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Gene expression analyses in male and female Cln3Δex7/8 mouse brains following oral administration of flupirtine for 14 weeks

ABSTRACT: The Neuronal Ceroid Lipofuscinoses, one of the most common fatal neurodegenerative diseases of childhood, is caused by mutations of the CLN3 gene. Treatment regimens are symptomatic, highlighting the importance of disease-modifying drugs. Flupirtine confers neuroprotective effects in CLN3-deficient cells as well as in Cln3Δex7/8 mice. The purpose of the present study is to discern global expression programs embodying therapeutic targeting of CLN3 disease. Differential gene expression in the brains of 18-week-old Cln3Δex7/8 mice, a knock-in mouse model, was determined after daily oral supplementation of vehicle or flupirtine (30 mg/kg BW), for a period of 14 weeks, using GeneChip Mouse Genome 430 2.0 array platform from Affymetrix and quantitative real-time PCR. This manuscript sheds light on global expression programs that are associated with therapeutic treatment of CLN3 disease by flupirtine in male and female mice separately. Understanding the molecular effects of flupirtine treatment herein would pave the way for new personalized and sex-specific strategies for treatment of this debilitating genetic disease.

ORGANISM(S): Mus musculus

SUBMITTER: Joelle Makoukji

PROVIDER: E-MTAB-9812 | biostudies-arrayexpress |

REPOSITORIES: biostudies-arrayexpress

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