P3 Ko

(29,30)

90% die within 24 h, 57% cleft palate Survivors fertile, but not nursing Animal model for Angelman syndrome spontaneous seizures, hyperactive, hyper-responsive Heterozygous mice show almost normal phenotype

Loss of ~50% of all GABAa receptors

Y2 KO (31-33)

Normal embryonic development Early postnatal lethality

72+/- mice are animal model for chronic anxiety with enhanced response in trace fear conditioning and ambiguous cue discrimination

Reduced postsynaptic clustering of GABAA receptors and gephyrin, which can be partially rescued by overexpression of 73

Y2 floxed X CaMKIIa-Cre (34)

Conditional knockout in 3-week-old mouse forebrain Lethal epilepsy

Rapid loss of postsynaptic GABAA receptors

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