Literature DB >> 20739808

Clinical and genetic study of a Chinese family with spinocerebellar ataxia type 7.

Yan Han1, Benqiang Deng, Mingyuan Liu, Jianming Jiang, Shuai Wu, Yangtai Guan.   

Abstract

UNLABELLED: Spinocerebellar ataxia 7 (SCA7) is a rare disease, and only few SCA7 families have been reported, especially from East Asia. Clinical features of a genetically confirmed SCA7 Chinese family were evaluated. The onset of the disease varied from 4 years to 48 years, and the initial presenting feature was cerebellar ataxia or visual impairment, or both. There were abnormal findings on fundus photography, electroretinogram, flash visual evoked potential and oscillatory potentials. Abnormal mitochondria were also found in skeletal muscle or liver biopsies. The number of cytosine adenine guanine (CAG) repeats ranged from 50 to 97, and the length of CAG repeat was inversely correlated with the age of onset (r=-0.867, P=0.025).
CONCLUSION: The clinical manifestations and SCA7 gene of SCA7 patients were homogeneous in this study. Larger CAG repeats had not only resulted in earlier onset, but also related to the rapid progression and severity of the disease. Abnormal mitochondria may be a common finding in biopsy studies of various organs in SCA7 patients.

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Year:  2010        PMID: 20739808     DOI: 10.4103/0028-3886.68674

Source DB:  PubMed          Journal:  Neurol India        ISSN: 0028-3886            Impact factor:   2.117


  4 in total

1.  Spinocerebellar ataxia type 7: Report of an Indian family.

Authors:  Gurusidheshwar M Wali
Journal:  Ann Indian Acad Neurol       Date:  2013-10       Impact factor: 1.383

2.  Metabolic and Organelle Morphology Defects in Mice and Human Patients Define Spinocerebellar Ataxia Type 7 as a Mitochondrial Disease.

Authors:  Jacqueline M Ward; Colleen A Stoyas; Pawel M Switonski; Farid Ichou; Weiwei Fan; Brett Collins; Christopher E Wall; Isaac Adanyeguh; Chenchen Niu; Bryce L Sopher; Chizuru Kinoshita; Richard S Morrison; Alexandra Durr; Alysson R Muotri; Ronald M Evans; Fanny Mochel; Albert R La Spada
Journal:  Cell Rep       Date:  2019-01-29       Impact factor: 9.423

Review 3.  The Molecular Basis of Spinocerebellar Ataxia Type 7.

Authors:  Rituparna Goswami; Abudu I Bello; Joe Bean; Kara M Costanzo; Bwaar Omer; Dayanne Cornelio-Parra; Revan Odah; Amit Ahluwalia; Shefaa K Allan; Nghi Nguyen; Taylor Shores; N Ahmad Aziz; Ryan D Mohan
Journal:  Front Neurosci       Date:  2022-03-24       Impact factor: 4.677

4.  Functional characterization of variants of unknown significance in a spinocerebellar ataxia patient using an unsupervised machine learning pipeline.

Authors:  Siddharth Nath; Nicholas S Caron; Linda May; Oxana B Gluscencova; Jill Kolesar; Lauren Brady; Brett A Kaufman; Gabrielle L Boulianne; Amadeo R Rodriguez; Mark A Tarnopolsky; Ray Truant
Journal:  Hum Genome Var       Date:  2022-04-14
  4 in total

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