نتایج جستجو برای: smarcal1

تعداد نتایج: 96  

2016
Marie Morimoto Clara Myung Kimberly Beirnes Kunho Choi Yumi Asakura Arend Bokenkamp Dominique Bonneau Milena Brugnara Joel Charrow Estelle Colin Amira Davis Georges Deschenes Mattia Gentile Mario Giordano Andrew K. Gormley Rajeshree Govender Mark Joseph Kory Keller Evelyne Lerut Elena Levtchenko Laura Massella Christy Mayfield Behzad Najafian David Parham Jurgen Spranger Peter Stenzel Uluc Yis Zhongxin Yu Jonathan Zonana Glenda Hendson Cornelius F. Boerkoel

BACKGROUND Schimke immuno-osseous dysplasia (SIOD) is a multisystemic disorder caused by biallelic mutations in the SWI/SNF-related matrix-associated actin-dependent regulator of chromatin, subfamily A-like 1 (SMARCAL1) gene. Changes in gene expression underlie the arteriosclerosis and T-cell immunodeficiency of SIOD; therefore, we hypothesized that SMARCAL1 deficiency causes the focal segmenta...

2014
Luisa Santangelo Maddalena Gigante Giuseppe Stefano Netti Sterpeta Diella Flora Puteo Vincenza Carbone Giuseppe Grandaliano Mario Giordano Loreto Gesualdo

BACKGROUND Schimke immuno-osseous dysplasia (SIOD, OMIM #242900) is an autosomal-recessive pleiotropic disorder characterized by spondyloepiphyseal dysplasia, renal dysfunction and T-cell immunodeficiency. SIOD is caused by mutations in the gene SMARCAL1. CASE PRESENTATION We report the clinical and genetic diagnosis of a 5-years old girl with SIOD, referred to our Center because of nephrotic...

Journal: :Journal of medical genetics 2002
S Lou P Lamfers N McGuire C F Boerkoel

Schimke immuno-osseous dysplasia (SIOD) is characterised by autosomal recessive inheritance, spondyloepiphyseal dysplasia causing growth retardation, defective cellular immunity, progressive nephropathy leading to renal failure, hyperpigmented macules, and dysmorphic facial features. Half of SIOD patients also have hypothyroidism, half episodic cerebral ischaemia, and a tenth bone marrow failur...

Journal: :Genomics 2000
M A Coleman J A Eisen H W Mohrenweiser

The SNF2 gene family consists of a large group of proteins involved in transcriptional regulation, maintenance of chromosome integrity, and various aspects of DNA repair. We cloned a novel SNF2 family human cDNA, with sequence identity to the Escherichia coli RNA polymerase-binding protein HepA and named the human hepA-related protein (HHARP/SMARCAL1). In addition, the mouse ortholog (Mharp/Sma...

Journal: :Journal of neuropathology and experimental neurology 2008
Kimiko Deguchi Johanna M Clewing Leah I Elizondo Ryuki Hirano Cheng Huang Kunho Choi Emily A Sloan Thomas Lücke Katja M Marwedel Ralph D Powell Karen Santa Cruz Sandrine Willaime-Morawek Ken Inoue Shu Lou Jennifer L Northrop Yonehiro Kanemura Derek van der Kooy Hideyuki Okano Dawna L Armstrong Cornelius F Boerkoel

Schimke immuno-osseous dysplasia (OMIM 242900) is an uncommon autosomal-recessive multisystem disease caused by mutations in SMARCAL1 (swi/snf-related, matrix-associated, actin-dependent regulator of chromatin, subfamily a-like 1), a gene encoding a putative chromatin remodeling protein. Neurologic manifestations identified to date relate to enhanced atherosclerosis and cerebrovascular disease....

Journal: :iranian journal of medical sciences 0
mitra basiratnia shiraz nephrology-urology research center, shiraz university of medical sciences, shiraz, iran alireza baradaran-heravi child and family research institute, department of medical genetics, university of british columbia, vancouver, canada majid yavarian hematology research center, shiraz university of medical sciences, shiraz, iran bita geramizadeh department of pathology, shiraz university of medical sciences, shiraz, iran mehran karimi hematology research center, shiraz university of medical sciences, shiraz, iran

schimke immuno-osseous dysplasia is a rare autosomal recessive multisystem disorder characterized by steroid-resistant nephrotic syndrome, immunodeficiency, and spondyloepiphy-seal dysplasia. mutations in swi/snf2 related, matrix associated, actin dependent regulator of chromatin, subfamily a-like 1 (smarcal1) gene are responsible for the disease. the present report describes, for the first tim...

Journal: :The Journal of biological chemistry 2016
Akosua Badu-Nkansah Aaron C Mason Brandt F Eichman David Cortez

DNA damage and other forms of replication stress can cause replication forks to stall. Replication stress response proteins stabilize and resolve stalled forks by mechanisms that include fork remodeling to facilitate repair or bypass of damaged templates. Several enzymes including SMARCAL1, HLTF, and ZRANB3 catalyze these reactions. SMARCAL1 and HLTF utilize structurally distinct accessory doma...

Alireza Baradaran-Heravi Bita Geramizadeh, Majid Yavarian Mehran Karimi, Mitra Basiratnia,

Schimke immuno-osseous dysplasia is a rare autosomal recessive multisystem disorder characterized by steroid-resistant nephrotic syndrome, immunodeficiency, and spondyloepiphy-seal dysplasia. Mutations in SWI/SNF2 related, matrix associated, actin dependent regulator of chromatin, subfamily a-like 1 (SMARCAL1) gene are responsible for the disease. The present report describes, for the f...

2017
Arun Mouli Kolinjivadi Vincenzo Sannino Anna De Antoni Karina Zadorozhny Mairi Kilkenny Hervé Técher Giorgio Baldi Rong Shen Alberto Ciccia Luca Pellegrini Lumir Krejci Vincenzo Costanzo

Brca2 deficiency causes Mre11-dependent degradation of nascent DNA at stalled forks, leading to cell lethality. To understand the molecular mechanisms underlying this process, we isolated Xenopus laevis Brca2. We demonstrated that Brca2 protein prevents single-stranded DNA gap accumulation at replication fork junctions and behind them by promoting Rad51 binding to replicating DNA. Without Brca2...

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