Data from the European registry for patients with McArdle disease and other muscle glycogenoses (EUROMAC)

The European registry for patients with McArdle disease and other muscle glycogenoses (EUROMAC) was launched to register rare muscle glycogenoses in Europe, to facilitate recruitment for research trials and to learn about the phenotypes and disseminate knowledge about the diseases through workshops...

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Autores: Scalco, Renata S., Lucia, Alejandro|||0000-0002-3025-2060, Santalla, Alfredo|||0000-0001-9418-0564, Martinuzzi, Andrea, Vavla, Marinela, Reni, Gianluigi, Toscano, Antonio, Musumeci, Olimpia|||0000-0002-9208-1527, Voermans, Nicol C.|||0000-0002-5837-7295, Kouwenberg, Carlyn V., Laforêt, Pascal, San-Millán, Beatriz, Vieitez, Irene, Siciliano, Gabriele, Kühnle, Enrico, Trost, Rebeca, Sacconi, Sabrina, Stemmerik, Mads G., Durmus, Hacer, Kierdaszuk, Biruta, Wakelin, Andrew, Andreu Périz, Antoni Lluís, Pinós Figueras, Tomàs|||0000-0002-4379-0917, Martí, Ramon A.|||0000-0002-8273-9540, Quinlivan, Ros, Vissing, John|||0000-0001-6144-8544
Tipo de recurso: artículo
Fecha de publicación:2020
País:España
Institución:Universitat Autònoma de Barcelona
Repositorio:Dipòsit Digital de Documents de la UAB
Idioma:inglés
OAI Identifier:oai:ddd.uab.cat:253126
Acceso en línea:https://ddd.uab.cat/record/253126
https://dx.doi.org/urn:doi:10.1186/s13023-020-01562-x
Access Level:acceso abierto
Palabra clave:Myopathy
Rare diseases
International registry
McArdle disease
Metabolic diseases
Glycogen storage disease
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spelling Data from the European registry for patients with McArdle disease and other muscle glycogenoses (EUROMAC)Scalco, Renata S.Lucia, Alejandro|||0000-0002-3025-2060Santalla, Alfredo|||0000-0001-9418-0564Martinuzzi, AndreaVavla, MarinelaReni, GianluigiToscano, AntonioMusumeci, Olimpia|||0000-0002-9208-1527Voermans, Nicol C.|||0000-0002-5837-7295Kouwenberg, Carlyn V.Laforêt, PascalSan-Millán, BeatrizVieitez, IreneSiciliano, GabrieleKühnle, EnricoTrost, RebecaSacconi, SabrinaStemmerik, Mads G.Durmus, HacerKierdaszuk, BirutaWakelin, AndrewAndreu Périz, Antoni LluísPinós Figueras, Tomàs|||0000-0002-4379-0917Martí, Ramon A.|||0000-0002-8273-9540Quinlivan, RosVissing, John|||0000-0001-6144-8544MyopathyRare diseasesInternational registryMcArdle diseaseMetabolic diseasesGlycogen storage diseaseThe European registry for patients with McArdle disease and other muscle glycogenoses (EUROMAC) was launched to register rare muscle glycogenoses in Europe, to facilitate recruitment for research trials and to learn about the phenotypes and disseminate knowledge about the diseases through workshops and websites. A network of twenty full and collaborating partners from eight European countries and the US contributed data on rare muscle glycogenosis in the EUROMAC registry. After approximately 3 years of data collection, the data in the registry was analysed. Of 282 patients with confirmed diagnoses of muscle glycogenosis, 269 had McArdle disease. New phenotypic features of McArdle disease were suggested, including a higher frequency (51.4%) of fixed weakness than reported before, normal CK values in a minority of patients (6.8%), ptosis in 8 patients, body mass index above background population and number of comorbidities with a higher frequency than in the background population (hypothyroidism, coronary heart disease). The EUROMAC project and registry have provided insight into new phenotypic features of McArdle disease and the variety of co-comorbidities affecting people with McArdle disease. This should lead to better management of these disorders in the future, including controlling weight, and preventive screening for thyroid and coronary artery diseases, as well as physical examination with attention on occurrence of ptosis and fixed muscle weakness. Normal serum creatine kinase in a minority of patients stresses the need to not discard a diagnosis of McArdle disease even though creatine kinase is normal and episodes of myoglobinuria are absent.Universitat Autònoma de Barcelona 22020-01-0120202020-01-01Articlehttp://purl.org/coar/resource_type/c_6501VoRhttp://purl.org/coar/version/c_970fb48d4fbd8a85info:eu-repo/semantics/articleapplication/pdfhttps://ddd.uab.cat/record/253126https://dx.doi.org/urn:doi:10.1186/s13023-020-01562-xreponame:Dipòsit Digital de Documents de la UABinstname:Universitat Autònoma de BarcelonaInglésengMinisterio de Economía y Competitividad https://doi.org/10.13039/501100003329 PI16/01492Instituto de Salud Carlos III https://doi.org/10.13039/501100004587 PI19/01313open accesshttp://purl.org/coar/access_right/c_abf2Aquest document està subjecte a una llicència d'ús Creative Commons. Es permet la reproducció total o parcial, la distribució, la comunicació pública de l'obra i la creació d'obres derivades, fins i tot amb finalitats comercials, sempre i quan es reconegui l'autoria de l'obra original.https://creativecommons.org/licenses/by/4.0/info:eu-repo/semantics/openAccessoai:ddd.uab.cat:2531262026-06-06T12:50:31Z
dc.title.none.fl_str_mv Data from the European registry for patients with McArdle disease and other muscle glycogenoses (EUROMAC)
title Data from the European registry for patients with McArdle disease and other muscle glycogenoses (EUROMAC)
spellingShingle Data from the European registry for patients with McArdle disease and other muscle glycogenoses (EUROMAC)
Scalco, Renata S.
Myopathy
Rare diseases
International registry
McArdle disease
Metabolic diseases
Glycogen storage disease
title_short Data from the European registry for patients with McArdle disease and other muscle glycogenoses (EUROMAC)
title_full Data from the European registry for patients with McArdle disease and other muscle glycogenoses (EUROMAC)
title_fullStr Data from the European registry for patients with McArdle disease and other muscle glycogenoses (EUROMAC)
title_full_unstemmed Data from the European registry for patients with McArdle disease and other muscle glycogenoses (EUROMAC)
title_sort Data from the European registry for patients with McArdle disease and other muscle glycogenoses (EUROMAC)
dc.creator.none.fl_str_mv Scalco, Renata S.
Lucia, Alejandro|||0000-0002-3025-2060
Santalla, Alfredo|||0000-0001-9418-0564
Martinuzzi, Andrea
Vavla, Marinela
Reni, Gianluigi
Toscano, Antonio
Musumeci, Olimpia|||0000-0002-9208-1527
Voermans, Nicol C.|||0000-0002-5837-7295
Kouwenberg, Carlyn V.
Laforêt, Pascal
San-Millán, Beatriz
Vieitez, Irene
Siciliano, Gabriele
Kühnle, Enrico
Trost, Rebeca
Sacconi, Sabrina
Stemmerik, Mads G.
Durmus, Hacer
Kierdaszuk, Biruta
Wakelin, Andrew
Andreu Périz, Antoni Lluís
Pinós Figueras, Tomàs|||0000-0002-4379-0917
Martí, Ramon A.|||0000-0002-8273-9540
Quinlivan, Ros
Vissing, John|||0000-0001-6144-8544
author Scalco, Renata S.
author_facet Scalco, Renata S.
Lucia, Alejandro|||0000-0002-3025-2060
Santalla, Alfredo|||0000-0001-9418-0564
Martinuzzi, Andrea
Vavla, Marinela
Reni, Gianluigi
Toscano, Antonio
Musumeci, Olimpia|||0000-0002-9208-1527
Voermans, Nicol C.|||0000-0002-5837-7295
Kouwenberg, Carlyn V.
Laforêt, Pascal
San-Millán, Beatriz
Vieitez, Irene
Siciliano, Gabriele
Kühnle, Enrico
Trost, Rebeca
Sacconi, Sabrina
Stemmerik, Mads G.
Durmus, Hacer
Kierdaszuk, Biruta
Wakelin, Andrew
Andreu Périz, Antoni Lluís
Pinós Figueras, Tomàs|||0000-0002-4379-0917
Martí, Ramon A.|||0000-0002-8273-9540
Quinlivan, Ros
Vissing, John|||0000-0001-6144-8544
author_role author
author2 Lucia, Alejandro|||0000-0002-3025-2060
Santalla, Alfredo|||0000-0001-9418-0564
Martinuzzi, Andrea
Vavla, Marinela
Reni, Gianluigi
Toscano, Antonio
Musumeci, Olimpia|||0000-0002-9208-1527
Voermans, Nicol C.|||0000-0002-5837-7295
Kouwenberg, Carlyn V.
Laforêt, Pascal
San-Millán, Beatriz
Vieitez, Irene
Siciliano, Gabriele
Kühnle, Enrico
Trost, Rebeca
Sacconi, Sabrina
Stemmerik, Mads G.
Durmus, Hacer
Kierdaszuk, Biruta
Wakelin, Andrew
Andreu Périz, Antoni Lluís
Pinós Figueras, Tomàs|||0000-0002-4379-0917
Martí, Ramon A.|||0000-0002-8273-9540
Quinlivan, Ros
Vissing, John|||0000-0001-6144-8544
author2_role author
author
author
author
author
author
author
author
author
author
author
author
author
author
author
author
author
author
author
author
author
author
author
author
author
dc.contributor.none.fl_str_mv Universitat Autònoma de Barcelona
dc.subject.none.fl_str_mv Myopathy
Rare diseases
International registry
McArdle disease
Metabolic diseases
Glycogen storage disease
topic Myopathy
Rare diseases
International registry
McArdle disease
Metabolic diseases
Glycogen storage disease
description The European registry for patients with McArdle disease and other muscle glycogenoses (EUROMAC) was launched to register rare muscle glycogenoses in Europe, to facilitate recruitment for research trials and to learn about the phenotypes and disseminate knowledge about the diseases through workshops and websites. A network of twenty full and collaborating partners from eight European countries and the US contributed data on rare muscle glycogenosis in the EUROMAC registry. After approximately 3 years of data collection, the data in the registry was analysed. Of 282 patients with confirmed diagnoses of muscle glycogenosis, 269 had McArdle disease. New phenotypic features of McArdle disease were suggested, including a higher frequency (51.4%) of fixed weakness than reported before, normal CK values in a minority of patients (6.8%), ptosis in 8 patients, body mass index above background population and number of comorbidities with a higher frequency than in the background population (hypothyroidism, coronary heart disease). The EUROMAC project and registry have provided insight into new phenotypic features of McArdle disease and the variety of co-comorbidities affecting people with McArdle disease. This should lead to better management of these disorders in the future, including controlling weight, and preventive screening for thyroid and coronary artery diseases, as well as physical examination with attention on occurrence of ptosis and fixed muscle weakness. Normal serum creatine kinase in a minority of patients stresses the need to not discard a diagnosis of McArdle disease even though creatine kinase is normal and episodes of myoglobinuria are absent.
publishDate 2020
dc.date.none.fl_str_mv 2
2020-01-01
2020
2020-01-01
dc.type.none.fl_str_mv Article
http://purl.org/coar/resource_type/c_6501
VoR
http://purl.org/coar/version/c_970fb48d4fbd8a85
dc.type.openaire.fl_str_mv info:eu-repo/semantics/article
format article
dc.identifier.none.fl_str_mv https://ddd.uab.cat/record/253126
https://dx.doi.org/urn:doi:10.1186/s13023-020-01562-x
url https://ddd.uab.cat/record/253126
https://dx.doi.org/urn:doi:10.1186/s13023-020-01562-x
dc.language.none.fl_str_mv Inglés
eng
language_invalid_str_mv Inglés
language eng
dc.relation.none.fl_str_mv Ministerio de Economía y Competitividad https://doi.org/10.13039/501100003329 PI16/01492
Instituto de Salud Carlos III https://doi.org/10.13039/501100004587 PI19/01313
dc.rights.none.fl_str_mv open access
http://purl.org/coar/access_right/c_abf2
https://creativecommons.org/licenses/by/4.0/
dc.rights.openaire.fl_str_mv info:eu-repo/semantics/openAccess
rights_invalid_str_mv open access
http://purl.org/coar/access_right/c_abf2
https://creativecommons.org/licenses/by/4.0/
eu_rights_str_mv openAccess
dc.format.none.fl_str_mv application/pdf
dc.source.none.fl_str_mv reponame:Dipòsit Digital de Documents de la UAB
instname:Universitat Autònoma de Barcelona
instname_str Universitat Autònoma de Barcelona
reponame_str Dipòsit Digital de Documents de la UAB
collection Dipòsit Digital de Documents de la UAB
repository.name.fl_str_mv
repository.mail.fl_str_mv
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