Jun Wang, Minhan Song, Zhimei Liu, Chaolong Xu, Ying Zou, Xin Duan, Yang Liu, Weihua Zhang, Jiuwei Li, Fang Fang
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引用次数: 0
摘要
本研究探讨了中国儿童单大规模线粒体DNA缺失(SLSMD)患者的临床和遗传学特征。我们使用远程PCR和下一代测序分析了28例患者(2004年7月- 2025年3月)。Spearman相关和方差分析评估了基因型与表型的关系。患者(平均年龄5.52±3.96岁)表现为多器官受累(5.43±1.87个器官)。常见的首发症状包括眼部(29%)、神经系统和内分泌功能紊乱。只有14.3%的患者有经典的4977 bp缺失,在25例患者中发现了23个新的缺失。较大的缺失与更多的MRC复合物缺失(r = 0.516, p = 0.0123)和更多的trna缺失(r = 0.534, p = 0.0103)相关。Kearns-Sayre综合征(KSS)患者比非KSS患者发病晚(p = 0.0337),缺失大(p = 0.0263), tRNA/MRC复合物受累(p = 0.0263, p = 0.0319)。中国儿童SLSMD主要导致KSS、Pearson综合征(PS)和累及多器官的进行性眼麻痹。基因型-表型相关性存在,特别是缺失大小、发病年龄和疾病严重程度之间。KSS患者表现出不同的遗传和临床特征,表明进展较慢。这项研究扩大了已知的SLSMD谱系,强调了儿科多器官疾病的线粒体检测。
Genotype-Phenotype Correlations in Chinese Pediatric Patients With Single Large-Scale Mitochondrial DNA Deletion Disorders.
This study investigated clinical and genetic characteristics of Chinese pediatric patients with single large-scale mitochondrial DNA deletions (SLSMD). We analyzed 28 patients (July 2004-March 2025) using long-range PCR and next-generation sequencing. Spearman correlation and ANOVA assessed genotype-phenotype relationships. Patients (mean age 5.52 ± 3.96 years) exhibited multi-organ involvement (5.43 ± 1.87 organs). Common initial presentations included ocular (29%), neurologic, and endocrine dysfunction. Only 14.3% had the classic 4977 bp deletion, and 23 novel deletions were identified in 25 patients. Larger deletions correlated with more deleted MRC complexes (r = 0.516, p = 0.0123) and more deleted tRNAs (r = 0.534, p = 0.0103). Kearns-Sayre syndrome (KSS) patients had later onset (p = 0.0337), larger deletions (p = 0.0263), and greater tRNA/MRC complex (p = 0.0263, p = 0.0319) involvement than non-KSS patients. SLSMD in Chinese children primarily causes KSS, Pearson syndrome (PS), and progressive ophthalmoplegia with multi-organ involvement. Genotype-phenotype correlations exist, particularly between deletion size, onset age, and disease severity. KSS patients show distinct genetic and clinical profiles, suggesting slower progression. This study expands the known SLSMD spectrum and underscores mitochondrial testing in pediatric multi-organ disorders.
期刊介绍:
Clinical Genetics links research to the clinic, translating advances in our understanding of the molecular basis of genetic disease for the practising clinical geneticist. The journal publishes high quality research papers, short reports, reviews and mini-reviews that connect medical genetics research with clinical practice.
Topics of particular interest are:
• Linking genetic variations to disease
• Genome rearrangements and disease
• Epigenetics and disease
• The translation of genotype to phenotype
• Genetics of complex disease
• Management/intervention of genetic diseases
• Novel therapies for genetic diseases
• Developmental biology, as it relates to clinical genetics
• Social science research on the psychological and behavioural aspects of living with or being at risk of genetic disease