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PMID: 20838799 Published · ppublish English Journal Article Research Support, N.I.H., Extramural Research Support, Non-U.S. Gov't

Glucocerebrosidase is present in α-synuclein inclusions in Lewy body disorders.

Acta neuropathologica ·Vol. 120 ·No. 5 ·2010-11-00 ·Pages 641-9

Goker-Alpan O, Stubblefield BK, Giasson BI, Sidransky E

Abstract

Mutations in the gene encoding the lysosomal enzyme glucocerebrosidase, known to cause Gaucher disease (GD), are a risk factor for the development of Parkinson disease (PD) and related disorders. This association is based on the concurrence of parkinsonism and GD, the identification of glucocerebrosidase mutations in cohorts with PD from centers around the world, and neuropathologic findings. The contribution of glucocerebrosidase to the development of parkinsonian pathology was explored by studying seven brain samples from subjects carrying glucocerebrosidase mutations with pathologic diagnoses of PD and/or Lewy body dementia. Three individuals had GD and four were heterozygous for glucocerebrosidase mutations. All cases had no known family history of PD and the mean age of disease onset was 59 years (range 42-77). Immunofluorescence studies on brain tissue samples from patients with parkinsonism associated with glucocerebrosidase mutations showed that glucocerebrosidase was present in 32-90% of Lewy bodies (mean 75%), some ubiquitinated and others non-ubiquitinated. In samples from seven subjects without mutations, <10% of Lewy bodies were glucocerebrosidase positive (mean 4%). This data demonstrates that glucocerebrosidase can be an important component of α-synuclein-positive pathological inclusions. Unraveling the role of mutant glucocerebrosidase in the development of this pathology will further our understanding of the lysosomal pathways that likely contribute to the formation and/or clearance of these protein aggregates.

MeSH Terms
Adult Aged Aged, 80 and over Blotting, Southern Brain/metabolism,pathology Female Fluorescent Antibody Technique Gaucher Disease/genetics,metabolism,pathology Glucosylceramidase/genetics,metabolism Humans Immunohistochemistry Lewy Bodies/chemistry,enzymology,pathology Lewy Body Disease/genetics,metabolism,pathology Male Microscopy, Confocal Middle Aged Mutation Neurites/chemistry,enzymology,pathology Parkinson Disease/genetics,metabolism,pathology Ubiquitination alpha-Synuclein/metabolism
Chemicals
alpha-Synuclein Glucosylceramidase
Authors & Affiliations
4 authors, click to expand affiliations / ORCID
Goker-Alpan Ozlem
Section on Molecular Neurogenetics, Medical Genetics Branch, NHGRI, National Institutes of Health, Bethesda, MD 20892-3708, USA.
Stubblefield Barbara K
Giasson Benoit I
Sidransky Ellen
References (33)
33 references, click to expand
  1. Genotype-phenotype correlations between GBA mutations and Parkinson disease risk and onset.
    Neurology. 2008 Jun 10;70(24):2277-83 PMID: 18434642
  2. Multicenter analysis of glucocerebrosidase mutations in Parkinson's disease.
    N Engl J Med. 2009 Oct 22;361(17):1651-61 PMID: 19846850
  3. Glucocerebrosidase mutations in subjects with parkinsonism.
    Mol Genet Metab. 2004 Jan;81(1):70-3 PMID: 14728994
  4. Alpha-synuclein: normal function and role in neurodegenerative diseases.
    Curr Top Dev Biol. 2004;60:17-54 PMID: 15094295
  5. Alpha-synuclein in Lewy bodies.
    Nature. 1997 Aug 28;388(6645):839-40 PMID: 9278044
  6. Glucocerebrosidase mutations in clinical and pathologically proven Parkinson's disease.
    Brain. 2009 Jul;132(Pt 7):1783-94 PMID: 19286695
  7. Parkinsonism among Gaucher disease carriers.
    J Med Genet. 2004 Dec;41(12):937-40 PMID: 15591280
  8. Lewy bodies.
    Proc Natl Acad Sci U S A. 2006 Feb 7;103(6):1661-8 PMID: 16449387
  9. Glucosylsphingosine accumulation in tissues from patients with Gaucher disease: correlation with phenotype and genotype.
    Mol Genet Metab. 2002 Aug;76(4):262-70 PMID: 12208131
  10. Diagnosis and management of dementia with Lewy bodies: third report of the DLB Consortium.
    Neurology. 2005 Dec 27;65(12):1863-72 PMID: 16237129
  11. Association of alpha-synuclein and mutants with lipid membranes: spin-label ESR and polarized IR.
    Biochemistry. 2006 Mar 14;45(10):3386-95 PMID: 16519533
  12. Association of glucocerebrosidase mutations with dementia with lewy bodies.
    Arch Neurol. 2009 May;66(5):578-83 PMID: 19433657
  13. Aggregation of alpha-synuclein in Lewy bodies of sporadic Parkinson's disease and dementia with Lewy bodies.
    Am J Pathol. 1998 Apr;152(4):879-84 PMID: 9546347
  14. Impaired degradation of mutant alpha-synuclein by chaperone-mediated autophagy.
    Science. 2004 Aug 27;305(5688):1292-5 PMID: 15333840
  15. Mutation in the alpha-synuclein gene identified in families with Parkinson's disease.
    Science. 1997 Jun 27;276(5321):2045-7 PMID: 9197268
  16. Double immunolabeling of neuropeptides in the human hypothalamus as analyzed by confocal laser scanning fluorescence microscopy.
    J Histochem Cytochem. 1999 Feb;47(2):229-36 PMID: 9889258
  17. Glucocerebrosidase mutations are an important risk factor for Lewy body disorders.
    Neurology. 2006 Sep 12;67(5):908-10 PMID: 16790605
  18. Genotypic heterogeneity and phenotypic variation among patients with type 2 Gaucher's disease.
    Pediatr Res. 1998 May;43(5):571-8 PMID: 9585001
  19. Lipid composition of microdomains is altered in a cell model of Gaucher disease.
    J Lipid Res. 2008 Aug;49(8):1725-34 PMID: 18427156
  20. Gaucher disease with parkinsonian manifestations: does glucocerebrosidase deficiency contribute to a vulnerability to parkinsonism?
    Mol Genet Metab. 2003 Jun;79(2):104-9 PMID: 12809640
  21. Glucocerebrosidase mutations in Chinese subjects from Taiwan with sporadic Parkinson disease.
    Mol Genet Metab. 2007 Jun;91(2):195-200 PMID: 17462935
  22. In situ and in vitro study of colocalization and segregation of alpha-synuclein, ubiquitin, and lipids in Lewy bodies.
    Exp Neurol. 2000 Dec;166(2):324-33 PMID: 11085897
  23. Staging of brain pathology related to sporadic Parkinson's disease.
    Neurobiol Aging. 2003 Mar-Apr;24(2):197-211 PMID: 12498954
  24. Alpha-synuclein and neurodegenerative diseases.
    Nat Rev Neurosci. 2001 Jul;2(7):492-501 PMID: 11433374
  25. Mutations in the glucocerebrosidase gene and Parkinson's disease in Ashkenazi Jews.
    N Engl J Med. 2004 Nov 4;351(19):1972-7 PMID: 15525722
  26. Immunohistochemical and biochemical studies demonstrate a distinct profile of alpha-synuclein permutations in multiple system atrophy.
    J Neuropathol Exp Neurol. 2000 Sep;59(9):830-41 PMID: 11005264
  27. Complete screening for glucocerebrosidase mutations in Parkinson disease patients from Portugal.
    Neurobiol Aging. 2009 Sep;30(9):1515-7 PMID: 18160183
  28. Accuracy of clinical diagnosis of idiopathic Parkinson's disease: a clinico-pathological study of 100 cases.
    J Neurol Neurosurg Psychiatry. 1992 Mar;55(3):181-4 PMID: 1564476
  29. Mutations for Gaucher disease confer high susceptibility to Parkinson disease.
    Arch Neurol. 2009 May;66(5):571-6 PMID: 19433656
  30. Analysis of the glucocerebrosidase gene in Parkinson's disease.
    Mov Disord. 2005 Mar;20(3):367-70 PMID: 15517592
  31. The spectrum of parkinsonian manifestations associated with glucocerebrosidase mutations.
    Arch Neurol. 2008 Oct;65(10):1353-7 PMID: 18852351
  32. Neuropathology provides clues to the pathophysiology of Gaucher disease.
    Mol Genet Metab. 2004 Jul;82(3):192-207 PMID: 15234332
  33. Mutations in GBA are associated with familial Parkinson disease susceptibility and age at onset.
    Neurology. 2009 Jan 27;72(4):310-6 PMID: 18987351
Article Info
Journal
Acta neuropathologica
Abbr.
Acta Neuropathol
ISSN
1432-0533
Published
2010-11-00
Epub
2010-00-14
Pages
641-9
Language
English
Region
Germany
NLM ID
0412041
PMCID
PMC3352317
Subset
IM
Grants
NINDS NIH HHS · P50 NS053488 · United States
Intramural NIH HHS · ZIA HG200336-06 · United States
NINDS NIH HHS · NS053488 · United States
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