Center for Health Outcomes and Interdisciplinary Research (CHOIR), Department of Psychiatry, Massachusetts General Hospital, One Bowdoin Square, 1st Floor, Suite 100, Boston, MA, 02114, USA.
Harvard Medical School, Boston, MA, USA.
J Neurooncol. 2024 Apr;167(2):315-322. doi: 10.1007/s11060-024-04610-9. Epub 2024 Feb 27.
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.
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.
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.
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.
神经纤维瘤病（NF）与生活质量（QoL）低下有关。学习障碍在 NF 患者中较为普遍，进一步降低了 QoL，并可能影响身心和教育干预的获益，但针对该人群的研究却很少。在这里，我们通过比较 NF 患者有无学习障碍在基线时的 QoL 以及两种干预措施后与 QoL 相关的获益，来填补这一空白。
对一项针对 NF 患者的身心干预（NF 松弛反应弹性计划，3RP-NF）与教育干预（NF 健康增强计划，HEP-NF）的完全有效 RCT 进行二次分析。共有 228 名 NF 患者参与。参与者报告了四个领域的 QoL（身体健康、心理、社会关系和环境）。我们在基线、治疗后和 12 个月随访时比较了数据，同时控制了干预类型。
在基线时，与没有学习障碍的个体相比，有学习障碍的 NF 患者心理（T=-3.0，p=0.001）和环境（T=-3.8，p<0.001）QoL 较低。无论是否存在学习障碍，两个方案均显著改善了所有 QoL 领域（p<0.0001-0.002）。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 QoL）中超过了最小临床重要差异，而没有学习障碍的个体在三个领域中均超过了最小临床重要差异。此外，有学习障碍的个体在 12 个月时未能维持心理 QoL 的统计学显著获益，而没有学习障碍的个体则维持了所有获益。
有学习障碍的 NF 成年患者的心理和环境 QoL 较低。尽管干预措施显示无论是否存在学习障碍都能改善 QoL，但对于有学习障碍的患者，可能需要额外的措施来增强临床获益和可持续性。
