Institute for Neurosciences of Montpellier (INM), INSERM U 1051 Montpellier, France ; "Integrative Biology of Neurodegeneration," IKERBASQUE Basque Foundation for Science, Neuroscience Department, University of the Basque Country Bilbao, Spain.
Institute for Neurosciences of Montpellier (INM), INSERM U 1051 Montpellier, France.
Front Cell Neurosci. 2013 Dec 27;7:280. doi: 10.3389/fncel.2013.00280. eCollection 2013.
Amyotrophic lateral sclerosis (ALS) is a fatal neurodegenerative disorder typified by a massive loss of motor neurons with few therapeutic options. The exact cause of neuronal degeneration is unknown but it is now admitted that ALS is a multifactorial disease with several mechanisms involved including glutamate excitotoxicity. More specifically, N-methyl-D-aspartate (NMDA)-mediated cell death and impairment of the glutamate-transport has been suggested to play a key role in ALS pathophysiology. Thus, evaluating NMDAR antagonists is of high therapeutic interest. Gacyclidine, also named GK11, is a high affinity non-competitive NMDAR antagonist that may protect against motor neuron death in an ALS context. Moreover, GK11 presents a low intrinsic neurotoxicity and has already been used in two clinical trials for CNS lesions. In the present study, we investigated the influence of chronic administration of two doses of GK11 (0.1 and 1 mg/kg) on the survival and the functional motor activity of hSOD1(G93A) mice, an animal model of ALS. Treatment started at early symptomatic age (60 days) and was applied bi-weekly until the end stage of the disease. We first confirmed that functional alteration of locomotor activity was evident in the hSOD1(G93A) transgenic female mice by 60 days of age. A low dose of GK11 improved the survival of the mice by 4.3% and partially preserved body weight. Improved life span was associated with a delay in locomotor function impairment. Conversely, the high dose treatment worsened motor functions. These findings suggest that chronic administration of GK11 beginning at early symptomatic stage may be beneficial for patients with ALS.
肌萎缩侧索硬化症(ALS)是一种致命的神经退行性疾病,其特征是运动神经元大量丧失,治疗选择很少。神经元退化的确切原因尚不清楚,但现在人们承认 ALS 是一种多因素疾病,涉及多种机制,包括谷氨酸兴奋性毒性。更具体地说,N-甲基-D-天冬氨酸(NMDA)介导的细胞死亡和谷氨酸转运的损害被认为在 ALS 病理生理学中起关键作用。因此,评估 NMDA 受体拮抗剂具有很高的治疗意义。加替沙星,也称为 GK11,是一种高亲和力的非竞争性 NMDA 受体拮抗剂,可能在 ALS 背景下保护运动神经元死亡。此外,GK11 表现出低固有神经毒性,并且已经在两项针对 CNS 病变的临床试验中使用。在本研究中,我们研究了两种剂量的 GK11(0.1 和 1mg/kg)慢性给药对 hSOD1(G93A)小鼠(ALS 的动物模型)的存活和功能性运动活动的影响。治疗从早期症状期(60 天)开始,并每两周应用一次,直到疾病的终末期。我们首先证实,60 天龄的 hSOD1(G93A)转基因雌性小鼠的运动活动功能改变明显。低剂量的 GK11 使小鼠的存活率提高了 4.3%,并部分维持了体重。寿命的延长与运动功能障碍的延迟有关。相反,高剂量治疗恶化了运动功能。这些发现表明,从早期症状期开始慢性给予 GK11 可能对 ALS 患者有益。