Osteoclast stimulation factor 1 (Ostf1) KNOCKOUT increases trabecular bone mass in mice

Vermeren, Matthieu and Lyraki, Rodanthi and Wani, Sachin and Airik, Rannar and Albagha, Omar and Mort, Richard and Hildebrandt, Friedhelm and Hurd, Toby (2017) Osteoclast stimulation factor 1 (Ostf1) KNOCKOUT increases trabecular bone mass in mice. Mammalian genome : official journal of the International Mammalian Genome Society, 28 (11-12). pp. 498-514. ISSN 0938-8990

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Abstract

Osteoclast stimulation factor 1 (OSTF1) is an SH3-domain containing protein that was initially identified as a factor involved in the indirect activation of osteoclasts. It has been linked to spinal muscular atrophy in humans through its interaction with SMN1, and is one of six genes deleted in a human developmental microdeletion syndrome. To investigate the function of OSTF1, we generated an Ostf1 knockout mouse model, with exons 3 and 4 of Ostf1 replaced by a LacZ orf. Extensive X-Gal staining was performed to examine the developmental and adult expression pattern, followed by phenotyping. We show widespread expression of the gene in the vasculature of most organs and in a number of cell types in adult and embryonic mouse tissues. Furthermore, whilst SHIRPA testing revealed no behavioural defects, we demonstrate increased trabecular mass in the long bones, confirming a role for OSTF1 in bone development.

Item Type:
Journal Article
Journal or Publication Title:
Mammalian genome : official journal of the International Mammalian Genome Society
Uncontrolled Keywords:
/dk/atira/pure/subjectarea/asjc/1300/1311
Subjects:
ID Code:
87966
Deposited By:
Deposited On:
06 Oct 2017 19:36
Refereed?:
Yes
Published?:
Published
Last Modified:
20 Oct 2020 05:23