Literature DB >> 17630040

Achondroplasia.

William A Horton1, Judith G Hall2, Jacqueline T Hecht3.   

Abstract

Achondroplasia is the most common form of short limb dwarfism in human beings, affecting more than 250,000 individuals worldwide. More than 95% of patients have the same point mutation in the gene for fibroblast growth factor receptor 3 (FGFR3) and more than 80% of these are new mutations. The mutation, which causes gain of FGFR3 function, affects many tissues, most strikingly the cartilaginous growth plate in the growing skeleton, leading to a variety of manifestations and complications. The biology of FGFR3 and the molecular and cellular consequences of the achondroplasia mutation are being elucidated, providing a more complete understanding of the disorder and a basis for future treatments targeted directly at relevant pathogenetic pathways. Furthermore, the natural history of the condition, which has been well delineated in childhood and adolescence, is being defined more fully in adults with achondroplasia; most of the serious complications can be modified favourably or prevented by anticipation and early treatment. Possible future treatments include chemical inhibition of receptor signalling, antibody blockade of receptor activation, and alteration of pathways that modulate the downstream propagation of FGFR3 signals.

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Year:  2007        PMID: 17630040     DOI: 10.1016/S0140-6736(07)61090-3

Source DB:  PubMed          Journal:  Lancet        ISSN: 0140-6736            Impact factor:   79.321


  144 in total

1.  Genetic inactivation of ERK1 and ERK2 in chondrocytes promotes bone growth and enlarges the spinal canal.

Authors:  Arjun Sebastian; Takehiko Matsushita; Aya Kawanami; Susan Mackem; Gary E Landreth; Shunichi Murakami
Journal:  J Orthop Res       Date:  2010-10-04       Impact factor: 3.494

Review 2.  Sixteen years and counting: the current understanding of fibroblast growth factor receptor 3 (FGFR3) signaling in skeletal dysplasias.

Authors:  Silvie Foldynova-Trantirkova; William R Wilcox; Pavel Krejci
Journal:  Hum Mutat       Date:  2011-11-16       Impact factor: 4.878

Review 3.  FGFR3-related dwarfism and cell signaling.

Authors:  Daisuke Harada; Yoshitaka Yamanaka; Koso Ueda; Hiroyuki Tanaka; Yoshiki Seino
Journal:  J Bone Miner Metab       Date:  2008-12-09       Impact factor: 2.626

Review 4.  Advances in Skeletal Dysplasia Genetics.

Authors:  Krista A Geister; Sally A Camper
Journal:  Annu Rev Genomics Hum Genet       Date:  2015-04-22       Impact factor: 8.929

5.  Surgical technique and outcomes for bilateral humeral lengthening for achondroplasia: 26-year experience.

Authors:  I Ginebreda; D Campillo-Recio; C Cárdenas; J Tapiolas; P Rovira; A Isart
Journal:  Musculoskelet Surg       Date:  2018-12-10

6.  Functional characteristics of mesenchymal stem cells derived from the adipose tissue of a patient with achondroplasia.

Authors:  Jeong-Ran Park; Hanbyeol Lee; Chung-Hyo Kim; Seok-Ho Hong; Kwon-Soo Ha; Se-Ran Yang
Journal:  In Vitro Cell Dev Biol Anim       Date:  2016-04-08       Impact factor: 2.416

7.  FGF signaling in the osteoprogenitor lineage non-autonomously regulates postnatal chondrocyte proliferation and skeletal growth.

Authors:  Kannan Karuppaiah; Kai Yu; Joohyun Lim; Jianquan Chen; Craig Smith; Fanxin Long; David M Ornitz
Journal:  Development       Date:  2016-04-06       Impact factor: 6.868

Review 8.  Disorders of the growth plate.

Authors:  Chanika Phornphutkul; Philip A Gruppuso
Journal:  Curr Opin Endocrinol Diabetes Obes       Date:  2009-12       Impact factor: 3.243

9.  FGFR3/fibroblast growth factor receptor 3 inhibits autophagy through decreasing the ATG12-ATG5 conjugate, leading to the delay of cartilage development in achondroplasia.

Authors:  Xiaofeng Wang; Huabing Qi; Quan Wang; Ying Zhu; Xianxing Wang; Min Jin; Qiaoyan Tan; Qizhao Huang; Wei Xu; Xiaogang Li; Liang Kuang; Yubing Tang; Xiaolan Du; Di Chen; Lin Chen
Journal:  Autophagy       Date:  2015-11-02       Impact factor: 16.016

10.  FGFR3 induces degradation of BMP type I receptor to regulate skeletal development.

Authors:  Huabing Qi; Min Jin; Yaqi Duan; Xiaolan Du; Yuanquan Zhang; Fangli Ren; Yinyin Wang; Qingyun Tian; Xiaofeng Wang; Quan Wang; Ying Zhu; Yangli Xie; Chuanju Liu; Xu Cao; Yuji Mishina; Di Chen; Chu-xia Deng; Zhijie Chang; Lin Chen
Journal:  Biochim Biophys Acta       Date:  2014-03-20
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