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保加利亚原发性免疫缺陷病 - PID 国家专家中心的成就与挑战。

Primary immunodeficiencies in Bulgaria - achievements and challenges of the PID National Expert Center.

机构信息

Clinic of Clinical Immunology with Stem Cell Bank, Expert Center for Rare Diseases-PID, University Hospital "Alexandrovska", Sofia, Bulgaria.

Department of Clinical Immunology, Faculty of Medicine, Medical University, Sofia, Bulgaria.

出版信息

Front Immunol. 2022 Sep 22;13:922752. doi: 10.3389/fimmu.2022.922752. eCollection 2022.

Abstract

Tremendous progress has been made in the recognition of primary immune deficiencies (PIDs) in Bulgaria since in 2005 we have joined the J Project Central-Eastern European collaborative program. Ten years later an Expert Centre (ExpC) for Rare Diseases - Primary Immune Deficiencies at the University Hospital "Alexandrovska"- Sofia was established. In May 2017 The National Register of Patients with Rare Diseases also became operational as a database containing clinical and genetic information for Bulgarian patients with PID. The transfer of data and information on Bulgarian PID patients to the European Primary Immunodeficiency Database, managed by the European Society for Primary Immunodeficiency (ESID) has started in 2020. The total number of registered patients now is 191 (100 men and 91 women), with more than half of them being children (106; 55.5%). Regular updating of the information in the register showed that 5.2% of patients are deceased and the majority (94.8%) is a subject to continuous monitoring as it has been reported for other European countries as well. With the establishment of the ExpC, the dynamics in the diagnosis and registration of patients with PID significantly intensified. For a period of 5 years (2016-2021) 101 patients were evaluated and registered in comparison with previous period - before ExpC establishment when only 89 patients were diagnosed. The most common pathology was humoral immune deficiency (85 patients; 44.5%). Ninety-six (50.3%) of the patients underwent genetic testing, and 66. 7% had genetically confirmed diagnosis. Three of the variants have not been reported in population databases. Following genetic investigation confirmation of the initial phenotypic diagnosis was achieved in 82.8% of cases and change in the diagnosis - in 17%. Sixty-two patients were on regular replacement or specific therapy, and the rest received symptomatic and supportive treatment. In summary, we present the first epidemiological report of PIDs in Bulgaria, based on the National PID register. Data on the clinical, phenotypic and genetic characteristics of PID patients provided important information about the nature of primary immunodeficiency diseases in our country.

摘要

自 2005 年加入 J 项目中东欧合作计划以来,保加利亚在原发性免疫缺陷(PID)的认识方面取得了巨大进展。十年后,在索非亚的“亚历山德拉夫斯卡”大学医院成立了一个罕见疾病专家中心(ExpC)-原发性免疫缺陷。2017 年 5 月,国家罕见疾病患者登记处也开始运作,成为一个包含保加利亚 PID 患者临床和遗传信息的数据库。2020 年开始向欧洲原发性免疫缺陷数据库(由欧洲原发性免疫缺陷学会(ESID)管理)传输保加利亚 PID 患者的数据和信息。目前已登记的患者总数为 191 人(100 名男性和 91 名女性),其中一半以上是儿童(106 人;55.5%)。定期更新登记处的信息表明,有 5.2%的患者死亡,与其他欧洲国家一样,大多数(94.8%)患者需要持续监测。随着 ExpC 的建立,PID 患者的诊断和登记动态明显加强。在 5 年期间(2016-2021 年),对 101 名患者进行了评估和登记,与之前的时期(ExpC 成立之前)相比,当时只诊断出 89 名患者。最常见的病理是体液免疫缺陷(85 名患者;44.5%)。96 名(50.3%)患者接受了基因检测,其中 66.7%的患者基因诊断得到确认。有 3 种变体尚未在人群数据库中报道。在进行基因调查后,82.8%的病例得到了初始表型诊断的确认,17%的病例改变了诊断。62 名患者定期接受替代或特定治疗,其余患者接受对症和支持治疗。总之,我们根据国家 PID 登记处提出了保加利亚首例 PID 的流行病学报告。PID 患者的临床、表型和遗传特征数据提供了有关我国原发性免疫缺陷疾病性质的重要信息。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/a0fa/9535737/5ca1f880b8d9/fimmu-13-922752-g001.jpg

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