Battery of Behavioral Tests Assessing General Locomotion, Muscular Strength, and Coordination in Mice

Osmon, Karlaina J.L.; Vyas, Meera; Woodley, Evan; Thompson, Patrick; Walia, Jagdeep S.

doi:10.3791/55491-v

Cited by 6 publications

(4 citation statements)

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“…Behavioral testing sessions included the use of OFT to assess general locomotion, and an accelerated RR protocol to evaluate motor coordination as previously described. 59 …”

Section: Methodsmentioning

confidence: 99%

Intrathecal delivery of a bicistronic AAV9 vector expressing β-hexosaminidase A corrects Sandhoff disease in a murine model: A dosage study

Ryckman,

Deschenes,

Quinville

et al. 2024

Molecular Therapy - Methods & Clinical Development

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“…Behavioral testing sessions included the use of OFT to assess general locomotion, and an accelerated RR protocol to evaluate motor coordination as previously described. 59 …”

Section: Methodsmentioning

confidence: 99%

Intrathecal delivery of a bicistronic AAV9 vector expressing β-hexosaminidase A corrects Sandhoff disease in a murine model: A dosage study

Ryckman,

Deschenes,

Quinville

et al. 2024

Molecular Therapy - Methods & Clinical Development

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“…Rotarod testing was utilized to evaluate overall motor function, including motor coordination and motor learning in mice [18][19][20]. A self-made Rotarod apparatus with a rotating rod 2.8 cm in diameter positioned 23 cm above the platform and with an adjustable rotation speed was used.…”

Section: ・Rotarod Testmentioning

confidence: 99%

Differences in exercise capacity and muscle glycogen metabolism in C57BL/6J and BALB/cA mice

Miyata,

Shogatsudani,

Igarashi

et al. 2024

Exp. Anim.

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This study compared differences in exercise capacity as well as muscle glycogen content and degradation, and mitochondrial enzyme activity between C57BL/6J and BALB/cA mice. In exercise tests, grip strength was higher in BALB/cA mice. In Rotarod and Inverted screen test, C57BL/6J mice had significantly longer exercise durations and showed differences in motor function and muscle endurance time. Glycogen in the liver and muscle of C57BL/6J mice was significantly decreased after 20 min of swimming. Muscle glycogen content in BALB/cA mice was higher than in C57BL/6J, but swimming induced no decrease in glycogen content. Glycogen phosphorylase in muscle was inactive in the absence of AMP, and its activity increased in a concentration-dependent manner with the addition of AMP in C57BL/6J mice. In BALB/cA mice, phosphorylase activity was increased by AMP, but not further increased by higher concentrations of AMP. The citrate synthase activity in muscle did not differ between C57BL/6J and BALB/cA mice. The results of this study suggested that the reactivity of muscle glycogen phosphorylase to AMP differs among strains of mice and affects glycogen availability during exercise.

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“…Rotarod Motor coordination of the mice was assessed using a RR apparatus (IITC Life Sciences, Woodland Hills, CA, USA) as per a previously published protocol [61]. Briefly, mice were placed on moving cylinders that accelerated from 4 to 40 rotations per minute (rpm) over 5 min.…”

Section: Behavioral Testingmentioning

confidence: 99%

“…Open Field Test Mice are tested using the OFT (ActiMot, TSE Systems, Berlin, Germany) to assess motor skills [61]. Animals were placed into a square box with high walls for 5 min each to record several parameters.…”

Section: Behavioral Testingmentioning

confidence: 99%

Efficacy of Adeno-Associated Virus Serotype 9-Mediated Gene Therapy for AB-Variant GM2 Gangliosidosis

Vyas,

Deschenes,

Osmon

et al. 2023

IJMS

Self Cite

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GM2 gangliosidoses are a group of neurodegenerative lysosomal storage disorders that are characterized by the accumulation of GM2 gangliosides (GM2), leading to rapid neurological decline and death. The hydrolysis of GM2 requires the specific synthesis, processing, and combination of products of three genes—HEXA, HEXB, and GM2A—within the cell’s lysosomes. Mutations in these genes result in Tay-Sachs disease, Sandhoff disease, or AB-variant GM2 gangliosidosis (ABGM2), respectively. ABGM2, the rarest of the three types, is characterized by a mutation in the GM2A gene, which encodes the GM2 activator (GM2A) protein. Being a monogenic disease, gene therapy is a plausible and likely effective method of treatment for ABGM2. This study aimed at assessing the effects of administering a one-time intravenous treatment of single-stranded Adeno-associated virus serotype 9 (ssAAV9)-GM2A viral vector at a dose of 1 × 1014 vector genomes (vg) per kilogram per mouse in an ABGM2 mouse model (Gm2a−/−). ssAAV9-GM2A was administered at 1-day (neonatal) or 6-weeks of age (adult-stage). The results demonstrated that, in comparison to Gm2a−/− mice that received a vehicle injection, the treated mice had reduced GM2 accumulation within the central nervous system and had long-term persistence of vector genomes in the brain and liver. This proof-of-concept study is a step forward towards the development of a clinically therapeutic approach for the treatment of patients with ABGM2.

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Battery of Behavioral Tests Assessing General Locomotion, Muscular Strength, and Coordination in Mice

Cited by 6 publications

References 0 publications

Intrathecal delivery of a bicistronic AAV9 vector expressing β-hexosaminidase A corrects Sandhoff disease in a murine model: A dosage study

Intrathecal delivery of a bicistronic AAV9 vector expressing β-hexosaminidase A corrects Sandhoff disease in a murine model: A dosage study

Differences in exercise capacity and muscle glycogen metabolism in C57BL/6J and BALB/cA mice

Efficacy of Adeno-Associated Virus Serotype 9-Mediated Gene Therapy for AB-Variant GM2 Gangliosidosis

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