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Cultivating resiliency in patients with neurofibromatosis 2 who are deafened or have severe hearing loss: a live‑video randomized control trial

  • Jonathan Greenberg
  • Sarah Carter
  • Ethan Lester
  • Christopher J. Funes
  • Eric A. Macklin
  • Scott Plotkin
  • Ana-Maria VranceanuEmail author
Clinical Study

Abstract

Introduction

Patients with NF2 who are deaf or have significant hearing loss face numerous and unique challenges which lead to poor quality of life, and thus may benefit from resiliency programs.

Methods

We performed secondary data analyses on a single blind, randomized controlled trial of an 8 week mind–body resiliency program (the Relaxation Response and Resiliency program for Deaf NF2; d3RP-NF2) versus a health education control (Health Enhancement Program for Deaf NF2;dHEP-NF2) which showed improvement in quality of life (Funes in JAMA 2019,  https://doi.org/10.1007/s11060-019-03182-3). Here we report on improvements in resiliency factors (i.e. optimism, gratitude, perceived social support, mindfulness, and perceived coping abilities) assessed at baseline, post-test and 6-month follow-up. Both programs were delivered via Skype using Communication Access Real-Time Translation.

Results

Patients who were randomized to the d3RP-NF2 program exhibited significant improvements from baseline to post-program in gratitude (Mdifference = 4.04, 95% CI 1.58–6.50; p = 0.002), perceived social support (Mdifference = 16.36, 95% CI 9.20–23.51; p < 0.001), mindfulness (Mdifference = 4.02, 95% CI 1.10–6.94; p = 0.008), perceived coping (Mdifference = 15.25, 95% CI 10.21–20.28; p < 0.001), and a non-significant trend of improvement in optimism (Mdifference = 1.15, 95% CI −0.14–12.44; p = 0.079). These improvements were all maintained through the 6-month follow up. Improvements in perceived coping (Mdifference = 12.34, 95% CI 4.75–19.93; p = 0.002), social support (Mdifference = 13.11, 95% CI 2.19–24.03; p = 0.02), and gratitude (Mdifference = 4.59, 95% CI 0.83–8.36; p = 0.018) were over and above the changes observed in those randomized to dHEP-NF2.

Conclusion

The d3RP-NF2 sustainably improves multiple dimensions of resiliency. Promoting resiliency may be of utmost importance for this uderserved population.

Keywords

Neurofibromatosis Deaf Resiliency Mind–body program Communication access real-time translation Videoconferencing 

Notes

Funding

This study was possible due to a Clinical Trials Award from the Children’s Tumor Foundation (CTF) and a Department of Defense Grant (W81XWH-17–1-0121) to Ana-Maria Vranceanu.

Compliance with ethical standards

Conflict of interest

All authors declare that they have no conflict of interest.

Ethical approval

All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional and/or national research committee and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards.

Informed consent

Informed consent was obtained from all individual participants included in the study.

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Copyright information

© Springer Science+Business Media, LLC, part of Springer Nature 2019

Authors and Affiliations

  1. 1.Integrated Brain Health Clinical and Research Program, Department of PsychiatryMassachusetts General Hospital/Harvard Medical SchoolBostonUSA
  2. 2.Harvard Medical SchoolBostonUSA
  3. 3.Division of Neuropsychiatry, Department of Psychiatry & Athinoula A Martinos Center for Biomedical ImagingMassachusetts General HospitalBostonUSA
  4. 4.Biostatistics CenterMassachusetts General HospitalBostonUSA
  5. 5.Neurofibromatosis ClinicMassachusetts General HospitalBostonUSA

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