S2255 Oral Viscous Budesonide Ameliorates PPI-Refractory Lymphocytic Esophagitis: A Longitudinal Clinical, Endoscopic, and Histologic Outcome Case Report
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Introduction: Lymphocytic esophagitis (LyE) is a new and emerging immune-mediated esophageal disease, manifesting clinically as dysphagia. Its natural history and effective treatments remain poorly characterized. We present a patient with LyE complicated by esophageal strictures refractory to proton-pump inhibitor (PPI) therapy, who demonstrated clinical, endoscopic and histologic improvement on swallowed oral viscous budesonide (OVB). Case Description/Methods: A 77-year-old nonsmoker nonatopic female with a history of remote buccal lichen planus, lymphocytic colitis, depression, anxiety, osteoarthritis, presented with chronic progressively worsening dysphagia to solids refractory to omeprazole 20 mg twice daily. Initial EGD elsewhere showed a narrowed esophagus with diffuse pallor, edema, decreased vascularity, diffuse rings, mucosal scarring, and distal strictures status post dilation. Esophageal biopsies showed LyE with >25 intraepithelial lymphocytes (IEL)/hpf proximally and distally. Our index EGD, 5 months after the initial outside EGD, revealed similar findings requiring esophageal dilation to 15-mm. Optimization of omeprazole was attempted but was limited to 20 mg daily due to dizziness. Repeat EGD after 3 months (month 8) showed worsening disease. OVB at 2 mg twice daily was added at month 9 to low-dose PPI, with clinical improvement within 2 weeks. OVB was reduced to 1 mg twice daily after 3 months due to joint pain. After 5 months on OVB and low-dose PPI, EGD showed partial improvement, with histologic remission proximally but not distally. EGD after 2 additional months on OVB monotherapy, off PPI, showed continued endoscopic improvement with decreased edema, improved vascularity, and improved esophageal caliber with no mucosal scarring. Mild diffuse esophageal rings were still present but appeared improved. Two distal esophageal strictures were dilated to 15-mm. Pan-esophageal biopsies improved histologically. Patient remains clinically improved on reduced OVB dose at 0.5 mg twice daily. Discussion: LyE has clinical and endoscopic features reminiscent of eosinophilic esophagitis (EoE); however, esophageal non-granulocytic infiltration with >20 peripapillary IEL/hpf is a predominant histologic feature within a spongiotic epithelium. Guidance for LyE therapy has not been established. Using OVB dosing regimen from EoE clinical trials, our case details longitudinal clinical, endoscopic and histologic outcomes in PPI-refractory LyE with treatment response to OVB, serving a basis for future studies.Keywords:
Esophagogastroduodenoscopy
Esophagitis
Esophageal stricture
Eosinophilic Esophagitis
Abstract: Over the past decade, the role of proton pump inhibitor (PPI) medication has evolved from a diagnostic tool for Eosinophilic Esophagitis (EoE), by excluding patients with PPI responsive esophageal eosinophilia (PPI-REE), to a therapy for EoE. This transition resulted from the Updated International Consensus Diagnostic Criteria for Eosinophilic Esophagitis: Proceedings of the Appraisal of Guidelines for Research and Evaluation II (AGREE) Conference to support PPI therapy for EoE in children and adults. Additional recent advances have suggested a role for genetic variations that might impact response to PPI therapy for EoE. This review article will explore a brief background of EoE, the evolution of PPI therapy for EoE and its proposed mechanisms, efficacy and safety in children and adults, and considerations for future PPI precision medicine in patients with EoE. Keywords: eosinophilic esophagitis, EoE, proton pump inhibitor medication, PPI, precision medicine
Eosinophilic Esophagitis
Dietary therapy
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Eosinophilic Esophagitis
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Eosinophilic Esophagitis
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Eosinophilic Esophagitis
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Eosinophilic esophagitis is characterized by eosinophil inflammation restricted to the esophagus and the resulting symptoms of esophageal dysfunction. Critical to the diagnosis of eosinophilic esophagitis is a trial of proton pump inhibitor therapy to exclude alternative causes of esophageal eosinophilia such as proton pump inhibitor-responsive esophageal eosinophilia. While consensus guidelines recommend a proton pump inhibitor trial prior to diagnosis, little is known about its implementation in clinical practice. The primary aim of this study is to assess the frequency of proton pump inhibitor trial prior to the diagnosis of eosinophilic esophagitis in community practice. The secondary aim is to assess the frequency of other treatments for eosinophilic esophagitis, including topical steroids and/or dietary therapy, in patients who did not undergo a proton pump inhibitor trial prior to diagnosis or who had an alternative diagnosis to eosinophilic esophagitis upon completed workup. We conducted a single-center, case series of patients referred to the Hospital of the University of Pennsylvania for eosinophilic esophagitis management between 2010 and 2015. This case series consisted of 125 patients who were referred from community practitioners with a presumptive diagnosis of eosinophilic esophagitis. Upon review, 90 out of 125 (72%) patients had not had a proton pump inhibitor trial or esophageal pH testing prior to the diagnosis of eosinophilic esophagitis being made. Of these patients, 77.8% (70/90) had already received either topical steroid or dietary therapy for presumed eosinophilic esophagitis. Of the 125 patients initially diagnosed with eosinophilic esophagitis, 32 (25.6%) were found to have an alternative diagnosis, and 79.2% of this subset of patients (25/32) had previously received topical steroid or dietary therapy. This study demonstrates that a substantial number of patients with presumed eosinophilic esophagitis have not had a proton pump inhibitor trial prior to diagnosis in community practice. This led to the misclassification of patients and potentially to the use of less optimal medical therapies in a substantial number of these patients.
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Eosinophilic esophagitis (EoE) is a chronic, immune/antigen-mediated esophageal disease, with eosinophilic infiltration limited to the esophagus. A minority of EoE patients respond well to proton pump inhibitor (PPI) therapy alone, and that condition is labelled PPI-responsive esophageal eosinophilia (PPI-REE). The prevalence of PPI-REE among EoE cases is unknown. We aimed to identify clinical manifestations of PPI-REE, and the proportion of PPI-REE among all EoE cases.
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Esophagogastroduodenoscopy
Esophageal stricture
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Objective: Eosinophilic esophagitis is a chronic inflammatory disease characterized by eosinophilic infiltration and esophagus dysfunction symptoms. Proton pump inhibitor responsive esophageal eosinophilia is similar to eosinophilic esophagitis in terms of clinical, laboratory, genetic expression profile, endoscopic and histopathological features. In this study, we aimed to share demographic features, clinical, laboratory and histopathological findings, and treatment outcomes of patient with eosinophilic esophagitis and proton pump inhibitor responsive esophageal eosinophilia. Materials and Methods: Demographic features, laboratory, endoscopic and histopathological findings, and treatment outcomes of patients followed in our clinic were recorded retrospectively since January 2010. Results: Four thousand six hundred fifty five patients underwent esophagogastroscopy since 2010 in our clinic and 0.4% (n=18) of these patients were diagnosed with eosinophilic esophagitis, and 0.2% (n=8) with proton pump inhibitor-responsive esophageal eosinophilia. The main symptom of patients with eosinophilic esophagitis were food impaction/dysphagia (n=5, 27.8%) and chronic abdominal pain (n=5, 27.8%). Allergen sensitization was found in 14 (77.8%), increased IgE in 12 (66.7%), peripheral eosinophilia in 12 (66.7%), and food allergen sensitization in 10 patients (55.6%) with eosinophilic esophagitis. On histopathological examination, the mean intraepithelial eosinophil count was 48.9 ± 30.9 cells / HPF (400x). When patients with eosinophilic esophagitis (group 1) and proton pump inhibitor-responsive esophageal eosinophilia (group 2) were compared, it was found that chronic abdominal pain was more common in the proton pump inhibitor-responsive esophageal eosinophilia group and food allergen sensitization in the eosinophilic esophagitis group (p<0.05). Total IgE, peripheral eosinophil count and intraepithelial eosinophil count were higher in the eosinophilic esophagitis group, but the differences were not statistically significant. Diet (n=11), medical (n=17) and dilatation (n=1) therapies were used in the eosinophilic esophagitis group. Fibrosis was detected on the histopathological examination in two patients who underwent TED and then SED was started. No side effect was seen any group in long term. Conclusion: Eosinophilic esophagitis and proton pump inhibitor-responsive esophageal eosinophilia have similar laboratory and histopathological features but eosinophilic esophagitis should be suspected more frequently in the presence of food allergy. Long-term followup is essential in patients with eosinophilic esophagitis.
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