Advertisement
Rapid Communication| Volume 47, ISSUE 6, e9-e13, June 2012

Eosinophilic esophagitis after esophageal atresia: is there an association? Case presentation and literature review

      Abstract

      Eosinophilic esophagitis (EoE) is a relatively new condition resulting in dysphagia or symptoms resembling gastroesophageal reflux disease, symptoms that also are common in patients with a history of esophageal atresia. We present 2 patients with persistent dysphagia after repair of esophageal atresia that was caused by EoE. Although the exact etiology and pathogenesis of EoE remain unclear, it is now generally accepted that it is the result of a T-helper cell 2–type immune response with a crucial role for the eosinophil-specific chemotaxis factor eotaxin 3 and eosinophils. Because there are genetic similarities between esophageal atresia and EoE, we speculate that patients with esophageal atresia are at increased risk for developing EoE.

      Key words

      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:

      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

      References

        • Straumann A.
        • Simon H.U.
        Eosinophilic esophagitis: escalating epidemiology?.
        J Allergy Clin Immunol. 2005; 115: 418-419
        • Bonis P.A.L.
        Putting the puzzle together: epidemiological and clinical clues in the aetiology of eosinophilic esophagitis.
        Immunol Allergy Clin North Am. 2009; 29: 41-52
        • Noel R.J.
        • Rothenberg M.E.
        Eosinophilic esophagitis.
        Curr Opin Pediatr. 2005; 17: 690-694
        • Blanchard C.
        • Wang N.
        • Stringer K.F.
        • et al.
        Eotaxin-3 and a uniquely conserved gene-expression profile in eosinophilic esophagitis.
        J Clin Invest. 2006; 116: 536-547
        • Furuta G.T.
        • Liacouras C.A.
        • Collins M.H.
        • et al.
        Eosinophilic esophagitis in children and adults: a systematic review and consensus recommendations for diagnosis and treatment.
        Gastroenterol. 2007; 133: 1342-1363
        • Atkins D.
        • Kramer R.
        • Capocelli K.
        • et al.
        Eosinophilic esophagitis: the newest esophageal inflammatory disease.
        Nat Rev Gastroenterol Hepatol. 2009; 6: 267-278
        • Torfs C.P.
        • Curry C.J.
        • Bateson T.F.
        Population-based study of tracheo-esophageal fistula and esophageal atresia.
        Teratology. 1995; 52: 220-232
        • Deurloo J.A.
        • Ekkelkamp S.
        • Bartelsman J.F.W.M.
        • et al.
        Gastro-esophageal reflux: prevalence in adults older than 28 years after correction of esophageal atresia.
        Ann Surg. 2003; 238: 686-689
        • Deurloo J.A.
        • Klinkenberg E.C.
        • Ekkelkamp S.
        • et al.
        Adults with corrected esophageal atresia: are complaints predictive of esophageal function and/or quality of life?.
        Pediatr Surg Int. 2008; 24: 537-541
        • Markowitz J.E.
        • Spergel J.M.
        • Ruchelli E.
        • et al.
        Elemental diet is an effective treatment for eosinophilic esophagitis in children and adolescents.
        Am J Gastroenterol. 2003; 98: 777-782
        • Rafiee P.
        • Therriot M.E.
        • Nelson V.M.
        • et al.
        Human esophageal micro vascular endothelial cells respond to acidic stress by PI3K/AKT and p38 MAPK-regulated induction of Hsp 70 and Hsp27.
        Am J Physiol Cell Physiol. 2006; 291: C931-C945
        • Sayej W.N.
        • Patel R.
        • Baker R.D.
        • et al.
        Treatment with high dose proton pump inhibitors helps distinguish eosinophilic esophagitis from noneosinophilic esophagitis.
        J Pediatr Gastroenterol Nutr. 2009; 49: 393-399
        • Spechler S.J.
        • Genta R.M.
        • Souza R.F.
        Thoughts on the complex relationship between gastroesophageal reflux disease and eosinophilic esophagitis.
        Am J Gastroenterol. 2007; 102: 1301-1306
        • Oliveira C.
        • Zamakhshary M.
        • Marcon P.
        • et al.
        Eosinophilic esophagitis and intermediate esophagitis after tracheoesophageal fistula repair: a case series.
        J Pediatr Surg. 2008; 43: 810-814
        • Batres L.A.
        • Liacouras C.
        • Schnaufer L.
        • et al.
        Eosinophilic esophagitis associated with anastomotic strictures after esophageal atresia repair.
        J Pediatr Gastroenterol Nutr. 2002; 35: 224-226
        • Remedios M.
        • Campbell C.
        • Jones D.M.
        • et al.
        Eosinophilic esophagitis in adults: clinical, endoscopic, histological findings and response to treatment with fluticasone propionate.
        Gastrointest Endosc. 2006; 63: 3-12
        • Putnam P.E.
        Eosinophilic esophagitis in children: clinical manifestations.
        Gastroenterol Clin North Am. 2008; 37: 369-381
        • Sant'Anna A.M.
        • Rolland S.
        • Fournet J.C.
        • et al.
        Eosinophilic esophagitis in children: symptoms, histology and pH probe results.
        J Pediatr Gastroenterol Nutr. 2004; 39: 373-377
        • Steiner S.J.
        • Kernek K.M.
        • Fitzgerald J.F.
        Severity of basal cell hyperplasia differs in reflux versus eosinophilic esophagitis.
        J Pediatr Gastroenterol Nutr. 2006; 42: 506-509
        • Tobey N.A.
        • Carson J.L.
        • Alkiek R.A.
        • et al.
        Dilated intercellular spaces: a morphological feature of acid-reflux damaged human esophageal epithelium.
        Gastroenterology. 1996; 111: 1200-1205
        • El-Gohary Y.
        • Gittes G.K.
        • Tovar J.A.
        Congenital anomalies of the esophagus.
        Semin Pediatr Surg. 2010; 19: 186-193
        • de Jong E.M.
        • Felix J.F.
        • de Klein A.
        • et al.
        Aetiology of esophageal atresia and tracheooesophageal fistula: “mind the gap..
        Cur Gastroenterol Rep. 2010; 12: 215-222
        • Felix J.F.
        • van Dooren M.F.
        • Klaassens M.
        • et al.
        Environmental factors in the aetiology of esophageal atresia and congenital diaphragmatic hernia: results of a case-control study.
        Birth Defects Res A Clin Mol Teratol. 2008; 82: 98-105
        • Ramalho-Santos M.
        • Melton D.A.
        • McMahon A.P.
        Hedgehog signals regulate multiple aspects of gastrointestinal development.
        Development. 2000; 127: 2763-2772
        • Shaw-Smith C.
        Genetic factors in esophageal atresia, tracheo-esophageal fistula and the Vacterl association: roles for FOXF1 And the 16q24.1 FOX transcription factor gene cluster, and review of the literature.
        Eur J Med Genet. 2010; 53: 6-13
        • Stankiewicz P.
        • Sen P.
        • Bhatt S.S.
        • et al.
        Genomic and genetic deletions of the FOX gene cluster on 16q24.1 and inactivating mutations of FOXF1 cause alveolar capillary dysplasia and other malformations.
        Am J Hum Genet. 2009; 84: 780-791
        • Madison B.B.
        • McKenna L.B.
        • Dolson D.
        • et al.
        FoxF1 and FoxL1 link hedgehog signalling and the control of epithelial proliferation in the developing stomach and intestine.
        J Biol Chem. 2009; 284: 5936-5944
        • Costa R.H.
        • Kalinichenko V.V.
        • Lim L.
        Transcription factors in mouse lung development and function.
        Am J Physiol Cell Mol Physiol. 2001; 280: L823-L838
        • Motoyama J.
        • Lui J.
        • MO R.
        • et al.
        Essential function of GLI 2 and GLI 3 in the formation of lung, trachea and oesophagus.
        Nat Genet. 1998; 20: 54-57
        • Lim L.
        • Kalinichenko V.V.
        • Whitsett J.A.
        • et al.
        Fusion of lung lobes and vessels in mouse embryos heterozygous for the forkhead box f1 targeted allele.
        Am J Physiol Lung Cell Mol Physiol. 2002; 282: L1012-L1022
        • Mahlapuu M.
        • Enerback S.
        • Carlsson P.
        Haploinsufficiency of the forkhead gene Fox f1, a target for sonic hedgehog signalling, causes lung and foregut malformations.
        Development. 2001; 128: 2397-2406
        • Whelan J.
        Selectin synthesis and inflammation.
        Trends Biochem Sci. 1996; 21: 65-69