{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,4,15]],"date-time":"2026-04-15T18:10:56Z","timestamp":1776276656277,"version":"3.50.1"},"reference-count":51,"publisher":"Public Library of Science (PLoS)","issue":"4","license":[{"start":{"date-parts":[[2026,4,15]],"date-time":"2026-04-15T00:00:00Z","timestamp":1776211200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["www.plosone.org"],"crossmark-restriction":false},"short-container-title":["PLoS One"],"abstract":"<jats:sec id=\"sec001\">\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>Sickle cell disease (SCD) is one of the most common monogenic disorders in the world. Patients with SCD have chronic hemolytic anemia and experience episodic pain due to vaso-occlusion resulting in, amongst other, musculoskeletal and cardiopulmonary complications. We hypothesize that adults with SCD are less physically active, but limited information is available on the movement behavior of adults with SCD. In the current study, we aimed to evaluate movement behavior in patients with SCD as compared to healthy controls.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec id=\"sec002\">\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>\n                      Movement behavior of patients with SCD (\u226516 years) was prospectively collected in two University Medical Centers in the Netherlands. Movement behavior was measured for seven consecutive days with an accelerometer (Activ8), distinguishing between lying\/non-wear, sitting, walking, running, and biking. Time spent on activities was compared between patients with SCD and healthy adults with a migration background and between HbSS\/HbS\u03b2\n                      <jats:sup>0<\/jats:sup>\n                      and HbSC\/Hb\u03b2\n                      <jats:sup>+<\/jats:sup>\n                      using the Man-Whitney U test. To adjust for multiple testing, season, sex, and age, a linear regression for each activity was performed.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec id=\"sec003\">\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>Data of 30 patients with SCD during steady state (median age 32.1 (IQR 25\u221247)) and 57 healthy adults (median age 37 (30\u221250)) were analyzed. Patients with SCD walked 0.68 (CI \u22121.22 to \u22120.14) h\/day less than healthy adults. No differences were identified for other postures and activities. Every point increase in hemoglobin level was associated with 0.26 (CI 0.01 to 0.52) h\/day increase in walking duration.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec id=\"sec004\">\n                    <jats:title>Conclusion and key findings<\/jats:title>\n                    <jats:p>Patients with SCD appear to be less physically active compared to healthy adults, which is mainly reflected by reduced walking time (around 3500 steps less a day), highlighting the need for tailored physical activity support.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1371\/journal.pone.0336932","type":"journal-article","created":{"date-parts":[[2026,4,15]],"date-time":"2026-04-15T17:29:08Z","timestamp":1776274148000},"page":"e0336932","update-policy":"https:\/\/doi.org\/10.1371\/journal.pone.corrections_policy","source":"Crossref","is-referenced-by-count":0,"title":["Movement behavior in adults with sickle cell disease compared to healthy adults: a cross-sectional study"],"prefix":"10.1371","volume":"21","author":[{"given":"Aaron A.","family":"Heeren","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Eduard J.","family":"van Beers","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Bart J.","family":"Biemond","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Myrthe J.","family":"van Dijk","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Erfan","family":"Nur","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Charlotte F.J.","family":"van Tuijn","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1910-999X","authenticated-orcid":true,"given":"Merel A.","family":"Timmer","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"340","published-online":{"date-parts":[[2026,4,15]]},"reference":[{"key":"pone.0336932.ref001","doi-asserted-by":"crossref","first-page":"18010","DOI":"10.1038\/nrdp.2018.10","article-title":"Sickle cell disease","volume":"4","author":"GJ Kato","year":"2018","journal-title":"Nat Rev Dis Primers"},{"issue":"9757","key":"pone.0336932.ref002","doi-asserted-by":"crossref","first-page":"2018","DOI":"10.1016\/S0140-6736(10)61029-X","article-title":"Sickle-cell disease","volume":"376","author":"DC Rees","year":"2010","journal-title":"The Lancet"},{"key":"pone.0336932.ref003","unstructured":"Beacher S, Beers van EJ, Biemond P. Richtlijn behandeling sikkelcelziekte. 2017. https:\/\/hematologienederland.nl\/wp-content\/uploads\/2019\/07\/richtlijn_sikkelcelziekte_2017.pdf"},{"key":"pone.0336932.ref004","first-page":"149","article-title":"Pathofysiologie en behandeling van sikkelcelziekte","volume":"1144","author":"EJ Beers","year":"2005","journal-title":"Ned Tijdschr Geneeskd"},{"issue":"10","key":"pone.0336932.ref005","doi-asserted-by":"crossref","first-page":"1033","DOI":"10.1001\/jama.2014.10517","article-title":"Management of sickle cell disease: summary of the 2014 evidence-based report by expert panel members","volume":"312","author":"BP Yawn","year":"2014","journal-title":"JAMA"},{"issue":"9861","key":"pone.0336932.ref006","doi-asserted-by":"crossref","first-page":"142","DOI":"10.1016\/S0140-6736(12)61229-X","article-title":"Global epidemiology of sickle haemoglobin in neonates: a contemporary geostatistical model-based map and population estimates","volume":"381","author":"FB Piel","year":"2013","journal-title":"Lancet"},{"key":"pone.0336932.ref007","article-title":"Neonatale hielprik-screening: veranderinge in het screeningsprogramma","author":"Rijksinstituut voor Volksgezondheid en Milieu","year":"2011"},{"issue":"8","key":"pone.0336932.ref008","article-title":"Sickle cell disease landscape and challenges in the EU: the ERN-EuroBloodNet perspective","volume":"10","author":"MDM Ma\u00f1\u00fa Pereira","year":"2023","journal-title":"Lancet Haematol"},{"key":"pone.0336932.ref009","doi-asserted-by":"crossref","DOI":"10.1016\/j.amepre.2011.09.006","article-title":"Disability among individuals with sickle cell disease: literature review from a public health perspective","volume":"41","author":"ME Swanson","year":"2011","journal-title":"Am J Prev Med"},{"issue":"1","key":"pone.0336932.ref010","doi-asserted-by":"crossref","first-page":"418","DOI":"10.1182\/asheducation-2018.1.418","article-title":"Balancing exercise risk and benefits: lessons learned from sickle cell trait and sickle cell anemia","volume":"2018","author":"RI Liem","year":"2018","journal-title":"Hematology Am Soc Hematol Educ Program"},{"issue":"6","key":"pone.0336932.ref011","doi-asserted-by":"crossref","first-page":"527","DOI":"10.1097\/AJP.0000000000000289","article-title":"Psychosocial and Functional Outcomes in Youth With Chronic Sickle Cell Pain","volume":"32","author":"S Sil","year":"2016","journal-title":"Clin J Pain"},{"key":"pone.0336932.ref012","doi-asserted-by":"crossref","first-page":"313","DOI":"10.2147\/JPR.S150065","article-title":"Presence of pain on three or more days of the week is associated with worse patient reported outcomes in adults with sickle cell disease","volume":"11","author":"N Bakshi","year":"2018","journal-title":"J Pain Res"},{"issue":"3","key":"pone.0336932.ref013","doi-asserted-by":"crossref","first-page":"416","DOI":"10.1016\/j.jpainsymman.2009.12.027","article-title":"A review of the literature on the multiple dimensions of chronic pain in adults with sickle cell disease","volume":"40","author":"LEV Taylor","year":"2010","journal-title":"J Pain Symptom Manage"},{"issue":"46","key":"pone.0336932.ref014","doi-asserted-by":"crossref","first-page":"1269","DOI":"10.15585\/mmwr.mm6646a2","article-title":"CDC Grand Rounds: Improving the Lives of Persons with Sickle Cell Disease","volume":"66","author":"M Hulihan","year":"2017","journal-title":"MMWR Morb Mortal Wkly Rep"},{"issue":"11","key":"pone.0336932.ref015","doi-asserted-by":"crossref","DOI":"10.1001\/jamanetworkopen.2019.15374","article-title":"Estimated Life Expectancy and Income of Patients With Sickle Cell Disease Compared With Those Without Sickle Cell Disease","volume":"2","author":"D Lubeck","year":"2019","journal-title":"JAMA Netw Open"},{"issue":"1","key":"pone.0336932.ref016","doi-asserted-by":"crossref","first-page":"3","DOI":"10.1016\/j.jshs.2012.07.009","article-title":"Chronic disease and the link to physical activity","volume":"2","author":"JL Durstine","year":"2013","journal-title":"Journal of Sport and Health Science"},{"key":"pone.0336932.ref017","doi-asserted-by":"crossref","first-page":"80","DOI":"10.1186\/1479-5868-8-80","article-title":"How many steps\/day are enough? For older adults and special populations","volume":"8","author":"C Tudor-Locke","year":"2011","journal-title":"Int J Behav Nutr Phys Act"},{"issue":"1","key":"pone.0336932.ref018","doi-asserted-by":"crossref","first-page":"75","DOI":"10.1186\/s12966-017-0525-8","article-title":"Sedentary Behavior Research Network (SBRN) - Terminology Consensus Project process and outcome","volume":"14","author":"MS Tremblay","year":"2017","journal-title":"Int J Behav Nutr Phys Act"},{"issue":"2","key":"pone.0336932.ref019","doi-asserted-by":"crossref","first-page":"185","DOI":"10.2522\/ptj.20130175","article-title":"Physical activity and sedentary behaviors in people with stroke living in the community: a systematic review","volume":"94","author":"C English","year":"2014","journal-title":"Phys Ther"},{"issue":"8","key":"pone.0336932.ref020","doi-asserted-by":"crossref","first-page":"819","DOI":"10.1002\/ajh.23752","article-title":"Exercise tolerance, lung function abnormalities, anemia, and cardiothoracic ratio in sickle cell patients","volume":"89","author":"EJ van Beers","year":"2014","journal-title":"Am J Hematol"},{"issue":"3","key":"pone.0336932.ref021","doi-asserted-by":"crossref","first-page":"324","DOI":"10.1016\/j.htct.2020.06.018","article-title":"Physical exercise in sickle cell anemia: a systematic review","volume":"43","author":"DMR Pinto","year":"2021","journal-title":"Hematol Transfus Cell Ther"},{"issue":"12","key":"pone.0336932.ref022","article-title":"Objectively measured physical activity levels and sedentary time in children and adolescents with sickle cell anemia","volume":"13","author":"HN Melo","year":"2018","journal-title":"PLoS One"},{"issue":"3","key":"pone.0336932.ref023","doi-asserted-by":"crossref","first-page":"388","DOI":"10.1123\/pes.2016-0276","article-title":"Self-Reported Physical Activity and Exercise Patterns in Children With Sickle Cell Disease","volume":"29","author":"OA Omwanghe","year":"2017","journal-title":"Pediatr Exerc Sci"},{"key":"pone.0336932.ref024","article-title":"Safety and efficacy of mitapivat, an oral pyruvate kinase activator, in sickle cell disease: A phase 2, open-label study","author":"MJ van Dijk","year":"2022","journal-title":"Am J Hematol"},{"issue":"3","key":"pone.0336932.ref025","doi-asserted-by":"crossref","first-page":"445","DOI":"10.1111\/hae.13441","article-title":"Movement behaviour in adults with haemophilia compared to healthy adults","volume":"24","author":"MA Timmer","year":"2018","journal-title":"Haemophilia"},{"key":"pone.0336932.ref026","doi-asserted-by":"crossref","first-page":"85","DOI":"10.1186\/1479-5868-7-85","article-title":"Large ethnic variations in recommended physical activity according to activity domains in amsterdam, the netherlands","volume":"7","author":"JS de Munter","year":"2010","journal-title":"Int J Behav Nutr Phys Act"},{"key":"pone.0336932.ref027","doi-asserted-by":"crossref","first-page":"378","DOI":"10.1086\/671200","article-title":"Racial, ethnic, and gender differences in physical activity","volume":"7","author":"H Saffer","year":"2013","journal-title":"J Hum Cap"},{"key":"pone.0336932.ref028","unstructured":"Activ8, Vitamove. Activ8 Professional Activity Monitor. https:\/\/www.activ8all.com\/product\/activ8-professional-activity-monitor\/ 2021 October 4."},{"key":"pone.0336932.ref029","first-page":"2055668319890535","article-title":"The Activ8 activity monitor: Validation of posture and movement classification","volume":"7","author":"H Horemans","year":"2020","journal-title":"J Rehabil Assist Technol Eng"},{"key":"pone.0336932.ref030","doi-asserted-by":"crossref","DOI":"10.1249\/01.mss.0000185657.86065.98","article-title":"Conducting accelerometer-based activity assessments in field-based research","volume":"37","author":"SG Trost","year":"2005","journal-title":"Med Sci Sports Exerc"},{"issue":"1","key":"pone.0336932.ref031","doi-asserted-by":"crossref","first-page":"24","DOI":"10.1186\/s12966-021-01091-1","article-title":"Seasons, weather, and device-measured movement behaviors: a scoping review from 2006 to 2020","volume":"18","author":"TB Turrisi","year":"2021","journal-title":"Int J Behav Nutr Phys Act"},{"key":"pone.0336932.ref032","unstructured":"Central Bureau voor de Statistiek. CBS - Statistics Netherlands. 2016; http:\/\/www.cbs.nl\/en\/"},{"key":"pone.0336932.ref033","unstructured":"IBM. IBM SPSS software. http:\/\/www-01.ibm.com\/software\/analytics\/spss\/ 2015. 2015 December 1."},{"key":"pone.0336932.ref034","volume-title":"Basishandboek SPSS 25","author":"A de Vocht","year":"2019"},{"key":"pone.0336932.ref035","volume-title":"The Practice of Statistics in the Life Sciences","author":"B Baldi","year":"2018","edition":"4"},{"key":"pone.0336932.ref036","volume-title":"Foundations of Clinical Research Applications to Practice","author":"LG Portney","year":"2014","edition":"3"},{"issue":"1","key":"pone.0336932.ref037","doi-asserted-by":"crossref","DOI":"10.1371\/journal.pone.0228399","article-title":"Sickle cell disease: A distinction of two most frequent genotypes (HbSS and HbSC)","volume":"15","author":"CC da Guarda","year":"2020","journal-title":"PLoS One"},{"key":"pone.0336932.ref038","doi-asserted-by":"crossref","first-page":"7","DOI":"10.14302\/issn.2372-6601.jhor-15-776","article-title":"Reduced physical activity patterns in patients with thalassemia compared to healthy controls","volume":"2","author":"E Fung","year":"2015","journal-title":"J Hematol Oncol Res"},{"issue":"8","key":"pone.0336932.ref039","doi-asserted-by":"crossref","first-page":"707","DOI":"10.1097\/AJP.0b013e31814da3eb","article-title":"Fear of movement (kinesiophobia), pain, and psychopathology in patients with sickle cell disease","volume":"23","author":"J Pells","year":"2007","journal-title":"Clin J Pain"},{"issue":"9","key":"pone.0336932.ref040","doi-asserted-by":"crossref","first-page":"1502","DOI":"10.1249\/mss.0b013e3180a76de5","article-title":"Physical activity and inactivity in an adult population assessed by accelerometry","volume":"39","author":"M Hagstr\u00f6mer","year":"2007","journal-title":"Med Sci Sports Exerc"},{"issue":"12","key":"pone.0336932.ref041","doi-asserted-by":"crossref","first-page":"1151","DOI":"10.1001\/jama.2020.1382","article-title":"Association of Daily Step Count and Step Intensity With Mortality Among US Adults","volume":"323","author":"PF Saint-Maurice","year":"2020","journal-title":"JAMA"},{"issue":"5","key":"pone.0336932.ref042","doi-asserted-by":"crossref","first-page":"01285","DOI":"10.1183\/23120541.01285-2024","article-title":"Minimal clinically important difference in physical activity in people with asthma","volume":"11","author":"FF de Lima","year":"2025","journal-title":"ERJ Open Res"},{"key":"pone.0336932.ref043","doi-asserted-by":"crossref","DOI":"10.2196\/63153","article-title":"Reliability of Average Daily Steps Measured Through a Consumer Smartwatch in Parkinson Disease Phenotypes, Stages, and Severities: Cross-Sectional Study","volume":"9","author":"E Bianchini","year":"2025","journal-title":"JMIR Form Res"},{"key":"pone.0336932.ref044","article-title":"Minimal clinically important difference for daily pedometer step count in COPD. ERJ open Res; 7","author":"O Polgar","year":"2021","journal-title":"Epub ahead of print"},{"issue":"12","key":"pone.0336932.ref045","doi-asserted-by":"crossref","first-page":"1887","DOI":"10.1002\/acr.22398","article-title":"Does increasing steps per day predict improvement in physical function and pain interference in adults with fibromyalgia?","volume":"66","author":"AS Kaleth","year":"2014","journal-title":"Arthritis Care Res (Hoboken)"},{"issue":"4","key":"pone.0336932.ref046","doi-asserted-by":"crossref","first-page":"292","DOI":"10.1007\/s12170-008-0054-8","article-title":"Too Little Exercise and Too Much Sitting: Inactivity Physiology and the Need for New Recommendations on Sedentary Behavior","volume":"2","author":"MT Hamilton","year":"2008","journal-title":"Curr Cardiovasc Risk Rep"},{"issue":"2","key":"pone.0336932.ref047","doi-asserted-by":"crossref","first-page":"123","DOI":"10.7326\/M14-1651","article-title":"Sedentary time and its association with risk for disease incidence, mortality, and hospitalization in adults: a systematic review and meta-analysis","volume":"162","author":"A Biswas","year":"2015","journal-title":"Ann Intern Med"},{"key":"pone.0336932.ref048","unstructured":"Centraal Bureau voor de Statistiek. Half of Dutch population meet physical activity guidelines. https:\/\/www.cbs.nl\/en-gb\/news\/2020\/17\/half-of-dutch-population-meet-physical-activity-guidelines 2020. 2022 June 23."},{"issue":"1","key":"pone.0336932.ref049","doi-asserted-by":"crossref","first-page":"117","DOI":"10.1186\/s12966-020-01009-3","article-title":"The effectiveness of sedentary behaviour interventions on sitting time and screen time in children and adults: an umbrella review of systematic reviews","volume":"17","author":"P Nguyen","year":"2020","journal-title":"Int J Behav Nutr Phys Act"},{"key":"pone.0336932.ref050","first-page":"505","article-title":"Knowledge of patients with falciform disease about physiotherapeutic treatment","volume":"8","author":"J Petto","year":"2018","journal-title":"Bahiana Journal"},{"issue":"3","key":"pone.0336932.ref051","doi-asserted-by":"crossref","first-page":"371","DOI":"10.1007\/s40279-019-01063-3","article-title":"Physical Literacy, Physical Activity and Health: Toward an Evidence-Informed Conceptual Model","volume":"49","author":"J Cairney","year":"2019","journal-title":"Sports Med"}],"container-title":["PLOS One"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/dx.plos.org\/10.1371\/journal.pone.0336932","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,4,15]],"date-time":"2026-04-15T17:29:16Z","timestamp":1776274156000},"score":1,"resource":{"primary":{"URL":"https:\/\/dx.plos.org\/10.1371\/journal.pone.0336932"}},"subtitle":[],"editor":[{"given":"Samuel Kofi","family":"Tchum","sequence":"first","affiliation":[],"role":[{"role":"editor","vocabulary":"crossref"}]}],"short-title":[],"issued":{"date-parts":[[2026,4,15]]},"references-count":51,"journal-issue":{"issue":"4","published-online":{"date-parts":[[2026,4,15]]}},"URL":"https:\/\/doi.org\/10.1371\/journal.pone.0336932","relation":{},"ISSN":["1932-6203"],"issn-type":[{"value":"1932-6203","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,4,15]]}}}