Fink Kyle D, Rossignol Julien, Crane Andrew T, Davis Kendra K, Bavar Angela M, Dekorver Nicholas W, Lowrance Steven A, Reilly Mark P, Sandstrom Michael I, von Hörsten Stephan, Lescaudron Laurent, Dunbar Gary L
Program in Neuroscience, Central Michigan University, USA.
Behav Neurosci. 2012 Jun;126(3):479-87. doi: 10.1037/a0028028.
Huntington's disease (HD) is a neurodegenerative disorder in humans caused by an expansion of a CAG trinucleotide repeat that produces choreic movements, which are preceded by cognitive deficits. The HD transgenic rat (tgHD), which contains the human HD mutation with a 51 CAG repeat allele, exhibits motor deficits that begin when these rats are 12 months of age. However, there are no reports of cognitive dysfunction occurring prior to this. To assess whether cognitive dysfunction might precede motor deficits in tgHD rats, one group of 9-month-old male rats with homozygotic mutated genes and one group of wild-type (WT) rats underwent three testing phases in a unique Spatial Operant Reversal Test (SORT) paradigm, as well as assessment of spontaneous motor activity. After testing, morphological and histological examination of the brains were made. Results indicated that tgHD rats acquired the cued-response (Phase 1) portion of the SORT, but made significantly more errors during the reversal (Phase 2) and during the pseudorandomized reversals (Phase 3) portion of the study, when compared to WT rats. Analysis of the data using mathematical principles of reinforcement revealed no memory, motor, or motivational deficits. These results indicate that early cognitive dysfunction, as measured by the SORT, occur prior to motor deficits, gross anatomical changes, or cell loss in the tgHD rat with 51 CAG repeats, and suggest that this protocol could provide a useful screen for therapeutic studies.
亨廷顿舞蹈症(HD)是一种人类神经退行性疾病,由CAG三核苷酸重复序列扩增引起,会导致舞蹈样动作,且在此之前会出现认知缺陷。携带51个CAG重复等位基因的人类HD突变的HD转基因大鼠(tgHD),在12个月大时开始出现运动缺陷。然而,此前并无关于在此之前出现认知功能障碍的报道。为了评估tgHD大鼠的认知功能障碍是否可能先于运动缺陷出现,一组9个月大的纯合突变基因雄性大鼠和一组野生型(WT)大鼠在独特的空间操作性逆转试验(SORT)范式中经历了三个测试阶段,并对自发运动活动进行了评估。测试后,对大脑进行了形态学和组织学检查。结果表明,与WT大鼠相比,tgHD大鼠在SORT的线索反应(第1阶段)部分表现正常,但在研究的逆转(第2阶段)和伪随机逆转(第3阶段)部分出现了明显更多的错误。使用强化数学原理对数据进行分析,未发现记忆、运动或动机缺陷。这些结果表明,通过SORT测量,早期认知功能障碍在具有51个CAG重复序列的tgHD大鼠的运动缺陷、大体解剖学变化或细胞丢失之前就已出现,这表明该方案可为治疗研究提供有用的筛选方法。