A cell-autonomous role for primary cilium-mediated signaling in long-range commissural axon guidance.

ABSTRACT: Ciliopathies are characterized by the absence or dysfunction of primary cilia. Despite the fact that cognitive impairments are a common feature of ciliopathies, how cilia dysfunction affects neuronal development has not been characterized in detail. Here, we show that primary cilium-mediated signaling is required cell-autonomously by neurons during neural circuit formation. In particular, a functional primary cilium is crucial during axonal pathfinding for the switch in responsiveness of axons at a choice point or intermediate target. Using different animal models and in vivo, ex vivo and in vitro experiments, we provide evidence for a crucial role of primary cilium-mediated signaling in long-range axon guidance. The primary cilium on the cell body of commissural neurons transduces long-ra

SUBMITTER: Dumoulin A 

PROVIDER: S-EPMC11423920 | biostudies-literature | 2024 Sep

REPOSITORIES: biostudies-literature