Department of Biochemistry and Molecular Biology, Thomas Jefferson University, Philadelphia, PA 19107, USA.
Dis Model Mech. 2012 Jan;5(1):141-5. doi: 10.1242/dmm.007849. Epub 2011 Oct 4.
Evidence from multiple animal models demonstrates that testosterone plays a crucial role in the progression of symptoms in spinal and bulbar muscular atrophy (SBMA), a condition that results in neurodegeneration and muscle atrophy in affected men. Mice bearing a transgene encoding a human androgen receptor (AR) that contains a stretch of 112 glutamines (expanded polyglutamine tract; AR112Q mice) reproduce several aspects of the human disease. We treated transgenic male AR112Q mice with testosterone for 6 months. Surprisingly, testosterone treatment of AR112Q males did not exacerbate the disease. Although transgenic AR112Q males exhibited functional deficits when compared with non-transgenics, long-term testosterone treatment had no effect on motor function. Testosterone treatment also failed to affect cellular markers of disease, including inclusion formation (the accumulation of large nuclear aggregates of mutant AR protein) and levels of unphosphorylated neurofilament heavy chain. These data suggest that the mechanism of disease in SBMA saturates at close to endogenous hormone levels and that individuals with SBMA who take, or have taken, testosterone for its putative therapeutic properties are unlikely to suffer adverse effects.
多项动物模型研究表明,睾丸酮在脊髓延髓肌肉萎缩症(SBMA)症状的发展中起着至关重要的作用,这种疾病会导致受影响男性的神经退行性变和肌肉萎缩。携带编码人类雄激素受体(AR)的转基因小鼠,该受体包含一段 112 个谷氨酰胺(扩展多谷氨酰胺链;AR112Q 小鼠),可复制人类疾病的多个方面。我们用睾丸酮治疗了转基因雄性 AR112Q 小鼠 6 个月。令人惊讶的是,睾丸酮治疗并没有使 AR112Q 雄性的疾病恶化。尽管与非转基因小鼠相比,转基因 AR112Q 雄性表现出功能缺陷,但长期睾丸酮治疗对运动功能没有影响。睾丸酮治疗也未能影响疾病的细胞标志物,包括包涵体形成(突变型 AR 蛋白的大核聚集物的积累)和未磷酸化神经丝重链的水平。这些数据表明,SBMA 中的疾病机制在接近内源性激素水平时达到饱和,并且患有 SBMA 并因潜在的治疗特性而服用或已服用睾丸酮的个体不太可能遭受不良影响。