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- Description
- Phenotype
- Genes & alleles
- Genotyping
- Health & husbandry
- References
- Purchasing information
- Terms of Use
Description
The genotypes of the animals provided may not reflect those discussed in the strain description or the mating scheme utilized by The Jackson Laboratory prior to cryopreservation. Please inquire for possible genotypes for this specific strain.
Strain Information
Former Names B6CBACa-Aw-J/A hy3/+ (Changed: 15-DEC-04 ) B6CBACa-Aw-J/A-hy3/+ (Changed: 15-DEC-04 ) Type Mutant Strain; Spontaneous Mutation; Additional information on Genetically Engineered and Mutant Mice. Visit our online Nomenclature tutorial. Species laboratory mouse Generation N2F2 Appearance
white-bellied agouti
Related Genotype: Aw-J/?
agouti
Related Genotype: A/ADescription
Mice homozygous for the hydrocephalus 3 spontaneous mutation (Hydinhy3) are usually identifiable at 3 to 5 days. Those with frank hydrocephalus die by 4 or 5 weeks of age. The lateral ventricles and the third ventricle are enlarged, the aqueduct of Sylvius and the fourth ventricle are only slightly affected, and there is some dilatation of the ventral subarachnoid cistern. The hydrocephalus seems to be due to a defect in the subarachnoid space under the calvarium caused by an abnormal postnatal differentiation of the arachnoid mater and pia mater which prevents their separation. Penetrance is incomplete.Development
Hydrocephalus 3 (Hydinhy3) arose spontaneously in a heterogeneous stock of laboratory mice (Gruneberg, H.1943.). The mutation was received at The Jackson Laboratory by Dr. M.C. Green from Dr. Gruneberg in 1963. The strain was brother by sister bred by test matings and was at F76 in 1983. In 1995 the inbred strain stopped breeding and an outcross was made to B6CBACaF1 Aw/A. Tested males were then continuously backcrossed to the hybrid. In 1996 untyped +/? Females were bred with proven heterozygous males to generate embryos for cryopreservation.
Control Information
| Control | ||
|---|---|---|
| Untyped from the colony | ||
| Considerations for Choosing Controls | ||
Related Strains
Strains carrying Aw-J allele
View Strains carrying Aw-J (31 strains)
008343 FVB.Cg-Hydinhy3/MlrJ
Phenotype
Phenotype Information
assigned by genotype
The following phenotype information may relate to a genetic background differing from this JAX® Mice strain.
Hydinhy3/Hydinhy3
involves: CBA
- life span-post-weaning/aging
- premature death (MGI Ref ID J:13035)
- usually die before 2 months of age, generally around 4-6 week
- craniofacial phenotype
- abnormal skull morphology (MGI Ref ID J:5426)
- thin skull
- abnormal cranial suture morphology (MGI Ref ID J:5426)
- open sutures
- domed skull (MGI Ref ID J:13035)
- develops after the first week of life
- enlarged parietal bone (MGI Ref ID J:13103)
- enlarged skull (MGI Ref ID J:13035)
- skull volume doubled at birth, 0.88cc vs 0.44cc
- does not develop before 3-5 days after birth or possible the 2nd week of life
- nervous system phenotype
- abnormal brain meninges morphology (MGI Ref ID J:13103)
- calvarium tends to adhere more tightly to brain
- reduction of meningeal layers at 8 days of age
- swelling of cells in meninges in the area of the foramina of Luschka and the otic capsules
- abnormal cerebral aqueduct (MGI Ref ID J:5377)
- aqueduct of Sylvius occluded around 16 days of age
- abnormal ependyma morphology (MGI Ref ID J:5426)
- flattened cells in the ependymal lining
- abnormal telencephalon morphology (MGI Ref ID J:5377)
- enlargement of extracellular spaces in the white matter after occlusion of the aqueduct of Sylvius
- abnormal lateral ventricle morphology (MGI Ref ID J:13103)
- foramina of Monro wider than normal
- dilated lateral ventricles (MGI Ref ID J:13035)
- enormously dilated at birth
- thinning of pallium but basal ganglia less affected
- not apparent before 3-5 days of age
- thin cerebral cortex (MGI Ref ID J:5426)
- cerebral cortex smooth and very thin
- hydroencephaly (MGI Ref ID J:13035)
- growth/size phenotype
- cachexia (MGI Ref ID J:13035)
- eventually loose weight
- in an emaciated condition at the time of death
- postnatal growth retardation (MGI Ref ID J:13035)
- general growth retardation
- never reach normal size
- behavior/neurological phenotype
- abnormal aggression-related behavior (MGI Ref ID J:5426)
- episodes of aggressive behavior
- abnormal fear/anxiety-related behavior (MGI Ref ID J:5426)
- "cowering" behavior observed at 21 days of age
- ataxia (MGI Ref ID J:5426)
- hypoactivity (MGI Ref ID J:13035)
- become inactive as they age
- reproductive system phenotype
- male infertility (MGI Ref ID J:13035)
- probably (N=1)
- muscle phenotype
- muscle spasm (MGI Ref ID J:13103)
- develop spasticity of the hind limbs
- respiratory system phenotype
- abnormal respiratory system physiology (MGI Ref ID J:13035)
- nasal discharge composed of squamous epithelia and polymorphonuclear leukocytes
- nasal discharge not seen in later studies
- skeleton phenotype
- abnormal skull morphology (MGI Ref ID J:5426)
- thin skull
- abnormal cranial suture morphology (MGI Ref ID J:5426)
- open sutures
- domed skull (MGI Ref ID J:13035)
- develops after the first week of life
- enlarged parietal bone (MGI Ref ID J:13103)
- enlarged skull (MGI Ref ID J:13035)
- skull volume doubled at birth, 0.88cc vs 0.44cc
- does not develop before 3-5 days after birth or possible the 2nd week of life
- kyphosis (MGI Ref ID J:5426)
Hydinhy3/Hydinhy3
involves: FVB/N
- lethality-postnatal
- postnatal lethality (MGI Ref ID J:135322)
- mice rarely survive beyond 3 weeks of age
- nervous system phenotype
- abnormal brainstem morphology (MGI Ref ID J:135322)
- small
- abnormal ependyma morphology (MGI Ref ID J:135322)
- in the central axoneme apparatus of the ependymal cilia, the C2b projection is absent, the C1b projection is displaced or altered in shape and the C2c projection is reduced or absent compared to in wild-type cilia
- hydroencephaly (MGI Ref ID J:135322)
- hydroencephaly is associated with defective ependymal cilia bending, delayed switching between forward and backward motion, lower beat frequency, and impaired cilia-generated flow
- small cerebellum (MGI Ref ID J:135322)
- small olfactory bulb (MGI Ref ID J:135322)
- respiratory system phenotype
- abnormal mucociliary clearance (MGI Ref ID J:135322)
- tracheal cilia beating is impaired with delayed switching between forward and backward motion, lower beat frequency, and impaired cilia-generated flow
- abnormal tracheal ciliated epithelium morphology (MGI Ref ID J:135322)
- in the central axoneme apparatus of the tracheal cilia, the C2b projection is absent and the C1b and C2c projections are displaced or altered in compared to in wild-type cilia
- skeleton phenotype
- domed skull (MGI Ref ID J:135322)
- cardiovascular system phenotype
- hemorrhage (MGI Ref ID J:135322)
- some mice exhibit severe hemorrhage below the skull
- growth/size phenotype
- postnatal growth retardation (MGI Ref ID J:135322)
- at P4
- craniofacial phenotype
- domed skull (MGI Ref ID J:135322)
This mouse can be used to support research in many areas including:Hydinhy3 related
Developmental Biology Research
Neurodevelopmental Defects
Neurobiology Research
Neurodevelopmental Defects
Genes & Alleles
Gene & Allele Information
| Allele Symbol | Aw-J | ||
|---|---|---|---|
| Allele Name | white bellied agouti Jackson | ||
| Allele Type | Spontaneous | ||
| Common Name(s) | AWJ; | ||
| Strain of Origin | C57BL/6J | ||
| Gene Symbol and Name | a, nonagouti | ||
| Chromosome | 2 | ||
| Gene Common Name(s) | AGSW; AGTI; AGTIL; ASP; As; MGC126092; MGC126093; SHEP9; agouti; agouti signal protein; agouti suppressor; | ||
| Allele Symbol | Hydinhy3 | ||
| Allele Name | hydrocephalus 3 | ||
| Allele Type | Spontaneous | ||
| Common Name(s) | hy-3; | ||
| Gene Symbol and Name | Hydin, hydrocephalus inducing | ||
| Chromosome | 8 | ||
| Gene Common Name(s) | 1700034M11Rik; 4930545D19Rik; AC069308.21, gene model 4; AC069308.21gm4; DKFZp434D0513; DKFZp434L0850; FLJ12871; FLJ14665; HYDIN1; KIAA1864; RIKEN cDNA 1700034M11 gene; RIKEN cDNA 4930545D19 gene; hy-3; hy3; hydrocephalus 3; | ||
| Molecular Note | Matings between mice carrying a transgenic insertion allele in this gene and hy3 mice demonstrated that these two mutations fail to complement each other, and suggests that these two mutations are allelic and very likely result from disrupted function ofthe same gene or set of genes. [MGI Ref ID J:83437] | ||
Genotyping
Genotyping Information
Genotyping Protocols
Hydinhy3, Pyrosequencing
Helpful Links
Genotyping resources and troubleshooting
References
References
Additional References
Davy BE; Robinson ML. 2003. Congenital hydrocephalus in hy3 mice is caused by a frameshift mutation in Hydin, a large novel gene. Hum Mol Genet 12(10):1163-70. [PubMed: 12719380] [MGI Ref ID J:83437]
Robinson ML; Allen CE; Davy BE; Durfee WJ; Elder FF; Elliott CS; Harrison WR. 2002. Genetic mapping of an insertional hydrocephalus-inducing mutation allelic to hy3. Mamm Genome 13(11):625-32. [PubMed: 12461648] [MGI Ref ID J:80595]
Aw-J relatedHydinhy3 relatedAberg T; Wang XP; Kim JH; Yamashiro T; Bei M; Rice R; Ryoo HM; Thesleff I. 2004. Runx2 mediates FGF signaling from epithelium to mesenchyme during tooth morphogenesis. Dev Biol 270(1):76-93. [PubMed: 15136142] [MGI Ref ID J:92174]
Barsh GS; Epstein CJ. 1989. Physical and genetic characterization of a 75-kilobase deletion associated with al, a recessive lethal allele at the mouse agouti locus. Genetics 121(4):811-8. [PubMed: 2566558] [MGI Ref ID J:9799]
Baurle J; Vogten H; Grusser-Cornehls U. 1998. Course and targets of the calbindin D-28k subpopulation of primary vestibular afferents. J Comp Neurol 402(1):111-28. [PubMed: 9831049] [MGI Ref ID J:118430]
Boran T; Lesot H; Peterka M; Peterkova R. 2005. Increased apoptosis during morphogenesis of the lower cheek teeth in tabby/EDA mice. J Dent Res 84(3):228-33. [PubMed: 15723861] [MGI Ref ID J:112546]
Chinta SJ; Rane A; Yadava N; Andersen JK; Nicholls DG; Polster BM. 2009. Reactive oxygen species regulation by AIF- and complex I-depleted brain mitochondria. Free Radic Biol Med 46(7):939-47. [PubMed: 19280713] [MGI Ref ID J:145908]
Cui CY; Hashimoto T; Grivennikov SI; Piao Y; Nedospasov SA; Schlessinger D. 2006. Ectodysplasin regulates the lymphotoxin-beta pathway for hair differentiation. Proc Natl Acad Sci U S A 103(24):9142-7. [PubMed: 16738056] [MGI Ref ID J:111051]
Cui CY; Kunisada M; Esibizione D; Grivennikov SI; Piao Y; Nedospasov SA; Schlessinger D. 2007. Lymphotoxin-beta regulates periderm differentiation during embryonic skin development. Hum Mol Genet 16(21):2583-90. [PubMed: 17673451] [MGI Ref ID J:129949]
Dickie MM. 1969. Mutations at the agouti locus in the mouse. J Hered 60(1):20-5. [PubMed: 5798139] [MGI Ref ID J:30922]
Esibizione D; Cui CY; Schlessinger D. 2008. Candidate EDA targets revealed by expression profiling of primary keratinocytes from Tabby mutant mice. Gene 427(1-2):42-6. [PubMed: 18848976] [MGI Ref ID J:143603]
Granholm DE; Reese RN; Granholm NH. 1996. Agouti alleles alter cysteine and glutathione concentrations in hair follicles and serum of mice (A y/a, A wJ/A wJ, and a/a). J Invest Dermatol 106(3):559-63. [PubMed: 8648194] [MGI Ref ID J:32132]
Granholm DE; Reese RN; Granholm NH. 1995. Agouti alleles influence thiol concentrations in hair follicles and extrafollicular tissues of mice (Ay/a, AwJ/AwJ, a/a). Pigment Cell Res 8(6):302-6. [PubMed: 8789738] [MGI Ref ID J:31403]
Jones JM; Huang JD; Mermall V; Hamilton BA; Mooseker MS; Escayg A; Copeland NG; Jenkins NA; Meisler MH. 2000. The mouse neurological mutant flailer expresses a novel hybrid gene derived by exon shuffling between Gnb5 and Myo5a. Hum Mol Genet 9(5):821-8. [PubMed: 10749990] [MGI Ref ID J:61324]
Kappenman KE; Dvoracek MA; Harvison GA; Fuller BB; Granholm NH. 1992. Tyrosinase abundance and activity in murine hairbulb melanocytes of agouti mutants (C57BL/6J-a/a, Ay/a, and AwJ/AwJ). Pigment Cell Res Suppl 2:79-83. [PubMed: 1409442] [MGI Ref ID J:1295]
Katoh A; Yoshida T; Himeshima Y; Mishina M; Hirano T. 2005. Defective control and adaptation of reflex eye movements in mutant mice deficient in either the glutamate receptor delta2 subunit or Purkinje cells. Eur J Neurosci 21(5):1315-26. [PubMed: 15813941] [MGI Ref ID J:101081]
Knapp PE; Adjan VV; Hauser KF. 2009. Cell-specific loss of kappa-opioid receptors in oligodendrocytes of the dysmyelinating jimpy mouse. Neurosci Lett 451(2):114-8. [PubMed: 19110031] [MGI Ref ID J:146365]
Lee M; Kim A; Chua SC Jr; Obici S; Wardlaw SL. 2007. Transgenic MSH overexpression attenuates the metabolic effects of a high-fat diet. Am J Physiol Endocrinol Metab 293(1):E121-31. [PubMed: 17374695] [MGI Ref ID J:126508]
Lu W; Tsirka SE. 2002. Partial rescue of neural apoptosis in the Lurcher mutant mouse through elimination of tissue plasminogen activator. Development 129(8):2043-50. [PubMed: 11934869] [MGI Ref ID J:111363]
Mayer TC; Fishbane JL. 1972. Mesoderm-ectoderm interaction in the production of the agouti pigmentation pattern in mice. Genetics 71(2):297-303. [PubMed: 4558326] [MGI Ref ID J:5288]
Mitsumori K; Yasuhara K; Mori I; Hayashi S; Shimo T; Onodera H; Nomura T; Hayashi Y. 1998. Pulmonary fibrosis caused by N-methyl-N-nitrosourethane inhibits lung tumorigenesis by urethane in transgenic mice carrying the human prototype c-Ha-ras gene. Cancer Lett 129(2):181-90. [PubMed: 9719460] [MGI Ref ID J:52138]
Monroe DG; Wipf LP; Diggins MR; Matthees DP; Granholm NH. 1998. Agouti-related maturation and tissue distribution of alpha-Melanocyte Stimulating Hormone in wild-type (AwJ/AwJ) and mutant (Ay/a,a/a) mice. Pigment Cell Res 11(5):310-3. [PubMed: 9877102] [MGI Ref ID J:52183]
Mullen RJ. 1974. A<w-J> - white-bellied agouti-J Mouse News Lett 50:38. [MGI Ref ID J:64104]
Mustonen T; Ilmonen M; Pummila M; Kangas AT; Laurikkala J; Jaatinen R; Pispa J; Gaide O; Schneider P; Thesleff I; Mikkola ML. 2004. Ectodysplasin A1 promotes placodal cell fate during early morphogenesis of ectodermal appendages. Development 131(20):4907-19. [PubMed: 15371307] [MGI Ref ID J:128256]
O'donnell SM; Hansberger MW; Connolly JL; Chappell JD; Watson MJ; Pierce JM; Wetzel JD; Han W; Barton ES; Forrest JC; Valyi-Nagy T; Yull FE; Blackwell TS; Rottman JN; Sherry B; Dermody TS. 2005. Organ-specific roles for transcription factor NF-kappaB in reovirus-induced apoptosis and disease. J Clin Invest 115(9):2341-2350. [PubMed: 16100570] [MGI Ref ID J:100906]
Peng J; Wu Z; Wu Y; Hsu M; Stevenson FF; Boonplueang R; Roffler-Tarlov SK; Andersen JK. 2002. Inhibition of caspases protects cerebellar granule cells of the weaver mouse from apoptosis and improves behavioral phenotype. J Biol Chem 277(46):44285-91. [PubMed: 12221097] [MGI Ref ID J:119427]
Peng J; Xie L; Stevenson FF; Melov S; Di Monte DA; Andersen JK. 2006. Nigrostriatal dopaminergic neurodegeneration in the weaver mouse is mediated via neuroinflammation and alleviated by minocycline administration. J Neurosci 26(45):11644-51. [PubMed: 17093086] [MGI Ref ID J:114943]
Poole TW. 1975. Dermal-epidermal interactions and the action of alleles at the agouti locus in the mouse. Dev Biol 42(2):203-10. [PubMed: 1090472] [MGI Ref ID J:5519]
Probst FJ; Cooper ML; Cheung SW; Justice MJ. 2008. Genotype, phenotype, and karyotype correlation in the XO mouse model of Turner Syndrome. J Hered 99(5):512-7. [PubMed: 18499648] [MGI Ref ID J:138994]
Smith DE; Xu SG. 2003. Ultrastructural organization of GABA-like immunoreactive profiles in the weaver substantia nigra. J Neurocytol 32(3):293-303. [PubMed: 14724391] [MGI Ref ID J:121345]
Vandenput L; Swinnen JV; Boonen S; Van Herck E; Erben RG; Bouillon R; Vanderschueren D. 2004. Role of the androgen receptor in skeletal homeostasis: the androgen-resistant testicular feminized male mouse model. J Bone Miner Res 19(9):1462-70. [PubMed: 15312246] [MGI Ref ID J:111491]
Wu Q; Miller RH; Ransohoff RM; Robinson S; Bu J; Nishiyama A. 2000. Elevated levels of the chemokine GRO-1 correlate with elevated oligodendrocyte progenitor proliferation in the jimpy mutant. J Neurosci 20(7):2609-17. [PubMed: 10729341] [MGI Ref ID J:109469]
Yamago G; Takata Y; Furuta I; Urase K; Momoi T; Huh N. 2001. Suppression of hair follicle development inhibits induction of sonic hedgehog, patched, and patched-2 in hair germs in mice. Arch Dermatol Res 293(9):435-41. [PubMed: 11758785] [MGI Ref ID J:116953]
Yoshida T; Katoh A; Ohtsuki G; Mishina M; Hirano T. 2004. Oscillating Purkinje neuron activity causing involuntary eye movement in a mutant mouse deficient in the glutamate receptor delta2 subunit. J Neurosci 24(10):2440-8. [PubMed: 15014119] [MGI Ref ID J:97010]
van Empel VP; Bertrand AT; van der Nagel R; Kostin S; Doevendans PA; Crijns HJ; de Wit E; Sluiter W; Ackerman SL; De Windt LJ. 2005. Downregulation of apoptosis-inducing factor in harlequin mutant mice sensitizes the myocardium to oxidative stress-related cell death and pressure overload-induced decompensation. Circ Res 96(12):e92-e101. [PubMed: 15933268] [MGI Ref ID J:110278]
Berry RJ. 1961. The inheritance and pathogenesis of hydrocephalus-3 in the mouse J Pathol Bacteriol 81:157-167. [MGI Ref ID J:13103]
Davy BE; Robinson ML. 2003. Congenital hydrocephalus in hy3 mice is caused by a frameshift mutation in Hydin, a large novel gene. Hum Mol Genet 12(10):1163-70. [PubMed: 12719380] [MGI Ref ID J:83437]
Gruneberg H. 1943. Two new mutant genes in the house mouse. J Genet 45:22-28. [MGI Ref ID J:13035]
Kabos P; Matundan H; Zandian M; Bertolotto C; Robinson ML; Davy BE; Yu JS; Krueger RC Jr. 2004. Neural precursors express multiple chondroitin sulfate proteoglycans, including the lectican family. Biochem Biophys Res Commun 318(4):955-63. [PubMed: 15147965] [MGI Ref ID J:90103]
Lechtreck KF; Delmotte P; Robinson ML; Sanderson MJ; Witman GB. 2008. Mutations in Hydin impair ciliary motility in mice. J Cell Biol 180(3):633-43. [PubMed: 18250199] [MGI Ref ID J:135322]
McLone DG; Bondareff W; Raimondi AJ. 1973. Hydrocephalus-3, a Murine mutant. II. Changes in the brain extracellular space. Surg Neurol 1(4):233-42. [PubMed: 4732188] [MGI Ref ID J:5377]
Raimondi AJ; Bailey OT; McLone DG; Lawson RF; Echeverry A. 1973. The pathophysiology and morphology of murine hydrocephalus in Hy-3 and Ch mutants. Surg Neurol 1(1):50-5. [PubMed: 4784576] [MGI Ref ID J:5426]
Sakuragawa N; Yokoyama Y. 1994. Clinical and molecular genetics of inherited hydrocephalus Cong Anom 34:303-10. [MGI Ref ID J:24172]
Health & husbandry
Health & Colony Maintenance Information
Currently there no information available for this strain. This may be due to the supply level of this strain.
Purchasing information
Pricing, Supply Level & Notes, Controls, General Terms & Conditions
Pricing
| Pricing for USA, Canada and Mexico shipping destinations |
|
Animals Provided
Price (US dollars $) Cryorecovery Fee $1900.00 At least two mice that carry the mutation (if it is a mutant strain) will be provided. Their genotypes may not reflect those discussed in the strain description. Please inquire for possible genotypes and see additional details below.
Additional Supply Details
| Pricing for International shipping destinations |
|
Animals Provided
Price (US dollars $) Cryorecovery Fee $2470.00 At least two mice that carry the mutation (if it is a mutant strain) will be provided. Their genotypes may not reflect those discussed in the strain description. Please inquire for possible genotypes and see additional details below.
Additional Supply Details
|
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|
Supply Details
| Standard Supply | Cryopreserved. Ready for recovery. Please refer to pricing and supply notes for further information. |
|---|---|
| Supply Notes |
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Control Information
| Control | ||
|---|---|---|
| Untyped 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. | ||
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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.Ordering and Purchasing Information
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