Loss-of-function of SHARPIN causes an osteopenic phenotype in mice
SHARPIN is a novel protein thought to interact with SHANK family and is widely expressed in multiple tissues/cells, including osteoblasts and osteoclasts. Loss-of-function of Sharpin develops the chronic proliferative dermatitis mutation (CPDM) in mice as well as a severe inflammation in other organs. The actual function of SHARPIN is poorly understood. Our aim was to determine the functional roles of SHARPIN in bone metabolism by using CPDM mice. The skeletal phenotypes were determined by peripheral quantitative computed tomography, micro-computed tomography, and quantitative real-time RT-PCR, the cellular functions of osteoblasts and osteoclasts were investigated by ex vivo cell culture. Compared to wild-type controls, CPDM mice demonstrated significantly lower total and cortical bone mineral content and bone mineral density, trabecular and cortical bone volume, and trabecular number. The mRNA expression of Runx2, osterix, type I collagen, and osteocalcin was significantly lower in the bone from CPDM mice. Osteoclasts and osteoblasts from CPDM mice were functionally defective. Our result suggests that SHARPIN plays important regulating roles in bone metabolism. These functional roles may either come from systemic chronic inflammatory or directly signaling pathway within bone cells.
Medienart: |
E-Artikel |
---|
Erscheinungsjahr: |
2011 |
---|---|
Erschienen: |
2011 |
Enthalten in: |
Zur Gesamtaufnahme - volume:39 |
---|---|
Enthalten in: |
Endocrine - 39(2011), 2 vom: 14. Apr., Seite 104-12 |
Sprache: |
Englisch |
---|
Beteiligte Personen: |
Xia, Tian [VerfasserIn] |
---|
Links: |
---|
Anmerkungen: |
Date Completed 04.08.2011 Date Revised 20.10.2021 published: Print-Electronic Citation Status MEDLINE |
---|
doi: |
10.1007/s12020-010-9418-1 |
---|
funding: |
|
---|---|
Förderinstitution / Projekttitel: |
|
PPN (Katalog-ID): |
NLM203552962 |
---|
LEADER | 01000naa a22002652 4500 | ||
---|---|---|---|
001 | NLM203552962 | ||
003 | DE-627 | ||
005 | 20231223230333.0 | ||
007 | cr uuu---uuuuu | ||
008 | 231223s2011 xx |||||o 00| ||eng c | ||
024 | 7 | |a 10.1007/s12020-010-9418-1 |2 doi | |
028 | 5 | 2 | |a pubmed24n0679.xml |
035 | |a (DE-627)NLM203552962 | ||
035 | |a (NLM)21069580 | ||
040 | |a DE-627 |b ger |c DE-627 |e rakwb | ||
041 | |a eng | ||
100 | 1 | |a Xia, Tian |e verfasserin |4 aut | |
245 | 1 | 0 | |a Loss-of-function of SHARPIN causes an osteopenic phenotype in mice |
264 | 1 | |c 2011 | |
336 | |a Text |b txt |2 rdacontent | ||
337 | |a ƒaComputermedien |b c |2 rdamedia | ||
338 | |a ƒa Online-Ressource |b cr |2 rdacarrier | ||
500 | |a Date Completed 04.08.2011 | ||
500 | |a Date Revised 20.10.2021 | ||
500 | |a published: Print-Electronic | ||
500 | |a Citation Status MEDLINE | ||
520 | |a SHARPIN is a novel protein thought to interact with SHANK family and is widely expressed in multiple tissues/cells, including osteoblasts and osteoclasts. Loss-of-function of Sharpin develops the chronic proliferative dermatitis mutation (CPDM) in mice as well as a severe inflammation in other organs. The actual function of SHARPIN is poorly understood. Our aim was to determine the functional roles of SHARPIN in bone metabolism by using CPDM mice. The skeletal phenotypes were determined by peripheral quantitative computed tomography, micro-computed tomography, and quantitative real-time RT-PCR, the cellular functions of osteoblasts and osteoclasts were investigated by ex vivo cell culture. Compared to wild-type controls, CPDM mice demonstrated significantly lower total and cortical bone mineral content and bone mineral density, trabecular and cortical bone volume, and trabecular number. The mRNA expression of Runx2, osterix, type I collagen, and osteocalcin was significantly lower in the bone from CPDM mice. Osteoclasts and osteoblasts from CPDM mice were functionally defective. Our result suggests that SHARPIN plays important regulating roles in bone metabolism. These functional roles may either come from systemic chronic inflammatory or directly signaling pathway within bone cells | ||
650 | 4 | |a Journal Article | |
650 | 4 | |a Research Support, Non-U.S. Gov't | |
650 | 7 | |a Collagen Type I |2 NLM | |
650 | 7 | |a Core Binding Factor Alpha 1 Subunit |2 NLM | |
650 | 7 | |a Nerve Tissue Proteins |2 NLM | |
650 | 7 | |a RNA, Messenger |2 NLM | |
650 | 7 | |a Runx2 protein, mouse |2 NLM | |
650 | 7 | |a Sp7 Transcription Factor |2 NLM | |
650 | 7 | |a Sp7 protein, mouse |2 NLM | |
650 | 7 | |a Transcription Factors |2 NLM | |
650 | 7 | |a sharpin |2 NLM | |
650 | 7 | |a Osteocalcin |2 NLM | |
650 | 7 | |a 104982-03-8 |2 NLM | |
700 | 1 | |a Liang, Yanhua |e verfasserin |4 aut | |
700 | 1 | |a Ma, Junrong |e verfasserin |4 aut | |
700 | 1 | |a Li, Mi |e verfasserin |4 aut | |
700 | 1 | |a Gong, Meng |e verfasserin |4 aut | |
700 | 1 | |a Yu, Xijie |e verfasserin |4 aut | |
773 | 0 | 8 | |i Enthalten in |t Endocrine |d 1995 |g 39(2011), 2 vom: 14. Apr., Seite 104-12 |w (DE-627)NLM091795540 |x 1559-0100 |7 nnns |
773 | 1 | 8 | |g volume:39 |g year:2011 |g number:2 |g day:14 |g month:04 |g pages:104-12 |
856 | 4 | 0 | |u http://dx.doi.org/10.1007/s12020-010-9418-1 |3 Volltext |
912 | |a GBV_USEFLAG_A | ||
912 | |a GBV_NLM | ||
951 | |a AR | ||
952 | |d 39 |j 2011 |e 2 |b 14 |c 04 |h 104-12 |