title: Tshz1 is required for axial skeleton, soft palate and middle ear development in mice.
abstract: Members of the Tshz gene family encode putative zinc fingers transcription factors that are broadly expressed during mouse embryogenesis. Tshz1 is detected from E9.5 in the somites, the spinal cord, the limb buds and the branchial arches. In order to assess the function of Tshz1 during mouse development, we generated Tshz1-deficient mice. Tshz1 inactivation leads to neonatal lethality and causes multiple developmental defects. In the craniofacial region, loss of Tshz1 function leads to specific malformations of middle ear components, including the malleus and the tympanic ring. Tshz1(-/-) mice exhibited Hox-like vertebral malformations and homeotic transformations in the cervical and thoracic regions, suggesting that Tshz1 and Hox genes are involved in common pathways to control skeletal morphogenesis. Finally, we demonstrate that Tshz1 is required for the development of the soft palate.
authors.affiliation: Institut de Biologie du Développement de Marseille-Luminy (IBDML), UMR6216, CNRS, Université de la Méditerranée, F-13288 Marseille cedex 09, France. ncore@ibdml.univ-mrs.fr
authors.forename: Nathalie
authors.initials: N
authors.lastname: Coré
authors.forename: Xavier
authors.initials: X
authors.lastname: Caubit
authors.forename: Aïcha
authors.initials: A
authors.lastname: Metchat
authors.forename: Annie
authors.initials: A
authors.lastname: Boned
authors.forename: Malek
authors.initials: M
authors.lastname: Djabali
authors.forename: Laurent
authors.initials: L
authors.lastname: Fasano
mesh_terms: D000818:Animals; D000831:Animals, Newborn; D001483:Base Sequence; D019521:Body Patterning; D001846:Bone Development / Q000235:genetics / Q000502:physiology*; D001842:Bone and Bones / Q000002:abnormalities; D017931:DNA Primers / Q000235:genetics; D004432:Ear, Middle / Q000002:abnormalities / Q000196:embryology*; D005260:Female; D018507:Gene Expression Regulation, Developmental; D005801:Genes, Homeobox; D018398:Homeodomain Proteins; D008297:Male; D051379:Mice; D008810:Mice, Inbred C57BL; D018345:Mice, Knockout; D010160:Palate, Soft / Q000002:abnormalities / Q000196:embryology*; D011247:Pregnancy; D012097:Repressor Proteins / Q000235:genetics / Q000502:physiology*; D014157:Transcription Factors / Q000172:deficiency / Q000235:genetics / Q000502:physiology*
chemical_list: D017931:DNA Primers; D018398:Homeodomain Proteins; D012097:Repressor Proteins; D014157:Transcription Factors; C489361:Tshz1 protein, mouse