نتایج جستجو برای: spondyloepiphyseal dysplasia

تعداد نتایج: 28617  

2015
Daniele Merico Maian Roifman Ulrich Braunschweig Ryan K C Yuen Roumiana Alexandrova Andrea Bates Brenda Reid Thomas Nalpathamkalam Zhuozhi Wang Bhooma Thiruvahindrapuram Paul Gray Alyson Kakakios Jane Peake Stephanie Hogarth David Manson Raymond Buncic Sergio L Pereira Jo-Anne Herbrick Benjamin J Blencowe Chaim M Roifman Stephen W Scherer

Roifman Syndrome is a rare congenital disorder characterized by growth retardation, cognitive delay, spondyloepiphyseal dysplasia and antibody deficiency. Here we utilize whole-genome sequencing of Roifman Syndrome patients to reveal compound heterozygous rare variants that disrupt highly conserved positions of the RNU4ATAC small nuclear RNA gene, a minor spliceosome component that is essential...

Journal: :iranian endodontic journal 0
hengameh ashraf department of endodontics, iranian center for endodontic research, dental school, shahid beheshti university of medical sciences, tehran, iran. mahsa eskandarinezhad department of endodontics, dental school, shahid beheshti university of medical sciences, tehran, iran.

this case report describes treatment of a necrotic immature permanent mandibular first molar with pulpal necrosis in 9-year old female with spondyloepiphyseal dysplasia. the coronal half of the root canal was debrided with a file #30 to remove necrotic tissue, and irrigated with chlorhexidine 0.12%. bleeding was evoked to form an intracanal blood clot; the wound was then dressed with calcium hy...

Journal: :Biochemical and biophysical research communications 2010
Katarzyna Gawron Deborah A Jensen Andrzej Steplewski Andrzej Fertala

Mutations in collagen II are associated with spondyloepiphyseal dysplasia, a group of heritable diseases whose common features include aberrations of skeletal growth. The mechanisms through which mutations in collagen II affect the cartilaginous tissues are complex and include both intracellular and extracellular processes. One of those mechanisms involves cellular stress caused by excessive ac...

Journal: :Cell 1995
Miikka Vikkula Edwin C.M Madman Vincent C.H Lui Natalia I Zhidkova George E Tiller Mary B Goldring Sylvia E.C van Beersum Maarten C de Waal Malefijt Frank H.J van den Hoogen Hans-Hilger Ropers Richard Mayne Kathryn S.E Cheah Bjorn R Olsen Matthew L Warman Han G Brunner

Identifying mutations that cause specific osteochondrodysplasias will provide novel insights into the function of genes that are essential for skeletal morphogenesis. We report here that an autosomal dominant form of Stickler syndrome, characterized by mild spondyloepiphyseal dysplasia, osteoarthritis, and sensorineural hearing loss, but no eye involvement, is caused by a splice donor site muta...

2013
Hye Won Min Kyo Yeon Koo Chul Ho Lee Jeong Yoon Yang Jin-Sung Lee

induce type II achondrogenesis-hydrochondrogenesis, spondyloepiphyseal dysplasia congenita, and Stickler syndrome, along with Kniest syndrome . 6-8) The clinical features of Kniest syndrome include a disproportionately short stature with a short, barrel-shaped chest, kyphoscoliosis, enlargement of joints with limited range of motion, a flat face with prominent eyes, low nasal bridge, myopia tha...

2003
S Brickwood D T Bonthron L I Al-Gazali K Piper T Hearn D I Wilson N A Hanley

W olcott-Rallison syndrome (OMIM 226980) is a rare autosomal recessive disorder characterised by permanent insulin requiring diabetes developing in the newborn period or early infancy, an early tendency to skeletal fractures, and spondyloepiphyseal dysplasia. The syndrome results from mutations in the gene encoding the eukaryotic translation initiation factor 2-a kinase 3 (EIF2AK3, also called ...

Journal: :American journal of physiology. Cell physiology 2003
Ottavia Barbieri Simonetta Astigiano Monica Morini Sara Tavella Anna Schito Alessandro Corsi Davide Di Martino Paolo Bianco Ranieri Cancedda Silvio Garofalo

We have generated transgenic mice harboring the deletion of exon 48 in the mouse alpha1(II) procollagen gene (Col2a1). This was the first dominant negative mutation identified in the human alpha1(II) procollagen gene (COL2A1). Patients carrying a single allele with this mutation suffer from a severe skeletal disorder called spondyloepiphyseal dysplasia congenita (SED). Transgenic mice phenotype...

Journal: :Journal of medical genetics 2003
S Brickwood D T Bonthron L I Al-Gazali K Piper T Hearn D I Wilson N A Hanley

W olcott-Rallison syndrome (OMIM 226980) is a rare autosomal recessive disorder characterised by permanent insulin requiring diabetes developing in the newborn period or early infancy, an early tendency to skeletal fractures, and spondyloepiphyseal dysplasia. The syndrome results from mutations in the gene encoding the eukaryotic translation initiation factor 2-a kinase 3 (EIF2AK3, also called ...

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