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食管颗粒细胞瘤与嗜酸性食管炎之间的统计学显著关联:对两家大型医院167434例食管胃镜检查的16年分析

A Statistically Significant Association Between Esophageal Granular Cell Tumors and Eosinophilic Esophagitis: A 16-year Analysis at Two Large Hospitals of 167,434 EGDs.

作者信息

Nojkov Borko, Amin Mitual, Ghaith Gehad, Cappell Mitchell S

机构信息

Division of Gastroenterology and Hepatology, University of Michigan Health System, Ann Arbor, MI, USA.

Department of Pathology, William Beaumont Hospital, Royal Oak, MI, 48073, USA.

出版信息

Dig Dis Sci. 2017 Dec;62(12):3517-3524. doi: 10.1007/s10620-017-4802-9. Epub 2017 Oct 24.

DOI:10.1007/s10620-017-4802-9
PMID:29064014
Abstract

BACKGROUND

Esophageal granular cell tumor (eGCT) is a rare, usually benign, neoplasm of neuroectodermic origin. Eosinophilic esophagitis (EoE) is a relatively uncommon, immune-mediated, chronic disease. Both diseases commonly present with dysphagia. One case has been reported of simultaneous occurrence of both diseases.

AIMS

To determine the association between diseases.

METHODS

The present study was an IRB-approved, retrospective review of esophagogastroduodenoscopies (EGDs) with esophageal biopsies from two large hospitals, 1999-2014.

RESULTS

Among 29,235 EGDs with esophageal biopsies for 16 years (167,434 total EGDs), 16 patients had pathologically diagnosed eGCT, and 1225 patients had pathologically diagnosed EoE. Five (31%) of 16 patients with eGCT had concomitant EoE (p = 0.001, OR 10.43, 95% ORCI 3.16-32.44, Fisher's exact test). Patients with simultaneous eGCT and EoE were young (mean age = 33.6 ± 12.9 years). Three were female. Dysphagia was presenting symptom in 4 (80%) of patients. Three had asthma. All five patients had > 20 eosinophils/hpf in esophageal biopsy specimens. Three patients had endoscopic esophageal abnormalities suggesting EoE. Four patients were treated with a PPI (before and after diagnosis of EoE), and 2 patients underwent six-food-elimination diet with partial symptomatic improvement. The eGCTs averaged 13.4 ± 4.2 mm in maximal diameter and were located in upper-2, middle-2, and lower esophagus-2 (1 patient had 2 eGCTs). eGCTs were endoscopically resected-3 patients, and monitored-2 patients. Surveillance endoscopies revealed no recurrence or growth of eGCTs after resection (mean follow-up = 4.6 years).

CONCLUSIONS

This novel report of 5 patients with simultaneous EoE and eGCT adds to one, previously published case and suggests these two diseases are associated, and have a common pathophysiologic link, despite apparently different pathogenesis. Large, prospective, endoscopic and pathologic studies are warranted to further investigate this association.

摘要

背景

食管颗粒细胞瘤(eGCT)是一种罕见的、通常为良性的神经外胚层起源的肿瘤。嗜酸性粒细胞性食管炎(EoE)是一种相对不常见的免疫介导的慢性疾病。这两种疾病通常都表现为吞咽困难。已有一例两种疾病同时发生的报道。

目的

确定这两种疾病之间的关联。

方法

本研究经机构审查委员会(IRB)批准,对1999年至2014年两家大型医院进行的食管胃十二指肠镜检查(EGD)及食管活检进行回顾性分析。

结果

在16年中进行的29235例有食管活检的EGD(共167434例EGD)中,16例患者经病理诊断为eGCT,1225例患者经病理诊断为EoE。16例eGCT患者中有5例(31%)同时患有EoE(p = 0.001,比值比10.43,95%可信区间3.16 - 32.44,Fisher精确检验)。同时患有eGCT和EoE的患者较为年轻(平均年龄 = 33.6 ± 12.9岁)。3例为女性。4例(80%)患者的主要症状为吞咽困难。3例有哮喘。所有5例患者食管活检标本中每高倍视野嗜酸性粒细胞均>20个。3例患者内镜下食管异常提示EoE。4例患者接受质子泵抑制剂治疗(在诊断EoE前后),2例患者接受六种食物排除饮食,症状部分改善。eGCT最大直径平均为13.4 ± 4.2毫米,位于食管上段2例、中段2例、下段2例(1例患者有2个eGCT)。3例患者的eGCT通过内镜切除,2例患者进行监测。监测内镜检查显示切除后eGCT无复发或生长(平均随访 = 4.6年)。

结论

这份关于5例同时患有EoE和eGCT患者的新报告补充了之前发表的1例病例,表明这两种疾病有关联,且有共同的病理生理联系,尽管其发病机制明显不同。有必要进行大规模的前瞻性内镜和病理研究以进一步探究这种关联。

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