Document Detail


Facioaudiosymphalangism syndrome and growth acceleration associated with a heterozygous NOG mutation.
MedLine Citation:
PMID:  20503332     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
We report on a father and son with facioaudiosymphalangism syndrome, an alternative designation for multiple synostoses syndrome, type I. This syndrome includes synostosis, brachydactyly, craniofacial dysmorphic features, stapes fixation, hyperopia, and growth retardation. In contrast to the typical presentation, the height of the 10-year-old son was above the 97th centile from the age of 3.5 years and he had markers of an activated bone metabolism. The father and son had a novel heterozygous missense mutation c.696C > G, p.Cys232Trp, in the NOG gene. While not yet described in human NOG-related disorders, there is experimental evidence that suppression of noggin might accelerate osteogenesis, which could explain the phenotype in the family reported here.
Authors:
Sabine Rudnik-Schöneborn; Tsutomu Takahashi; Sabine Busse; Thorsten Schmidt; Jan Senderek; Thomas Eggermann; Klaus Zerres
Publication Detail:
Type:  Case Reports; Journal Article    
Journal Detail:
Title:  American journal of medical genetics. Part A     Volume:  152A     ISSN:  1552-4833     ISO Abbreviation:  Am. J. Med. Genet. A     Publication Date:  2010 Jun 
Date Detail:
Created Date:  2010-05-26     Completed Date:  2010-08-30     Revised Date:  2010-09-28    
Medline Journal Info:
Nlm Unique ID:  101235741     Medline TA:  Am J Med Genet A     Country:  United States    
Other Details:
Languages:  eng     Pagination:  1540-4     Citation Subset:  IM    
Copyright Information:
(c) 2010 Wiley-Liss, Inc.
Affiliation:
Medical Faculty, Institute of Human Genetics, RWTH Aachen University, Aachen, Germany. srudnik-schoeneborn@ukaachen.de
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MeSH Terms
Descriptor/Qualifier:
Carrier Proteins / genetics*
Child
Craniofacial Abnormalities / genetics*
Finger Phalanges / abnormalities*,  radiography
Growth Disorders / genetics*
Heterozygote
Humans
Male
Mutation, Missense
Pedigree
Syndrome
Synostosis / genetics*,  radiography
Young Adult
Chemical
Reg. No./Substance:
0/Carrier Proteins; 148294-77-3/noggin protein

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