Mouse Genome Informatics
hm1
    Magel2tm1Stw/Magel2tm1Stw
C57BL/6-Magel2tm1Stw
Key:
phenotype observed in females WTSI Wellcome Trust Sanger Institute
phenotype observed in males EuPh Europhenome
N normal phenotype
mortality/aging
• when the Magel2tm1Stw allele is inherited paternally, 10% fewer mice survive to weaning than expected

reproductive system
• when the Magel2tm1Stw allele is inherited paternally, males reproduce but with decreasing frequency and cease mating by 16 weeks unlike wild-type mice (J:125637)
• however, no defects in spermatogenesis and testes weight are observed (J:125637)

behavior/neurological
• when the Magel2tm1Stw allele is inherited paternally, mice run significantly less than wild-type mice (3047+/-930 counts per day compared to 12770+/-2343 counts per day for wild-type mice)
• when the Magel2tm1Stw allele is inherited paternally, mice run in more frequent and shorter bouts (8.5+/-0.7 bouts per day compared to 4.9+/-0.4 bouts per day for wild-type mice and 21.8+/-4.3 minutes per bout compared to 83.1+/-15.4 minutes per bout for wild-type mice)
• when the Magel2tm1Stw allele is inherited paternally, mice exhibit less daily activity during the subjective night (76.9+/-2.8% compared to 86.6+/-2.5% for wild-type mice)when the Magel2tm1Stw allele is inherited paternally, mice exhibit less daily activity during the subjective night (76.9+/-2.8% compared to 86.6+/-2.5% for wild-type mice)
• when the Magel2tm1Stw allele is inherited paternally, rhythms have lower chi-squared periodogram amplitudes than in wild-type mice
• when the Magel2tm1Stw allele is inherited paternally, after 24 hours starvation mice consume less food than wild-type mice
• when the Magel2tm1Stw allele is inherited paternally, mice run significantly less than wild-type mice (3047+/-930 counts per day compared to 12770+/-2343 counts per day for wild-type mice)
• when the Magel2tm1Stw allele is inherited paternally, mice run in more frequent and shorter bouts (8.5+/-0.7 bouts per day compared to 4.9+/-0.4 bouts per day for wild-type mice and 21.8+/-4.3 minutes per bout compared to 83.1+/-15.4 minutes per bout for wild-type mice)

growth/size
N
• when the Magel2tm1Stw allele is inherited paternally, surviving mice display no abnormalities in size or weight up to 2 years of age (J:125637)

cellular
• only the paternally inherited allele is expressed


Mouse Genome Informatics
ht2
    Magel2tm1Stw/Magel2+
C57BL/6-Magel2tm1Stw
Key:
phenotype observed in females WTSI Wellcome Trust Sanger Institute
phenotype observed in males EuPh Europhenome
N normal phenotype

Older Magel2tm1Stw/Magel2+ females that inherit the mutant allele paternally lack corpora lutea in the ovaries

mortality/aging
• only 50-60% of pups born to female mice that inherit the mutant allele paternally survive until weaning
• infertility occurs in both sexes by 24 weeks of age in mice that inherit the mutant allele paternally
• fertility rates are normal between 7-14 weeks of age, drops to about 20% at 19-24 weeks of age, with no litters are born after 24 weeks of age
• at younger ages, there is also a significant increase in the mean time between pairing and successful breeding (males: 9 days vs. 4 days for controls, females: 12 days vs. 4 days in controls)

cellular
• only the paternally inherited allele is expressed

reproductive system
• an absence of corpus lutea is noted in 10 of 14 female mice that are over 24 weeks of age and have inherited the mutant allele paternally (J:144836)
• infertility occurs in both sexes by 24 weeks of age in mice that inherit the mutant allele paternally
• fertility rates are normal between 7-14 weeks of age, drops to about 20% at 19-24 weeks of age, with no litters are born after 24 weeks of age
• at younger ages, there is also a significant increase in the mean time between pairing and successful breeding (males: 9 days vs. 4 days for controls, females: 12 days vs. 4 days in controls)
• in female mice inheriting the mutant allele paternally, vaginal opening is significantly delayed by 1.4 days
• in female mice inheriting the mutant allele paternally, the age of first estrus is delayed by 5.3 days
• only 25% of mice experience proestrus in female mice that are 26 weeks of age and have inherited the mutant allele paternally (J:144836)
• estrous cycle is prolonged and irregular in female mice that have inherited the mutant allele paternally (J:144836)
• a mean of 6.4 pups is born to female mice that inherit the mutant allele paternally compared to 7.8 pups for controls

taste/olfaction
• latency time to find buried food is more than twice that of controls for mice that are over 24 weeks of age and have inherited the mutant allele paternally
• fasted male mice that have inherited the mutant allele paternally only investigate a dried vanilla spot for 0.75 s compared to 6.7 s for controls
• sexually-experienced male mice that have inherited the mutant allele paternally show no preference for female soiled bedding unlike their wild-type controls

behavior/neurological
• female mice with paternal inheritance of the mutant allele frequently cannibalize their pups (J:144836)

endocrine/exocrine glands
• an absence of corpus lutea is noted in 10 of 14 female mice that are over 24 weeks of age and have inherited the mutant allele paternally (J:144836)

homeostasis/metabolism
• mean serum testosterone levels are significantly lower in male mice that inherit the mutant allele paternally (6.1 ng/ml versus 20.2 ng/ml in controls)

Mouse Models of Human Disease
OMIM IDRef(s)
Prader-Willi Syndrome; PWS 176270 J:144836