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A mutation in Tpst2 encoding tyrosylprotein sulfotransferase causes dwarfism associated with hypothyroidism.

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Title: A mutation in Tpst2 encoding tyrosylprotein sulfotransferase causes dwarfism associated with hypothyroidism.
Authors: Sasaki, Nobuya Browse this author →KAKEN DB
Hosoda, Yayoi Browse this author
Nagata, Aogu Browse this author
Ding, Ming Browse this author
Cheng, Ji-Ming Browse this author
Miyamoto, Tomomi Browse this author
Okano, Shinya Browse this author
Asano, Atsushi Browse this author
Miyoshi, Ichiro Browse this author
Agui, Takashi Browse this author →KAKEN DB
Issue Date: Jul-2007
Publisher: The Endocrine Society
Journal Title: Molecular Endocrinology
Volume: 21
Issue: 7
Start Page: 1713
End Page: 1721
Publisher DOI: 10.1210/me.2007-0040
PMID: 17456791
Abstract: The growth-retarded (grt) mouse has an autosomal recessive, fetal-onset, severe thyroid hypoplasia related to TSH hyporesponsiveness. Through genetic mapping and complementation experiments, we show that grt is a missense mutation of a highly conserved region of the tyrosylprotein sulfotransferase 2 (Tpst2) gene, encoding one of the two Tpst genes implicated in posttranslational tyrosine O-sulfation. We present evidence that the grt mutation leads to a loss of TPST2 activity, and TPST2 isoform has a high degree of substrate preference for TSH receptor (TSHR). The expression of TPST2 can restore TSH-TSHR-mediated cAMP production in fibroblasts derived from grt mice. Therefore, we propose that the tyrosine sulfation of TSHR by TPST2 is crucial for TSH signaling and resultant thyroid gland function.
Type: article (author version)
URI: http://hdl.handle.net/2115/26433
Appears in Collections:獣医学院・獣医学研究院 (Graduate School of Veterinary Medicine / Faculty of Veterinary Medicine) > 雑誌発表論文等 (Peer-reviewed Journal Articles, etc)

Submitter: 安居院 高志

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