| Phenotypic Profile |
| Category |
Entity |
Quantity |
Experimental Paradigm |
Age at Testing |
| Motor phenotype |
Motor coordination and balance1 |
Restored
Description: Restored latency to fall off the rotarod; delaying or skipping training sessions erased the benefits
|
Accelerating rotarod test |
8-32 weeks |
| |
|
Description: Restored latency to fall off the rotarod; delaying or skipping training sessions erased the benefits
|
|
|
| Motor phenotype |
Grip strength1 |
Refractory
Description: No decrease in foot slips
|
Wire hang test |
24 weeks |
| Motor phenotype |
General locomotor activity: Ambulatory activity1 |
Refractory
Description: No increase in total distance travelled
|
Open field test |
25 weeks |
| |
|
Description: No increase in total distance travelled
|
|
|
| Neuroanatomy / Ultrastructure / Cytoarchitecture |
Neuronal morphology1 |
Ameliorated
Description: No increased in soma and nuclear area; morphological changes were not due to nonspecific improvements across the hippocampus
|
Immunofluorescence staining |
24 weeks |
| |
|
Description: No increased in soma and nuclear area; morphological changes were not due to nonspecific improvements across the hippocampus
|
|
|
| Neuroanatomy / Ultrastructure / Cytoarchitecture |
Dendritic architecture: spine density1 |
Ameliorated
Description: Enhanced spine density
|
Immunofluorescence staining |
24 weeks |
| |
|
Description: Enhanced spine density
|
|
|
| Neuroanatomy / Ultrastructure / Cytoarchitecture |
Dendritic architecture: dendritic tree complexity1 |
Ameliorated
Description: Enhanced dendritic complexity
|
Immunofluorescence staining |
24 weeks |
| |
|
Description: Enhanced dendritic complexity
|
|
|
| Neurophysiology |
Neuronal activation following behavioral stimulation1 |
Ameliorated
Description: More tdtomato+cfos+ neurons in mice that were retested in the water maze; activation of task-specific neurons is necessary for presymptomatic training to benefit rett mice; inhibiting or activating a random subpopulation of neurons did not affect the memory of wild-type or rett mice expressing camk2acreer
Exp Paradigm: used cFos-targeted recombination in active populations (Fos-TRAP) to label task-specific neurons activated during water-maze
|
Immunofluorescence staining |
24 weeks |
| |
|
Description: More tdtomato+cfos+ neurons in mice that were retested in the water maze; activation of task-specific neurons is necessary for presymptomatic training to benefit rett mice; inhibiting or activating a random subpopulation of neurons did not affect the memory of wild-type or rett mice expressing camk2acreer
Exp Paradigm: used cFos-targeted recombination in active populations (Fos-TRAP) to label task-specific neurons activated during water-maze |
|
|
| Neurophysiology |
Spontaneous post synaptic event frequency: excitatory currents1 |
Ameliorated
Description: Presymptomatic training improved the frequencies of sipscs and sepscs of mecp2 negative neurons in rett mice (fig. 4f, h), but not the amplitudes (fig. 4g, i).
|
Whole-cell patch clamp |
13 weeks |
| |
|
Description: Presymptomatic training improved the frequencies of sipscs and sepscs of mecp2 negative neurons in rett mice (fig. 4f, h), but not the amplitudes (fig. 4g, i).
|
|
|
| Neurophysiology |
Spontaneous post synaptic event amplitude: inhibitory currents1 |
Refractory
Description: Presymptomatic training improved the frequencies of sipscs and sepscs of mecp2 negative neurons in rett mice (fig. 4f, h), but not the amplitudes (fig. 4g, i).
|
Whole-cell patch clamp |
13 weeks |
| |
|
Description: Presymptomatic training improved the frequencies of sipscs and sepscs of mecp2 negative neurons in rett mice (fig. 4f, h), but not the amplitudes (fig. 4g, i).
|
|
|
| Neurophysiology |
Spontaneous post synaptic event amplitude: excitatory currents1 |
Refractory
Description: Presymptomatic training improved the frequencies of sipscs and sepscs of mecp2 negative neurons in rett mice (fig. 4f, h), but not the amplitudes (fig. 4g, i).
|
Whole-cell patch clamp |
13 weeks |
| |
|
Description: Presymptomatic training improved the frequencies of sipscs and sepscs of mecp2 negative neurons in rett mice (fig. 4f, h), but not the amplitudes (fig. 4g, i).
|
|
|
| Neurophysiology |
Spontaneous post synaptic event frequency: inhibitory currents1 |
Ameliorated
Description: Presymptomatic training improved the frequencies of sipscs and sepscs of mecp2 negative neurons in rett mice (fig. 4f, h), but not the amplitudes (fig. 4g, i).
|
Whole-cell patch clamp |
13 weeks |
| |
|
Description: Presymptomatic training improved the frequencies of sipscs and sepscs of mecp2 negative neurons in rett mice (fig. 4f, h), but not the amplitudes (fig. 4g, i).
|
|
|
| Sensory |
Startle response: acoustic stimulus1 |
Refractory
Description: No increase in startle amplitude
|
Acoustic startle reflex test |
24 weeks |
| |
|
Description: No increase in startle amplitude
|
|
|
| Developmental profile |
Size/growth1 |
No adverse effect
Description: No change in body weight
|
Body weight measurement |
Adult |
| |
|
Description: No change in body weight
|
|
|
| Emotion |
Anxiety1 |
Refractory
Description: No decrease in time spent in open arms
|
Elevated plus maze test |
24 weeks |
| |
|
Description: No decrease in time spent in open arms
|
|
|
| Learning & memory |
Spatial working memory1 |
Restored
Description: Restored latency for memory acquisition
|
Morris water maze test |
24 weeks |
| |
|
Description: Restored latency for memory acquisition
|
|
|
| Learning & memory |
Spatial reference memory1 |
Restored
Description: Restored time in target quadrant in probe trial
|
Morris water maze test |
24 weeks |
| |
|
Description: Restored time in target quadrant in probe trial
|
|
|
| Learning & memory |
Cued or contextual fear conditioning: Memory of cue1 |
No adverse effect
Description: No change in time spent freezing
|
Fear conditioning test |
24 weeks |
| |
|
Description: No change in time spent freezing
|
|
|
| Learning & memory |
Cued or contextual fear conditioning: Memory of context1 |
Refractory
Description: No increase in time spent freezing
|
Fear conditioning test |
24 weeks |
| |
|
Description: No increase in time spent freezing
|
|
|
|
| Not Reported: |
|
