نتایج جستجو برای: autozygosity mapping

تعداد نتایج: 198456  

2014
James A. Poulter Gina Murillo Steven J. Brookes Claire E. L. Smith David A. Parry Sandra Silva Jennifer Kirkham Chris F. Inglehearn Alan J. Mighell

Amelogenesis imperfecta (AI) describes a heterogeneous group of inherited dental enamel defects reflecting failure of normal amelogenesis. Ameloblastin (AMBN) is the second most abundant enamel matrix protein expressed during amelogenesis. The pivotal role of AMBN in amelogenesis has been confirmed experimentally using mouse models. However, no AMBN mutations have been associated with human AI....

Journal: :American journal of human genetics 1999
K W Broman J L Weber

Using genotypes from nearly 8,000 short tandem-repeat polymorphisms typed in eight of the reference families from the Centre d'Etude du Polymorphisme Humain (CEPH), we identified numerous long chromosomal segments of marker homozygosity in many CEPH individuals. These segments are likely to represent autozygosity, the result of the mating of related individuals. Confidence that the complete seg...

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...

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: :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...

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