نتایج جستجو برای: shprintzen syndrome

تعداد نتایج: 621913  

Journal: :Development 2008
David A Lyons Stephen G Naylor Sara Mercurio Claudia Dominguez William S Talbot

Mutations in Kif1-binding protein/KIAA1279 (KBP) cause the devastating neurological disorder Goldberg-Shprintzen syndrome (GSS) in humans. The cellular function of KBP and the basis of the symptoms of GSS, however, remain unclear. Here, we report the identification and characterization of a zebrafish kbp mutant. We show that kbp is required for axonal outgrowth and maintenance. In vivo time-lap...

Journal: :Human brain mapping 2012
Siddharth Srivastava Michael H Buonocore Tony J Simon

Chromosome 22q11.2 deletion syndrome (22q11.2DS) is a neurogenetic disorder associated with neurocognitive impairments. This article focuses on the cortical gyrification changes that are associated with the genetic disorder in 6-15-year-old children with 22q11.2DS, when compared with a group of age-matched typically developing (TD) children. Local gyrification index (lGI; Schaer et al. [2008]: ...

Journal: :Annals of internal medicine 2009
Irwin M Braverman Donald B Redford Philip A Mackowiak

Akhenaten was one of Egypt's most controversial pharaohs, in part because of his strange appearance in images produced after he had declared Aten, the Sun-disc, his one-and-only god. Whether these were symbolic representations or realistic ones that indicate a deforming genetic disorder is the subject of continuing debate. The authors present evidence that the bizarre physical features portraye...

Journal: :Developmental disabilities research reviews 2008
Tony J Simon

In this article, I present an updated account that attempts to explain, in cognitive processing and neural terms, the nonverbal intellectual impairments experienced by most children with deletions of chromosome 22q11.2. Specifically, I propose that this genetic syndrome leads to early developmental changes in the structure and function of clearly delineated neural circuits for basic spatiotempo...

Journal: :Archives of disease in childhood 1996
D G de Silva T P Gunawardena F M Law

Marfan's syndrome has diverse manifestations that overlap with those seen in other connective tissue disorders. Visceral diverticula have been described only once in four adults with marfanoid features of recessive inheritance. Two siblings of a consanguineous marriage with marfanoid features, visceral diverticula, and diaphragmatic eventration are reported.

2016
Kin Y Mok Una Sheerin Javier Simón-Sánchez Afnan Salaka Lucy Chester Valentina Escott-Price Kiran Mantripragada Karen M Doherty Alastair J Noyce Niccolo E Mencacci Steven J Lubbe Caroline H Williams-Gray Roger A Barker Karin D van Dijk Henk W Berendse Peter Heutink Jean-Christophe Corvol Florence Cormier Suzanne Lesage Alexis Brice Kathrin Brockmann Claudia Schulte Thomas Gasser Thomas Foltynie Patricia Limousin Karen E Morrison Carl E Clarke Stephen Sawcer Tom T Warner Andrew J Lees Huw R Morris Mike A Nalls Andrew B Singleton John Hardy Andrey Y Abramov Vincent Plagnol Nigel M Williams Nicholas W Wood

BACKGROUND Parkinson's disease has been reported in a small number of patients with chromosome 22q11.2 deletion syndrome. In this study, we screened a series of large, independent Parkinson's disease case-control studies for deletions at 22q11.2. METHODS We used data on deletions spanning the 22q11.2 locus from four independent case-control Parkinson's disease studies (UK Wellcome Trust Case ...

2013
Marianne B.M. van den Bree Gregory Miller Elizabeth Mansell Anita Thapar Frances Flinter Michael J. Owen

With advances in laboratory technology, an increasing number of potentially pathogenic CNVs is recognised. The phenotypic effects of some CNVs are well characterised, however, it remains unclear how much information reaches the parents of affected children and by what route. The 22q11.2 deletion syndrome (del22q11.2) is caused by the deletion of approximately 40 genes from the long arm of chrom...

Journal: :The International journal of developmental biology 2006
Maria Zoupa Maisa Seppala Thimios Mitsiadis Martyn T Cobourne

TBX1 encodes a T-box-containing transcription factor, which is thought to be a key player in the aetiology of the DiGeorge and Velocardiofacial syndromes (DGS/VCFS). In addition to defects affecting structures derived from the pharyngeal pouches, these patients exhibit varying degrees of facial dysmorphology and cleft palate. We have analysed the expression of murine Tbx1 during early facial de...

Journal: :American journal of medical genetics. Part A 2008
Elaine Tam Edwin J Young Colleen A Morris Christian R Marshall Wayne Loo Stephen W Scherer Carolyn B Mervis Lucy R Osborne

Williams-Beuren syndrome (WBS) is caused by a approximately 1.5 million base pair deletion at 7q11.23. A common inversion of the region, WBSinv-1, exists as a polymorphism but was also found in individuals with WBS-like features but no deletion, suggesting it could cause clinical symptoms. We performed a full clinical, developmental and genetic assessment of two previously reported individuals ...

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