清晨好,您是今天最早来到科研通的研友!由于当前在线用户较少,发布求助请尽量完整的填写文献信息,科研通机器人24小时在线,伴您科研之路漫漫前行!

SLC gene mutations and pediatric neurological disorders: diverse clinical phenotypes in a Saudi Arabian population

生物 溶质载体族 肌张力障碍 人口 表型 癫痫 遗传学 生物信息学 基因 医学 神经科学 运输机 环境卫生
作者
Ali Mir,Montaha Almudhry,Fouad Alghamdi,Raidah Albaradie,Mona H. Ibrahim,Fatimah Aldurayhim,Abdullah Alhedaithy,Mushari Alamr,Maryam Bawazir,Sahar Mohammad,Salma Abdelhay,Shahid Bashir,Yousef Housawi
出处
期刊:Human Genetics [Springer Nature]
卷期号:141 (1): 81-99 被引量:19
标识
DOI:10.1007/s00439-021-02404-x
摘要

The uptake and efflux of solutes across a plasma membrane is controlled by transporters. There are two main superfamilies of transporters, adenosine 5'-triphosphate (ATP) binding cassettes (ABCs) and solute carriers (SLCs). In the brain, SLC transporters are involved in transporting various solutes across the blood-brain barrier, blood-cerebrospinal fluid barrier, astrocytes, neurons, and other brain cell types including oligodendrocytes and microglial cells. SLCs play an important role in maintaining normal brain function. Hence, mutations in the genes that encode SLC transporters can cause a variety of neurological disorders. We identified the following SLC gene variants in 25 patients in our cohort: SLC1A2, SLC2A1, SLC5A1, SLC6A3, SLC6A5, SLC6A8, SLC9A6, SLC9A9, SLC12A6, SLC13A5, SLC16A1, SLC17A5, SLC19A3, SLC25A12, SLC25A15, SLC27A4, SLC45A1, SLC46A1, and SLC52A3. Eight patients harbored pathogenic or likely pathogenic mutations (SLC5A1, SLC9A6, SLC12A6, SLC16A1, SLC19A3, and SLC52A3), and 12 patients were found to have variants of unknown clinical significance (VOUS); these variants occurred in 11 genes (SLC1A2, SLC2A1, SLC6A3, SLC6A5, SLC6A8, SLC9A6, SLC9A9, SLC13A5, SLC25A12, SLC27A4, and SLC45A1). Five patients were excluded as they were carriers. In the remaining 20 patients with SLC gene variants, we identified 16 possible distinct neurological disorders. Based on the clinical presentation, we categorized them into genes causing intellectual delay (ID) or autism spectrum disorder (ASD), those causing epilepsy, those causing vitamin-related disorders, and those causing other neurological diseases. Several variants were detected that indicated possible personalized therapies: SLC2A1 led to dystonia or epilepsy, which can be treated with a ketogenic diet; SLC6A3 led to infantile parkinsonism-dystonia 1, which can be treated with levodopa; SLC6A5 led to hyperekplexia 3, for which unnecessary treatment with antiepileptic drugs should be avoided; SLC6A8 led to creatine deficiency syndrome type 1, which can be treated with creatine monohydrate; SLC16A1 led to monocarboxylate transporter 1 deficiency, which causes seizures that should not be treated with a ketogenic diet; SLC19A3 led to biotin-thiamine-responsive basal ganglia disease, which can be treated with biotin and thiamine; and SLC52A3 led to Brown-Vialetto-Van-Laere syndrome 1, which can be treated with riboflavin. The present study examines the prevalence of SLC gene mutations in our cohort of children with epilepsy and other neurological disorders. It highlights the diverse phenotypes associated with mutations in this large family of SLC transporter proteins, and an opportunity for personalized genomics and personalized therapeutics.
最长约 10秒,即可获得该文献文件

科研通智能强力驱动
Strongly Powered by AbleSci AI
科研通是完全免费的文献互助平台,具备全网最快的应助速度,最高的求助完成率。 对每一个文献求助,科研通都将尽心尽力,给求助人一个满意的交代。
实时播报
1秒前
yuntong完成签到 ,获得积分10
7秒前
yzw发布了新的文献求助10
8秒前
月儿完成签到 ,获得积分10
12秒前
朽木完成签到 ,获得积分10
18秒前
深情安青应助rotator采纳,获得10
21秒前
畅快的毛衣应助yzw采纳,获得10
31秒前
万能图书馆应助yzw采纳,获得10
32秒前
48秒前
在水一方应助科研通管家采纳,获得20
48秒前
57秒前
maclogos完成签到,获得积分10
58秒前
焚心结完成签到 ,获得积分0
59秒前
雪花完成签到 ,获得积分10
1分钟前
Young完成签到 ,获得积分10
1分钟前
rotator发布了新的文献求助10
1分钟前
1分钟前
Neew完成签到 ,获得积分10
1分钟前
迅速的幻雪完成签到 ,获得积分10
1分钟前
蒋灵馨完成签到 ,获得积分10
1分钟前
科研临床两手抓完成签到 ,获得积分10
1分钟前
naczx完成签到,获得积分0
1分钟前
露露完成签到 ,获得积分10
1分钟前
怕黑道消完成签到 ,获得积分10
2分钟前
digger2023完成签到 ,获得积分10
2分钟前
乐仔发布了新的文献求助10
2分钟前
chcmy完成签到 ,获得积分0
2分钟前
hqq完成签到,获得积分10
2分钟前
自然的含蕾完成签到 ,获得积分10
2分钟前
风起枫落完成签到 ,获得积分10
2分钟前
2分钟前
xuli21315完成签到 ,获得积分10
2分钟前
枫林摇曳完成签到 ,获得积分10
2分钟前
闲云野鹤发布了新的文献求助10
2分钟前
宇文雨文完成签到 ,获得积分10
2分钟前
2分钟前
闲云野鹤完成签到,获得积分10
3分钟前
今天开心吗完成签到 ,获得积分10
3分钟前
李爱国应助乐仔采纳,获得10
3分钟前
ananan完成签到,获得积分10
3分钟前
高分求助中
Continuum thermodynamics and material modelling 3000
Production Logging: Theoretical and Interpretive Elements 2500
Healthcare Finance: Modern Financial Analysis for Accelerating Biomedical Innovation 2000
Applications of Emerging Nanomaterials and Nanotechnology 1111
Covalent Organic Frameworks 1000
Les Mantodea de Guyane Insecta, Polyneoptera 1000
Theory of Block Polymer Self-Assembly 750
热门求助领域 (近24小时)
化学 医学 材料科学 生物 工程类 有机化学 生物化学 纳米技术 内科学 物理 化学工程 计算机科学 复合材料 基因 遗传学 物理化学 催化作用 细胞生物学 免疫学 电极
热门帖子
关注 科研通微信公众号,转发送积分 3477509
求助须知:如何正确求助?哪些是违规求助? 3068936
关于积分的说明 9110233
捐赠科研通 2760462
什么是DOI,文献DOI怎么找? 1514928
邀请新用户注册赠送积分活动 700486
科研通“疑难数据库(出版商)”最低求助积分说明 699617