Morphogenesis of Doublefoot (Dbf), a mouse mutant with polydactyly and craniofacial defects

被引:13
作者
Hayes, C
Lyon, MF
Morriss-Kay, GM
机构
[1] Univ Oxford, Dept Human Anat, Oxford OX1 3QX, England
[2] MRC, Mammalian Genet Unit, Harwell, Berks, England
关键词
limb development; Sonic hedgehog;
D O I
10.1046/j.1469-7580.1998.19310081.x
中图分类号
R602 [外科病理学、解剖学]; R32 [人体形态学];
学科分类号
100101 ;
摘要
We report the morphogenesis of a new mouse mutant, Doublefoot (Dbf). The major phenotypic features involve the limb and craniofacial regions. There is polydactyly of all 4 limbs, with typically 6-8 digits per limb. All of the digits are triphalangeal; some show bifurcations and some are not attached to the carpus/tarsus. The carpus and tarsus are broader than normal, and their elements are partially fused. There are also tibial defects. Mutant embryos show a diencephalic bulge on d 10.0, with older animals exhibiting broadened and bulbous skulls sometimes with an additional midline skeletal element, shortened snouts and bulging eyes. Homozygotes, which do not survive beyond d 15, show midline facial clefting. In this study of the embryonic and fetal development of Dbf animals, we focus on the morphogenesis of the limbs and head, and discuss the possible molecular developmental mechanisms.
引用
收藏
页码:81 / 91
页数:11
相关论文
共 31 条
  • [1] CHAN DC, 1995, DEVELOPMENT, V121, P1971
  • [2] Cyclopia and defective axial patterning in mice lacking Sonic hedgehog gene function
    Chiang, C
    Ying, LTT
    Lee, E
    Young, KE
    Corden, JL
    Westphal, H
    Beachy, PA
    [J]. NATURE, 1996, 383 (6599) : 407 - 413
  • [3] Polarity, proliferation and the hedgehog pathway
    Dean, M
    [J]. NATURE GENETICS, 1996, 14 (03) : 245 - 247
  • [4] DIETZ FR, 1985, LANCET, P388
  • [5] DUNN PM, 1985, LANCET, P635
  • [6] FromentalRamain C, 1996, DEVELOPMENT, V122, P2997
  • [7] Altered neural cell fates and medulloblastoma in mouse patched mutants
    Goodrich, LV
    Milenkovic, L
    Higgins, KM
    Scott, MP
    [J]. SCIENCE, 1997, 277 (5329) : 1109 - 1113
  • [8] Hayes C, 1998, DEVELOPMENT, V125, P351
  • [9] A MOUSE MODEL OF GREIG CEPHALOPOLYSYNDACTYLY SYNDROME - THE EXTRA-TOES(J) MUTATION CONTAINS AN INTRAGENIC DELETION OF THE GLI3 GENE
    HUI, CC
    JOYNER, AL
    [J]. NATURE GENETICS, 1993, 3 (03) : 241 - 246
  • [10] EXPERIMENTAL-ANALYSIS OF THE INVIVO CHONDROGENIC POTENTIAL OF THE INTERDIGITAL MESENCHYME OF THE CHICK LEG BUD SUBJECTED TO LOCAL ECTODERMAL REMOVAL
    HURLE, JM
    GANAN, Y
    MACIAS, D
    [J]. DEVELOPMENTAL BIOLOGY, 1989, 132 (02) : 368 - 374