Developmental YAPdeltaC determines adult pathology in a model of spinocerebellar ataxia type 1

Kyota Fujita; Ying Mao; Shigenori Uchida; Xigui Chen; Hiroki Shiwaku; Takuya Tamura; Hikaru Ito; Kei Watase; Hidenori Homma; Kazuhiko Tagawa; Marius Sudol; Hitoshi Okazawa

doi:10.1038/s41467-017-01790-z

Nature Communications (Nov 2017)

Developmental YAPdeltaC determines adult pathology in a model of spinocerebellar ataxia type 1

Kyota Fujita,
Ying Mao,
Shigenori Uchida,
Xigui Chen,
Hiroki Shiwaku,
Takuya Tamura,
Hikaru Ito,
Kei Watase,
Hidenori Homma,
Kazuhiko Tagawa,
Marius Sudol,
Hitoshi Okazawa

Affiliations

Kyota Fujita: Department of Neuropathology, Medical Research Institute, Tokyo Medical and Dental University
Ying Mao: Department of Neuropathology, Medical Research Institute, Tokyo Medical and Dental University
Shigenori Uchida: Department of Neuropathology, Medical Research Institute, Tokyo Medical and Dental University
Xigui Chen: Department of Neuropathology, Medical Research Institute, Tokyo Medical and Dental University
Hiroki Shiwaku: Department of Neuropathology, Medical Research Institute, Tokyo Medical and Dental University
Takuya Tamura: Department of Neuropathology, Medical Research Institute, Tokyo Medical and Dental University
Hikaru Ito: Department of Neuropathology, Medical Research Institute, Tokyo Medical and Dental University
Kei Watase: Center for Brain Integration Research, Tokyo Medical and Dental University
Hidenori Homma: Department of Neuropathology, Medical Research Institute, Tokyo Medical and Dental University
Kazuhiko Tagawa: Department of Neuropathology, Medical Research Institute, Tokyo Medical and Dental University
Marius Sudol: Mechanobiology Institute, National University of Singapore
Hitoshi Okazawa: Department of Neuropathology, Medical Research Institute, Tokyo Medical and Dental University

DOI: https://doi.org/10.1038/s41467-017-01790-z
Journal volume & issue: Vol. 8, no. 1
pp. 1 – 15

Abstract

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Ataxin-1, linked to spinocerebellar ataxia type 1, is known to interact with the orphan nuclear receptor RORα. Here, Fujita and colleagues show that genetic supplementation of RORα-interacting protein YAPdeltaC during early development can rescue the adult pathologies of SCA1 mouse model.

Published in Nature Communications

ISSN: 2041-1723 (Online)
Publisher: Nature Portfolio
Country of publisher: United Kingdom
LCC subjects: Science
Website: https://www.nature.com/ncomms/

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