Shoc2 controls ERK1/2-driven neural crest development by balancing components of the extracellular matrix

• DOI: 10.1101/2022.06.13.495941
• Publication Date: 2022-06-14T11:45:19Z
• Authors: Rebecca G. Norcross; Lina Abdelmoti; Eric C. Rouchka; Kalina Andreeva; Olivia Tussey; Daileen Landestoy; Emilia Galperin
• Keywords: 0301 basic medicine; 03 medical and health sciences; MAP Kinase Signaling System; Neural Crest; Noonan Syndrome; Intracellular Signaling Peptides and Proteins; Loose Anagen Hair Syndrome; Animals; Humans; Zebrafish; Extracellular Matrix

ABSTRACT The extracellular signal-regulated kinase (ERK1/2) pathway is essential in embryonic development. scaffold protein Shoc2 a critical modulator of ERK1/2 signals, and mutations the shoc2 gene lead to human developmental disease known as Noonan-like syndrome with loose anagen hair (NSLH). loss NSLH-causing affect tissues neural crest (NC) origin. In this study, we utilized zebrafish model dissect role Shoc2-ERK1/2 signals development NC. These studies established that significantly altered expression transcription factors regulating specification differentiation NC cells. Using comparative transcriptome analysis NC-derived cells from CRISPR/Cas9 mutant larvae, found Shoc2-mediated regulate programs at several levels, including genes coding for proteins matrix (ECM) ECM regulators. Together, our results demonstrate an regulator This study also indicates disbalance turnover may abnormalities NSLH patients.