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

Journal Article · · American Journal of Medical Genetics
 [1];  [2];  [3]
  1. Pathologisches Institut der Universitaet Muenchen (Germany)
  2. Kinderlinik und Poliklinik der Technischen Universitaet, Muenchen (Germany)
  3. Institut Necker, Paris (France)

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. 16 refs., 4 figs., 1 tab.

OSTI ID:
441194
Journal Information:
American Journal of Medical Genetics, Journal Name: American Journal of Medical Genetics Journal Issue: 1 Vol. 63; ISSN 0148-7299; ISSN AJMGDA
Country of Publication:
United States
Language:
English

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