Home LiteratureArticle Details
PMID: 19632874 Published · ppublish English Journal Article Research Support, N.I.H., Extramural Research Support, Non-U.S. Gov't

A Swedish family with de novo alpha-synuclein A53T mutation: evidence for early cortical dysfunction.

Parkinsonism & related disorders ·Vol. 15 ·No. 9 ·2009-11-00 ·Pages 627-32

Puschmann A, Ross OA, Vilariño-Güell C, Lincoln SJ, Kachergus JM, Cobb SA, Lindquist SG, Nielsen JE, Wszolek ZK, Farrer M, Widner H, van Westen D, Hägerström D, Markopoulou K, Chase BA, Nilsson K, Reimer J, Nilsson C

Abstract

A de novo alpha-synuclein A53T (p.Ala53 Th; c.209G > A) mutation has been identified in a Swedish family with autosomal dominant Parkinson's disease (PD). Two affected individuals had early-onset (before 31 and 40 years), severe levodopa-responsive PD with prominent dysphasia, dysarthria, and cognitive decline. Longitudinal clinical follow-up, EEG, SPECT and CSF biomarker examinations suggested an underlying encephalopathy with cortical involvement. The mutated allele (c.209A) was present within a haplotype different from that shared among mutation carriers in the Italian (Contursi) and the Greek-American Family H kindreds. One unaffected family member carried the mutation haplotype without the c.209A mutation, strongly suggesting its de novo occurrence within this family. Furthermore, a novel mutation c.488G > A (p.Arg163His; R163H) in the presenilin-2 (PSEN2) gene was detected, but was not associated with disease state.

MeSH Terms
Adult Cerebral Cortex/pathology,physiopathology Female Humans Intermediate Filament Proteins/genetics Male Mutation Parkinson Disease/genetics,pathology,physiopathology Pedigree Polymerase Chain Reaction Sweden
Chemicals
Intermediate Filament Proteins desmuslin
Authors & Affiliations
18 authors, click to expand affiliations / ORCID
Puschmann Andreas
Department of Neurology, Lund University Hospital, Sweden; Department of Clinical Science, Section of Geriatric Psychiatry, Lund University, Sweden. andreas.puschmann@med.lu.se
Ross Owen A
Vilariño-Güell Carles
Lincoln Sarah J
Kachergus Jennifer M
Cobb Stephanie A
Lindquist Suzanne G
Nielsen Jørgen E
Wszolek Zbigniew K
Farrer Matthew
Widner Håkan
van Westen Danielle
Hägerström Douglas
Markopoulou Katerina
Chase Bruce A
Nilsson Karin
Reimer Jan
Nilsson Christer
References (26)
26 references, click to expand
  1. Mutation in the alpha-synuclein gene identified in families with Parkinson's disease.
    Science. 1997 Jun 27;276(5321):2045-7 PMID: 9197268
  2. Alpha synuclein is present in Lewy bodies in sporadic Parkinson's disease.
    Mol Psychiatry. 1998 Nov;3(6):493-9 PMID: 9857974
  3. Unverricht-Lundborg disease-a misnomer?
    Mov Disord. 2009 Mar 15;24(4):629-30; author reply 630 PMID: 18512745
  4. Mutated alpha-synuclein gene in two Greek kindreds with familial PD: incomplete penetrance?
    Neurology. 1999 Feb;52(3):651-4 PMID: 10025809
  5. Concurrence of alpha-synuclein and tau brain pathology in the Contursi kindred.
    Acta Neuropathol. 2002 Jul;104(1):7-11 PMID: 12070658
  6. The Ala53Thr mutation in the alpha-synuclein gene in a Korean family with Parkinson disease.
    Clin Genet. 2007 May;71(5):471-3 PMID: 17489854
  7. Novel alpha-synuclein-immunoreactive proteins in brain samples from the Contursi kindred, Parkinson's, and Alzheimer's disease.
    Exp Neurol. 1998 Dec;154(2):684-90 PMID: 9878203
  8. A case of late onset sporadic Parkinson's disease with an A53T mutation in alpha-synuclein.
    J Neurol Neurosurg Psychiatry. 2005 Apr;76(4):596-7 PMID: 15774457
  9. Clinical genetic analysis of Parkinson's disease in the Contursi kindred.
    Ann Neurol. 1996 Nov;40(5):767-75 PMID: 8957018
  10. Clinical features of parkinsonian patients with the alpha-synuclein (G209A) mutation.
    Mov Disord. 2001 Nov;16(6):1007-13 PMID: 11748731
  11. Diagnostic criteria for Parkinson disease.
    Arch Neurol. 1999 Jan;56(1):33-9 PMID: 9923759
  12. Validation of a telephone questionnaire for Parkinson's disease.
    J Clin Epidemiol. 1998 Jun;51(6):517-23 PMID: 9636001
  13. Clinical and pathological features of a Parkinsonian syndrome in a family with an Ala53Thr alpha-synuclein mutation.
    Ann Neurol. 2001 Mar;49(3):313-9 PMID: 11261505
  14. LRRK2 mutations are a common cause of Parkinson's disease in Spain.
    Eur J Neurol. 2006 Apr;13(4):391-4 PMID: 16643318
  15. A Greek-American kindred with autosomal dominant, levodopa-responsive parkinsonism and anticipation.
    Ann Neurol. 1995 Sep;38(3):373-8 PMID: 7668822
  16. A large kindred with autosomal dominant Parkinson's disease.
    Ann Neurol. 1990 Mar;27(3):276-82 PMID: 2158268
  17. Alpha-synuclein and Parkinson's disease: implications from the screening of more than 1,900 patients.
    Mov Disord. 2005 Sep;20(9):1191-4 PMID: 15895422
  18. Accelerated in vitro fibril formation by a mutant alpha-synuclein linked to early-onset Parkinson disease.
    Nat Med. 1998 Nov;4(11):1318-20 PMID: 9809558
  19. Clinical, neuropathological and genotypic variability in SNCA A53T familial Parkinson's disease. Variability in familial Parkinson's disease.
    Acta Neuropathol. 2008 Jul;116(1):25-35 PMID: 18389263
  20. Genetic analysis of families with Parkinson disease that carry the Ala53Thr mutation in the gene encoding alpha-synuclein.
    Am J Hum Genet. 1999 Aug;65(2):555-8 PMID: 10417297
  21. Clinical characteristics of the alpha-synuclein mutation (G209A)-associated Parkinson's disease in comparison with other forms of familial Parkinson's disease in Greece.
    Eur J Neurol. 2003 May;10(3):281-6 PMID: 12752402
  22. Fibrillization of alpha-synuclein and tau in familial Parkinson's disease caused by the A53T alpha-synuclein mutation.
    Exp Neurol. 2004 Jun;187(2):279-88 PMID: 15144854
  23. Alpha-synuclein multiplications with parkinsonism, dementia or progressive myoclonus?
    Parkinsonism Relat Disord. 2009 Jun;15(5):390-2 PMID: 18824390
  24. Criteria for diagnosing Parkinson's disease.
    Ann Neurol. 1992;32 Suppl:S125-7 PMID: 1510370
  25. Reduced expression of the G209A alpha-synuclein allele in familial Parkinsonism.
    Ann Neurol. 1999 Sep;46(3):374-81 PMID: 10482268
  26. Phenotypic variation in a large Swedish pedigree due to SNCA duplication and triplication.
    Neurology. 2007 Mar 20;68(12):916-22 PMID: 17251522
Article Info
Journal
Parkinsonism & related disorders
Abbr.
Parkinsonism Relat Disord
ISSN
1873-5126
Published
2009-11-00
Epub
2009-00-25
Pages
627-32
Language
English
Region
England
NLM ID
9513583
PMCID
PMC2783246
Subset
IM
Grants
NIA NIH HHS · P01 AG017216-100005 · United States
NINDS NIH HHS · R15 NS043162-01A2 · United States
NIA NIH HHS · R01 AG015866-09 · United States
NINDS NIH HHS · R15 NS043162 · United States
NIA NIH HHS · R01AG015866 · United States
NIA NIH HHS · P01 AG017216 · United States
NINDS NIH HHS · P50 NS040256 · United States
NINDS NIH HHS · P50 NS40256 · United States
NIA NIH HHS · P01AG017216 · United States
NIA NIH HHS · R01 AG015866 · United States
Analysis Services
Analysis Services

Contact

No. 2 Wenbo Road, Zhangqiu District, Jinan, Shandong

Qilu Normal University · Genelibs Bioinformatics Lab

750 Shunhua Rd, Jinan

2F, Bldg F, University Science Park

Tel: 0531-88819269

WeChat Official Account

Follow our WeChat subscription account for real-time updates and the latest in medical and biological research.


Business Email

E-mail: product@genelibs.com