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精脒/精胺 N1-乙酰转移酶样 1(SATL1)基因功能丧失性变异是自闭症谱系障碍的潜在病因。

Loss-of-function variant in spermidine/spermine N1-acetyl transferase like 1 (SATL1) gene as an underlying cause of autism spectrum disorder.

机构信息

Department of Basic Medical Sciences, Taibah University Medina, Almadinah Almunawwarah, Saudi Arabia.

Department of Physiology, Faculty of Medicine, King Saud University, Riyadh, Saudi Arabia.

出版信息

Sci Rep. 2024 Mar 8;14(1):5765. doi: 10.1038/s41598-024-56253-5.

DOI:10.1038/s41598-024-56253-5
PMID:38459140
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC10923806/
Abstract

Autism spectrum disorder (ASD) is a complicated, lifelong neurodevelopmental disorder affecting verbal and non-verbal communication and social interactions. ASD signs and symptoms appear early in development before the age of 3 years. It is unlikely for a person to acquire autism after a period of normal development. However, we encountered an 8-year-old child who developed ASD later in life although his developmental milestones were normal at the beginning of life. Sequencing the complete coding part of the genome identified a hemizygous nonsense mutation (NM_001367857.2):c.1803C>G; (p.Tyr601Ter) in the gene (SATL1) encoding spermidine/spermine N1-acetyl transferase like 1. Screening an ASD cohort of 28 isolated patients for the SATL1 gene identified another patient with the same variant. Although SATL1 mutations have not been associated with any human diseases, our data suggests that a mutation in SATL1 is the underlying cause of ASD in our cases. In mammals, mutations in spermine synthase (SMS), an enzyme needed for the synthesis of spermidine polyamine, have been reported in a syndromic form of the X-linked mental retardation. Moreover, SATL1 gene expression studies showed a relatively higher expression of SATL1 transcripts in ASD related parts of the brain including the cerebellum, amygdala and frontal cortex. Additionally, spermidine has been characterized in the context of learning and memory and supplementations with spermidine increase neuroprotective effects and decrease age-induced memory impairment. Furthermore, spermidine biosynthesis is required for spontaneous axonal regeneration and prevents α-synuclein neurotoxicity in invertebrate models. Thus, we report, for the first time, that a mutation in the SATL1 gene could be a contributing factor in the development of autistic symptoms in our patients.

摘要

自闭症谱系障碍 (ASD) 是一种复杂的、终身性神经发育障碍,影响言语和非言语交流以及社交互动。ASD 的症状和体征在 3 岁前的发育早期出现。一个人在正常发育一段时间后不太可能患上自闭症。然而,我们遇到了一个 8 岁的孩子,他在生命早期的发育里程碑正常,但后来出现了自闭症。对基因组的完整编码部分进行测序,确定了一个半合子无义突变 (NM_001367857.2):c.1803C>G; (p.Tyr601Ter) 在基因 (SATL1) 中,该基因编码精脒/精胺 N1-乙酰转移酶样 1。对 28 名孤立 ASD 患者进行 SATL1 基因筛查,发现另一名患者携带相同的变异。尽管 SATL1 突变与任何人类疾病无关,但我们的数据表明,SATL1 中的突变是我们病例中 ASD 的潜在原因。在哺乳动物中,已报道多胺合成酶 (SMS) 中的突变,该酶是精脒合成所必需的,其与 X 连锁智力低下的综合征形式有关。此外,SATL1 基因表达研究显示,SATL1 转录本在与 ASD 相关的大脑区域(包括小脑、杏仁核和额叶皮质)中的表达相对较高。此外,精脒已在学习和记忆的背景下进行了研究,并且补充精脒可以增加神经保护作用并减少年龄引起的记忆障碍。此外,精脒生物合成是自发轴突再生所必需的,并可以防止α-突触核蛋白在无脊椎动物模型中的神经毒性。因此,我们首次报道 SATL1 基因突变可能是我们患者自闭症症状发展的一个因素。

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本文引用的文献

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