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Mcrs1 is required for branchial arch and cranial cartilage development.
Keer, Stephanie; Cousin, Helene; Jourdeuil, Karyn; Neilson, Karen M; Tavares, Andre L P; Alfandari, Dominique; Moody, Sally A.
Afiliación
  • Keer S; Department of Anatomy and Cell Biology, The George Washington University School of Medicine and Health Sciences, 2300 I (eye) Street, NW, Washington, DC, 20037, USA.
  • Cousin H; Department of Animal Science, University of Massachusetts Amherst, Integrated Science Building, 661 N. Pleasant Street, Amherst, MA, 01003, USA.
  • Jourdeuil K; Department of Anatomy and Cell Biology, The George Washington University School of Medicine and Health Sciences, 2300 I (eye) Street, NW, Washington, DC, 20037, USA.
  • Neilson KM; Department of Anatomy and Cell Biology, The George Washington University School of Medicine and Health Sciences, 2300 I (eye) Street, NW, Washington, DC, 20037, USA.
  • Tavares ALP; Department of Anatomy and Cell Biology, The George Washington University School of Medicine and Health Sciences, 2300 I (eye) Street, NW, Washington, DC, 20037, USA.
  • Alfandari D; Department of Animal Science, University of Massachusetts Amherst, Integrated Science Building, 661 N. Pleasant Street, Amherst, MA, 01003, USA.
  • Moody SA; Department of Anatomy and Cell Biology, The George Washington University School of Medicine and Health Sciences, 2300 I (eye) Street, NW, Washington, DC, 20037, USA. Electronic address: samoody@gwu.edu.
Dev Biol ; 489: 62-75, 2022 09.
Article en En | MEDLINE | ID: mdl-35697116
Mcrs1 is a multifunctional protein that is critical for many cellular processes in a wide range of cell types. Previously, we showed that Mcrs1 binds to the Six1 transcription factor and reduces the ability of the Six1-Eya1 complex to upregulate transcription, and that Mcrs1 loss-of-function leads to the expansion of several neural plate genes, reduction of neural border and pre-placodal ectoderm (PPR) genes, and pleiotropic effects on various neural crest (NC) genes. Because the affected embryonic structures give rise to several of the cranial tissues affected in Branchio-otic/Branchio-oto-renal (BOR) syndrome, herein we tested whether these gene expression changes subsequently alter the development of the proximate precursors of BOR affected structures - the otic vesicles (OV) and branchial arches (BA). We found that Mcrs1 is required for the expression of several OV genes involved in inner ear formation, patterning and otic capsule cartilage formation. Mcrs1 knockdown also reduced the expression domains of many genes expressed in the larval BA, derived from either NC or PPR, except for emx2, which was expanded. Reduced Mcrs1 also diminished the length of the expression domain of tbx1 in BA1 and BA2 and interfered with cranial NC migration from the dorsal neural tube; this subsequently resulted in defects in the morphology of lower jaw cartilages derived from BA1 and BA2, including the infrarostral, Meckel's, and ceratohyal as well as the otic capsule. These results demonstrate that Mcrs1 plays an important role in processes that lead to the formation of craniofacial cartilages and its loss results in phenotypes consistent with reduced Six1 activity associated with BOR.
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Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Región Branquial / Síndrome Branquio Oto Renal Idioma: En Revista: Dev Biol Año: 2022 Tipo del documento: Article País de afiliación: Estados Unidos Pais de publicación: Estados Unidos
Texto completo
Añadir a Mi BVS
Imprimir
XML
PubMed Links
Buscar en Google

Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Región Branquial / Síndrome Branquio Oto Renal Idioma: En Revista: Dev Biol Año: 2022 Tipo del documento: Article País de afiliación: Estados Unidos Pais de publicación: Estados Unidos