Spns1-dependent endocardial lysosomal function drives valve morphogenesis through Notch1-signaling.

ABSTRACT: Autophagy-lysosomal degradation is a conserved homeostatic process considered to be crucial for cardiac morphogenesis. However, both its cell specificity and functional role during heart development remain unclear. Here, we introduced zebrafish models to visualize autophagic vesicles in vivo and track their temporal and cellular localization in the larval heart. We observed a significant accumulation of autolysosomal and lysosomal vesicles in the atrioventricular and bulboventricular regions and their respective valves. We addressed the role of lysosomal degradation based on the Spinster homolog 1 (spns1) mutant (not really started, nrs). n rs larvae displayed morphological and functional cardiac defects, including abnormal endocardial organization

SUBMITTER: Chavez MN 

PROVIDER: S-EPMC11667069 | biostudies-literature | 2024 Dec

REPOSITORIES: biostudies-literature