All Phenotypes En1<tm2(cre)Wrst> MGI Mouse

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Phenotypes associated with this allele

Allele Symbol
Allele Name
Allele ID

En1^tm2(cre)Wrst
targeted mutation 2, Wolfgang Wurst
MGI:2446434

Summary

29 genotypes

Jump to	Allelic Composition	Genetic Background	Genotype ID
cn1	En1^tm2(cre)Wrst/En1^tm8.1Alj	involves: 129/Sv * Black Swiss * C57BL/6 * SJL	MGI:3789299
cn2	En1^tm2(cre)Wrst/En1⁺ Mfn2^tm1Dcc/Mfn2^tm3Dcc	involves: 129 * 129S4/SvJaeSor * Black Swiss	MGI:3779095
cn3	Dnm1l^tm1.1Hise/Dnm1l^tm1.2Hise En1^tm2(cre)Wrst/En1⁺	involves: 129 * C57BL/6 * FVB/N * SJL	MGI:4366518
cn4	Wnt1^tm1.1Mze/Wnt1^tm1.1Mze Gt(ROSA)26Sor^{tm14(CAG-tdTomato)Hze}/Gt(ROSA)26Sor⁺ En1^tm2(cre)Wrst/En1⁺	involves: 129 * C57BL/6N	MGI:5495280
cn5	Upf2^tm1Btp/Upf2^tm1Btp En1^tm2(cre)Wrst/En1⁺	involves: 129P2/OlaHsd * 129S1/Sv * 129X1/SvJ	MGI:6718865
cn6	En1^tm2(cre)Wrst/En1⁺ Tor1a^tm1Wtd/Tor1a^tm3.1Wtd	involves: 129P2/OlaHsd * 129S7/SvEvBrd * C57BL/6J	MGI:5605974
cn7	Drd2^tm3.1Ebo/Drd2^tm3.1Ebo En1^tm2(cre)Wrst/En1⁺	involves: 129S1/Sv * 129S2/SvPas * 129X1/SvJ * C57BL/6	MGI:5431882
cn8	Hbs1l^{tm1c(KOMP)Wtsi}/Hbs1l^{tm1c(KOMP)Wtsi} En1^tm2(cre)Wrst/En1⁺	involves: 129S1/Sv * 129S4/SvJaeSor * 129X1/SvJ * C57BL/6NTac	MGI:6718853
cn9	Gt(ROSA)26Sor^{tm7(CAG-mCherry,-EGFP/tetX)Dym}/Gt(ROSA)26Sor⁺ En1^tm2(cre)Wrst/En1⁺ Tg(Fev-flpe)1Dym/0	involves: 129S1/Sv * 129S6/SvEvTac * 129X1/SvJ	MGI:4412291
cn10	En1^tm2(cre)Wrst/? Gli3^tm1Alj/Gli3^tm1Alj Smo^tm2Amc/Smo^tm2Amc	involves: 129S1/Sv * 129S6/SvEvTac * 129X1/SvJ * C57BL/6	MGI:3803100
cn11	En1^tm2(cre)Wrst/? Gli3^tm1Alj/Gli3^tm1Alj	involves: 129S1/Sv * 129S6/SvEvTac * 129X1/SvJ * C57BL/6	MGI:3803098
cn12	Gt(ROSA)26Sor^{tm7(CAG-mCherry,-EGFP/tetX)Dym}/Gt(ROSA)26Sor⁺ En1^tm2(cre)Wrst/En1⁺ Tg(ACTFLPe)9205Dym/0	involves: 129S1/Sv * 129S6/SvEvTac * 129X1/SvJ * C57BL/6 * SJL	MGI:4412289
cn13	En1^tm2(cre)Wrst/En1⁺ Tor1a^tm2Wtd/Tor1a^tm3.1Wtd	involves: 129S1/Sv * 129S6/SvEvTac * C57BL/6J * SJL	MGI:5605977
cn14	En1^tm2(cre)Wrst/En1⁺ Lhx1^tm1Tmj/Lhx1^tm2.1Bhr Lhx5^tm1Lmgd/Lhx5^tm1Lmgd	involves: 129S1/Sv * 129S7/SvEvBrd * 129X1/SvJ	MGI:3719690
cn15	En1^tm2(cre)Wrst/En1⁺ Lhx1^tm2.1Bhr/Lhx1^tm2.1Bhr	involves: 129S1/Sv * 129S7/SvEvBrd * 129X1/SvJ	MGI:3719688
cn16	En1^tm2(cre)Wrst/En1⁺ Fgfr1^tm1Jpa/Fgfr1^tm1Jpa	involves: 129S1/Sv * 129X1/SvJ	MGI:3702736
cn17	En1^tm2(cre)Wrst/? Gt(ROSA)26Sor^{tm5(CAG-EGFP,-lacZ)Dym}/? Tg(Fev-flpe)1Dym/?	involves: 129S1/Sv * 129X1/SvJ	MGI:3804425
cn18	En1^tm2(cre)Wrst/? Gt(ROSA)26Sor^{tm5(CAG-EGFP,-lacZ)Dym}/? Nkx2-2^tm1Jlr/Nkx2-2^tm1Jlr Tg(Fev-flpe)1Dym/?	involves: 129S1/Sv * 129X1/SvJ	MGI:3804428
cn19	En1^tm2(cre)Wrst/En1⁺ Gata2^tm1Msal/Gata2^tm1Msal	involves: 129S1/Sv * 129X1/SvJ	MGI:4818570
cn20	Slc12a2^tm1.1Jheb/Slc12a2^tm1.1Jheb En1^tm2(cre)Wrst/En1⁺	involves: 129S1/Sv * 129X1/SvJ * C57BL/6	MGI:5538345
cn21	En1^tm2(cre)Wrst/En1⁺ Fgfr2^tm1Wrst/Fgfr2^tm1Wrst	involves: 129S1/Sv * 129X1/SvJ * C57BL/6	MGI:3700937
cn22	Ankrd16^tm1.1Slac/Ankrd16^tm1.2Slac En1^tm2(cre)Wrst/En1⁺	involves: 129S1/Sv * 129X1/SvJ * C57BL/6J	MGI:6197726
cn23	Pelo^tm1Slac/Pelo^tm1Slac En1^tm2(cre)Wrst/En1⁺	involves: 129S1/Sv * 129X1/SvJ * C57BL/6J	MGI:6718863
cn24	Wnt1^tm1.1Mze/Wnt1^tm1.1Mze En1^tm2(cre)Wrst/En1⁺	involves: 129S1/Sv * 129X1/SvJ * C57BL/6N	MGI:5495279
cn25	Atg7^tm1Tchi/Atg7^tm1Tchi En1^tm2(cre)Wrst/En1⁺	involves: 129S1/Sv * 129X1/SvJ * C57BL/6NCrlj * CBA/JNCrlj	MGI:5471363
cn26	Dnm1l^tm1.1Hise/Dnm1l^tm1.1Hise En1^tm2(cre)Wrst/En1⁺	involves: 129S/SvEv * 129S1/Sv * 129X1/SvJ * C57BL/6 * SJL	MGI:4366517
cn27	En1^tm2(cre)Wrst/En1^tm8.1Alj En2^tm2Alj/En2^tm2Alj	involves: 129/Sv * Black Swiss * C57BL/6 * SJL * Swiss Webster	MGI:3789301
cn28	En1^tm2(cre)Wrst/En1^tm8.1Alj En2^tm2Alj/En2⁺	involves: 129/Sv * Black Swiss * C57BL/6 * SJL * Swiss Webster	MGI:3789300
cn29	En1^tm2(cre)Wrst/En1⁺ Gbx2^tm1.1Mrt/Gbx2^tm1.1Alj	involves: 129/Sv * C57BL/6 * SJL * Swiss Webster	MGI:3790208

Allelic Composition	En1^tm2(cre)Wrst/En1^tm8.1Alj
Genetic Background	involves: 129/Sv * Black Swiss * C57BL/6 * SJL

Find Mice

Using the International Mouse Strain Resource (IMSR)
Mouse lines carrying:
En1^tm2(cre)Wrst mutation (1 available); any En1 mutation (32 available)
En1^tm8.1Alj mutation (1 available); any En1 mutation (32 available)

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

nervous system

abnormal inferior colliculus morphology ( J:134516 )

abnormal cerebellum anterior vermis morphology ( J:134516 )

• in most adult mice, mild foliation defect is observed

• fissure between anterior-most folia I/II and III either fails to form or is shallower than normal in most animals

• at E18.5, vermis may be slightly delayed in forming folia

decreased anterior vermis size ( J:134516 )

• overall size in some mutants is slightly reduced relative to wild-type at E18.5

Allelic Composition	En1^tm2(cre)Wrst/En1⁺ Mfn2^tm1Dcc/Mfn2^tm3Dcc
Genetic Background	involves: 129 * 129S4/SvJaeSor * Black Swiss

Find Mice

Using the International Mouse Strain Resource (IMSR)
Mouse lines carrying:
En1^tm2(cre)Wrst mutation (1 available); any En1 mutation (32 available)
Mfn2^tm1Dcc mutation (0 available); any Mfn2 mutation (26 available)
Mfn2^tm3Dcc mutation (2 available); any Mfn2 mutation (26 available)

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

mortality/aging

postnatal lethality, complete penetrance ( J:132329 )

• mutant pups are born at appropriate Mendelian ratio

• after birth, about one third of mutant mice die on postnatal day 1

• all remaining mutant mice die by P17

behavior/neurological

impaired righting response ( J:132329 )

• mice surviving beyond P1 cannot easily regain posture when placed on their backs

impaired limb coordination ( J:132329 )

• mice surviving beyond P1 display uncoordinated limb movements, and move primarily by writhing on their abdomen

growth/size/body

decreased body size ( J:132329 )

• mice surviving beyond P1 are severely runted, likely due to feeding problems secondary to their movement disorder

nervous system

abnormal neuron mitochondrial morphology ( J:132329 )

• mitochondria in mutant Purkinje cells in mixed cerebellar cultures tend to cluster together in the cell body and do not enter the dendritic tracts

increased neuron apoptosis ( J:132329 )

• markedly higher levels of apoptosis in mutant cerebella as early as p6 and continuing through the second postnatal week

Purkinje cell degeneration ( J:132329 )

• widespread loss of Purkinje cell bodies py P15 resulting in reduction of the molecular layer

• Purkinje cell loss due to a degenerative process in mixed cerebellar cultures

abnormal Purkinje cell dendrite morphology ( J:132329 )

• reduced growth and deterioration of Purkinje cell dendrite during the second postnatal week

small cerebellum ( J:132329 )

• severe defect in postnatal cerebellar growth

• between P7 and P16, the mutant cerebellum reduces in size

• lobular organization and formation of the three cellular layers is relatively intact

cellular

abnormal neuron mitochondrial morphology ( J:132329 )

• mitochondria in mutant Purkinje cells in mixed cerebellar cultures tend to cluster together in the cell body and do not enter the dendritic tracts

increased neuron apoptosis ( J:132329 )

• markedly higher levels of apoptosis in mutant cerebella as early as p6 and continuing through the second postnatal week

Allelic Composition	Dnm1l^tm1.1Hise/Dnm1l^tm1.2Hise En1^tm2(cre)Wrst/En1⁺
Genetic Background	involves: 129 * C57BL/6 * FVB/N * SJL

Find Mice

Using the International Mouse Strain Resource (IMSR)
Mouse lines carrying:
Dnm1l^tm1.1Hise mutation (0 available); any Dnm1l mutation (43 available)
Dnm1l^tm1.2Hise mutation (0 available); any Dnm1l mutation (43 available)
En1^tm2(cre)Wrst mutation (1 available); any En1 mutation (32 available)

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

Defects in cerebellar development in Dnm1l^tm1.1Hise/Dnm1l^tm1.2Hise En1^tm2(cre)Wrst/En1⁺ and Dnm1l^tm1.2Hise/Dnm1l^tm1.2Hise En1^tm2(cre)Wrst/En1⁺ mice

cellular

abnormal Purkinje cell mitochondrial morphology ( J:153835 )

• mitochondria in Purkinje cells are enlarged compared to in wild-type cells

mortality/aging

postnatal lethality, complete penetrance ( J:153835 )

• mice die within 36 hours of birth

nervous system

abnormal cerebellum development ( J:153835 )

• at P0, cerebellum are smooth and exhibit a 60% decreased in size compared to in wild-type mice

reduced cerebellar foliation ( J:153835 )

• at P0, cerebellum lack foliation

• at P0.5, lobule fissures are barely detectable

abnormal cerebellar Purkinje cell layer ( J:153835 )

• Purkinje cells in the cerebellum are decreased in number and form a discontinuous cell layer unlike in wild-type mice

abnormal Purkinje cell mitochondrial morphology ( J:153835 )

• mitochondria in Purkinje cells are enlarged compared to in wild-type cells

decreased Purkinje cell number ( J:153835 )

• in the cerebellum

small cerebellum ( J:153835 )

• at P0, cerebellum are 60% smaller than in wild-type mice due to decreased cell proliferation

behavior/neurological

absent gastric milk in neonates ( J:153835 )

• mice die without milk in their stomach

• however, mice exhibit normal swallowing when manually fed milk

Allelic Composition	Wnt1^tm1.1Mze/Wnt1^tm1.1Mze Gt(ROSA)26Sor^{tm14(CAG-tdTomato)Hze}/Gt(ROSA)26Sor⁺ En1^tm2(cre)Wrst/En1⁺
Genetic Background	involves: 129 * C57BL/6N

Find Mice

Using the International Mouse Strain Resource (IMSR)
Mouse lines carrying:
En1^tm2(cre)Wrst mutation (1 available); any En1 mutation (32 available)
Gt(ROSA)26Sor^{tm14(CAG-tdTomato)Hze} mutation (5 available); any Gt(ROSA)26Sor mutation (942 available)
Wnt1^tm1.1Mze mutation (0 available); any Wnt1 mutation (30 available)

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

nervous system

abnormal rhombomere morphology ( J:194842 )

• rhombomere 1 is diminished with only a small domain adjacent to axonal bundles connected to the trigeminal ganglia

• dorsal rhombomere 1 is severely depleted but the ventral portion is less severely affected

abnormal midbrain morphology ( J:194842 )

• substantial depletion of the En1 lineage derived mesencephalon at E12.5

• dorsal mesencephalon is severely depleted but the ventral portion is less severely affected

abnormal hindbrain morphology ( J:194842 )

• severe truncation of the lateral anterior hindbrain

abnormal innervation ( J:194842 )

• aberrantly patterned whisker fields innervated by serotonergic neurons

decreased neuronal precursor cell number ( J:194842 )

• absence of LMX1a+ progenitors throughout the medial-lateral extent of the ventral mesencephalon and decreased numbers in the ventral diencephalon

decreased neuron number ( J:194842 )

• only a small disorganized cohort of TH+ MbDA neurons remain at E8.5

embryo

abnormal rhombomere morphology ( J:194842 )

• rhombomere 1 is diminished with only a small domain adjacent to axonal bundles connected to the trigeminal ganglia

• dorsal rhombomere 1 is severely depleted but the ventral portion is less severely affected

Allelic Composition	Upf2^tm1Btp/Upf2^tm1Btp En1^tm2(cre)Wrst/En1⁺
Genetic Background	involves: 129P2/OlaHsd * 129S1/Sv * 129X1/SvJ

Find Mice

Using the International Mouse Strain Resource (IMSR)
Mouse lines carrying:
En1^tm2(cre)Wrst mutation (1 available); any En1 mutation (32 available)
Upf2^tm1Btp mutation (0 available); any Upf2 mutation (62 available)

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

nervous system

increased neuron apoptosis ( J:306384 )

• increased apoptotic cells in the cerebellum in both the ventricular zone and the prospective white matter

increased neuronal precursor proliferation ( J:306384 )

• in the ventricular zone progenitors at E13.5

abnormal cerebellum external granule cell layer morphology ( J:306384 )

• missing at E13.5 and P0

decreased inferior colliculus size ( J:306384 )

• grossly hypoplastic being nearly absent

decreased superior colliculus size ( J:306384 )

• grossly hypoplastic being nearly absent

cerebellum hypoplasia ( J:306384 )

• grossly being nearly absent

loss of GABAergic neurons ( J:306384 )

• at E13.5 in the cerebellum

cellular

increased neuron apoptosis ( J:306384 )

• increased apoptotic cells in the cerebellum in both the ventricular zone and the prospective white matter

increased neuronal precursor proliferation ( J:306384 )

• in the ventricular zone progenitors at E13.5

Allelic Composition	En1^tm2(cre)Wrst/En1⁺ Tor1a^tm1Wtd/Tor1a^tm3.1Wtd
Genetic Background	involves: 129P2/OlaHsd * 129S7/SvEvBrd * C57BL/6J

Find Mice

Using the International Mouse Strain Resource (IMSR)
Mouse lines carrying:
En1^tm2(cre)Wrst mutation (1 available); any En1 mutation (32 available)
Tor1a^tm1Wtd mutation (1 available); any Tor1a mutation (22 available)
Tor1a^tm3.1Wtd mutation (1 available); any Tor1a mutation (22 available)

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

nervous system

abnormal red nucleus morphology ( J:213785 )

gliosis ( J:213785 )

• reactive gliosis is observed in superior cerebellar peduncle and deep cerebellar nuclei

decreased neuron number ( J:213785 )

• decrease in neuron number in red nucleus and deep cerebellar nuclei (DCN) as compared to controls

• neuron loss in DCN is 2 fold higher in this genotype as compared to mice carrying Tor1a^tm2Wtd allele

neurodegeneration ( J:213785 )

• neurodegeneration is observed in midbrain/hindbrain

behavior/neurological

limb grasping ( J:213785 )

• hindlimb and forelimb clasping observed by P15

• forepaw clenching during tail suspension observed by P15

dystonia ( J:213785 )

• twisted truncal postures observed by P15, however, mice do not exhibit spontaneous hind paw twisting

growth/size/body

decreased body weight ( J:213785 )

• mice are weigh less by P28 than littermate controls

muscle

dystonia ( J:213785 )

• twisted truncal postures observed by P15, however, mice do not exhibit spontaneous hind paw twisting

Allelic Composition	Drd2^tm3.1Ebo/Drd2^tm3.1Ebo En1^tm2(cre)Wrst/En1⁺
Genetic Background	involves: 129S1/Sv * 129S2/SvPas * 129X1/SvJ * C57BL/6

Find Mice

Using the International Mouse Strain Resource (IMSR)
Mouse lines carrying:
Drd2^tm3.1Ebo mutation (0 available); any Drd2 mutation (70 available)
En1^tm2(cre)Wrst mutation (1 available); any En1 mutation (32 available)

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

behavior/neurological

enhanced behavioral response to xenobiotic ( J:185643 )

• mice fed cocaine exhibit increased locomotion compared with control mice

• mice exhibit enhanced response to nomifensine with greater relative contribution of dopamine transporter activity to peak amplitude compared with control mice

enhanced behavioral response to cocaine ( J:185643 )

• mice treated with cocaine exhibit increased hyperactivity compared with control mice

enhanced behavioral response to anesthetic ( J:185643 )

• mice exhibit increased catalepsy induced by haloperidol compared with control mice

hyperactivity ( J:185643 )

• in an open field

increased vertical activity ( J:185643 )

induced hyperactivity ( J:185643 )

• in mice fed cocaine

nervous system

abnormal nervous system electrophysiology ( J:185643 )

homeostasis/metabolism

abnormal dopamine level ( J:185643 )

• dopamine overflow evoked by a single stimulus in the dorsal striatum (DSt) is decreased compared to in control mice

• mice exhibit lower AMP evoked dopamine release compared with control mice

• dopamine reuptake is increased compared to in control mice

• mice exhibit enhanced response to nomifensine with greater relative contribution of dopamine transporter activity to peak amplitude compared with control mice

• following paired pulse, dopamine overflow is higher than in control mice

• single-pulse evoke dopamine overflow is decreased compared to in control mice

• multiple-pulse evoked dopamine overflow is increased compared to in wild-type mice

• however, quinpirole-treated mice exhibit normal dose-dependent inhibition of dopamine overflow

increased dopamine level ( J:185643 )

• in the nucleus accumbens (NAcc) and cortex

decreased physiological sensitivity to xenobiotic ( J:185643 )

• mice treated with quinpirole exhibit less of a decrease in locomotion compared with control mice

• mice treated with quinpirole in a novel environment fail to exhibit a decrease in locomotion compared with control mice

Allelic Composition	Hbs1l^{tm1c(KOMP)Wtsi}/Hbs1l^{tm1c(KOMP)Wtsi} En1^tm2(cre)Wrst/En1⁺
Genetic Background	involves: 129S1/Sv * 129S4/SvJaeSor * 129X1/SvJ * C57BL/6NTac

Find Mice

Using the International Mouse Strain Resource (IMSR)
Mouse lines carrying:
En1^tm2(cre)Wrst mutation (1 available); any En1 mutation (32 available)
Hbs1l^{tm1c(KOMP)Wtsi} mutation (0 available); any Hbs1l mutation (49 available)

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

nervous system

increased neuronal precursor proliferation ( J:306384 )

• more GABAergic precursors are retained between E12.5 and E16.5 due to increased proliferation

abnormal cerebellum morphology ( J:306384 )

• cerebellar defects are not rescued by restoration or complete deletion of n-TRtct5

abnormal cerebellar foliation ( J:306384 )

• delayed at P0 with failure of secondary fissures to form

• however, the trilaminar structure is normal at P21

decreased Purkinje cell number ( J:306384 )

• reduced ventricular zone-derived Lhx1/5+ Purkinje cells between E12.5 and E16.5

small cerebellum ( J:306384 )

• by E13.5

abnormal GABAergic neuron morphology ( J:306384 )

• more GABAergic precursors are retained between E12.5 and E16.5 due to increased proliferation

decreased neuronal precursor cell number ( J:306384 )

• reduced Pax2+ interneuron precursors between E12.5 and E16.5

• reduced granule cell precursors at E13.5 to E16.5

• reduced oligodendroglial progenitors at P5

• however, granule cell precursor proliferation is normal at E16.5 and P5

cellular

increased neuronal precursor proliferation ( J:306384 )

• more GABAergic precursors are retained between E12.5 and E16.5 due to increased proliferation

Allelic Composition	Gt(ROSA)26Sor^{tm7(CAG-mCherry,-EGFP/tetX)Dym}/Gt(ROSA)26Sor⁺ En1^tm2(cre)Wrst/En1⁺ Tg(Fev-flpe)1Dym/0
Genetic Background	involves: 129S1/Sv * 129S6/SvEvTac * 129X1/SvJ

Find Mice

Using the International Mouse Strain Resource (IMSR)
Mouse lines carrying:
En1^tm2(cre)Wrst mutation (1 available); any En1 mutation (32 available)
Gt(ROSA)26Sor^{tm7(CAG-mCherry,-EGFP/tetX)Dym} mutation (0 available); any Gt(ROSA)26Sor mutation (942 available)
Tg(Fev-flpe)1Dym mutation (0 available)

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

behavior/neurological

behavior/neurological phenotype ( J:154944 )

N	• mice exhibit normal contextual fear conditioning and prepulse mediated inhibition of acoustic startle reflex

decreased anxiety-related response ( J:154944 )

nervous system

abnormal axon morphology ( J:154944 )

• 5HT+ axon varicosities are enlarged compared to in wild-type mice

Allelic Composition	En1^tm2(cre)Wrst/? Gli3^tm1Alj/Gli3^tm1Alj Smo^tm2Amc/Smo^tm2Amc
Genetic Background	involves: 129S1/Sv * 129S6/SvEvTac * 129X1/SvJ * C57BL/6

Find Mice

Using the International Mouse Strain Resource (IMSR)
Mouse lines carrying:
En1^tm2(cre)Wrst mutation (1 available); any En1 mutation (32 available)
Gli3^tm1Alj mutation (1 available); any Gli3 mutation (80 available)
Smo^tm2Amc mutation (1 available); any Smo mutation (39 available)

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

nervous system

nervous system phenotype ( J:137136 )

N	• inferior colliculus normal • isthmus generally normal • superior colliculus normal

abnormal rhombomere morphology ( J:137136 )

• Purkinje projections into posterior isthmus region

• some Purkinje cells in anterior isthmus region

• dorsal mesencephalon, isthmus, and r1 not distinct at E10.5 and E12.5

• isthmus flexure less prominent

• thickness of ventricular zone increased

enlarged cerebral aqueduct ( J:137136 )

• expanded mesencephalic ventricle at E10.5 and E12.5

abnormal tectum morphology ( J:137136 )

• misshapen

enlarged tectum ( J:137136 )

abnormal hindbrain morphology ( J:137136 )

abnormal cerebellum morphology ( J:137136 )

abnormal cerebellar foliation ( J:137136 )

• abnormal at E18.5

thin external granule cell layer ( J:137136 )

• external granule cell layer thinner at birth

small cerebellum ( J:137136 )

• at birth

embryo

abnormal rhombomere morphology ( J:137136 )

• some Purkinje cells in anterior isthmus region

• Purkinje projections into posterior isthmus region

• dorsal mesencephalon, isthmus, and r1 not distinct at E10.5 and E12.5

• isthmus flexure less prominent

• thickness of ventricular zone increased

Allelic Composition	En1^tm2(cre)Wrst/? Gli3^tm1Alj/Gli3^tm1Alj
Genetic Background	involves: 129S1/Sv * 129S6/SvEvTac * 129X1/SvJ * C57BL/6

Find Mice

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

nervous system

nervous system phenotype ( J:137136 )

N	• isthmus generally normal • superior colliculus normal • inferior colliculus normal

abnormal rhombomere morphology ( J:137136 )

• some Purkinje cells in anterior isthmus region

• Purkinje projections into posterior isthmus region

• dorsal mesencephalon, isthmus, and r1 not distinct at E10.5 and E12.5

• isthmus flexure less prominent

• thickness of ventricular zone increased

enlarged cerebral aqueduct ( J:137136 )

• expanded mesencephalic ventricle at E10.5 and E12.5

abnormal tectum morphology ( J:137136 )

• misshapen

enlarged tectum ( J:137136 )

abnormal hindbrain morphology ( J:137136 )

abnormal cerebellar foliation ( J:137136 )

• abnormal at E18.5

• cerebellum otherwise normal

embryo

abnormal rhombomere morphology ( J:137136 )

• some Purkinje cells in anterior isthmus region

• Purkinje projections into posterior isthmus region

• dorsal mesencephalon, isthmus, and r1 not distinct at E10.5 and E12.5

• isthmus flexure less prominent

• thickness of ventricular zone increased

Allelic Composition	Gt(ROSA)26Sor^{tm7(CAG-mCherry,-EGFP/tetX)Dym}/Gt(ROSA)26Sor⁺ En1^tm2(cre)Wrst/En1⁺ Tg(ACTFLPe)9205Dym/0
Genetic Background	involves: 129S1/Sv * 129S6/SvEvTac * 129X1/SvJ * C57BL/6 * SJL

Find Mice

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

mortality/aging

perinatal lethality ( J:154944 )

Allelic Composition	En1^tm2(cre)Wrst/En1⁺ Tor1a^tm2Wtd/Tor1a^tm3.1Wtd
Genetic Background	involves: 129S1/Sv * 129S6/SvEvTac * C57BL/6J * SJL

Find Mice

Using the International Mouse Strain Resource (IMSR)
Mouse lines carrying:
En1^tm2(cre)Wrst mutation (1 available); any En1 mutation (32 available)
Tor1a^tm2Wtd mutation (1 available); any Tor1a mutation (22 available)
Tor1a^tm3.1Wtd mutation (1 available); any Tor1a mutation (22 available)

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

behavior/neurological

limb grasping ( J:213785 )

• hindlimb and forelimb clasping observed by P15, however, with maturity clasping decreases

• forepaw clenching during tail suspension observed by P15

dystonia ( J:213785 )

• twisted truncal postures observed by P15, however, mice do not exhibit spontaneous twisting of hind paws

impaired coordination ( J:213785 )

• mice exhibit an increase in number of footslip/cross in beam crossing

muscle

dystonia ( J:213785 )

• twisted truncal postures observed by P15, however, mice do not exhibit spontaneous twisting of hind paws

nervous system

abnormal red nucleus morphology ( J:213785 )

gliosis ( J:213785 )

decreased neuron number ( J:213785 )

• decrease in neuron number in red nucleus and deep cerebellar nuclei (DCN) as compared to controls

• neuron loss in DCN is 2 fold less in this genotype as compared to mice carrying Tor1a^tm1Wtd allele

abnormal facial nerve morphology ( J:213785 )

neurodegeneration ( J:213785 )

• neurodegeneration is milder in in this genotype as compared to mice carrying Tor1a^tm1Wtd allele

Allelic Composition	En1^tm2(cre)Wrst/En1⁺ Lhx1^tm1Tmj/Lhx1^tm2.1Bhr Lhx5^tm1Lmgd/Lhx5^tm1Lmgd
Genetic Background	involves: 129S1/Sv * 129S7/SvEvBrd * 129X1/SvJ

Find Mice

Using the International Mouse Strain Resource (IMSR)
Mouse lines carrying:
En1^tm2(cre)Wrst mutation (1 available); any En1 mutation (32 available)
Lhx1^tm1Tmj mutation (0 available); any Lhx1 mutation (22 available)
Lhx1^tm2.1Bhr mutation (0 available); any Lhx1 mutation (22 available)
Lhx5^tm1Lmgd mutation (0 available); any Lhx5 mutation (18 available)

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

nervous system

decreased Purkinje cell number ( J:123471 )

• largely absent at E18.5

absent Purkinje cell layer ( J:123471 )

• layer is absent at E18.5

• however, the external granule cell layer appears normal

small cerebellum ( J:123471 )

• small at E18.5 compared to controls

Allelic Composition	En1^tm2(cre)Wrst/En1⁺ Lhx1^tm2.1Bhr/Lhx1^tm2.1Bhr
Genetic Background	involves: 129S1/Sv * 129S7/SvEvBrd * 129X1/SvJ

Find Mice

Using the International Mouse Strain Resource (IMSR)
Mouse lines carrying:
En1^tm2(cre)Wrst mutation (1 available); any En1 mutation (32 available)
Lhx1^tm2.1Bhr mutation (0 available); any Lhx1 mutation (22 available)

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

normal phenotype

no abnormal phenotype detected ( J:123471 )

• viable and fertile

Allelic Composition	En1^tm2(cre)Wrst/En1⁺ Fgfr1^tm1Jpa/Fgfr1^tm1Jpa
Genetic Background	involves: 129S1/Sv * 129X1/SvJ

Find Mice

Using the International Mouse Strain Resource (IMSR)
Mouse lines carrying:
En1^tm2(cre)Wrst mutation (1 available); any En1 mutation (32 available)
Fgfr1^tm1Jpa mutation (0 available); any Fgfr1 mutation (221 available)

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

nervous system

abnormal midbrain morphology ( J:83004 )

• extensive deletions are seen in the posterior midbrain

• however, no extensive deletions or alterations are seen in the basal plate of the midbrain-hindbrain region and no change in apoptosis is seen in the dorsal midbrain at E9.5

absent inferior colliculus ( J:83004 )

• absence of the inferior colliculus in the posterior midbrain in newborn mice

abnormal locus ceruleus morphology ( J:83004 )

• appears disorganized

abnormal cerebellum morphology ( J:83004 )

• abnormal foliation of the hemispheres

absent cerebellum vermis ( J:83004 )

• absent in newborns and adults

abnormal trochlear nerve morphology ( J:83004 )

• however, in adults the trochlear motoneurons are distinguishable and the oculomotor nerve is normal in both adults and E10.5 embryos

• at E10.5 the dorsal part of the trochlear nerve can not be distinguished

behavior/neurological

impaired coordination ( J:83004 )

• impaired performance in stationary beam and rotarod assays

abnormal posture ( J:83004 )

• adults have a wide stance

abnormal gait ( J:83004 )

Allelic Composition	En1^tm2(cre)Wrst/? Gt(ROSA)26Sor^{tm5(CAG-EGFP,-lacZ)Dym}/? Tg(Fev-flpe)1Dym/?
Genetic Background	involves: 129S1/Sv * 129X1/SvJ

Find Mice

Using the International Mouse Strain Resource (IMSR)
Mouse lines carrying:
En1^tm2(cre)Wrst mutation (1 available); any En1 mutation (32 available)
Gt(ROSA)26Sor^{tm5(CAG-EGFP,-lacZ)Dym} mutation (0 available); any Gt(ROSA)26Sor mutation (942 available)
Tg(Fev-flpe)1Dym mutation (0 available)

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

normal phenotype

no abnormal phenotype detected ( J:136096 )

• alleles are used in intersectional and subtractive cell lineage fate mapping to visualize serotonin (5-HT) producing neurons and their precursors in the E12.5 embryonic hindbrain or mature brainstem

• rhombomere-1 (r1) derived 5-HT precursor cells are labeled by beta-gal while 5-HT precursors arising caudal to r1 are labeled by eGFP

Allelic Composition	En1^tm2(cre)Wrst/? Gt(ROSA)26Sor^{tm5(CAG-EGFP,-lacZ)Dym}/? Nkx2-2^tm1Jlr/Nkx2-2^tm1Jlr Tg(Fev-flpe)1Dym/?
Genetic Background	involves: 129S1/Sv * 129X1/SvJ

Find Mice

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

nervous system

abnormal rhombomere morphology ( J:136096 )

• alleles are used in intersectional and subtractive cell lineage fate mapping to visualize serotonin (5-HT) producing neurons and their precursors in the P0.5 brainstem

• rhombomere-1 (r1) derived 5-HT precursor cells are not found in the B1-B3 areas of the brainstem

embryo

abnormal rhombomere morphology ( J:136096 )

• alleles are used in intersectional and subtractive cell lineage fate mapping to visualize serotonin (5-HT) producing neurons and their precursors in the P0.5 brainstem

• rhombomere-1 (r1) derived 5-HT precursor cells are not found in the B1-B3 areas of the brainstem

Allelic Composition	En1^tm2(cre)Wrst/En1⁺ Gata2^tm1Msal/Gata2^tm1Msal
Genetic Background	involves: 129S1/Sv * 129X1/SvJ

Find Mice

Using the International Mouse Strain Resource (IMSR)
Mouse lines carrying:
En1^tm2(cre)Wrst mutation (1 available); any En1 mutation (32 available)
Gata2^tm1Msal mutation (1 available); any Gata2 mutation (26 available)

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

nervous system

nervous system phenotype ( J:143527 )

N	• mice exhibit normal midbrain morphology and neuronal precursor proliferation, survival, patterning, and cell cycle exit

abnormal neuron differentiation ( J:143527 )

• GABAergic precursor cells adopt marker expression similar to precursor cells in glutamatergic regions unlike in wild-type mice

abnormal GABAergic neuron morphology ( J:143527 )

• mice lack GABAergic neuron precursors at the dorsoventral and anteroposterior levels of the midbrain unlike in wild-type mice

• however, GABAergic neurons form in the rhombomere 1 and ventral dopaminergic nuclei

abnormal serotonergic neuron morphology ( J:143527 )

• rhombomere 1 lack serotonergic neurons unlike in wild-type mice

cellular

abnormal neuron differentiation ( J:143527 )

• GABAergic precursor cells adopt marker expression similar to precursor cells in glutamatergic regions unlike in wild-type mice

Allelic Composition	Slc12a2^tm1.1Jheb/Slc12a2^tm1.1Jheb En1^tm2(cre)Wrst/En1⁺
Genetic Background	involves: 129S1/Sv * 129X1/SvJ * C57BL/6

Find Mice

Using the International Mouse Strain Resource (IMSR)
Mouse lines carrying:
En1^tm2(cre)Wrst mutation (1 available); any En1 mutation (32 available)
Slc12a2^tm1.1Jheb mutation (0 available); any Slc12a2 mutation (58 available)

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

behavior/neurological

behavior/neurological phenotype ( J:201156 )

N	• no behavioral abnormalities are detected

Allelic Composition	En1^tm2(cre)Wrst/En1⁺ Fgfr2^tm1Wrst/Fgfr2^tm1Wrst
Genetic Background	involves: 129S1/Sv * 129X1/SvJ * C57BL/6

Find Mice

Using the International Mouse Strain Resource (IMSR)
Mouse lines carrying:
En1^tm2(cre)Wrst mutation (1 available); any En1 mutation (32 available)
Fgfr2^tm1Wrst mutation (0 available); any Fgfr2 mutation (87 available)

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

nervous system

nervous system phenotype ( J:118802 )

N	• mutants do not show defects in brain morphology and development (mid/hindbrain patterning, boundary development, neuronal subpopulations, or oligodendrocyte development)

Allelic Composition	Ankrd16^tm1.1Slac/Ankrd16^tm1.2Slac En1^tm2(cre)Wrst/En1⁺
Genetic Background	involves: 129S1/Sv * 129X1/SvJ * C57BL/6J

Find Mice

Using the International Mouse Strain Resource (IMSR)
Mouse lines carrying:
Ankrd16^tm1.1Slac mutation (0 available); any Ankrd16 mutation (25 available)
Ankrd16^tm1.2Slac mutation (0 available); any Ankrd16 mutation (25 available)
En1^tm2(cre)Wrst mutation (1 available); any En1 mutation (32 available)

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

nervous system

neuron degeneration ( J:262421 )

• of interneurons in the molecular layer and neurons in the granule cell layer with protein aggregates

• of postnatal cortical and hippocampal neurons

Purkinje cell degeneration ( J:262421 )

• with protein aggregates

Allelic Composition	Pelo^tm1Slac/Pelo^tm1Slac En1^tm2(cre)Wrst/En1⁺
Genetic Background	involves: 129S1/Sv * 129X1/SvJ * C57BL/6J

Find Mice

Using the International Mouse Strain Resource (IMSR)
Mouse lines carrying:
En1^tm2(cre)Wrst mutation (1 available); any En1 mutation (32 available)
Pelo^tm1Slac mutation (0 available); any Pelo mutation (14 available)

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

nervous system

increased neuron apoptosis ( J:306384 )

• increased apoptotic cells in the cerebellum in both the ventricular zone and the prospective white matter

increased neuronal precursor proliferation ( J:306384 )

• in the ventricular zone progenitors at E13.5

abnormal cerebellum external granule cell layer morphology ( J:306384 )

• missing at E13.5 and P0

abnormal brain interneuron morphology ( J:306384 )

• decreased Pax2+ interneurons

decreased inferior colliculus size ( J:306384 )

• grossly hypoplastic being nearly absent

decreased superior colliculus size ( J:306384 )

• grossly hypoplastic being nearly absent

decreased Purkinje cell number ( J:306384 )

• reduced Lhx1/5+ Purkinje cells

cerebellum hypoplasia ( J:306384 )

• grossly being nearly absent

loss of GABAergic neurons ( J:306384 )

• at E13.5 in the cerebellum

cellular

increased neuron apoptosis ( J:306384 )

• increased apoptotic cells in the cerebellum in both the ventricular zone and the prospective white matter

increased neuronal precursor proliferation ( J:306384 )

• in the ventricular zone progenitors at E13.5

Allelic Composition	Wnt1^tm1.1Mze/Wnt1^tm1.1Mze En1^tm2(cre)Wrst/En1⁺
Genetic Background	involves: 129S1/Sv * 129X1/SvJ * C57BL/6N

Find Mice

Using the International Mouse Strain Resource (IMSR)
Mouse lines carrying:
En1^tm2(cre)Wrst mutation (1 available); any En1 mutation (32 available)
Wnt1^tm1.1Mze mutation (0 available); any Wnt1 mutation (30 available)

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

nervous system

decreased rhombomere 1 size ( J:194842 )

• severe deletion of rhombomere 1 at E10.5

abnormal midbrain morphology ( J:194842 )

• the ventral mesencephalon is dysmorphic

decreased midbrain size ( J:194842 )

• severe deletion of the putative mesencephalon at E10.5

embryo

decreased rhombomere 1 size ( J:194842 )

• severe deletion of rhombomere 1 at E10.5

Allelic Composition	Atg7^tm1Tchi/Atg7^tm1Tchi En1^tm2(cre)Wrst/En1⁺
Genetic Background	involves: 129S1/Sv * 129X1/SvJ * C57BL/6NCrlj * CBA/JNCrlj

Find Mice

Using the International Mouse Strain Resource (IMSR)
Mouse lines carrying:
Atg7^tm1Tchi mutation (3 available); any Atg7 mutation (51 available)
En1^tm2(cre)Wrst mutation (1 available); any En1 mutation (32 available)

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

mortality/aging

decreased survivor rate ( J:192452 )

• mice show a 76% reduction in survival by 1 year of age

premature death ( J:192452 )

• mice show a 76% reduction in survival by 1 year of age

growth/size/body

weight loss ( J:192452 )

behavior/neurological

tremors ( J:192452 )

• mice exhibit tremulousness

abnormal locomotor behavior ( J:192452 )

• mice show decreased locomotor activity

ataxia ( J:192452 )

abnormal gait ( J:192452 )

• mice exhibit a wide-based and ataxic gait

homeostasis/metabolism

decreased dopamine level ( J:192452 )

• mice exhibit an age dependent decrease in striatal dopamine content in the brain, with a 64.5% reduction by 7-9 months of age compared to controls

nervous system

abnormal brain morphology ( J:192452 )

• while alpha-synuclein inclusions are not seen, accumulation of low-molecular weight alpha-synuclein is seen in the brain

abnormal substantia nigra pars compacta morphology ( J:192452 )

• 60% loss of substantia nigra pars compacta dopamine neurons by 7-9 months of age

astrocytosis ( J:192452 )

• a modest increase in reactive astrocytes in the of substantia nigra pars compacta

abnormal neuron morphology ( J:192452 )

• neuronal inclusions are present in the substantia nigra pars compacta

• inclusions are often perinuclear but are also in the neuropil and are positive for ubiquitin

• neuronal inclusions are present in juveniles but are smaller in size than at older ages

neuron degeneration ( J:192452 )

• 60% loss of substantia nigra pars compacta dopamine neurons by 7-9 months of age

Purkinje cell degeneration ( J:192452 )

Mouse Models of Human Disease		DO ID	OMIM ID(s)	Ref(s)
Parkinson's disease		DOID:14330	OMIM:PS168600	J:192452

Allelic Composition	Dnm1l^tm1.1Hise/Dnm1l^tm1.1Hise En1^tm2(cre)Wrst/En1⁺
Genetic Background	involves: 129S/SvEv * 129S1/Sv * 129X1/SvJ * C57BL/6 * SJL

Find Mice

Using the International Mouse Strain Resource (IMSR)
Mouse lines carrying:
Dnm1l^tm1.1Hise mutation (0 available); any Dnm1l mutation (43 available)
En1^tm2(cre)Wrst mutation (1 available); any En1 mutation (32 available)

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

Defects in cerebellar development in Dnm1l^tm1.1Hise/Dnm1l^tm1.2Hise En1^tm2(cre)Wrst/En1⁺ and Dnm1l^tm1.2Hise/Dnm1l^tm1.2Hise En1^tm2(cre)Wrst/En1⁺ mice

cellular

abnormal Purkinje cell mitochondrial morphology ( J:153835 )

• mitochondria in Purkinje cells are enlarged compared to in wild-type cells

mortality/aging

postnatal lethality, complete penetrance ( J:153835 )

• mice die within 36 hours of birth

nervous system

abnormal cerebellum development ( J:153835 )

• at P0, cerebellum are smooth and exhibit a 60% decreased in size compared to in wild-type mice

reduced cerebellar foliation ( J:153835 )

• at P0, cerebellum lack foliation

• at P0.5, lobule fissures are barely detectable

abnormal cerebellar Purkinje cell layer ( J:153835 )

• Purkinje cells in the cerebellum are decreased in number and form a discontinuous cell layer unlike in wild-type mice

abnormal Purkinje cell mitochondrial morphology ( J:153835 )

• mitochondria in Purkinje cells are enlarged compared to in wild-type cells

decreased Purkinje cell number ( J:153835 )

• in the cerebellum

small cerebellum ( J:153835 )

• at P0, cerebellum are 60% smaller than in wild-type mice due to decreased cell proliferation

behavior/neurological

absent gastric milk in neonates ( J:153835 )

• mice die without milk in their stomach

• however, mice exhibit normal swallowing when manually fed milk

Allelic Composition	En1^tm2(cre)Wrst/En1^tm8.1Alj En2^tm2Alj/En2^tm2Alj
Genetic Background	involves: 129/Sv * Black Swiss * C57BL/6 * SJL * Swiss Webster

Find Mice

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

nervous system

absent inferior colliculus ( J:134516 )

• absent at E18.5

absent superior colliculus ( J:134516 )

• absent at E18.5

absent colliculi ( J:134516 )

absent tectum ( J:134516 )

absent cerebellum ( J:134516 )

• at E18.5, no cerebellum is present

Allelic Composition	En1^tm2(cre)Wrst/En1^tm8.1Alj En2^tm2Alj/En2⁺
Genetic Background	involves: 129/Sv * Black Swiss * C57BL/6 * SJL * Swiss Webster

Find Mice

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

nervous system

absent inferior colliculus ( J:134516 )

• absent at E18.5

decreased superior colliculus size ( J:134516 )

• reduced in size at E18.5

absent cerebellum ( J:134516 )

• at E18.5, no cerebellum is present

Allelic Composition	En1^tm2(cre)Wrst/En1⁺ Gbx2^tm1.1Mrt/Gbx2^tm1.1Alj
Genetic Background	involves: 129/Sv * C57BL/6 * SJL * Swiss Webster

Find Mice

Using the International Mouse Strain Resource (IMSR)
Mouse lines carrying:
En1^tm2(cre)Wrst mutation (1 available); any En1 mutation (32 available)
Gbx2^tm1.1Alj mutation (1 available); any Gbx2 mutation (27 available)
Gbx2^tm1.1Mrt mutation (0 available); any Gbx2 mutation (27 available)

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

mortality/aging

mortality/aging ( J:79431 )

N	• more than half of the mice survive past weaning, are fertile and nurture their offspring normally

postnatal lethality, incomplete penetrance ( J:79431 )

• some mutants are found dead prior to weaning age

growth/size/body

decreased body weight ( J:79431 )

• surviving mutants weigh less than wild-type littermates

abnormal postnatal growth ( J:79431 )

• surviving mutants (adults) are smaller than littermates

nervous system

nervous system phenotype ( J:79431 )

N	• cerebellar hemisphere development and cytoarchitecture of cerebellum are essentially normal

decreased embryonic neuroepithelium thickness ( J:79431 )

• at E12.5, neuroepithelium of medial cerebellar anlage is thinner than in wild-type with abnormal indents found in the ventricular layer

abnormal cerebellum development ( J:79431 )

• at E10.5, mesencephalon is expanded caudally and alar plate of r1 is significantly reduced

• medial region of cerebellar primordium is reduced in size from E12.5 to 18.5

• increased cell proliferation is observed in indents into ventricular layer, resulting in small cell aggregates in ventricular layer at E14.5; large cell aggregates are observed in medial cerebella of mutants at E18.5

• however, no cell aggregates are seen in cerebella of 8-week old mutants

abnormal cerebellar foliation ( J:79431 )

• foliation pattern is disrupted in adults

• in adult mutants all lobules are reduced in size to varying extents with lobules V and IX less affected

abnormal midbrain development ( J:79431 )

• at E10.5, the mesencephalon is expanded caudally

abnormal midbrain-hindbrain boundary morphology ( J:79431 )

• at E9.5, isthmic constriction dividing the mesencephalon and rhombomere 1 (r1) at dorsal midline is less prominent than in wild-type

increased inferior colliculus size ( J:79431 )

cerebellum vermis hypoplasia ( J:79431 )

decreased embryonic neuroepithelial cell proliferation ( J:79431 )

• proliferation in the medial region of the cerebellum is reduced compared to wild-type

embryo

decreased embryonic neuroepithelium thickness ( J:79431 )

• at E12.5, neuroepithelium of medial cerebellar anlage is thinner than in wild-type with abnormal indents found in the ventricular layer

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