低于亨廷顿病阈值的亨廷廷蛋白 CAG 重复大小变异:与抑郁、焦虑和基底神经节结构的关联。

IF 3.7 2区 生物学 Q2 BIOCHEMISTRY & MOLECULAR BIOLOGY European Journal of Human Genetics Pub Date : 2024-11-21 DOI:10.1038/s41431-024-01737-1
Magdalena Vater, Nicolas Rost, Gertrud Eckstein, Susann Sauer, Alina Tontsch, Angelika Erhardt, Susanne Lucae, Tanja Brückl, Thomas Klopstock, Philipp G Sämann, Elisabeth B Binder
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引用次数: 0

摘要

亨廷顿氏病(Huntington's disease,HD)与精神症状密切相关,但亨廷丁基因(HTT)CAG重复序列大小变异与HD综合征以外的精神表型之间的关系仍未得到充分研究。在这项遗传病例对照研究中,我们比较了重度抑郁症(MDD)(2136 人)和焦虑症(ANX)(493 人)患者与健康对照组(CON)(1566 人)之间 HTT CAG 重复大小在预定范围内的分布情况。我们使用回归模型研究了等位基因之间的相互作用以及与细粒度临床表型和基底节结构的关联。在患者中,处于不完全渗透范围(36-39 个重复序列)的 HD 基因突变所占比例并不高。在 48 岁以上的参与者中,两个 HTT 等位基因上的 13-20 个重复序列与 ANX 风险降低有关,而 13-20 | 21-26 组合与 ANX 风险增加有关。事后分析证实,在 21 个等位基因重复点附近存在一个转折点,而在 MDD 方面也发现了相同的趋势。对所有等位基因组合进行的患者 | CON 联合分析证实了相互作用效应和与年龄相关的等位基因 | 风险特征。短等位基因与长等位基因的相互作用效应以及短等位基因与年龄的负相关关系被检测到,且与诊断组无关。总之,我们发现在非高密度范围内,两个等位基因的 HTT CAG 重复大小与常见精神疾病的风险调节以及基底神经节结构的差异有关,且与年龄相关,这可能意味着功能多样的野生型亨廷蛋白的正常变异可能会影响大脑功能。
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Huntingtin CAG repeat size variations below the Huntington's disease threshold: associations with depression, anxiety and basal ganglia structure.

Huntington's disease (HD) is strongly associated with psychiatric symptoms, yet, associations between huntingtin gene (HTT) CAG repeat size variations and psychiatric phenotypes outside the HD complex are still under-investigated. In this genetic case-control study we compared the distribution of HTT CAG repeat sizes in predefined ranges between patients with major depressive disorder (MDD) (n = 2136) and anxiety disorders (ANX) (n = 493), and healthy controls (CON) (n = 1566). We used regression models to study interactions between the alleles and associations with fine-granular clinical phenotypes and basal ganglia structure. HD mutations in the range of incomplete penetrance (36-39 repeats) were not overrepresented in patients. In participants older than 48 years, 13-20 repeats on both HTT alleles were associated with a reduced ANX risk whereas a 13-20 | 21-26 combination was associated with an increased ANX risk. Post-hoc analyses confirmed a turning point around 21 repeats and trends in the same direction were detected for MDD. The joint patient | CON analysis of the full spectrum of allele combinations confirmed interaction effects and age-dependent allele | risk profiles. A short-by-long interaction effect and an age-dependent negative correlation of the short allele on the nucleus accumbens volume was detected, independently of the diagnostic group. In conclusion, we revealed that HTT CAG repeat sizes of both alleles in the non-HD range are associated with a risk modulation for common psychiatric disorders as well as basal ganglia structure differences in an age-dependent way, possibly implying that normal variation of the functionally diverse wildtype huntingtin protein may impact brain function.

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来源期刊
European Journal of Human Genetics
European Journal of Human Genetics 生物-生化与分子生物学
CiteScore
9.90
自引率
5.80%
发文量
216
审稿时长
2 months
期刊介绍: The European Journal of Human Genetics is the official journal of the European Society of Human Genetics, publishing high-quality, original research papers, short reports and reviews in the rapidly expanding field of human genetics and genomics. It covers molecular, clinical and cytogenetics, interfacing between advanced biomedical research and the clinician, and bridging the great diversity of facilities, resources and viewpoints in the genetics community. Key areas include: -Monogenic and multifactorial disorders -Development and malformation -Hereditary cancer -Medical Genomics -Gene mapping and functional studies -Genotype-phenotype correlations -Genetic variation and genome diversity -Statistical and computational genetics -Bioinformatics -Advances in diagnostics -Therapy and prevention -Animal models -Genetic services -Community genetics
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