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Biology subjects

Meilhac, S. M.

Publications and source records attributed to Meilhac, S. M..

3 recordsLinked to original sources

Wnt and Nodal asymmetries stratify mouse laterality phenotypes in the absence of node flow

Left-right symmetry breaking in mice is considered to occur via node leftward fluid flow. The molecular cascade within the node has been reconstructed at high resolution. However, its importance for organogenesis remains poorly understood. Here we show in mutants for the motile cilium component CCDC40 that 70% of mice develop normal situs at birth despite abrogation of node flow. The discrete morphospace output, including situs inversus totalis and heterotaxy with left isomerism, supports a novel model of symmetry-breaking, in which node flow only biases asymmetry orientation, while another mechanism, potentially self-amplifying, generates it. Longitudinal, quantitative and paired transcriptomic analyses uncover the molecular signature of laterality clusters, highlighting WNT in addition to NODAL pathway asymmetry. We identify asymmetry of cardiopulmonary progenitors, the disruption of which is associated with combined heart and lung defects in mutants. Our work refines the role of the node in the establishment of asymmetry.

developmental biology↗

VANGL2 shapes the mouse heart tube from adjacent epithelia and without planar polarity

Disruption of the core Planar Cell Polarity component VANGL2 in mice is associated with congenital heart defects and impaired morphogenesis of the embryonic heart tube. However, the underlying mechanisms have remained unclear. Here we quantified in 3D the heart geometry and adjacent tissue architecture in a series of mutants to reveal a dual role of Vangl2 in shaping the heart tube. Together with cell labelling in the chick, we show that VANGL2 in multicellular junctions promotes second heart field cell rearrangements and thus the elongation of the arterial pole. In addition, apically localised VANGL2 and its downstream actin-binding effector SHROOM3 control the bilateral symmetry of the splanchnic mesoderm caudal to the venous pole. Disorganisation of this midline anchor is associated with a rotation of the heart tube and abnormal left ventricle position. Our work overall uncovers novel specific epithelial roles of VANGL2 unrelated to planar polarity during heart morphogenesis.

developmental biology↗

Intraflagellar transport complex B proteins regulate the Hippo effector Yap1 during cardiogenesis

Cilia and the intraflagellar transport (IFT) proteins involved in ciliogenesis are associated with congenital heart diseases (CHD). However, the molecular links between cilia, IFT proteins and cardiogenesis are yet to be established. Using a combination of biochemistry, genetics, and live imaging methods, we show that IFT complex B proteins (Ift88, Ift54 and Ift20) modulate the Hippo pathway effector YAP1 in zebrafish and mouse. We demonstrate that this interaction is key to restrict the formation of the proepicardium and the myocardium. In cellulo experiments suggest that IFT88 and IFT20 interact with YAP1 in the cytoplasm and functionally modulates its activity, identifying a molecular link between cilia related proteins and the Hippo pathway. Taken together, our results highlight a novel role for IFT complex B proteins during cardiogenesis and shed light on an unexpected mechanism of action for ciliary proteins in YAP1 regulation. These findings provide mechanistic insights into a non-canonical role for cilia related proteins during cardiogenesis.

developmental biology↗