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Pax3 Hypomorphs Reveal Hidden Pax7 Functional Genetic Compensation in Utero.


ABSTRACT: Pax3 and Pax7 transcription factors are paralogs within the Pax gene family that that are expressed in early embryos in partially overlapping expression domains and have distinct functions. Significantly, mammalian development is largely unaffected by Pax7 systemic deletion but systemic Pax3 deletion results in defects in neural tube closure, neural crest emigration, cardiac outflow tract septation, muscle hypoplasia and in utero lethality by E14. However, we previously demonstrated that Pax3 hypomorphs expressing only 20% functional Pax3 protein levels exhibit normal neural tube and heart development, but myogenesis is selectively impaired. To determine why only some Pax3-expressing cell lineages are affected and to further titrate Pax3 threshold levels

SUBMITTER: Zhou HM 

PROVIDER: S-EPMC9149870 | biostudies-literature | 2022 May

REPOSITORIES: biostudies-literature