ArticleDevelopmental dynamics : an official publication of the American Association of Anatomists2019
Nuclear FGFR2 regulates musculoskeletal integration within the developing limb.
Article in Developmental dynamics : an official publication of the American Association of Anatomists, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 6 papers.
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Who cites it
6 citing papers in PubMed, 8 citations in OpenAlex.
- The heterozygous c.2241G>C variation in FGFR2 may cause autosomal dominant Kirner's deformity in a Chinese Han pedigree.BMC medical genomics · 2026Article
- New developments in the biology of fibroblast growth factors.WIREs mechanisms of disease · 2022Review
- Genomic adaptations for arboreal locomotion in Asian flying treefrogs.Proceedings of the National Academy of Sciences of the United States of America · 2022Article
- FGF7-FGFR2 autocrine signaling increases growth and chemoresistance of fusion-positive rhabdomyosarcomas.Molecular oncology · 2022Article
- FGF/FGFR signaling in health and disease.Signal transduction and targeted therapy · 2020Review
- Nuclear Fibroblast Growth Factor Receptor Signaling in Skeletal Development and Disease.Current osteoporosis reports · 2019Review
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Authors and funding
4 authors at 1 institution in 1 country.
Funding
Abstract
backgroundBent bone dysplasia syndrome (BBDS), a congenital skeletal disorder caused by dominant mutations in fibroblast growth factor receptor 2 (FGFR2), is characterized by bowed long bones within the limbs. We previously showed that the FGFR2 mutations in BBDS enhance nuclear and nucleolar localization of the receptor; however, exactly how shifts in subcellular distribution of FGFR2 affect limb development remained unknown.
resultsTargeted expression of the BBDS mutations in the lateral plate mesoderm of the developing chick induced angulated hindlimbs, a hallmark feature of the disease. Whole-mount analysis of the underlying skeleton revealed bent long bones with shortened bone collars and, in severe cases, dysmorphic epiphyses. Epiphyseal changes were also correlated with joint dislocations and contractures. Histological analysis revealed that bent long bones and joint defects were closely associated with irregularities in skeletal muscle patterning and tendon-to-bone attachment. The spectrum of limb phenotypes induced by the BBDS mutations were recapitulated by targeted expression of wild-type FGFR2 appended with nuclear and nucleolar localization signals.
conclusionsOur results indicate that the bent long bones in BBDS arise from disruptions in musculoskeletal integration and that increased nuclear and nucleolar localization of FGFR2 plays a mechanistic role in the disease phenotype. 248:233-246, 2019. © 2018 Wiley Periodicals, Inc.
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