نتایج جستجو برای: wolfram syndrome

تعداد نتایج: 623455  

Journal: :QJM: An International Journal of Medicine 2019

Journal: :Turkish Journal of Pediatric Disease 2015

Journal: :Orphanet Journal of Rare Diseases 2020

2016
Dagmar Prochazkova Zuzana Hruba Petra Konecna Jarmila Skotakova Lenka Fajkusova

Wolfram-like syndrome (WFSL) is a rare autosomal dominant disease characterised by congenital progressive hearing loss, diabetes mellitus, and optic atrophy. The patient was a boy with the juvenile form of diabetes mellitus and findings which clinically matched the symptoms of Wolfram syndrome. At the age of 3 1/4 years, diabetes mellitus was diagnosed in this boy who also had severe psychomoto...

2016
Michal Cagalinec Mailis Liiv Zuzana Hodurova Miriam Ann Hickey Annika Vaarmann Merle Mandel Akbar Zeb Vinay Choubey Malle Kuum Dzhamilja Safiulina Eero Vasar Vladimir Veksler Allen Kaasik

Deficiency of the protein Wolfram syndrome 1 (WFS1) is associated with multiple neurological and psychiatric abnormalities similar to those observed in pathologies showing alterations in mitochondrial dynamics. The aim of this study was to examine the hypothesis that WFS1 deficiency affects neuronal function via mitochondrial abnormalities. We show that down-regulation of WFS1 in neurons leads ...

2012
Concetta Aloi Alessandro Salina Lorenzo Pasquali Francesca Lugani Katia Perri Chiara Russo Ramona Tallone Gian Marco Ghiggeri Renata Lorini Giuseppe d'Annunzio

BACKGROUND Wolfram Syndrome (WS) is an autosomal recessive neurodegenerative disorder characterized by Diabetes Insipidus, Diabetes Mellitus, Optic Atrophy, and Deafness identified by the acronym "DIDMOAD". The WS gene, WFS1, encodes a transmembrane protein called Wolframin, which recent evidence suggests may serve as a novel endoplasmic reticulum calcium channel in pancreatic β-cells and neuro...

2013
Bess A Marshall M Alan Permutt Alexander R Paciorkowski James Hoekel Roanne Karzon Jon Wasson Amy Viehover Neil H White Joshua S Shimony Linda Manwaring Paul Austin Timothy E Hullar Tamara Hershey

BACKGROUND Wolfram Syndrome (WFS:OMIM 222300) is an autosomal recessive, progressive, neurologic and endocrinologic degenerative disorder caused by mutations in the WFS1 gene, encoding the endoplasmic reticulum (ER) protein wolframin, thought to be involved in the regulation of ER stress. This paper reports a cross section of data from the Washington University WFS Research Clinic, a longitudin...

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