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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.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.issn 1422-0067
dc.identifier.uri http://hdl.handle.net/10230/57221
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.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.uri http://creativecommons.org/licenses/by/4.0/
dc.title Innovative computerized dystrophin quantification method based on spectral confocal microscopy
dc.type info:eu-repo/semantics/article
dc.identifier.doi http://dx.doi.org/10.3390/ijms24076358
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.rights.accessRights info:eu-repo/semantics/openAccess
dc.type.version info:eu-repo/semantics/publishedVersion


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