| Autor: |
Dunn, N.Ray, Winnier, Glenn E., Hargett, Linda K., Schrick, Jeffrey J., Fogo, Agnes B., Hogan, Brigid L.M. |
| Zdroj: |
Developmental Biology; August 1997, Vol. 188 Issue: 2 p235-247, 13p |
| Abstrakt: |
Bone morphogenetic protein 4(Bmp4), a vertebrate homolog ofDrosophila decapentaplegic(dpp), encodes a signaling protein with multiple functions during embryogenesis. Most mouse embryos homozygous for theBmp4tm1blhnull allele die around the time of gastrulation, with little or no mesoderm. Two independently derivedBmp4tm1mutations were backcrossed onto the C57BL/6 genetic background. Several independently expressed, incompletely penetrant abnormalities were observed in heterozygotes, including cystic kidney, craniofacial malformations, microphthalmia, and preaxial polydactyly of the right hindlimb. In addition, heterozygotes were consistently underrepresented at weaning. These results indicate thatBmp4gene dosage is essential for the normal development of a variety of organs and for neonatal viability. Two mutations that enhance the penetrance and expressivity of the polydactylous phenotype were identified:Gli3XtJ,a deletion mutation involving a gene encoding a zinc-finger protein related toDrosophilacubitus interruptus, andAlx4tm1rwm,a targeted null mutation in a gene encoding a paired class homeoprotein related toDrosophilaaristaless. All doubleBmp4tm1; Gli3XtJheterozygotes have extensive anterior digit abnormalities of both fore- and hindlimbs, while all doubleBmp4tm1; Alx4tm1heterozygotes display ectopic anterior digits only on the hindlimbs. These genetic interactions suggest a model for the multigenic control of anterior digit patterning during vertebrate limb development. |
| Databáze: |
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