Literature DB >> 31175630

Eye Movement Abnormalities Are Ubiquitous in the Spinocerebellar Ataxias.

Christopher D Stephen1,2, Jeremy D Schmahmann3,4,5.   

Abstract

Oculomotor abnormalities are common in the spinocerebellar ataxias (SCAs). In studies of SCAs 1, 2, 3, and 6, eye movement abnormalities correlate with disease severity. Oculomotor abnormalities may be the sole motor manifestation of early and/or premanifest disease; however, not all ataxia rating scales include oculomotor assessment. We sought to identify the prevalence and characteristics of oculomotor abnormalities at first presentation in a large SCA cohort, including those in earlier stages of disease. We performed a retrospective assessment of initial clinical examinations of SCA patients followed in the Massachusetts General Hospital Ataxia Unit and assessed with the Brief Ataxia Rating Scale (BARS). One hundred thirty-four SCA patients were assessed: 17 SCA1, 13 SCA2, 55 SCA3, 2 SCA5, 22 SCA6, 11 SCA7, 9 SCA8, and 5 SCA17, mainly in the early stages of disease (67.2% stage 0-1). Oculomotor abnormalities were present on initial assessment in 94.8%, including 7/9 stage 0 and 77/81 stage 1 patients. Stage 0/1 patients had frequent saccadic intrusions, nystagmus, and hypo/hypermetric saccades. Saccadic slowing was present even in early stage SCA7 and SCA2, eventually leading to ophthalmoplegia. The burden of oculomotor abnormalities correlated with disease stage, duration, and severity, remaining highly significant even when controlling for age. The ubiquitous presence of oculomotor abnormalities in the SCAs, particularly early in the course, underscores the importance of oculomotor assessment in ataxia rating scales such as BARS. These findings highlight the potential for quantitative physiological oculomotor measures as clinical biomarkers in natural history studies and clinical trials.

Entities:  

Keywords:  Clinical neurology examination; Ocular motility, ocular physiological phenomena; Spinocerebellar ataxia

Mesh:

Year:  2019        PMID: 31175630     DOI: 10.1007/s12311-019-01044-2

Source DB:  PubMed          Journal:  Cerebellum        ISSN: 1473-4222            Impact factor:   3.847


  13 in total

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Authors:  Roderick P P W M Maas; Judith van Gaalen; Thomas Klockgether; Bart P C van de Warrenburg
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2.  The natural history of degenerative ataxia: a retrospective study in 466 patients.

Authors:  T Klockgether; R Lüdtke; B Kramer; M Abele; K Bürk; L Schöls; O Riess; F Laccone; S Boesch; I Lopes-Cendes; A Brice; R Inzelberg; N Zilber; J Dichgans
Journal:  Brain       Date:  1998-04       Impact factor: 13.501

3.  Measuring Friedreich ataxia: complementary features of examination and performance measures.

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Journal:  Neurology       Date:  2006-06-13       Impact factor: 9.910

4.  Scale for the assessment and rating of ataxia: development of a new clinical scale.

Authors:  T Schmitz-Hübsch; S Tezenas du Montcel; L Baliko; J Berciano; S Boesch; C Depondt; P Giunti; C Globas; J Infante; J-S Kang; B Kremer; C Mariotti; B Melegh; M Pandolfo; M Rakowicz; P Ribai; R Rola; L Schöls; S Szymanski; B P van de Warrenburg; A Dürr; T Klockgether; Roberto Fancellu
Journal:  Neurology       Date:  2006-06-13       Impact factor: 9.910

5.  Inventory of Non-Ataxia Signs (INAS): validation of a new clinical assessment instrument.

Authors:  H Jacobi; M Rakowicz; R Rola; R Fancellu; C Mariotti; P Charles; A Dürr; M Küper; D Timmann; C Linnemann; L Schöls; O Kaut; C Schaub; A Filla; L Baliko; B Melegh; J-S Kang; P Giunti; B P C van de Warrenburg; R Fimmers; T Klockgether
Journal:  Cerebellum       Date:  2013-06       Impact factor: 3.847

6.  Saccade velocity is reduced in presymptomatic spinocerebellar ataxia type 2.

Authors:  L Velázquez-Pérez; C Seifried; M Abele; F Wirjatijasa; R Rodríguez-Labrada; N Santos-Falcón; G Sánchez-Cruz; L Almaguer-Mederos; R Tejeda; N Canales-Ochoa; M Fetter; U Ziemann; T Klockgether; J Medrano-Montero; J Rodríguez-Díaz; J M Laffita-Mesa; G Auburger
Journal:  Clin Neurophysiol       Date:  2009-02-07       Impact factor: 3.708

7.  Ocular motor characteristics of different subtypes of spinocerebellar ataxia: distinguishing features.

Authors:  Ji Sun Kim; Ji Soo Kim; Jinyoung Youn; Dae-Won Seo; Yuri Jeong; Ji-Hoon Kang; Jeong Ho Park; Jin Whan Cho
Journal:  Mov Disord       Date:  2013-04-22       Impact factor: 10.338

8.  Oculomotor phenotypes in autosomal dominant ataxias.

Authors:  N Buttner; D Geschwind; J C Jen; S Perlman; S M Pulst; R W Baloh
Journal:  Arch Neurol       Date:  1998-10

9.  Cerebellum and ocular motor control.

Authors:  Amir Kheradmand; David S Zee
Journal:  Front Neurol       Date:  2011-09-01       Impact factor: 4.003

10.  Nystagmus as an early ocular alteration in Machado-Joseph disease (MJD/SCA3).

Authors:  Mafalda Raposo; João Vasconcelos; Conceição Bettencourt; Teresa Kay; Paula Coutinho; Manuela Lima
Journal:  BMC Neurol       Date:  2014-01-22       Impact factor: 2.474

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1.  Evolution of the vestibular function during head impulses in spinocerebellar ataxia type 6.

Authors:  Sun-Uk Lee; Ji-Soo Kim; Hyo-Jung Kim; Jeong-Yoon Choi; Ji-Yun Park; Jong-Min Kim; Xu Yang
Journal:  J Neurol       Date:  2020-02-17       Impact factor: 4.849

2.  Quantitative oculomotor and nonmotor assessments in late-onset GM2 gangliosidosis.

Authors:  Christopher D Stephen; David Balkwill; Peter James; Elizabeth Haxton; Kenneth Sassower; Jeremy D Schmahmann; Florian Eichler; Richard Lewis
Journal:  Neurology       Date:  2020-01-21       Impact factor: 9.910

3.  Establish a Nomogram to Predict Falls in Spinocerebellar Ataxia Type 3.

Authors:  Junyu Lin; Lingyu Zhang; Bei Cao; Qianqian Wei; Ruwei Ou; Yanbing Hou; Xinran Xu; Kuncheng Liu; Xiaojing Gu; Huifang Shang
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4.  Abnormal eye movements in spinocerebellar ataxia type 3.

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Authors:  Giulia Coarelli; Thomas Wirth; Christine Tranchant; Michel Koenig; Alexandra Durr; Mathieu Anheim
Journal:  J Neurol       Date:  2022-09-24       Impact factor: 6.682

6.  Accurate detection of cerebellar smooth pursuit eye movement abnormalities via mobile phone video and machine learning.

Authors:  Zhuoqing Chang; Ziyu Chen; Christopher D Stephen; Jeremy D Schmahmann; Hau-Tieng Wu; Guillermo Sapiro; Anoopum S Gupta
Journal:  Sci Rep       Date:  2020-10-29       Impact factor: 4.379

7.  Region-specific preservation of Purkinje cell morphology and motor behavior in the ATXN1[82Q] mouse model of spinocerebellar ataxia 1.

Authors:  Joshua J White; Laurens W J Bosman; Francois G C Blot; Catarina Osório; Bram W Kuppens; Wilhelmina H J J Krijnen; Charlotte Andriessen; Chris I De Zeeuw; Dick Jaarsma; Martijn Schonewille
Journal:  Brain Pathol       Date:  2021-03-16       Impact factor: 6.508

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