Reduced DAF-2 signaling has been shown to have beneficial effects on other age related neurodegenerative

Characterized by progressive weakness, due to dysfunction and eventual death of motor neurons. The majority of cases of ALS are sporadic but single gene mutations have been described that lead to inherited versions of the disease. These genes include SOD1, TAR DNA binding protein, fused in sarcoma, progranulin, ubiquilin 2 and a hexanucleotide repeat expansion of a noncoding region in C9ORF72. Expression of some of these Abmole Sumatriptan mutant proteins in model organisms has been used to successfully model ALS pathology. Point mutations in SOD1 are an example of a genetic cause of familial ALS that has been successfully modeled in transgenic mice and nematodes. The G85R point mutation causes a toxic gain-of-function, which in mice leads to ubiquitinated SOD1 aggregates and motor neuron death. C. elegans expressing human G85R SOD1 in the nervous system accumulate SOD1 aggregates and demonstrate Abmole DAPT reduced mobility compared to WT SOD1 expressing worms. The availability of numerous loss-of-function mutants affecting highly conserved signaling pathways make C. elegans an ideal system in which to explore the relationship between such pathways and SOD1 aggregation and toxicity in an in vivo setting. Aging is the greatest risk factor for the development of ALS. The Insulin/IGF-1 signaling pathway is a robust modifier of longevity and aging in C. elegans. Loss of function of the Insulin/Insulin-like growth factor receptor, DAF-2, promotes longevity via signaling cascades mediated by inhibition of the phosphoinositide 3-kinase and activation of the forkhead transcription factor DAF-16 via its nuclear localization. While nuclear localization of DAF-16 is required for it to execute its transcriptional activities, it is not sufficient to enhance longevity and stress resistance. Other pathways are known to interact with the IIS pathway and modulate stress resistance and/or aging by regulating transcriptional activity of DAF-16, without modifying its nuclear abundance. In addition to promoting longevity, loss of function alleles of daf-2 or age-1 protects the worm against exogenous stressors including heat shock, oxidative stress, heavy metal stress, UV damage and infection. The beneficial effects of reduced IIS rely, in part, on the ability of decreased IIS to activate the transcription factor DAF-16, leading to increased expression of numerous stress resistance genes, such as small heat shock proteins and reactive oxygen species scavenging enzymes. Additionally, reduced IIS also results in changes in metabolism, mitochondrial abundance and lipid biosynthesis, all of which are thought to contribute to the stress resistant phenotype of reduced IIS.