The L444P Gba1 mutation enhances alpha-synuclein induced loss of nigral dopaminergic neurons in mice

被引:50
作者
Migdalska-Richards, Anna [1 ]
Wegrzynowicz, Michal [2 ]
Rusconi, Raffaella [3 ]
Deangeli, Giulio [2 ,4 ,5 ]
Di Monte, Donato A. [3 ]
Spillantini, Maria G. [2 ]
Schapira, Anthony H. V. [1 ]
机构
[1] UCL, Inst Neurol, Dept Clin Neurosci, London NW3 2PF, England
[2] Univ Cambridge, Dept Clin Neurosci, Clifford Allhutt Bldg, Cambridge CB2 0AH, England
[3] German Ctr Neurodegenerat Dis DZNE, Sigmund Freud Str 27, D-53127 Bonn, Germany
[4] Scuola Super Sant Anna, Piazza Martini Liberty 33, I-56127 Pisa, Italy
[5] Univ Pisa, Lungarno Antonio Pacinotti 43, I-56126 Pisa, Italy
基金
英国医学研究理事会;
关键词
glucocerebrosidase; GBA1; alpha-synuclein; Parkinson's disease; neurodegeneration; PARKINSON-LIKE NEURODEGENERATION; NEURONOPATHIC GAUCHER-DISEASE; GLUCOCEREBROSIDASE MUTATIONS; MOUSE MODEL; CARRIERS; RISK; EXPRESSION; GENE; PHOSPHORYLATION; OVEREXPRESSION;
D O I
10.1093/brain/awx221
中图分类号
R74 [神经病学与精神病学];
学科分类号
摘要
Mutations in glucocerebrosidase 1 (GBA1) represent the most prevalent risk factor for Parkinson's disease. The molecular mechanisms underlying the link between GBA1 mutations and Parkinson's disease are incompletely understood. We analysed two aged (24-month-old) Gba1 mouse models, one carrying a knock-out mutation and the other a L444P knock-in mutation. A significant reduction of glucocerebrosidase activity was associated with increased total alpha-synuclein accumulation in both these models. Gba1 mutations alone did not alter the number of nigral dopaminergic neurons nor striatal dopamine levels. We then investigated the effect of overexpression of human alpha-synuclein in the substantia nigra of aged (18 to 21-month-old) L444P Gba1 mice. Following intraparenchymal injections of human alpha-synuclein carrying viral vectors, pathological accumulation of phosphorylated alpha-synuclein occurred within the transduced neurons. Stereological counts of nigral dopaminergic neurons revealed a significantly greater cell loss in Gba1-mutant than wild-type mice. These results indicate that Gba1 deficiency enhances neuronal vulnerability to neurodegenerative processes triggered by increased alpha-synuclein expression.
引用
收藏
页码:2706 / 2721
页数:16
相关论文
共 57 条
  • [11] Progressive neurodegenerative and behavioural changes induced by AAV-mediated overexpression of α-synuclein in midbrain dopamine neurons
    Decressac, M.
    Mattsson, B.
    Lundblad, M.
    Weikop, P.
    Bjorklund, A.
    [J]. NEUROBIOLOGY OF DISEASE, 2012, 45 (03) : 939 - 953
  • [12] Murine models of acute neuronopathic Gaucher disease
    Enquist, Ida Berglin
    Lo Bianco, Christophe
    Ooka, Andreas
    Nilsson, Eva
    Mansson, Jan-Eric
    Ehinger, Mats
    Richter, Johan
    Brady, Roscoe O.
    Kirik, Deniz
    Karlsson, Stefan
    [J]. PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA, 2007, 104 (44) : 17483 - 17488
  • [13] No evidence for activation of the unfolded protein response in neuronopathic models of Gaucher disease
    Farfel-Becker, Tamar
    Vitner, Einat
    Dekel, Hani
    Leshem, Noa
    Enquist, Ida Berglin
    Karlsson, Stefan
    Futerman, Anthony H.
    [J]. HUMAN MOLECULAR GENETICS, 2009, 18 (08) : 1482 - 1488
  • [14] Augmentation of phenotype in a transgenic Parkinson mouse heterozygous for a Gaucher mutation
    Fishbein, Ianai
    Kuo, Yien-Ming
    Giasson, Benoit I.
    Nussbaum, Robert L.
    [J]. BRAIN, 2014, 137 : 3235 - 3247
  • [15] α-Synuclein is phosphorylated in synucleinopathy lesions
    Fujiwara, H
    Hasegawa, M
    Dohmae, N
    Kawashima, A
    Masliah, E
    Goldberg, MS
    Shen, J
    Takio, K
    Iwatsubo, T
    [J]. NATURE CELL BIOLOGY, 2002, 4 (02) : 160 - 164
  • [16] Glucocerebrosidase deficiency in substantia nigra of parkinson disease brains
    Gegg, Matthew E.
    Burke, Derek
    Heales, Simon J. R.
    Cooper, J. Mark
    Hardy, John
    Wood, Nicholas W.
    Schapira, Anthony H. V.
    [J]. ANNALS OF NEUROLOGY, 2012, 72 (03) : 455 - 463
  • [17] THE EFFICIENCY OF SYSTEMATIC-SAMPLING IN STEREOLOGY AND ITS PREDICTION
    GUNDERSEN, HJG
    JENSEN, EB
    [J]. JOURNAL OF MICROSCOPY-OXFORD, 1987, 147 : 229 - 263
  • [18] Brain propagation of transduced α-synuclein involves non-fibrillar protein species and is enhanced in α-synuclein null mice
    Helwig, Michael
    Klinkenberg, Michael
    Rusconi, Raffaella
    Musgrove, Ruth E.
    Majbour, Nour K.
    El-Agnaf, Omar M. A.
    Ulusoy, Ayse
    Di Monte, Donato A.
    [J]. BRAIN, 2016, 139 : 856 - 870
  • [19] Houck SA, 2012, METHODS MOL BIOL, V832, P455, DOI 10.1007/978-1-61779-474-2_32
  • [20] KILPATRICK IC, 1986, J NEUROCHEM, V46, P1865