نتایج جستجو برای: progeroid syndromes

تعداد نتایج: 81654  

2011
Oliver Dreesen Colin L. Stewart

Hutchinson-Gilford Progeria (HGPS) and Werner syndromes are diseases that clinically resemble some aspects of accelerated aging. HGPS is caused by mutations in theLMNA gene resulting in post-translational processing defects that trigger Progeria in children. Werner syndrome, arising from mutations in the WRN helicase gene, causes premature aging in young adults. What are the molecular mechanism...

2010
York Kamenisch Maria Fousteri Jennifer Knoch Anna-Katharina von Thaler Birgit Fehrenbacher Hiroki Kato Thomas Becker Martijn E.T. Dollé Raoul Kuiper Marc Majora Martin Schaller Gijsbertus T.J. van der Horst Harry van Steeg Martin Röcken Doron Rapaport Jean Krutmann Leon H. Mullenders Mark Berneburg

Defects in the DNA repair mechanism nucleotide excision repair (NER) may lead to tumors in xeroderma pigmentosum (XP) or to premature aging with loss of subcutaneous fat in Cockayne syndrome (CS). Mutations of mitochondrial (mt)DNA play a role in aging, but a link between the NER-associated CS proteins and base excision repair (BER)-associated proteins in mitochondrial aging remains enigmatic. ...

Journal: :Hormone research in paediatrics 2014
Michael H Guo Yiping Shen Emily C Walvoord Timothy C Miller Jennifer E Moon Joel N Hirschhorn Andrew Dauber

BACKGROUND/AIMS Short stature is a common reason for presentation to pediatric endocrinology clinics. However, for most patients, no cause for the short stature can be identified. As genetics plays a strong role in height, we sought to identify known and novel genetic causes of short stature. METHODS We recruited 14 children with severe short stature of unknown etiology. We conducted whole ex...

2015
Ashley M Wood Kyle Laster Ellen L Rice Steven T Kosak

Ever since the first demonstration of their repetitive sequence and unique replication pathway, telomeres have beguiled researchers with how they function in protecting chromosome ends. Of course much has been learned over the years, and we now appreciate that telomeres are comprised of the multimeric protein/DNA shelterin complex and that the formation of t-loops provides protection from DNA d...

2015
David Araujo-Vilar Sofía Sánchez-Iglesias Cristina Guillín-Amarelle Ana Castro Mary Lage Marcos Pazos José Manuel Rial Javier Blasco Encarna Guillén-Navarro Rosario Domingo-Jiménez María Ruiz del Campo Blanca González-Méndez Felipe F. Casanueva

Lipodystrophies are a group of diseases mainly characterized by a loss of adipose tissue and frequently associated with insulin resistance, hypertriglyceridemia, and hepatic steatosis. In uncommon lipodystrophies, these complications frequently are difficult to control with conventional therapeutic approaches. This retrospective study addressed the effectiveness of recombinant methionyl leptin ...

2016
Rafal Czapiewski Michael I. Robson Eric C. Schirmer

It is well established that the nuclear envelope has many distinct direct connections to chromatin that contribute to genome organization. The functional consequences of genome organization on gene regulation are less clear. Even less understood is how interactions of lamins and nuclear envelope transmembrane proteins (NETs) with chromatin can produce anchoring tethers that can withstand the ph...

Journal: :Proceedings of the National Academy of Sciences 1973

Journal: :Genetics 2013
Jeffrey D Stumpf William C Copeland

The importance of mitochondrial DNA (mtDNA) deletions in the progeroid phenotype of exonuclease-deficient DNA polymerase γ mice has been intensely debated. We show that disruption of Mip1 exonuclease activity increases mtDNA deletions 160-fold, whereas disease-associated polymerase variants were mostly unaffected, suggesting that exonuclease activity is vital to avoid deletions during mtDNA rep...

2014
Ismene Karakasilioti George A. Garinis

asymmetry is a fundamental aspect of all living beings. This also applies to single celled organisms that reproduce symmetrically into two identical halves; the cell that inherits the old pole, it grows at a slower rate, produces less offspring, and shows a decrease in overall cell viability (Stewart EJ et al., PloS Biol 2005; 2:e45). Functional asymmetry becomes clearly visible when one consid...

2015
Isabelle Schrauwen Szabolcs Szelinger Ashley L. Siniard Ahmet Kurdoglu Jason J. Corneveaux Ivana Malenica Ryan Richholt Guy Van Camp Matt De Both Shanker Swaminathan Mari Turk Keri Ramsey David W. Craig Vinodh Narayanan Matthew J. Huentelman Markus Schuelke

A 3-year-old female patient presenting with an unknown syndrome of a neonatal progeroid appearance, lipodystrophy, pulmonary hypertension, cutis marmorata, feeding disorder and failure to thrive was investigated by whole-genome sequencing. This revealed a de novo, heterozygous, frame-shift mutation in the Caveolin1 gene (CAV1) (p.Phe160X). Mutations in CAV1, encoding the main component of the c...

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