Pre-training performance of Cdkl5 deletion and knock-in mice. All Cdkl5 mutant mice successfully initiated trials during 2 days of habituation, met the criteria of completing 30+ trials for 2 consecutive days in stage 3 and required similar number of days to reach criterion in stage 4 when compared with wild-type, sex-matched littermate controls
| Pre-training stages . | Cdkl5 exon6+/+F N = 10 . | Cdkl5 exon6−/+F N = 11 . | Cdkl5 exon6+/yM N = 10 . | Cdkl5 exon6−/yM N = 11 . | Cdkl5 R59X+/+F N = 10 . | Cdkl5 R59X−/+F N = 11 . | Cdkl5 R59X+/yM N = 10 . | Cdkl5 R59X−/yM N = 11 . |
|---|---|---|---|---|---|---|---|---|
| Stage 1: Habituation day 1 number of trials | 11.10 ± 1.51 | 23.73 ± 2.87 | 15.10 ± 1.16 | 34.91 ± 4.96 | 9.80 ± 0.95 | 19.00 ± 1.88 | 19.60 ± 5.64 | 42.82 ± 5.18 |
| Stage 1: Habituation day 2 number of trials | 34.10 ± 10.13 | 86.55 ± 17.79 | 67.30 ± 14.03 | 99.09 ± 18.13 | 47.70 ± 8.12 | 63.27 ± 12.28 | 82.90 ± 8.14 | 138 ± 15.33 |
| Stage 2: Number of trials | 27.60 ± 3.49 | 59.45 ± 6.26 | 26.50 ± 5.99 | 58.55 ± 7.64 | 27.80 ± 2.928 | 48.18 ± 3.56 | 32.10 ± 3.37 | 72.18 ± 6.03 |
| Stage 3: Number of trials | 53.13 ± 7.31 | 94.18 ± 5.61 | 57.97 ± 6.68 | 90.00 ± 5.51 | 56.59 ± 7.50 | 76.45 ± 8.34 | 51.87 ± 5.66 | 69.20 ± 6.86 |
| Stage 4: Days to reach criterion | 3.70 ± 0.47 | 3.64 ± 0.34 | 2.90 ± 0.10 | 4.18 ± 0.40 | 2.20 ± 0.20 | 2.46 ± 0.25 | 2.60 ± 0.16 | 2.64 ± 0.34 |
| Pre-training stages . | Cdkl5 exon6+/+F N = 10 . | Cdkl5 exon6−/+F N = 11 . | Cdkl5 exon6+/yM N = 10 . | Cdkl5 exon6−/yM N = 11 . | Cdkl5 R59X+/+F N = 10 . | Cdkl5 R59X−/+F N = 11 . | Cdkl5 R59X+/yM N = 10 . | Cdkl5 R59X−/yM N = 11 . |
|---|---|---|---|---|---|---|---|---|
| Stage 1: Habituation day 1 number of trials | 11.10 ± 1.51 | 23.73 ± 2.87 | 15.10 ± 1.16 | 34.91 ± 4.96 | 9.80 ± 0.95 | 19.00 ± 1.88 | 19.60 ± 5.64 | 42.82 ± 5.18 |
| Stage 1: Habituation day 2 number of trials | 34.10 ± 10.13 | 86.55 ± 17.79 | 67.30 ± 14.03 | 99.09 ± 18.13 | 47.70 ± 8.12 | 63.27 ± 12.28 | 82.90 ± 8.14 | 138 ± 15.33 |
| Stage 2: Number of trials | 27.60 ± 3.49 | 59.45 ± 6.26 | 26.50 ± 5.99 | 58.55 ± 7.64 | 27.80 ± 2.928 | 48.18 ± 3.56 | 32.10 ± 3.37 | 72.18 ± 6.03 |
| Stage 3: Number of trials | 53.13 ± 7.31 | 94.18 ± 5.61 | 57.97 ± 6.68 | 90.00 ± 5.51 | 56.59 ± 7.50 | 76.45 ± 8.34 | 51.87 ± 5.66 | 69.20 ± 6.86 |
| Stage 4: Days to reach criterion | 3.70 ± 0.47 | 3.64 ± 0.34 | 2.90 ± 0.10 | 4.18 ± 0.40 | 2.20 ± 0.20 | 2.46 ± 0.25 | 2.60 ± 0.16 | 2.64 ± 0.34 |
Pre-training performance of Cdkl5 deletion and knock-in mice. All Cdkl5 mutant mice successfully initiated trials during 2 days of habituation, met the criteria of completing 30+ trials for 2 consecutive days in stage 3 and required similar number of days to reach criterion in stage 4 when compared with wild-type, sex-matched littermate controls
| Pre-training stages . | Cdkl5 exon6+/+F N = 10 . | Cdkl5 exon6−/+F N = 11 . | Cdkl5 exon6+/yM N = 10 . | Cdkl5 exon6−/yM N = 11 . | Cdkl5 R59X+/+F N = 10 . | Cdkl5 R59X−/+F N = 11 . | Cdkl5 R59X+/yM N = 10 . | Cdkl5 R59X−/yM N = 11 . |
|---|---|---|---|---|---|---|---|---|
| Stage 1: Habituation day 1 number of trials | 11.10 ± 1.51 | 23.73 ± 2.87 | 15.10 ± 1.16 | 34.91 ± 4.96 | 9.80 ± 0.95 | 19.00 ± 1.88 | 19.60 ± 5.64 | 42.82 ± 5.18 |
| Stage 1: Habituation day 2 number of trials | 34.10 ± 10.13 | 86.55 ± 17.79 | 67.30 ± 14.03 | 99.09 ± 18.13 | 47.70 ± 8.12 | 63.27 ± 12.28 | 82.90 ± 8.14 | 138 ± 15.33 |
| Stage 2: Number of trials | 27.60 ± 3.49 | 59.45 ± 6.26 | 26.50 ± 5.99 | 58.55 ± 7.64 | 27.80 ± 2.928 | 48.18 ± 3.56 | 32.10 ± 3.37 | 72.18 ± 6.03 |
| Stage 3: Number of trials | 53.13 ± 7.31 | 94.18 ± 5.61 | 57.97 ± 6.68 | 90.00 ± 5.51 | 56.59 ± 7.50 | 76.45 ± 8.34 | 51.87 ± 5.66 | 69.20 ± 6.86 |
| Stage 4: Days to reach criterion | 3.70 ± 0.47 | 3.64 ± 0.34 | 2.90 ± 0.10 | 4.18 ± 0.40 | 2.20 ± 0.20 | 2.46 ± 0.25 | 2.60 ± 0.16 | 2.64 ± 0.34 |
| Pre-training stages . | Cdkl5 exon6+/+F N = 10 . | Cdkl5 exon6−/+F N = 11 . | Cdkl5 exon6+/yM N = 10 . | Cdkl5 exon6−/yM N = 11 . | Cdkl5 R59X+/+F N = 10 . | Cdkl5 R59X−/+F N = 11 . | Cdkl5 R59X+/yM N = 10 . | Cdkl5 R59X−/yM N = 11 . |
|---|---|---|---|---|---|---|---|---|
| Stage 1: Habituation day 1 number of trials | 11.10 ± 1.51 | 23.73 ± 2.87 | 15.10 ± 1.16 | 34.91 ± 4.96 | 9.80 ± 0.95 | 19.00 ± 1.88 | 19.60 ± 5.64 | 42.82 ± 5.18 |
| Stage 1: Habituation day 2 number of trials | 34.10 ± 10.13 | 86.55 ± 17.79 | 67.30 ± 14.03 | 99.09 ± 18.13 | 47.70 ± 8.12 | 63.27 ± 12.28 | 82.90 ± 8.14 | 138 ± 15.33 |
| Stage 2: Number of trials | 27.60 ± 3.49 | 59.45 ± 6.26 | 26.50 ± 5.99 | 58.55 ± 7.64 | 27.80 ± 2.928 | 48.18 ± 3.56 | 32.10 ± 3.37 | 72.18 ± 6.03 |
| Stage 3: Number of trials | 53.13 ± 7.31 | 94.18 ± 5.61 | 57.97 ± 6.68 | 90.00 ± 5.51 | 56.59 ± 7.50 | 76.45 ± 8.34 | 51.87 ± 5.66 | 69.20 ± 6.86 |
| Stage 4: Days to reach criterion | 3.70 ± 0.47 | 3.64 ± 0.34 | 2.90 ± 0.10 | 4.18 ± 0.40 | 2.20 ± 0.20 | 2.46 ± 0.25 | 2.60 ± 0.16 | 2.64 ± 0.34 |
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