Stüve-Wiedemann syndrome: long-term follow-up and genetic heterogeneity.

Details

Serval ID
serval:BIB_2B7FF34737C7
Type
Article: article from journal or magazin.
Publication sub-type
Case report (case report): feedback on an observation with a short commentary.
Collection
Publications
Title
Stüve-Wiedemann syndrome: long-term follow-up and genetic heterogeneity.
Journal
Clinical Genetics
Author(s)
Jung C., Dagoneau N., Baujat G., Le Merrer M., David A., Di Rocco M., Hamel B., Mégarbané A., Superti-Furga A., Unger S., Munnich A., Cormier-Daire V.
ISSN
1399-0004 (Electronic)
ISSN-L
0009-9163
Publication state
Published
Issued date
2010
Volume
77
Number
3
Pages
266-272
Language
english
Abstract
Stüve-Wiedemann syndrome (SWS, OMIM 601559) is a severe autosomal recessive condition caused by mutations in the leukemia inhibitory receptor (LIFR) gene. The main characteristic features are bowing of the long bones, neonatal respiratory distress, swallowing/sucking difficulties and dysautonomia symptoms including temperature instability often leading to death in the first years of life. We report here four patients with SWS who have survived beyond 36 months of age with no LIFR mutation. These patients have been compared with six unreported SWS survivors carrying null LIFR mutations. We provide evidence of clinical homogeneity of the syndrome in spite of the genetic heterogeneity.
Keywords
Abnormalities, Multiple/genetics, Abnormalities, Multiple/physiopathology, Female, Follow-Up Studies, Genes, Recessive, Genetic Heterogeneity, Humans, Leukemia Inhibitory Factor Receptor alpha Subunit/genetics, Male, Osteochondrodysplasias/genetics, Osteochondrodysplasias/physiopathology, Syndrome
Pubmed
Web of science
Create date
14/03/2011 17:09
Last modification date
20/08/2019 14:10
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