Virtual mind-body treatment for geographically diverse youth with neurofibromatosis: A pilot randomized controlled trial

Lester, Ethan G.; DiStefano, Sofia; Mace, Ryan A.; Macklin, Eric A.; Plotkin, Scott R.; Vranceanu, Ana‐Maria

doi:10.1016/j.genhosppsych.2019.12.001

Cited by 16 publications

(51 citation statements)

References 30 publications

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“…In an RCT adapted for NF2 patients with deafness, Funes et al 17 found that the d3RP‐NF program significantly improved physical, psychological, and environmental QoL domains among NF2 patients with deafness, when compared to an attention‐placebo control. Similarly, in an RCT by Lester et al, 20 an RY‐NF program adapted for youth with NF resulted in significantly greater improvements in physical, psychological, social, and environmental QoL domains, when compared to an attention‐placebo control. Meta‐analysis results demonstrated that MBT was significantly associated with improved physical (MD = 13.63, 95%CI 6.95‐20.30, P < .0001, I 2 = 24%), psychological (MD = 14.11, 95%CI 6.44‐21.78, P = .0003, I 2 = 38%), social (MD = 9.63, 95%CI 2.93‐16.33, P = .005, I 2 = 0%), and environmental QoL (MD = 14.14, 95%CI 8.28‐20.00, P < .00001.…”

Section: Resultsmentioning

confidence: 90%

Impact of mind‐body treatment interventions on quality of life in neurofibromatosis patients: A systematic review and meta‐analysis

Wei

Farooq

Kumar

2020

Dermatologic Therapy

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Individuals with neurofibromatosis (NF) experience poorer quality of life (QoL), in part contributed by the clinical manifestations of NF, such as functional disability, chronic pain, and altered physical appearance. Mind‐body therapies (MBTs) tailored to NF have been developed, and have demonstrated promising potential to improve QoL in this population. We sought to systematically review current evidence on the effectiveness of MBTs in addressing QoL deficits in NF patients. Databases were reviewed between the date of inception and June 2020, using search terms: neurofibromatosis, schwannomatosis, psychotherapy, mind‐body, mindfulness, meditation, resiliency, and behavioral therapy. Quality appraisal was assessed using the Cochrane Risk of Bias Tools and National Institutes of Health Study Quality Assessment Tools. We conducted a meta‐analysis of mean differences and reported aggregate effect estimates with 95% confidence intervals. A total of 10 articles, including randomized‐controlled trials and pre‐post studies, were identified. Meta‐analytic results of randomized‐controlled trial data from six citations demonstrated MBTs were associated with improved physical (MD = 13.63, 95%CI 6.95‐20.30, P < .0001, I2 = 24%), psychological (MD = 14.11, 95%CI 6.44‐21.78, P = .0003, I2 = 38%), social (MD = 9.63, 95%CI 2.93‐16.33, P = .005, I2 = 0%), and environmental QoL (MD = 14.14, 95%CI 8.28‐20.00, P < .00001. I2 = 0%) in NF patients. These associations were maintained at 6‐months follow‐up for physical, psychological, and environmental QoL (P < .05). Our findings suggest that NF‐adapted MBT strategies are associated with improving QoL in diverse NF populations, including NF2 patients experiencing deafness and youth NF patients. Providers and caregivers for NF should be aware of the potential benefits of MBT in chronic NF management.

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Section: Resultsmentioning

confidence: 90%

Impact of mind‐body treatment interventions on quality of life in neurofibromatosis patients: A systematic review and meta‐analysis

Wei

Farooq

Kumar

2020

Dermatologic Therapy

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“…Patients diagnosed with NF have a lower but modifiable QoL. Virtual mind–body interventions developed to improve stress management and QoL in people with NF are feasible, acceptable, and beneficial [ 3 , 5 , 6 ]. However, most patients with NF still struggle to access specialized, evidence-based psychological treatment, even with virtual adaptations.…”

Section: Discussionmentioning

confidence: 99%

“…Despite this need, patients with NF do not have access to tailored, in-person evidence-based psychological care because of barriers such as distance, cost, time or scheduling, and lack of trained providers. Our team at Massachusetts General Hospital used mixed methods approaches to adapt an evidence-based mind–body program for the specific needs of adults with NF (Relaxation Response Resiliency Program for Neurofibromatosis [3RP-NF]; NF1, NF2, or schwannomatosis) [ 3 ], adolescents with NF (Resilient Youth with NF) [ 5 ], and adults with NF2 who are deaf (Relaxation Response Resiliency Program for NF2) [ 6 ] for virtual delivery to bypass these barriers to care. In three single-blinded pilot randomized controlled trials (RCTs) [ 3 , 5 , 6 ], we have shown that these programs are feasible, acceptable, and superior to an attention placebo health education counterpart in improving QoL, pain, and psychosocial functioning.…”

Section: Introductionmentioning

confidence: 99%

“…Our team at Massachusetts General Hospital used mixed methods approaches to adapt an evidence-based mind–body program for the specific needs of adults with NF (Relaxation Response Resiliency Program for Neurofibromatosis [3RP-NF]; NF1, NF2, or schwannomatosis) [ 3 ], adolescents with NF (Resilient Youth with NF) [ 5 ], and adults with NF2 who are deaf (Relaxation Response Resiliency Program for NF2) [ 6 ] for virtual delivery to bypass these barriers to care. In three single-blinded pilot randomized controlled trials (RCTs) [ 3 , 5 , 6 ], we have shown that these programs are feasible, acceptable, and superior to an attention placebo health education counterpart in improving QoL, pain, and psychosocial functioning. With funding from the United States Department of Defense (DoD), we are currently conducting fully powered efficacy trials of the 3RP-NF intervention in adults (aged 18 years or older) and adolescents (aged 12-17 years).…”

Section: Introductionmentioning

confidence: 99%

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Adaptation of a Live Video Mind–Body Program to a Web-Based Platform for English-Speaking Adults With Neurofibromatosis: Protocol for the NF-Web Study

Lester¹,

Hopkins²,

Popok³

et al. 2021

JMIR Res Protoc

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Background Neurofibromatosis (NF) is a rare genetic condition associated with lower but modifiable quality of life (QoL). Although a virtual live video program (Relaxation Response Resiliency Program for Neurofibromatosis [3RP-NF]; efficacy randomized controlled trial underway) that we created has been made available, ongoing barriers impede some patients from engaging in this intervention. A necessary next step is to develop a stand-alone web-based intervention that reduces barriers to accessing NF-specific psychosocial care. Objective First, we aim to develop a web-based platform (Neurofibromatosis-Web [NF-Web]) of our mind–body resiliency program (3RP-NF) through qualitative interviews with participants from an adult efficacy randomized controlled trial. Second, we aim to iteratively optimize the feasibility, acceptability, credibility, and satisfaction of the NF-Web platform through open pilot trials with participant exit interviews and explore quantitative outcomes within this sample. Here, we describe the protocol and study design, intervention, and analysis plan. Methods For aim 1, we will invite completers from our efficacy trial to participate in qualitative interviews. We will use data from these interviews to adapt the content of the live video program for asynchronous delivery and understand how to create a user-friendly format for an engaging web platform. For aim 2, we will enroll eligible participants recruited for the efficacy trial who could not enroll because of treatment barriers. Eligible participants will complete QoL, depression, anxiety, pain, treatment satisfaction, and program credibility measures at baseline and posttest. Inclusion criteria are identical to those for the efficacy trial, including stress and coping difficulties (self-report), no change in antidepressant medication in the past 3 months, no psychotherapy in the past 3 months, no major upcoming surgeries in the next 12 months, English speaking, ability to complete questionnaires on the web and participate in live video interventions, and consent before participation. The primary outcomes are feasibility, treatment satisfaction, and credibility. The secondary outcomes include physical, psychological, social, and environmental QoL; depression; anxiety; pain intensity; and pain interference. We will enroll at least two group cohorts and iteratively refine the program based on participant feedback after each cohort completes the open pilot trial. Results This trial is ongoing. We have completed the interviews (n=23) and analyzed the data to construct the website. Afterward, we will recruit our cohorts for the trial (approximately n=15/cohort; total=30). Recruitment will end by May 2021, with plans to analyze the data by October 2021. Conclusions We will develop the first web platform for people with NF with difficulties managing stress and NF symptoms and report on feasibility and preliminary effects in improving QoL and psychosocial functioning. NF-Web has potential to extend the reach of our 3RP-NF intervention by removing barriers to care, including lack of trained providers, scheduling difficulties, and appearance concerns. International Registered Report Identifier (IRRID) DERR1-10.2196/27526

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“…23 It has been translated into 19 languages and used in hundreds of psychosocial clinical trials, including NF. 7,8,24 Limitations include no normative data as yet for patients with NF. In addition, 25 the KINDL-R questionnaire, 26 and the DISABKIDS questionnaire.…”

Section: Adult Psychosocial Trialsmentioning

confidence: 99%

Current Recommendations for Patient-Reported Outcome Measures Assessing Domains of Quality of Life in Neurofibromatosis Clinical Trials

et al. 2021

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Objective:To review and recommend patient-reported outcome (PRO) measures assessing multidimensional domains of quality of life (QOL) to use as clinical endpoints in medical and psychosocial trials for children and adults with neurofibromatosis type 1 (NF1), neurofibromatosis type 2 (NF2), and schwannomatosis.Methods:The PRO working group of the Response Evaluation in Neurofibromatosis and Schwannomatosis (REiNS) International Collaboration used systematic methods to review, rate, and recommend existing self-report and parent-report PRO measures of generic and disease-specific QOL for neurofibromatosis (NF) clinical trials. Recommendations were based on four main criteria: patient characteristics, item content, psychometric properties, and feasibility.Results:The highest rated generic measures were 1) the Pediatric Quality of Life Inventory (PedsQL™) Generic Core Scales for NF clinical trials for children or for children through adults, 2) the Functional Assessment of Cancer Therapy-General (FACT-G) for adult medical trials, and 3) the World Health Organization Quality of Life-BREF (WHOQOL-BREF) for adult psychosocial trials. The highest rated disease-specific measures were 1) the PedsQL NF1 Module for NF1 trials, 2) the NF2 Impact on Quality of Life (NFTI-QOL) Scale for NF2 trials, and 3) the Penn Acoustic Neuroma Quality of Life (PANQOL) Scale for NF2 trials targeting vestibular schwannomas. To date, there are no disease-specific tools assessing multidimensional domains of QOL for schwannomatosis.Conclusions:The REiNS Collaboration currently recommends these generic and disease-specific PRO measures to assess multidimensional domains of QOL for NF clinical trials. Additional research is needed to further evaluate the use of these measures in both medical and psychosocial trials.

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Virtual mind-body treatment for geographically diverse youth with neurofibromatosis: A pilot randomized controlled trial

Cited by 16 publications

References 30 publications

Impact of mind‐body treatment interventions on quality of life in neurofibromatosis patients: A systematic review and meta‐analysis

Impact of mind‐body treatment interventions on quality of life in neurofibromatosis patients: A systematic review and meta‐analysis

Adaptation of a Live Video Mind–Body Program to a Web-Based Platform for English-Speaking Adults With Neurofibromatosis: Protocol for the NF-Web Study

Current Recommendations for Patient-Reported Outcome Measures Assessing Domains of Quality of Life in Neurofibromatosis Clinical Trials

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