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Barriers to enrollment included a limited pool of eligible participants, restrictive entry criteria, and a diverse age range that required pediatric and adult expertise. Retention was complicated by a 3-year commitment to a complex study and medication regimen. The Network partnered with the Marfan Foundation, bridging the community with the research. The aims of this study are to report protocol and medication adherence and associated predictive factors, and to describe recruitment and retention strategies.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods:<\/jats:title>\n                    <jats:p>Recruitment, retention, and adherence to protocol activities related to the primary outcome were measured. Retention was measured by percentage of enrolled participants with 3-year outcome data. Protocol adherence was calculated by completion rates of study visits, ambulatory electrocardiography (Holter monitoring), and quarterly calls. Medication adherence was assessed by the number of tablets or the amount of liquid in bottles returned. Centers were ranked according to adherence (high, medium, and low tertiles). Recruitment, retention, and adherence questionnaires were completed by sites. Descriptive statistics summarized recruitment, retention, and adherence, as well as questionnaire results. Regression modeling assessed predictors of adherence.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results:<\/jats:title>\n                    <jats:p>Completion rates for visits, Holter monitors, and quarterly calls were 99%, 94%, and 96%, respectively. Primary outcome data at 3 years were obtained for 88% of participants. The mean percentage of medication taken was estimated at 89%. Site and age were associated with all measures of adherence. Young adult and African American participants had lower levels of adherence. Higher adherence sites employed more strategies; had more staffing resources, less key staff turnover, and more collaboration with referring providers; utilized the Foundation\u2019s resources; and used a greater number of strategies to recruit, retain, and promote protocol and medication adherence.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion:<\/jats:title>\n                    <jats:p>Overall adherence was excellent for this trial conducted within a National Institutes of Health\u2013funded clinical trial network. Strategies specifically targeted to young adults and African Americans may have been beneficial. Many strategies employed by higher adherence sites are ones that any site could easily use, such as greeting families at non-study hospital visits, asking for family feedback, providing calendars for tracking schedules, and recommending apps for medication reminders. Additional key learnings include adherence differences by age, race, and site, the value of collaborative learning, and the importance of partnerships with patient advocacy groups. These lessons could shape recruitment, retention, and adherence to improve the quality of future complex trials involving rare conditions.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1177\/1740774520945988","type":"journal-article","created":{"date-parts":[[2020,8,21]],"date-time":"2020-08-21T08:15:02Z","timestamp":1597997702000},"page":"684-695","update-policy":"https:\/\/doi.org\/10.1177\/sage-journals-update-policy","source":"Crossref","is-referenced-by-count":7,"title":["Recruitment, retention, and adherence in a clinical trial: The Pediatric Heart Network\u2019s Marfan Trial experience"],"prefix":"10.1177","volume":"17","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-0018-057X","authenticated-orcid":false,"given":"Michelle S","family":"Hamstra","sequence":"first","affiliation":[{"name":"Heart Institute Administration, Cincinnati Children\u2019s Hospital Medical Center, Cincinnati, OH, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Victoria L","family":"Pemberton","sequence":"additional","affiliation":[{"name":"National Heart, Lung, and Blood Institute, NIH, Bethesda, MD, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nicholas","family":"Dagincourt","sequence":"additional","affiliation":[{"name":"New England Research Institutes, Inc., Watertown, MA, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Danielle","family":"Hollenbeck-Pringle","sequence":"additional","affiliation":[{"name":"New England Research Institutes, Inc., Watertown, MA, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Felicia L","family":"Trachtenberg","sequence":"additional","affiliation":[{"name":"New England Research Institutes, Inc., Watertown, MA, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6732-8949","authenticated-orcid":false,"given":"James F","family":"Cnota","sequence":"additional","affiliation":[{"name":"Heart Institute Administration, Cincinnati Children\u2019s Hospital Medical Center, Cincinnati, OH, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Andrew M","family":"Atz","sequence":"additional","affiliation":[{"name":"Medical University of South Carolina, Charleston, SC, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Elizabeth","family":"Cappella","sequence":"additional","affiliation":[{"name":"Ann and Robert H. Lurie Children\u2019s Hospital, Chicago, IL, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sylvia","family":"De Nobele","sequence":"additional","affiliation":[{"name":"Ghent University Hospital, Ghent, Belgium"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Josephine","family":"Grima","sequence":"additional","affiliation":[{"name":"The Marfan Foundation, Port Washington, NY, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Martha","family":"King","sequence":"additional","affiliation":[{"name":"Harvard Medical School, Boston Children\u2019s Hospital, Boston, MA, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rosalind","family":"Korsin","sequence":"additional","affiliation":[{"name":"Children\u2019s Hospital of New York, New York, NY, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Linda M","family":"Lambert","sequence":"additional","affiliation":[{"name":"Primary Children\u2019s Hospital, University of Utah, Salt Lake City, UT, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Meghan K","family":"MacNeal","sequence":"additional","affiliation":[{"name":"Icahn School of Medicine at Mount Sinai, New York, NY, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Larry W","family":"Markham","sequence":"additional","affiliation":[{"name":"The Monroe Carell Jr. Children\u2019s Hospital at Vanderbilt, Nashville, TN, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Gretchen","family":"MacCarrick","sequence":"additional","affiliation":[{"name":"Johns Hopkins School of Medicine, Baltimore, MD, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Donna M","family":"Sylvester","sequence":"additional","affiliation":[{"name":"Children\u2019s Hospital of Philadelphia, Philadelphia, PA, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Patricia","family":"Walter","sequence":"additional","affiliation":[{"name":"Hospital for Sick Children, University of Toronto, Toronto, ON, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mingfen","family":"Xu","sequence":"additional","affiliation":[{"name":"Duke University School of Medicine, Durham, NC, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ronald V","family":"Lacro","sequence":"additional","affiliation":[{"name":"Harvard Medical School, Boston Children\u2019s Hospital, Boston, MA, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"name":"for the Pediatric Heart Network Investigators","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"179","published-online":{"date-parts":[[2020,8,21]]},"reference":[{"key":"bibr1-1740774520945988","unstructured":"Dietz H. 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