Syndactyly and preaxial synpolydactyly in the single Sfrp2 deleted mutant mice

Developmental Dynamics : an Official Publication of the American Association of Anatomists
Masaya IkegawaKei Tashiro

Abstract

Secreted Frizzled-related protein 2 (Sfrp2) or Stromal Cell Derived Factor-5 (SDF-5) is highly expressed in the developing limbs. Here we showed the single Sfrp2 inactivation in mice resulted in syndactyly and preaxial synpolydactyly, predominantly in the hindlimbs. Tails were often kinked. A penetrance of the syndactyly was highest in 129/SvJ or CBA/N x 129/SvJ background and the phenotype was haploinsufficient. Preaxial synpolydactyly was seen in homozygous mutants in C57BL/6 x 129/SvJ. Of note, syndactyly showed retarded apoptosis of the second and the third interdigital spaces; concomitantly, mesodermal Msx2 expression was down-regulated. Impaired digital anlagen maturation was also noticeable in the same position. Preaxial synpolydactyly of the Sfrp2 mutants was a non-mirror image type and Shh independent. Although joint formation was not disrupted, chondrocyte maturation was preaxially disturbed. Our results suggest that the Sfrp2 deleted mice can be a useful animal model to study human syndactyly/preaxial synpolydactyly defects.

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Citations

Nov 6, 2014·BMC Research Notes·Yuichi TokudaKei Tashiro
Mar 27, 2012·The Open Orthopaedics Journal·D JordanW Khan
Aug 17, 2020·Developmental Dynamics : an Official Publication of the American Association of Anatomists·Juan A MonteroJuan M Hurle
May 15, 2015·Molecular and Cellular Biochemistry·Mi-Jin KimSeung-Yoon Park
Nov 4, 2020·Cancer Metastasis Reviews·Karlijn van LoonArjan W Griffioen

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