Four Cancer Cases After Esophageal Atresia Repair: Time to Start Screening the Upper Gastrointestinal Tract

Overview

Journal World J Gastroenterol

Publisher Baishideng Publishing Group

Specialty Gastroenterology

Date 2018 Mar 14

PMID 29531469

Citations 11

Authors

Floor Wt Vergouwe

Madeleine Gottrand

Bas Pl Wijnhoven

Hanneke IJsselstijn

Guillaume Piessen

Marco J Bruno

Rene Mh Wijnen

Manon Cw Spaander

Affiliations

Soon will be listed here.

Abstract

Esophageal atresia (EA) is one of the most common congenital digestive malformations and requires surgical correction early in life. Dedicated centers have reported survival rates up to 95%. The most frequent comorbidities after EA repair are dysphagia (72%) and gastroesophageal reflux (GER) (67%). Chronic GER after EA repair might lead to mucosal damage, esophageal stricturing, Barrett's esophagus and eventually esophageal adenocarcinoma. Several long-term follow-up studies found an increased risk of Barrett's esophagus and esophageal carcinoma in EA patients, both at a relatively young age. Given these findings, the recent ESPGHAN-NASPGHAN guideline recommends routine endoscopy in adults born with EA. We report a series of four EA patients who developed a carcinoma of the gastrointestinal tract: three esophageal carcinoma and one colorectal carcinoma in a colonic interposition. These cases emphasize the importance of lifelong screening of the upper gastrointestinal tract in EA patients.

Citing Articles

Current practice and barriers for transition of care (TOC) in pediatric surgery: perspectives of adult surgeons from different subspecialties.

Sia W, Tay J, Lee T, Nah S, A Nallusamy M, Mahendran H Pediatr Surg Int. 2025; 41(1):76.

PMID: 39873765 PMC: 11774974. DOI: 10.1007/s00383-025-05969-0.

Long-term outcome of oesophageal atresia in adolescence (TransEAsome): a national French cohort study protocol.

Leroy M, Aumar M, Duhamel M, Dauchet L, Figeac M, Gaillard S BMJ Open. 2025; 15(1):e086303.

PMID: 39800411 PMC: 11749844. DOI: 10.1136/bmjopen-2024-086303.

Follow-up and transition practices in esophageal atresia: a review of European Reference Network on rare Inherited and Congenital Anomalies (ERNICA) centres and affiliates.

Durkin N, Pellegrini M, Gorter R, Slater G, Cross K, Ure B Pediatr Surg Int. 2024; 40(1):300.

PMID: 39521743 PMC: 11550284. DOI: 10.1007/s00383-024-05865-z.

The International Network on Oesophageal Atresia (INoEA) consensus guidelines on the transition of patients with oesophageal atresia-tracheoesophageal fistula.

Krishnan U, Dumont M, Slater H, Gold B, Seguy D, Bouin M Nat Rev Gastroenterol Hepatol. 2023; 20(11):735-755.

PMID: 37286639 DOI: 10.1038/s41575-023-00789-w.

Systematic review of long term follow-up and transitional care in adolescents and adults with esophageal atresia - why is transitional care mandatory?.

Brooks G, Gazzaneo M, Bertozzi M, Riccipetitoni G, Raffaele A Eur J Pediatr. 2023; 182(5):2057-2066.

PMID: 36905437 PMC: 10175361. DOI: 10.1007/s00431-023-04893-6.

References

Vergouwe F, IJsselstijn H, Wijnen R, Bruno M, Spaander M . Screening and Surveillance in Esophageal Atresia Patients: Current Knowledge and Future Perspectives. Eur J Pediatr Surg. 2015; 25(4):345-52. DOI: 10.1055/s-0035-1559817. View

Pajecki D, Zilberstein B, Cecconello I, Dos Santos M, Yagi O, Gama-Rodrigues J . Larger amounts of nitrite and nitrate-reducing bacteria in megaesophagus of Chagas' disease than in controls. J Gastrointest Surg. 2007; 11(2):199-203. DOI: 10.1007/s11605-006-0066-y. View

Deurloo J, van Lanschot J, Drillenburg P, Aronson D . Esophageal squamous cell carcinoma 38 years after primary repair of esophageal atresia. J Pediatr Surg. 2001; 36(4):629-30. DOI: 10.1053/jpsu.2001.22305. View

Taylor A, Breen K, Auldist A, Catto-Smith A, Clarnette T, Crameri J . 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):702-6. DOI: 10.1016/j.cgh.2007.03.012. View

Pultrum B, Bijleveld C, de Langen Z, Plukker J . Development of an adenocarcinoma of the esophagus 22 years after primary repair of a congenital atresia. J Pediatr Surg. 2005; 40(12):e1-4. DOI: 10.1016/j.jpedsurg.2005.08.042. View

Sandler R, Nyren O, Ekbom A, Eisen G, Yuen J, Josefsson S . The risk of esophageal cancer in patients with achalasia. A population-based study. JAMA. 1995; 274(17):1359-62. View

Solomon B, Baker L, Bear K, Cunningham B, Giampietro P, Hadigan C . An approach to the identification of anomalies and etiologies in neonates with identified or suspected VACTERL (vertebral defects, anal atresia, tracheo-esophageal fistula with esophageal atresia, cardiac anomalies, renal anomalies, and limb.... J Pediatr. 2013; 164(3):451-7.e1. PMC: 3943871. DOI: 10.1016/j.jpeds.2013.10.086. View

Kamangar F, Chow W, Abnet C, Dawsey S . Environmental causes of esophageal cancer. Gastroenterol Clin North Am. 2009; 38(1):27-57, vii. PMC: 2685172. DOI: 10.1016/j.gtc.2009.01.004. View

Sulkowski J, Cooper J, Lopez J, Jadcherla Y, Cuenot A, Mattei P . Morbidity and mortality in patients with esophageal atresia. Surgery. 2014; 156(2):483-91. PMC: 4099299. DOI: 10.1016/j.surg.2014.03.016. View

10.

Alfaro L, Bermas H, Fenoglio M, Parker R, Janik J . Are patients who have had a tracheoesophageal fistula repair during infancy at risk for esophageal adenocarcinoma during adulthood?. J Pediatr Surg. 2005; 40(4):719-20. DOI: 10.1016/j.jpedsurg.2005.01.001. View

11.

Krishnan U, Mousa H, DallOglio L, Homaira N, Rosen R, Faure C . 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):550-570. DOI: 10.1097/MPG.0000000000001401. View

12.

Chapuy L, Pomerleau M, Faure C . Topical mitomycin-C application in recurrent esophageal strictures after surgical repair of esophageal atresia. J Pediatr Gastroenterol Nutr. 2014; 59(5):608-11. DOI: 10.1097/MPG.0000000000000352. View

13.

Adzick N, Fisher J, Winter H, Sandler R, HENDREN W . Esophageal adenocarcinoma 20 years after esophageal atresia repair. J Pediatr Surg. 1989; 24(8):741-4. DOI: 10.1016/s0022-3468(89)80528-7. View

14.

Sistonen S, Koivusalo A, Lindahl H, Pukkala E, Rintala R, Pakarinen M . Cancer after repair of esophageal atresia: population-based long-term follow-up. J Pediatr Surg. 2008; 43(4):602-5. DOI: 10.1016/j.jpedsurg.2007.12.002. View

15.

Pedersen R, Calzolari E, Husby S, Garne E . Oesophageal atresia: prevalence, prenatal diagnosis and associated anomalies in 23 European regions. Arch Dis Child. 2012; 97(3):227-32. DOI: 10.1136/archdischild-2011-300597. View

16.

Torre L, Bray F, Siegel R, Ferlay J, Lortet-Tieulent J, Jemal A . Global cancer statistics, 2012. CA Cancer J Clin. 2015; 65(2):87-108. DOI: 10.3322/caac.21262. View

17.

Wang B, Tashiro J, Allan B, Sola J, Parikh P, Hogan A . A nationwide analysis of clinical outcomes among newborns with esophageal atresia and tracheoesophageal fistulas in the United States. J Surg Res. 2014; 190(2):604-12. DOI: 10.1016/j.jss.2014.04.033. View

18.

Jayasekera C, Desmond P, Holmes J, Kitson M, Taylor A . Cluster of 4 cases of esophageal squamous cell cancer developing in adults with surgically corrected esophageal atresia--time for screening to start. J Pediatr Surg. 2012; 47(4):646-51. DOI: 10.1016/j.jpedsurg.2011.09.065. View

19.

Sistonen S, Koivusalo A, Nieminen U, Lindahl H, Lohi J, Kero M . Esophageal morbidity and function in adults with repaired esophageal atresia with tracheoesophageal fistula: a population-based long-term follow-up. Ann Surg. 2010; 251(6):1167-73. DOI: 10.1097/SLA.0b013e3181c9b613. View

20.

Zhang S, Guo L, Chen Q, Zhang M, Liu S, Quan P . The association between human papillomavirus 16 and esophageal cancer in Chinese population: a meta-analysis. BMC Cancer. 2015; 15:1096. PMC: 4352244. DOI: 10.1186/s12885-015-1096-1. View