Loxl2 and Loxl3 Paralogues Play Redundant Roles during Mouse Development

Lysyl oxidase-like 2 (LOXL2) and 3 (LOXL3) are members of the lysyl oxidase family of enzymes involved in the maturation of the extracellular matrix. Both enzymes share a highly conserved catalytic domain, but it is unclear whether they perform redundant functions in vivo. In this study, we show tha...

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Detalles Bibliográficos
Autores: Santamaría, Patricia G., Dubus, Pierre R., Bustos-Tauler, José, Floristán, Alfredo, Vázquez-Naharro, Alberto, Morales, Saleta, Cano, Amparo, Portillo, Francisco
Tipo de recurso: artículo
Estado:Versión publicada
Fecha de publicación:2022
País:España
Institución:Consejo Superior de Investigaciones Científicas (CSIC)
Repositorio:DIGITAL.CSIC. Repositorio Institucional del CSIC
OAI Identifier:oai:digital.csic.es:10261/295444
Acceso en línea:http://hdl.handle.net/10261/295444
Access Level:acceso abierto
Palabra clave:Lysyl oxidases
Loxl2
Loxl3
Epistasis analysis
Embryonic lethality
Descripción
Sumario:Lysyl oxidase-like 2 (LOXL2) and 3 (LOXL3) are members of the lysyl oxidase family of enzymes involved in the maturation of the extracellular matrix. Both enzymes share a highly conserved catalytic domain, but it is unclear whether they perform redundant functions in vivo. In this study, we show that mice lacking Loxl3 exhibit perinatal lethality and abnormal skeletal development. Additionally, analysis of the genotype of embryos carrying double knockout of Loxl2 and Loxl3 genes suggests that both enzymes have overlapping functions during mouse development. Furthermore, we also show that ubiquitous expression of Loxl2 suppresses the lethality associated with Loxl3 knockout mice.