All Phenotypes Vangl1<Gt(XL802)Byg> MGI Mouse

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Phenotypes associated with this allele

Allele Symbol
Allele Name
Allele ID

Vangl1^Gt(XL802)Byg
gene trap XL802, BayGenomics
MGI:3778822

Summary

6 genotypes

Jump to	Allelic Composition	Genetic Background	Genotype ID
hm1	Vangl1^Gt(XL802)Byg/Vangl1^Gt(XL802)Byg	involves: 129P2/OlaHsd * C57BL/6J	MGI:3778823
cx2	Vangl1^Gt(XL802)Byg/Vangl1⁺ Vangl2^ska17/Vangl2⁺	involves: 129P2/OlaHsd * 129S6/SvEvTac * C57BL/6J	MGI:4941950
cx3	Vangl1^Gt(XL802)Byg/Vangl1^Gt(XL802)Byg Vangl2^Lp/Vangl2⁺	involves: 129P2/OlaHsd * A	MGI:4819599
cx4	Vangl1^Gt(XL802)Byg/Vangl1^Gt(XL802)Byg Vangl2^tm1.2Yy/Vangl2^tm1.2Yy	involves: 129P2/OlaHsd * C57BL/6 * CBA	MGI:4819601
cx5	Vangl1^Gt(XL802)Byg/Vangl1^Gt(XL802)Byg Vangl2^tm1.2Yy/Vangl2⁺	involves: 129P2/OlaHsd * C57BL/6 * CBA	MGI:4819600
cx6	Vangl1^Gt(XL802)Byg/Vangl1⁺ Vangl2^Lp/Vangl2⁺	involves: 129P2/OlaHsd * C57BL/6J * LPT/LeJ	MGI:3778824

Allelic Composition	Vangl1^Gt(XL802)Byg/Vangl1^Gt(XL802)Byg
Genetic Background	involves: 129P2/OlaHsd * C57BL/6J

Find Mice

Using the International Mouse Strain Resource (IMSR)
Mouse lines carrying:
Vangl1^Gt(XL802)Byg mutation (1 available); any Vangl1 mutation (85 available)

♀	phenotype observed in females
♂	phenotype observed in males
N	normal phenotype

Abnormal orientation of hair cells of the cochlea in Vangl1^Gt(XL802)Byg/Vangl1^Gt(XL802)Byg embryos

hearing/vestibular/ear

abnormal orientation of cochlear hair cell stereociliary bundles ( J:132697 )

• modest but significant misorientation in all hair cell layers

• however, the number and organization of hair cell rows is not different from wild-type controls

cardiovascular system

cardiovascular system phenotype ( J:132697 )

N	• unlike mice homozygous for Vangl2^Lp alone, nor outflow tract abnormalities are detected

nervous system

nervous system phenotype ( J:132697 )

N	• unlike mice with mutations in Vangl2, no neural tube defects are detected

abnormal orientation of cochlear hair cell stereociliary bundles ( J:132697 )

• modest but significant misorientation in all hair cell layers

• however, the number and organization of hair cell rows is not different from wild-type controls

Mouse Models of Human Disease		DO ID	OMIM ID(s)	Ref(s)
NOT	neural tube defect	DOID:0080074	OMIM:301410 OMIM:601634	J:132697

Allelic Composition	Vangl1^Gt(XL802)Byg/Vangl1⁺ Vangl2^ska17/Vangl2⁺
Genetic Background	involves: 129P2/OlaHsd * 129S6/SvEvTac * C57BL/6J

Find Mice

Using the International Mouse Strain Resource (IMSR)
Mouse lines carrying:
Vangl1^Gt(XL802)Byg mutation (1 available); any Vangl1 mutation (85 available)
Vangl2^ska17 mutation (0 available); any Vangl2 mutation (34 available)

♀	phenotype observed in females
♂	phenotype observed in males
N	normal phenotype

normal phenotype

no abnormal phenotype detected ( J:169664 )

• no tail, neural tube or eyelid fusion defects are detected

Allelic Composition	Vangl1^Gt(XL802)Byg/Vangl1^Gt(XL802)Byg Vangl2^Lp/Vangl2⁺
Genetic Background	involves: 129P2/OlaHsd * A

Find Mice

Using the International Mouse Strain Resource (IMSR)
Mouse lines carrying:
Vangl1^Gt(XL802)Byg mutation (1 available); any Vangl1 mutation (85 available)
Vangl2^Lp mutation (2 available); any Vangl2 mutation (34 available)

♀	phenotype observed in females
♂	phenotype observed in males
N	normal phenotype

reproductive system

vagina atresia ( J:162640 )

female infertility ( J:162640 )

nervous system

abnormal cochlear inner hair cell morphology ( J:162640 )

• inner hair cells are misoriented compared to in wild-type mice

abnormal cochlear outer hair cell morphology ( J:162640 )

• outer hair cells are misoriented compared to in wild-type mice

limbs/digits/tail

short tail ( J:162640 )

• compared with Vangl1^Gt(XL802)Byg/Vangl1^Gt(XL802)Byg Vangl2^tm1.2Yy/Vangl2⁺

hearing/vestibular/ear

abnormal cochlear inner hair cell morphology ( J:162640 )

• inner hair cells are misoriented compared to in wild-type mice

abnormal cochlear outer hair cell morphology ( J:162640 )

• outer hair cells are misoriented compared to in wild-type mice

Allelic Composition	Vangl1^Gt(XL802)Byg/Vangl1^Gt(XL802)Byg Vangl2^tm1.2Yy/Vangl2^tm1.2Yy
Genetic Background	involves: 129P2/OlaHsd * C57BL/6 * CBA

Find Mice

Using the International Mouse Strain Resource (IMSR)
Mouse lines carrying:
Vangl1^Gt(XL802)Byg mutation (1 available); any Vangl1 mutation (85 available)
Vangl2^tm1.2Yy mutation (0 available); any Vangl2 mutation (34 available)

♀	phenotype observed in females
♂	phenotype observed in males
N	normal phenotype

embryo

abnormal embryonic cilium location or orientation ( J:162640 )

• positioning of the posterior notochord (node) cilia is randomized unlike in wild-type mice

• in the posterior notochord (node), cell density is slightly reduced and cilia localization is altered compared to in wild-type mice

abnormal motile primary cilium physiology ( J:162640 )

• nodal flow produces swirling vortices and time to cross the posterior notochord (node) is lengthened compared to in wild-type mice

• unilateral nodal flow in the posterior notochord (node) is compromised compared to in wild-type mice

• however, cilia in the posterior notochord beat at the same speed and direction as in wild-type mice

abnormal embryo turning ( J:162640 )

• embryos fail to turn unlike wild-type mice

abnormal notochord morphology ( J:162640 )

• cell density and the number of posteriorly localized basal bodies in the posterior notochord (node) are decreased compared to in wild-type mice

growth/size/body

abnormal prenatal body size ( J:162640 )

• embryos exhibit a reduction in length to width ratio compared with wild-type mice

heterotaxia ( J:162640 )

• mice exhibit left-right asymmetry defects compared with wild-type mice

• however, nodal cilia appear normal

left pulmonary isomerism ( J:162640 )

• mild

nervous system

abnormal cochlear inner hair cell morphology ( J:162640 )

• inner hair cells are misoriented compared to in wild-type mice

abnormal cochlear outer hair cell morphology ( J:162640 )

• outer hair cells are misoriented compared to in wild-type mice

cardiovascular system

failure of heart looping ( J:162640 )

respiratory system

left pulmonary isomerism ( J:162640 )

• mild

hearing/vestibular/ear

abnormal cochlear inner hair cell morphology ( J:162640 )

• inner hair cells are misoriented compared to in wild-type mice

abnormal cochlear outer hair cell morphology ( J:162640 )

• outer hair cells are misoriented compared to in wild-type mice

cellular

abnormal embryonic cilium location or orientation ( J:162640 )

• positioning of the posterior notochord (node) cilia is randomized unlike in wild-type mice

• in the posterior notochord (node), cell density is slightly reduced and cilia localization is altered compared to in wild-type mice

abnormal motile primary cilium physiology ( J:162640 )

• nodal flow produces swirling vortices and time to cross the posterior notochord (node) is lengthened compared to in wild-type mice

• unilateral nodal flow in the posterior notochord (node) is compromised compared to in wild-type mice

• however, cilia in the posterior notochord beat at the same speed and direction as in wild-type mice

Allelic Composition	Vangl1^Gt(XL802)Byg/Vangl1^Gt(XL802)Byg Vangl2^tm1.2Yy/Vangl2⁺
Genetic Background	involves: 129P2/OlaHsd * C57BL/6 * CBA

Find Mice

♀	phenotype observed in females
♂	phenotype observed in males
N	normal phenotype

nervous system

abnormal cochlear outer hair cell morphology ( J:162640 )

• outer hair cells in rows 3 and 2 are misoriented compared to in wild-type mice

limbs/digits/tail

long tail ( J:162640 )

• compared with Vangl2^Lp/Vangl2^Lp Vangl2^tm1.2Yy/Vangl2⁺

hearing/vestibular/ear

abnormal cochlear outer hair cell morphology ( J:162640 )

• outer hair cells in rows 3 and 2 are misoriented compared to in wild-type mice

Allelic Composition	Vangl1^Gt(XL802)Byg/Vangl1⁺ Vangl2^Lp/Vangl2⁺
Genetic Background	involves: 129P2/OlaHsd * C57BL/6J * LPT/LeJ

Find Mice

♀	phenotype observed in females
♂	phenotype observed in males
N	normal phenotype

Vangl1^Gt(XL802)Byg/Vangl1⁺ Vangl2^Lp/Vangl2⁺ and Vangl2^Lp/Vangl2^Lp embryos exhibit aberrant right subclavian artery

mortality/aging

perinatal lethality, incomplete penetrance ( J:132697 )

• fewer than expected double heterozygotes are found at weaning (20% rather than the expected 50%) given the presence of craniorachischisis late embryonic lethality is probably the cause of the distorted ratio

nervous system

craniorachischisis ( J:132697 )

• seen in over 60% of double heterozygotes at E13.5 - E18.5

• phenotype is as severe as in mice homozygous for Vangl2^Lp alone

• no obvious neural tube defects are seen in surviving mice

abnormal cochlear hair cell stereociliary bundle morphology ( J:132697 )

• profoundly distorted

abnormal orientation of inner hair cell stereociliary bundles ( J:132697 )

• 20% of inner hair cell bundles are misoriented

abnormal orientation of outer hair cell stereociliary bundles ( J:132697 )

• some bundles in all 3 layers are misoriented especially at the apical turn (over 50% of bundles in OHC1, 65% in OHC2, over 80% in OHC3)

• vertices are randomly oriented with rotation angles of 40 - 180 degrees

cardiovascular system

cardiovascular system phenotype ( J:132697 )

N	• unlike mice homozygous for Vangl2^Lp alone, no outflow tract abnormalities are detected in double heterozygotes

retroesophageal right subclavian artery ( J:132697 )

• at E14.5, the right subclavian artery is positioned dorsal to the esophagus

hearing/vestibular/ear

abnormal cochlea morphology ( J:132697 )

• reduced in size at E18.5 in mice displaying craniorachischisis

abnormal cochlear hair cell stereociliary bundle morphology ( J:132697 )

• profoundly distorted

abnormal orientation of inner hair cell stereociliary bundles ( J:132697 )

• 20% of inner hair cell bundles are misoriented

abnormal orientation of outer hair cell stereociliary bundles ( J:132697 )

• some bundles in all 3 layers are misoriented especially at the apical turn (over 50% of bundles in OHC1, 65% in OHC2, over 80% in OHC3)

• vertices are randomly oriented with rotation angles of 40 - 180 degrees

limbs/digits/tail

curly tail ( J:132697 )

embryo

craniorachischisis ( J:132697 )

• seen in over 60% of double heterozygotes at E13.5 - E18.5

• phenotype is as severe as in mice homozygous for Vangl2^Lp alone

• no obvious neural tube defects are seen in surviving mice

Mouse Models of Human Disease		DO ID	OMIM ID(s)	Ref(s)
neural tube defect		DOID:0080074	OMIM:301410 OMIM:601634	J:132697

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