نتایج جستجو برای: cdkl5

تعداد نتایج: 205  

Journal: :Human molecular genetics 2005
Clark Lin Brunella Franco Marsha Rich Rosner

X-linked cyclin-dependent kinase-like 5 (CDKL5 or STK9) has recently been implicated in atypical Rett and X-linked West syndromes, severe neurological disorders associated with mental retardation, loss of communication and motor skills and infantile spasms and seizures in predominantly females. Besides CDKL5, these disease phenotypes are also linked to mutations in the MECP2 and ARX genes. Here...

Journal: :The Journal of neuroscience : the official journal of the Society for Neuroscience 2010
Qian Chen Yong-Chuan Zhu Jing Yu Sheng Miao Jing Zheng Li Xu Yang Zhou Dan Li Chi Zhang Jiong Tao Zhi-Qi Xiong

Mutations in cyclin-dependent kinase-like 5 (CDKL5), also known as serine/threonine kinase 9 (STK9), have been identified in patients with Rett syndrome (RTT) and X-linked infantile spasm. However, the function of CDKL5 in the brain remains unknown. Here, we report that CDKL5 is a critical regulator of neuronal morphogenesis. We identified a neuron-specific splicing variant of CDKL5 whose expre...

2016
Mohammad Sarfaraz Nawaz Elisa Giarda Francesco Bedogni Paolo La Montanara Sara Ricciardi Dalila Ciceri Tiziana Alberio Nicoletta Landsberger Laura Rusconi Charlotte Kilstrup-Nielsen Robert Blum

In the last years, the X-linked cyclin-dependent kinase-like 5 (CDKL5) gene has been associated with epileptic encephalopathies characterized by the early onset of intractable epilepsy, severe developmental delay, autistic features, and often the development of Rett syndrome-like features. Still, the role of CDKL5 in neuronal functions is not fully understood. By way of a yeast two hybrid scree...

Journal: :Human molecular genetics 2005
Francesca Mari Sara Azimonti Ilaria Bertani Fabrizio Bolognese Elena Colombo Rossella Caselli Elisa Scala Ilaria Longo Salvatore Grosso Chiara Pescucci Francesca Ariani Giuseppe Hayek Paolo Balestri Anna Bergo Gianfranco Badaracco Michele Zappella Vania Broccoli Alessandra Renieri Charlotte Kilstrup-Nielsen Nicoletta Landsberger

Rett syndrome (RTT) is a severe neurodevelopmental disorder almost exclusively affecting females and characterized by a wide spectrum of clinical manifestations. Most patients affected by classic RTT and a smaller percentage of patients with the milder form 'preserved speech variant' have either point mutations or deletions/duplications in the MECP2 gene. Recently, mutations in the CDKL5 gene, ...

2014
Elena Amendola Yang Zhan Camilla Mattucci Enrico Castroflorio Eleonora Calcagno Claudia Fuchs Giuseppina Lonetti Davide Silingardi Alexei L. Vyssotski Dominika Farley Elisabetta Ciani Tommaso Pizzorusso Maurizio Giustetto Cornelius T. Gross

Mutations in cyclin-dependent kinase-like 5 (CDKL5) cause early-onset epileptic encephalopathy, a neurodevelopmental disorder with similarities to Rett Syndrome. Here we describe the physiological, molecular, and behavioral phenotyping of a Cdkl5 conditional knockout mouse model of CDKL5 disorder. Behavioral analysis of constitutive Cdkl5 knockout mice revealed key features of the human disorde...

Journal: :Gene 2017
Ralph D Hector Owen Dando Tuula E Ritakari Peter C Kind Mark E S Bailey Stuart R Cobb

CDKL5 deficiency is a severe neurological disorder caused by mutations in the X-linked Cyclin-Dependent Kinase-Like 5 gene (CDKL5). The predominant human CDKL5 brain isoform is a 9.7kb transcript comprised of 18 exons with a large 6.6kb 3'-untranslated region (UTR). Mammalian models of CDKL5 disorder are currently limited to mouse, and little is known about Cdkl5 in other organisms used to mode...

Journal: :Neurobiology of disease 2015
Claudia Fuchs Roberto Rimondini Rocchina Viggiano Stefania Trazzi Marianna De Franceschi Renata Bartesaghi Elisabetta Ciani

Mutations in the X-linked cyclin-dependent kinase-like 5 (CDKL5) gene have been identified in a rare neurodevelopmental disorder characterized by early-onset seizures, severe developmental delay, intellectual disability and Rett syndrome-like features. CDKL5 is highly expressed in the brain during early postnatal stages, suggesting its importance for brain maturation. Using a newly-generated Cd...

Journal: :The Journal of biological chemistry 2006
Ilaria Bertani Laura Rusconi Fabrizio Bolognese Greta Forlani Barbara Conca Lucia De Monte Gianfranco Badaracco Nicoletta Landsberger Charlotte Kilstrup-Nielsen

Mutations in the X-linked cyclin-dependent kinase-like 5 (CDKL5) gene have been identified in patients with Rett syndrome, West syndrome, and X-linked infantile spasms sharing the common features of generally intractable early seizures and mental retardation. Disease-causing mutations are distributed in both the catalytic domain and in the large COOH terminus. In this report, we examine the fun...

2016
Ralph D Hector Owen Dando Nicoletta Landsberger Charlotte Kilstrup-Nielsen Peter C Kind Mark E S Bailey Stuart R Cobb

Mutations in the X-linked Cyclin-Dependent Kinase-Like 5 gene (CDKL5) cause early onset infantile spasms and subsequent severe developmental delay in affected children. Deleterious mutations have been reported to occur throughout the CDKL5 coding region. Several studies point to a complex CDKL5 gene structure in terms of exon usage and transcript expression. Improvements in molecular diagnosis ...

Journal: :Human molecular genetics 2009
Sara Ricciardi Charlotte Kilstrup-Nielsen Thierry Bienvenu Aurélia Jacquette Nicoletta Landsberger Vania Broccoli

Mutations in the human X-linked cyclin-dependent kinase-like 5 (CDKL5) gene have been shown to cause severe neurodevelopmental disorders including infantile spasms, encephalopathy, West-syndrome and an early-onset variant of Rett syndrome. CDKL5 is a serine/threonine kinase whose involvement in Rett syndrome can be inferred by its ability to directly bind and mediate phosphorylation of MeCP2. H...

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