Literature DB >> 28617590

Structure and Dynamics of RNA Repeat Expansions That Cause Huntington's Disease and Myotonic Dystrophy Type 1.

Jonathan L Chen1, Damian M VanEtten2, Matthew A Fountain2, Ilyas Yildirim1,3, Matthew D Disney1,3.   

Abstract

RNA repeat expansions cause a host of incurable, genetically defined diseases. The most common class of RNA repeats consists of trinucleotide repeats. These long, repeating transcripts fold into hairpins containing 1 × 1 internal loops that can mediate disease via a variety of mechanism(s) in which RNA is the central player. Two of these disorders are Huntington's disease and myotonic dystrophy type 1, which are caused by r(CAG) and r(CUG) repeats, respectively. We report the structures of two RNA constructs containing three copies of a r(CAG) [r(3×CAG)] or r(CUG) [r(3×CUG)] motif that were modeled with nuclear magnetic resonance spectroscopy and simulated annealing with restrained molecular dynamics. The 1 × 1 internal loops of r(3×CAG) are stabilized by one-hydrogen bond (cis Watson-Crick/Watson-Crick) AA pairs, while those of r(3×CUG) prefer one- or two-hydrogen bond (cis Watson-Crick/Watson-Crick) UU pairs. Assigned chemical shifts for the residues depended on the identity of neighbors or next nearest neighbors. Additional insights into the dynamics of these RNA constructs were gained by molecular dynamics simulations and a discrete path sampling method. Results indicate that the global structures of the RNA are A-form and that the loop regions are dynamic. The results will be useful for understanding the dynamic trajectory of these RNA repeats but also may aid in the development of therapeutics.

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Year:  2017        PMID: 28617590      PMCID: PMC5810133          DOI: 10.1021/acs.biochem.7b00252

Source DB:  PubMed          Journal:  Biochemistry        ISSN: 0006-2960            Impact factor:   3.162


  88 in total

1.  Design of a bioactive small molecule that targets the myotonic dystrophy type 1 RNA via an RNA motif-ligand database and chemical similarity searching.

Authors:  Raman Parkesh; Jessica L Childs-Disney; Masayuki Nakamori; Amit Kumar; Eric Wang; Thomas Wang; Jason Hoskins; Tuan Tran; David Housman; Charles A Thornton; Matthew D Disney
Journal:  J Am Chem Soc       Date:  2012-03-05       Impact factor: 15.419

2.  Expansion of a CUG trinucleotide repeat in the 3' untranslated region of myotonic dystrophy protein kinase transcripts results in nuclear retention of transcripts.

Authors:  B M Davis; M E McCurrach; K L Taneja; R H Singer; D E Housman
Journal:  Proc Natl Acad Sci U S A       Date:  1997-07-08       Impact factor: 11.205

3.  Mexiletine is an effective antimyotonia treatment in myotonic dystrophy type 1.

Authors:  E L Logigian; W B Martens; R T Moxley; M P McDermott; N Dilek; A W Wiegner; A T Pearson; C A Barbieri; C L Annis; C A Thornton; R T Moxley
Journal:  Neurology       Date:  2010-05-04       Impact factor: 9.910

4.  NMRPipe: a multidimensional spectral processing system based on UNIX pipes.

Authors:  F Delaglio; S Grzesiek; G W Vuister; G Zhu; J Pfeifer; A Bax
Journal:  J Biomol NMR       Date:  1995-11       Impact factor: 2.835

5.  Aberrant regulation of insulin receptor alternative splicing is associated with insulin resistance in myotonic dystrophy.

Authors:  R S Savkur; A V Philips; T A Cooper
Journal:  Nat Genet       Date:  2001-09       Impact factor: 38.330

6.  Actinomycin D Specifically Reduces Expanded CUG Repeat RNA in Myotonic Dystrophy Models.

Authors:  Ruth B Siboni; Masayuki Nakamori; Stacey D Wagner; Adam J Struck; Leslie A Coonrod; Shanee A Harriott; Daniel M Cass; Matthew K Tanner; J Andrew Berglund
Journal:  Cell Rep       Date:  2015-12-10       Impact factor: 9.423

Review 7.  Molecular therapy in myotonic dystrophy: focus on RNA gain-of-function.

Authors:  Susan A M Mulders; Baziel G M van Engelen; Bé Wieringa; Derick G Wansink
Journal:  Hum Mol Genet       Date:  2010-04-20       Impact factor: 6.150

8.  Precise small-molecule recognition of a toxic CUG RNA repeat expansion.

Authors:  Suzanne G Rzuczek; Lesley A Colgan; Yoshio Nakai; Michael D Cameron; Denis Furling; Ryohei Yasuda; Matthew D Disney
Journal:  Nat Chem Biol       Date:  2016-12-12       Impact factor: 15.040

9.  The nuclear RNase III Drosha initiates microRNA processing.

Authors:  Yoontae Lee; Chiyoung Ahn; Jinju Han; Hyounjeong Choi; Jaekwang Kim; Jeongbin Yim; Junho Lee; Patrick Provost; Olof Rådmark; Sunyoung Kim; V Narry Kim
Journal:  Nature       Date:  2003-09-25       Impact factor: 49.962

10.  DSSR: an integrated software tool for dissecting the spatial structure of RNA.

Authors:  Xiang-Jun Lu; Harmen J Bussemaker; Wilma K Olson
Journal:  Nucleic Acids Res       Date:  2015-07-15       Impact factor: 16.971

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  5 in total

1.  Computational Investigation of RNA A-Bulges Related to the Microtubule-Associated Protein Tau Causing Frontotemporal Dementia and Parkinsonism.

Authors:  David J Wales; Matthew D Disney; Ilyas Yildirim
Journal:  J Phys Chem B       Date:  2019-01-02       Impact factor: 2.991

Review 2.  RNA Structural Dynamics As Captured by Molecular Simulations: A Comprehensive Overview.

Authors:  Jiří Šponer; Giovanni Bussi; Miroslav Krepl; Pavel Banáš; Sandro Bottaro; Richard A Cunha; Alejandro Gil-Ley; Giovanni Pinamonti; Simón Poblete; Petr Jurečka; Nils G Walter; Michal Otyepka
Journal:  Chem Rev       Date:  2018-01-03       Impact factor: 60.622

3.  CAG RNAs induce DNA damage and apoptosis by silencing NUDT16 expression in polyglutamine degeneration.

Authors:  Shaohong Peng; Pei Guo; Xiao Lin; Ying An; Kong Hung Sze; Matthew Ho Yan Lau; Zhefan Stephen Chen; Qianwen Wang; Wen Li; Jacquelyne Ka-Li Sun; Sum Yi Ma; Ting-Fung Chan; Kwok-Fai Lau; Jacky Chi Ki Ngo; Kin Ming Kwan; Chun-Ho Wong; Sik Lok Lam; Steven C Zimmerman; Tiziano Tuccinardi; Zhong Zuo; Ho Yu Au-Yeung; Hei-Man Chow; Ho Yin Edwin Chan
Journal:  Proc Natl Acad Sci U S A       Date:  2021-05-11       Impact factor: 11.205

4.  Computational Investigation of Bending Properties of RNA AUUCU, CCUG, CAG, and CUG Repeat Expansions Associated With Neuromuscular Disorders.

Authors:  Amirhossein Taghavi; Ilyas Yildirim
Journal:  Front Mol Biosci       Date:  2022-04-11

5.  Design of a small molecule that stimulates vascular endothelial growth factor A enabled by screening RNA fold-small molecule interactions.

Authors:  Hafeez S Haniff; Laurent Knerr; Xiaohui Liu; Gogce Crynen; Jonas Boström; Daniel Abegg; Alexander Adibekian; Elizabeth Lekah; Kye Won Wang; Michael D Cameron; Ilyas Yildirim; Malin Lemurell; Matthew D Disney
Journal:  Nat Chem       Date:  2020-08-24       Impact factor: 24.427

  5 in total

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