Phenotypes associated with this allele
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| Find Mice |
Using the International Mouse Strain Resource (IMSR)
Mouse lines carrying:
Zdhhc7tm1.2Lusc mutation
(1 available);
any
Zdhhc7 mutation
(25 available)
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growth/size/body
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• both males and females exhibit normal body weight at 1-2 months of age, with no overt physical phenotypes
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behavior/neurological
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N |
• mice exhibit no overt behavioral phenotypes
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homeostasis/metabolism
mortality/aging
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• a few double knockout (DKO) mice survive to 1 year of age
• however, no viable postnatal DKO mice are recovered past 3 generations of brother-sister matings of double heterozygotes
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• unexpectedly, most DKO mice die perinatally with a few surviving to adulthood
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growth/size/body
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• DKO mice appear severely runted by 1 year of age
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• by 1 year of age, surviving DKO mice display a severely reduced body weight relative to controls (~54% of that in double heterozygotes and single Zdhhc7tm1.2Lusc homozygotes)
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nervous system
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• by 1 year of age, surviving DKO mice display a severely reduced brain weight relative to controls (~75% of that in double heterozygotes and single Zdhhc7tm1.2Lusc homozygotes)
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• under competitive culture conditions, DKO primary cortical neurons co-cultured with wild-type neurons exhibit defects in synaptic localization of GABAARs resulting in loss of innervation by wild-type neurons
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• under competitive culture conditions, DKO primary cortical neurons co-cultured with wild-type neurons show deficits in GABAARs clustering and inhibitory synapse density, with a drastic reduction in the density of both presynaptic GAD puncta and postsynaptic gamma2 puncta, no change in the size of gamma2 puncta, but significantly decreased colocalization of gamma2 and GAD puncta
• under competitive culture conditions, the gamma2 clustering defect in co-cultured DKO neurons is comparable to that in seen in co-cultures of Zdhhc3tm1.2Lusc neurons with wild-type neurons
• however, in pure cultures, DKO primary cortical neurons show normal density and colocalization of punctate immunoreactivity for the gamma2 subunit and GAD
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• under competitive culture conditions, DKO primary cortical neurons co-cultured with an excess of wild-type neurons exhibit decreased accumulation of GABAARs at synapses along with reduced GABAergic innervation and synaptic function
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• patch clamp recordings of DKO neurons grown in competition with wild-type neurons show that the frequency, but not the amplitude, of mEPSCs is reduced in DKO versus wild-type neurons
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• patch clamp recordings of DKO neurons grown in competition with wild-type neurons show that the frequency and amplitude of mIPSCs recorded from DKO neurons are significantly reduced relative to wild-type neurons
• however, GABA- and glutamate-evoked whole-cell currents in DKO neurons are normal
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homeostasis/metabolism
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