User:Rockpocket/MGP/CSRP2BP

Model organisms

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|+ Csrp2bp knockout mouse phenotype

CharacteristicPhenotype
Homozygote viabilitybgcolor="#C40000"|Abnormal
Recessive lethal studybgcolor="#C40000"|Abnormal
Homozygous Fertilitybgcolor="#488ED3"|Normal
Body weightbgcolor="#488ED3"|Normal
Anxietybgcolor="#488ED3"|Normal
Neurological assessmentbgcolor="#C40000"|Abnormal{{cite web |url=http://www.sanger.ac.uk/mouseportal/phenotyping/MBDX/neurological-assessment/ |title=Neurological assessment data for Csrp2bp |publisher=Wellcome Trust Sanger Institute}}
Grip strengthbgcolor="#488ED3"|Normal
Hot platebgcolor="#488ED3"|Normal
Dysmorphologybgcolor="#C40000"|Abnormal{{cite web |url=http://www.sanger.ac.uk/mouseportal/phenotyping/MBDX/dysmorphology/ |title=Dysmorphology data for Csrp2bp |publisher=Wellcome Trust Sanger Institute}}
Indirect calorimetrybgcolor="#488ED3"|Normal
Glucose tolerance testbgcolor="#488ED3"|Normal
Auditory brainstem responsebgcolor="#488ED3"|Normal
DEXAbgcolor="#C40000"|Abnormal{{cite web |url=http://www.sanger.ac.uk/mouseportal/phenotyping/MBDX/body-composition-dexa/ |title=DEXA data for Csrp2bp |publisher=Wellcome Trust Sanger Institute}}
Radiographybgcolor="#C40000"|Abnormal{{cite web |url=http://www.sanger.ac.uk/mouseportal/phenotyping/MBDX/x-ray-imaging/ |title=Radiography data for Csrp2bp |publisher=Wellcome Trust Sanger Institute}}
Body temperaturebgcolor="#488ED3"|Normal
Eye morphologybgcolor="#C40000"|Abnormal{{cite web |url=http://www.sanger.ac.uk/mouseportal/phenotyping/MBDX/eye-morphology/ |title=Eye morphology data for Csrp2bp |publisher=Wellcome Trust Sanger Institute}}
Clinical chemistrybgcolor="#488ED3"|Normal{{cite web |url=http://www.sanger.ac.uk/mouseportal/phenotyping/MBDX/plasma-chemistry/ |title=Clinical chemistry data for Csrp2bp |publisher=Wellcome Trust Sanger Institute}}
Plasma immunoglobulinsbgcolor="#488ED3"|Normal
Haematologybgcolor="#488ED3"|Normal
Peripheral blood lymphocytesbgcolor="#488ED3"|Normal
Micronucleus testbgcolor="#488ED3"|Normal
Heart weightbgcolor="#488ED3"|Normal
Tail epidermis wholemountbgcolor="#488ED3"|Normal
Skin Histopathologybgcolor="#488ED3"|Normal
Brain histopathologybgcolor="#488ED3"|Normal
MicroCT & Quantitative Faxitronbgcolor="#488ED3"|Normal
Salmonella infectionbgcolor="#C40000"|Abnormal{{cite web |url=http://www.sanger.ac.uk/mouseportal/phenotyping/MBDX/salmonella-challenge/ |title=Salmonella infection data for Csrp2bp |publisher=Wellcome Trust Sanger Institute}}
Citrobacter infectionbgcolor="#488ED3"|Normal{{cite web |url=http://www.sanger.ac.uk/mouseportal/phenotyping/MBDX/citrobacter-challenge/ |title=Citrobacter infection data for Csrp2bp |publisher=Wellcome Trust Sanger Institute}}
colspan=2; style="text-align: center;" | All tests and analysis from{{cite web |url=http://onlinelibrary.wiley.com/doi/10.1111/j.1755-3768.2010.4142.x/abstract |title=The Sanger Mouse Genetics Programme: high throughput characterisation of knockout mice |author=Gerdin AK |year=2010 |location=Acta Opthalmologica 88: 925-7.doi:10.1111/j.1755-3768.2010.4142.x |publisher=Wiley}}[http://www.sanger.ac.uk/mouseportal/ Mouse Resources Portal], Wellcome Trust Sanger Institute.

Model organisms have been used in the study of CSRP2BP function. A conditional knockout mouse line, called Csrp2bptm1a(KOMP)Wtsi{{cite web |url=http://www.knockoutmouse.org/martsearch/search?query=Csrp2bp |title=International Knockout Mouse Consortium}}{{cite web |url=http://www.informatics.jax.org/searchtool/Search.do?query=MGI:4363521 |title=Mouse Genome Informatics}} 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.{{Cite journal| last1 = Skarnes |first1 =W. C.| doi = 10.1038/nature10163 | last2 = Rosen | first2 = B.| last3 = West | first3 = A. P.| last4 = Koutsourakis | first4 = M.| last5 = Bushell | first5 = W.| last6 = Iyer | first6 = V.| last7 = Mujica | first7 = A. O.| last8 = Thomas | first8 = M.| last9 = Harrow | first9 = J.| last10 = Cox | first10 = T.| last11 = Jackson | first11 = D.| last12 = Severin | first12 = J.| last13 = Biggs | first13 = P.| last14 = Fu | first14 = J.| last15 = Nefedov | first15 = M.| last16 = De Jong | first16 = P. J.| last17 = Stewart | first17 = A. F.| last18 = Bradley | first18 = A. | title = A conditional knockout resource for the genome-wide study of mouse gene function | journal = Nature | volume = 474 | issue = 7351 | pages = 337–342 | year = 2011 | pmid = 21677750 | pmc =3572410 }}{{cite web |url=http://www.nature.com/news/2011/110615/full/474262a.html |title=Mouse library set to be knockout |author=Dolgin E |year=June 2011 |location=Nature 474: 262-263. doi:10.1038/474262a}}{{cite book |title=A mouse for all reasons |author=Collins FS, Rossant J, Wurst W |year=January 2007 |location=Cell 128(1): 9-13. doi:10.1016/j.cell.2006.12.018 PMID 17218247}}

Male and female animals underwent a standardized phenotypic screen to determine the effects of deletion.{{cite journal| author=van der Weyden L, White JK, Adams DJ, Logan DW| title=The mouse genetics toolkit: revealing function and mechanism. | journal=Genome Biol | year= 2011 | volume= 12 | issue= 6 | pages= 224 | pmid=21722353 | doi=10.1186/gb-2011-12-6-224 | url=http://www.ncbi.nlm.nih.gov/entrez/eutils/elink.fcgi?dbfrom=pubmed&tool=sumsearch.org/cite&retmode=ref&cmd=prlinks&id=21722353 }}

Twenty seven tests were carried out on mutant mice and eight significant abnormalities were observed. 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. 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).

References

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