Abstract
Purpose
Neurofibromatoses (NF; NF1, NF2, and schwannomatosis) are incurable tumor suppressor syndromes with heterogeneous symptoms. Emotional distress (e.g., depression, anxiety, stress) is common in NF and impairs quality of life (QoL). Several modifiable dimensions of resiliency can contribute to enhanced QoL in medical populations but have been overlooked as treatment mechanisms for NF. Our goal was to determine, using data from an ongoing efficacy RCT testing a mind–body program for NF, if resiliency explains the relationship between emotional distress and QoL.
Methods
We performed structural equation modeling mediation analysis on baseline measures of QoL (physical health, psychological, social relationships, environmental), emotional distress (depression, anxiety, stress), and resiliency (gratitude, optimism, coping, mindfulness, empathy) completed by adults with NF (N = 228). We controlled for variables known to impact psychosocial functioning in NF (age, diagnosis, learning disability, and education).
Results
After adjusting for covariates, resiliency had a significant and large indirect effect on the negative relationship between emotional distress and QoL (CSIE = − 0.31, 95% CI = − 0.59 to − 0.19, p = .001). The direct effect of emotional distress on QoL was smaller but remained significant (β = − 0.23, 95% CI = − 0.44 to − 0.03, p = .03), suggesting partial mediation through resiliency.
Conclusions
Resiliency may buffer the high rates of emotional distress in NF. Mind–body interventions targeting multiple modifiable resiliency factors may be a promising path toward promoting QoL in adults with NF.
Trial registration
Clinical Trials.gov Identifier: NCT03406208.
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Data availability
The authors have full control of all primary data and that they agree to allow the journal to review their data if requested.
Code availability
Not applicable.
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This work was supported by the U.S. Department of Defense (Grant Number: W81XWH-17-1-0121).
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RM conducted the analyses and wrote the manuscript. JD assisted results interpretation and manuscript writing. JB assisted data analysis, results interpretation, and manuscript writing. JC assisted manuscript writing and preparation. AMV conceptualized the study and guided manuscript preparation. All authors reviewed and agreed with the final manuscript prior to submission.
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Partners Healthcare Human Research Committee has approved the procedures (IRB # 2020P003067 and 2020P004087). The authors certify that the study was performed in accordance with the ethical standards in the 1964 Declaration of Helsinki and its amendments.
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Mace, R.A., Doorley, J., Bakhshaie, J. et al. Psychological resiliency explains the relationship between emotional distress and quality of life in neurofibromatosis. J Neurooncol 155, 125–132 (2021). https://doi.org/10.1007/s11060-021-03852-1
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DOI: https://doi.org/10.1007/s11060-021-03852-1