Literature DB >> 8723102

Radiological and histological variants of thanatophoric dysplasia are associated with common mutations in FGFR-3.

A G Nerlich1, P Freisinger, J Bonaventure.   

Abstract

We describe two fetuses of the 21st week of gestation that share some macroscopic, radiologic, and histologic findings of thanatophoric dysplasia (TD), but also show distinct differences from the usual subtypes of TD. These differences mainly comprise the lack of facial abnormality, only mild reduction of chondrocyte proliferation and hypertrophy, and the lack of fibrous tissue interposition between cartilage and periosteal bone. Thus, these two cases may represent a distinct variant of thanatophoric dysplasia. The molecular analysis of the FGF-R-3 gene demonstrated in both cases mutations which were not significantly different from those of other cases of TD. Thus, the phenotypic modulation within the subtypes of TD may be influenced by additional and yet unknown factors.

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Year:  1996        PMID: 8723102     DOI: 10.1002/(SICI)1096-8628(19960503)63:1<155::AID-AJMG27>3.0.CO;2-M

Source DB:  PubMed          Journal:  Am J Med Genet        ISSN: 0148-7299


  2 in total

Review 1.  Molecular basis for skeletal variation: insights from developmental genetic studies in mice.

Authors:  C Kappen; A Neubüser; R Balling; R Finnell
Journal:  Birth Defects Res B Dev Reprod Toxicol       Date:  2007-12

Review 2.  Survival to adulthood and dominant inheritance of platyspondylic skeletal dysplasia, Torrance-Luton type.

Authors:  Luitgard Neumann; Jürgen Kunze; Markus Uhl; Brigitte Stöver; Bernhard Zabel; Jürgen Spranger
Journal:  Pediatr Radiol       Date:  2003-09-05
  2 in total

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