Eosinophilic Esophagitis Complicated by a Distal Esophageal Stricture in a 15-Month-Old Infant

Eosinophilic esophagitis (EoE) is a chronic immune mediated disease characterized by dysphagia, food impactions, and esophageal eosinophilia, culminating in fibrosis and stricture formation. In children, fibrostenotic complication is rare. Studies have reported up to 86% of adults with dysphagia with esophageal narrowing as compared to 6%of children. Further, the incidence of stricture in children is low, as studies report rates of 3.4%.The youngest age previously reported was 8.4 years. Risk factors related to fibrostenosis include older age, longer duration and increased frequency of symptoms. We describe a case of a 15 month old with dysphagia, who was diagnosed with EoE and a distal esophageal stricture. A 6 month old ex 25 week female was originally seen for evaluation for gastroesophageal reflux and cow's milk protein allergy. Symptoms were well controlled on Ranitidine and Erythromycin. She progressed to Stage 2 baby foods without event. However, at 13 months old she had dysphagia, vomiting and food refusal when advancing to higher textures and a table food diet. Proton Pump inhibitor (PPI) was started for treatment of reflux and for EoE evaluation, per the guidelines. An upper GI (UGI) done at 2 months of age indicated no abnormalities. A repeat UGI showed luminal narrowing of the distal esophagus and dilation of the mid esophagus. She had upper endoscopy at 15 months old, while on 2mg/kg/day PPI. Stenosis was found 20cm from the incisors, 50 eosinophils per high power field), degranulation, surface layering and eosinophilic micro-abscesses. She was started on a steroid course with PPI. After diagnosis, she required dilations every 4-6 weeks, with each subsequent dilation demonstrating improvement in stenosis. EoE was initially treated with targeted diet elimination however ultimately required oral viscous budesonide due to difficulties with diet adherence. Esophageal eosinophilia improved as well as symptoms and she was able to advance to a toddler diet. This case challenges existing thought that longstanding eosinophilic tissue remodelling leads to fibrotic changes given a de novo esophageal stricture in a 15 month old with EoE. This is the youngest age we are aware of involving such a presentation. It suggests that fibrostenosis may represent a phenotype separate, rather than a progression from the inflammatory phenotype of disease. It informs of the need for timely evaluation of EoE due to the potential for early development of stricture.1822_A Figure 1. A. Normal epiglottis at index scope (July 2017) B. Longitudinal furrows at in the mid esophagus at the index scope (July 2017) C. Stricture at 20 cm from incisors with a soft plastic foreign body at index scope measuring 4 mm (July 2017)1822_B Figure 2. A. Stricture at 20 cm from the incisors, 8 mm in diameter five weeks after completing prednisone taper (October 2017) B. Stricture 8 mm in diameter one month after last endoscopy and dilation with improved distensibility than prior (November 2017) C. Stricture measuring 15 mm in diameter three months after starting diet elimination therapy (January 2018)