δEF1, a zinc finger and homeodomain transcription factor, is required for skeleton patterning in multiple lineages
δEF1 is a DNA binding protein containing a homeodomain and two zinc finger clusters, and is regarded as a vertebrate homologue of zfh-1 (zinc finger homeodomain-containing factor-1) in Drosophila. In the developing embryo, δEF1 is expressed in the notochord, somites, limb, neural crest derivatives a...
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Veröffentlicht in: | Development (Cambridge) 1998-01, Vol.125 (1), p.21-31 |
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Sprache: | eng |
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Zusammenfassung: | δEF1 is a DNA binding protein containing a homeodomain and two zinc finger clusters, and is regarded as a vertebrate homologue of zfh-1 (zinc finger homeodomain-containing factor-1) in Drosophila. In the developing embryo, δEF1 is expressed in the notochord, somites, limb, neural crest derivatives and a few restricted sites of the brain and spinal cord. To elucidate the regulatory function of δEF1 in mouse embryogenesis, we generated δEF1 null mutant (δEF1null(lacZ)) mice. The δEF1null(lacZ) homozygotes developed to term, but never survived postnatally. In addition to severe T cell deficiency of the thymus, the δEF1null(lacZ) homozygotes exhibited skeletal defects of various lineages. (1) Craniofacial abnormalities of neural crest origin: cleft palate, hyperplasia of Meckel’s cartilage, dysplasia of nasal septum and shortened mandible. (2) Limb defects: shortening and broadening of long bones, fusion of carpal/tarsal bone and fusion of joints. (3) Fusion of ribs. (4) Sternum defects: split and asymmetric ossification pattern of the sternebrae associated with irregular sternocostal junctions. (5) Hypoplasia of intervertebral discs. These results indicate that δEF1 has an essential role in regulating development of these skeletal structures. Since the skeletal defects were not observed in δEF1ΔC727 mice, δEF1 bears distinct regulatory activities which are dependent on different domains of the molecule. |
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ISSN: | 0950-1991 1477-9129 |
DOI: | 10.1242/dev.125.1.21 |