- organ of Corti degeneration / MGI
- abnormal stria vascularis morphology / MGI
- impaired swimming / MGI
- decreased litter size / MGI
- deafness / MGI
- increased B cell derived lymphoma incidence / MGI
- seizures / MGI
- premature death / MGI
- abnormal CNS synaptic transmission / MGI
- abnormal nervous system electrophysiology / MGI
- abnormal spleen germinal center morphology / MGI
- abnormal spleen marginal zone morphology / MGI
- cochlear ganglion degeneration / MGI
- abnormal excitatory postsynaptic currents / MGI
- abnormal cell migration / MGI
- enlarged otoliths / MGI
- decreased otolith number / MGI
- abnormal tectorial membrane morphology / MGI
- abnormal tumor morphology / MGI
- nervous system phenotype / MGI
- increased tumor growth/size / MGI
- tonic-clonic seizures / MGI
- abnormal Rosenthal canal morphology / MGI
- vestibular saccular degeneration / MGI
- cochlear hair cell degeneration / MGI
- thin stria vascularis / MGI
- abnormal strial basal cell morphology / MGI
- abnormal strial marginal cell morphology / MGI
- abnormal strial intermediate cell morphology / MGI
- abnormal stria vascularis vasculature morphology / MGI
- cochlear outer hair cell degeneration / MGI
- abnormal vestibular labyrinth morphology / MGI
- absent distortion product otoacoustic emissions / MGI
- abnormal vestibular system physiology / MGI
- otosclerosis / MGI
- abnormal brain wave pattern / MGI
- increased B cell number / MGI
- increased T cell number / MGI
- increased B cell proliferation / MGI
- head tilt / MGI
- cardiovascular system phenotype / MGI
- abnormal action potential / MGI
- increased angiogenesis / MGI
- detached Reissner membrane / MGI
- absent startle reflex / MGI
- increased lung tumor incidence / MGI
- increased germinal center B cell number / MGI
- small scala media / MGI
- increased spleen germinal center number / MGI
- increased spleen germinal center size / MGI
- decreased B cell apoptosis / MGI
- abnormal tumor vascularization / MGI
- postnatal lethality, incomplete penetrance / MGI
- increased or absent threshold for auditory brainstem response / MGI
C57BL/6N-S1pr2stdf/WtsiOulu
Status | Available to order |
EMMA ID | EM:10854 |
International strain name | C57BL/6N-S1pr2stdf/WtsiOulu |
Alternative name | C57BL/6N Stonedeaf mutation |
Strain type | Spontaneous |
Allele/Transgene symbol | stdf |
Gene/Transgene symbol | S1pr2 |
Information from provider
Provider | Karen Steel |
Provider affiliation | Centre for Age-Related Diseases, King |
Additional owner | Wellcome Trust Sanger Institute |
Genetic information | This mutation is a G>C transition in the S1pr2 gene, resulting in a threonine to arginine change (T289R). |
Phenotypic information | Homozygous:These mutants show an early-onset, rapidly-progressive hearing loss, with Auditory Brainstem Response thresholds increasing from two weeks onwards. No other features were detected.Heterozygous:No detected phenotype in heterozygotes. |
Breeding history | The mutation arose spontaneously during targeting of a separate gene (Mms22l). The frozen embryos carrying the stonedeaf mutation do not carry the targeted Mms22l mutation as this was bred out of the line prior to producing embryos for freezing. |
References |
|
Homozygous fertile | yes |
Homozygous viable | yes |
Homozygous matings required | no |
Immunocompromised | no |
Information from EMMA
Archiving centre | University of Oulu, Oulu, Finland |
Disease and phenotype information
Orphanet associated rare diseases, based on orthologous gene matching
- Autosomal recessive non-syndromic sensorineural deafness type DFNB / Orphanet_90636
MGI phenotypes (gene matching)
Literature references
- Identification and characterisation of spontaneous mutations causing deafness from a targeted knockout programme.;Lewis Morag A, Ingham Neil J, Chen Jing, Pearson Selina, Di Domenico Francesca, Rekhi Sohinder, Allen Rochelle, Drake Matthew, Willaert Annelore, Rook Victoria, Pass Johanna, Keane Thomas, Adams David J, Tucker Abigail S, White Jacqueline K, Steel Karen P, ;2022;BMC biology;20;67; 35296311
- S1PR2 variants associated with auditory function in humans and endocochlear potential decline in mouse.;Ingham Neil J, Carlisle Francesca, Pearson Selina, Lewis Morag A, Buniello Annalisa, Chen Jing, Isaacson Rivka L, Pass Johanna, White Jacqueline K, Dawson Sally J, Steel Karen P, ;2016;Scientific reports;6;28964; 27383011
Information on how we integrate external resources can be found here
INFRAFRONTIER® and European Mouse Mutant Archive - EMMA® are registered trademarks at the European Union Intellectual Property Office (EUIPO).