Document Detail


Spondylar dysplasia in type X collagenopathy.
MedLine Citation:
PMID:  11214689     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
BACKGROUND: The type X collagen gene (COL10A1) is currently known as the disease-causing gene of metaphyseal dysplasia type Schmid (MDS), whereas a mutation of COL10A1 has been reported to cosegregate with a disease phenotype of mild spondylometaphyseal dysplasia (SMD) in a Japanese family. OBJECTIVE: To elucidate whether or not spondylar dysplasia is common in patients with mutations of COL10A1. MATERIALS AND METHODS: We re-evaluated the radiological manifestations in six patients with mutations of COL10A1, who had been previously reported as having MDS. RESULTS: Two of six patients showed mild platyspondyly in infancy and early childhood. In both patients, the spondylar dysplasia tended to normalize with age, but mild alterations of the vertebral bodies persisted, even into late childhood. The other radiological manifestations of both patients were identical to those of MDS. CONCLUSION: Our observation suggests that mild spondylar dysplasia may not be uncommon in MDS.
Authors:
G Nishimura; N Manabe; K Kosaki; N Haga; H Ohashi; K Nakamura; S Ikegawa
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Publication Detail:
Type:  Case Reports; Journal Article    
Journal Detail:
Title:  Pediatric radiology     Volume:  31     ISSN:  0301-0449     ISO Abbreviation:  Pediatr Radiol     Publication Date:  2001 Feb 
Date Detail:
Created Date:  2001-02-14     Completed Date:  2001-04-26     Revised Date:  2004-11-17    
Medline Journal Info:
Nlm Unique ID:  0365332     Medline TA:  Pediatr Radiol     Country:  Germany    
Other Details:
Languages:  eng     Pagination:  76-80     Citation Subset:  IM    
Affiliation:
Nishi-Tama Hospital, Tokyo, Japan. gen-n@dokkpmed.ac.jp
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MeSH Terms
Descriptor/Qualifier:
Bones of Upper Extremity / abnormalities,  radiography
Collagen / genetics*
Heterozygote
Humans
Infant
Japan
Leg Bones / abnormalities,  radiography
Male
Mutation
Osteochondrodysplasias / genetics*,  radiography*
Spine / abnormalities,  radiography
Chemical
Reg. No./Substance:
9007-34-5/Collagen

From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine


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