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Z_NBEA_1_KO_HM
Model Details
Change
No Change
     
Phenotypic Profile
Category Entity Quantity Experimental Paradigm Age at Testing
Motor phenotype Motor coordination and balance1
Decreased
Description: nbea mutants frequently lie on their sides and are unable to balance using their pectoral fins, unlike controls.

General observations 6 dpf
Motor phenotype General locomotor activity1
Decreased
Description: nbea mutants show decreased overall motion compared to controls.

General observations 6 dpf
Neuroanatomy / Ultrastructure / Cytoarchitecture Dendritic architecture: dendritic tree complexity1
Decreased
Description: nbea mutant shows decreased dendritic tree complexity of Mauthner neurons compared to controls. Time course analysis shows nbea is required to maintain the extensive branching and fine dendritic processes found in mature neurons.

Immunofluorescence staining Adult
Neuroanatomy / Ultrastructure / Cytoarchitecture Synaptic morphology1
Decreased
Description: nbea mutant shows decreased expression of glycine receptor at Mauther neuron synapses in the hindbrain and spinal cord, compared to controls. Ablation of M circuit led to a loss of GlyR associated with M dendrites in the hindbrain but did not affect GlyR in the CoLo/CoLo synapses in the spinal cord. At CoLo/CoLo synapses in nbea mutants GlyR was reduced, compared to controls.

Immunofluorescence staining 3-14 dpf, larva
Neuroanatomy / Ultrastructure / Cytoarchitecture Synaptic morphology1
Decreased
Description: nbea mutant shows decreased expression of connexin36, a marker of electrical synapses, at sensory/Mauthner neurons and Mauthner/Commissural local neuron synapses, compared to controls. nbea mutants show decreased connexin36 staining in the M circuit, (3-14 dpf) and also at electrical synapses in the forebrain, midbrain, hindbrain and spinal cord compared to controls.

Immunofluorescence staining 3 dpf, 3-14 dpf, Larva
Neuroanatomy / Ultrastructure / Cytoarchitecture Synaptic morphology1
Decreased
Description: nbea mutant shows decreased expression of the cytosolic scaffold protein ZO-1, found at electrical synapses and non-synaptic tight junctons, compared to controls. ZO-1 colocalized with Cx36 and within the M circuit.

Immunofluorescence staining Larva
Neuroanatomy / Ultrastructure / Cytoarchitecture Dendritic architecture: dendritic tree complexity1
Decreased
Description: nbea mutants cannot maintain the fine processes resulting in loss of dendritic complexity compared to controls.

Immunofluorescence staining 5 dpf
Neuroanatomy / Ultrastructure / Cytoarchitecture Synaptic morphology1
Decreased
Description: nbea mutant shows decreased expression of the cytosolic scaffold protein Gephyrin, found at glycinergic inhibitory synapses, compared to controls.

Immunofluorescence staining Larva
Neurophysiology Synaptic transmission1
Decreased
Description: nbea mutant shows decreased electrical synapse function compared to controls.

Retrograde labeling assay Larva
Sensory Startle response: acoustic stimulus1
Decreased
Description: nbea mutants show decreased frequency of response in acoustic startle reponse test compared to wildtype controls although mutants could perform escape responses that were indistinguishable from wildtype but responded in fewer trials.

Acoustic startle reflex test 6 dpf
Sensory Touch1
Decreased
Description: nbea mutants show decreased responsiveness to touch compared to wildtype controls.

Gentle touch test 6 dpf
Developmental profile Mortality/lethality1
Decreased
Description: nbea mutants surivive to at least 14dpf but die before they reach adulthood.

General observations adult
Molecular profile Targeted expression1
Decreased
Description: nbea mutant shows decreased expression of nbea protein compared to wildtype controls.

Immunofluorescence staining Larva
Developmental profile Developmental trajectory1 No change General observations Larva
Developmental profile Developmental trajectory1 No change Macroscopic analysis 5 dpf
Developmental profile General characteristics1 No change General observations Larva
Neuroanatomy / Ultrastructure / Cytoarchitecture Anatomical projections and connectivity1 No change Immunofluorescence staining 5 dpf
Neuroanatomy / Ultrastructure / Cytoarchitecture Anatomical projections and connectivity1 No change Immunofluorescence staining 5 dpf
Neuroanatomy / Ultrastructure / Cytoarchitecture Dendritic architecture: dendritic tree complexity1 No change Immunofluorescence staining 1-4 dpf
Neuroanatomy / Ultrastructure / Cytoarchitecture Neuronal number1 No change Immunofluorescence staining 5 dpf
Neuroanatomy / Ultrastructure / Cytoarchitecture Synaptic morphology: active zone1 No change Immunofluorescence staining Larva
Neuroanatomy / Ultrastructure / Cytoarchitecture Synaptic morphology: active zone1 No change Immunofluorescence staining Larva
Not Reported: Circadian sleep/wake cycle,  Communications,  Emotion,  Immune response,  Learning & memory,  Maternal behavior,  Physiological parameters,  Repetitive behavior,  Seizure,  Social behavior,  
 
References: 1:  Miller AC , et al. 2014
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