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      Msx1 deficient mice exhibit cleft palate and abnormalities of craniofacial and tooth development.

      Nature genetics

      Transcription Factors, genetics, embryology, Tooth Abnormalities, abnormalities, Skull, Phenotype, Morphogenesis, Molecular Sequence Data, Mice, Mutant Strains, Mice, pathology, Mesoderm, Malleus, Male, MSX1 Transcription Factor, Jaw Abnormalities, Humans, Homeodomain Proteins, Head, Genes, Recessive, Genes, Lethal, Genes, Homeobox, Female, Facial Bones, Embryonic Induction, Embryo Transfer, Disease Models, Animal, Dental Papilla, physiology, DNA-Binding Proteins, Cleft Palate, Chimera, Cell Line, Animals, Abnormalities, Multiple, Base Sequence

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          Abstract

          The Msx1 homeobox gene is expressed at diverse sites of epithelial-mesenchymal interaction during vertebrate embryogenesis, and has been implicated in signalling processes between tissue layers. To determine the phenotypic consequences of its deficiency, we prepared mice lacking Msx1 function. All Msx1- homozygotes manifest a cleft secondary palate, a deficiency of alveolar mandible and maxilla and a failure of tooth development. These mice also exhibit abnormalities of the nasal, frontal and parietal bones, and of the malleus in the middle ear. Msx1 thus has a critical role in mediating epithelial-mesenchymal interactions during craniofacial bone and tooth development. The Msx1-/Msx1- phenotype is similar to human cleft palate, and provides a genetic model for cleft palate and oligodontia in which the defective gene is known.

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          Most cited references 29

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              Differential staining of cartilage and bone in whole mouse fetuses by alcian blue and alizarin red S.

              The procedure described by Inouye ('76) for the staining of full-term mouse fetal skeletons has been adapted for use with mouse embryos and fetuses of days 14-18 of gestation. The main adaptations for younger specimens involve a longer time in acetone, in lieu of skinning, and omission of the aqueous KOH step. These adaptations require more time but result in consistently good staining of intact specimens.
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                Author and article information

                Journal
                10.1038/ng0494-348
                7914451

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