Beyond the brain: widespread pathology in Huntington's disease

被引:259
作者
van der Burg, Jorien M. M. [1 ]
Bjorkqvist, Maria [1 ]
Brundin, Patrik [1 ]
机构
[1] Lund Univ, Neuronal Survival Unit, Dept Expt Med Sci, Wallenberg Neurosci Ctr, S-22184 Lund, Sweden
基金
瑞典研究理事会;
关键词
TRANSGENIC MOUSE MODEL; CHOLESTEROL-BIOSYNTHESIS PATHWAY; PITUITARY-ADRENAL AXIS; PERIPHERAL-BLOOD CELLS; EXPANDED CAG REPEAT; R6/2; MOUSE; GENE-EXPRESSION; WEIGHT-LOSS; INCREASED APOPTOSIS; DIABETES-MELLITUS;
D O I
10.1016/S1474-4422(09)70178-4
中图分类号
R74 [神经病学与精神病学];
学科分类号
摘要
Huntington's disease (HD) is an inherited neurodegenerative disorder caused by a polyglutamine stretch in the huntingtin protein. Today, more than 15 years after the genetic defect underlying HD was discovered, the pathogenesis is still not well understood and there is no adequate treatment. Research into this disorder has conventionally focused on neurological symptoms and brain pathology, particularly neurodegeneration in the basal ganglia and cerebral cortex. Mutant huntingtin is, however, ubiquitously expressed throughout the body. Indeed, contrary to earlier thinking, HD is associated with abnormalities in peripheral tissues. These abnormal changes are not all secondary to brain dysfunction, but most seem to be directly caused by expression of mutant huntingtin in peripheral tissues. In this article, we highlight this emerging field of research and how it might affect our understanding of the pathogenesis of this disease, the development of novel biomarkers of disease progression, and the identification of new potential treatments.
引用
收藏
页码:765 / 774
页数:10
相关论文
共 110 条
[51]   Metformin therapy in a transgenic mouse model of Huntington's disease [J].
Ma, Thong C. ;
Buescher, Jessica L. ;
Oatis, Benjamin ;
Funk, Jason A. ;
Nash, Andrew J. ;
Carrier, Raeann L. ;
Hoyt, Kari R. .
NEUROSCIENCE LETTERS, 2007, 411 (02) :98-103
[52]   Huntingtin fragmentation and increased caspase 3, 8 and 9 activities in lymphoblasts with heterozygous and homozygous Huntington's disease mutation [J].
Maglione, V ;
Cannella, M ;
Gradini, R ;
Cislaghi, G ;
Squitieri, F .
MECHANISMS OF AGEING AND DEVELOPMENT, 2006, 127 (02) :213-216
[53]   Exon 1 of the HD gene with an expanded CAG repeat is sufficient to cause a progressive neurological phenotype in transgenic mice [J].
Mangiarini, L ;
Sathasivam, K ;
Seller, M ;
Cozens, B ;
Harper, A ;
Hetherington, C ;
Lawton, M ;
Trottier, Y ;
Lehrach, H ;
Davies, SW ;
Bates, GP .
CELL, 1996, 87 (03) :493-506
[54]   Plasma testosterone in male patients with Huntington's disease: Relations to severity of illness and dementia [J].
Markianos, M ;
Panas, M ;
Kalfakis, N ;
Vassilopoulos, D .
ANNALS OF NEUROLOGY, 2005, 57 (04) :520-525
[55]  
Markianos M, 2007, NEUROENDOCRINOL LETT, V28, P199
[56]   Exendin-4 Improves Glycemic Control, Ameliorates Brain and Pancreatic Pathologies, and Extends Survival in a Mouse Model of Huntington's Disease [J].
Martin, Bronwen ;
Golden, Erin ;
Carlson, Olga D. ;
Pistell, Paul ;
Zhou, Jie ;
Kim, Wook ;
Frank, Brittany P. ;
Thomas, Sam ;
Chadwick, Wayne A. ;
Greig, Nigel H. ;
Bate, Gillian P. ;
Sathasivam, Kirupa ;
Bernier, Michel ;
Maudsley, Stnart ;
Mattson, Mark P. ;
Egan, Josephine M. .
DIABETES, 2009, 58 (02) :318-328
[57]  
MARULLO M, 2008, BRAIN PATHOL, DOI DOI 10.1111/J.1750=3639.2008.00249.X
[58]   Cardiac dysfunction in the R6/2 mouse model of Huntington's disease [J].
Mihm, Michael J. ;
Amann, Deborah M. ;
Schanbacher, Brandon L. ;
Altschuld, Ruth A. ;
Bauer, John Anthony ;
Hoyt, Karl R. .
NEUROBIOLOGY OF DISEASE, 2007, 25 (02) :297-308
[59]   Early Energy Deficit in Huntington Disease: Identification of a Plasma Biomarker Traceable during Disease Progression [J].
Mochel, Fanny ;
Charles, Perrine ;
Seguin, Francois ;
Barritault, Julie ;
Coussieu, Christiane ;
Perin, Laurence ;
Le Bouc, Yves ;
Gervais, Christiane ;
Carcelain, Guislaine ;
Vassault, Anne ;
Feingold, Josue ;
Rabier, Daniel ;
Durr, Alexandra .
PLOS ONE, 2007, 2 (07)
[60]   NUTRITIONAL-EVALUATION OF HUNTINGTON DISEASE PATIENTS [J].
MORALES, LM ;
ESTEVEZ, J ;
SUAREZ, H ;
VILLALOBOS, R ;
DEBONILLA, LC ;
BONILLA, E .
AMERICAN JOURNAL OF CLINICAL NUTRITION, 1989, 50 (01) :145-150