Intramural esophageal dissection, a rare complication of eosinophilic esophagitis

Abstract

Eosinophilic esophagitis (EoE) is characterized clinically by typical endoscopic features and the presence of more than 15 eosinophils visible per high-power field in esophageal mucosal biopsy samples. Due to deep chronic eosinophilic inflammation, it can lead to complications, such as esophageal stenosis and rarely perforation. Intramural esophageal dissection (IED) is a previously undescribed complication of EoE in children. This article presents a clinical case of IED in a 15-year-old boy with clinical debut as sudden epigastric pain and nausea against the background of acute respiratory viral infection. Endoscopy, computed tomography, and X-ray examination revealed a double-barrel esophagus with two cavities, the esophagus itself and a blind diverticulum (pouch or “pocket”), in which granulation tissue and active inflammation with many neutrophils were detected. Initial histological assessment showed no significant eosinophilic infiltration in the esophagus. After 6 months, endoscopy revealed characteristic signs of EoE: longitudinal grooves and erosions, transverse rings, narrowing of the esophageal lumen. In the middle third, the picture of double-barrel esophagus remained unchanged, but without signs of acute detachment and with a dense septum between two esophageal cavities. Histologically, the infiltrate contained more than 50 eosinophils per high-power field. The diagnosis of EoE complicated by IED was established. A strict hypoallergenic diet and an oral gel budesonide were prescribed. Endoscopic dissection of the esophageal septum was performed, its patency was restored. In 2 months, during a re-examination after a course of pharmacotherapy and diet, there were no complaints, radiological evaluation of esophageal patency showed no abnormalities. Endoscopically, the esophagus was freely passable, there was a small fold at the surgical site, longitudinal grooves and small erosions in the lower third maintained. Histologically, the number of eosinophils decreased significantly. The therapy was continued. This clinical case demonstrates the possibility of IED development as a complication of EoE. The course of EoE can be asymptomatic, while IED can serve as the first manifestation of the disease. The endoscopic presentation of double-barrel 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 IED. Key words: eosinophilic esophagitis, intramural esophageal dissection, double-barrel esophagus