Literature DB >> 33836316

Rating scales and biomarkers for CAG-repeat spinocerebellar ataxias: Implications for therapy development.

Meng-Ling Chen1, Chih-Chun Lin1, Liana S Rosenthal2, Puneet Opal3, Sheng-Han Kuo4.   

Abstract

Spinocerebellar ataxias (SCAs) are a group of dominantly-inherited cerebellar ataxias, among which CAG expansion-related SCAs are most common. These diseases have very high penetrance with defined disease progression, and emerging therapies are being developed to provide either symptomatic or disease-modifying benefits. In clinical trial design, it is crucial to incorporate biomarkers to test target engagement or track disease progression in response to therapies, especially in rare diseases such as SCAs. In this article, we review the available rating scales and recent advances of biomarkers in CAG-repeat SCAs. We divided biomarkers into neuroimaging, body fluid, and physiological studies. Understanding the utility of each biomarker will facilitate the design of robust clinical trials to advance therapies for SCAs.
Copyright © 2021 Elsevier B.V. All rights reserved.

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Year:  2021        PMID: 33836316      PMCID: PMC8111569          DOI: 10.1016/j.jns.2021.117417

Source DB:  PubMed          Journal:  J Neurol Sci        ISSN: 0022-510X            Impact factor:   3.181


  141 in total

1.  Selection and coordination of human locomotor forms following cerebellar damage.

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Journal:  J Neurophysiol       Date:  2001-02       Impact factor: 2.714

2.  Vestibulo-ocular reflex dynamics with head-impulses discriminates spinocerebellar ataxias types 1, 2 and 3 and Friedreich ataxia.

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Journal:  J Vestib Res       Date:  2016-07-02       Impact factor: 2.435

3.  The cerebellum does more than sensory prediction error-based learning in sensorimotor adaptation tasks.

Authors:  Peter A Butcher; Richard B Ivry; Sheng-Han Kuo; David Rydz; John W Krakauer; Jordan A Taylor
Journal:  J Neurophysiol       Date:  2017-06-21       Impact factor: 2.714

4.  Neurochemical abnormalities in premanifest and early spinocerebellar ataxias.

Authors:  James M Joers; Dinesh K Deelchand; Tianmeng Lyu; Uzay E Emir; Diane Hutter; Christopher M Gomez; Khalaf O Bushara; Lynn E Eberly; Gülin Öz
Journal:  Ann Neurol       Date:  2018-04-10       Impact factor: 10.422

5.  Atrophy of the cerebellum and brainstem in dentatorubral pallidoluysian atrophy. Influence of CAG repeat size on MRI findings.

Authors:  R Koide; O Onodera; T Ikeuchi; R Kondo; H Tanaka; S Tokiguchi; A Tomoda; T Miike; F Isa; H Beppu; N Shimizu; Y Watanabe; Y Horikawa; T Shimohata; K Hirota; A Ishikawa; S Tsuji
Journal:  Neurology       Date:  1997-12       Impact factor: 9.910

6.  Spinal cord damage in Machado-Joseph disease.

Authors:  Camila N Fahl; Lucas Melo T Branco; Felipe P G Bergo; Anelyssa D'Abreu; Iscia Lopes-Cendes; Marcondes C França
Journal:  Cerebellum       Date:  2015-04       Impact factor: 3.847

7.  Serum insulin-like system alterations in patients with spinocerebellar ataxia type 3.

Authors:  Jonas Alex Morales Saute; Andrew Chaves Feitosa da Silva; Alexandre Pastoris Muller; Gisele Hansel; Alexandre Silva de Mello; Fábio Maeda; Leonardo Vedolin; Maria Luiza Saraiva-Pereira; Diogo Onofre Souza; Javier Arpa; Ignacio Torres-Aleman; Luis Valmor Cruz Portela; Laura Bannach Jardim
Journal:  Mov Disord       Date:  2010-11-10       Impact factor: 10.338

8.  Wide Profiling of Circulating MicroRNAs in Spinocerebellar Ataxia Type 7.

Authors:  Verónica M Borgonio-Cuadra; Claudia Valdez-Vargas; Sandra Romero-Córdoba; Alfredo Hidalgo-Miranda; Yessica Tapia-Guerrero; César M Cerecedo-Zapata; Oscar Hernández-Hernández; Bulmaro Cisneros; Jonathan J Magaña
Journal:  Mol Neurobiol       Date:  2019-02-05       Impact factor: 5.590

9.  Quantitative assessment of cerebral blood flow in genetically confirmed spinocerebellar ataxia type 6.

Authors:  Kie Honjo; Tomohiko Ohshita; Hideshi Kawakami; Hiromitsu Naka; Yukari Imon; Hirofumi Maruyama; Yasuyo Mimori; Masayasu Matsumoto
Journal:  Arch Neurol       Date:  2004-06

10.  Clinical characteristics of patients with spinocerebellar ataxias 1, 2, 3 and 6 in the US; a prospective observational study.

Authors:  Tetsuo Ashizawa; Karla P Figueroa; Susan L Perlman; Christopher M Gomez; George R Wilmot; Jeremy D Schmahmann; Sarah H Ying; Theresa A Zesiewicz; Henry L Paulson; Vikram G Shakkottai; Khalaf O Bushara; Sheng-Han Kuo; Michael D Geschwind; Guangbin Xia; Pietro Mazzoni; Jeffrey P Krischer; David Cuthbertson; Amy Roberts Holbert; John H Ferguson; Stefan M Pulst; S H Subramony
Journal:  Orphanet J Rare Dis       Date:  2013-11-13       Impact factor: 4.123

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  3 in total

Review 1.  Current and emerging treatment modalities for spinocerebellar ataxias.

Authors:  Shaila D Ghanekar; Sheng-Han Kuo; Joseph S Staffetti; Theresa A Zesiewicz
Journal:  Expert Rev Neurother       Date:  2022-02-10       Impact factor: 4.287

2.  Brain structural abnormalities in the preclinical stage of Machado-Joseph disease/spinocerebellar ataxia type 3 (MJD/SCA3): evaluation by MRI morphometry, diffusion tensor imaging and neurite orientation dispersion and density imaging.

Authors:  Mengcheng Li; Xinyuan Chen; Hao-Ling Xu; Ziqiang Huang; Naping Chen; Yuqing Tu; Shirui Gan; Jianping Hu
Journal:  J Neurol       Date:  2021-11-16       Impact factor: 6.682

Review 3.  Spinocerebellar ataxia clinical trials: opportunities and challenges.

Authors:  Sarah M Brooker; Chandrakanth Reddy Edamakanti; Sara M Akasha; Sheng-Han Kuo; Puneet Opal
Journal:  Ann Clin Transl Neurol       Date:  2021-05-21       Impact factor: 4.511

  3 in total

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