Hedgehog (HH) signalling is crucial for nervous system patterning. Genetic variants in key HH regulators, such as Gpr161, have been clinically linked to cranial neural tube closure defects (exencephaly) – a congenitally lethal condition. However, the role of Gpr161 in cranial neural tube closure remains unclear. In their latest study, Eric Brooks, Saikat Mukhopadhyay and colleagues present a substantial advance to our understanding of HH pathway logic across the developing murine nervous system by showing that ciliary GPR161 regulates HH signalling in different ways along the neural tube, with different consequences for cell remodelling and tube-closure. To learn more about this work and the people behind it, we talked to first author Sun-Hee Hwang, corresponding author Saikat Mukhopadhyay, and both first and corresponding author Eric Brooks.
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