{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,13]],"date-time":"2026-10-13T13:22:33Z","timestamp":1791897753379,"version":"4.3.4"},"reference-count":27,"publisher":"American Medical Association (AMA)","issue":"6","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["JAMA Netw Open"],"abstract":"<jats:sec><jats:title>Importance<\/jats:title><jats:p>Neurofibromatoses (NF; NF1, NF2, and schwannomatosis) are hereditary tumor predisposition syndromes with a risk for poor quality of life (QOL) and no evidence-based treatments.<\/jats:p><\/jats:sec><jats:sec><jats:title>Objective<\/jats:title><jats:p>To compare a mind-body skills training program, the Relaxation Response Resiliency Program for NF (3RP-NF), with a health education program (Health Enhancement Program for NF; HEP-NF) for improvement of quality of life among adults with NF.<\/jats:p><\/jats:sec><jats:sec><jats:title>Design, Setting, and Participants<\/jats:title><jats:p>This single-blind, remote randomized clinical trial randomly assigned 228 English-speaking adults with NF from around the world on a 1:1 basis, stratified by NF type, between October 1, 2017, and January 31, 2021, with the last follow-up February 28, 2022.<\/jats:p><\/jats:sec><jats:sec><jats:title>Interventions<\/jats:title><jats:p>Eight 90-minute group virtual sessions of 3RP-NF or HEP-NF.<\/jats:p><\/jats:sec><jats:sec><jats:title>Main Outcomes and Measures<\/jats:title><jats:p>Outcomes were collected at baseline, after treatment, and at 6-month and 1-year follow-up. The primary outcomes were physical health and psychological domain scores of the World Health Organization Quality of Life Brief Version (WHOQOL-BREF). Secondary outcomes were the social relationships and environment domain scores of the WHOQOL-BREF. Scores are reported as transformed domain scores (range, 0-100, with higher scores indicating higher QOL). Analysis was performed on an intention-to-treat basis.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Of 371 participants who underwent screening, 228 were randomized (mean [SD] age, 42.7 [14.5] years; 170 women [75%]), and 217 attended 6 or more of 8 sessions and provided posttest data. Participants in both programs improved from baseline to after treatment in primary outcomes of physical health QOL score (3RP-NF, 5.1; 95% CI, 3.2-7.0; <jats:italic>P<\/jats:italic>\u2009&amp;amp;lt;\u2009.001; HEP-NF, 6.4; 95% CI, 4.6-8.3; <jats:italic>P<\/jats:italic>\u2009&amp;amp;lt;\u2009.001) and psychological QOL score (3RP-NF, 8.5; 95% CI, 6.4-10.7; <jats:italic>P<\/jats:italic>\u2009&amp;amp;lt;\u2009.001; HEP-NF, 9.2; 95% CI, 7.1-11.2; <jats:italic>P<\/jats:italic>\u2009&amp;amp;lt;\u2009.001). Participants in the 3RP-NF group showed sustained improvements after treatment to 12 months; posttreatment improvements for the HEP-NF group diminished (between-group difference for physical health QOL score, 4.9; 95% CI, 2.1-7.7; <jats:italic>P<\/jats:italic>\u2009=\u2009.001; effect size [ES]\u2009=\u20090.3; and psychological QOL score, 3.7; 95% CI, 0.2-7.6; <jats:italic>P<\/jats:italic>\u2009=\u2009.06; ES\u2009=\u20090.2). Results were similar for secondary outcomes of social relationships and environmental QOL. There were significant between-group differences from baseline to 12 months in favor of the 3RP-NF for physical health QOL score (3.6; 95% CI, 0.5-6.6; <jats:italic>P<\/jats:italic>\u2009=\u2009.02; ES\u2009=\u20090.2), social relationships QOL score (6.9; 95% CI, 1.2-12.7; <jats:italic>P<\/jats:italic>\u2009=\u2009.02; ES\u2009=\u20090.3), and environmental QOL score (3.5; 95% CI, 0.4-6.5; <jats:italic>P<\/jats:italic>\u2009=\u2009.02; ES\u2009=\u20090.2).<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusions and Relevance<\/jats:title><jats:p>In this randomized clinical trial of 3RP-NF vs HEP-NF, benefits from 3RP-NF and HEP-NF were comparable after treatment, but at 12 months from baseline, 3RP-NF was superior to HEP-NF on all primary and secondary outcomes. Results support the implementation of 3RP-NF in routine care.<\/jats:p><\/jats:sec><jats:sec><jats:title>Trial Registration<\/jats:title><jats:p>ClinicalTrials.gov Identifier: <jats:ext-link xmlns:xlink=\"http:\/\/www.w3.org\/1999\/xlink\" ext-link-type=\"uri\" xlink:href=\"https:\/\/clinicaltrials.gov\/ct2\/show\/NCT03406208\">NCT03406208<\/jats:ext-link><\/jats:p><\/jats:sec>","DOI":"10.1001\/jamanetworkopen.2023.20599","type":"journal-article","created":{"date-parts":[[2023,6,28]],"date-time":"2023-06-28T15:34:25Z","timestamp":1687966465000},"page":"e2320599","source":"Crossref","is-referenced-by-count":18,"title":["Effect of Mind-Body Skills Training on Quality of Life for Geographically Diverse Adults With Neurofibromatosis"],"prefix":"10.1001","volume":"6","author":[{"given":"Ana-Maria","family":"Vranceanu","sequence":"first","affiliation":[{"name":"Center for Health Outcomes and Interdisciplinary Research, Department of Psychiatry, Massachusetts General Hospital, Boston"},{"name":"Harvard Medical School, Boston, Massachusetts"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Heena R.","family":"Manglani","sequence":"additional","affiliation":[{"name":"Center for Health Outcomes and Interdisciplinary Research, Department of Psychiatry, Massachusetts General Hospital, Boston"},{"name":"Harvard Medical School, Boston, Massachusetts"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nathaniel R.","family":"Choukas","sequence":"additional","affiliation":[{"name":"Center for Health Outcomes and Interdisciplinary Research, Department of Psychiatry, Massachusetts General Hospital, Boston"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Millan R.","family":"Kanaya","sequence":"additional","affiliation":[{"name":"Center for Health Outcomes and Interdisciplinary Research, Department of Psychiatry, Massachusetts General Hospital, Boston"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ethan","family":"Lester","sequence":"additional","affiliation":[{"name":"Center for Health Outcomes and Interdisciplinary Research, Department of Psychiatry, Massachusetts General Hospital, Boston"},{"name":"Harvard Medical School, Boston, Massachusetts"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Emily L.","family":"Zale","sequence":"additional","affiliation":[{"name":"Department of Psychology, Harpur College of Arts and Sciences, Binghamton University, Binghamton, New York"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Scott R.","family":"Plotkin","sequence":"additional","affiliation":[{"name":"Department of Neurology and Cancer Center, Massachusetts General Hospital, Boston"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Justin","family":"Jordan","sequence":"additional","affiliation":[{"name":"Pappas Center for Neuro-Oncology, Massachusetts General Hospital, Boston"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Eric","family":"Macklin","sequence":"additional","affiliation":[{"name":"Harvard Medical School, Boston, Massachusetts"},{"name":"Biostatistics Center, Massachusetts General Hospital, Boston"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jafar","family":"Bakhshaie","sequence":"additional","affiliation":[{"name":"Center for Health Outcomes and Interdisciplinary Research, Department of Psychiatry, Massachusetts General Hospital, Boston"},{"name":"Harvard Medical School, Boston, Massachusetts"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"10","published-online":{"date-parts":[[2023,6,28]]},"reference":[{"issue":"5","key":"zoi230612r1","doi-asserted-by":"publisher","first-page":"1932","DOI":"10.1016\/j.ajpath.2010.12.056","article-title":"The pathoetiology of neurofibromatosis 1.","volume":"178","author":"Jouhilahti","year":"2011","journal-title":"Am J Pathol"},{"issue":"2","key":"zoi230612r4","doi-asserted-by":"publisher","first-page":"171","DOI":"10.1080\/02688697.2020.1836323","article-title":"What to know about schwannomatosis: a literature review.","volume":"36","author":"Schraepen","year":"2022","journal-title":"Br J Neurosurg"},{"issue":"11","key":"zoi230612r5","doi-asserted-by":"publisher","first-page":"616","DOI":"10.1038\/nrclinonc.2013.144","article-title":"Advances in the treatment of neurofibromatosis-associated tumours.","volume":"10","author":"Lin","year":"2013","journal-title":"Nat Rev Clin Oncol"},{"issue":"3","key":"zoi230612r6","doi-asserted-by":"publisher","first-page":"257","DOI":"10.1007\/s11060-013-1195-2","article-title":"Quality of life among adult patients with neurofibromatosis 1, neurofibromatosis 2 and schwannomatosis: a systematic review of the literature.","volume":"114","author":"Vranceanu","year":"2013","journal-title":"J Neurooncol"},{"issue":"7","key":"zoi230612r7","doi-asserted-by":"publisher","first-page":"1704","DOI":"10.1002\/ajmg.a.v182.7","article-title":"Clinical characteristics and quality of life, depression, and anxiety in adults with neurofibromatosis type 1: a nationwide study.","volume":"182","author":"Doser","year":"2020","journal-title":"Am J Med Genet A"},{"issue":"12","key":"zoi230612r8","doi-asserted-by":"publisher","first-page":"977","DOI":"10.1038\/gim.2012.85","article-title":"Emotional functioning of patients with neurofibromatosis tumor suppressor syndrome.","volume":"14","author":"Wang","year":"2012","journal-title":"Genet Med"},{"issue":"9","key":"zoi230612r9","doi-asserted-by":"publisher","first-page":"2672","DOI":"10.1002\/ajmg.a.v188.9","article-title":"Understanding barriers to diagnosis in a rare, genetic disease: delays and errors in diagnosing schwannomatosis.","volume":"188","author":"Merker","year":"2022","journal-title":"Am J Med Genet A"},{"issue":"1","key":"zoi230612r10","doi-asserted-by":"publisher","first-page":"19","DOI":"10.1080\/13548506.2010.516363","article-title":"A qualitative study of the impact of living with neurofibromatosis type 2.","volume":"16","author":"Patel","year":"2011","journal-title":"Psychol Health Med"},{"issue":"1","key":"zoi230612r11","doi-asserted-by":"publisher","first-page":"103","DOI":"10.1007\/s11060-014-1522-2","article-title":"The Relaxation Response Resiliency Program (3RP) in patients with neurofibromatosis 1, neurofibromatosis 2, and schwannomatosis: results from a pilot study.","volume":"120","author":"Vranceanu","year":"2014","journal-title":"J Neurooncol"},{"issue":"8","key":"zoi230612r12","doi-asserted-by":"publisher","first-page":"806","DOI":"10.1212\/WNL.0000000000003005","article-title":"Mind-body therapy via videoconferencing in patients with neurofibromatosis: an RCT.","volume":"87","author":"Vranceanu","year":"2016","journal-title":"Neurology"},{"issue":"10","key":"zoi230612r13","doi-asserted-by":"publisher","DOI":"10.2196\/11008","article-title":"Mind-body treatment for international English-speaking adults with neurofibromatosis via live videoconferencing: protocol for a single-blind randomized controlled trial.","volume":"7","author":"Vranceanu","year":"2018","journal-title":"JMIR Res Protoc"},{"issue":"4","key":"zoi230612r14","doi-asserted-by":"publisher","first-page":"385","DOI":"10.2307\/2136404","article-title":"A global measure of perceived stress.","volume":"24","author":"Cohen","year":"1983","journal-title":"J Health Soc Behav"},{"issue":"2","key":"zoi230612r16","doi-asserted-by":"publisher","first-page":"299","DOI":"10.1023\/B:QURE.0000018486.91360.00","article-title":"The World Health Organization\u2019s WHOQOL-BREF quality of life assessment: psychometric properties and results of the international field trial: a report from the WHOQOL Group.","volume":"13","author":"Skevington","year":"2004","journal-title":"Qual Life Res"},{"issue":"3","key":"zoi230612r17","doi-asserted-by":"publisher","first-page":"551","DOI":"10.1017\/S0033291798006667","article-title":"Development of the World Health Organization WHOQOL-BREF quality of life assessment.","volume":"28","author":"The WHOQOL Group","year":"1998","journal-title":"Psychol Med"},{"issue":"7","key":"zoi230612r18","doi-asserted-by":"publisher","first-page":"S50","DOI":"10.1212\/WNL.0000000000012421","article-title":"Current recommendations for patient-reported outcome measures assessing domains of quality of life in neurofibromatosis clinical trials.","volume":"97","author":"Wolters","year":"2021","journal-title":"Neurology"},{"issue":"5","key":"zoi230612r19","doi-asserted-by":"publisher","first-page":"1295","DOI":"10.1007\/s00520-012-1664-8","article-title":"The minimal clinical important difference in the World Health Organization Quality of Life instrument\u2013100.","volume":"21","author":"Den Oudsten","year":"2013","journal-title":"Support Care Cancer"},{"issue":"2","key":"zoi230612r20","doi-asserted-by":"publisher","first-page":"377","DOI":"10.1016\/j.jbi.2008.08.010","article-title":"Research electronic data capture (REDCap)\u2014a metadata-driven methodology and workflow process for providing translational research informatics support.","volume":"42","author":"Harris","year":"2009","journal-title":"J Biomed Inform"},{"issue":"5","key":"zoi230612r21","doi-asserted-by":"publisher","first-page":"443","DOI":"10.1037\/0278-6133.23.5.443","article-title":"Enhancing treatment fidelity in health behavior change studies: best practices and recommendations from the NIH Behavior Change Consortium.","volume":"23","author":"Bellg","year":"2004","journal-title":"Health Psychol"},{"key":"zoi230612r22","author":"Liang","year":"2000","journal-title":"Sankhya"},{"issue":"1","key":"zoi230612r23","doi-asserted-by":"publisher","first-page":"43","DOI":"10.1037\/a0014699","article-title":"Effect sizes for growth-modeling analysis for controlled clinical trials in the same metric as for classical analysis.","volume":"14","author":"Feingold","year":"2009","journal-title":"Psychol Methods"},{"issue":"1","key":"zoi230612r24","doi-asserted-by":"publisher","first-page":"111","DOI":"10.1037\/a0030048","article-title":"A regression framework for effect size assessments in longitudinal modeling of group differences.","volume":"17","author":"Feingold","year":"2013","journal-title":"Rev Gen Psychol"},{"issue":"2","key":"zoi230612r25","doi-asserted-by":"publisher","first-page":"96","DOI":"10.20982\/tqmp.15.2.p096","article-title":"New approaches for estimation of effect sizes and their confidence intervals for treatment effects from randomized controlled trials.","volume":"15","author":"Feingold","year":"2019","journal-title":"Quant Method Psychol"},{"issue":"1","key":"zoi230612r26","doi-asserted-by":"publisher","first-page":"37","DOI":"10.1007\/s11205-005-5552-1","article-title":"Interpreting the WHOQOL-Br\u00e8f: preliminary population norms and effect sizes.","volume":"77","author":"Hawthorne","year":"2006","journal-title":"Soc Indic Res"},{"key":"zoi230612r2","volume-title":"GeneReviews","author":"Friedman","year":"1993"},{"key":"zoi230612r3","volume-title":"GeneReviews","author":"Evans"},{"key":"zoi230612r15","author":"Cohen","year":"1988"},{"key":"zoi230612r27","first-page":"29","volume-title":"Statistical Analysis with Missing Data","year":"2019","edition":"3rd ed"}],"container-title":["JAMA Network Open"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/jamanetwork.com\/journals\/jamanetworkopen\/articlepdf\/2806486\/vranceanu_2023_oi_230612_1686854138.21499.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,6,28]],"date-time":"2023-06-28T15:34:29Z","timestamp":1687966469000},"score":1,"resource":{"primary":{"URL":"https:\/\/jamanetwork.com\/journals\/jamanetworkopen\/fullarticle\/2806486"}},"subtitle":["A Fully Remote Randomized Clinical Trial"],"short-title":[],"issued":{"date-parts":[[2023,6,28]]},"references-count":27,"journal-issue":{"issue":"6","published-print":{"date-parts":[[2023,6,1]]}},"URL":"https:\/\/doi.org\/10.1001\/jamanetworkopen.2023.20599","relation":{},"ISSN":["2574-3805"],"issn-type":[{"value":"2574-3805","type":"electronic"}],"subject":[],"published":{"date-parts":[[2023,6,28]]}}}