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Dnm2 downregulation by antisense oligonucleotide (ASO1) injections rescue a murine model of recessive centronuclear myopathy (Bin1mck-/-)

ABSTRACT: In this study, we performed RNA sequencing on tibialis anterior muscle of male mice. WT (injected with PBS) and Bin1mck-/- (injected with PBS) mice as well as ASO1 injected Bin1mck-/- mice and ASO1 injected WT mice were sequenced at 7 weeks of age. ASO1 targets specifically Dnm2 ARN.

ORGANISM(S): Mus musculus

PROVIDER: GSE160077 | GEO | 2020/12/15

REPOSITORIES: GEO

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Dnm2 downregulation by antisense oligonucleotide (ASO1) injections rescue a murine model of autosomal dominant centronuclear myopathy (Dnm2SL/+)

Project description:In this study, we performed RNA sequencing on tibialis anterior muscle of male mice. WT (injected with ASO control) and Dnm2SL/+ (injected with ASO control) mice as well as ASO1 injected Dnm2SL/+ mice and ASO1 injected WT mice were sequenced at 7w of age. ASO1 targets specifically Dnm2 ARN.

2020-12-15 | GSE160078 | GEO

Genetic downregulation of Dnm2 as a therapeutic approach in a murine model of X-linked myotubular myopathy (Mtm1-/y)

Project description:In this study, we performed mass spectometry on tibialis anterior muscle of male mice. WT and Mtm1 deficient (Mtm1-/y) mice as well as on Dnm2+/- mice and on Mtm1-/y mice crossed with Dnm2+/- (Mtm1-/yDnm2+/-) were analyzed. A longitudinal follow up was established by analyzing all the genotypes at E18.5, 2w and 7w.

2022-10-13 | PXD021725 | Pride

Genetic downregulation of Dnm2 as a therapeutic approach in a murine model of X-linked myotubular myopathy (Mtm1-/y)

Project description:In this study, we performed RNA sequencing on tibialis anterior muscle of male mice. WT and Mtm1 deficient (Mtm1-/y) mice as well as on Dnm2+/- mice and on Mtm1-/y mice crossed with Dnm2+/- (Mtm1-/yDnm2+/-) were sequenced. A longitudinal follow up was established by sequencing all the genotypes at E18.5, 2w and 7w.

2020-12-15 | GSE160079 | GEO

Dnm2 downregulation by antisense oligonucleotide (ASO1) injections rescue a murine model of recessive centronuclear myopathy (Bin1mck-/-)

Project description:Dnm2 downregulation by antisense oligonucleotide (ASO1) injections rescue a murine model of recessive centronuclear myopathy (Bin1mck-/-)

| PRJNA671741 | ENA

Dnm2 downregulation by antisense oligonucleotide (ASO1) injections rescue a murine model of autosomal dominant centronuclear myopathy (Dnm2SL/+)

Project description:Dnm2 downregulation by antisense oligonucleotide (ASO1) injections rescue a murine model of autosomal dominant centronuclear myopathy (Dnm2SL/+)

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RNA sequencing of control and dnm2-knockout mouse sciatic nerves

Project description:In this study, we analyzed the transcriptome profiles of mouse sciatic nerves after developmental (P1 and P5) or inducible (4 weeks post Tamoxifen) deletion of Dnm2 conditionally in Schwann cells (using a P0Cre-driven or a P0CreERT2-driven recombination of floxed alleles, respectively) as compared to controls (floxed Dnm2 homozygous, Cre-negative).

2019-01-16 | GSE113106 | GEO

Reducing dynamin 2 (DNM2) rescues <i>DNM2</i>-related dominant centronuclear myopathy.

Project description:Centronuclear myopathies (CNM) are a group of severe muscle diseases for which no effective therapy is currently available. We have previously shown that reduction of the large GTPase DNM2 in a mouse model of the X-linked form, due to loss of myotubularin phosphatase MTM1, prevents the development of the skeletal muscle pathophysiology. As DNM2 is mutated in autosomal dominant forms, here we tested whether DNM2 reduction can rescue DNM2-related CNM in a knock-in mouse harboring the p.R465W mutation (Dnm2 RW/+) and displaying a mild CNM phenotype similar to patients with the same mutation. A single intramuscular injection of adeno-associated virus-shRNA targeting Dnm2 resulted in reduction in protein levels 5 wk post injection, with a corresponding improvement in muscle mass and fiber size distribution, as well as an improvement in histopathological CNM features. To establish a systemic treatment, weekly i.p. injections of antisense oligonucleotides targeting Dnm2 were administered to Dnm2 RW/+mice for 5 wk. While muscle mass, histopathology, and muscle ultrastructure were perturbed in Dnm2 RW/+mice compared with wild-type mice, these features were indistinguishable from wild-type mice after reducing DNM2. Therefore, DNM2 knockdown via two different strategies can efficiently correct the myopathy due to DNM2 mutations, and it provides a common therapeutic strategy for several forms of centronuclear myopathy. Furthermore, we provide an example of treating a dominant disease by targeting both alleles, suggesting that this strategy may be applied to other dominant diseases.

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Transcriptomic analysis of the soleus muscle extracted from Dnm2 K562E point-mutant heterozygous mice

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Some DNM2 mutations cause extremely severe congenital myopathy and phenocopy myotubular myopathy.

Project description: Not available

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Effect of Bmal1 depletion on gene expression in poly(I:C)-stimulated myeloid peritoneal cells.

Project description:To investigate the effect of Bmal1 on poly(I:C) response, we isolate mRNA from PBS or poly(I:C)-injected WT and myeloid Bmal1 KO mice. We then compare the poly(I:C)-induced transcriptomic change between WT and Bmal1 KO peritoneal myeloid cells.

2024-01-31 | GSE222759 | GEO

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