Abstract

Introduction: Adalimumab is an anti-tumor necrosis factor-alpha (TNF-alpha) used in the treatment of Crohn’s disease (CD). There have been reported associations of CD and Eosinophilic Esophagitis (EoE); A recent study found that the prevalence of Inflammatory Bowel Disease (IBD) in the EoE population was 2.2% and the prevalence of EoE in the IBD population was 1.5%. A second study found that patients with IBD are four times more likely to develop EoE than the general population. In a small case series, 10 children with CD were diagnosed with EoE while on biologic therapy with infliximab. We describe two adolescents who developed EoE while on adalimumab therapy for CD. Case Description/Methods: Case 1: A 16-year-old male with CD and Down’s syndrome underwent an upper endoscopy and colonoscopy for monitoring of IBD activity. On gross examination, the esophagus appeared erythematous and biopsies of the mid and distal esophagus revealed 50-100 eos/hpf consistent with EoE. He had initiated adalimumab therapy three years prior to EoE diagnosis when esophageal biopsies were normal. Adalimumab level at this time was 6.79 ug/mL. He had no issues with dysphagia, but his weight gain was sub-optimal. After initiation of topical esophageal steroid therapy his weight gain improved and adalimumab therapy has been continued. Due to the current pandemic, follow up biopsies have been postponed. Case 2: A 14-year-old female with CD underwent an upper endoscopy and colonoscopy for monitoring of IBD activity. On gross examination, the esophagus had furrowing and biopsies of the mid and distal esophagus revealed 25-50 eos/hpf consistent with EoE. She had initiated adalimumab therapy one year prior to EoE diagnosis when esophageal biopsies showed mild chronic esophagitis (0-2 eos/hpf). Colonic biopsies showed active disease. Adalimumab level at this time was 17 ug/mL. She reported symptoms of dysphagia. Adalimumab therapy was changed to infliximab due to insufficient disease control and she was started on a high dose proton pump inhibitor to treat EoE with resolution of dysphagia. Due to the current pandemic, follow up biopsies have been postponed. Discussion: These two cases highlight the development of EoE in children with IBD on anti-TNF therapy. To our knowledge, based on a PubMed search, this is the first case report of children developing EoE while on adalimumab therapy for CD.

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