Hypocretin and melanin-concentrating hormone in patients with Huntington disease

被引:86
作者
Aziz, Ahmad [1 ,4 ]
Fronczek, Rolf [1 ,4 ]
Maat-Schieman, Marion [1 ]
Unmehopa, Unga [4 ]
Roelandse, Freek [2 ]
Overeem, Sebastiaan [1 ,5 ]
van Duinen, Sjoerd [3 ]
Lammers, Gert-Jan [1 ]
Swaab, Dick [4 ]
Roos, Raymund [1 ]
机构
[1] Leiden Univ, Med Ctr, Dept Neurol, NL-2300 RC Leiden, Netherlands
[2] Leiden Univ, Med Ctr, Dept Clin Chem, NL-2300 RC Leiden, Netherlands
[3] Leiden Univ, Med Ctr, Dept Pathol, NL-2300 RC Leiden, Netherlands
[4] Netherlands Inst Neurosci, Amsterdam, ZO, Netherlands
[5] Radboud Univ Nijmegen, Med Ctr, Dept Neurol, NL-6525 ED Nijmegen, Netherlands
关键词
Huntington; hypocretin; hypothalamus; melanin-concentrating hormone; orexin;
D O I
10.1111/j.1750-3639.2008.00135.x
中图分类号
R74 [神经病学与精神病学];
学科分类号
摘要
To evaluate whether hypocretin-1 (orexin-A) and melanin-concentrating hormone (MCH) neurotransmission are affected in patients with Huntington disease (HD), we immunohistochemically stained hypocretin and MCH neurons and estimated their total numbers in the lateral hypothalamus of both HD patients and matched controls. In addition, hypocretin-1 levels were determined in prefrontal cortical tissue and post-mortem ventricular cerebrospinal fluid (CSF) using a radioimmunoassay. The total number of hypocretin-1 neurons was significantly reduced by 30% in HD brains (P = 0.015), while the total number of MCH neurons was not significantly altered (P = 0.100). Levels of hypocretin-1 were 33% lower in the prefrontal cortex of the HD patients (P = 0.025), but ventricular CSF levels were similar to the control values (P = 0.306). Neuronal intranuclear and cytoplasmic inclusions of mutant huntingtin were present in all HD hypothalami, although with a variable distribution across different hypothalamic structures. We found a specific reduction in hypocretin signaling in patients with HD as MCH cell number was not significantly affected. It remains to be shown whether the moderate decrease in hypocretin neurotransmission could contribute to clinical symptoms. As the number of MCH-expressing neurons was not affected, alterations in MCH signaling are unlikely to have clinical effects in HD patients.
引用
收藏
页码:474 / 483
页数:10
相关论文
共 43 条
[1]   Hypocretin/orexin-containing neurons are produced in one sharp peak in the developing ventral diencephalon [J].
Amiot, C ;
Brischoux, F ;
Colard, C ;
La Roche, A ;
Fellmann, D ;
Risold, PY .
EUROPEAN JOURNAL OF NEUROSCIENCE, 2005, 22 (02) :531-534
[2]  
Aziz NA, 2007, REV NEUROSCIENCE, V18, P223
[3]   Hypocretin-1 (orexin A) levels are normal in Huntington's disease [J].
Baumann, Christian R. ;
Hersberger, Martin ;
Bassetti, Claudio L. .
JOURNAL OF NEUROLOGY, 2006, 253 (09) :1232-1233
[4]   Cerebrospinal fluid levels of orexin-A are not a clinically useful biomarker for Huntington disease [J].
Bjorkqvist, M. ;
Petersen, A. ;
Carroll, J. ;
Nielsen, J. ;
Ecker, D. ;
Mulder, H. ;
Hayden, M. R. ;
Landwehrmeyer, B. ;
Brundin, P. ;
Leavitt, B. R. .
CLINICAL GENETICS, 2006, 70 (01) :78-79
[5]   Orexin and psychiatric symptoms in suicide attempters [J].
Brundin, Lena ;
Petersen, Asa ;
Bjorkqvist, Maria ;
Traskman-Bendz, Lil .
JOURNAL OF AFFECTIVE DISORDERS, 2007, 100 (1-3) :259-263
[6]   Narcolepsy in orexin knockout mice:: Molecular genetics of sleep regulation [J].
Chemelli, RM ;
Willie, JT ;
Sinton, CM ;
Elmquist, JK ;
Scammell, T ;
Lee, C ;
Richardson, JA ;
Williams, SC ;
Xiong, YM ;
Kisanuki, Y ;
Fitch, TE ;
Nakazato, M ;
Hammer, RE ;
Saper, CB ;
Yanagisawa, M .
CELL, 1999, 98 (04) :437-451
[7]   Orexin A-like immunoreactivity in the rat brain [J].
Chen, CT ;
Dun, SL ;
Kwok, EH ;
Dun, NJ ;
Chang, JK .
NEUROSCIENCE LETTERS, 1999, 260 (03) :161-164
[8]   Aggregation of huntingtin in neuronal intranuclear inclusions and dystrophic neurites in brain [J].
DiFiglia, M ;
Sapp, E ;
Chase, KO ;
Davies, SW ;
Bates, GP ;
Vonsattel, JP ;
Aronin, N .
SCIENCE, 1997, 277 (5334) :1990-1993
[9]   The number of hypothalamic hypocretin (orexin) neurons is not affected in Prader-Willi syndrome [J].
Fronczek, R ;
Lammers, GJ ;
Balesar, R ;
Unmehopa, UA ;
Swaab, DF .
JOURNAL OF CLINICAL ENDOCRINOLOGY & METABOLISM, 2005, 90 (09) :5466-5470
[10]   Hypocretin (orexin) loss in Parkinson's disease [J].
Fronczek, Rolf ;
Overeem, Sebastiaan ;
Lee, Sandy Y. Y. ;
Hegeman, Ingrid. M. ;
van Pelt, Johannes ;
van Duinen, Sjoerd. G. ;
Lammers, Gert Jan ;
Swaab, Dick F. .
BRAIN, 2007, 130 :1577-1585