Muragaki Y, Mundlos S, Upton J, Olsen B R
Department of Cell Biology, Harvard Medical School, Boston, MA 02115, USA.
Science. 1996 Apr 26;272(5261):548-51. doi: 10.1126/science.272.5261.548.
Hox genes regulate patterning during limb development. It is believed that they function in the determination of the timing and extent of local growth rates. Here, it is demonstrated that synpolydactyly, an inherited human abnormality of the hands and feet, is caused by expansions of a polyalanine stretch in the amino-terminal region of HOXD13. The homozygous phenotype includes the transformation of metacarpal and metatarsal bones to short carpal- and tarsal-like bones. The mutations identify the polyalanine stretch outside of the DNA binding domain of HOXD13 as a region necessary for proper protein function.
Hox基因在肢体发育过程中调控模式形成。据信它们在决定局部生长速率的时间和程度方面发挥作用。在此,研究表明并指多指畸形(一种人类手部和足部的遗传性异常)是由HOXD13氨基末端区域的聚丙氨酸序列延伸扩增所引起的。纯合子表型包括掌骨和跖骨转变为短的腕骨样和跗骨样骨骼。这些突变确定了HOXD13 DNA结合域之外的聚丙氨酸序列延伸是蛋白质正常功能所必需的区域。