Abstract

Purpose

Neurofibromatosis (NF) is associated with low quality-of-life (QoL). Learning disabilities are prevalent among those with NF, further worsening QoL and potentially impacting benefits from mind–body and educational interventions, yet research on this population is scarce. Here, we address this gap by comparing NF patients with and without learning disabilities on QoL at baseline and QoL-related gains following two interventions.

Methods

Secondary analysis of a fully-powered RCT of a mind–body program (Relaxation Response Resiliency Program for NF; 3RP-NF) versus an educational program (Health Enhancement Program for NF; HEP-NF) among 228 adults with NF. Participants reported QoL in four domains (Physical Health, Psychological, Social Relationships, and Environmental). We compare data at baseline, post-treatment, and 12-month follow-up, controlling for intervention type.

Results

At baseline, individuals with NF and learning disabilities had lower Psychological (T = -3.0, p = .001) and Environmental (T = -3.8, p < .001) QoL compared to those without learning disabilities. Both programs significantly improved all QoL domains (ps < .0001–0.002) from baseline to post-treatment, regardless of learning disability status. However, those with learning disabilities exceeded the minimal clinically important difference in only one domain (Psychological QoL) compared to three domains in individuals without learning disabilities. Moreover, those with learning disabilities failed to sustain statistically significant gains in Psychological QoL at 12-months, while those without learning disabilities sustained all gains.

Conclusion

Adults with NF and learning disabilities have lower Psychological and Environmental QoL. While interventions show promise in improving QoL regardless of learning disabilities, additional measures may bolster clinical benefit and sustainability among those with learning disabilities.

中文翻译：

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多样化的学习者：神经纤维瘤病成人身心和健康教育干预后的学习障碍和生活质量

摘要

目的

神经纤维瘤病 (NF) 与低生活质量 (QoL) 相关。学习障碍在神经功能障碍患者中普遍存在，这进一步恶化了生活质量，并可能影响身心和教育干预的益处，但针对这一人群的研究却很少。在这里，我们通过比较有和没有学习障碍的 NF 患者的基线生活质量以及两种干预措施后的生活质量相关收益来解决这一差距。

方法

对 228 名 NF 成人的身心计划（神经纤维瘤放松反应恢复计划；3RP-NF）与教育计划（神经纤维瘤健康增强计划；HEP-NF）的全面随机对照试验进行二次分析。参与者报告了四个领域的生活质量（身体健康、心理、社会关系和环境）。我们比较基线、治疗后和 12 个月随访的数据，并控制干预类型。

结果

 在基线时，与没有学习障碍的人相比，患有 NF 和学习障碍的人的心理 (T = -3.0， p  = .001) 和环境 (T = -3.8，p < .001) 生活质量较低。无论学习障碍状况如何，从基线到治疗后，这两个项目均显着改善了所有生活质量领域 ( p s < .0001–0.002)。然而，与没有学习障碍的个体相比，有学习障碍的个体仅在一个领域（心理生活质量）超过了三个领域的最小临床重要差异。此外，有学习障碍的人在 12 个月时未能维持心理生活质量的统计显着改善，而没有学习障碍的人则维持了所有改善。

结论

患有 NF 和学习障碍的成年人的心理和环境生活质量较低。尽管无论学习障碍如何，干预措施都有望改善生活质量，但额外的措施可能会增强学习障碍患者的临床获益和可持续性。