Insights into the role of ribonuclease 4 polymorphisms in amyotrophic lateral sclerosis

被引:11
作者
Padhi, Aditya K. [1 ,2 ]
Narain, Priyam [1 ]
Dave, Upma [1 ]
Satija, Rohit [1 ]
Patir, Anirudh [1 ]
Gomes, James [1 ]
机构
[1] Indian Inst Technol Delhi, Kusuma Sch Biol Sci, Block 1A,Room 307,Hauz Khas, New Delhi 110016, India
[2] RIKEN, Ctr Life Sci Technol, Div Struct & Synthet Biol, Struct Bioinformat Team,Tsurumi Ku, 1-7-22 Suehiro Cho, Yokohama, Kanagawa 2300045, Japan
关键词
amyotrophic lateral sclerosis; loss-of-functions; molecular dynamics; polymorphism; RNA processing pathway; ribonuclease; 4; ANGIOGENIN VARIANTS; MOLECULAR-DYNAMICS; MUTATIONS; INTEGRATION; SURVIVAL;
D O I
10.1080/07391102.2017.1419147
中图分类号
Q5 [生物化学]; Q7 [分子生物学];
学科分类号
071010 ; 081704 ;
摘要
Mutations in certain genes of the Ribonuclease (RNASE) superfamily can cause amyotrophic lateral sclerosis (ALS) through altered RNA processing mechanisms. About 30 of these missense mutations in RNASE5/ANG gene have already been reported in ALS patients. In another gene of the ribonuclease superfamily, ribonuclease 4 (RNASE4), missense mutations and single nucleotide polymorphisms have been identified in patients suffering from ALS. However, their plausible molecular mechanisms of association with ALS are not known. Here, we present the molecular mechanisms of RNASE4 polymorphisms with ALS using all-atom molecular dynamics (MD) simulations followed by functional assay experiments. As most ALS causing mutations in RNASE superfamily proteins affect either the ribonucleolytic or nuclear translocation activity, we examined these functional properties of wild-type and known RNASE4 variants, R10W, A98V, E48D and V75I, using MD simulations. Our simulation predicted that these variants would retain nuclear translocation activity and that E48D would exhibit loss of ribonucleolytic activity, which was subsequently validated by ribonucleolytic assay. Our results give a mechanistic insight into the association of RNASE4 polymorphisms with ALS and show that E48D-RNASE4 would probably be deleterious and cause ALS in individuals harbouring this polymorphism.
引用
收藏
页码:116 / 130
页数:15
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