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- W2951356483 endingPage "e1007765" @default.
- W2951356483 startingPage "e1007765" @default.
- W2951356483 abstract "Rare individuals with inactivating mutations in the Huntington’s disease gene (HTT) exhibit variable abnormalities that imply essential HTT roles during organ development. Here we report phenotypes produced when increasingly severe hypomorphic mutations in the murine HTT orthologue Htt, (HdhneoQ20, HdhneoQ50, HdhneoQ111), were placed over a null allele (Hdhex4/5). The most severe hypomorphic allele failed to rescue null lethality at gastrulation, while the intermediate, though still severe, alleles yielded recessive perinatal lethality and a variety of fetal abnormalities affecting body size, skin, skeletal and ear formation, and transient defects in hematopoiesis. Comparative molecular analysis of wild-type and Htt-null retinoic acid-differentiated cells revealed gene network dysregulation associated with organ development that nominate polycomb repressive complexes and miRNAs as molecular mediators. Together these findings demonstrate that Htt is required both pre- and post-gastrulation to support normal development." @default.
- W2951356483 created "2019-06-27" @default.
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- W2951356483 date "2019-03-21" @default.
- W2951356483 modified "2023-09-29" @default.
- W2951356483 title "Hypomorphic mutation of the mouse Huntington’s disease gene orthologue" @default.
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- W2951356483 doi "https://doi.org/10.1371/journal.pgen.1007765" @default.
- W2951356483 hasPubMedCentralId "https://www.ncbi.nlm.nih.gov/pmc/articles/6445486" @default.