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Neural crest cells require Meis2 for patterning the mandibular arch via the Sonic hedgehog pathway
- Source :
- Biology Open, Vol 9, Iss 6 (2020), Biology Open, article-version (VoR) Version of Record
- Publication Year :
- 2020
- Publisher :
- The Company of Biologists, 2020.
-
Abstract
- Cranial neural crest cells (cNCCs) originate in the anterior neural tube and populate pharyngeal arches in which they contribute to formation of bone and cartilage. This cell population also provides molecular signals for the development of tissues of non-neural crest origin, such as the tongue muscles, teeth enamel or gland epithelium. Here we show that the transcription factor Meis2 is expressed in the oral region of the first pharyngeal arch (PA1) and later in the tongue primordium. Conditional inactivation of Meis2 in cNCCs resulted in loss of Sonic hedgehog signalling in the oropharyngeal epithelium and impaired patterning of PA1 along the lateral–medial and oral–aboral axis. Failure of molecular specification of PA1, illustrated by altered expression of Hand1/2, Dlx5, Barx1, Gsc and other markers, led to hypoplastic tongue and ectopic ossification of the mandible. Meis2-mutant mice thus display craniofacial defects that are reminiscent of several human syndromes and patients with mutations in the Meis2 gene.<br />Summary: Wnt1-Cre2;Meis2fl/fl mutants have perturbed Shh activity in the oral region of the mandibular arch, which leads to specification failure of NCCs in the tongue primordium and ectopic ossification at the expense of glossogenesis, which are hallmarks of several mouse mutants with eliminated Shh activity.
- Subjects :
- QH301-705.5
Organogenesis
Science
Mice, Transgenic
Mandible
craniofacial
General Biochemistry, Genetics and Molecular Biology
meis
Mice
Cranial neural crest
Dental Arch
Tongue
medicine
Animals
Hedgehog Proteins
Sonic hedgehog
Biology (General)
Alleles
Body Patterning
Homeodomain Proteins
biology
First pharyngeal arch
Neural tube
Neural crest
Calcinosis
sonic hedgehog (shh) signalling
Anatomy
Immunohistochemistry
Hedgehog signaling pathway
pharyngeal arch
medicine.anatomical_structure
Phenotype
Neural Crest
embryonic structures
biology.protein
General Agricultural and Biological Sciences
Pharyngeal arch
Biomarkers
Gene Deletion
Signal Transduction
Research Article
Subjects
Details
- Language :
- English
- ISSN :
- 20466390
- Volume :
- 9
- Issue :
- 6
- Database :
- OpenAIRE
- Journal :
- Biology Open
- Accession number :
- edsair.doi.dedup.....6b3fb6c53d4525861cf4a90aee55daf6