The two domain hypothesis of limb prepattern and its relevance to congenital limb anomalies.
Functional annotation of mutations that cause human limb anomalies is enabled by basic developmental studies. In this study, we focus on the prepatterning stage of limb development and discuss a recent model that proposes anterior and posterior domains of the early limb bud generate two halves of the future skeleton. By comparing phenotypes in humans with those in model organisms, we evaluate whether this prepatterning concept helps to annotate human disease alleles. WIREs Dev Biol 2017, 6:e270. doi: 10.1002/wdev.270 For further resources related to this article, please visit the WIREs website.
Program in Developmental and Stem Cell Biology, Research Institute, The Hospital for Sick Children, Toronto, Canada.
Hirotaka
H
Tao
Department of Genetics, Cell Biology and Development, Stem Cell Institute, University of Minnesota, Minneapolis, MN, USA.
Yasuhiko
Y
Kawakami
Program in Developmental and Stem Cell Biology, Research Institute, The Hospital for Sick Children, Toronto, Canada.|Department of Molecular Genetics, University of Toronto, Toronto, Canada.
Chi-Chung
CC
Hui
Program in Developmental and Stem Cell Biology, Research Institute, The Hospital for Sick Children, Toronto, Canada.|Department of Molecular Genetics, University of Toronto, Toronto, Canada.|Division of Orthopaedic Surgery, Hospital for Sick Children and University of Toronto, Toronto, Canada.
Sevan
S
Hopyan
D000818:Animals; D005121:Extremities / Q000196:embryology* / Q000502:physiology*; D018507:Gene Expression Regulation, Developmental; D006801:Humans; D017880:Limb Deformities, Congenital / Q000473:pathology*