Literature DB >> 24646433

Personalized gene silencing therapeutics for Huntington disease.

C Kay1, N H Skotte, A L Southwell, M R Hayden.   

Abstract

Gene silencing offers a novel therapeutic strategy for dominant genetic disorders. In specific diseases, selective silencing of only one copy of a gene may be advantageous over non-selective silencing of both copies. Huntington disease (HD) is an autosomal dominant disorder caused by an expanded CAG trinucleotide repeat in the Huntingtin gene (HTT). Silencing both expanded and normal copies of HTT may be therapeutically beneficial, but preservation of normal HTT expression is preferred. Allele-specific methods can selectively silence the mutant HTT transcript by targeting either the expanded CAG repeat or single nucleotide polymorphisms (SNPs) in linkage disequilibrium with the expansion. Both approaches require personalized treatment strategies based on patient genotypes. We compare the prospect of safe treatment of HD by CAG- and SNP-specific silencing approaches and review HD population genetics used to guide target identification in the patient population. Clinical implementation of allele-specific HTT silencing faces challenges common to personalized genetic medicine, requiring novel solutions from clinical scientists and regulatory authorities.
© 2014 John Wiley & Sons A/S. Published by John Wiley & Sons Ltd.

Entities:  

Keywords:  Huntington disease; SNPs; antisense oligonucleotides; gene silencing; haplotypes; personalized medicine; population genetics; single-stranded siRNA; trinucleotide repeat

Mesh:

Substances:

Year:  2014        PMID: 24646433     DOI: 10.1111/cge.12385

Source DB:  PubMed          Journal:  Clin Genet        ISSN: 0009-9163            Impact factor:   4.438


  25 in total

Review 1.  Huntington Disease: Linking Pathogenesis to the Development of Experimental Therapeutics.

Authors:  Tiago A Mestre; Cristina Sampaio
Journal:  Curr Neurol Neurosci Rep       Date:  2017-02       Impact factor: 5.081

Review 2.  Toward precision medicine in amyotrophic lateral sclerosis.

Authors:  Zhang-Yu Zou; Chang-Yun Liu; Chun-Hui Che; Hua-Pin Huang
Journal:  Ann Transl Med       Date:  2016-01

3.  A Comprehensive Haplotype-Targeting Strategy for Allele-Specific HTT Suppression in Huntington Disease.

Authors:  Chris Kay; Jennifer A Collins; Nicholas S Caron; Luciana de Andrade Agostinho; Hailey Findlay-Black; Lorenzo Casal; Dulika Sumathipala; Vajira H W Dissanayake; Mario Cornejo-Olivas; Fiona Baine; Amanda Krause; Jacquie L Greenberg; Carmen Lúcia Antão Paiva; Ferdinando Squitieri; Michael R Hayden
Journal:  Am J Hum Genet       Date:  2019-11-07       Impact factor: 11.025

4.  The targetable A1 Huntington disease haplotype has distinct Amerindian and European origins in Latin America.

Authors:  Chris Kay; Indira Tirado-Hurtado; Mario Cornejo-Olivas; Jennifer A Collins; Galen Wright; Miguel Inca-Martinez; Diego Veliz-Otani; Maria E Ketelaar; Ramy A Slama; Colin J Ross; Pilar Mazzetti; Michael R Hayden
Journal:  Eur J Hum Genet       Date:  2016-12-21       Impact factor: 4.246

Review 5.  Translation of MicroRNA-Based Huntingtin-Lowering Therapies from Preclinical Studies to the Clinic.

Authors:  Jana Miniarikova; Melvin M Evers; Pavlina Konstantinova
Journal:  Mol Ther       Date:  2018-02-08       Impact factor: 11.454

6.  RNAi-Based GluN3A Silencing Prevents and Reverses Disease Phenotypes Induced by Mutant huntingtin.

Authors:  Sonia Marco; Alvaro Murillo; Isabel Pérez-Otaño
Journal:  Mol Ther       Date:  2018-06-15       Impact factor: 11.454

Review 7.  Huntington's Disease-Update on Treatments.

Authors:  Kara J Wyant; Andrew J Ridder; Praveen Dayalu
Journal:  Curr Neurol Neurosci Rep       Date:  2017-04       Impact factor: 5.081

Review 8.  Non-coding RNAs as drug targets.

Authors:  Masayuki Matsui; David R Corey
Journal:  Nat Rev Drug Discov       Date:  2016-07-22       Impact factor: 84.694

9.  Genetic characterization of Spinocerebellar ataxia 1 in a South Indian cohort.

Authors:  Dhanya Kumaran; Krishnan Balagopal; Reginald George Alex Tharmaraj; Sanjith Aaron; Kuryan George; Jayaprakash Muliyil; Ajith Sivadasan; Sumita Danda; Mathew Alexander; Gaiti Hasan
Journal:  BMC Med Genet       Date:  2014-10-25       Impact factor: 2.103

Review 10.  RNA-based therapies for genodermatoses.

Authors:  Olivier Bornert; Patricia Peking; Jeroen Bremer; Ulrich Koller; Peter C van den Akker; Annemieke Aartsma-Rus; Anna M G Pasmooij; Eva M Murauer; Alexander Nyström
Journal:  Exp Dermatol       Date:  2017-01       Impact factor: 3.960

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