Nuclear inclusions of the androgen receptor protein in spinal and bulbar muscular atrophy
- PMID: 9708548
- DOI: 10.1002/ana.410440216
Nuclear inclusions of the androgen receptor protein in spinal and bulbar muscular atrophy
Abstract
Spinal and bulbar muscular atrophy (SBMA) is an X-linked motor neuronopathy caused by the expansion of an unstable CAG repeat in the coding region of the androgen receptor (AR) gene. To study AR protein expression in normal and SBMA individuals, we used several antibodies that recognize AR protein, and analyzed neural and nonneural tissues by immunohistochemistry and western blotting. Both the normal and the mutant AR proteins were widely distributed, predominantly, but not exclusively, in the cytoplasm of neurons regardless of the pathological involvement, and predominantly in the nuclei of the nonneural tissues in both normal and SBMA individuals, with different expression levels of AR protein among different tissues. In the motor neurons of SBMA patients, there were AR-immunoreactive ubiquitinated nuclear inclusions that were detected by antibodies that recognize a small portion of the N terminus of the AR protein. Absence of other immunoreactive AR epitopes within the inclusion may be due to altered AR configuration, or masking of AR epitopes by other proteins, or proteolytic cleavage of the AR. Our data show that, in addition to the normal cellular distribution of the AR protein, mutant AR-bearing nuclear inclusions are present in SBMA.
Similar articles
-
Nonneural nuclear inclusions of androgen receptor protein in spinal and bulbar muscular atrophy.Am J Pathol. 1998 Sep;153(3):695-701. doi: 10.1016/S0002-9440(10)65612-X. Am J Pathol. 1998. PMID: 9736019 Free PMC article.
-
Mutant androgen receptor accumulation in spinal and bulbar muscular atrophy scrotal skin: a pathogenic marker.Ann Neurol. 2006 Mar;59(3):520-6. doi: 10.1002/ana.20735. Ann Neurol. 2006. PMID: 16358333 Clinical Trial.
-
Loss of endogenous androgen receptor protein accelerates motor neuron degeneration and accentuates androgen insensitivity in a mouse model of X-linked spinal and bulbar muscular atrophy.Hum Mol Genet. 2006 Jul 15;15(14):2225-38. doi: 10.1093/hmg/ddl148. Epub 2006 Jun 13. Hum Mol Genet. 2006. PMID: 16772330
-
Spinal and bulbar muscular atrophy: ligand-dependent pathogenesis and therapeutic perspectives.J Mol Med (Berl). 2004 May;82(5):298-307. doi: 10.1007/s00109-004-0530-7. Epub 2004 Feb 27. J Mol Med (Berl). 2004. PMID: 15133611 Review.
-
Spinal and bulbar muscular atrophy.Handb Clin Neurol. 2018;148:625-632. doi: 10.1016/B978-0-444-64076-5.00040-5. Handb Clin Neurol. 2018. PMID: 29478604 Review.
Cited by
-
Mutant androgen receptor induces neurite loss and senescence independently of ARE binding in a neuronal model of SBMA.Proc Natl Acad Sci U S A. 2024 Jul 16;121(29):e2321408121. doi: 10.1073/pnas.2321408121. Epub 2024 Jul 8. Proc Natl Acad Sci U S A. 2024. PMID: 38976730 Free PMC article.
-
The localization and interactions of huntingtin.Philos Trans R Soc Lond B Biol Sci. 1999 Jun 29;354(1386):1021-7. doi: 10.1098/rstb.1999.0454. Philos Trans R Soc Lond B Biol Sci. 1999. PMID: 10434301 Free PMC article. Review.
-
Heat shock protein 70 chaperone overexpression ameliorates phenotypes of the spinal and bulbar muscular atrophy transgenic mouse model by reducing nuclear-localized mutant androgen receptor protein.J Neurosci. 2003 Mar 15;23(6):2203-11. doi: 10.1523/JNEUROSCI.23-06-02203.2003. J Neurosci. 2003. PMID: 12657679 Free PMC article.
-
CHIP overexpression reduces mutant androgen receptor protein and ameliorates phenotypes of the spinal and bulbar muscular atrophy transgenic mouse model.J Neurosci. 2007 May 9;27(19):5115-26. doi: 10.1523/JNEUROSCI.1242-07.2007. J Neurosci. 2007. PMID: 17494697 Free PMC article.
-
B2 attenuates polyglutamine-expanded androgen receptor toxicity in cell and fly models of spinal and bulbar muscular atrophy.J Neurosci Res. 2010 Aug 1;88(10):2207-16. doi: 10.1002/jnr.22389. J Neurosci Res. 2010. PMID: 20336775 Free PMC article.
Publication types
MeSH terms
Substances
LinkOut - more resources
Full Text Sources
Other Literature Sources
Medical
Research Materials