نتایج جستجو برای: autozygosity mapping

تعداد نتایج: 198456  

Journal: :Cancer research 2009
Sarah L Spain Jean-Baptiste Cazier Richard Houlston Luis Carvajal-Carmona Ian Tomlinson

Genome-wide association studies have identified several common single nucleotide polymorphisms (SNP) associated with an increased risk of colorectal cancer (CRC), although they have failed to identify any recessively acting alleles that contribute to disease risk. However, two recent studies have suggested that inbreeding and runs of homozygosity (ROH) increase the risk of developing cancer, pe...

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
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...

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...

2014
Anas M Alazami Mohammed Zain Seidahmed Fatema Alzahrani Adam O Mohammed Fowzan S Alkuraya

Cranioectodermal dysplasia (CED) is a very rare autosomal recessive disorder characterized by a recognizable craniofacial profile in addition to ectodermal manifestations involving the skin, hair, and teeth. Four genes are known to be mutated in this disorder, all involved in the ciliary intraflagellar transport confirming that CED is a ciliopathy. In a multiplex consanguineous family with typi...

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