Literature DB >> 9853502

Genotype phenotype correlation in achondroplasia and hypochondroplasia.

Y Matsui1, N Yasui, T Kimura, N Tsumaki, H Kawabata, T Ochi.   

Abstract

Recent studies of the fibroblast growth factor receptor 3 (FGFR3) gene have established that achondroplasia and hypochondroplasia are allelic disorders of different mutations. To determine whether the genotype could be distinguished on the basis of the phenotype, we analysed height, arm span, and skeletal radiographs from 23 patients with achondroplasia and the G380R mutation of FGFR3 and eight with hypochondroplasia and the N540K mutation. Both conditions share the classical pathological features of micromelic short stature, reduced or unchanged interpedicular distances in the lumbar spine, disproportionately long fibulae, and squared and shortened pelvic ilia. These were significantly more severe in the G380R patients than in the N540K patients. Our findings have shown a firm statistical correlation between the genotype and the phenotype, although there were a few exceptional cases in which there was phenotypic overlap between the two conditions.

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Year:  1998        PMID: 9853502     DOI: 10.1302/0301-620x.80b6.9277

Source DB:  PubMed          Journal:  J Bone Joint Surg Br        ISSN: 0301-620X


  12 in total

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7.  FGFR3 mutation frequency in 324 cases from the International Skeletal Dysplasia Registry.

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10.  Concomitant achondroplasia and developmental dysplasia of the hip.

Authors:  Tennison L Malcolm; Duy L Phan; Ran Schwarzkopf
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