PMID:7720575

Citation	Fawcett, D, Pasceri, P, Fraser, R, Colbert, M, Rossant, J and Giguère, V (1995) Postaxial polydactyly in forelimbs of CRABP-II mutant mice. Development 121:671-9
Abstract	The cytoplasmic retinoic acid (RA)-binding protein CRABP-II is expressed widely throughout early morphogenesis in mouse embryo, but its expression becomes more restricted as organogenesis progresses. CRABP-II expression remains strong in the developing limb bud suggesting a role for this protein in limb patterning. Here, we show that the CRABP-II promoter can direct expression of a lacZ transgene in a specific posterior domain during limb bud development. In order to investigate in more detail the role played by CRABP-II in RA signal transduction, we have also generated mice homozygous for a null mutation of this gene. CRABPII-/- mice are viable and fertile but show a developmental defect of the forelimb, specifically an additional, postaxial digit. This digit is generally, but not exclusively, limited to a single forepaw of an individual animal. The penetrance of the phenotype varies according to the genetic background, occurring most frequently on the inbred 129Sv background (50%), less frequently on the C57Bl/6 background (30%) and rarely on the outbred CD1 background (10%). This developmental abnormality implies a role for CRABP-II in normal patterning of the limb.
Links	PubMed
Keywords	Animals; Forelimb/abnormalities; Gene Expression; Immunohistochemistry; Mice; Mice, Transgenic; Morphogenesis/drug effects; Morphogenesis/genetics; Phenotype; Polydactyly/embryology; Polydactyly/genetics; Promoter Regions, Genetic; Receptors, Retinoic Acid/genetics; Signal Transduction; Tretinoin/pharmacology
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Gene product	Qualifier	GO ID	GO term name	Evidence Code	with/from	Aspect	Notes	Status
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