نتایج جستجو برای: autozygosity

تعداد نتایج: 143  

2009
Mehrnaz Narooie-Nejad Fereshteh Chitsazian Betsabeh Khoramian Tusi Faride Mousavi Massoud Houshmand Mohammad R. Rohani Azam S. Hosseinipour Akram Rismanchian Elahe Elahi

PURPOSE To assess whether loci other than GLC3A, GLC3B, and GLC3C are linked to primary congenital glaucoma (PCG). METHODS The gene CYP1B1 at GLC3A was screened in 19 Iranian PCG probands who had been recruited mostly from among individuals of Turkish ethnicity and individuals from central and eastern Iran. The gene MYOC was screened in patients from this cohort who lacked CYP1B1 mutations an...

2015
Christopher M. Watson Laura A. Crinnion Juliana Gurgel‐Gianetti Sally M. Harrison Catherine Daly Agne Antanavicuite Carolina Lascelles Alexander F. Markham Sergio D. J. Pena David T. Bonthron Ian M. Carr

Autozygosity mapping is a powerful technique for the identification of rare, autosomal recessive, disease-causing genes. The ease with which this category of disease gene can be identified has greatly increased through the availability of genome-wide SNP genotyping microarrays and subsequently of exome sequencing. Although these methods have simplified the generation of experimental data, its a...

Journal: :Proceedings of the National Academy of Sciences of the United States of America 2004
Erik G Puffenberger Diane Hu-Lince Jennifer M Parod David W Craig Seth E Dobrin Andrew R Conway Elizabeth A Donarum Kevin A Strauss Travis Dunckley Javier F Cardenas Kara R Melmed Courtney A Wright Winnie Liang Phillip Stafford C Robert Flynn D Holmes Morton Dietrich A Stephan

We have identified a lethal phenotype characterized by sudden infant death (from cardiac and respiratory arrest) with dysgenesis of the testes in males [Online Mendelian Inheritance in Man (OMIM) accession no. 608800]. Twenty-one affected individuals with this autosomal recessive syndrome were ascertained in nine separate sibships among the Old Order Amish. High-density single-nucleotide polymo...

Journal: :Human molecular genetics 2013
Salma Awad Mohammed S Al-Dosari Nadya Al-Yacoub Dilek Colak Mustafa A Salih Fowzan S Alkuraya Coralie Poizat

Primary microcephaly (PM) is a developmental disorder of early neuroprogenitors that results in reduction of the brain mass, particularly the cortex. To gain fresh insight into the pathogenesis of PM, we describe a consanguineous family with a novel genetic variant responsible for the disease. We performed autozygosity mapping followed by exome sequencing to detect the causal genetic variant. S...

Journal: :American journal of human genetics 2013
Xiaowu Gai Daniele Ghezzi Mark A Johnson Caroline A Biagosch Hanan E Shamseldin Tobias B Haack Aurelio Reyes Mai Tsukikawa Claire A Sheldon Satish Srinivasan Matteo Gorza Laura S Kremer Thomas Wieland Tim M Strom Erzsebet Polyak Emily Place Mark Consugar Julian Ostrovsky Sara Vidoni Alan J Robinson Lee-Jun Wong Neal Sondheimer Mustafa A Salih Emtethal Al-Jishi Christopher P Raab Charles Bean Francesca Furlan Rossella Parini Costanza Lamperti Johannes A Mayr Vassiliki Konstantopoulou Martina Huemer Eric A Pierce Thomas Meitinger Peter Freisinger Wolfgang Sperl Holger Prokisch Fowzan S Alkuraya Marni J Falk Massimo Zeviani

Whole-exome sequencing and autozygosity mapping studies, independently performed in subjects with defective combined mitochondrial OXPHOS-enzyme deficiencies, identified a total of nine disease-segregating FBXL4 mutations in seven unrelated mitochondrial disease families, composed of six singletons and three siblings. All subjects manifested early-onset lactic acidemia, hypotonia, and developme...

2015
Amit Sud Rosie Cooke Anthony J. Swerdlow Richard S. Houlston

Recent studies have reported that regions of homozygosity (ROH) in the genome are detectable in outbred populations and can be associated with an increased risk of malignancy. To examine whether homozygosity is associated with an increased risk of developing Hodgkin lymphoma (HL) we analysed 589 HL cases and 5,199 controls genotyped for 484,072 tag single nucleotide polymorphisms (SNPs). Across...

2009
Walid El-Sayed David A. Parry Roger C. Shore Mushtaq Ahmed Hussain Jafri Yasmin Rashid Suhaila Al-Bahlani Sharifa Al Harasi Jennifer Kirkham Chris F. Inglehearn Alan J. Mighell

Healthy dental enamel is the hardest and most highly mineralized human tissue. Though acellular, nonvital, and without capacity for turnover or repair, it can nevertheless last a lifetime. Amelogenesis imperfecta (AI) is a collective term for failure of normal enamel development, covering diverse clinical phenotypes that typically show Mendelian inheritance patterns. One subset, known as hypoma...

2015
Konstantinos Nikopoulos Almudena Avila-Fernandez Marta Corton Maria Isabel Lopez-Molina Raquel Perez-Carro Lara Bontadelli Silvio Alessandro Di Gioia Olga Zurita Blanca Garcia-Sandoval Carlo Rivolta Carmen Ayuso

Inherited retinal dystrophies present extensive phenotypic and genetic heterogeneity, posing a challenge for patients' molecular and clinical diagnoses. In this study, we wanted to clinically characterize and investigate the molecular etiology of an atypical form of autosomal recessive retinal dystrophy in two consanguineous Spanish families. Affected members of the respective families exhibite...

Journal: :Brain : a journal of neurology 2007
Erik G Puffenberger Kevin A Strauss Keri E Ramsey David W Craig Dietrich A Stephan Donna L Robinson Christine L Hendrickson Steven Gottlieb David A Ramsay Victoria M Siu Gregory G Heuer Peter B Crino D Holmes Morton

We used single nucleotide polymorphism (SNP) microarrays to investigate the cause of a symptomatic epilepsy syndrome in a group of seven distantly related Old Order Mennonite children. Autozygosity mapping was inconclusive, but closer inspection of the data followed by formal SNP copy number analyses showed that all affected patients had homozygous deletions of a single SNP (rs721575) and their...

Journal: :Molecular genetics & genomic medicine 2016
Philip M Boone Bo Yuan Shen Gu Zhiwei Ma Tomasz Gambin Claudia Gonzaga-Jauregui Mahim Jain Todd J Murdock Janson J White Shalini N Jhangiani Kimberly Walker Qiaoyan Wang Donna M Muzny Richard A Gibbs J Fielding Hejtmancik James R Lupski Jennifer E Posey Richard A Lewis

BACKGROUND Juvenile-onset cataracts are known among the Hutterites of North America. Despite being identified over 30 years ago, this autosomal recessive condition has not been mapped, and the disease gene is unknown. METHODS We performed whole exome sequencing of three Hutterite-type cataract trios and follow-up genotyping and mapping in four extended kindreds. RESULTS Trio exomes enabled ...

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