小儿自身免疫性全血细胞减少症与炎症性肠病的关联表明两者有共同的遗传起源。

IF 5.1 2区 医学 Q1 HEMATOLOGY
M Gilton, H Fernandes, C Martinez, G Leverger, W Abou Chahla, V Li Thiao Te, M Deparis, C Armari Alla, N Garnier, J Benadiba, A Marie-Cardine, F Rieux-Laucat, C Picard, N Aladjidi, T Leblanc
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引用次数: 0

摘要

小范围报道了自身免疫性细胞减少症 (AIC) 与炎症性肠病 (IBD) 的关联,但 AIC 儿童中 IBD 的发病率和风险因素尚不清楚。本研究纳入了前瞻性 OBS'CEREVANCE 队列中的 169 名患有慢性免疫性血小板减少性紫癜、自身免疫性溶血性贫血或埃文斯综合征的儿童。共有 15 名儿童被诊断为 IBD,其中 14 名儿童在 AIC 诊断后发展为 IBD(中位延迟时间:21 个月)。IBD发病的唯一风险因素是AIC年龄超过10岁。在接受基因检测的 10 名儿童中,发现了 3 个与自身免疫性疾病相关的种系变异(CTLA4:2 个,DOCK11:1 个)。对于因 AIC 或既往 AIC 病史而接受监测的儿童和青少年,尤其是 10 岁以上的儿童,胃肠道(GI)症状(反复腹痛、消化道出血、慢性腹泻、体重减轻)应提示 IBD,并应进行专门的检查和基因研究。确定病因种系变异将有助于进行有针对性的治疗。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Association of paediatric autoimmune cytopenia and inflammatory bowel disease suggests a common genetic origin.

The association of autoimmune cytopenia (AIC) and inflammatory bowel disease (IBD) has been reported in small series, but the incidence of and risk factors for IBD in children with AIC are not known. One thousand six hundred nine children with chronic immune thrombocytopenic purpura, autoimmune haemolytic anaemia or Evans syndrome from the prospective OBS'CEREVANCE cohort are included in this study. Overall, 15 children were diagnosed with IBD, including 14 who developed IBD after AIC diagnosis (median delay: 21 months). The only risk factor for IBD development is age at AIC over 10 years. Out of 10 children genetically tested, germline variants associated with autoimmune disorders were identified in three (CTLA4: two, DOCK11: one). In children and adolescents monitored for AIC or past history of AIC, especially children over 10 years, gastro-intestinal (GI) symptoms (recurrent abdominal pains, GI bleeding, chronic diarrhoea, weight loss) should suggest IBD and deserve specific work-up and genetic studies. Identification of a causal germline variant will allow targeted therapy.

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来源期刊
CiteScore
8.60
自引率
4.60%
发文量
565
审稿时长
1 months
期刊介绍: The British Journal of Haematology publishes original research papers in clinical, laboratory and experimental haematology. The Journal also features annotations, reviews, short reports, images in haematology and Letters to the Editor.
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