Quality of sequences obtained with CASAVA 1.8.1 (Illumina) workflow. PF reads passing Illumina chastity filter. (XLSX 46 kb

Christelle Etard

Marco Ferg

Olivier Armant

Urmas Roostalu

Uwe Strähle

Victor Gourain

PubMed

Crossref

Loss of function of myosin chaperones triggers Hsf1-mediated transcriptional response in skeletal muscle cells

BACKGROUND: Mutations in myosin chaperones Unc45b and Hsp90aa1.1 as well as in the Unc45b-binding protein Smyd1b impair formation of myofibrils in skeletal muscle and lead to the accumulation of misfolded myosin. The concomitant transcriptional response involves up-regulation of the three genes encoding these proteins, as well as genes involved in muscle development. The transcriptional up-regulation of unc45b, hsp90aa1.1 and smyd1b is specific to zebrafish mutants with myosin folding defects, and is not triggered in other zebrafish myopathy models. RESULTS: By dissecting the promoter of unc45b, we identify a Heat shock factor 1 (Hsf1) binding element as a mediator of unc45b up-regulation in myofibers lacking myosin folding proteins. Loss-of-function of Hsf1 abolishes unc45b up-regulation in mutants with defects in myosin folding. CONCLUSIONS: Taken together, our data show that skeletal muscle cells respond to defective myosin chaperones with a complex gene program and suggest that this response is mediated by Hsf1 activation

Etard, Christelle

Armant, Olivier

Roostalu, Urmas

Gourain, Victor

Ferg, Marco

Strähle, Uwe

The University of Manchester - Institutional Repository

Loss of function of myosin chaperones triggers Hsf1-mediated transcriptional response in skeletal muscle cells.

Christelle Etard (3538202)

Olivier Armant (63733)

Urmas Roostalu (3538199)

Victor Gourain (3538193)

Marco Ferg (693553)

Uwe StrĂ¤hle (3538196)

The Francis Crick Institute

Additional file 1: Table S1. of Loss of function of myosin chaperones triggers Hsf1-mediated transcriptional response in skeletal muscle cells

The 1411 genes regulated in the unc45b mutant compared with wild-type embryos at 72 hpf with FDR  1.5. (XLSX 120 kb

Uwe Strähle (38270)

Additional file 3: Table S2. of Loss of function of myosin chaperones triggers Hsf1-mediated transcriptional response in skeletal muscle cells

Springer - Publisher Connector

https://figshare.com/articles/Additional_file_1_Table_S1_of_Loss_of_function_of_myosin_chaperones_triggers_Hsf1-mediated_transcriptional_response_in_skeletal_muscle_cells/4426550

Loss of function of myosin chaperones triggers Hsf1-mediated transcriptional response in skeletal muscle cells

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The University of Manchester - Institutional Repository

The University of Manchester - Institutional Repository

The Francis Crick Institute

The Francis Crick Institute

Springer - Publisher Connector