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Baseline characteristics of adults with neurofibromatosis enrolled on a psychosocial randomized controlled trial

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Abstract

Purpose

Neurofibromatosis (NF) is an incurable genetic neurological condition. Psychosocial interventions that promote resiliency are a promising approach to address the high emotional distress and low quality of life (QoL) associated with NF. However, no studies have examined the psychosocial needs of treatment-seeking adults with NF. Our goal was to explore, using data from the largest efficacy trial of a psychosocial intervention for NF, differences in QoL, emotional distress, resiliency, and pain-related outcomes compared to other chronic medical populations and within subtypes (NF1, NF2, schwannomatosis; SCHW).

Methods

Enrolled participants (N = 228) were geographically diverse adults with NF and elevated stress. We performed secondary analysis on baseline measures of QoL, emotional distress, resiliency, and pain-related outcomes. We reported descriptive statistics and normative comparisons to understand the psychosocial characteristics of the overall sample and performed between-group analyses to explore differences within NF type.

Results

Our sample endorsed worse QoL, emotional distress, resilience, and pain-related outcomes than similar chronic illness populations. Within NF types, participants with NF1 reported lower QoL and resilience compared to those with NF2. Participants with SCHW reported higher pain intensity than those with NF1. Participants with SCHW reported higher pain interference and lower physical QoL compared to those with NF1 and NF2.

Conclusions

Our findings support the urgent need for psychosocial interventions targeting deficits in QoL, emotional distress, resilience, and pain-related outcomes in adults with NF. We recommend efforts to enhance sample diversity, prepare clinicians to provide high-levels of support, and attune skills training to each NF type.

Trial registration

ClinicalTrials.gov NCT03406208; https://clinicaltrials.gov/ct2/show/NCT03406208 (Archived by WebCite at http://www.webcitation.org/72ZoTDQ6h).

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Acknowledgements

The Department of Defense funded this study (DOD; W81XWH-17-1-0121). The sponsor was not involved in the review or approval of this manuscript for publication. We are grateful to the study participants, the Children’s Tumor Foundation, and the other organizations who aided this work.

Funding

This work was supported by the Department of the Defense (Grant No. W81XWH-17-1-0121).

Author information

Authors and Affiliations

  1. Integrated Brain Health Clinical and Research Program, Massachusetts General Hospital, Boston, MA, USA

    Nathan S. Fishbein, Ana-Maria Vranceanu & Ryan A. Mace

  2. Center for Health Outcomes and Interdisciplinary Research, Massachusetts General Hospital, Boston, MA, USA

    Nathan S. Fishbein, Ana-Maria Vranceanu & Ryan A. Mace

  3. Harvard Medical School, Boston, MA, USA

    Ana-Maria Vranceanu & Ryan A. Mace

Authors
  1. Nathan S. Fishbein
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Contributions

Material preparation, data collection and analysis were performed by Mr. Fishbein, Dr. Vranceanu, and Dr. Mace. The first draft of the manuscript was written by Mr. Fishbein and Dr. Mace, and all authors critically edited the manuscript. Dr. Vranceanu secured the funding for this project. All authors read and approved the final manuscript.

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Correspondence to Ryan A. Mace.

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Fishbein, N.S., Vranceanu, AM. & Mace, R.A. Baseline characteristics of adults with neurofibromatosis enrolled on a psychosocial randomized controlled trial. J Neurooncol 159, 637–646 (2022). https://doi.org/10.1007/s11060-022-04104-6

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