A mutation in Tpst2 encoding tyrosylprotein sulfotransferase causes dwarfism associated with hypothyroidism.
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概要
- 論文の詳細を見る
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.
- The Endocrine Societyの論文
著者
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Asano Atsushi
Laboratory Of Experimental Animal Science Graduate School Of Veterinary Medicine Hokkaido University
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Asano Atsushi
Mobile Computing And Communications Development Center Digital Media Equipment And Services Company
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Asano Atsushi
北海道大学 獣医学研究科動物疾病制御学講座実験動物学
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Tachi Atsushi
Laboratory Of Public Health Graduate School Of Veterinary Medicine Hokkaido University
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Agui T
Laboratory Of Laboratory Animal Science And Medicine Department Of Disease Control Graduate School O
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Hosoda Yayoi
Laboratory Of Laboratory Animal Science And Medicine Department Of Disease Control Graduate School O
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