{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,1,22]],"date-time":"2026-01-22T02:53:03Z","timestamp":1769050383568,"version":"3.49.0"},"reference-count":41,"publisher":"Public Library of Science (PLoS)","issue":"5","license":[{"start":{"date-parts":[[2018,5,24]],"date-time":"2018-05-24T00:00:00Z","timestamp":1527120000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/100000050","name":"National Heart, Lung, and Blood Institute","doi-asserted-by":"publisher","award":["1 R01 HL107406-01"],"award-info":[{"award-number":["1 R01 HL107406-01"]}],"id":[{"id":"10.13039\/100000050","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100002651","name":"Barth Syndrome Foundation","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100002651","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100002651","name":"Barth Syndrome Foundation","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100002651","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100006108","name":"National Center for Advancing Translational Sciences","doi-asserted-by":"publisher","award":["UL1 TR002345"],"award-info":[{"award-number":["UL1 TR002345"]}],"id":[{"id":"10.13039\/100006108","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["www.plosone.org"],"crossmark-restriction":false},"short-container-title":["PLoS ONE"],"DOI":"10.1371\/journal.pone.0197776","type":"journal-article","created":{"date-parts":[[2018,5,24]],"date-time":"2018-05-24T17:56:50Z","timestamp":1527184610000},"page":"e0197776","update-policy":"https:\/\/doi.org\/10.1371\/journal.pone.corrections_policy","source":"Crossref","is-referenced-by-count":19,"title":["Peak oxygen uptake (VO2peak) across childhood, adolescence and young adulthood in Barth syndrome: Data from cross-sectional and longitudinal studies"],"prefix":"10.1371","volume":"13","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-2329-7623","authenticated-orcid":true,"given":"William Todd","family":"Cade","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kathryn L.","family":"Bohnert","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Dominic N.","family":"Reeds","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Linda R.","family":"Peterson","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Adam J.","family":"Bittel","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Adil","family":"Bashir","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Barry J.","family":"Byrne","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Carolyn L.","family":"Taylor","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"340","published-online":{"date-parts":[[2018,5,24]]},"reference":[{"key":"ref1","unstructured":"Foundation BS. About Barth Syndrome-Frequently Asked Questions 2018. <ext-link xmlns:xlink=\"http:\/\/www.w3.org\/1999\/xlink\" ext-link-type=\"uri\" xlink:href=\"https:\/\/www.barthsyndrome.org\/about-barth-syndrome\" xlink:type=\"simple\">https:\/\/www.barthsyndrome.org\/about-barth-syndrome<\/ext-link>."},{"key":"ref2","doi-asserted-by":"crossref","first-page":"23","DOI":"10.1186\/1750-1172-8-23","article-title":"Barth syndrome","volume":"8","author":"SL Clarke","year":"2013","journal-title":"Orphanet J Rare Dis"},{"issue":"1\u20133","key":"ref3","doi-asserted-by":"crossref","first-page":"327","DOI":"10.1016\/0022-510X(83)90209-5","article-title":"An X-linked mitochondrial disease affecting cardiac muscle, skeletal muscle and neutrophil leucocytes","volume":"62","author":"PG Barth","year":"1983","journal-title":"J Neurol Sci"},{"issue":"4","key":"ref4","doi-asserted-by":"crossref","first-page":"385","DOI":"10.1038\/ng0496-385","article-title":"A novel X-linked gene, G4.5. is responsible for Barth syndrome","volume":"12","author":"S Bione","year":"1996","journal-title":"Nat Genet"},{"issue":"6","key":"ref5","doi-asserted-by":"crossref","first-page":"616","DOI":"10.1038\/nm.3545","article-title":"Modeling the mitochondrial cardiomyopathy of Barth syndrome with induced pluripotent stem cell and heart-on-chip technologies","volume":"20","author":"G Wang","year":"2014","journal-title":"Nature medicine"},{"issue":"8","key":"ref6","doi-asserted-by":"crossref","first-page":"641","DOI":"10.1038\/nchembio.2113","article-title":"Loss of protein association causes cardiolipin degradation in Barth syndrome","volume":"12","author":"Y Xu","year":"2016","journal-title":"Nature chemical biology"},{"issue":"46","key":"ref7","doi-asserted-by":"crossref","first-page":"43553","DOI":"10.1074\/jbc.C200551200","article-title":"Gluing the respiratory chain together. Cardiolipin is required for supercomplex formation in the inner mitochondrial membrane","volume":"277","author":"M Zhang","year":"2002","journal-title":"J Biol Chem"},{"key":"ref8","doi-asserted-by":"crossref","first-page":"135","DOI":"10.1093\/qjmed\/os-16.62.135","article-title":"Muscular exercise, lactic acid, and the supply and utilization of oxygen","volume":"16","author":"AV Hill","year":"1923","journal-title":"Q J Med"},{"key":"ref9","doi-asserted-by":"crossref","first-page":"155","DOI":"10.1098\/rspb.1924.0048","article-title":"Muscular exercise, lactic acid and the supply and utilisation of oxygen: Parts VII-VIII","volume":"97","author":"AV Hill","year":"1924","journal-title":"Proc Roy Soc B"},{"issue":"1","key":"ref10","doi-asserted-by":"crossref","first-page":"70","DOI":"10.1097\/00005768-200001000-00012","article-title":"Limiting factors for maximum oxygen uptake and determinants of endurance performance","volume":"32","author":"DR Bassett Jr.","year":"2000","journal-title":"Med Sci Sports Exerc"},{"issue":"1","key":"ref11","doi-asserted-by":"crossref","first-page":"62","DOI":"10.1249\/00005768-199002000-00011","article-title":"Physiological determinants of pulmonary gas exchange kinetics during exercise","volume":"22","author":"BJ Whipp","year":"1990","journal-title":"Med Sci Sports Exerc"},{"issue":"1","key":"ref12","doi-asserted-by":"crossref","first-page":"25","DOI":"10.1113\/jphysiol.2007.147629","article-title":"VO2max: what do we know, and what do we still need to know?","volume":"586","author":"BD Levine","year":"2008","journal-title":"J Physiol"},{"issue":"24","key":"ref13","doi-asserted-by":"crossref","first-page":"e653","DOI":"10.1161\/CIR.0000000000000461","article-title":"Importance of Assessing Cardiorespiratory Fitness in Clinical Practice: A Case for Fitness as a Clinical Vital Sign: A Scientific Statement From the American Heart Association","volume":"134","author":"R Ross","year":"2016","journal-title":"Circulation"},{"issue":"3","key":"ref14","doi-asserted-by":"crossref","first-page":"143","DOI":"10.1111\/j.1600-0838.1995.tb00027.x","article-title":"A maximal cycle exercise protocol to predict maximal oxygen uptake","volume":"5","author":"LB Andersen","year":"1995","journal-title":"Scand J Med Sci Sports"},{"issue":"3","key":"ref15","doi-asserted-by":"crossref","first-page":"345","DOI":"10.1080\/02701367.1996.10607962","article-title":"The reliability of aerobic capacity (VO2max) testing in adolescent girls","volume":"67","author":"JM Pivarnik","year":"1996","journal-title":"Res Q Exerc Sport"},{"issue":"12","key":"ref16","doi-asserted-by":"crossref","first-page":"1413","DOI":"10.1093\/aje\/kwm031","article-title":"Cardiorespiratory fitness as a predictor of nonfatal cardiovascular events in asymptomatic women and men","volume":"165","author":"X Sui","year":"2007","journal-title":"Am J Epidemiol"},{"issue":"19","key":"ref17","doi-asserted-by":"crossref","first-page":"2024","DOI":"10.1001\/jama.2009.681","article-title":"Cardiorespiratory fitness as a quantitative predictor of all-cause mortality and cardiovascular events in healthy men and women: a meta-analysis","volume":"301","author":"S Kodama","year":"2009","journal-title":"JAMA"},{"issue":"17","key":"ref18","doi-asserted-by":"crossref","first-page":"2395","DOI":"10.1001\/jama.1989.03430170057028","article-title":"Physical fitness and all-cause mortality. A prospective study of healthy men and women","volume":"262","author":"SN Blair","year":"1989","journal-title":"JAMA"},{"issue":"4 Suppl","key":"ref19","first-page":"27","article-title":"Mortality trends in the general population: the importance of cardiorespiratory fitness","volume":"24","author":"DC Lee","year":"2010","journal-title":"J Psychopharmacol"},{"issue":"Pt 2","key":"ref20","doi-asserted-by":"crossref","first-page":"413","DOI":"10.1093\/brain\/awg028","article-title":"The spectrum of exercise tolerance in mitochondrial myopathies: a study of 40 patients","volume":"126","author":"T Taivassalo","year":"2003","journal-title":"Brain"},{"issue":"1","key":"ref21","doi-asserted-by":"crossref","first-page":"86","DOI":"10.1002\/ana.10594","article-title":"Oxidative capacity correlates with muscle mutation load in mitochondrial myopathy","volume":"54","author":"TD Jeppesen","year":"2003","journal-title":"Ann Neurol"},{"issue":"5","key":"ref22","doi-asserted-by":"crossref","first-page":"H2122","DOI":"10.1152\/ajpheart.00479.2010","article-title":"Impaired cardiac reserve and severely diminished skeletal muscle O(2) utilization mediate exercise intolerance in Barth syndrome","volume":"301","author":"CT Spencer","year":"2011","journal-title":"Am J Physiol Heart Circ Physiol"},{"issue":"3","key":"ref23","doi-asserted-by":"crossref","DOI":"10.14814\/phy2.13130","article-title":"Impaired cardiac and skeletal muscle bioenergetics in children, adolescents, and young adults with Barth syndrome","volume":"5","author":"A Bashir","year":"2017","journal-title":"Physiol Rep"},{"key":"ref24","article-title":"Children\u2019s Exercise Physiology","author":"TW Rowland","year":"2005"},{"issue":"8","key":"ref25","doi-asserted-by":"crossref","first-page":"1455","DOI":"10.1097\/00005768-200008000-00014","article-title":"Tracking of activity and fitness and the relationship with cardiovascular disease risk factors","volume":"32","author":"JW Twisk","year":"2000","journal-title":"Med Sci Sports Exerc"},{"key":"ref26","doi-asserted-by":"crossref","first-page":"435","DOI":"10.1249\/00003677-199401000-00016","article-title":"Assessment and interpretation of aerobic fitness in children and adolescents","volume":"22","author":"N Armstrong","year":"1994","journal-title":"Exercise and sport sciences reviews"},{"issue":"10","key":"ref27","doi-asserted-by":"crossref","first-page":"1001","DOI":"10.1038\/gim.2015.204","article-title":"New targets for monitoring and therapy in Barth syndrome","volume":"18","author":"WR Thompson","year":"2016","journal-title":"Genet Med"},{"key":"ref28","doi-asserted-by":"crossref","first-page":"31","DOI":"10.1186\/1471-2466-10-31","article-title":"The six minute walk test accurately estimates mean peak oxygen uptake","volume":"10","author":"RM Ross","year":"2010","journal-title":"BMC Pulm Med"},{"key":"ref29","doi-asserted-by":"crossref","first-page":"15","DOI":"10.1007\/8904_2016_553","article-title":"Endurance Exercise Training in Young Adults with Barth Syndrome: A Pilot Study","volume":"32","author":"WT Cade","year":"2017","journal-title":"JIMD Rep"},{"key":"ref30","article-title":"ACSM\u2019s guidelines for exercise testing and prescription","year":"2000"},{"key":"ref31","article-title":"Principles of Exercise Testing and Interpretation: Including Pathophysiology and Clinical Applications","author":"K Wasserman","year":"1999"},{"key":"ref32","article-title":"Growth, maturation and physical activity","author":"RM Malina","year":"1991"},{"key":"ref33","doi-asserted-by":"crossref","first-page":"503","DOI":"10.1249\/00003677-198500130-00015","article-title":"Developmental aspects of maximal aerobic power in children","volume":"13","author":"GS Krahenbuhl","year":"1985","journal-title":"Exerc Sport Sci Rev"},{"issue":"1","key":"ref34","doi-asserted-by":"crossref","first-page":"19","DOI":"10.1080\/03014469700004742","article-title":"Maturity-associated variation in peak oxygen uptake in active adolescent boys and girls","volume":"24","author":"RM Malina","year":"1997","journal-title":"Ann Hum Biol"},{"issue":"1","key":"ref35","doi-asserted-by":"crossref","first-page":"165","DOI":"10.1097\/00005768-199801000-00023","article-title":"Peak oxygen uptake and maturation in 12-yr olds","volume":"30","author":"N Armstrong","year":"1998","journal-title":"Med Sci Sports Exerc"},{"key":"ref36","unstructured":"International Council for Harmonisation of Technical Requirements for Pharmaceuticals for Human Use. ICH Harmonised Tripartite Guideline: Statistical Principles for Clinical Trials (E9)1996 January 10, 2018. <ext-link xmlns:xlink=\"http:\/\/www.w3.org\/1999\/xlink\" ext-link-type=\"uri\" xlink:href=\"http:\/\/www.ich.org\/fileadmin\/Public_Web_Site\/ICH_Products\/\u2026\/E9_Guideline.pdf\" xlink:type=\"simple\">www.ich.org\/fileadmin\/Public_Web_Site\/ICH_Products\/\u2026\/E9_Guideline.pdf<\/ext-link>."},{"issue":"2","key":"ref37","doi-asserted-by":"crossref","first-page":"137","DOI":"10.1080\/02739610902813344","article-title":"Psychosocial Functioning in Youth with Barth Syndrome","volume":"38","author":"EA Storch","year":"2009","journal-title":"Child Health Care"},{"issue":"11","key":"ref38","doi-asserted-by":"crossref","first-page":"2726","DOI":"10.1002\/ajmg.a.35609","article-title":"The Barth Syndrome Registry: distinguishing disease characteristics and growth data from a longitudinal study","volume":"158A","author":"AE Roberts","year":"2012","journal-title":"Am J Med Genet A"},{"issue":"1","key":"ref39","doi-asserted-by":"crossref","first-page":"40","DOI":"10.1016\/0002-9149(92)90673-M","article-title":"Physiologic responses to recumbent versus upright cycle ergometry, and implications for exercise prescription in patients with coronary artery disease","volume":"69","author":"SC Bonzheim","year":"1992","journal-title":"Am J Cardiol"},{"issue":"3","key":"ref40","doi-asserted-by":"crossref","first-page":"257","DOI":"10.1016\/S0003-9993(95)80612-1","article-title":"Physiologic responses of cardiac patients to supine, recumbent, and upright cycle ergometry","volume":"76","author":"TJ Quinn","year":"1995","journal-title":"Arch Phys Med Rehabil"},{"issue":"1","key":"ref41","doi-asserted-by":"crossref","first-page":"167","DOI":"10.1007\/s00246-015-1260-z","article-title":"Clinical Characteristics and Outcomes of Cardiomyopathy in Barth Syndrome: The UK Experience","volume":"37","author":"SL Kang","year":"2016","journal-title":"Pediatr Cardiol"}],"container-title":["PLOS ONE"],"original-title":[],"language":"en","link":[{"URL":"http:\/\/dx.plos.org\/10.1371\/journal.pone.0197776","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2020,5,9]],"date-time":"2020-05-09T14:06:51Z","timestamp":1589033211000},"score":1,"resource":{"primary":{"URL":"https:\/\/dx.plos.org\/10.1371\/journal.pone.0197776"}},"subtitle":[],"editor":[{"given":"Jacobus P.","family":"van Wouwe","sequence":"first","affiliation":[],"role":[{"role":"editor","vocabulary":"crossref"}]}],"short-title":[],"issued":{"date-parts":[[2018,5,24]]},"references-count":41,"journal-issue":{"issue":"5","published-online":{"date-parts":[[2018,5,24]]}},"URL":"https:\/\/doi.org\/10.1371\/journal.pone.0197776","relation":{},"ISSN":["1932-6203"],"issn-type":[{"value":"1932-6203","type":"electronic"}],"subject":[],"published":{"date-parts":[[2018,5,24]]}}}