نتایج جستجو برای: congenital stationary night blindness

تعداد نتایج: 223938  

2017
Liheng Shi Janet Ya-An Chang Fei Yu Michael L. Ko Gladys Y.-P. Ko

L-type voltage-gated calcium channels (LTCCs) regulate tonic neurotransmitter release from sensory neurons including retinal photoreceptors. There are three types of LTCCs (Cav1.2, Cav1.3, and Cav1.4) expressed in the retina. While Cav1.2 is expressed in all retinal cells including the Müller glia and neurons, Cav1.3 and Cav1.4 are expressed in the retinal neurons with Cav1.4 exclusively expres...

2012
Muhammad Ajmal Muhammad Imran Khan Kornelia Neveling Yar Muhammad Khan Syeda Hafiza Benish Ali Waqas Ahmed Muhammad Safdar Iqbal Maleeha Azam Anneke I. den Hollander Rob W.J. Collin Raheel Qamar Frans P.M. Cremers

PURPOSE To identify the underlying genetic causes of fundus albipunctatus (FA), a rare form of congenital stationary night blindness that is characterized by the presence of white dots in the midperiphery of the retina and delayed dark adaptation, in Pakistan. METHODS Two families with FA were identified by fundus examination, and genome-wide single nucleotide polymorphism genotyping was perf...

2011
Mineo Kondo Rikako Sanuki Shinji Ueno Yuji Nishizawa Naozumi Hashimoto Hiroshi Ohguro Shuichi Yamamoto Shigeki Machida Hiroko Terasaki Grazyna Adamus Takahisa Furukawa

BACKGROUND Paraneoplastic retinopathy (PR), including cancer-associated retinopathy (CAR) and melanoma-associated retinopathy (MAR), is a progressive retinal disease caused by antibodies generated against neoplasms not associated with the eye. While several autoantibodies against retinal antigens have been identified, there has been no known autoantibody reacting specifically against bipolar ce...

Journal: :Investigative ophthalmology & visual science 1995
T P Dryja E L Berson

in response to a genetic defect, the human retina has two major categories of response: to degenerate or not to work well. Ophthalmologists and vision scientists have been successful during the last century in discovering variations in clinical course, symptoms, funduscopic appearance, and measurements of visual function that distinguish scores of hereditary retinal diseases. Most of this categ...

2013
Xiaoni Liu Vasily Kerov Françoise Haeseleer Anurima Majumder Nikolai Artemyev Sheila A Baker Amy Lee

Mutations in the gene encoding Cav 1.4, CACNA1F, are associated with visual disorders including X-linked incomplete congenital stationary night blindness type 2 (CSNB2). In mice lacking Cav 1.4 channels, there are defects in the development of "ribbon" synapses formed between photoreceptors (PRs) and second-order neurons. However, many CSNB2 mutations disrupt the function rather than expression...

Journal: :Human molecular genetics 2014
Sujuan Jia Akira Muto Wilda Orisme Hannah E Henson Chaithanyarani Parupalli Bensheng Ju Herwig Baier Michael R Taylor

Mutations in the human CACNA1F gene cause incomplete congenital stationary night blindness type 2 (CSNB2), a non-progressive, clinically heterogeneous retinal disorder. However, the molecular mechanisms underlying CSNB2 have not been fully explored. Here, we describe the positional cloning of a blind zebrafish mutant, wait until dark (wud), which encodes a zebrafish homolog of human CACNA1F. We...

2012
Nadia K. Waheed Ahmed H. Qavi Sarah N. Malik Maleeha Maria Moeen Riaz Frans P. M. Cremers Maleeha Azam Raheel Qamar

PURPOSE Genetic studies were performed to identify the causative mutation in a 15-year-old girl diagnosed with congenital stationary night blindness (CSNB) presenting Mizuo-Nakamura phenomenon, a typical Oguchi disease symptom. The patient also had dural sinus thrombosis (DST), thrombocytopenia, and systemic lupus erythematosus (SLE). METHODS Mutation analysis was done by sequencing two candi...

Journal: :Vision Research 1997
Steve H. Kim Ronald A. Bush Paul A. Sieving

The 30 Hz flicker electroretinogram (ERG) response was studied in seven patients with Schubert-Bornschein complete-type congenital stationary night blindness (SB-CSNB) by measuring conventional peak implicit times and by harmonic analysis. Responses were elicited with xenon photostrobe flashes. The fundamental flicker component showed a significant increased phase lag (P = 0.002), even though t...

2013
Rebecca R. Bellone Heather Holl Vijayasaradhi Setaluri Sulochana Devi Nityanand Maddodi Sheila Archer Lynne Sandmeyer Arne Ludwig Daniel Foerster Melanie Pruvost Monika Reissmann Ralf Bortfeldt David L. Adelson Sim Lin Lim Janelle Nelson Bianca Haase Martina Engensteiner Tosso Leeb George Forsyth Michael J. Mienaltowski Padmanabhan Mahadevan Michael Hofreiter Johanna L. A. Paijmans Gloria Gonzalez-Fortes Bruce Grahn Samantha A. Brooks

Leopard complex spotting is a group of white spotting patterns in horses caused by an incompletely dominant gene (LP) where homozygotes (LP/LP) are also affected with congenital stationary night blindness. Previous studies implicated Transient Receptor Potential Cation Channel, Subfamily M, Member 1 (TRPM1) as the best candidate gene for both CSNB and LP. RNA-Seq data pinpointed a 1378 bp inser...

Journal: :Journal of neurophysiology 2005
Naheed W Khan Mineo Kondo Kelaginamane T Hiriyanna Jeff A Jamison Ronald A Bush Paul A Sieving

Retinal on-pathway dysfunction is implicated in human complete-type congenital stationary night blindness (CSNB1), a Mendelian genetic condition that results from mutations in the NYX gene encoding the protein nyctalopin. We probed cone pathway dysfunction in four human genotyped CSNB1 affected males by electroretinogram (ERG) recordings elicited with photopic sinusoidal and rapid-on/off-ramp f...

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