumont M. Incidence of gastroesophageal reflux during transpyloric feeds. J Pediatr Gastroenterol Nutr. 2011;52(5):532–5.

    Article  PubMed  Google Scholar 

  113. Arnold BN, Dunst CM, Gill AB, Goers TA, Swanstrom LL. Postoperative impedance-pH testing is unreliable after Nissen fundoplication with or without giant hiatal hernia repair. J Gastrointest Surg. 2011;15(9):1506–12.

    Article  PubMed  Google Scholar 

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Correspondence to Rachel Rosen MD, MPH.

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Lusine Ambartsumyan, Samuel Nurko, and Rachel Rosen declare no conflict of interest.

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Ambartsumyan, L., Nurko, S. & Rosen, R. Gastrointestinal Dysmotility and the Implications for Respiratory Disease. Curr Treat Options Peds 5, 197–214 (2019). https://doi.org/10.1007/s40746-019-00158-3

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Keywords

  • Dysmotility
  • Fundoplication
  • High-resolution esophageal manometry
  • Aerodigestive
  • Gastroesophageal reflux
  • Impedance