نتایج جستجو برای: progeroid syndromes

تعداد نتایج: 81654  

2012
Maureen Bowles John Lally Andrew J. Fadden Stephane Mouilleron Timothy Hammonds Neil Q. McDonald

The structure-specific endonuclease activity of the human XPF-ERCC1 complex is essential for a number of DNA processing mechanisms that help to maintain genomic integrity. XPF-ERCC1 cleaves DNA structures such as stem-loops, bubbles or flaps in one strand of a duplex where there is at least one downstream single strand. Here, we define the minimal substrate requirements for cleavage of stem-loo...

2013
Lolita S. Nidadavolu Laura J. Niedernhofer Saleem A. Khan

XFE progeroid syndrome, a disease of accelerated aging caused by deficiency in the DNA repair endonuclease XPF-ERCC1, is modeled by Ercc1 knockout and hypomorphic mice. Tissues and primary cells from these mice senesce prematurely, offering a unique opportunity to identify factors that regulate senescence and aging. We compared microRNA (miRNA) expression in Ercc1-/- primary mouse embryonic fib...

Journal: :Pathobiology of Aging & Age-related Diseases 2011

Journal: :Journal of cell science 2016
Yuexia Wang Uta Lichter-Konecki Kwame Anyane-Yeboa Jessica E Shaw Jonathan T Lu Cecilia Östlund Ji-Yeon Shin Lorraine N Clark Gregg G Gundersen Peter L Nagy Howard J Worman

In 1994 in the Journal of Cell Science, Hennekes and Nigg reported that changing valine to arginine at the endoproteolytic cleavage site in chicken prelamin A abolishes its conversion to lamin A. The consequences of this mutation in an organism have remained unknown. We now report that the corresponding mutation in a human subject leads to accumulation of prelamin A and causes a progeroid disor...

Journal: :Proceedings of the National Academy of Sciences of the United States of America 2016
José Rivera-Torres Conrado J Calvo Anna Llach Gabriela Guzmán-Martínez Ricardo Caballero Cristina González-Gómez Luis J Jiménez-Borreguero Juan A Guadix Fernando G Osorio Carlos López-Otín Adela Herraiz-Martínez Nuria Cabello Alex Vallmitjana Raul Benítez Leslie B Gordon José Jalife José M Pérez-Pomares Juan Tamargo Eva Delpón Leif Hove-Madsen David Filgueiras-Rama Vicente Andrés

Hutchinson-Gilford progeria syndrome (HGPS) is a rare genetic disease caused by defective prelamin A processing, leading to nuclear lamina alterations, severe cardiovascular pathology, and premature death. Prelamin A alterations also occur in physiological aging. It remains unknown how defective prelamin A processing affects the cardiac rhythm. We show age-dependent cardiac repolarization abnor...

2013
Sven Starke Peter Meinke Daria Camozzi Elisabetta Mattioli Roland Pfaeffle Manuela Siekmeyer Wolfgang Hirsch Lars Christian Horn Uwe Paasch Diana Mitter Giovanna Lattanzi Manfred Wehnert Wieland Kiess

The clinical course of a female patient affected by a progeroid syndrome with Restrictive Dermopathy (RD)-like features was followed up. Besides missing hairiness, stagnating weight and growth, RD-like features including progressive skin swelling and solidification, acrocontractures, osteolysis and muscular hypotension were observed until the patient died at the age of 11 months. A homozygousLM...

Journal: :American journal of human genetics 2015
Björn Fischer-Zirnsak Nathalie Escande-Beillard Jaya Ganesh Yu Xuan Tan Mohammed Al Bughaili Angela E Lin Inderneel Sahai Paulina Bahena Sara L Reichert Abigail Loh Graham D Wright Jaron Liu Elisa Rahikkala Eniko K Pivnick Asim F Choudhri Ulrike Krüger Tomasz Zemojtel Conny van Ravenswaaij-Arts Roya Mostafavi Irene Stolte-Dijkstra Sofie Symoens Leila Pajunen Lihadh Al-Gazali David Meierhofer Peter N Robinson Stefan Mundlos Camilo E Villarroel Peter Byers Amira Masri Stephen P Robertson Ulrike Schwarze Bert Callewaert Bruno Reversade Uwe Kornak

Progeroid disorders overlapping with De Barsy syndrome (DBS) are collectively denoted as autosomal-recessive cutis laxa type 3 (ARCL3). They are caused by biallelic mutations in PYCR1 or ALDH18A1, encoding pyrroline-5-carboxylate reductase 1 and pyrroline-5-carboxylate synthase (P5CS), respectively, which both operate in the mitochondrial proline cycle. We report here on eight unrelated individ...

Journal: :acta medica iranica 0
m. mohammadi

the clinical and phenotypic features of two siblings (a 12 years old girl and her 7 year old brother) with cockayne syndrome are described. the main problems were mild to moderate mental retardation, dwarfism, clumsy gait, photosensitive skin lesions and progeroid (senile like) appearance. brain ct - scans revealed symmetrical, well defined areas of calcification mainly located at lenticular nu...

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