title: Severe defects in the formation of epaxial musculature in open brain (opb) mutant mouse embryos.
abstract: The differentiation of somite derivatives is dependent on signals from neighboring axial structures. While ventral signals have been described extensively, little is known about dorsal influences, especially those from the dorsal half of the neural tube. Here, we describe severe phenotypic alterations in dorsal somite derivatives of homozygous open brain (opb) mutant mouse embryos which suggest crucial interactions between dorsal neural tube and dorsal somite regions. At Theiler stage 17 (day 10.5 post coitum) of development, strongly altered expression patterns of Pax3 and Myf5 were observed in dorsal somite regions indicating that the dorsal myotome and dermomyotome were not differentiating properly. These abnormalities were later followed by the absence of epaxial (dorsal) musculature; whereas, body wall and limb musculature formed normally. Analysis of Mox1 and Pax1 expression in opb embryos revealed additional defects in the differentiation of the dorsal sclerotome. The observed abnormalities coincided with defects in differentiation of dorsal neural tube regions. The implications of our findings for interactions between dorsal neural tube, surface ectoderm and dorsomedial somite regions in specifying epaxial musculature are discussed.
authors.affiliation: GSF - Forschungszentrum für Umwelt und Gesundheit, Institut für Säugetiergenetik, Neuherberg, Oberschleissheim, Germany.
authors.forename: R
authors.initials: R
authors.lastname: Spörle
authors.forename: T
authors.initials: T
authors.lastname: Günther
authors.forename: M
authors.initials: M
authors.lastname: Struwe
authors.forename: K
authors.initials: K
authors.lastname: Schughart
mesh_terms: D000818:Animals; D001921:Brain / Q000196:embryology*; D004268:DNA-Binding Proteins / Q000235:genetics; D005260:Female; D017403:In Situ Hybridization; D051379:Mice; D008817:Mice, Mutant Strains; D009124:Muscle Proteins / Q000235:genetics; D018482:Muscle, Skeletal / Q000196:embryology*; D009154:Mutation; D051787:Myogenic Regulatory Factor 5; D009436:Neural Tube Defects / Q000196:embryology* / Q000235:genetics*; D000072041:PAX3 Transcription Factor; D051761:Paired Box Transcription Factors; D010641:Phenotype; D011247:Pregnancy; D011506:Proteins / Q000235:genetics; D013116:Spinal Cord / Q000196:embryology; D015534:Trans-Activators; D014157:Transcription Factors; D051153:Wnt Proteins; D060508:Wnt3 Protein
chemical_list: D004268:DNA-Binding Proteins; D009124:Muscle Proteins; C497418:Myf5 protein, mouse; D051787:Myogenic Regulatory Factor 5; D000072041:PAX3 Transcription Factor; D051761:Paired Box Transcription Factors; D011506:Proteins; D015534:Trans-Activators; D014157:Transcription Factors; D051153:Wnt Proteins; D060508:Wnt3 Protein; C068686:Pax3 protein, mouse