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十二指肠狭窄——嗜酸性粒细胞性胃肠炎的一种罕见表现:病例报告

Duodenal stenosis, an unusual presentation of eosinophilic gastroenteritis: a case report.

作者信息

Di Mari Clelia, Pozzi Elena, Mantegazza Cecilia, Destro Francesca, Meroni Milena, Coletta Marina, Sorge Andrea, Pelizzo Gloria, Zuccotti Gian Vincenzo

机构信息

Department of Pediatrics, Buzzi Children's Hospital, Milan, Italy.

Department of Pediatric Surgery, Buzzi Children's Hospital, Milan, Italy.

出版信息

Front Pediatr. 2024 Apr 18;12:1390946. doi: 10.3389/fped.2024.1390946. eCollection 2024.

Abstract

Eosinophilic gastrointestinal diseases (EGIDs) are rare, chronic inflammatory disorders characterized by eosinophilic infiltration of the gastrointestinal tract. Symptoms and clinical presentations vary depending on the site and layer of the gastrointestinal wall infiltrated by eosinophils. Gastrointestinal obstruction is a serious, though uncommon, presentation. Management can be extremely challenging because of the rarity of the condition and the lack of robust scientific evidence. Current treatment approaches for EGIDs mainly focus on elimination diets, proton pump inhibitors and corticosteroids, which present high refractoriness rates. Novel targeted therapies are being investigated but not routinely used. Surgery should be avoided as far as possible; however, it may be the only option in gastrointestinal obstruction when long-term remission cannot be attained by any medical strategy. Herein we report the case of an adolescent boy affected by an eosinophilic gastrointestinal disease with progressive duodenal stenosis, refractory to medical therapy, who successfully benefitted from surgical management. He presented with a one-year history of gastrointestinal obstructive symptoms with feeding intolerance. After the diagnostic workup, he was diagnosed with an eosinophilic gastrointestinal disease (esophagitis and enteritis) with a duodenal involvement causing a progressive duodenal stenosis. Due to refractoriness to the conventional medical therapies and the consequent high impact on his quality of life, related both to the need for enteral nutrition and repeated hospitalizations, we decided to perform a gastro-jejunum anastomosis, which allowed us to obtain a clinical and endoscopic long-term remission. The early discussion of the case and the involvement of all experienced specialists, pediatricians and pediatric surgeons is essential.

摘要

嗜酸性粒细胞性胃肠疾病(EGIDs)是一种罕见的慢性炎症性疾病,其特征是胃肠道出现嗜酸性粒细胞浸润。症状和临床表现因嗜酸性粒细胞浸润的胃肠道部位和层次而异。胃肠道梗阻是一种严重但不常见的表现。由于该疾病罕见且缺乏有力的科学证据,其管理极具挑战性。目前EGIDs的治疗方法主要集中在排除饮食、质子泵抑制剂和皮质类固醇,但这些方法的难治率很高。新型靶向治疗正在研究中,但尚未常规使用。应尽可能避免手术;然而,当任何药物治疗策略都无法实现长期缓解时,手术可能是胃肠道梗阻的唯一选择。在此,我们报告一例患有嗜酸性粒细胞性胃肠疾病并伴有进行性十二指肠狭窄的青少年男性病例,该病例对药物治疗无效,最终通过手术治疗成功获益。他有一年的胃肠道梗阻症状病史,伴有喂养不耐受。经过诊断检查,他被诊断为嗜酸性粒细胞性胃肠疾病(食管炎和肠炎),十二指肠受累导致进行性十二指肠狭窄。由于对传统药物治疗无效,且由于肠内营养需求和反复住院对其生活质量产生了严重影响,我们决定进行胃空肠吻合术,从而实现了临床和内镜下的长期缓解。对该病例进行早期讨论并让所有经验丰富的专家、儿科医生和小儿外科医生参与至关重要。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ef9e/11063303/48ac99a55774/fped-12-1390946-g001.jpg

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