{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,10,29]],"date-time":"2025-10-29T19:31:41Z","timestamp":1761766301467,"version":"3.37.3"},"reference-count":29,"publisher":"Wiley","issue":"2","license":[{"start":{"date-parts":[[2017,12,20]],"date-time":"2017-12-20T00:00:00Z","timestamp":1513728000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"},{"start":{"date-parts":[[2017,12,20]],"date-time":"2017-12-20T00:00:00Z","timestamp":1513728000000},"content-version":"unspecified","delay-in-days":0,"URL":"http:\/\/www.springer.com\/tdm"}],"funder":[{"DOI":"10.13039\/100006093","name":"Patient-Centered Outcomes Research Institute","doi-asserted-by":"publisher","award":["PPRN-1306-0460"],"award-info":[{"award-number":["PPRN-1306-0460"]}],"id":[{"id":"10.13039\/100006093","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["Journal of Genetic Counseling"],"published-print":{"date-parts":[[2018,4]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:p>Duchenne\/Becker muscular dystrophy (DBMD) and spinal muscular atrophy (SMA) are rare neuromuscular disorders that present challenges to therapeutic and clinical trial decision making. We developed an interactive, evidence\u2010based online tool designed to encourage thoughtful deliberation of the pros and cons of trial participation and to inform meaningful discussions with healthcare providers. Prior research demonstrates the importance of tool availability at the time each family is considering trial participation, which may be prior to the informed consent process. The tool is intended to be easily modified to other pediatric disease communities. Tool development was informed by prior qualitative research, literature reviews, and stakeholder input. Specific items were derived based on an online exploratory questionnaire of parents whose children participated in a trial for DBMD or SMA to understand motivations for participation. Parent participants in the exploratory survey reported strong impact of altruistic and individual benefit motivations and placed much greater emphasis on anticipated trial benefits than on harms when making participation decisions. We used this data to develop the evidence\u2010based deliberation tool using a community\u2010engaged approach. We initially targeted the tool for DBMD while using SMA survey data to evaluate ease of transition to that population. We conducted two iterative sets of activities to inform development and refinement of the tool: (1) community engagement of key stakeholders and (2) user experience testing. These activities suggest that the tool may increase deliberation and the weighing of benefits and harms. Ongoing evaluation will determine the acceptability and efficacy of this online intervention.<\/jats:p>","DOI":"10.1007\/s10897-017-0190-8","type":"journal-article","created":{"date-parts":[[2017,12,20]],"date-time":"2017-12-20T01:41:55Z","timestamp":1513734115000},"page":"416-425","update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":9,"title":["An Evidence\u2010Based, Community\u2010Engaged Approach to Develop an Interactive Deliberation Tool for Pediatric Neuromuscular Trials"],"prefix":"10.1002","volume":"27","author":[{"given":"Rebecca R.","family":"Moultrie","sequence":"first","affiliation":[{"name":"RTI International P.O. Box 12194, 3040 E. Cornwallis Road Research Triangle Park NC 27709 USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Megan A.","family":"Lewis","sequence":"additional","affiliation":[{"name":"RTI International P.O. Box 12194, 3040 E. Cornwallis Road Research Triangle Park NC 27709 USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ryan S.","family":"Paquin","sequence":"additional","affiliation":[{"name":"RTI International P.O. Box 12194, 3040 E. Cornwallis Road Research Triangle Park NC 27709 USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ann","family":"Lucas","sequence":"additional","affiliation":[{"name":"Parent Project Muscular Dystrophy Hackensack NJ USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jill","family":"Jarecki","sequence":"additional","affiliation":[{"name":"Cure SMA Elk Grove Village IL USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Holly L.","family":"Peay","sequence":"additional","affiliation":[{"name":"RTI International P.O. Box 12194, 3040 E. Cornwallis Road Research Triangle Park NC 27709 USA"},{"name":"Parent Project Muscular Dystrophy Hackensack NJ USA"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2017,12,20]]},"reference":[{"key":"e_1_2_9_2_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.pec.2010.01.002"},{"key":"e_1_2_9_3_1","doi-asserted-by":"publisher","DOI":"10.1007\/s10897-015-9859-z"},{"key":"e_1_2_9_4_1","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.0000000000002445"},{"key":"e_1_2_9_5_1","doi-asserted-by":"publisher","DOI":"10.1038\/nn.4508"},{"key":"e_1_2_9_6_1","doi-asserted-by":"publisher","DOI":"10.1038\/nbt0217-99"},{"key":"e_1_2_9_7_1","doi-asserted-by":"publisher","DOI":"10.1136\/bmj.38926.629329.AE"},{"key":"e_1_2_9_8_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.nmd.2007.06.058"},{"key":"e_1_2_9_9_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0140-6736(16)31408-8"},{"key":"e_1_2_9_10_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ncl.2014.05.002"},{"key":"e_1_2_9_11_1","doi-asserted-by":"publisher","DOI":"10.1177\/0883073815627882"},{"key":"e_1_2_9_12_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jval.2016.02.014"},{"key":"e_1_2_9_13_1","unstructured":"International Patient Decision Aid Standards Collaboration [IPDAS].(2005).Criteria for judging the quality of patient decision aids [Online report] Retrieved May 1 2017. Retrieved fromhttp:\/\/ipdas.ohri.ca\/IPDAS_checklist.pdf"},{"key":"e_1_2_9_14_1","doi-asserted-by":"publisher","DOI":"10.1001\/jama.2016.16437"},{"key":"e_1_2_9_15_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ncl.2015.07.004"},{"key":"e_1_2_9_16_1","doi-asserted-by":"publisher","DOI":"10.1177\/0272989X0002000114"},{"key":"e_1_2_9_17_1","doi-asserted-by":"publisher","DOI":"10.1093\/oxfordjournals.jncimonographs.a024212"},{"key":"e_1_2_9_18_1","doi-asserted-by":"publisher","DOI":"10.1377\/hlthaff.26.3.716"},{"key":"e_1_2_9_19_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1525-1497.2004.30047.x"},{"key":"e_1_2_9_20_1","doi-asserted-by":"crossref","unstructured":"Peay H. L. Biesecker B. B. Wilfond B. S. Jarecki J. Umstead K. L. Escolar D. et al. (2017).Barriers and facilitators to clinical trial participation among parents of children with pediatric neuromuscular disorders. Manuscript submitted for publication.","DOI":"10.1177\/1740774517751118"},{"key":"e_1_2_9_21_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.cct.2015.11.006"},{"key":"e_1_2_9_22_1","doi-asserted-by":"publisher","DOI":"10.1177\/1740774513512726"},{"volume-title":"Participatory design:principles and practices.","year":"1993","author":"Schuler D.","key":"e_1_2_9_23_1"},{"key":"e_1_2_9_24_1","doi-asserted-by":"publisher","DOI":"10.1007\/s40124-014-0044-x"},{"key":"e_1_2_9_25_1","first-page":"CD001431","article-title":"Decision aids for people facing health treatment or screening decisions","volume":"10","author":"Stacey D.","year":"2011","journal-title":"Cochrane Database of Systematic Reviews"},{"key":"e_1_2_9_26_1","first-page":"264","volume-title":"Spinal muscular atrophy: disease mechanisms and therapy","author":"Sunshine S.","year":"2016"},{"key":"e_1_2_9_27_1","first-page":"37","volume-title":"Research ethics: a psychological approach","author":"Thompson W. C.","year":"1996"},{"key":"e_1_2_9_28_1","doi-asserted-by":"publisher","DOI":"10.1258\/jms.2008.007110"},{"key":"e_1_2_9_29_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00431-016-2715-9"},{"key":"e_1_2_9_30_1","doi-asserted-by":"publisher","DOI":"10.4236\/ojped.2012.21001"}],"container-title":["Journal of Genetic Counseling"],"original-title":[],"language":"en","link":[{"URL":"http:\/\/link.springer.com\/article\/10.1007\/s10897-017-0190-8\/fulltext.html","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"http:\/\/link.springer.com\/content\/pdf\/10.1007\/s10897-017-0190-8.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"http:\/\/link.springer.com\/content\/pdf\/10.1007\/s10897-017-0190-8.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1007\/s10897-017-0190-8","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,9,15]],"date-time":"2023-09-15T12:31:15Z","timestamp":1694781075000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1007\/s10897-017-0190-8"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2017,12,20]]},"references-count":29,"journal-issue":{"issue":"2","published-print":{"date-parts":[[2018,4]]}},"alternative-id":["10.1007\/s10897-017-0190-8"],"URL":"https:\/\/doi.org\/10.1007\/s10897-017-0190-8","archive":["Portico"],"relation":{},"ISSN":["1059-7700","1573-3599"],"issn-type":[{"type":"print","value":"1059-7700"},{"type":"electronic","value":"1573-3599"}],"subject":[],"published":{"date-parts":[[2017,12,20]]}}}