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
  • [1] Comparison of Parkinson Risk in Ashkenazi Jewish Patients With Gaucher Disease and GBA Heterozygotes
    Alcalay, Roy N.
    Dinur, Tama
    Quinn, Timothy
    Sakanaka, Karina
    Levy, Oren
    Waters, Cheryl
    Fahn, Stanley
    Dorovski, Tsvyatko
    Chung, Wendy K.
    Pauciulo, Michael
    Nichols, William
    Rana, Huma Q.
    Balwani, Manisha
    Bier, Louise
    Elstein, Deborah
    Zimran, Ari
    [J]. JAMA NEUROLOGY, 2014, 71 (06) : 752 - 757
  • [2] Penetrance of Parkinson disease in glucocerebrosidase gene mutation carriers
    Anheim, M.
    Elbaz, A.
    Lesage, S.
    Durr, A.
    Condroyer, C.
    Viallet, F.
    Pollak, P.
    Bonaiti, B.
    Bonaiti-Pellie, C.
    Brice, A.
    [J]. NEUROLOGY, 2012, 78 (06) : 417 - 420
  • [3] Evaluation of α-synuclein immunohistochemical methods used by invited experts
    Beach, Thomas G.
    White, Charles L.
    Hamilton, Ronald L.
    Duda, John E.
    Iwatsubo, Takeshi
    Dickson, Dennis W.
    Leverenz, James B.
    Roncaroli, Federico
    Buttini, Manuel
    Hladik, Christa L.
    Sue, Lucia I.
    Noorigian, Joseph V.
    Adler, Charles H.
    [J]. ACTA NEUROPATHOLOGICA, 2008, 116 (03) : 277 - 288
  • [4] GAUCHER DISEASE - NEW MOLECULAR APPROACHES TO DIAGNOSIS AND TREATMENT
    BEUTLER, E
    [J]. SCIENCE, 1992, 256 (5058) : 794 - 799
  • [5] GBA-associated PD presents with nonmotor characteristics
    Brockmann, K.
    Srulijes, K.
    Hauser, A. -K.
    Schulte, C.
    Csoti, I.
    Gasser, T.
    Berg, D.
    [J]. NEUROLOGY, 2011, 77 (03) : 276 - 280
  • [6] The risk of Parkinson's disease in type 1 Gaucher disease
    Bultron, Gilberto
    Kacena, Katherine
    Pearson, Daniel
    Boxer, Michael
    Yang, Ruhua
    Sathe, Swati
    Pastores, Gregory
    Mistry, Pramod K.
    [J]. JOURNAL OF INHERITED METABOLIC DISEASE, 2010, 33 (02) : 167 - 173
  • [7] Synaptic failure and α-synuclein
    Calo, Laura
    Wegrzynowicz, Michal
    Santivanez-Perez, Jessica
    Spillantini, Maria Grazia
    [J]. MOVEMENT DISORDERS, 2016, 31 (02) : 169 - 177
  • [8] Glucocerebrosidase inhibition causes mitochondrial dysfunction and free radical damage
    Cleeter, Michael W. J.
    Chau, Kai-Yin
    Gluck, Caroline
    Mehta, Atul
    Hughes, Derralynn A.
    Duchen, Michael
    Wood, Nicholas William
    Hardy, John
    Cooper, J. Mark
    Schapira, Anthony Henry
    [J]. NEUROCHEMISTRY INTERNATIONAL, 2013, 62 (01) : 1 - 7
  • [9] Acid β-Glucosidase Mutants Linked to Gaucher Disease, Parkinson Disease, and Lewy Body Dementia Alter α-Synuclein Processing
    Cullen, Valerie
    Sardi, Pablo
    Ng, Juliana
    Xu, You-Hai
    Sun, Ying
    Tomlinson, Julianna J.
    Kolodziej, Piotr
    Kahn, Ilana
    Saftig, Paul
    Woulfe, John
    Rochet, Jean-Christophe
    Glicksman, Marcie A.
    Cheng, Seng H.
    Grabowski, Gregory A.
    Shihabuddin, Lamya S.
    Schlossmacher, Michael G.
    [J]. ANNALS OF NEUROLOGY, 2011, 69 (06) : 940 - 953
  • [10] Glucocerebrosidase Deficiency in Drosophila Results in α-Synuclein-Independent Protein Aggregation and Neurodegeneration
    Davis, Marie Y.
    Trinh, Kien
    Thomas, Ruth E.
    Yu, Selina
    Germanos, Alexandre A.
    Whitley, Brittany N.
    Sardi, Sergio Pablo
    Montine, Thomas J.
    Pallanck, Leo J.
    [J]. PLOS GENETICS, 2016, 12 (03):