遗传性还是获得性?全面的基因检测有助于对血管性水肿患者进行分层。

IF 2.6 4区 医学 Q2 ALLERGY
Marija Rozevska, Adine Kanepa, Signe Purina, Linda Gailite, Inga Nartisa, Henriette Farkas, Dmitrijs Rots, Natalja Kurjane
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引用次数: 0

摘要

遗传性血管性水肿(HAE)因其发作性、非特异性症状和病情重叠而给诊断带来挑战。本研究的重点是 HAE 的遗传基础,尤其是未解决的病例和 C1 抑制剂水平正常的病例(nC1-INH HAE)。本研究显示,传统检测仅在 10 例患者(n = 32)中发现了致病变异,这强调了采用基因组、外显子组和转录组测序的综合方法的必要性。尽管进行了广泛的基因分析,但在我们的研究中,nC1-INH HAE 的诊断率仍然很低,仅在 1 名患者(n = 21)中发现了 nC1-INH HAE 的致病变体。对候选基因的调查没有发现致病变异体,这促使我们对患者的诊断进行重新评估。本研究提倡对基因检测采取细致入微的方法,承认其局限性,并强调需要持续进行临床评估。nC1-INH HAE 的遗传情况十分复杂,需要进一步研究,以获得更全面的认识。总之,这项研究对 HAE 遗传学的复杂性提出了宝贵的见解,强调了诊断中的挑战和该疾病不断演变的性质。研究结果强调了先进测序技术和综合诊断策略在揭示 HAE 复杂性方面的重要性,尤其是在 nС1-INH HAE 病例中。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Hereditary or acquired? Comprehensive genetic testing assists in stratifying angioedema patients.

Hereditary angioedema (HAE) poses diagnostic challenges due to its episodic, non-specific symptoms and overlapping conditions. This study focuses on the genetic basis of HAE, particularly focusing on unresolved cases and those with normal C1-inhibitor levels (nC1-INH HAE). This study reveals that conventional testing identified pathogenic variants in only 10 patients (n = 32), emphasizing the necessity for an integrative approach using genome, exome, and transcriptome sequencing. Despite extensive genetic analyses, the diagnostic yield for nC1-INH HAE remains low in our study, the pathogenic variant for nC1-INH HAE was identified in only 1 patient (n = 21). Investigation into candidate genes yielded no pathogenic variants, prompting a re-evaluation of patients' diagnoses. This study advocates for a nuanced approach to genetic testing, recognizing its limitations and emphasizing the need for continuous clinical assessment. The complex genetic landscape of nC1-INH HAE necessitates further research for a more comprehensive understanding. In conclusion, this study contributes valuable insights into the genetic intricacies of HAE, highlighting the challenges in diagnosis and the evolving nature of the disease. The findings underscore the importance of advanced sequencing techniques and an integrated diagnostic strategy in unravelling the complexities of HAE, particularly in nС1-INH HAE cases.

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来源期刊
CiteScore
4.30
自引率
3.70%
发文量
96
审稿时长
12 weeks
期刊介绍: Allergy, Asthma & Clinical Immunology (AACI), the official journal of the Canadian Society of Allergy and Clinical Immunology (CSACI), is an open access journal that encompasses all aspects of diagnosis, epidemiology, prevention and treatment of allergic and immunologic disease. By offering a high-visibility forum for new insights and discussions, AACI provides a platform for the dissemination of allergy and clinical immunology research and reviews amongst allergists, pulmonologists, immunologists and other physicians, healthcare workers, medical students and the public worldwide. AACI reports on basic research and clinically applied studies in the following areas and other related topics: asthma and occupational lung disease, rhinoconjunctivitis and rhinosinusitis, drug hypersensitivity, allergic skin diseases, urticaria and angioedema, venom hypersensitivity, anaphylaxis and food allergy, immunotherapy, immune modulators and biologics, immune deficiency and autoimmunity, T cell and B cell functions, regulatory T cells, natural killer cells, mast cell and eosinophil functions, complement abnormalities.
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