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Former Names C57BL/6J-Aqp2ndi/J (Changed: 22-MAR-06 ) Type Chemically Induced Mutation; Coisogenic; Mutant Strain; Species laboratory mouse Donating Investigator Nicholas Gekakis, Genomics Inst of the Novartis Res Fndtn Description
Mice homozygous for this mutation are viable with normal body size and lifespan. Homozygotes are fertile, but females usually die during labor. The predicted valine for phenylalanine substitution at amino acid 204 of the mutant protein (F204V) leads to an enrichment in kidney cells of a normally short-lived, intermediately glycosylated 31 kDa isoform and to a reduction in the mature, glycosylated protein level. The mutant AQP2F204V protein is mis-localized in kidney collecting duct cells, and fails to translocate to the apical cell surface in response to desmopressin. Homozygous mice exhibit excessive water consumption and urination, normal plasma glucose levels, and no glucose in the urine. Mutant mice are unable to concentrate urine and exhibit severe hydronephrosis. The presence of some mature, glycosylated protein and a defective, but not completely absent, desmopressin response in homozygous mutant mice likely permits their survival. Heterozygous mice are viable and fertile, and their kidneys appear normal by immunohistochemical analysis. This strain may be useful in studies of diabetes insipidus, kidney metabolism, or intracellular protein translocation, and represents the first published mouse model of nephrogenic diabetes insipidus to survive past the first week of life.Development
Following multidose ethylnitrosourea (ENU) treatments to induce mutations in founder C57BL/6J mice, a forward genetic screen was utilized to identify offspring for abnormal whole body metabolism. This screening identified a family of mice that urinated and drank excessively. The disorder in these mice segregated in a monogenic, autosomal recessive manner. Sequencing of the endogenous gene coding region of affected mice identified a thymine to guanine (T to G) transversion. This leads to a valine for phenylalanine substitution at amino acid 204 of the protein (F204V).
| Control | ||
|---|---|---|
| Wild-type from the colony | ||
| 000664 C57BL/6J | ||
| Considerations for Choosing Controls | ||
Strains carrying other alleles of Aqp2
006881 B6.Cg-Tg(Aqp2-cre)1Dek/J 000530 C57BL/6J-Aqp2cph/J View Strains carrying other alleles of Aqp2 (2 strains)
Related Disease (OMIM) Terms
Diabetes Insipidus, Nephrogenic, Autosomal Mammalian Phenotype Terms assigned by genotype
Aqp2F204V/Aqp2F204V
C57BL/6-Aqp2F204V
- life span-post-weaning/aging
- *normal* life span-post-weaning/aging (MGI Ref ID J:105809)
- mutants appear to have normal lifespans, unlike Aqp2 null mice
- homeostasis/metabolism phenotype
- decreased urine osmolality (MGI Ref ID J:105809)
- mice produce very dilute urine; concentration is approximately 161 mOsm compared to about 1,293 mOsm in wild-type
- behavior/neurological phenotype
- polydipsia (MGI Ref ID J:105809)
- water consumption is dramatically higher than in heterozygotes or wild-type
- renal/urinary system phenotype
- abnormal kidney physiology (MGI Ref ID J:105809)
- treatment with 1-deamino-8-D-arginine vasopressin produces a proportionally smaller increase in urine osmolarity compared to wild-type
- polyuria (MGI Ref ID J:105809)
- mice have dramatically increased urine output, producing more than their body weight in urine in 24 hours
- decreased urine osmolality (MGI Ref ID J:105809)
- mice produce very dilute urine; concentration is approximately 161 mOsm compared to about 1,293 mOsm in wild-type
- hydronephrosis (MGI Ref ID J:105809)
- mice exhibit severe hydronephrosis that is detectable at 4 weeks of age and worsens with age
- ureter morphology is normal despite dilation of the renal pelvis
Research Applications
This mouse can be used to support research in many areas including:
Cell Biology Research
Channel and Transporter Defects
Developmental Biology Research
Internal/Organ Defects (hydronephrosis)
Internal/Organ Defects (kidney)
Internal/Organ Research
Kidney Defects (diabetes insipidus)
| Allele Symbol | Aqp2F204V | ||
|---|---|---|---|
| Allele Name | Phe204Val | ||
| Common Name(s) | Aqp2ndi; | ||
| Mutation Made By | Nicholas Gekakis, Genomics Inst of the Novartis Res Fndtn | ||
| Strain of Origin | C57BL/6 | ||
| Gene Symbol and Name | Aqp2, aquaporin 2 | ||
| Chromosome | 15 | ||
| Gene Common Name(s) | AQP-2; AQP-CD; MGC156502; MGC34501; WCH-CD; aquaporin-2; congenital progressive hydronephrosis; cph; jpk; juvenile polycystic kidneys; | ||
| Molecular Note | A T to G transversion in the coding region of the gene led to a valine to phenylalanine substitution at postion 204 of the protein. The mutant protein was distributed randomly throughout collecting duct cells of the kidney rather than localized to the subapical region of collecting duct cells in the wild-type kidney. [MGI Ref ID J:105809] | ||
Genotyping Protocols
Aqp2F204V, SEP PCR, vers. 1
Helpful Links
Optimizing PCR Protocols
Lloyd DJ; Hall FW; Tarantino LM; Gekakis N. 2005. Diabetes Insipidus in Mice with a Mutation in Aquaporin-2. PLoS Genet 1(2):e20. [PubMed: 16121255] [MGI Ref ID J:105809]
Colony Maintenance
Breeding & Husbandry When maintaining a live colony, these mice are bred as heterozygotes. Homozygous mice are fertile, but females almost always die during labor. Homozygous mice exhibit excessive water consumption and urination.
*Price(s) in US dollars ($)
Weeks of Age Price* Gender Cryorecovery Fee $1900.00 Cryopreserved Embryos Fee $1600.00
| Standard Supply | Repository-Cryopreserved. Must Be Recovered. Please refer to pricing and supply notes for further information. |
|---|---|
| Supply Notes |
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*Price(s) in US dollars ($)
Weeks of Age Price* Gender Cryorecovery Fee $2470.00 Cryopreserved Embryos Fee $2080.00
| Standard Supply | Repository-Cryopreserved. Must Be Recovered. Please refer to pricing and supply notes for further information. |
|---|---|
| Supply Notes |
|
| Control | ||
|---|---|---|
| Wild-type from the colony | ||
| 000664 C57BL/6J | ||
| 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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