APHP- Necker Enfants Malades Hospital 1 article published in JoVE Developmental Biology 2D and 3D Human Induced Pluripotent Stem Cell-Based Models to Dissect Primary Cilium Involvement during Neocortical Development Lucile Boutaud*1, Marie Michael*1, Céline Banal2, Damelys Calderon1, Sarah Farcy1, Julie Pernelle1, Nicolas Goudin3, Camille Maillard1, Clémantine Dimartino1, Cécile Deleschaux1, Sébastien Dupichaud4, Corinne Lebreton1, Sophie Saunier1, Tania Attié-Bitach1,5, Nadia Bahi-Buisson1,6, Nathalie Lefort2, Sophie Thomas1 1Imagine Institute, INSERM UMR 1163, Université de Paris, 2Imagine Institute, iPSC Core Facility, INSERM UMR U1163, Université de Paris, 3Necker Bio-image Analysis platform of the SFR Necker, 4Imagine Institute, Cell Imaging Platform, INSERM-US24-CNRS UMS 3633 Structure Fédérative de Recherche Necker, INSERM UMR U1163, Université de Paris, 5Fédération de Génétique, Hôpital Necker-Enfants Malades, Assistance Publique Hôpitaux de Paris, 6Pediatric Neurology, APHP- Necker Enfants Malades Hospital We present detailed protocols for the generation and characterization of 2D and 3D human induced pluripotent stem cell (hIPSC)-based models of neocortical development as well as complementary methodologies enabling qualitative and quantitative analysis of primary cilium (PC) biogenesis and function.