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患有乳糜泻的儿童的食管嗜酸性粒细胞增多症。

Oesophageal eosinophilia in children with coeliac disease.

作者信息

Ari Anne, Morgenstern Sara, Chodick Gabriel, Matar Manar, Silbermintz Ari, Assa Amit, Mozer-Glassberg Yael, Rinawi Firas, Nachmias-Friedler Vered, Shamir Raanan, Zevit Noam

机构信息

Institute of Gastroenterology, Nutrition and Liver Diseases, Schneider Children's Medical Center of Israel, Petach Tikvah, Israel.

Pediatrics C, Schneider Children's Medical Center of Israel, Petach Tikvah, Israel.

出版信息

Arch Dis Child. 2017 Sep;102(9):825-829. doi: 10.1136/archdischild-2016-311944. Epub 2017 Apr 12.

Abstract

OBJECTIVES

An association between coeliac disease (CD) and eosinophilic oesophagitis (EoE)/oesophageal eosinophilia (EE) has been suggested. We sought to characterise children with CD+EE in-depth and assess the contribution of each condition to the clinical presentation and treatment response.

STUDY DESIGN

Medical records of children with both CD+EE, or isolated EoE diagnosed between 2000 and 2014, were retrospectively reviewed and compared with patients with isolated CD or epigastric pain. Frequency of EE was calculated from endoscopy results of patients with suspected CD or epigastric pain between 2011 and 2014. Missing data were obtained via a telephone questionnaire.

SETTING

Single large, tertiary paediatric centre.

PATIENTS

17 CD+EE, 46 EoE, 302 isolated CD and 247 epigastric pain.

RESULTS

The patients with CD+EE shared characteristics of both individual conditions. While age at diagnosis, family history of autoimmunity/CD and anaemia were similar to patients with CD, other characteristics such as male gender, personal/family history of atopy, peripheral eosinophilia and oesophageal white papules were more similar to patients with EoE. Combined patients (CD+EE) tended to present with CD-associated symptoms; the majority (63%) later developed typical EoE symptoms. Only a minority (21%) of combined patients had EE that resolved after a gluten-free diet; another 21% had normalisation of EE upon proton pump inhibitor treatment. The remainder required EoE-specific treatment.

CONCLUSION

Patients with CD found to have EE share characteristics with both isolated CD and EoE. It appears that these are two coexisting entities presenting in the same patient rather than eosinophilia associated with CD, and therefore, interventions separately addressing each condition may be considered.

摘要

目的

乳糜泻(CD)与嗜酸性食管炎(EoE)/食管嗜酸性粒细胞增多症(EE)之间的关联已被提出。我们试图深入描述患有CD+EE的儿童,并评估每种疾病对临床表现和治疗反应的影响。

研究设计

对2000年至2014年间诊断为CD+EE或孤立性EoE的儿童的病历进行回顾性分析,并与孤立性CD或上腹部疼痛的患者进行比较。根据2011年至2014年间疑似CD或上腹部疼痛患者的内镜检查结果计算EE的发生率。通过电话问卷获取缺失数据。

研究地点

单一大型三级儿科中心。

患者

17例CD+EE患者、46例EoE患者、302例孤立性CD患者和247例上腹部疼痛患者。

结果

CD+EE患者具有两种疾病各自的特征。虽然诊断时的年龄、自身免疫/CD家族史和贫血情况与CD患者相似,但其他特征如男性、特应性个人/家族史、外周嗜酸性粒细胞增多和食管白色丘疹更类似于EoE患者。合并患者(CD+EE)倾向于出现与CD相关的症状;大多数(63%)后来出现典型的EoE症状。只有少数(21%)合并患者的EE在无麸质饮食后得到缓解;另外21%的患者在质子泵抑制剂治疗后EE恢复正常。其余患者需要针对EoE的特异性治疗。

结论

被发现患有EE的CD患者具有孤立性CD和EoE两者的特征。看来这是同一患者中并存的两种疾病,而非与CD相关的嗜酸性粒细胞增多,因此,可以考虑分别针对每种疾病进行干预。

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