The　c-Jun　N-terminal　kinase　(JNK)　is　highly　evolutionarily　conserved　and　plays　important　roles　in　a　broad　range　of　physiological　and　pathological　processes.　The　WD40-repeat　protein　62　(WDR62)　is　a　scaffold　protein　that　recruits　different　components　of　the　JNK　signaling　pathway　to　regulate　several　human　diseases　including　neurological　disorders,　infertility,　and　tumorigenesis.　Recent　studies　revealed　that　WDR62　regulates　the　process　of　neural　stem　cell　mitosis　and　germ　cell　meiosis　through　JNK　signaling.　In　this　review　we　summarize　the　roles　of　WDR62　and　JNK　signaling　in　neuronal　and　non-neuronal　contexts　and　discuss　how　JNK-dependent　signaling　regulates　both　processes.　WDR62　is　involved　in　various　human　disorders　via　JNK　signaling　regulation,　and　may　represent　a　promising　therapeutic　strategy　for　the　treatment　of　related　diseases.