Sylvie Schneider‐Maunoury

Spinal lumen remodeling under the control of Gli signaling mechanically drives roof plate cells extension

The paper shows that Gli signaling controls lumen retraction in the zebrafish spinal cord, which drives roof plate cell extension during development.

The Joubert gene TMEM67 is required for the correct establishment of spinal dorsal identities in human organoids.

This study shows that TMEM67 is essential for proper dorsal spinal cord patterning in human organoids, with loss of TMEM67 disrupting interneuron specification and altering BMP4 signaling.

The primary cilium controls programmed cell death via its proteasome-regulating function

The paper shows that primary cilia regulate programmed cell death by controlling the proteasomal degradation of MOAP1 through ciliopathy proteins like RPGRIP1L, TCTN1, and CEP290.

Regulation of FGF/MAPK signaling at the primary cilium base controls the proliferation-neurogenesis balance in human cerebellar organoids

This study shows that ciliary dysfunction in human cerebellar organoids leads to overactivation of FGF/MAPK signaling, impairing neurogenesis and increasing progenitor proliferation.

A differential requirement for ciliary transition zone proteins in human and mouse neural progenitor fate specification

This study compares the role of RPGRIP1L in human and mouse neural progenitor fate specification, finding distinct effects on motoneuron identity and SHH signaling.

Astrogliosis And Neuroinflammation Underlie Scoliosis Upon Cilia Dysfunction

This study identifies astrogliosis and neuroinflammation as pathogenic mechanisms underlying scoliosis in zebrafish caused by cilia dysfunction.

PLANAR POLARIZATION OF CILIA IN THE ZEBRAFISH FLOOR PLATE INVOLVES Par3-MEDIATED POSTERIOR LOCALIZATION OF HIGHLY MOTILE BASAL BODIES.

Primary cilium-dependent cAMP/PKA signaling at the centrosome regulates neuronal migration

The paper shows that primary cilia regulate neuronal migration through cAMP/PKA signaling at the centrosome, with genetic and imaging experiments demonstrating the role of ciliary Adenylate Cyclase 3 and centrosomal PKA.

A transient role of primary cilia in controlling direct versus indirect neurogenesis in the developing cerebral cortex

This study shows that primary cilia regulate direct versus indirect neurogenesis in the developing cerebral cortex, with Inpp5e mutations causing increased direct neurogenesis and layer V neuron overproduction.

Loss of the Reissner Fiber and increased URP neuropeptide signaling underliescoliosis in a zebrafish ciliopathy mutant

The Ciliopathy Gene Ftm/Rpgrip1l Controls Mouse Forebrain Patterning Via Region-Specific Modulation Of Hedgehog/Gli Signaling.

This study investigates the role of the ciliopathy gene Ftm/Rpgrip1l in mouse forebrain development, revealing its region-specific modulation of Hedgehog/Gli signaling in the diencephalon, hypothalamus, and eyes.

An in vivo translation-reporter system for the study of protein synthesis in zebrafish embryos

The paper introduces a method to visualize protein synthesis in zebrafish embryos by using a fluorescent reporter system to identify sites of translation.