Intramural dissection of the esophagus is a rare complication of eosinophilic esophagitis

Eosinophilic esophagitis (EoE) is characterized by endoscopic signs and detection of more than 15 eosinophils per high-power field of view in biopsies of the esophageal mucosa. Due to deep chronic eosinophilic inflammation, it can cause complications: esophageal stenosis, rarely perforation. Intramural esophageal dissection (IED) is a previously undescribed complication of EoE in children. We present a case of IED in a 15-year-old boy, which clinically manifested itself as sudden epigastric pain radiating to the back and nausea against the background of acute respiratory viral infection. According to the endoscopy, CT and X-ray contrast study, a double-barreled esophagus with two cavities was found - the esophagus itself and a blind «pocket», histologically in which granulation tissue and active inflammation with a large number of neutrophils were found. The initial histological examination did not reveal significant eosinophilic infiltration in the esophagus. The boy received a course of esomeprazole, was re-examined after 6 months. The radiographic picture was the same, and endoscopically characteristic signs of EoE were detected: longitudinal grooves and erosions, transverse rings, narrowing of the esophageal lumen. In the middle third, the picture of a double-barreled esophagus was still preserved, but without signs of acute detachment, but with a dense septum between the two cavities of the esophagus. Histologically, the infiltrate contained more than 50 eosinophils in the field of view. The diagnosis was: eosinophilic esophagitis complicated by RTI. A strict hypoallergenic diet was prescribed, excluding obligate allergens, budesonide 1 mg 2 times a day in the form of a gel. Endoscopic dissection of the esophageal septum was performed, its patency was restored. After 2 months, against the background of ongoing drug therapy and diet, the boy was examined again. He had no complaints, radiological patency of the esophagus was not impaired. Endoscopically, the esophagus is freely passable, there is only a small fold at the site of the operation, longitudinal grooves and small erosions in the lower third remain. Histologically, the number of eosinophils has significantly decreased. Therapy was continued. This clinical case demonstrates the possibility of developing RDI as a complication of Eo E. The course of EoE can be asymptomatic, and RDI is the first manifestation of the disease. The endoscopic picture of a double-barreled esophagus is a consequence of dissection. The combination of a hypoallergenic diet, topical steroids and endoscopic septectomy is effective in the treatment of EoE with RDI. Eosinophilic esophagitis (EoE) is characterized by endoscopic signs and detection of more than 15 eosinophils per high-power field of view in biopsies of the esophageal mucosa. Due to deep chronic eosinophilic inflammation, it can cause complications: esophageal stenosis, rarely perforation. Intramural esophageal dissection (IED) is a previously undescribed complication of EoE in children. We present a case of IED in a 15-year-old boy, which clinically manifested itself as sudden epigastric pain radiating to the back and nausea against the background of acute respiratory viral infection. According to the endoscopy, CT and X-ray contrast study, a double-barreled esophagus with two cavities was found - the esophagus itself and a blind «pocket», histologically in which granulation tissue and active inflammation with a large number of neutrophils were found. The initial histological examination did not reveal significant eosinophilic infiltration in the esophagus. The boy received a course of esomeprazole, was re-examined after 6 months. The radiographic picture was the same, and endoscopically characteristic signs of EoE were detected: longitudinal grooves and erosions, transverse rings, narrowing of the esophageal lumen. In the middle third, the picture of a double-barreled esophagus was still preserved, but without signs of acute detachment, but with a dense septum between the two cavities of the esophagus. Histologically, the infiltrate contained more than 50 eosinophils in the field of view. The diagnosis was: eosinophilic esophagitis complicated by RTI. A strict hypoallergenic diet was prescribed, excluding obligate allergens, budesonide 1 mg 2 times a day in the form of a gel. Endoscopic dissection of the esophageal septum was performed, its patency was restored. After 2 months, against the background of ongoing drug therapy and diet, the boy was examined again. He had no complaints, radiological patency of the esophagus was not impaired. Endoscopically, the esophagus is freely passable, there is only a small fold at the site of the operation, longitudinal grooves and small erosions in the lower third remain. Histologically, the number of eosinophils has significantly decreased. Therapy was continued. This clinical case demonstrates the possibility of developing RDI as a complication of Eo E. The course of EoE can be asymptomatic, and RDI is the first manifestation of the disease. The endoscopic picture of a double-barreled esophagus is a consequence of dissection. The combination of a hypoallergenic diet, topical steroids and endoscopic septectomy is effective in the treatment of EoE with RDI.

eosinophilic esophagitis, intramural esophageal dissection, double-barreled esophagus

1. Hruz P., Straumann A., Bussmann C., Heer P., Simon H. U., Zwahlen M., Beglinger C., Schoepfer A. M.; Swiss EoE study group. Escalating incidence of eosinophilic esophagitis: a 20-year prospective, population-based study in Olten County, Switzerland. J Allergy Clin Immunol. 2011 Dec;128(6):1349-1350.e5. doi: 10.1016/j.jaci.2011.09.013.

2. Al-Horani R. A., Chiles R. First Therapeutic Approval for Eosinophilic Esophagitis. Gastroenterol Insights. 2022 Sep;13(3):238-244. doi: 10.3390/gastroent13030024.

3. Dellon E. S., Gonsalves N., Hirano I., Furuta G. T., Liacouras C. A., Katzka D. A.; American College of Gastroenterology. ACG clinical guideline: Evidenced based approach to the diagnosis and management of esophageal eosinophilia and eosinophilic esophagitis (EoE). Am J Gastroenterol. 2013 May;108(5):679-92; quiz 693. doi: 10.1038/ajg.2013.71.

4. Liacouras C. A., Furuta G. T., Hirano I. et al. Eosinophilic esophagitis: updated consensus recommendations for children and adults. J Allergy Clin Immunol. 2011 Jul;128(1):3-20.e6; quiz 21-2. doi: 10.1016/j.jaci.2011.02.040.

5. Furuta G. T., Katzka D. A. Eosinophilic Esophagitis. N Engl J Med. 2015 Oct 22;373(17):1640-8. doi: 10.1056/NEJMra1502863.

6. Marfani G. M., Kashikar S. V., Singhania S. Double barrel oesophagus - a case report. J. Clin Diagn. Research. 2018; v.12(8): TD01-TD02.

7. Sethi S. S., Sachdeva S., Singh A. K. Double-Barrel Esophagus: Deglutition at the Crossroads. Clin Gastroenterol Hepatol. 2022 Jan;20(1): e1. doi: 10.1016/j.cgh.2020.07.045.

8. Hsu C. C., Changchien C. S. Endoscopic and radiological features of intramural esophageal dissection. Endoscopy. 2001 Apr;33(4):379-81. doi: 10.1055/s-2001-13696.

9. Young C. A., Menias C. O., Bhalla S., Prasad S. R. CT features of esophageal emergencies. Radiographics. 2008 Oct;28(6):1541-53. doi: 10.1148/rg.286085520.

10. Cullen S. N., McIntyre A. S. Dissecting intramural haematoma of the oesophagus. Eur J Gastroenterol Hepatol. 2000 Oct;12(10):1151-62. doi: 10.1097/00042737-200012100-00014.

11. Santiago Garcia J., Duffy J., Ortiz-Fernandez-Sordo J., Budihal S., Parra-Blanco A., Ragunath K. Endoscopic management of eosinophilic esophagitis presenting as a double lumen esophagus. Endoscopy. 2020 Sep;52(9): E316-E317. doi: 10.1055/a-1119-0894.