Model organisms

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Model organisms have been used in the study of CSRP2BP function. A conditional knockout mouse line, called Csrp2bptm1a(KOMP)Wtsi[11][12] was generated as part of the International Knockout Mouse Consortium program — a high-throughput mutagenesis project to generate and distribute animal models of disease to interested scientists.[13][14][15]

Male and female animals underwent a standardized phenotypic screen to determine the effects of deletion.[9][16]

Twenty seven tests were carried out on mutant mice and eight significant abnormalities were observed.[9] Fewer than expected homozygous mutant embryos were identified during gestation. Fewer also survived until weaning. Male homozygous mutant's eyelids fail to open, they had abnormal eye size, a decreased susceptibility to bacterial infection and a decreased body length.[9] Female homozygous mutants had a decreased lean body mass. Animals of both sex also had corneal opacity and spinal abnormalities (including scoliosis and fusion of vertebral arches).[9]

References

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  1. ^ "Neurological assessment data for Csrp2bp". Wellcome Trust Sanger Institute.
  2. ^ "Dysmorphology data for Csrp2bp". Wellcome Trust Sanger Institute.
  3. ^ "DEXA data for Csrp2bp". Wellcome Trust Sanger Institute.
  4. ^ "Radiography data for Csrp2bp". Wellcome Trust Sanger Institute.
  5. ^ "Eye morphology data for Csrp2bp". Wellcome Trust Sanger Institute.
  6. ^ "Clinical chemistry data for Csrp2bp". Wellcome Trust Sanger Institute.
  7. ^ "Salmonella infection data for Csrp2bp". Wellcome Trust Sanger Institute.
  8. ^ "Citrobacter infection data for Csrp2bp". Wellcome Trust Sanger Institute.
  9. ^ a b c d e Gerdin AK (2010). "The Sanger Mouse Genetics Programme: high throughput characterisation of knockout mice". Acta Opthalmologica 88: 925-7.doi:10.1111/j.1755-3768.2010.4142.x: Wiley.{{cite web}}: CS1 maint: location (link)
  10. ^ Mouse Resources Portal, Wellcome Trust Sanger Institute.
  11. ^ "International Knockout Mouse Consortium".
  12. ^ "Mouse Genome Informatics".
  13. ^ Skarnes, W. C.; Rosen, B.; West, A. P.; Koutsourakis, M.; Bushell, W.; Iyer, V.; Mujica, A. O.; Thomas, M.; Harrow, J.; Cox, T.; Jackson, D.; Severin, J.; Biggs, P.; Fu, J.; Nefedov, M.; De Jong, P. J.; Stewart, A. F.; Bradley, A. (2011). "A conditional knockout resource for the genome-wide study of mouse gene function". Nature. 474 (7351): 337–342. doi:10.1038/nature10163. PMC 3572410. PMID 21677750.
  14. ^ Dolgin E (June 2011). "Mouse library set to be knockout". Nature 474: 262-263. doi:10.1038/474262a.{{cite web}}: CS1 maint: location (link) CS1 maint: year (link)
  15. ^ Collins FS, Rossant J, Wurst W (January 2007). A mouse for all reasons. Cell 128(1): 9-13. doi:10.1016/j.cell.2006.12.018 PMID 17218247.{{cite book}}: CS1 maint: location (link) CS1 maint: location missing publisher (link) CS1 maint: multiple names: authors list (link) CS1 maint: year (link)
  16. ^ van der Weyden L, White JK, Adams DJ, Logan DW (2011). "The mouse genetics toolkit: revealing function and mechanism". Genome Biol. 12 (6): 224. doi:10.1186/gb-2011-12-6-224. PMID 21722353.{{cite journal}}: CS1 maint: multiple names: authors list (link) CS1 maint: unflagged free DOI (link)