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遗传性还是后天性?全面的基因检测有助于对血管性水肿患者进行分层。

Hereditary or acquired? Comprehensive genetic testing assists in stratifying angioedema patients.

作者信息

Rozevska Marija, Kanepa Adine, Purina Signe, Gailite Linda, Nartisa Inga, Farkas Henriette, Rots Dmitrijs, Kurjane Natalja

机构信息

Riga Stradiņš University, Riga, Latvia.

Children's clinical university hospital, Riga, Latvia.

出版信息

Allergy Asthma Clin Immunol. 2024 Mar 30;20(1):28. doi: 10.1186/s13223-024-00889-5.

Abstract

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.

摘要

遗传性血管性水肿(HAE)因其发作性、非特异性症状以及存在重叠病症而带来诊断挑战。本研究聚焦于HAE的遗传基础,尤其关注未确诊病例以及C1抑制剂水平正常的病例(nC1-INH HAE)。该研究表明,传统检测仅在10例患者中(n = 32)发现了致病变异,这凸显了采用基因组、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的诊断率仍然较低,仅在1例患者中(n = 21)鉴定出了nC1-INH HAE的致病变异。对候选基因的研究未发现致病变异,促使对患者的诊断进行重新评估。本研究主张采用细致入微的基因检测方法,认识到其局限性,并强调持续临床评估的必要性。nC1-INH HAE复杂的遗传图谱需要进一步研究以获得更全面的理解。总之,本研究为HAE的遗传复杂性提供了有价值见解,突出了诊断中的挑战以及该疾病不断演变的特性。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。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/4e00/10981324/b2815d9782b3/13223_2024_889_Fig1_HTML.jpg

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