نتایج جستجو برای: autozygosity

تعداد نتایج: 143  

Journal: :American journal of human genetics 2010
Ludovica Volpi Gaia Roversi Elisa Adele Colombo Nico Leijsten Daniela Concolino Andrea Calabria Maria Antonietta Mencarelli Michele Fimiani Fabio Macciardi Rolph Pfundt Eric F P M Schoenmakers Lidia Larizza

Next-generation sequencing is a straightforward tool for the identification of disease genes in extended genomic regions. Autozygosity mapping was performed on a five-generation inbred Italian family with three siblings affected with Clericuzio-type poikiloderma with neutropenia (PN [MIM %604173]), a rare autosomal-recessive genodermatosis characterised by poikiloderma, pachyonychia, and chroni...

2015
Eui-Soo Kim Tad S. Sonstegard Marcos V. G. B. da Silva Louis C. Gasbarre Curtis P. Van Tassell

Genetic markers associated with parasite indicator traits are ideal targets for study of marker assisted selection aimed at controlling infections that reduce herd use of anthelminthics. For this study, we collected gastrointestinal (GI) nematode fecal egg count (FEC) data from post-weaning animals of an Angus resource population challenged to a 26 week natural exposure on pasture. In all, data...

2014
Sali M K Farhan Jian Wang John F Robinson Piya Lahiry Victoria M Siu Chitra Prasad Jonathan B Kronick David A Ramsay C Anthony Rupar Robert A Hegele

Iron-sulfur (Fe-S) clusters are a class of highly conserved and ubiquitous prosthetic groups with unique chemical properties that allow the proteins that contain them, Fe-S proteins, to assist in various key biochemical pathways. Mutations in Fe-S proteins often disrupt Fe-S cluster assembly leading to a spectrum of severe disorders such as Friedreich's ataxia or iron-sulfur cluster assembly en...

Journal: :Human molecular genetics 2015
Abolfazl Heidari Chanakan Tongsook Reza Najafipour Luciana Musante Nasim Vasli Masoud Garshasbi Hao Hu Kirti Mittal Amy J M McNaughton Kumudesh Sritharan Melissa Hudson Henning Stehr Saeid Talebi Mohammad Moradi Hossein Darvish Muhammad Arshad Rafiq Hossein Mozhdehipanah Ali Rashidinejad Shahram Samiei Mohsen Ghadami Christian Windpassinger Gabriele Gillessen-Kaesbach Andreas Tzschach Iltaf Ahmed Anna Mikhailov D James Stavropoulos Melissa T Carter Soraya Keshavarz Muhammad Ayub Hossein Najmabadi Xudong Liu Hans Hilger Ropers Peter Macheroux John B Vincent

Histamine (HA) acts as a neurotransmitter in the brain, which participates in the regulation of many biological processes including inflammation, gastric acid secretion and neuromodulation. The enzyme histamine N-methyltransferase (HNMT) inactivates HA by transferring a methyl group from S-adenosyl-l-methionine to HA, and is the only well-known pathway for termination of neurotransmission actio...

2013
Neil V Morgan Jane L Hartley Kenneth DR Setchell Michael A Simpson Rachel Brown Louise Tee Sian Kirkham Shanaz Pasha Richard C Trembath Eamonn R Maher Paul Gissen Deirdre A Kelly

Infantile cholestatic diseases can be caused by mutations in a number of genes involved in different hepatocyte molecular pathways. Whilst some of the essential pathways have a well understood function, such as bile biosynthesis and transport, the role of the others is not known. Here we report the findings of a clinical, biochemical and molecular study of a family with three patients affected ...

Journal: :Genetics 2000
N Bierne A Tsitrone P David

Associative overdominance, the fitness difference between heterozygotes and homozygotes at a neutral locus, is classically described using two categories of models: linkage disequilibrium in small populations or identity disequilibrium in infinite, partially selfing populations. In both cases, only equilibrium situations have been considered. In the present study, associative overdominance is r...

Journal: :Molecular Vision 2009
Esther Meyer Fatimah Rahman Jessica Owens Shanaz Pasha Neil V. Morgan Richard C. Trembath Edwin M. Stone Anthony T. Moore Eamonn R. Maher

PURPOSE To identify the molecular basis for autosomal recessively inherited congenital non-syndromic pulverulent cataracts in a consanguineous family with four affected children. METHODS An autozygosity mapping strategy using high density SNP microarrays and microsatellite markers was employed to detect regions of homozygosity. Subsequently good candidate genes were screened for mutations by ...

2016
Leman Damla Kotan Charlton Cooper Şükran Darcan Ian M. Carr Samim Özen Yi Yan Mohammad K. Hamedani Fatih Gürbüz Eda Mengen İhsan Turan Ayça Ulubay Gamze Akkuş Bilgin Yüksel A. Kemal Topaloğlu Etienne Leygue

OBJECTIVE What initiates the pubertal process in humans and other mammals is still unknown. We hypothesized that gene(s) taking roles in triggering human puberty may be identified by studying a cohort of idiopathic hypogonadotropic hypogonadism (IHH). METHODS A cohort of IHH cases was studied based on autozygosity mapping coupled with whole exome sequencing. RESULTS Our studies revealed thr...

2010

Development of the human brain occurs in a number of complex preand postnatal stages which are governed by both genetic and environmental factors. Aberrant brain development due to inherited defects may result in a wide spectrum of neurological disorders which are commonly encountered in the clinical field of paediatric neurology. In the work for this thesis, I have investigated the molecular b...

2015
Biljana Ilkovski Alistair T. Pagnamenta Gina L. O'Grady Taroh Kinoshita Malcolm F. Howard Monkol Lek Brett Thomas Anne Turner John Christodoulou David Sillence Samantha J.L. Knight Niko Popitsch David A. Keays Consuelo Anzilotti Anne Goriely Leigh B. Waddell Fabienne Brilot Kathryn N. North Noriyuki Kanzawa Daniel G. Macarthur Jenny C. Taylor Usha Kini Yoshiko Murakami Nigel F. Clarke

Glycosylphosphatidylinositol (GPI)-anchored proteins are ubiquitously expressed in the human body and are important for various functions at the cell surface. Mutations in many GPI biosynthesis genes have been described to date in patients with multi-system disease and together these constitute a subtype of congenital disorders of glycosylation. We used whole exome sequencing in two families to...

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