The use of Caenorhabditis elegans in neurological research has been instrumental in advancing our understanding of multiple biological processes. The model has been employed with success towards obtaining an understanding of human brain function and diseases. With more than 60% of its genes having human counterparts and 42% of human disease-associated genes finding orthologues in C. elegans, this model organism offers unique insights into the conserved mechanisms of neuronal function and disease. The development of transgenic C. elegans lines has further facilitated detailed studies into neurodegenerative diseases (NDs) such as Parkinson’s disease (PD), Alzheimer’s disease (AD), and Amyotrophic Lateral Sclerosis (ALS), highlighting the roles of neuronal loss and dysfunctional protein homeostasis. Latest research efforts employing C. elegans as a model system have expanded into studying “glial cells” as well. This important cell population is now known to be critically associated with multiple processes, including remodelling into neurons and neuronal repair. Additionally, C. elegans has proven to be a vital tool for neurotoxicological assessments and the pharmacological screening of potential drug therapies. This chapter delineates the critical milestones achieved through C. elegans research, emphasizing its impact on the discovery of disease pathways and the exploration of therapeutic strategies within the domain of neurological research.