TY  - JOUR
TI  - Genetic interaction of <I>Gli3 </I> and <I>Alx4 </I> during limb development
AU  - Panman, Lia
AU  - Drenth, Thijs
AU  - Tewelscher, Pascal
AU  - Zuniga, Aimee
AU  - Zeller, Rolf
T2  - The International Journal of Developmental Biology
AB  - The Gli3  and Alx4  transcriptional regulators are expressed in the anterior limb bud mesenchyme and their disruption in mice results in preaxial polydactyly. While the polydactylous phenotype of Alx4  deficient limb buds depends on SHH, the one of Gli3  deficient limb buds is completely independent of SHH signalling, suggesting that these genes act in parallel pathways. Analysis of limb buds lacking both Gli3  and Alx4  now shows that these two genes interact during limb skeletal morphogenesis. In addition to the defects in single mutants, the stylopod is severely malformed and the anterior element of the zeugopod is lost in double mutant limbs. However, limb bud patterning in Gli3-/- ; Alx4-/-  double mutant embryos is not affected more than in single mutants as the expression domains of key regulators remain the same. Most interestingly, the loss of the severe preaxial polydactyly characteristic of Gli3 -/- limbs in double mutant embryos establishes that this type of polydactyly requires Alx4 function.
PY  - 2005
DO  - 10.1387/ijdb.051984lp
VL  - 49
IS  - 4
SP  - 443
EP  - 448
J2  - Int. J. Dev. Biol.
LA  - en
SN  - 0214-6282
SN  - 1696-3547
UR  - https://ijdb.ehu.eus/article/051984lp
Y2  - 2024/04/26/23:46:28
ER  -