Quality of life among adult patients with neurofibromatosis 1, neurofibromatosis 2 and schwannomatosis: a systematic review of the literature

Vranceanu, Ana‐Maria; Merker, Vanessa L.; Park, Elyse Richelle; Plotkin, Scott R.

doi:10.1007/s11060-013-1195-2

Cited by 86 publications

(104 citation statements)

References 22 publications

(23 reference statements)

Supporting

Mentioning

Contrasting

Unclassified

Order By: Relevance

“…In NF1, MPNST is the most commonly found malignancy with a lifetime risk of 8–13% in NF1 (Evans et al, 2002). In the current study groups, problems with self‐acceptance, low self‐esteem, and limited socioeconomic participation also seemed associated with the NF1‐related cosmetic burden (Vranceanu et al, 2013) and mental health problems.…”

Section: Discussionmentioning

confidence: 55%

See 1 more Smart Citation

Worries and needs of adults and parents of adults with neurofibromatosis type 1

Rietman

Helden²,

Both³

et al. 2018

American J of Med Genetics Pt A

View full text Add to dashboard Cite

Neurofibromatosis type 1 (NF1) is a neurocutaneous disorder associated with lifelong tumor growth propensity and neurocognitive impairments. Although follow‐up of adults with NF1 often focuses on tumor growth, follow‐up of cognitive or social problems and other NF1‐related comorbidity is often not a part of standardized care. In order to provide optimal care services for these patients, we explored the care needs of adults with NF1. A qualitative study was performed using semi‐structured group interviews, exploring worries and care needs in medical, psychological, and socioeconomic domains, also focusing on the transition from pediatric to adult care. Four focus groups were conducted, including young adult patients, patients over age 30, and parents of young adult patients. In total, 30 patients and 12 parents participated. Data were transcribed verbatim and analyzed by computerized thematic analysis. Themes were organized using the World Health Organization International classification of functioning, disability, and health (ICF). Results indicated many and diverse worries and care needs both during the transitional period and in adulthood in medical, mental health, and socioeconomic domains. Worries could be categorized into 13 themes. Parents reported high stress levels and difficulties with their parental role. Participants expressed the need for more information, access to NF1 experts, daily living support, care for mental health and socioeconomic participation, and closer communication between health‐care providers. In conclusion, worries and needs of patients and parents underline the importance of multidisciplinary follow‐up and continuity of care during and after the transitional period. Additionally, parental stress requires more attention from care providers.

show abstract

Section: Discussionmentioning

confidence: 55%

“…At the same time, they have a high complication rate and neuropsychiatric and socioeconomic problems persisted or worsened (Oates et al, 2013). Adult patients with NF1 experience decreased quality of life (Vranceanu et al, 2013) and they require developmentally appropriate care (Farre et al, 2016). …”

Section: Introductionmentioning

confidence: 99%

Worries and needs of adults and parents of adults with neurofibromatosis type 1

Rietman

Helden²,

Both³

et al. 2018

American J of Med Genetics Pt A

View full text Add to dashboard Cite

show abstract

“…Despite their distinct pathophysiology patients' psychosocial profile is similar regardless of NF type [8].…”

Section: Introductionmentioning

confidence: 97%

“…As a group, patients with NF have lower quality of life (QoL), and more pain when compared to general population norms [8,9], and comparable rates of depression, anxiety, and stress to patients with cancer and coronary heart disease [9,10]. Despite this heavy psychological burden, there are no evidence-based psychosocial treatments that directly address the specific needs of this population.…”

Section: Introductionmentioning

confidence: 99%

“…Using a sequential approach that included focus groups, in-person open-pilot testing with exit interviews [8], and a preliminary randomized controlled trial (RCT) [11], we adapted an evidence-based mind-body intervention [12], for the specific needs of patients with NF and for live video delivery. The transition from inperson to live videoconferencing delivery was done based on feedback from patients about burdens of traveling for weekly visits and to increase feasibility and extend our reach to patients from across the US and internationally.…”

Section: Introductionmentioning

confidence: 99%

See 1 more Smart Citation

Mind-Body Treatment for International English-Speaking Adults With Neurofibromatosis via Live Videoconferencing: Protocol for a Single-Blind Randomized Controlled Trial (Preprint)

Vranceanu¹,

Zale²,

Vranceanu³

et al. 2018

Preprint

Self Cite

View full text Add to dashboard Cite

show abstract

Effect of Mind-Body Skills Training on Quality of Life for Geographically Diverse Adults With Neurofibromatosis

et al. 2023

Self Cite

View full text Add to dashboard Cite

ImportanceNeurofibromatoses (NF; NF1, NF2, and schwannomatosis) are hereditary tumor predisposition syndromes with a risk for poor quality of life (QOL) and no evidence-based treatments.ObjectiveTo compare a mind-body skills training program, the Relaxation Response Resiliency Program for NF (3RP-NF), with a health education program (Health Enhancement Program for NF; HEP-NF) for improvement of quality of life among adults with NF.Design, Setting, and ParticipantsThis single-blind, remote randomized clinical trial randomly assigned 228 English-speaking adults with NF from around the world on a 1:1 basis, stratified by NF type, between October 1, 2017, and January 31, 2021, with the last follow-up February 28, 2022.InterventionsEight 90-minute group virtual sessions of 3RP-NF or HEP-NF.Main Outcomes and MeasuresOutcomes were collected at baseline, after treatment, and at 6-month and 1-year follow-up. The primary outcomes were physical health and psychological domain scores of the World Health Organization Quality of Life Brief Version (WHOQOL-BREF). Secondary outcomes were the social relationships and environment domain scores of the WHOQOL-BREF. Scores are reported as transformed domain scores (range, 0-100, with higher scores indicating higher QOL). Analysis was performed on an intention-to-treat basis.ResultsOf 371 participants who underwent screening, 228 were randomized (mean [SD] age, 42.7 [14.5] years; 170 women [75%]), and 217 attended 6 or more of 8 sessions and provided posttest data. Participants in both programs improved from baseline to after treatment in primary outcomes of physical health QOL score (3RP-NF, 5.1; 95% CI, 3.2-7.0; P &lt; .001; HEP-NF, 6.4; 95% CI, 4.6-8.3; P &lt; .001) and psychological QOL score (3RP-NF, 8.5; 95% CI, 6.4-10.7; P &lt; .001; HEP-NF, 9.2; 95% CI, 7.1-11.2; P &lt; .001). Participants in the 3RP-NF group showed sustained improvements after treatment to 12 months; posttreatment improvements for the HEP-NF group diminished (between-group difference for physical health QOL score, 4.9; 95% CI, 2.1-7.7; P = .001; effect size [ES] = 0.3; and psychological QOL score, 3.7; 95% CI, 0.2-7.6; P = .06; ES = 0.2). Results were similar for secondary outcomes of social relationships and environmental QOL. There were significant between-group differences from baseline to 12 months in favor of the 3RP-NF for physical health QOL score (3.6; 95% CI, 0.5-6.6; P = .02; ES = 0.2), social relationships QOL score (6.9; 95% CI, 1.2-12.7; P = .02; ES = 0.3), and environmental QOL score (3.5; 95% CI, 0.4-6.5; P = .02; ES = 0.2).Conclusions and RelevanceIn this randomized clinical trial of 3RP-NF vs HEP-NF, benefits from 3RP-NF and HEP-NF were comparable after treatment, but at 12 months from baseline, 3RP-NF was superior to HEP-NF on all primary and secondary outcomes. Results support the implementation of 3RP-NF in routine care.Trial RegistrationClinicalTrials.gov Identifier: NCT03406208

show abstract

Quality of life among adult patients with neurofibromatosis 1, neurofibromatosis 2 and schwannomatosis: a systematic review of the literature

Cited by 86 publications

References 22 publications

Worries and needs of adults and parents of adults with neurofibromatosis type 1

Worries and needs of adults and parents of adults with neurofibromatosis type 1

Mind-Body Treatment for International English-Speaking Adults With Neurofibromatosis via Live Videoconferencing: Protocol for a Single-Blind Randomized Controlled Trial (Preprint)

Effect of Mind-Body Skills Training on Quality of Life for Geographically Diverse Adults With Neurofibromatosis

Contact Info

Product

Resources

About