title: Cellular analysis of limb development in the mouse mutant hypodactyly.
abstract: The limb defect in the mouse Hypodactyly (Hd) affects only the distal structures. Heterozygotes (Hd/+) lack all or part of the distal phalanx and the terminal claw of digit on the hindlimbs; mice homozygous (Hd/Hd) for the mutation have just one digit on each of the four limbs. Early limb development in the mutant appears normal and a change in morphology can only be detected later. Limb buds of Hd/+ and Hd/Hd embryos become reduced in width, with Hd/Hd buds becoming very pointed instead of rounded. This change in bud shape is correlated with an increase in cell death anteriorly in Hd/+ hindlimbs and both anteriorly and posteriorly in Hd/Hd fore- and hindlimb buds. The apical ectodermal ridge is very pronounced in pointed Hd/Hd limb buds. Mesenchyme cells from the Hd/Hd mutant in culture show a cell-autonomous change in behaviour and less cartilage differentiates.
authors.affiliation: Department of Anatomy and Developmental Biology, University College London, United Kingdom.
authors.forename: K E
authors.initials: KE
authors.lastname: Robertson
authors.forename: M H
authors.initials: MH
authors.lastname: Chapman
authors.forename: A
authors.initials: A
authors.lastname: Adams
authors.forename: C
authors.initials: C
authors.lastname: Tickle
authors.forename: S M
authors.initials: SM
authors.lastname: Darling
mesh_terms: D000818:Animals; D001842:Bone and Bones / Q000166:cytology / Q000196:embryology; D002356:Cartilage / Q000166:cytology / Q000196:embryology; D016923:Cell Death; D002478:Cells, Cultured; D000013:Congenital Abnormalities / Q000235:genetics*; D004622:Embryo, Mammalian / Q000648:ultrastructure; D005314:Embryonic and Fetal Development / Q000235:genetics*; D005260:Female; D005838:Genotype; D005865:Gestational Age; D006614:Hindlimb / Q000002:abnormalities*; D018878:Limb Buds / Q000166:cytology; D008297:Male; D051379:Mice; D008809:Mice, Inbred C3H; D008810:Mice, Inbred C57BL; D008817:Mice, Mutant Strains; D008855:Microscopy, Electron, Scanning; D011247:Pregnancy