نتایج جستجو برای: congenital stationary night blindness

تعداد نتایج: 223938  

Journal: :Acta biochimica Polonica 2000
B Dworakowska K Dołowy

There are many diseases related to ion channels. Mutations in muscle voltage-gated sodium, potassium, calcium and chloride channels, and acetylcholine-gated channel may lead to such physiological disorders as hyper- and hypokalemic periodic paralysis, myotonias, long QT syndrome, Brugada syndrome, malignant hyperthermia and myasthenia. Neuronal disorders, e.g., epilepsy, episodic ataxia, famili...

2012
Neal S. Peachey Jillian N. Pearring Pasano Bojang Matthew E. Hirschtritt Thomas A. Ray Takahisa Furukawa Chieko Koike Andrew F.X. Goldberg Yin Shen Maureen A. McCall Scott Nawy Patsy M. Nishina Ronald G. Gregg

Mutations in TRPM1 are found in humans with an autosomal recessive form of 25 complete congenital stationary night blindness (cCSNB). The Trpm1 mouse has been an 26 important animal model for this condition. Here we report a new mouse mutant, tvrm27, 27 identified in a chemical mutagenesis screen. Genetic mapping of the no b-wave 28 electroretinogram (ERG) phenotype of tvrm27 localized the muta...

2017
Kentaro Kurata Katsuhiro Hosono Yoshihiro Hotta

BACKGROUND This report describes a 45-year-old man with complete congenital stationary night blindness (CSNB1) who has been followed up for 38 years. CASE The patient first visited our hospital as a 7-year-old boy with a complaint of low visual acuity. Best corrected visual acuity (BCVA) was 0.5 in the right eye and 0.6 in the left eye. The refractive error was approximately -5.0 D in both ey...

2014
Sivasankar Malaichamy Parveen Sen Ramya Sachidanandam Tharigopala Arokiasamy Marie Elise Lancelot Isabelle Audo Christina Zeitz Nagasamy Soumittra

PURPOSE Congenital stationary night blindness (CSNB) is a non-progressive retinal disorder that shows genetic and clinical heterogeneity. CSNB is inherited as an autosomal recessive, autosomal dominant, or X-linked recessive trait and shows a good genotype-phenotype correlation. Clinically, CSNB is classified as the Riggs type and the Schubert-Bornschein type. The latter form is further sub-cla...

Journal: :American journal of human genetics 2012
Isabelle Audo Kinga Bujakowska Elise Orhan Charlotte M Poloschek Sabine Defoort-Dhellemmes Isabelle Drumare Susanne Kohl Tien D Luu Odile Lecompte Eberhart Zrenner Marie-Elise Lancelot Aline Antonio Aurore Germain Christelle Michiels Claire Audier Mélanie Letexier Jean-Paul Saraiva Bart P Leroy Francis L Munier Saddek Mohand-Saïd Birgit Lorenz Christoph Friedburg Markus Preising Ulrich Kellner Agnes B Renner Veselina Moskova-Doumanova Wolfgang Berger Bernd Wissinger Christian P Hamel Daniel F Schorderet Elfride De Baere Dror Sharon Eyal Banin Samuel G Jacobson Dominique Bonneau Xavier Zanlonghi Guylene Le Meur Ingele Casteels Robert Koenekoop Vernon W Long Francoise Meire Katrina Prescott Thomy de Ravel Ian Simmons Hoan Nguyen Hélène Dollfus Olivier Poch Thierry Léveillard Kim Nguyen-Ba-Charvet José-Alain Sahel Shomi S Bhattacharya Christina Zeitz

Congenital stationary night blindness (CSNB) is a heterogeneous retinal disorder characterized by visual impairment under low light conditions. This disorder is due to a signal transmission defect from rod photoreceptors to adjacent bipolar cells in the retina. Two forms can be distinguished clinically, complete CSNB (cCSNB) or incomplete CSNB; the two forms are distinguished on the basis of th...

2010
Makoto Nakamura Rikako Sanuki Tetsuhiro R. Yasuma Akishi Onishi Koji M. Nishiguchi Chieko Koike Mikiko Kadowaki Mineo Kondo Yozo Miyake Takahisa Furukawa

PURPOSE To identify human transient receptor potential cation channel, subfamily M, member 1 (TRPM1) gene mutations in patients with congenital stationary night blindness (CSNB). METHODS We analyzed four different Japanese patients with complete CSNB in whom previous molecular examination revealed no mutation in either nyctalopin (NYX) or glutamate receptor, metabotropic 6 (GRM6). The ophthal...

Journal: :Investigative ophthalmology & visual science 1996
F Tremblay I De Becker C Cheung G R LaRoche

PURPOSE To investigate a proposed postretinal defect in patients with the incomplete form of congenital stationary night blindness (CSNB2) and to compare visual evoked potential (VEP) results with those found in various forms of albinism. METHODS Visual evoked potentials were performed in 10 patients with a diagnosis of CSNB2, 10 subjects with albinism, and 17 normal subjects. Visual evoked p...

2014
Marion Neuillé Said El Shamieh Elise Orhan Christelle Michiels Aline Antonio Marie-Elise Lancelot Christel Condroyer Kinga Bujakowska Olivier Poch José-Alain Sahel Isabelle Audo Christina Zeitz

Mutations in LRIT3, coding for a Leucine-Rich Repeat, immunoglobulin-like and transmembrane domains 3 protein lead to autosomal recessive complete congenital stationary night blindness (cCSNB). The role of the corresponding protein in the ON-bipolar cell signaling cascade remains to be elucidated. Here we genetically and functionally characterize a commercially available Lrit3 knock-out mouse, ...

Journal: :Vision Research 1995
François Tremblay Robert G. Laroche Inge De Becker

Fifteen patients with the incomplete form of congenital stationary night blindness (iCSNB) were reviewed to better characterize their electroretinographic (ERG) findings in view of differential diagnosis with other retinal conditions also presenting with negative bright-flash ERG responses. In all 15 patients, in dark-adapted conditions, the bright-flash ERG response had a normal a-wave followe...

Journal: :American journal of human genetics 2009
Maria M van Genderen Mieke M C Bijveld Yvonne B Claassen Ralph J Florijn Jillian N Pearring Francoise M Meire Maureen A McCall Frans C C Riemslag Ronald G Gregg Arthur A B Bergen Maarten Kamermans

Congenital stationary night blindness (CSNB) is a clinically and genetically heterogeneous group of retinal disorders characterized by nonprogressive impaired night vision and variable decreased visual acuity. We report here that six out of eight female probands with autosomal-recessive complete CSNB (cCSNB) had mutations in TRPM1, a retinal transient receptor potential (TRP) cation channel gen...

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