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Revista Española de Enfermedades Digestivas

versión impresa ISSN 1130-0108

Rev. esp. enferm. dig. vol.110 no.7 Madrid jul. 2018

http://dx.doi.org/10.17235/reed.2018.5577/2018 

SCIENTIFIC LETTERS

Intramural esophageal dissection. A rare occurrence in pediatric eosinophilic esophagitis

Susana Castanhinha1  , João Lopes2  , Ana-Isabel Lopes1  3 

1Gastroenterology Unit. Department of Pediatrics. Santa Maria University Hospital. Lisbon Academical Medical Centre. Lisbon, Portugal

2Department of Gastroenterology. Santa Maria University Hospital. Lisbon Academical Medical Centre. Lisbon, Portugal

3Medical Faculty. University of Lisbon. Lisbon Academic Medical Centre. Lisbon, Portugal

Key words: Esophageal dissection; Eosinophilic esophagitis; Endoscopy; Children; Pediatrics

Dear Editor,

Eosinophilic esophagitis (EoE) is a chronic immune-mediated esophageal disease, with a rising incidence in childhood. The diagnosis is confirmed by esophageal mucosa eosinophil-predominant inflammation. Rare complications, such as intramural esophageal dissection (IED) due to progressive transmural inflammatory and fibrotic process, may occur in young adults 1,2,3,4. While severe cases can require immediate surgical intervention, conservative management may be also effective, as recently reported by Ibáñez-Sanz et al. 3, decreasing the risk of surgery-related complications.

Case report

We report a pediatric case of EoE-associated esophageal dissection with a favorable outcome after a conservative management. An 11-year-old boy presented to the Emergency Department with retrosternal pain, odynophagia/dysphagia and sialorrhea of a one-week duration. He had an atopic background (allergic asthma and rhinitis), allergic rhinitis and a previous EoE diagnosis at the age of eight years (food impaction episode). He did not have any food allergies. Despite the voluntary interruption of swallowed fluticasone, he had remained asymptomatic.

On admission, the patient was febrile and a laboratory evaluation identified leukocytosis and elevated C-reactive protein. A chest computed tomography (CT) scan excluded mediastinitis, and endoscopy showed extensive ulceration and dissection of the esophageal posterior wall that extended to the muscular layer (Fig. 1). The symptoms resolved over the next two weeks with a conservative treatment of enteral nutrition, proton pump inhibitors (PPI), prednisolone, broad-spectrum antibiotic therapy. The child was discharged on day 35. Follow-up endoscopy revealed mucosal healing and no strictures. Twelve months later, the patient remained asymptomatic and compliant to maintenance treatment with swallowed fluticasone.

Fig. 1 Upper gastrointestinal endoscopy showing extensive ulceration and dissection of the esophageal posterior wall that reaches the muscular layer (~30% of the circumferential diameter). 

Discussion

This case illustrates a quite rare and serious complication of pediatric EoE. To our knowledge this is the youngest reported case of IED in this setting, with favorable outcome and a conservative management that allowed an esophageal wall restitution. Furthermore, it provides further insights into its natural history 1,2,3. The role of conservative management, particularly in this age group, despite an extensive dissection, and the treatment compliance of EoE are noteworthy, considering the progressive course of a transmural chronic inflammatory process.

References

1. Liguori G, Cortale M, Cimino F, et al. Circumferential mucosal dissection and esophageal perforation in a patient with eosinophilic esophagitis. World J Gastroenterol 2008;14:803-4. DOI: 10.3748/wjg.14.803. PMID: 18205276. [ Links ]

2. Sgrò A, Betalli P, Battaglia G, et al. An unusual complication of eosinophilic esophagitis in an adolescent: intramural esophageal dissection. Endoscopy 2012;44:E419-20. DOI: 10.1055/s-0032-1325777 [ Links ]

3. Ibáñez-Sanz G, Rodríguez-Alonso L, Romero NM. Spontaneous intramural esophageal dissection: an unusual onset of eosinophilic esophagitis. Rev Esp Enferm Dig 2016;108(3):156-7. [ Links ]

4. Lucendo AJ, Arias A, De Rezende LC, et al. Subepithelial collagen deposition, profibrogenic cytokine gene expression, and changes after prolonged fluticasone propionate treatment in adult eosinophilic esophagitis: a prospective study. J Allergy Clin Immunol 2011;128:1037-46. DOI: 10.1016/j.jaci.2011.08.007 [ Links ]

Author's contribution

Susana Castanhinha and Ana Isabel Lopes have equally contributed as authors.