{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,10,21]],"date-time":"2025-10-21T00:39:10Z","timestamp":1761007150630,"version":"build-2065373602"},"reference-count":38,"publisher":"Elsevier BV","license":[{"start":{"date-parts":[[2023,11,1]],"date-time":"2023-11-01T00:00:00Z","timestamp":1698796800000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/tdm\/userlicense\/1.0\/"},{"start":{"date-parts":[[2023,11,1]],"date-time":"2023-11-01T00:00:00Z","timestamp":1698796800000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/legal\/tdmrep-license"},{"start":{"date-parts":[[2023,11,1]],"date-time":"2023-11-01T00:00:00Z","timestamp":1698796800000},"content-version":"stm-asf","delay-in-days":0,"URL":"https:\/\/doi.org\/10.15223\/policy-017"},{"start":{"date-parts":[[2023,11,1]],"date-time":"2023-11-01T00:00:00Z","timestamp":1698796800000},"content-version":"stm-asf","delay-in-days":0,"URL":"https:\/\/doi.org\/10.15223\/policy-037"},{"start":{"date-parts":[[2023,11,1]],"date-time":"2023-11-01T00:00:00Z","timestamp":1698796800000},"content-version":"stm-asf","delay-in-days":0,"URL":"https:\/\/doi.org\/10.15223\/policy-012"},{"start":{"date-parts":[[2023,11,1]],"date-time":"2023-11-01T00:00:00Z","timestamp":1698796800000},"content-version":"stm-asf","delay-in-days":0,"URL":"https:\/\/doi.org\/10.15223\/policy-029"},{"start":{"date-parts":[[2023,11,1]],"date-time":"2023-11-01T00:00:00Z","timestamp":1698796800000},"content-version":"stm-asf","delay-in-days":0,"URL":"https:\/\/doi.org\/10.15223\/policy-004"}],"funder":[{"DOI":"10.13039\/501100007380","name":"Funda\u00e7\u00e3o Instituto de Pesquisas Econ\u00f4micas","doi-asserted-by":"publisher","award":["170562"],"award-info":[{"award-number":["170562"]}],"id":[{"id":"10.13039\/501100007380","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100002322","name":"CAPES","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100002322","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100003593","name":"Conselho Nacional de Desenvolvimento Cient\u00edfico e Tecnol\u00f3gico","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100003593","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100004263","name":"Funda\u00e7\u00e3o de Amparo \u00e0 Pesquisa do Estado do Rio Grande do Sul","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100004263","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["clinicalkey.com","clinicalkey.com.au","clinicalkey.es","clinicalkey.fr","clinicalkey.jp","elsevier.com","sciencedirect.com"],"crossmark-restriction":true},"short-container-title":["Cardiovascular Pathology"],"published-print":{"date-parts":[[2023,11]]},"DOI":"10.1016\/j.carpath.2023.107575","type":"journal-article","created":{"date-parts":[[2023,9,18]],"date-time":"2023-09-18T20:14:37Z","timestamp":1695068077000},"page":"107575","update-policy":"https:\/\/doi.org\/10.1016\/elsevier_cm_policy","source":"Crossref","is-referenced-by-count":1,"special_numbering":"C","title":["Characterization of heart disease in mucopolysaccharidosis type II mice"],"prefix":"10.1016","volume":"67","author":[{"given":"Angela Maria Vicente","family":"Tavares","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Esteban Alberto","family":"Gonzalez","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Isabelle Souza","family":"Viana","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Fernanda","family":"Visioli","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Luisa Natalia Pimentel","family":"Vera","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Guilherme","family":"Baldo","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"78","reference":[{"key":"10.1016\/j.carpath.2023.107575_bib0001","unstructured":"Hashmi MS, Gupta V. Mucopolysaccharidosis type II. 2022."},{"key":"10.1016\/j.carpath.2023.107575_bib0002","doi-asserted-by":"crossref","first-page":"66","DOI":"10.1186\/s12881-019-0807-x","article-title":"Case report: a rare case of Hunter syndrome (type II mucopolysaccharidosis) in a girl","volume":"20","author":"Semyachkina","year":"2019","journal-title":"BMC Med Genet"},{"key":"10.1016\/j.carpath.2023.107575_bib0003","doi-asserted-by":"crossref","first-page":"90","DOI":"10.1002\/ajmg.a.34371","article-title":"Retrospective analysis of the clinical manifestations and survival of Korean patients with mucopolysaccharidosis type II: emphasis on the cardiovascular complication and mortality cases","volume":"158 A","author":"Sohn","year":"2012","journal-title":"Am J Med Genet A"},{"key":"10.1016\/j.carpath.2023.107575_bib0004","doi-asserted-by":"crossref","first-page":"1643","DOI":"10.1093\/hmg\/ddx069","article-title":"Perturbations in cell signaling elicit early cardiac defects in mucopolysaccharidosis type II","volume":"26","author":"Costa","year":"2017","journal-title":"Hum Mol Genet"},{"key":"10.1016\/j.carpath.2023.107575_bib0005","doi-asserted-by":"crossref","first-page":"290","DOI":"10.1007\/s00380-018-1242-1","article-title":"Aortic root dilatation in patients with mucopolysaccharidoses and the impact of enzyme replacement therapy","volume":"34","author":"Poswar F de","year":"2019","journal-title":"Heart Vessels"},{"key":"10.1016\/j.carpath.2023.107575_bib0006","doi-asserted-by":"crossref","first-page":"924","DOI":"10.1007\/s10545-007-0641-8","article-title":"The characterization of a murine model of mucopolysaccharidosis II (Hunter syndrome)","volume":"30","author":"Garcia","year":"2007","journal-title":"J Inherit Metab Dis"},{"key":"10.1016\/j.carpath.2023.107575_bib0007","doi-asserted-by":"crossref","first-page":"124","DOI":"10.1186\/s13052-018-0564-z","article-title":"New treatments for the mucopolysaccharidoses: from pathophysiology to therapy","volume":"44","author":"Fecarotta","year":"2018","journal-title":"Ital J Pediatr"},{"key":"10.1016\/j.carpath.2023.107575_bib0008","doi-asserted-by":"crossref","DOI":"10.3389\/fcvm.2021.801147","article-title":"Progression of cardiovascular manifestations in adults and children with mucopolysaccharidoses with and without enzyme replacement therapy","volume":"8","author":"Poswar","year":"2022","journal-title":"Front Cardiovasc Med"},{"key":"10.1016\/j.carpath.2023.107575_bib0009","doi-asserted-by":"crossref","DOI":"10.1016\/j.carpath.2017.01.001","article-title":"Progressive heart disease in mucopolysaccharidosis type I mice may be mediated by increased cathepsin B activity","volume":"27","author":"Baldo","year":"2017","journal-title":"Cardiovasc Pathol"},{"key":"10.1016\/j.carpath.2023.107575_bib0010","doi-asserted-by":"crossref","first-page":"102","DOI":"10.1016\/j.lfs.2018.01.020","article-title":"Cathepsin B inhibition attenuates cardiovascular pathology in mucopolysaccharidosis I mice","volume":"196","author":"Gonzalez","year":"2018","journal-title":"Life Sci"},{"key":"10.1016\/j.carpath.2023.107575_bib0011","doi-asserted-by":"crossref","DOI":"10.1016\/j.ymgme.2013.03.005","article-title":"Enzyme replacement therapy started at birth improves outcome in difficult-to-treat organs in mucopolysaccharidosis I mice","volume":"109","author":"Baldo","year":"2013","journal-title":"Mol Genet Metab"},{"key":"10.1016\/j.carpath.2023.107575_bib0012","doi-asserted-by":"crossref","DOI":"10.1007\/s11011-017-0110-5","article-title":"Aversive and non-aversive memory impairment in the mucopolysaccharidosis II mouse model","volume":"33","author":"Azambuja","year":"2018","journal-title":"Metab Brain Dis"},{"key":"10.1016\/j.carpath.2023.107575_bib0013","doi-asserted-by":"crossref","DOI":"10.1161\/01.CIR.65.5.962","article-title":"Two-dimensional echocardiographic assessment of left ventricular volumes and ejection fraction in children","volume":"65","author":"Mercier","year":"1982","journal-title":"Circulation"},{"key":"10.1016\/j.carpath.2023.107575_bib0014","doi-asserted-by":"crossref","DOI":"10.1002\/9780470942390.mo100130","article-title":"Echocardiography in mice","volume":"1","author":"Gao","year":"2011","journal-title":"Curr Protoc Mouse Biol"},{"key":"10.1016\/j.carpath.2023.107575_bib0015","article-title":"Early loss of cardiac function in acute myocardial infarction is associated with redox imbalance","volume":"17","author":"Tavares","year":"2012","journal-title":"Exp Clin Cardiol"},{"key":"10.1016\/j.carpath.2023.107575_bib0016","doi-asserted-by":"crossref","DOI":"10.1152\/ajpheart.00979.2001","article-title":"Serial noninvasive assessment of progressive pulmonary hypertension in a rat model","volume":"283","author":"Jones","year":"2002","journal-title":"Am J Physiol Heart Circ Physiol"},{"key":"10.1016\/j.carpath.2023.107575_bib0017","doi-asserted-by":"crossref","DOI":"10.1016\/j.ymgme.2016.09.005","article-title":"Glycosaminoglycans detection methods: applications of mass spectrometry","volume":"120","author":"Kubaski","year":"2017","journal-title":"Mol Genet Metab"},{"key":"10.1016\/j.carpath.2023.107575_bib0018","doi-asserted-by":"crossref","DOI":"10.1016\/S0021-9258(19)52451-6","article-title":"Protein measurement with the Folin phenol reagent","volume":"193","author":"Lowry","year":"1951","journal-title":"J Biol Chem"},{"key":"10.1016\/j.carpath.2023.107575_bib0019","doi-asserted-by":"crossref","DOI":"10.3390\/ijms21041258","article-title":"Mucopolysaccharidosis type II: one hundred years of research, diagnosis, and treatment","volume":"21","author":"D\u02b9avanzo","year":"2020","journal-title":"Int J Mol Sci"},{"key":"10.1016\/j.carpath.2023.107575_bib0020","doi-asserted-by":"crossref","DOI":"10.1007\/s10545-011-9359-8","article-title":"Cardiac disease in patients with mucopolysaccharidosis: presentation, diagnosis and management","volume":"34","author":"Braunlin","year":"2011","journal-title":"J Inherit Metab Dis"},{"key":"10.1016\/j.carpath.2023.107575_bib0021","doi-asserted-by":"crossref","DOI":"10.3390\/ijms22157888","article-title":"Differences in MPS I and MPS II disease manifestations","volume":"22","author":"Hampe","year":"2021","journal-title":"Int J Mol Sci"},{"key":"10.1016\/j.carpath.2023.107575_bib0022","doi-asserted-by":"crossref","DOI":"10.1186\/s13023-022-02396-5","article-title":"Cardiac involvement in MPS patients: incidence and response to therapy in an Italian multicentre study","volume":"17","author":"Sestito","year":"2022","journal-title":"Orphanet J Rare Dis"},{"key":"10.1016\/j.carpath.2023.107575_bib0023","article-title":"Presentation and treatments for mucopolysaccharidosis type II (MPS II; Hunter Syndrome)","volume":"5","author":"Stapleton","year":"2017","journal-title":"Expert Opin Orphan Drugs"},{"key":"10.1016\/j.carpath.2023.107575_bib0024","doi-asserted-by":"crossref","DOI":"10.1017\/S104795110999062X","article-title":"Echocardiographic study of paediatric patients with mucopolysaccharidosis","volume":"20","author":"Leal","year":"2010","journal-title":"Cardiol Young"},{"key":"10.1016\/j.carpath.2023.107575_bib0025","article-title":"Cardiovascular abnormalities in Egyptian children with mucopolysaccharidoses","volume":"10","author":"Selim","year":"2016","journal-title":"J Clin Diagn Res"},{"issue":"1","key":"10.1016\/j.carpath.2023.107575_bib0035","doi-asserted-by":"crossref","first-page":"117","DOI":"10.36469\/jheor.2022.33801","article-title":"Clinical characteristics and healthcare resource utilization for patients with mucopolysaccharidosis II (MPS II) in the United States: a retrospective chart review","volume":"9","author":"Ayodele","year":"2022","journal-title":"J Health Econ Outcomes Res"},{"issue":"1","key":"10.1016\/j.carpath.2023.107575_bib0036","doi-asserted-by":"crossref","first-page":"323","DOI":"10.1186\/s13023-021-01875-5","article-title":"A review of the clinical outcomes in idursulfase-treated and untreated Filipino patients with mucopolysaccharidosis type II: data from the local lysosomal storage disease registry","volume":"16","author":"Racoma","year":"2021","journal-title":"Orphanet J Rare Dis"},{"key":"10.1016\/j.carpath.2023.107575_bib0037","doi-asserted-by":"crossref","first-page":"71","DOI":"10.1016\/bs.accb.2019.09.001","article-title":"Mucopolysaccharidosis type II (Hunter syndrome): clinical and biochemical aspects of the disease and approaches to its diagnosis and treatment","volume":"77","author":"Mohamed","year":"2020","journal-title":"Adv Carbohydr Chem Biochem"},{"issue":"2","key":"10.1016\/j.carpath.2023.107575_bib0038","doi-asserted-by":"crossref","first-page":"327","DOI":"10.1016\/j.jpeds.2011.01.054","article-title":"Prevalence and characterization of cardiac involvement in Hunter syndrome","volume":"159","author":"Kampmann","year":"2011","journal-title":"J Pediatr"},{"issue":"23-24","key":"10.1016\/j.carpath.2023.107575_bib0039","doi-asserted-by":"crossref","first-page":"1279","DOI":"10.1089\/hum.2022.141","article-title":"Phenotypic correction of murine mucopolysaccharidosis type II by engraftment of ex vivo lentiviral vector-transduced hematopoietic stem and progenitor cells","volume":"33","author":"Smith","year":"2022","journal-title":"Hum Gene Ther"},{"key":"10.1016\/j.carpath.2023.107575_bib0026","doi-asserted-by":"crossref","DOI":"10.1038\/s41434-019-0113-4","article-title":"Neonatal nonviral gene editing with the CRISPR\/Cas9 system improves some cardiovascular, respiratory, and bone disease features of the mucopolysaccharidosis I phenotype in mice","volume":"27","author":"Schuh","year":"2020","journal-title":"Gene Ther"},{"key":"10.1016\/j.carpath.2023.107575_bib0027","doi-asserted-by":"crossref","first-page":"170","DOI":"10.1017\/S1047951109003576","article-title":"The natural course and the impact of therapies of cardiac involvement in the mucopolysaccharidoses","volume":"19","author":"Fesslov\u00e1","year":"2009","journal-title":"Cardiol Young"},{"key":"10.1016\/j.carpath.2023.107575_bib0028","doi-asserted-by":"crossref","DOI":"10.1016\/j.cellsig.2020.109770","article-title":"Signalling pathways linking cysteine cathepsins to adverse cardiac remodelling","volume":"76","author":"O'Toole","year":"2020","journal-title":"Cell Signal"},{"key":"10.1016\/j.carpath.2023.107575_bib0030","doi-asserted-by":"crossref","DOI":"10.3390\/ijms21072515","article-title":"Pathogenesis of mucopolysaccharidoses, an update","volume":"21","author":"Fecarotta","year":"2020","journal-title":"Int J Mol Sci"},{"key":"10.1016\/j.carpath.2023.107575_bib0031","doi-asserted-by":"crossref","DOI":"10.3390\/cells9071679","article-title":"The ins and outs of cathepsins: physiological function and role in disease management","volume":"9","author":"Yadati","year":"2020","journal-title":"Cells"},{"key":"10.1016\/j.carpath.2023.107575_bib0032","doi-asserted-by":"crossref","DOI":"10.1016\/j.bbapap.2020.140360","article-title":"Cysteinyl cathepsins in cardiovascular diseases","volume":"1868","author":"Zhang","year":"2020","journal-title":"Biochim Biophys Acta Proteins Proteom"},{"key":"10.1016\/j.carpath.2023.107575_bib0033","doi-asserted-by":"crossref","DOI":"10.1161\/CIRCULATIONAHA.111.066712","article-title":"Role for cysteine protease cathepsins in heart disease: focus on biology and mechanisms with clinical implication","volume":"125","author":"Cheng","year":"2012","journal-title":"Circulation"},{"issue":"4","key":"10.1016\/j.carpath.2023.107575_bib0034","doi-asserted-by":"crossref","DOI":"10.3390\/cells9040979","article-title":"Cathepsins in the pathophysiology of mucopolysaccharidoses: new perspectives for therapy","volume":"9","author":"de Pasquale","year":"2020","journal-title":"Cells"}],"container-title":["Cardiovascular Pathology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S1054880723000595?httpAccept=text\/xml","content-type":"text\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S1054880723000595?httpAccept=text\/plain","content-type":"text\/plain","content-version":"vor","intended-application":"text-mining"}],"deposited":{"date-parts":[[2025,10,20]],"date-time":"2025-10-20T11:47:50Z","timestamp":1760960870000},"score":1,"resource":{"primary":{"URL":"https:\/\/linkinghub.elsevier.com\/retrieve\/pii\/S1054880723000595"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2023,11]]},"references-count":38,"alternative-id":["S1054880723000595"],"URL":"https:\/\/doi.org\/10.1016\/j.carpath.2023.107575","relation":{},"ISSN":["1054-8807"],"issn-type":[{"type":"print","value":"1054-8807"}],"subject":[],"published":{"date-parts":[[2023,11]]},"assertion":[{"value":"Elsevier","name":"publisher","label":"This article is maintained by"},{"value":"Characterization of heart disease in mucopolysaccharidosis type II mice","name":"articletitle","label":"Article Title"},{"value":"Cardiovascular Pathology","name":"journaltitle","label":"Journal Title"},{"value":"https:\/\/doi.org\/10.1016\/j.carpath.2023.107575","name":"articlelink","label":"CrossRef DOI link to publisher maintained version"},{"value":"article","name":"content_type","label":"Content Type"},{"value":"\u00a9 2023 Elsevier Inc. All rights reserved.","name":"copyright","label":"Copyright"}],"article-number":"107575"}}