Literature DB >> 19811945

Caring for Machado-Joseph disease: current understanding and how to help patients.

Anelyssa D'Abreu1, Marcondes C França, Henry L Paulson, Iscia Lopes-Cendes.   

Abstract

Machado-Joseph disease or spinocerebellar ataxia 3 (MJD/SCA3) is a clinically heterogeneous, neurodegenerative disorder characterized by varying degrees of ataxia, ophthalmoplegia, peripheral neuropathy, pyramidal dysfunction and movement disorder. MJD/SCA3 is caused by a CAG repeat expansion mutation in the protein coding region of the ATXN3 gene located at chromosome 14q32.1. Current hypotheses regarding pathogenesis favor the view that mutated ataxin-3, with its polyglutamine expansion, is prone to adopt an abnormal conformation, engage in altered protein-protein interactions and aggregate. Expanded CAG repeat length correlates with the range and severity of the clinical manifestations and inversely correlates with age of disease onset. Though MJD/SCA3 is classically described as affecting the cerebellum, brainstem and basal ganglia, recent neuropathology and neuroimaging series demonstrate involvement of other areas such as the thalamus and cerebral cortex. Clinically, much emphasis has been placed in the description and recognition of the non-motor symptoms observed in these patients, such as pain, cramps, fatigue and depression. Currently, no disease modifying treatment exists for MJD/SCA3. Standard of care includes genetic counseling, exercise/physical therapy programs, and speech and swallow evaluation. Symptomatic treatment for clinical findings such as depression, sleep disorders, parkinsonism, dystonia, cramps, and pain is important to improve the quality of life for those with MJD/SCA3.

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Year:  2009        PMID: 19811945      PMCID: PMC2818316          DOI: 10.1016/j.parkreldis.2009.08.012

Source DB:  PubMed          Journal:  Parkinsonism Relat Disord        ISSN: 1353-8020            Impact factor:   4.891


  49 in total

1.  A familial factor independent of CAG repeat length influences age at onset of Machado-Joseph disease.

Authors:  A L DeStefano; L A Cupples; P Maciel; C Gaspar; J Radvany; D M Dawson; L Sudarsky; L Corwin; P Coutinho; P MacLeod
Journal:  Am J Hum Genet       Date:  1996-07       Impact factor: 11.025

2.  Azorean disease of the nervous system.

Authors:  F C Romanul; H L Fowler; J Radvany; R G Feldman; M Feingold
Journal:  N Engl J Med       Date:  1977-06-30       Impact factor: 91.245

3.  Autosomal dominant striatonigral degeneration. A clinical, pathologic, and biochemical study of a new genetic disorder.

Authors:  R N Rosenberg; W L Nyhan; C Bay; P Shore
Journal:  Neurology       Date:  1976-08       Impact factor: 9.910

4.  Positron emission tomography in asymptomatic gene carriers of Machado-Joseph disease.

Authors:  B W Soong; R S Liu
Journal:  J Neurol Neurosurg Psychiatry       Date:  1998-04       Impact factor: 10.154

5.  Characteristic magnetic resonance imaging findings in Machado-Joseph disease.

Authors:  Y Murata; S Yamaguchi; H Kawakami; Y Imon; H Maruyama; T Sakai; T Kazuta; T Ohtake; M Nishimura; T Saida; S Chiba; T Oh-i; S Nakamura
Journal:  Arch Neurol       Date:  1998-01

6.  Progressive atrophy of cerebellum and brainstem as a function of age and the size of the expanded CAG repeats in the MJD1 gene in Machado-Joseph disease.

Authors:  O Onodera; J Idezuka; S Igarashi; Y Takiyama; K Endo; H Takano; M Oyake; H Tanaka; T Inuzuka; T Hayashi; T Yuasa; J Ito; T Miyatake; S Tsuji
Journal:  Ann Neurol       Date:  1998-03       Impact factor: 10.422

7.  Positron emission tomography (PET) in Machado-Joseph disease.

Authors:  T Taniwaki; T Sakai; T Kobayashi; Y Kuwabara; M Otsuka; Y Ichiya; K Masuda; I Goto
Journal:  J Neurol Sci       Date:  1997-01       Impact factor: 3.181

8.  CAG expansions in a novel gene for Machado-Joseph disease at chromosome 14q32.1.

Authors:  Y Kawaguchi; T Okamoto; M Taniwaki; M Aizawa; M Inoue; S Katayama; H Kawakami; S Nakamura; M Nishimura; I Akiguchi
Journal:  Nat Genet       Date:  1994-11       Impact factor: 38.330

9.  Molecular features of the CAG repeats and clinical manifestation of Machado-Joseph disease.

Authors:  H Maruyama; S Nakamura; Z Matsuyama; T Sakai; M Doyu; G Sobue; M Seto; M Tsujihata; T Oh-i; T Nishio
Journal:  Hum Mol Genet       Date:  1995-05       Impact factor: 6.150

10.  Machado-Joseph disease: A proposal of spastic paraplegic subtype.

Authors:  T Sakai; H Kawakami
Journal:  Neurology       Date:  1996-03       Impact factor: 9.910

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  20 in total

Review 1.  Neurological diseases and pain.

Authors:  David Borsook
Journal:  Brain       Date:  2011-11-08       Impact factor: 13.501

2.  Sleep disorders in machado-joseph disease: frequency, discriminative thresholds, predictive values, and correlation with ataxia-related motor and non-motor features.

Authors:  José Luiz Pedroso; Pedro Braga-Neto; André Carvalho Felício; Lívia Almeida Dutra; William A C Santos; Gilmar Fernandes do Prado; Orlando Graziani Povoas Barsottini
Journal:  Cerebellum       Date:  2011-06       Impact factor: 3.847

Review 3.  Machado-Joseph Disease: from first descriptions to new perspectives.

Authors:  Conceição Bettencourt; Manuela Lima
Journal:  Orphanet J Rare Dis       Date:  2011-06-02       Impact factor: 4.123

4.  Dysautonomia is frequent in Machado-Joseph disease: clinical and neurophysiological evaluation.

Authors:  Karen A G Takazaki; Anelyssa D'Abreu; Anamarli Nucci; Iscia Lopes-Cendes; Marcondes C França
Journal:  Cerebellum       Date:  2013-08       Impact factor: 3.847

Review 5.  Current concepts in the treatment of hereditary ataxias.

Authors:  Pedro Braga Neto; José Luiz Pedroso; Sheng-Han Kuo; C França Marcondes Junior; Hélio Afonso Ghizoni Teive; Orlando Graziani Povoas Barsottini
Journal:  Arq Neuropsiquiatr       Date:  2016-03       Impact factor: 1.420

6.  Serotonergic signalling suppresses ataxin 3 aggregation and neurotoxicity in animal models of Machado-Joseph disease.

Authors:  Andreia Teixeira-Castro; Ana Jalles; Sofia Esteves; Soosung Kang; Liliana da Silva Santos; Anabela Silva-Fernandes; Mário F Neto; Renée M Brielmann; Carlos Bessa; Sara Duarte-Silva; Adriana Miranda; Stéphanie Oliveira; Andreia Neves-Carvalho; João Bessa; Teresa Summavielle; Richard B Silverman; Pedro Oliveira; Richard I Morimoto; Patrícia Maciel
Journal:  Brain       Date:  2015-09-15       Impact factor: 13.501

7.  Nonmotor Symptoms in Patients with Spinocerebellar Ataxia Type 10.

Authors:  Adriana Moro; Renato P Munhoz; Mariana Moscovich; Walter O Arruda; Salmo Raskin; Laura Silveira-Moriyama; Tetsuo Ashizawa; Hélio A G Teive
Journal:  Cerebellum       Date:  2017-12       Impact factor: 3.847

8.  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

Review 9.  Pathogenic mechanisms and therapeutic strategies in spinobulbar muscular atrophy.

Authors:  Jason P Chua; Andrew P Lieberman
Journal:  CNS Neurol Disord Drug Targets       Date:  2013-12       Impact factor: 4.388

10.  Psychological well-being and family satisfaction levels five years after being confirmed as a carrier of the Machado-Joseph disease mutation.

Authors:  Carlos Gonzalez; Elisabete Gomes; Nadiya Kazachkova; Conceição Bettencourt; Mafalda Raposo; Teresa Taylor Kay; Patrick MacLeod; João Vasconcelos; Manuela Lima
Journal:  Genet Test Mol Biomarkers       Date:  2012-12
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