Mutant Huntingtin Does Not Affect the Intrinsic Phenotype of Human Huntington's Disease T Lymphocytes

被引:11
|
作者
Miller, James R. C. [1 ]
Traeger, Ulrike [1 ]
Andre, Ralph [1 ]
Tabrizi, Sarah J. [1 ]
机构
[1] UCL Inst Neurol, Dept Neurodegenerat Dis, London, England
来源
PLOS ONE | 2015年 / 10卷 / 11期
基金
英国医学研究理事会;
关键词
NF-KAPPA-B; IN-VIVO; PROLIFERATION; ACTIVATION; DIFFERENTIATION; TRANSCRIPTION; PATHWAY; ONSET;
D O I
10.1371/journal.pone.0141793
中图分类号
O [数理科学和化学]; P [天文学、地球科学]; Q [生物科学]; N [自然科学总论];
学科分类号
07 ; 0710 ; 09 ;
摘要
Huntington's disease is a fatal neurodegenerative condition caused by a CAG repeat expansion in the huntingtin gene. The peripheral innate immune system is dysregulated in Huntington's disease and may contribute to its pathogenesis. However, it is not clear whether or to what extent the adaptive immune system is also involved. Here, we carry out the first comprehensive investigation of human ex vivo T lymphocytes in Huntington's disease, focusing on the frequency of a range of T lymphocyte subsets, as well as analysis of proliferation, cytokine production and gene transcription. In contrast to the innate immune system, the intrinsic phenotype of T lymphocytes does not appear to be affected by the presence of mutant huntingtin, with Huntington's disease T lymphocytes exhibiting no significant functional differences compared to control cells. The transcriptional profile of T lymphocytes also does not appear to be significantly affected, suggesting that peripheral immune dysfunction in Huntington's disease is likely to be mediated primarily by the innate rather than the adaptive immune system. This study increases our understanding of the effects of Huntington's disease on peripheral tissues, while further demonstrating the differential effects of the mutant protein on different but related cell types. Finally, this study suggests that the potential use of novel therapeutics aimed at modulating the Huntington's disease innate immune system should not be extended to include the adaptive immune system.
引用
收藏
页数:16
相关论文
共 50 条
  • [31] Artificial miRNAs Reduce Human Mutant Huntingtin Throughout the Striatum in a Transgenic Sheep Model of Huntington's Disease
    Pfister, Edith L.
    DiNardo, Natalie
    Mondo, Erica
    Borel, Florie
    Conroy, Faith
    Fraser, Cara
    Gernoux, Gwladys
    Han, Xin
    Hu, Danjing
    Johnson, Emily
    Kennington, Lori
    Liu, PengPeng
    Reid, Suzanne J.
    Sapp, Ellen
    Vodicka, Petr
    Kuchel, Tim
    Morton, A. Jennifer
    Howland, David
    Moser, Richard
    Sena-Esteves, Miguel
    Gao, Guangping
    Mueller, Christian
    DiFiglia, Marian
    Aronin, Neil
    HUMAN GENE THERAPY, 2018, 29 (06) : 663 - 673
  • [32] Mutant huntingtin's effects on striatal gene expression in mice recapitulate changes observed in human Huntington's disease brain and do not differ with mutant huntingtin length or wild-type huntingtin dosage
    Kuhn, Alexandre
    Goldstein, Darlene R.
    Hodges, Angela
    Strand, Andrew D.
    Sengstag, Thierry
    Kooperberg, Charles
    Becanovic, Kristina
    Pouladi, Mahmoud A.
    Sathasivam, Kirupa
    Cha, Jang-Ho J.
    Hannan, Anthony J.
    Hayden, Michael R.
    Leavitt, Blair R.
    Dunnett, Stephen B.
    Ferrante, Robert J.
    Albin, Roger
    Shelbourne, Peggy
    Delorenzi, Mauro
    Augood, Sarah J.
    Faull, Richard L. M.
    Olson, James M.
    Bates, Gillian P.
    Jones, Lesley
    Luthi-Carter, Ruth
    HUMAN MOLECULAR GENETICS, 2007, 16 (15) : 1845 - 1861
  • [33] Mutant huntingtin and neurofilament light have distinct longitudinal dynamics in Huntington's disease
    Rodrigues, Filipe B.
    Byrne, Lauren M.
    Tortelli, Rosanna
    Johnson, Eileanoir B.
    Wijeratne, Peter A.
    Arridge, Marzena
    De Vita, Enrico
    Ghazaleh, Naghmeh
    Houghton, Richard
    Furby, Hannah
    Alexander, Daniel C.
    Tabrizi, Sarah J.
    Schobel, Scott
    Scahill, Rachael, I
    Heslegrave, Amanda
    Zetterberg, Henrik
    Wild, Edward J.
    SCIENCE TRANSLATIONAL MEDICINE, 2020, 12 (574)
  • [34] Oxidative stress in striatal cells expressing mutant huntingtin and in Huntington's disease cybrids
    Ribeiro, Marcio
    Nascimento, Maria V.
    Louros, Susana
    Ferreira, I. Luisa
    Almeida, Sandra
    Cardoso, Sandra M.
    Oliveira, Catarina R.
    Rego, A. Cristina
    FREE RADICAL RESEARCH, 2007, 41 : S50 - S50
  • [35] Mutant huntingtin-impaired degradation of β-catenin causes neurotoxicity in Huntington's disease
    Godin, Juliette D.
    Poizat, Ghislaine
    Hickey, Miriam A.
    Maschat, Florence
    Humbert, Sandrine
    EMBO JOURNAL, 2010, 29 (14): : 2433 - 2445
  • [36] Increased Steady-State Mutant Huntingtin mRNA in Huntington's Disease Brain
    Liu, Wanzhao
    Chaurette, Joanna
    Pfister, Edith L.
    Kennington, Lori A.
    Chase, Kathryn O.
    Bullock, Jocelyn
    Vonsattel, Jean Paul G.
    Faull, Richard L. M.
    Macdonald, Douglas
    DiFiglia, Marian
    Zamore, Phillip D.
    Aronin, Neil
    JOURNAL OF HUNTINGTONS DISEASE, 2013, 2 (04) : 491 - 500
  • [37] Cerebrospinal fluid mutant huntingtin is a biomarker for huntingtin lowering in the striatum of Huntington disease mice
    Caron, Nicholas S.
    Banos, Raul
    Aly, Amirah E.
    Xie, Yuanyun
    Ko, Seunghyun
    Potluri, Nalini
    Anderson, Christine
    Black, Hailey Findlay
    Anderson, Lisa M.
    Gordon, Benjamin
    Southwell, Amber L.
    Hayden, Michael R.
    NEUROBIOLOGY OF DISEASE, 2022, 166
  • [38] Genistein induces degradation of mutant huntingtin in fibroblasts from Huntington’s disease patients
    Karolina Pierzynowska
    Lidia Gaffke
    Zuzanna Cyske
    Grzegorz Węgrzyn
    Metabolic Brain Disease, 2019, 34 : 715 - 720
  • [39] Mutant huntingtin causes context-dependent neurodegeneration in mice with Huntington's disease
    Yu, ZX
    Li, SH
    Evans, J
    Pillarisetti, A
    Li, H
    Li, XJ
    JOURNAL OF NEUROSCIENCE, 2003, 23 (06): : 2193 - 2202
  • [40] Mutant huntingtin impairs immune cell migration in Huntington disease
    Kwan, Wanda
    Traeger, Ulrike
    Davalos, Dimitrios
    Chou, Austin
    Bouchard, Jill
    Andre, Ralph
    Miller, Aaron
    Weiss, Andreas
    Giorgini, Flaviano
    Cheah, Christine
    Moeller, Thomas
    Stella, Nephi
    Akassoglou, Katerina
    Tabrizi, Sarah J.
    Muchowski, Paul J.
    JOURNAL OF CLINICAL INVESTIGATION, 2012, 122 (12): : 4737 - 4747