Acceptance and commitment therapy in youth with neurofibromatosis type 1 (NF1) and chronic pain and their parents: A pilot study of feasibility and preliminary efficacy

Pediatric chronic pain is common and can result in substantial long-term disability. Previous studies on acceptance and commitment therapy (ACT) have shown promising results in improving functioning in affected children, but more research is still urgently needed. In the current clinical pilot study, we evaluated an ACT-based interdisciplinary outpatient intervention (14 sessions), including a parent support program (four sessions). Adolescents were referred to the clinic if they experienced disabling chronic pain. They were then randomized, along with their parents, to receive group (n = 12) or individual (n = 18) treatment. Adolescent pain interference, pain reactivity, depression, functional disability, pain intensity and psychological flexibility, along with parent anxiety, depression, pain reactivity and psychological flexibility were assessed using self-reported questionnaires. There were no significant differences in outcomes between individual and group treatment. Analyses illustrated significant (p < 0.01) improvements (medium to large effects) in pain interference, depression, pain reactivity and psychological flexibility post-treatment. Additionally, analyses showed significant (p < 0.01) improvements (large effects) in parent pain reactivity and psychological flexibility post-treatment. On all significant outcomes, clinically-significant changes were observed for 21%–63% of the adolescents across the different outcome measures and in 54%–76% of the parents. These results support previous findings and thus warrant the need for larger, randomized clinical trials evaluating the relative utility of individual and group treatment and the effects of parental interventions.

Objective: Tumors and other disease complications of neurofibromatosis (NF) can cause pain and negatively affect physical functioning. To document the clinical benefit of treatment in NF trials _targeting these manifestations, patient-reported outcomes (PROs) assessing pain and physical functioning should be included as study endpoints. Currently, there is no consensus on the selection and use of such measures in the NF population. This article presents the recommendations of the PRO group of the Response Evaluation in Neurofibromatosis and Schwannomatosis (REiNS) International Collaboration for assessing the domains of pain and physical functioning for NF clinical trials.Methods: The REiNS PRO group reviewed and rated existing PRO measures assessing pain intensity, pain interference, and physical functioning using their systematic method. Final recommendations are based primarily on 4 main criteria: patient characteristics, item content, psychometric properties, and feasibility for clinical trials.Results: The REiNS PRO group chose the Numeric Rating Scale-11 ($8 years) to assess pain intensity, the Pain Interference Index (6-24 years) and the Patient-Reported Outcome Measurement Information System (PROMIS) Pain Interference Scale ($18 years) to evaluate pain interference, and the PROMIS Physical Functioning Scale to measure upper extremity function and mobility ($5 years) for NF clinical trials. Conclusions:The REiNS Collaboration currently recommends these PRO measures to assess the domains of pain and physical functioning for NF clinical trials; however, further research is needed to evaluate their use in individuals with NF. A final consensus recommendation for the pain interference measure will be disseminated in a future publication based on findings from additional published research. Neurology ® 2016;87 (Suppl 1):S4-S12

Parents of children with the neurofibromatoses (NF; neurofibromatosis 1, neurofibromatosis 2, and schwannomatosis) are at an increased risk for emotional and physical health problems. This study aimed to determine parents' perceptions of stressors associated with parenting a child with NF in order to inform the development of a resiliency intervention. We conducted three live video semi structured focus groups with parents of youth with NF (N = 30), which were subsequently transcribed and coded using qualitative content analysis. Parents reported heightened stress associated with the child's educational, medical, and social needs, as well as concerns about their child's physical and mental health. They also reported stress associated with managing finances, multiple medical appointments, role challenges (i.e., being a parent or partner), and managing the uncertainty/unpredictability of their child's NF diagnosis. These stressors reportedly affected employment status (i.e., work scale backs), relationships (i.e., social, familial, with partner, other children), and the parents' physical and mental health. All participants expressed interest in a mind body program aimed at improving resiliency by teaching coping skills (e.g., mindfulness, adaptive thinking, positive psychology skills) and enhancing social support. Results show parents' enthusiasm for a resiliency intervention _targeting stress associated with parenting a child with NF, and provide valuable information for the content of the intervention and its delivery modality.