Mind-Body Treatment for International English-Speaking Adults With Neurofibromatosis via Live Videoconferencing: Protocol for a Single-Blind Randomized Controlled Trial

Ana-Maria Vranceanu, Emily L Zale, Christopher J Funes, Eric A Macklin, Jessica McCurley, Elyse R Park, Justin T Jordan, Ann Lin, Scott R Plotkin, Ana-Maria Vranceanu, Emily L Zale, Christopher J Funes, Eric A Macklin, Jessica McCurley, Elyse R Park, Justin T Jordan, Ann Lin, Scott R Plotkin

Abstract

Background: Neurofibromatoses (NF) are rare genetic conditions associated with substantial psychosocial burden and impaired quality of life (QoL). We developed the first NF-tailored mind-body program (the Relaxation Response Resiliency Program for NF) and adapted it for delivery via live videoconferencing to decrease participation barriers and increase its reach. In a pilot randomized controlled trial (RCT), we found that the Relaxation Response Resiliency Program for NF had excellent feasibility and acceptability when delivered via live videoconferencing; furthermore, the Relaxation Response Resiliency Program for NF showed proof of concept in improving QoL compared with an NF-tailored health education control program (the Health Enhancement Program for NF). A fully powered trial is needed to ascertain the efficacy and durability of the Relaxation Response Resiliency Program for NF delivered via secure live videoconferencing among geographically diverse patients.

Objective: The objective of this study is to evaluate the efficacy of the Relaxation Response Resiliency Program for NF versus the Health Enhancement Program for NF, both delivered in groups via secure live videoconferencing, among geographically diverse patients with NF across the United States and internationally. Here we describe the protocol, manualized treatments, evaluation plan, and study design.

Methods: This is a single-blind RCT. Patients are told that they will be randomized to one of the two stress management programs (stress management program 1: the Relaxation Response Resiliency Program for NF and stress management program 2: the Health Enhancement Program for NF). Patients are recruited from NF-specific national and international foundations and NF clinics across the United States through study ads and a video of participants who have completed the program as part of the pilot study or ongoing trial. Interested participants are screened for eligibility via secure live videoconferencing (self-reported stress and difficulties coping, no change in antidepressant medication within the past 3 months, no psychotherapy within the past 3 months, no major upcoming surgeries within the next 12 months, English speaking, and able to complete questionnaires online and participate in live video interventions) and consent obtained before participation. Both programs are manualized comprising 8 sessions delivered via secure live videoconferencing by trained clinical psychologists. Primary outcomes are physical health QoL and psychological health QoL. Secondary outcomes are social relationship QoL, environment QoL, and psychosocial and resiliency variables. Outcomes are assessed at baseline, posttraining, and 6- and 12-month follow-ups.

Results: The trial is ongoing. Thus far, we have recruited 55 patients and aim to recruit a total of 224. Recruitment will close in May 2020; we plan to complete data analyses by June 2021.

Conclusions: This trial will answer key questions about the efficacy and durability of the Relaxation Response Resiliency Program for NF via live videoconferencing with English-speaking adults with NF worldwide. If found efficacious, this program can be readily implemented through national and international NF foundations and NF-specific clinics. The virtual model of delivery has extensive applications for patients in rural areas, those with disability or illness that precludes travel to clinics, and those with rare diseases.

Trial registration: ClinicalTrials.gov NCT03406208; https://ichgcp.net/clinical-trials-registry/NCT03406208 (Archived by WebCite at http://www.webcitation.org/72ZoTDQ6h).

International registered report identifier (irrid): RR1-10.2196/11008.

Keywords: mind-body; mobile phone; neurofibromatosis; quality of life; resiliency; stress management; videoconferencing.

Conflict of interest statement

Conflicts of Interest: None declared.

©Ana-Maria Vranceanu, Emily L Zale, Christopher J Funes, Eric A Macklin, Jessica McCurley, Elyse R Park, Justin T Jordan, Ann Lin, Scott R Plotkin. Originally published in JMIR Research Protocols (http://www.researchprotocols.org), 23.10.2018.

Figures

Figure 1
Figure 1
Study design. SMP: stress management program.

References

    1. Friedman JM. Epidemiology of neurofibromatosis type 1. Am J Med Genet. 1999 Mar 26;89(1):1–6.
    1. Viskochil D, Buchberg AM, Xu G, Cawthon RM, Stevens J, Wolff RK, Culver M, Carey JC, Copeland NG, Jenkins NA. Deletions and a translocation interrupt a cloned gene at the neurofibromatosis type 1 locus. Cell. 1990 Jul 13;62(1):187–92.
    1. Jouhilahti E, Peltonen S, Heape AM, Peltonen J. The pathoetiology of neurofibromatosis 1. Am J Pathol. 2011 May;178(5):1932–9. doi: 10.1016/j.ajpath.2010.12.056.
    1. Friedman J. Neurofibromatosis 1. In: Adam MP, Ardinger HH, Pagon RA, Wallace SE, Bean LJ, Stephens K, Amemiya A, editors. GeneReviews. Seattle, WA: University of Washington, Seattle; 1993.
    1. Evans DG. Neurofibromatosis 2. In: Adam MP, Ardinger HH, Pagon RA, Wallace SE, Bean LJ, Stephens K, Amemiya A, editors. GeneReviews. Seattle (WA): University of Washington, Seattle; 1993.
    1. Hilton DA, Hanemann CO. Schwannomas and their pathogenesis. Brain Pathol. 2014 Apr;24(3):205–20. doi: 10.1111/bpa.12125.
    1. Lin AL, Gutmann DH. Advances in the treatment of neurofibromatosis-associated tumours. Nat Rev Clin Oncol. 2013 Nov;10(11):616–24. doi: 10.1038/nrclinonc.2013.144.
    1. Vranceanu AM, Merker VL, Park E, Plotkin SR. Quality of life among adult patients with neurofibromatosis 1, neurofibromatosis 2 and schwannomatosis: a systematic review of the literature. J Neurooncol. 2013 Sep;114(3):257–62. doi: 10.1007/s11060-013-1195-2.
    1. Vranceanu AM, Riklin E, Plotkin S, Park E. Psychosocial presentation of adults with neurofibromatosis 1, 2 and schwannomatosis enrolled in a stress reduction program. June; Childrens Tumor Foundation; June 2015; Austin, TX. 2015. Jun 21, p. 2015.
    1. Wang DL, Smith KB, Esparza S, Leigh FA, Muzikansky A, Park ER, Plotkin SR. Emotional functioning of patients with neurofibromatosis tumor suppressor syndrome. Genet Med. 2012 Dec;14(12):977–82. doi: 10.1038/gim.2012.85.
    1. Vranceanu AM, Riklin E, Merker VL, Macklin EA, Park ER, Plotkin SR. Mind-body therapy via videoconferencing in patients with neurofibromatosis: An RCT. Neurology. 2016 Aug 23;87(8):806–14. doi: 10.1212/WNL.0000000000003005.
    1. Park ER, Traeger L, Vranceanu AM, Scult M, Lerner JA, Benson H, Denninger J, Fricchione GL. The development of a patient-centered program based on the relaxation response: the Relaxation Response Resiliency Program (3RP) Psychosomatics. 2013;54(2):165–74. doi: 10.1016/j.psym.2012.09.001.
    1. Zale EL, Pierre-Louis C, Macklin EA, Riklin E, Vranceanu AM. The impact of a mind-body program on multiple dimensions of resiliency among geographically diverse patients with neurofibromatosis. J Neurooncol. 2018 Apr;137(2):321–329. doi: 10.1007/s11060-017-2720-5.
    1. Mahaffey B, Mackin D, Vranceanu AM, Lofaro M, Bromet E, Luft B, Gonzalez A. The stony brook health enhancement program (SB-HEP): The development of a multiple health behavior change program. J Health Psychol. 2018
    1. Rounsaville B, Carroll K, Onken L. A stage model of behavioral therapies research: Getting startedmoving on from stage I. Clin Psychol Sci Pract. 2006;8(2):133–142. doi: 10.1093/clipsy.8.2.133.
    1. Carroll KM, Rounsaville BJ. Bridging the gap: a hybrid model to link efficacy and effectiveness research in substance abuse treatment. Psychiatr Serv. 2003 Mar;54(3):333–9. doi: 10.1176/appi.ps.54.3.333.
    1. Cohen S, Kamarck T, Mermelstein R. A global measure of perceived stress. J Health Soc Behav. 1983 Dec;24(4):385–396.
    1. Cohen S. Perceived stress in a probability sample of the United States. In: Spacapan S, Oskamp S, editors. The Claremont Symposium on Applied Social Psychology: The Social Psychology of Health. Sage Publications: Thousand Oaks, CA; 1988. pp. 31–67.
    1. McGregor BA, Dolan ED, Murphy KM, Sannes TS, Highland KB, Albano DL, Ward AA, Charbonneau AM, Redman MW, Ceballos RM. Cognitive Behavioral Stress Management for Healthy Women at Risk for Breast Cancer: a Novel Application of a Proven Intervention. Ann Behav Med. 2015 Dec;49(6):873–84. doi: 10.1007/s12160-015-9726-z.
    1. Donner A, Klar N. Pitfalls of and controversies in cluster randomization trials. Am J Public Health. 2004 Mar;94(3):416–22.
    1. Benson H, Rosner BA, Marzetta BR, Klemchuk HM. Decreased blood-pressure in pharmacologically treated hypertensive patients who regularly elicited the relaxation response. Lancet. 1974 Feb 23;1(7852):289–91.
    1. Benson H, Klemchuk HP, Graham JR. The usefulness of the relaxation response in the therapy of headache. Headache. 1974 Apr;14(1):49–52.
    1. Benson H, Rosner BA, Marzetta BR, Klemchuk HP. Decreased blood pressure in borderline hypertensive subjects who practiced meditation. J Chronic Dis. 1974 Mar;27(3):163–9.
    1. Beary JF, Benson H. A simple psychophysiologic technique which elicits the hypometabolic changes of the relaxation response. Psychosom Med. 1974;36(2):115–20.
    1. Peters RK, Benson H, Peters JM. Daily relaxation response breaks in a working population: II. Effects on blood pressure. Am J Public Health. 1977 Oct;67(10):954–9.
    1. Greenwood MM, Benson H. The efficacy of progressive relaxation in systematic desensitization and a proposal for an alternative competitive response--the relaxation response. Behav Res Ther. 1977;15(4):337–43.
    1. Benson H, Klipper Mz. Relaxation Response. Boston: HarperCollins Publishers; 2000.
    1. Dusek JA, Otu HH, Wohlhueter AL, Bhasin M, Zerbini LF, Joseph MG, Benson H, Libermann TA. Genomic counter-stress changes induced by the relaxation response. PLoS One. 2008 Jul 02;3(7):e2576. doi: 10.1371/journal.pone.0002576.
    1. Harris PA, Taylor R, Thielke R, Payne J, Gonzalez N, Conde JG. Research electronic data capture (REDCap)—a metadata-driven methodology and workflow process for providing translational research informatics support. J Biomed Inform. 2009 Apr;42(2):377–81. doi: 10.1016/j.jbi.2008.08.010.
    1. Skevington SM, Lotfy M, O'Connell KA. The World Health Organization's WHOQOL-BREF quality of life assessment: psychometric properties and results of the international field trial. A report from the WHOQOL group. Qual Life Res. 2004 Mar;13(2):299–310.
    1. Kroenke K, Spitzer RL, Williams JB. The PHQ-9: validity of a brief depression severity measure. J Gen Intern Med. 2001 Sep;16(9):606–13.
    1. Spitzer RL, Kroenke K, Williams JBW, Löwe B. A brief measure for assessing generalized anxiety disorder: the GAD-7. Arch Intern Med. 2006 May 22;166(10):1092–7. doi: 10.1001/archinte.166.10.1092.
    1. Von KM, Ormel J, Keefe FJ, Dworkin SF. Grading the severity of chronic pain. Pain. 1992 Aug;50(2):133–49.
    1. Amtmann D, Cook KF, Jensen MP, Chen W, Choi S, Revicki D, Cella D, Rothrock N, Keefe F, Callahan L, Lai J. Development of a PROMIS item bank to measure pain interference. Pain. 2010 Jul;150(1):173–82. doi: 10.1016/j.pain.2010.04.025.
    1. Roberti J, Harrington L, Storch E. Further psychometric support for the 10-item version of the perceived stress scale. J Coll Couns. 2006;9(2):135–147. doi: 10.1002/j.2161-1882.2006.tb00100.x.
    1. Lee E. Review of the psychometric evidence of the perceived stress scale. Asian Nurs Res (Korean Soc Nurs Sci) 2012 Dec;6(4):121–7. doi: 10.1016/j.anr.2012.08.004.
    1. Sherbourne CD, Stewart AL. The MOS social support survey. Soc Sci Med. 1991;32(6):705–14.
    1. Mccullough ME, Emmons RA, Tsang J. The grateful disposition: a conceptual and empirical topography. J Pers Soc Psychol. 2002 Jan;82(1):112–27.
    1. Carver CS, Scheier MF, Segerstrom SC. Optimism. Clinical Psychology Review. 2010 Nov;30(7):879–889. doi: 10.1016/j.cpr.2010.01.006.
    1. Antoni MH, Lechner SC, Kazi A, Wimberly SR, Sifre T, Urcuyo KR, Phillips K, Glück S, Carver CS. How stress management improves quality of life after treatment for breast cancer. J Consult Clin Psychol. 2006 Dec;74(6):1143–52. doi: 10.1037/0022-006X.74.6.1152.
    1. Feldman G, Hayes A, Kumar S, Greeson J, Laurenceau J. Mindfulness and Emotion Regulation: The Development and Initial Validation of the Cognitive and Affective Mindfulness Scale-Revised (CAMS-R) J Psychopathol Behav Assess. 2006 Nov 7;29(3):177–190. doi: 10.1007/s10862-006-9035-8.
    1. Davis M. A multidimensional approach to individual differences in empathy. Cat Sel Doc Psychol. 1980;10
    1. ten Klooster Peter M, Drossaers-Bakker KW, Taal E, van de Laar Martin A F J. Patient-perceived satisfactory improvement (PPSI): interpreting meaningful change in pain from the patient's perspective. Pain. 2006 Mar;121(1-2):151–7. doi: 10.1016/j.pain.2005.12.021.
    1. Baron RM, Kenny DA. The moderator-mediator variable distinction in social psychological research: conceptual, strategic, and statistical considerations. J Pers Soc Psychol. 1986 Dec;51(6):1173–82.
    1. Valeri L, Vanderweele TJ. Mediation analysis allowing for exposure-mediator interactions and causal interpretation: theoretical assumptions and implementation with SAS and SPSS macros. Psychol Methods. 2013 Jun;18(2):137–50. doi: 10.1037/a0031034.
    1. Kraemer HC, Wilson GT, Fairburn CG, Agras WS. Mediators and moderators of treatment effects in randomized clinical trials. Arch Gen Psychiatry. 2002 Oct;59(10):877–83.

Source: PubMed

Sponsorzy i współpracownicy

Warunki medyczne

Interwencje lekowe

3
Subskrybuj