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在一名依赖胃造口管且使用氨基酸配方奶粉的儿科患者中,对度普利尤单抗有反应的组织学显著的嗜酸性食管炎

Histologically Remarkable Eosinophilic Esophagitis Responsive to Dupilumab in a Gastrostomy Tube-Dependent Pediatric Patient on Amino Acid Formula.

作者信息

Castro Kenny, Arostegui Dalia, Schwarz Steven, Gandhi Sonia, Peshimam Juveria, Rabinowitz Simon, Pittman Meredith, Wallach Thomas

机构信息

From the Division of Pediatric Gastroenterology, Department of Pediatrics, SUNY Downstate, Brooklyn, New York.

Department of Pediatrics, SUNY Downstate, Brooklyn, New York.

出版信息

JPGN Rep. 2022 Sep 1;3(4):e250. doi: 10.1097/PG9.0000000000000250. eCollection 2022 Nov.

Abstract

Eosinophilic esophagitis (EoE), an eosinophil predominant, TH2-mediated condition increasing in prevalence in pediatric and adult populations, is typically treated with dietary manipulations to avoid triggering antigens. However, identifying specific dietary causes remains a persistent challenge, and restrictive diets are burdensome. Total dietary modification using amino acid-based formula does not always produce symptomatic or histologic resolution, suggesting that exposure to ingested aeroallergens drives their disease. EoE patients demonstrate symptomatic exacerbation from July to September correlating with higher grass and ragweed pollen counts. We present a 7-year-old tracheostomy- and gastrostomy-dependent girl who was found on surveillance endoscopy to have profound eosinophilic infiltration throughout the esophagus with inflammatory changes including basal cell hyperplasia on histology. She responded partially to topical corticosteroid therapy with fluticasone and had complete resolution of esophageal eosinophilic infiltrate with subcutaneous dupilumab.

摘要

嗜酸性粒细胞性食管炎(EoE)是一种以嗜酸性粒细胞为主、由TH2介导的疾病,在儿童和成人中的患病率不断上升,通常采用饮食调整来避免触发抗原。然而,确定具体的饮食病因仍然是一个持续存在的挑战,而且限制性饮食负担较重。使用氨基酸配方进行全面饮食调整并不总能使症状或组织学得到缓解,这表明接触摄入性气传变应原会引发该病。EoE患者在7月至9月出现症状加重,这与较高的禾本科和豚草花粉计数相关。我们报告一名7岁的依赖气管造口术和胃造口术的女孩,在监测性内镜检查中发现其整个食管有严重的嗜酸性粒细胞浸润,并伴有包括组织学上基底细胞增生在内的炎症变化。她对氟替卡松局部皮质类固醇治疗有部分反应,皮下注射度普利尤单抗后食管嗜酸性粒细胞浸润完全消退。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/a5bd/10158404/902f8af2c3dd/pg9-3-e250-g001.jpg

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