Protocol for measuring force in skinned diaphragm muscle fibers of myopathic SEPN1 knockout mice following chronic tauroursodeoxycholic acid treatment

Summary: Selenoprotein N1 (SEPN1) is a type II endoplasmic reticulum (ER) glycoprotein. Loss-of-function mutations in the gene encoding for SEPN1 give rise to myopathy. Here, we present a protocol for evaluating the contractility of diaphragmatic muscle fibers of SEPN1 knockout mice following chroni...

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Main Authors: Leonardo Nogara, Serena Germani, Cosimo De Napoli, Bert Blaauw, Ester Zito
Format: Article
Language:English
Published: Elsevier 2025-09-01
Series:STAR Protocols
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Online Access:http://www.sciencedirect.com/science/article/pii/S2666166725003247
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author Leonardo Nogara
Serena Germani
Cosimo De Napoli
Bert Blaauw
Ester Zito
author_facet Leonardo Nogara
Serena Germani
Cosimo De Napoli
Bert Blaauw
Ester Zito
author_sort Leonardo Nogara
collection DOAJ
description Summary: Selenoprotein N1 (SEPN1) is a type II endoplasmic reticulum (ER) glycoprotein. Loss-of-function mutations in the gene encoding for SEPN1 give rise to myopathy. Here, we present a protocol for evaluating the contractility of diaphragmatic muscle fibers of SEPN1 knockout mice following chronic treatment with tauroursodeoxycholic acid (TUDCA). We describe steps for genotyping SEPN1 knockout mice, TUDCA in vivo treatment, diaphragm dissection, and chemical permeabilization. We then detail procedures for single muscle fiber isolation and tension measurement.For complete details on the use and execution of this protocol, please refer to Germani et al.1 : Publisher’s note: Undertaking any experimental protocol requires adherence to local institutional guidelines for laboratory safety and ethics.
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publishDate 2025-09-01
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spelling doaj-art-eb75f636a34a4e4ab7ada181f75703552025-08-20T03:32:12ZengElsevierSTAR Protocols2666-16672025-09-016310391810.1016/j.xpro.2025.103918Protocol for measuring force in skinned diaphragm muscle fibers of myopathic SEPN1 knockout mice following chronic tauroursodeoxycholic acid treatmentLeonardo Nogara0Serena Germani1Cosimo De Napoli2Bert Blaauw3Ester Zito4Department of Biomedical Sciences, University of Padua, Padua, Italy; Department of Pharmaceutical Sciences, University of Padua, Padua, Italy; Corresponding authorIstituto di Ricerche Farmacologiche Mario Negri IRCCS, Milan, Italy; Corresponding authorDepartment of Biomedical Sciences, University of Padua, Padua, ItalyDepartment of Biomedical Sciences, University of Padua, Padua, Italy; Veneto Institute of Molecular Medicine (VIMM), Padua, ItalyIstituto di Ricerche Farmacologiche Mario Negri IRCCS, Milan, Italy; Department of Biomolecular Sciences, University of Urbino Carlo Bo, Urbino, Italy; Corresponding authorSummary: Selenoprotein N1 (SEPN1) is a type II endoplasmic reticulum (ER) glycoprotein. Loss-of-function mutations in the gene encoding for SEPN1 give rise to myopathy. Here, we present a protocol for evaluating the contractility of diaphragmatic muscle fibers of SEPN1 knockout mice following chronic treatment with tauroursodeoxycholic acid (TUDCA). We describe steps for genotyping SEPN1 knockout mice, TUDCA in vivo treatment, diaphragm dissection, and chemical permeabilization. We then detail procedures for single muscle fiber isolation and tension measurement.For complete details on the use and execution of this protocol, please refer to Germani et al.1 : Publisher’s note: Undertaking any experimental protocol requires adherence to local institutional guidelines for laboratory safety and ethics.http://www.sciencedirect.com/science/article/pii/S2666166725003247Biotechnology and bioengineeringHealth SciencesModel Organisms
spellingShingle Leonardo Nogara
Serena Germani
Cosimo De Napoli
Bert Blaauw
Ester Zito
Protocol for measuring force in skinned diaphragm muscle fibers of myopathic SEPN1 knockout mice following chronic tauroursodeoxycholic acid treatment
STAR Protocols
Biotechnology and bioengineering
Health Sciences
Model Organisms
title Protocol for measuring force in skinned diaphragm muscle fibers of myopathic SEPN1 knockout mice following chronic tauroursodeoxycholic acid treatment
title_full Protocol for measuring force in skinned diaphragm muscle fibers of myopathic SEPN1 knockout mice following chronic tauroursodeoxycholic acid treatment
title_fullStr Protocol for measuring force in skinned diaphragm muscle fibers of myopathic SEPN1 knockout mice following chronic tauroursodeoxycholic acid treatment
title_full_unstemmed Protocol for measuring force in skinned diaphragm muscle fibers of myopathic SEPN1 knockout mice following chronic tauroursodeoxycholic acid treatment
title_short Protocol for measuring force in skinned diaphragm muscle fibers of myopathic SEPN1 knockout mice following chronic tauroursodeoxycholic acid treatment
title_sort protocol for measuring force in skinned diaphragm muscle fibers of myopathic sepn1 knockout mice following chronic tauroursodeoxycholic acid treatment
topic Biotechnology and bioengineering
Health Sciences
Model Organisms
url http://www.sciencedirect.com/science/article/pii/S2666166725003247
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