Description

Strain Information

Type Mutant Stock; Additional information on Genetically Engineered Mutant Mice. Species laboratory mouse Generation F28p

Appearance
agouti, spins
Related Genotype: A/A Myo15^sh2/Myo15^sh2

agouti, unaffected
Related Genotype: A/A Myo15^sh2/+

Description
Mice homozygous for the shaker 2 spontaneous mutation (Myo15^sh2) are viable and fertile, showing only a slight reduction in both compared to wildtype mice. Homozygous mutant mice display a phenotype very similar to the behavior and pathology to shaker-1 (Myo7a^sh1) with the exception that the abnormalities are observed a little earlier in shaker 2 mice. Homozygotes appear to be deaf from the beginning, and the saccular macula is abnormal at birth. The stria vascularis appears normal by light microscopy at 2 weeks, but begins to show degenerative changes shortly thereafter. Many of the cells contain electron-dense inclusions filled with a fine granular material. At 2.5 months, the type I hair cells of the cristae ampullares and maculae utriculi contain rod-shaped inclusion bodies composed of actin filaments. The visible phenotype of the shaker-2 Jackson (Myo15^sh2-J, Stock No. 002048) does not differ from that of the original shaker 2.

Control Information

	Control
	Heterozygote from the colony
Considerations for Choosing Controls

Related Strains

Strains carrying other alleles of Myo15

002048	B6 x C57BLKS-m Leprdb Myo15sh2-J/J
003304	STOCK Rb(16.17)7Bnr-Myo15sh2-2J/J

View Strains carrying other alleles of Myo15     (2 strains)

Phenotype

Phenotype Information

View Mammalian Phenotype Terms

Mammalian Phenotype Terms

      assigned by genotype

Myo15^sh2/Myo15^sh2

        Mixed stock

• hearing/vestibular/ear phenotype
• abnormal cochlear hair cell stereociliary bundle morphology (MGI Ref ID J:132633)
  • hair cell stereocilia are shorter than in wild-type mice
• nervous system phenotype
• abnormal cochlear hair cell stereociliary bundle morphology (MGI Ref ID J:132633)
  • hair cell stereocilia are shorter than in wild-type mice

View Research Applications

Research Applications

This mouse can be used to support research in many areas including:

Myo15^sh2 related

Mouse/Human Gene Homologs
deafness, neurosensory, autosomal recessive, 3

Neurobiology Research
Tremor Defects
Vestibular and Hearing Defects

Sensorineural Research
Vestibular and Hearing Defects

Genes & Alleles

Gene & Allele Information

Allele Symbol		Myo15sh2
Allele Name		shaker 2
Allele Type		Radiation induced
Common Name(s)		sh2;
Strain of Origin		Mixed stock
Gene Symbol and Name		Myo15, myosin XV
Chromosome		11
Gene Common Name(s)		DFNB3; DKFZp686N18198; FLJ17274; FLJ31311; RGD1561873; sh-2; sh2; shaker 2;
General Note		The Myo15sh2 mutation was discovered by Dobrovolskaia-Zavadskaia (J:15021) in descendants of an irradiated male. Viability and fertility are nearly normal. This mutant is very similar in behavior and pathology to Myo7ash1, with the exception that the abnormalities are observed a little earlier in Myo15sh2. Homozygotes appear to be deaf from the beginning, and the saccular macula is abnormal at birth (J:13046). The stria vascularis appears normal by light microscopy at 2 weeks of age, but begins to show degenerative changes shortly thereafter. Many of the cells contain electron-dense inclusions filled with a fine granular material (J:6781). At 2.5 months old, the type I hair cells of the cristae ampullares and maculae utriculi contain rod-shaped inclusion bodies composed of actin filaments (J:6873).Myo15sh2 was shown by complementation tests to be at a different locus from Myo7ash1 (J:28674), and linkage with Egfr on Chr 11 was established (J:303). Detailed linkage and physical maps of the Myo15sh2 region of Chr 11 have been constructed (J:40351).

Genotyping

Genotyping Information

This strain will not have a genotyping protocol or one is not currently available.

Helpful Links

Optimizing PCR Protocols

References

References

Additional References

Anderson DW; Probst FJ; Belyantseva IA; Fridell RA; Beyer L; Martin DM; Wu D; Kachar B; Friedman TB; Raphael Y; Camper SA. 2000. The motor and tail regions of myosin XV are critical for normal structure and function of auditory and vestibular hair cells Hum Mol Genet 9(12):1729-38. [PubMed: 10915760]  [MGI Ref ID J:63740]

Anniko M. 1982. Specific pathology of the stria vascularis in postnatal progressive genetic inner ear disorder. Hear Res 6(3):247-58. [PubMed: 6979536]  [MGI Ref ID J:6781]

Belyantseva IA; Boger ET; Friedman TB. 2003. Myosin XVa localizes to the tips of inner ear sensory cell stereocilia and is essential for staircase formation of the hair bundle. Proc Natl Acad Sci U S A 100(24):13958-63. [PubMed: 14610277]  [MGI Ref ID J:92621]

Erichsen S; Mikkola M; Sahlin L; Hultcrantz M. 2001. Cochlear distribution of Na,K-ATPase and corticosteroid receptors in two mouse strains with congenital hearing disorders. Acta Otolaryngol 121(7):794-802. [PubMed: 11718241]  [MGI Ref ID J:92589]

Lee DJ; Cahill HB; Ryugo DK. 2003. Effects of congenital deafness in the cochlear nuclei of Shaker-2 mice: an ultrastructural analysis of synapse morphology in the endbulbs of Held. J Neurocytol 32(3):229-43. [PubMed: 14724386]  [MGI Ref ID J:89801]

Probst FJ; Fridell RA; Raphael Y; Saunders TL; Wang A; Liang Y ; Morell RJ ; Touchman JW ; Lyons RH ; Noben-Trauth K ; Friedman TB ; Camper SA. 1998. Correction of deafness in shaker-2 mice by an unconventional myosin in a BAC transgene [see comments] Science 280(5368):1444-7. [PubMed: 9603735]  [MGI Ref ID J:48001]

Myo15^sh2 related

Anderson DW; Probst FJ; Belyantseva IA; Fridell RA; Beyer L; Martin DM; Wu D; Kachar B; Friedman TB; Raphael Y; Camper SA. 2000. The motor and tail regions of myosin XV are critical for normal structure and function of auditory and vestibular hair cells Hum Mol Genet 9(12):1729-38. [PubMed: 10915760]  [MGI Ref ID J:63740]

Anniko M. 1982. Specific pathology of the stria vascularis in postnatal progressive genetic inner ear disorder. Hear Res 6(3):247-58. [PubMed: 6979536]  [MGI Ref ID J:6781]

Belyantseva IA; Boger ET; Friedman TB. 2003. Myosin XVa localizes to the tips of inner ear sensory cell stereocilia and is essential for staircase formation of the hair bundle. Proc Natl Acad Sci U S A 100(24):13958-63. [PubMed: 14610277]  [MGI Ref ID J:92621]

Belyantseva IA; Boger ET; Naz S; Frolenkov GI; Sellers JR; Ahmed ZM; Griffith AJ; Friedman TB. 2005. Myosin-XVa is required for tip localization of whirlin and differential elongation of hair-cell stereocilia. Nat Cell Biol 7(2):148-56. [PubMed: 15654330]  [MGI Ref ID J:102099]

Beyer LA; Odeh H; Probst FJ; Lambert EH; Dolan DF; Camper SA; Kohrman DC; Raphael Y. 2000. Hair cells in the inner ear of the pirouette and shaker 2 mutant mice. J Neurocytol 29(4):227-40. [PubMed: 11276175]  [MGI Ref ID J:94385]

Bossi G; Booth S; Clark R; Davis EG; Liesner R; Richards K; Starcevic M; Stinchcombe J; Trambas C; Dell'Angelica EC; Griffiths GM. 2005. Normal lytic granule secretion by cytotoxic T lymphocytes deficient in BLOC-1, -2 and -3 and myosins Va, VIIa and XV. Traffic 6(3):243-51. [PubMed: 15702992]  [MGI Ref ID J:105404]

CARTER TC; PHILLIPS RJ. 1953. The sex distribution of waved-2, shaker-2 and Rex in the house mouse. Z Indukt Abstamm Vererbungsl 85(4):564-78. [PubMed: 13170359]  [MGI Ref ID J:225]

Calderon A; Derr A; Stagner BB; Johnson KR; Martin G; Noben-Trauth K. 2006. Cochlear developmental defect and background-dependent hearing thresholds in the Jackson circler (jc) mutant mouse. Hear Res 221(1-2):44-58. [PubMed: 16962269]  [MGI Ref ID J:113021]

Clark FH. 1935. Linkage relations of Zavadskaia Shaker in the house mouse (Mus musculus) Proc Natl Acad Sci U S A 21(5):247-51. [PubMed: 16577664]  [MGI Ref ID J:28674]

Deol MS. 1954. The anomalies of the labyrinth of the mutants varitint-waddler, shaker-2 and jerker in the mouse. J Genet 52:562-588.  [MGI Ref ID J:13046]

Dobrovolskaia-Zavasdkaia N. 1928. L'irradiation des testicules et l'heredite chez la souris Arch Biol (Liege) 38:457-501.  [MGI Ref ID J:15021]

Erichsen S; Mikkola M; Sahlin L; Hultcrantz M. 2001. Cochlear distribution of Na,K-ATPase and corticosteroid receptors in two mouse strains with congenital hearing disorders. Acta Otolaryngol 121(7):794-802. [PubMed: 11718241]  [MGI Ref ID J:92589]

KOCHER W. 1960. [Studies on the genetics and pathology of the development of 8 labyrinth mutants (deaf-waltzer-shaker mutants) in the mouse (Mus musculus).] Z Vererbungsl 91:114-40. [PubMed: 13853422]  [MGI Ref ID J:15164]

Kanzaki S; Beyer L; Karolyi IJ; Dolan DF; Fang Q; Probst FJ; Camper SA; Raphael Y. 2006. Transgene correction maintains normal cochlear structure and function in 6-month-old Myo15a mutant mice. Hear Res 214(1-2):37-44. [PubMed: 16580798]  [MGI Ref ID J:108202]

Karolyi IJ; Probst FJ; Beyer L; Odeh H; Dootz G; Cha KB; Martin DM; Avraham KB; Kohrman D; Dolan DF; Raphael Y; Camper SA. 2003. Myo15 function is distinct from Myo6, Myo7a and pirouette genes in development of cochlear stereocilia. Hum Mol Genet 12(21):2797-805. [PubMed: 12966030]  [MGI Ref ID J:86379]

Kikkawa Y; Mburu P; Morse S; Kominami R; Townsend S; Brown SD. 2005. Mutant analysis reveals whirlin as a dynamic organizer in the growing hair cell stereocilium. Hum Mol Genet 14(3):391-400. [PubMed: 15590699]  [MGI Ref ID J:95916]

Lee DJ; Cahill HB; Ryugo DK. 2003. Effects of congenital deafness in the cochlear nuclei of Shaker-2 mice: an ultrastructural analysis of synapse morphology in the endbulbs of Held. J Neurocytol 32(3):229-43. [PubMed: 14724386]  [MGI Ref ID J:89801]

Mburu P; Kikkawa Y; Townsend S; Romero R; Yonekawa H; Brown SD. 2006. Whirlin complexes with p55 at the stereocilia tip during hair cell development. Proc Natl Acad Sci U S A 103(29):10973-8. [PubMed: 16829577]  [MGI Ref ID J:111803]

Probst FJ; Fridell RA; Raphael Y; Saunders TL; Wang A; Liang Y ; Morell RJ ; Touchman JW ; Lyons RH ; Noben-Trauth K ; Friedman TB ; Camper SA. 1998. Correction of deafness in shaker-2 mice by an unconventional myosin in a BAC transgene [see comments] Science 280(5368):1444-7. [PubMed: 9603735]  [MGI Ref ID J:48001]

Prosser HM; Rzadzinska AK; Steel KP; Bradley A. 2008. Mosaic complementation demonstrates a regulatory role for myosin VIIa in actin dynamics of stereocilia. Mol Cell Biol 28(5):1702-12. [PubMed: 18160714]  [MGI Ref ID J:132633]

Snell GD; Law LW. 1939. A linkage between shaker-2 and wavy-2 in the house mouse J Hered 30:447.  [MGI Ref ID J:303]

Sobin A; Anniko M; Flock A. 1982. Rods of actin filaments in type I hair cells of the Shaker-2 mouse. Arch Otorhinolaryngol 236(1):1-6. [PubMed: 6889851]  [MGI Ref ID J:6873]

Wakabayashi Y; Kikkawa Y; Matsumoto Y; Shinbo T; Kosugi S; Chou D; Furuya M; Jishage K; Noda T; Yonekawa H; Kominami R. 1997. Genetic and physical delineation of the region of the mouse deafness mutation shaker-2. Biochem Biophys Res Commun 234(1):107-10. [PubMed: 9168970]  [MGI Ref ID J:40351]

Wakabayashi Y; Takahashi Y; Kikkawa Y; Okano H; Mishima Y; Ushiki T ; Yonekawa H ; Kominami R. 1998. A novel type of myosin encoded by the mouse deafness gene shaker-2. Biochem Biophys Res Commun 248(3):655-9. [PubMed: 9703981]  [MGI Ref ID J:49063]

Webster DB; Sobin A; Anniko M. 1986. Incomplete maturation of brainstem auditory nuclei in genetically induced early postnatal cochlear degeneration. Acta Otolaryngol 101(5-6):429-38. [PubMed: 3727977]  [MGI Ref ID J:94384]

Wright ME. 1947. Two sex-linkages in the house mouse, with unusual recombination values Heredity 1:349-54.  [MGI Ref ID J:313]

Health & husbandry

Health & Colony Maintenance Information

Colony Maintenance

Breeding & Husbandry	Myo15sh2/Myo15sh2 males may trample pups due to circling behavior; remove the male from the cage when the female is pregnant.

Purchasing information

Pricing, Supply Level & Notes, Controls, General Terms & Conditions

Pricing

Supply Details

Standard Supply

Repository-Cryopreserved. Must Be Recovered. Please refer to pricing and supply notes for further information.

Supply Notes

Cryorecovery - Standard. The recovery process begins when a signed agreement form is returned to the Customer Service Department after order placement. Although results vary by strain, at least two males and two females (two pairs) will be provided, typically within 15 weeks of our receipt of the signed agreement form. If the first recovery attempt is unsuccessful or only one pair is recovered, a second recovery will be done, extending the delivery time to approximately 25 weeks. At least one member of each pair will be of known genotype and will carry the mutation if it is a mutant strain. Please note that pairs may not reflect the mating scheme utilized by The Jackson Laboratory prior to cryopreservation of the strain. Mating schemes are sometimes modified for successful cryopreservation. Price represents a repository maintenance fee, which includes the cost of recovery of the strain from the cryopreservation resource and the periodic replacement of the frozen embryos used for recovery. Cryorecovery to establish a Dedicated Supply for greater quantities of mice. One to two pairs will be recovered to establish a Dedicated Supply of mice. Price by quotation. For more information on Dedicated Supply, please contact JAX® Services, Tel: 1-800-422-6423 or 1-207-288-5845. This strain is included in the Mouse Mutant Resource collection. Genomic DNA is available for this strain from the Mouse DNA Resource.

Control Information

	Control
	Heterozygote from the colony
Considerations for Choosing Controls
USA, Canada and Mexico - Control Pricing Information for Genetically Engineered Mutant Strains.
International - Control Pricing Information for Genetically Engineered Mutant Strains.

General Terms and Conditions

See Terms of Use

The Jackson Laboratory's Genotype Promise

The Jackson Laboratory has rigorous genetic quality control and mutant gene genotyping programs to ensure the genetic background of JAX^® Mice strains as well as the genotypes of strains with identified molecular mutations. JAX^® Mice strains are only made available to researchers after meeting our standards. However, the phenotype of each strain may not be fully characterized and/or captured in the strain data sheets. Therefore, we cannot guarantee a strain's phenotype will meet all expectations. To ensure that JAX^® Mice will meet the needs of individual research projects or when requesting a strain that is new to your research, we suggest ordering and performing tests on a small number of mice to determine suitability for your particular project.

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Ordering and Purchasing Information

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Fax: 207.288.6150
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Terms of Use

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General Terms and Conditions

Contact information

General inquiries

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phone:	207-288-6470
fax:	207-288-6655

JAX^® Mice & Services Conditions of Use

“Each recipient institution, including its employees and other researchers under its control (RECIPIENT), of mice or services using mice from The Jackson Laboratory (TJL) agrees that such mice, descendants of those mice derived by inbreeding or crossbreeding, including unmodified derivatives of those mice or their descendants (“MICE”) shall not be: (i) used for any purpose other than the internal research of the RECIPIENT, (ii) sold or otherwise provided to any third party for any use, or (iii) provided to any agent or other third party to provide breeding or other services with respect to MICE. Acceptance of MICE from TJL shall be deemed agreement by RECIPIENT to these conditions, and departure from these conditions requires The Jackson Laboratory’s prior written authorization.”

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