Literature DB >> 29019756

Imaging of Skeletal Disorders Caused by Fibroblast Growth Factor Receptor Gene Mutations.

Kiran M Sargar1, Achint K Singh1, Simon C Kao1.   

Abstract

Fibroblast growth factors and fibroblast growth factor receptors (FGFRs) play important roles in human axial and craniofacial skeletal development. FGFR1, FGFR2, and FGFR3 are crucial for both chondrogenesis and osteogenesis. Mutations in the genes encoding FGFRs, types 1-3, are responsible for various skeletal dysplasias and craniosynostosis syndromes. Many of these disorders are relatively common in the pediatric population, and diagnosis is often challenging. These skeletal disorders can be classified based on which FGFR is affected. Skeletal disorders caused by type 1 mutations include Pfeiffer syndrome (PS) and osteoglophonic dysplasia, and disorders caused by type 2 mutations include Crouzon syndrome (CS), Apert syndrome (AS), and PS. Disorders caused by type 3 mutations include achondroplasia, hypochondroplasia, thanatophoric dysplasia (TD), severe achondroplasia with developmental delay and acanthosis nigricans, Crouzonodermoskeletal syndrome, and Muenke syndrome. Most of these mutations are inherited in an autosomal dominant fashion and are gain-of-function-type mutations. Imaging plays a key role in the evaluation of these skeletal disorders. Knowledge of the characteristic imaging and clinical findings can help confirm the correct diagnosis and guide the appropriate molecular genetic tests. Some characteristics and clinical findings include premature fusion of cranial sutures and deviated broad thumbs and toes in PS; premature fusion of cranial sutures and syndactyly of the hands and feet in AS; craniosynostosis, ocular proptosis, and absence of hand and foot abnormalities in CS; rhizomelic limb shortening, caudal narrowing of the lumbar interpediculate distance, small and square iliac wings, and trident hands in achondroplasia; and micromelia, bowing of the femora, and platyspondyly in TD. ©RSNA, 2017.

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Year:  2017        PMID: 29019756     DOI: 10.1148/rg.2017170017

Source DB:  PubMed          Journal:  Radiographics        ISSN: 0271-5333            Impact factor:   5.333


  10 in total

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3.  Chiari 1 malformation and exome sequencing in 51 trios: the emerging role of rare missense variants in chromatin-remodeling genes.

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Journal:  Hum Genet       Date:  2020-12-18       Impact factor: 4.132

Review 4.  Pictorial review: imaging of the spinal manifestations of achondroplasia.

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5.  Mutational landscape and genetic signatures of cell-free DNA in tumour-induced osteomalacia.

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Journal:  J Cell Mol Med       Date:  2020-04-11       Impact factor: 5.310

6.  The association of genetic variants in FGFR2 with osteoporosis susceptibility in Chinese Han population.

Authors:  Yang Yang; Mengxue Fei; Xinying Zhou; Yuejun Li; Dadi Jin
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7.  The primary role of radiological imaging in the diagnosis of rare musculoskeletal diseases. Emphasis on ultrasound.

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Journal:  JBMR Plus       Date:  2021-11-09

Review 9.  Identification of a novel mutation of FGFR3 gene in a large Chinese pedigree with hypochondroplasia by next-generation sequencing: A case report and brief literature review.

Authors:  Guixiang Yao; Guangxin Wang; Dawei Wang; Guohai Su
Journal:  Medicine (Baltimore)       Date:  2019-01       Impact factor: 1.817

10.  HSPB7 regulates osteogenic differentiation of human adipose derived stem cells via ERK signaling pathway.

Authors:  Chanyuan Jin; Ting Shuai; Zhihui Tang
Journal:  Stem Cell Res Ther       Date:  2020-10-23       Impact factor: 6.832

  10 in total

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