Impaired bone resorption in cathepsin K-deficient mice is partially compensated for by enhanced osteoclastogenesis and increased expression of other proteases via an increased RANKL/OPG ratio

Bone ◽  
2005 ◽  
Vol 36 (1) ◽  
pp. 159-172 ◽  
Author(s):  
Riku Kiviranta ◽  
Jukka Morko ◽  
Sari L. Alatalo ◽  
Roisin NicAmhlaoibh ◽  
Juha Risteli ◽  
...  
Keyword(s):  
Bone Resorption ◽  
Cathepsin K ◽  
Deficient Mice

scholarly journals Impaired osteoclastic bone resorption leads to osteopetrosis in cathepsin-K-deficient mice

1998 ◽  
Vol 95 (23) ◽  
pp. 13453-13458 ◽  
Author(s):  
P. Saftig ◽  
E. Hunziker ◽  
O. Wehmeyer ◽  
S. Jones ◽  
A. Boyde ◽  
...  
Keyword(s):  
Bone Resorption ◽  
Cathepsin K ◽  
Osteoclastic Bone Resorption ◽  
Deficient Mice

Enhanced load adaptation in long bone of cathepsin K-deficient mice

2014 ◽  
Author(s):  
Nicolas Bonnet ◽  
Lee Duong ◽  
Serge Ferrari
Keyword(s):  
Cathepsin K ◽  
Long Bone ◽  
Deficient Mice

scholarly journals Pathogenic variants in GNPTAB and GNPTG encoding distinct subunits of GlcNAc-1-phosphotransferase differentially impact bone resorption in patients with mucolipidosis type II and III

2021 ◽  
Author(s):  
Giorgia Di Lorenzo ◽  
Lena M. Westermann ◽  
Timur A. Yorgan ◽  
Julian Stürznickel ◽  
Nataniel F. Ludwig ◽  
...  
Keyword(s):  
Bone Resorption ◽  
Bone Remodeling ◽  
Bone Cells ◽  
Collagen Crosslinks ◽  
Clinical Criteria ◽  
Pathogenic Variants ◽  
Bone Biopsies ◽  
Appropriate Therapy ◽  
Alpha Beta ◽  
Deficient Mice

Abstract Purpose Pathogenic variants in GNPTAB and GNPTG, encoding different subunits of GlcNAc-1-phosphotransferase, cause mucolipidosis (ML) II, MLIII alpha/beta, and MLIII gamma. This study aimed to investigate the cellular and molecular bases underlying skeletal abnormalities in patients with MLII and MLIII. Methods We analyzed bone biopsies from patients with MLIII alpha/beta or MLIII gamma by undecalcified histology and histomorphometry. The skeletal status of Gnptgkoand Gnptab-deficient mice was determined and complemented by biochemical analysis of primary Gnptgko bone cells. The clinical relevance of the mouse data was underscored by systematic urinary collagen crosslinks quantification in patients with MLII, MLIII alpha/beta, and MLIII gamma. Results The analysis of iliac crest biopsies revealed that bone remodeling is impaired in patients with GNPTAB-associated MLIII alpha/beta but not with GNPTG-associated MLIII gamma. Opposed to Gnptab-deficient mice, skeletal remodeling is not affected in Gnptgko mice. Most importantly, patients with variants in GNPTAB but not in GNPTG exhibited increased bone resorption. Conclusion The gene-specific impact on bone remodeling in human individuals and in mice proposes distinct molecular functions of the GlcNAc-1-phosphotransferase subunits in bone cells. We therefore appeal for the necessity to classify MLIII based on genetic in addition to clinical criteria to ensure appropriate therapy.

A highly potent inhibitor of cathepsin K (relacatib) reduces biomarkers of bone resorption both in vitro and in an acute model of elevated bone turnover in vivo in monkeys

Bone ◽  
2007 ◽  
Vol 40 (1) ◽  
pp. 122-131 ◽  
Author(s):  
S. Kumar ◽  
L. Dare ◽  
J.A. Vasko-Moser ◽  
I.E. James ◽  
S.M. Blake ◽  
...  
Keyword(s):  
Bone Resorption ◽  
Bone Turnover ◽  
Potent Inhibitor ◽  
Cathepsin K

Inhibition ofUlmus davidiana Planch (Ulmaceae) on bone resorption mediated by processing of Cathepsin K in cultured mouse osteoclasts

2008 ◽  
Vol 22 (4) ◽  
pp. 511-517 ◽  
Author(s):  
Kyung-Woon Kim ◽  
Jun-Sung Park ◽  
Kap-Sung Kim ◽  
Un-Ho Jin ◽  
June-Ki Kim ◽  
...  
Keyword(s):  
Bone Resorption ◽  
Cathepsin K

scholarly journals Cathepsin K Antisense Oligodeoxynucleotide Inhibits Osteoclastic Bone Resorption

1997 ◽  
Vol 272 (13) ◽  
pp. 8109-8112 ◽  
Author(s):  
Takashi Inui ◽  
Osamu Ishibashi ◽  
Tetsuya Inaoka ◽  
Yumi Origane ◽  
Masayoshi Kumegawa ◽  
...  
Keyword(s):  
Bone Resorption ◽  
Cathepsin K ◽  
Osteoclastic Bone Resorption ◽  
Antisense Oligodeoxynucleotide

Lipopolysaccharide-induced bone resorption is increased in TNF type 2 receptor-deficient mice in vivo

2008 ◽  
Vol 26 (5) ◽  
pp. 469-477 ◽  
Author(s):  
Anower Hussain Mian ◽  
Hiroaki Saito ◽  
Neil Alles ◽  
Hitoyata Shimokawa ◽  
Kazuhiro Aoki ◽  
...  
Keyword(s):  
Bone Resorption ◽  
Deficient Mice

scholarly journals Peptide Aldehyde Inhibitors of Cathepsin K Inhibit Bone Resorption Both In Vitro and In Vivo

1997 ◽  
Vol 12 (9) ◽  
pp. 1396-1406 ◽  
Author(s):  
Bartholomew J. Votta ◽  
Mark A. Levy ◽  
Alison Badger ◽  
Jeremy Bradbeer ◽  
Robert A. Dodds ◽  
...  
Keyword(s):  
Bone Resorption ◽  
Cathepsin K ◽  
Inhibit Bone Resorption
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