Innovative computerized dystrophin quantification method based on spectral confocal microscopy
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- dc.contributor.author Codina, Anna
- dc.contributor.author Roldán, Mònica
- dc.contributor.author Natera de Benito, Daniel
- dc.contributor.author Ortez, Carlos
- dc.contributor.author Planas, Robert
- dc.contributor.author Matalonga, Leslie
- dc.contributor.author Cuadras, Daniel
- dc.contributor.author Carrera, Laura
- dc.contributor.author Exposito, Jesica
- dc.contributor.author Marquez, Jesus
- dc.contributor.author Jimenez-Mallebrera, Cecilia
- dc.contributor.author Porta, Josep M.
- dc.contributor.author Nascimento, Andrés
- dc.contributor.author Jou-Muñoz, Cristina
- dc.date.accessioned 2023-06-19T06:05:39Z
- dc.date.available 2023-06-19T06:05:39Z
- dc.date.issued 2023
- dc.description.abstract Several clinical trials are working on drug development for Duchenne and Becker muscular dystrophy (DMD and BMD) treatment, and, since the expected increase in dystrophin is relatively subtle, high-sensitivity quantification methods are necessary. There is also a need to quantify dystrophin to reach a definitive diagnosis in individuals with mild BMD, and in female carriers. We developed a method for the quantification of dystrophin in DMD and BMD patients using spectral confocal microscopy. It offers the possibility to capture the whole emission spectrum for any antibody, ensuring the selection of the emission peak and allowing the detection of fluorescent emissions of very low intensities. Fluorescence was evaluated first on manually selected regions of interest (ROIs), proving the usefulness of the methodology. Later, ROI selection was automated to make it operator-independent. The proposed methodology correctly classified patients according to their diagnosis, detected even minimal traces of dystrophin, and the results obtained automatically were statistically comparable to the manual ones. Thus, spectral imaging could be implemented to measure dystrophin expression and it could pave the way for detailed analysis of how its expression relates to the clinical course. Studies could be further expanded to better understand the expression of dystrophin-associated protein complexes (DAPCs).
- dc.format.mimetype application/pdf
- dc.identifier.citation Codina A, Roldán M, Natera-de Benito D, Ortez C, Planas R, Matalonga L, Cuadras D, Carrera L, Exposito J, Marquez J, Jimenez-Mallebrera C, M Porta J, Nascimento A, Jou C. Innovative computerized dystrophin quantification method based on spectral confocal microscopy. Int J Mol Sci. 2023 Mar 28;24(7):6358. DOI: 10.3390/ijms24076358
- dc.identifier.doi http://dx.doi.org/10.3390/ijms24076358
- dc.identifier.issn 1422-0067
- dc.identifier.uri http://hdl.handle.net/10230/57221
- dc.language.iso eng
- dc.publisher MDPI
- dc.relation.ispartof Int J Mol Sci. 2023 Mar 28;24(7):6358
- dc.rights © 2023 by the authors. Licensee MDPI, Basel, Switzerland. This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution (CC BY) license (https://creativecommons.org/licenses/by/4.0/).
- dc.rights.accessRights info:eu-repo/semantics/openAccess
- dc.rights.uri http://creativecommons.org/licenses/by/4.0/
- dc.subject.keyword Becker muscular dystrophy
- dc.subject.keyword Duchenne muscular dystrophy
- dc.subject.keyword Confocal microscopy
- dc.subject.keyword Dystrophin
- dc.subject.keyword Fluorescence quantification
- dc.subject.keyword Spectral imaging
- dc.title Innovative computerized dystrophin quantification method based on spectral confocal microscopy
- dc.type info:eu-repo/semantics/article
- dc.type.version info:eu-repo/semantics/publishedVersion