Department of Natural Sciences, Black Hills State University, Spearfish, South Dakota, United States of America.
Department of Biology, University of Sioux Falls, Sioux Falls, South Dakota, United States of America.
PLoS One. 2023 Mar 9;18(3):e0278793. doi: 10.1371/journal.pone.0278793. eCollection 2023.
Chronic exposure to the Cyanobacteria biotoxin Beta-methylamino-L-alanine (BMAA) has been associated with development of a sporadic form of ALS called Amyotrophic Lateral Sclerosis/Parkinsonism-Dementia Complex (ALS/PDC), as observed within certain Indigenous populations of Guam and Japan. Studies in primate models and cell culture have supported the association of BMAA with ALS/PDC, yet the pathological mechanisms at play remain incompletely characterized, effectively stalling the development of rationally-designed therapeutics or application of preventative measures for this disease. In this study we demonstrate for the first time that sub-excitotoxic doses of BMAA modulate the canonical Wnt signaling pathway to drive cellular defects in human neuroblastoma cells, suggesting a potential mechanism by which BMAA may promote neurological disease. Further, we demonstrate here that the effects of BMAA can be reversed in cell culture by use of pharmacological modulators of the Wnt pathway, revealing the potential value of targeting this pathway therapeutically. Interestingly, our results suggest the existence of a distinct Wnt-independent mechanism activated by BMAA in glioblastoma cells, highlighting the likelihood that neurological disease may result from the cumulative effects of distinct cell-type specific mechanisms of BMAA toxicity.
慢性暴露于蓝藻生物毒素β-甲基氨基-L-丙氨酸(BMAA)与一种称为肌萎缩侧索硬化症/帕金森病痴呆复合征(ALS/PDC)的散发性 ALS 的发展有关,这种情况在关岛和日本的某些土著人群中观察到。灵长类动物模型和细胞培养研究支持 BMAA 与 ALS/PDC 的关联,但起作用的病理机制仍未完全描述,有效地阻碍了针对这种疾病的合理设计的治疗方法或预防措施的发展。在这项研究中,我们首次证明亚兴奋性剂量的 BMAA 可以调节经典的 Wnt 信号通路,从而导致人神经母细胞瘤细胞的细胞缺陷,这表明 BMAA 可能促进神经疾病的潜在机制。此外,我们在这里证明,在细胞培养中,Wnt 通路的药理学调节剂可逆转 BMAA 的作用,这揭示了靶向该通路治疗的潜在价值。有趣的是,我们的结果表明,BMAA 在神经胶质瘤细胞中激活了一种独特的 Wnt 非依赖性机制,这突出表明神经疾病可能是由 BMAA 毒性的不同细胞类型特异性机制的累积效应引起的。