Altered growth and branching patterns in synpolydactyly caused by mutations in HOXD13

Y Muragaki, S Mundlos, J Upton, BR Olsen - Science, 1996 - science.org
Y Muragaki, S Mundlos, J Upton, BR Olsen
Science, 1996science.org
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 …
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.
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