Back to Search Start Over

CDON contributes to Hedgehog-dependent patterning and growth of the developing limb

Authors :
Martha L, Echevarría-Andino
Nicole E, Franks
Hannah E, Schrader
Mingi, Hong
Robert S, Krauss
Benjamin L, Allen
Source :
Developmental Biology. 493:1-11
Publication Year :
2023
Publisher :
Elsevier BV, 2023.

Abstract

Hedgehog (HH) signaling is a major driver of tissue patterning during embryonic development through the regulation of a multitude of cell behaviors including cell fate specification, proliferation, migration, and survival. HH ligands signal through the canonical receptor PTCH1 and three co-receptors, GAS1, CDON and BOC. While previous studies demonstrated an overlapping and collective requirement for these co-receptors in early HH-dependent processes, the early embryonic lethality of Gas1;Cdon;Boc mutants precluded an assessment of their collective contribution to later HH-dependent signaling events. Specifically, a collective role for these co-receptors during limb development has yet to be explored. Here, we investigate the combined contribution of these co-receptors to digit specification, limb patterning and long bone growth through limb-specific conditional deletion of Cdon in a Gas1;Boc null background. Combined deletion of Gas1, Cdon and Boc in the limb results in digit loss as well as defects in limb outgrowth and long bone patterning. Taken together, these data demonstrate that GAS1, CDON and BOC are collectively required for HH-dependent patterning and growth of the developing limb.

Details

ISSN :
00121606
Volume :
493
Database :
OpenAIRE
Journal :
Developmental Biology
Accession number :
edsair.doi.dedup.....f95616c0a8b4d3e5eb9f6e34a001925f