Literature DB >> 19562770

Expanding the clinical phenotype of SNCA duplication carriers.

Kenya Nishioka1, Owen A Ross, Kenji Ishii, Jennifer M Kachergus, Kiichi Ishiwata, Mayumi Kitagawa, Satoshi Kono, Tomokazu Obi, Koichi Mizoguchi, Yuichi Inoue, Hisamasa Imai, Masashi Takanashi, Yoshikuni Mizuno, Matthew J Farrer, Nobutaka Hattori.   

Abstract

SNCA duplication is a recognized cause of familial Parkinson's disease (PD). We aimed to explore the genetic and clinical variability in the disease manifestation. Molecular characterization was performed using real-time PCR, SNP arrays, and haplotype analysis. We further studied those patients who were found to harbor SNCA duplication with olfactory function tests, polysomnography, and PET. We identified four new families and one sporadic patient with SNCA duplication. Eleven symptomatic patients from these four families presented with parkinsonism, of which three subsequently developed dementia. The lifetime estimate of overall penetrance was 43.8%. FDG-PET study of symptomatic patients showed hypometabolism in the occipital lobe, whereas asymptomatic carriers of SNCA duplication demonstrated normal glucose metabolism. Symptomatic patients showed abnormal olfactory function and polysomnography and asymptomatic carriers showed normal results. The clinical features of SNCA duplication include parkinsonism with or without dementia. Asymptomatic carriers displayed normal test results with the eldest individual aged 79 years; thus, even a carrier of SNCA duplication may escape the development of PD. This difference in age-associated penetrance may be due to the genetic background or environmental exposures. Further studies of SNCA duplication carriers will help identify disease-modifiers and may open novel avenues for future treatment.

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Year:  2009        PMID: 19562770     DOI: 10.1002/mds.22682

Source DB:  PubMed          Journal:  Mov Disord        ISSN: 0885-3185            Impact factor:   10.338


  49 in total

1.  Multisystem Lewy body disease and the other parkinsonian disorders.

Authors:  J William Langston; Birgitt Schüle; Linda Rees; R Jeremy Nichols; Carrolee Barlow
Journal:  Nat Genet       Date:  2015-12       Impact factor: 38.330

Review 2.  Autosomal dominant Parkinson's disease caused by SNCA duplications.

Authors:  Takuya Konno; Owen A Ross; Andreas Puschmann; Dennis W Dickson; Zbigniew K Wszolek
Journal:  Parkinsonism Relat Disord       Date:  2015-09-03       Impact factor: 4.891

Review 3.  The heterozygous A53T mutation in the alpha-synuclein gene in a Chinese Han patient with Parkinson disease: case report and literature review.

Authors:  Wei-Xi Xiong; Yi-Min Sun; Rong-Yuan Guan; Su-Shan Luo; Chen Chen; Yu An; Jian Wang; Jian-Jun Wu
Journal:  J Neurol       Date:  2016-07-08       Impact factor: 4.849

Review 4.  Circadian dysfunction may be a key component of the non-motor symptoms of Parkinson's disease: insights from a transgenic mouse model.

Authors:  L David Willison; Takashi Kudo; Dawn H Loh; Dika Kuljis; Christopher S Colwell
Journal:  Exp Neurol       Date:  2013-01-24       Impact factor: 5.330

Review 5.  Genetics of Parkinson disease and essential tremor.

Authors:  Christian Wider; Owen A Ross; Zbigniew K Wszolek
Journal:  Curr Opin Neurol       Date:  2010-08       Impact factor: 5.710

6.  Heritability of Parkinson disease in Swedish twins: a longitudinal study.

Authors:  Karin Wirdefeldt; Margaret Gatz; Chandra A Reynolds; Carol A Prescott; Nancy L Pedersen
Journal:  Neurobiol Aging       Date:  2011-04-11       Impact factor: 4.673

Review 7.  Genetics of synucleins in neurodegenerative diseases.

Authors:  José Brás; Elizabeth Gibbons; Rita Guerreiro
Journal:  Acta Neuropathol       Date:  2020-08-01       Impact factor: 17.088

8.  Increased expression of α-synuclein by SNCA duplication is associated with resistance to toxic stimuli.

Authors:  Han-Joon Kim; Beom S Jeon; Min-Yung Yoon; Sung-Sup Park; Kwang-Woo Lee
Journal:  J Mol Neurosci       Date:  2012-03-06       Impact factor: 3.444

9.  {alpha}-Synuclein gene duplication impairs reward learning.

Authors:  Szabolcs Kéri; Ahmed A Moustafa; Catherine E Myers; György Benedek; Mark A Gluck
Journal:  Proc Natl Acad Sci U S A       Date:  2010-08-23       Impact factor: 11.205

Review 10.  The mitochondrial kinase PINK1, stress response and Parkinson's disease.

Authors:  Marina Jendrach; Suzana Gispert; Filomena Ricciardi; Michael Klinkenberg; Rudolf Schemm; Georg Auburger
Journal:  J Bioenerg Biomembr       Date:  2009-12       Impact factor: 2.945

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