نتایج جستجو برای: autozygosity

تعداد نتایج: 143  

2015
Ronja Hollstein David A Parry Lisa Nalbach Clare V Logan Tim M Strom Verity L Hartill Ian M Carr Georg C Korenke Sandeep Uppal Mushtaq Ahmed Thomas Wieland Alexander F Markham Christopher P Bennett Gabriele Gillessen-Kaesbach Eamonn G Sheridan Frank J Kaiser David T Bonthron

BACKGROUND The genetic aetiology of neurodevelopmental defects is extremely diverse, and the lack of distinctive phenotypic features means that genetic criteria are often required for accurate diagnostic classification. We aimed to identify the causative genetic lesions in two families in which eight affected individuals displayed variable learning disability, spasticity and abnormal gait. ME...

2013
Tommaso Pippucci Antonia Parmeggiani Flavia Palombo Alessandra Maresca Andrea Angius Laura Crisponi Francesco Cucca Rocco Liguori Maria Lucia Valentino Marco Seri Valerio Carelli

Contribution to epileptic encephalopathy (EE) of mutations in CACNA2D2, encoding α2δ-2 subunit of Voltage Dependent Calcium Channels, is unclear. To date only one CACNA2D2 mutation altering channel functionality has been identified in a single family. In the same family, a rare CELSR3 polymorphism also segregated with disease. Involvement of CACNA2D2 in EE is therefore not confirmed, while that...

2012
Asli Sirmaci Yvonne J. K. Edwards Hatice Akay Mustafa Tekin

Whole exome sequencing provides unprecedented opportunities to identify causative DNA variants in rare Mendelian disorders. Finding the responsible mutation via traditional methods in families with hearing loss is difficult due to a high degree of genetic heterogeneity. In this study we combined autozygosity mapping and whole exome sequencing in a family with 3 affected children having nonsyndr...

Journal: :The Journal of allergy and clinical immunology 2012
Abdullah Alangari Abdulrahman Alsultan Nouran Adly Michel J Massaad Iram Shakir Kiani Abdulrahman Aljebreen Emad Raddaoui Abdul-Kareem Almomen Saleh Al-Muhsen Raif S Geha Fowzan S Alkuraya

BACKGROUND Clinical immunology has traditionally relied on accurate phenotyping of the patient's immune dysfunction for the identification of a candidate gene or genes for sequencing and molecular confirmation. Although this is also true for other branches of medicine, the marked variability in immune-related phenotypes and the highly complex network of molecules that confer normal host immunit...

2016
Laura Iacolina Astrid V Stronen Cino Pertoldi Małgorzata Tokarska Louise S Nørgaard Joaquin Muñoz Anders Kjærsgaard Aritz Ruiz-Gonzalez Stanisław Kamiński Deirdre C Purfield

Runs of homozygosity (ROH), uninterrupted stretches of homozygous genotypes resulting from parents transmitting identical haplotypes to their offspring, have emerged as informative genome-wide estimates of autozygosity (inbreeding). We used genomic profiles based on 698 K single nucleotide polymorphisms (SNPs) from nine breeds of domestic cattle (Bos taurus) and the European bison (Bison bonasu...

2016
Hanne S. Sorte Liv T. Osnes Børre Fevang Pål Aukrust Hans C. Erichsen Paul H. Backe Tore G. Abrahamsen Ole B. Kittang Torstein Øverland Shalini N. Jhangiani Donna M. Muzny Magnus D. Vigeland Pubudu Samarakoon Tomasz Gambin Zeynep H. C. Akdemir Richard A. Gibbs Olaug K. Rødningen Robert Lyle James R. Lupski Asbjørg Stray‐Pedersen

BACKGROUND Four patients from three Norwegian families presented with a common skin phenotype of warts, molluscum contagiosum, and dermatitis since early childhood, and various other immunological features. Warts are a common manifestation of human papilloma virus (HPV), but when they are overwhelming, disseminated and/or persistent, and presenting together with other immunological features, a ...

Journal: :Human molecular genetics 2010
Michael Nothnagel Timothy Tehua Lu Manfred Kayser Michael Krawczak

The availability of high-density panels of genetic polymorphisms has led to the discovery of extended regions of apparent autozygosity in the human genome. At the genotype level, these regions present as sizeable stretches, or 'runs', of homozygosity (ROH). Here, we investigated both the genomic and the geographic distribution of ROHs in a large European sample of individuals originating from 2...

Journal: :American journal of human genetics 2012
Muzammil Ahmad Khan Muhammad Arshad Rafiq Abdul Noor Shobbir Hussain Joana V Flores Verena Rupp Akshita K Vincent Roland Malli Ghazanfar Ali Falak Sher Khan Gisele E Ishak Dan Doherty Rosanna Weksberg Muhammad Ayub Christian Windpassinger Shahnaz Ibrahim Michaela Frye Muhammad Ansar John B Vincent

Causes of autosomal-recessive intellectual disability (ID) have, until very recently, been under researched because of the high degree of genetic heterogeneity. However, now that genome-wide approaches can be applied to single multiplex consanguineous families, the identification of genes harboring disease-causing mutations by autozygosity mapping is expanding rapidly. Here, we have mapped a di...

Journal: :Human molecular genetics 2005
Kirsten Heathcote Claire Braybrook Lulu Abushaban Michelle Guy Maher E Khetyar Michael A Patton Nicholas D Carter Peter J Scambler Petros Syrris

Persistent truncus arteriosus (PTA) is a failure of septation of the cardiac outflow tract (OFT) into the pulmonary artery and the aorta. A common arterial trunk (CAT) is often diagnosed as PTA in the absence of evidence of embryological mechanism. We have used autozygosity mapping of a large consanguineous family segregating CAT to map the causative locus to chromosome 8p21. An F151L mutation ...

Journal: :Collegium antropologicum 2004
Igor Rudan Harry Campbell

As the genetic architecture of common complex diseases of late onset is emerging through intensive research, it is intriguing to assess the predicted effect of inbreeding on those diseases. In this paper, we propose five reasons why we believe inbreeding may have a considerable effect on post-reproductive human health. (i) The joint effect of inbreeding depression on all polygenic quantitative ...

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