Source Paper
Atrophin-1 antisense oligonucleotide provides robust protection from pathology in a fully humanized DRPLA model
Smith VL, Gidi BZ, Bragg RM, Cantle JP, Ben-Varon A et al.
Mol Ther Nucleic Acids • 2025
Modified SHIRPA
Objective: Phenotypic screening assessment to evaluate lethargy, gait issues, tremors, pelvic and tail elevation, hindlimb and forelimb grasping, piloerection, and touch escape behaviors in mice
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Materials1
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Protocol Steps
Home cage observation
Observe mice inside their home cage to assess multiple behavioral phenotypes
View evidence from paper
“we observed mice inside of their home cage to assess their level of lethargy, gait issues, tremors, pelvic and tail elevation, hindlimb and forelimb grasping, piloerection, and touch escape”
Phenotype scoring
Score each assessed phenotype using individual scales, with higher scores indicating worsening of the phenotype
View evidence from paper
“Each phenotype that was assessed had an individual scale that was used to measure the behavior”
Assessment timing
Perform all assessments when mice are 4 weeks of age
View evidence from paper
“All mice were assessed at 4 weeks of age”