Document Detail


A novel Xp22.11 deletion causing a syndrome of craniosynostosis and periventricular nodular heterotopia.
MedLine Citation:
PMID:  22052819     Owner:  NLM     Status:  Publisher    
Abstract/OtherAbstract:
We report on a follow-up evaluation of a male with a phenotype including craniosynostosis, periventricular nodular heterotopia, and neurodevelopmental delay. He was initially assigned a clinical diagnosis of Fontaine-Farriaux syndrome (FFS) as an infant although now, with improved delineation of this entity, it is evident that this diagnosis is not applicable to this individual. Array comparative genomic hybridization has demonstrated a 300 kb interstitial deletion on Xp22.11 affecting all or part of three annotated genes, ZFX, PDK3, and PCYT1B in this subject. The deletion was inherited from the phenotypically normal mother who also exhibited markedly skewed X-inactivation. These findings implicate hemizygosity for one or all three of these genes as the cause of this phenotype. © 2011 Wiley Periodicals, Inc.
Authors:
Margriet van Kogelenberg; Margherita Lerone; Teresa De Toni; Maria T Divizia; Arjan P M de Brouwer; Joris A Veltman; Hans van Bokhoven; Stephen P Robertson
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Publication Detail:
Type:  LETTER     Date:  2011-11-3
Journal Detail:
Title:  American journal of medical genetics. Part A     Volume:  -     ISSN:  1552-4833     ISO Abbreviation:  -     Publication Date:  2011 Nov 
Date Detail:
Created Date:  2011-11-4     Completed Date:  -     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  101235741     Medline TA:  Am J Med Genet A     Country:  -    
Other Details:
Languages:  ENG     Pagination:  -     Citation Subset:  -    
Copyright Information:
Copyright © 2011 Wiley Periodicals, Inc.
Affiliation:
Department of Paediatrics and Child Health, Dunedin School of Medicine, Otago University, Dunedin, New Zealand.
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