نتایج جستجو برای: sandhoff disease

تعداد نتایج: 1490121  

Journal: :Brain : a journal of neurology 2003
M Jeyakumar R Thomas E Elliot-Smith D A Smith A C van der Spoel A d'Azzo V Hugh Perry T D Butters R A Dwek F M Platt

Mouse models of the GM2 gangliosidoses [Tay-Sachs, late onset Tay-Sachs (LOTS), Sandhoff] and GM1 gangliosidosis have been studied to determine whether there is a common neuro-inflammatory component to these disorders. During the disease course, we have: (i) examined the expression of a number of inflammatory markers in the CNS, including MHC class II, CD68, CD11b (CR3), 7/4, F4/80, nitrotyrosi...

2011
Kazuhiko Matsuoka Tomomi Tamura Daisuke Tsuji Yukie Dohzono Keisuke Kitakaze Kazuki Ohno Seiji Saito Hitoshi Sakuraba Kohji Itoh

To develop a novel enzyme replacement therapy for neurodegenerative Tay-Sachs disease (TSD) and Sandhoff disease (SD), which are caused by deficiency of β-hexosaminidase (Hex) A, we designed a genetically engineered HEXB encoding the chimeric human β-subunit containing partial amino acid sequence of the α-subunit by structure-based homology modeling. We succeeded in producing the modified HexB ...

2011
Karen M. Ashe Dinesh Bangari Lingyun Li Mario A. Cabrera-Salazar Scott D. Bercury Jennifer B. Nietupski Christopher G. F. Cooper Johannes M. F. G. Aerts Edward R. Lee Diane P. Copeland Seng H. Cheng Ronald K. Scheule John Marshall

The neuropathic glycosphingolipidoses are a subgroup of lysosomal storage disorders for which there are no effective therapies. A potential approach is substrate reduction therapy using inhibitors of glucosylceramide synthase (GCS) to decrease the synthesis of glucosylceramide and related glycosphingolipids that accumulate in the lysosomes. Genz-529468, a blood-brain barrier-permeant iminosugar...

Journal: :The Journal of biological chemistry 2004
Michael B Tropak Stephen P Reid Marianne Guiral Stephen G Withers Don Mahuran

Tay-Sachs and Sandhoff diseases are lysosomal storage disorders that result from an inherited deficiency of beta-hexosaminidase A (alphabeta). Whereas the acute forms are associated with a total absence of hexosaminidase A and early death, the chronic adult forms exist with activity and protein levels of approximately 5%, and unaffected individuals have been found with only 10% of normal levels...

Journal: :Proceedings of the National Academy of Sciences of the United States of America 1999
M Jeyakumar T D Butters M Cortina-Borja V Hunnam R L Proia V H Perry R A Dwek F M Platt

Sandhoff disease is a neurodegenerative disorder resulting from the autosomal recessive inheritance of mutations in the HEXB gene, which encodes the beta-subunit of beta-hexosaminidase. GM2 ganglioside fails to be degraded and accumulates within lysosomes in cells of the periphery and the central nervous system (CNS). There are currently no therapies for the glycosphingolipid lysosomal storage ...

Journal: :Journal of Biological Chemistry 1989

2014
Mauro Scarpelli Giuliano Tomelleri Laura Bertolasi Alessandro Salviati

An adult with Sandhoff disease presented with pure lower motor neuron phenotype. Twenty years later, he showed signs of upper motor neuron involvement. 25 years from the onset, his muscle weakness slightly worsened but he was fully independent in activities of daily living. GM2-gangliosidosis can manifest as a motor neuron disease with a slowly progressive course. The correct knowledge of the n...

2010
Seiichi Kanzaki Akira Yamaguchi Kayoko Yamaguchi Yoshitsugu Kojima Kyoko Suzuki Noriko Koumitsu Yoji Nagashima Kiyotaka Nagahama Michiko Ehara Yoshio Hirayasu Akihide Ryo Ichiro Aoki Shoji Yamanaka

BACKGROUND Sandhoff disease is a lysosomal storage disorder characterized by the absence of β-hexosaminidase and storage of GM2 ganglioside and related glycolipids. We have previously found that the progressive neurologic disease induced in Hexb(-/-) mice, an animal model for Sandhoff disease, is associated with the production of pathogenic anti-glycolipid autoantibodies. METHODOLOGY/PRINCIPA...

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