Dendrite development occupies a central position in the formation of nervous system. However, whether lysosomal degradative function is required for dendritogenesis of neurons remains unknown. We have recently demonstrated the critical role of Vps18 in the lysosomal degradation pathway in mice. Here, we report that Vps18 deficiency severely blocks the dendrite development of Pukinje cells but not cerebral cortical neurons. Furthermore, we also demonstrate that the lysyl oxidase (Lox) protein is degraded through lysosome and accumulated in the Vps18 deficient cerebellum but not in cerebral cortices. Our results suggest that lysosome regulates dendritogenesis of Purkinje cells though degrading Lox. (C) 2012 Elsevier Inc. All rights reserved.