Literature DB >> 31049826

First report of quality of life in adults with neurofibromatosis 2 who are deafened or have significant hearing loss: results of a live-video randomized control trial.

Christopher J Funes1, Ryan A Mace1,2, Erik A Macklin2,3, Scott R Plotkin2,4, Justin T Jordan2,4, Ana-Maria Vranceanu5,6,7.   

Abstract

PURPOSE: To test the feasibility, acceptability, and preliminary efficacy of a mind-body program for patients with neurofibromatosis 2 (NF2) who are deaf or have significant hearing loss (d3RP-NF2) against an attention placebo control (dHEP-NF2) in a single-blind randomized control trial. Both were delivered using Communication Access Real-Time Translation and live group videoconferencing.
METHODS: Forty-five adults with NF2 were randomized. Co-primary outcomes were physical quality of life (QoL) and psychological QoL and secondary outcomes were social QoL and environmental QoL, all measured with the World Health Organization Quality of Life Abbreviated Instrument (WHOQOL-BREF). Assessments were conducted at baseline, post-treatment, and six-month follow-up.
RESULTS: Forty-one participants (91%) completed the intervention, and 29 (64%) completed the six-month follow up. Participants in the d3RP-NF2 showed significantly greater improvements from baseline to post-treatment on physical QoL (14.79, 95% CI 5.41-24.18; p ≤ 0.001), psychological QoL (18.77, 95% CI 7.09-30.44, p ≤ 0.001), and environmental QoL (13.25, 95% CI 1.10-25.39, p = 0.03) compared to the dHEP-NF2. Social QoL also significantly increased in the d3RP-NF2 (16.32, 95% CI 6.66-25.97, p = 0.001), but improvement was not beyond the dHEP-NF2. Gains in QoL were clinically meaningful and maintained at the 6-month follow-up for d3RP-NF2 participants across all QoL domains. There were more treatment responders in the d3RP-NF2 compared to the dHEP-NF2.
CONCLUSIONS: The d3RP-NF2 was well accepted, highly feasible, and resulted in sustained improvements in QoL in patients with NF2 who are deaf or have significant hearing loss.

Entities:  

Keywords:  Communication access real-time translation; Deaf; Mind–body program; Neurofibromatosis; Quality of Life; Videoconferencing

Mesh:

Year:  2019        PMID: 31049826     DOI: 10.1007/s11060-019-03182-3

Source DB:  PubMed          Journal:  J Neurooncol        ISSN: 0167-594X            Impact factor:   4.130


  13 in total

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2.  Analysis of pilot and early phase studies with small sample sizes.

Authors:  Weichung Joseph Shih; Pamela A Ohman-Strickland; Yong Lin
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3.  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.

Authors:  S M Skevington; M Lotfy; K A O'Connell
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4.  Research electronic data capture (REDCap)--a metadata-driven methodology and workflow process for providing translational research informatics support.

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5.  Health-related Quality of Life of Individuals With Neurofibromatosis Type 2: Results From the NF2 Natural History Study.

Authors:  Vanessa L Merker; Amanda L Bergner; Ana-Maria Vranceanu; Alona Muzikansky; William Slattery; Scott R Plotkin
Journal:  Otol Neurotol       Date:  2016-06       Impact factor: 2.311

6.  The relationship between patients' perception of the effects of neurofibromatosis type 2 and the domains of the Short Form-36.

Authors:  W J Neary; V F Hillier; T Flute; S D G Stephens; R T Ramsden; D G R Evans
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7.  Psychological burden in adult neurofibromatosis type 1 patients: impact of disease visibility on body image.

Authors:  Sofia Granström; Anna Langenbruch; Matthias Augustin; Victor-Felix Mautner
Journal:  Dermatology       Date:  2012-04-12       Impact factor: 5.366

8.  Impact of neurofibromatosis 1 on Quality of Life: a cross-sectional study of 176 American cases.

Authors:  Patricia Z Page; Grier P Page; Emmanuel Ecosse; Bruce R Korf; Alain Leplege; Pierre Wolkenstein
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9.  Quality-of-life impairment in neurofibromatosis type 1: a cross-sectional study of 128 cases.

Authors:  P Wolkenstein; J Zeller; J Revuz; E Ecosse; A Leplège
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10.  Estimating the sample size for a pilot randomised trial to minimise the overall trial sample size for the external pilot and main trial for a continuous outcome variable.

Authors:  Amy L Whitehead; Steven A Julious; Cindy L Cooper; Michael J Campbell
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  6 in total

Review 1.  Can Cancer Education Programs Improve Health Literacy Among Deaf and Hard of Hearing Patients: a Systematic Review.

Authors:  Jan Münstermann; Jutta Hübner; Jens Büntzel
Journal:  J Cancer Educ       Date:  2022-09-19       Impact factor: 1.771

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

Authors:  Jonathan Greenberg; Sarah Carter; Ethan Lester; Christopher J Funes; Eric A Macklin; Scott Plotkin; Ana-Maria Vranceanu
Journal:  J Neurooncol       Date:  2019-11-01       Impact factor: 4.130

3.  Can a Dyadic Resiliency Program Improve Quality of Life in Cognitively Intact Dyads of Neuro-ICU Survivors and Informal Caregivers? Results from a Pilot RCT.

Authors:  Ethan G Lester; Ryan A Mace; Sarah M Bannon; Paula J Popok; Melissa V Gates; Emma Meyers; Tara Tehan; Danielle Sagueiro; Jonathan Rosand; Eric A Macklin; Ana-Maria Vranceanu
Journal:  Neurocrit Care       Date:  2021-04-21       Impact factor: 3.210

Review 4.  Current Understanding of Neurofibromatosis Type 1, 2, and Schwannomatosis.

Authors:  Ryota Tamura
Journal:  Int J Mol Sci       Date:  2021-05-29       Impact factor: 5.923

5.  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.

Authors:  Ethan Gabriel Lester; Sarah Whitall Hopkins; Paula Jean Popok; Ana-Maria Vranceanu
Journal:  JMIR Res Protoc       Date:  2021-06-10

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

Authors:  Pamela L Wolters; Ana-Maria Vranceanu; Heather L Thompson; Staci Martin; Vanessa L Merker; Andrea Baldwin; Carolina Barnett; Kimberley S Koetsier; Cynthia M Hingtgen; Christopher J Funes; James H Tonsgard; Elizabeth K Schorry; Taryn Allen; Taylor Smith; Barbara Franklin; Stephanie Reeve
Journal:  Neurology       Date:  2021-07-06       Impact factor: 11.800

  6 in total

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