Differences in neuronal ciliation rate and ciliary content revealed by systematic imaging-based analysis of hiPSC-derived models across protocols.

ABSTRACT:

Introduction

Ciliopathies are a group of human Mendelian disorders caused by dysfunction of primary cilia, small quasi-ubiquitous sensory organelles. Patients suffering from ciliopathies often display prominent neurodevelopmental phenotypes, underscoring the importance of primary cilia during development and for function of the central nervous system (CNS). Human tissues, in particular from the CNS, are very hard to obtain for research. Patient derived- or genetically engineered human induced pluripotent stem cells (hiPSCs) are therefore a precious resource for investigating the role of cilia in human neurons.

Methods

In this study we used a variety of 2D and 3D neuronal differentiation protocols in multiple hiPSC lines and systematically analyzed ciliation rates and ciliary

SUBMITTER: Haenseler W 

PROVIDER: S-EPMC12021924 | biostudies-literature | 2025

REPOSITORIES: biostudies-literature