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.
Highlights
Osteoclast stimulation factor 1 (OSTF1) was originally described as SH3P2 in a screen for Src-homology 3 (SH3)containing proteins by peptide array (Sparks et al 1996), and independently discovered in an expression cloning screen (Reddy et al 1998)
Exons 3 and 4 that encode most of the Osteoclast stimulation factor 1 (Ostf1) SH3 domain are replaced by a LacZ expression cassette
OSTF1 was previously described as a cytoplasmic protein with SH3 and ankyrin domains that indirectly activates osteoclast differentiation and regulates their bone-resorbing activity (Reddy et al 1998)
Summary
Osteoclast stimulation factor 1 (OSTF1) was originally described as SH3P2 in a screen for Src-homology 3 (SH3)containing proteins by peptide array (Sparks et al 1996), and independently discovered in an expression cloning screen (Reddy et al 1998). In a series of cell-based experiments, overexpression of OSTF1 in HeLa cells was found to have a negative impact on cell motility in transwell migration assays (Tanimura et al 2011). These transfected HeLa cells were found to be more rounded and had a smaller footprint that controls
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