**1. Introduction**

Although children with acquired brain injury (pediatric acquired brain injury—pABI) may suffer long-lasting physical, cognitive, behavioral and social symptoms as well as disturbances in psychological adaptation [1–3], few randomized controlled trials have

**Citation:** Holthe, I.L.;

Rohrer-Baumgartner, N.; Svendsen, E.J.; Hauger, S.L.; Forslund, M.V.; Borgen, I.M.H.; Øra, H.P.; Kleffelgård, I.; Strand-Saugnes, A.P.; Egeland, J.; et al. Feasibility and Acceptability of a Complex Telerehabilitation Intervention for Pediatric Acquired Brain Injury: The Child in Context Intervention (CICI). *J. Clin. Med.* **2022**, *11*, 2564. https://doi.org/10.3390/ jcm11092564

Academic Editor: Massimiliano Valeriani

Received: 29 March 2022 Accepted: 30 April 2022 Published: 3 May 2022

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**Copyright:** © 2022 by the authors. Licensee MDPI, Basel, Switzerland. This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution (CC BY) license (https:// creativecommons.org/licenses/by/ 4.0/).

explored the effectiveness of complex interventions targeting these children, their families and their everyday context at home and in school. This may be due to the considerable complexity of designing and conducting such studies where one must take a range of factors into account. These include the child's age and developmental level, the heterogeneity of symptoms that may follow a pABI, family factors that may influence the ability and willingness to participate in an intervention, and the heterogeneity in factors related to the child's everyday life in the community, such as school and other areas of participation. The possible pitfalls are many. Exploring the feasibility of a complex intervention is therefore important prior to large-scale RCTs. This paper describes the investigation of the feasibility of a complex, individualized and community-based intervention targeting children with pABI and their families.

Brain damage acquired after birth can be caused by traumatic (TBI) or non-traumatic injuries such as stroke (i.e., brain hemorrhage or infarction), tumor, inflammation, infection or hypoxia. pABI may cause a wide range of disruptions in the child's developmental course [1–3]. Deficits may be persistent, leading to reduced participation in school and social life [4–6], and reduced quality of life [2,7,8]. Prior studies have identified needs for long-term follow-up after ABI; psychological support for parents, siblings and the affected child; as well as support in the return to school [9]. Interestingly, reported needs may change over time, from primarily physical concerns in the first months post injury (79%), to cognitive (47%) and socioemotional needs (68%) later on [10]. Importantly, rehabilitation needs after pABI are described as largely unmet [11].

As family needs after a pediatric brain injury are complex and heterogeneous, rehabilitation services and follow-up care should be individualized to each patient's needs. In line with this, rehabilitation should be multidisciplinary and take place within a biopsychosocial framework [12]. In addition, rehabilitation needs to be flexible, monitoring the development and goals of the patient, which may change over time [12]. A recent study mapped treatment goals set by young people with ABI to the domains in the International Classification of Functioning, Disability and Health (ICF) [13], in which 52% were related to activities and participation, and 20% to environmental factors [14]. This highlights the need to include community-based rehabilitation that is individualized and contextsensitive [14]. The complexity of pediatric ABI rehabilitation may explain the small body of evidence-based recommendations. However, existing knowledge provides evidence for family/caregiver-focused interventions and the use of technology in rehabilitation [15]. In addition, direct interventions related to cognitive functions such as attention, memory, executive function and emotional/behavioral functioning have given positive results [15]. However, a critique against the direct training approach in cognitive rehabilitation (i.e., interventions directed toward retraining the child's abilities) is that generalization to everyday functioning is often unknown or disputed [16]. Therefore, indirect approaches such as behavioral compensation and modification, environmental modification and supports, educational supports and instructional strategies are recommended, including education for parents and caregiver involvement in handling executive dysfunction [17,18]. The effects of parenting/family interventions after pABI have been explored in randomized controlled trials (RCTs) by Wade and colleagues with promising results for online problem-solving training, improving behavior problems, executive functioning, and family functioning [15,19–22]. The use of technology in rehabilitation is relatively new, but telerehabilitation has shown to be a reliable alternative to in-person meetings [23,24] with promising results in cognitive rehabilitation [25], speech and language therapy [26] and family therapy [27,28]. Recent reviews on technology-based or -assisted rehabilitation programs for children with ABI have found that in addition to the mentioned evidence for online problem-solving, there is promising evidence for training of cognitive, social and behavioral skills [15,22,29,30]. The findings are characterized by heterogeneity and small samples, but the involvement of a clinician in addition to the technology-based intervention may be of importance for a positive result [30]. Although most of this research has been conducted in the USA, some of the technology-based intervention studies presented in the

literature reviews have also been performed in Europe. In addition, goal-oriented rehabilitation is considered a key ingredient in pABI rehabilitation [14,31], and goal attainment scaling can be a sensitive and meaningful way to measure rehabilitation outcomes [32]. Children's self-identified goals have been found to be as achievable as their parents' goals in a sample of children with a mean age of 9 years [33], pointing toward the importance of including the children in defining rehabilitation goals.

To our knowledge, no single intervention has included all these important perspectives simultaneously in a pABI-population. However, an intervention targeting individualized, everyday rehabilitation needs in a home setting has been carried out for the adult TBIpopulation in the US [34] and is currently ongoing in Norway [35]. This intervention defined individualized rehabilitation goals in collaboration with the patient and family member when available and worked on strategies to reach these goals over a period of four months. To meet the demands of holistic, person-centered and goal-oriented interventions for the pABI-population, this adult intervention was adapted to the pediatric population. In the Child in Context Intervention (CICI), the family and the child's school are included, and collaboration is established with the Norwegian Service for Special Needs Education ("Statped"). The intervention has largely been adapted to a telerehabilitation format to facilitate access to rehabilitation services, and to ease participation for families and schools.

The Medical Research Council [36] has pointed out the need to carry out feasibility studies as part of the development of complex interventions. This is especially important as the complexity of such studies is influenced not only by the intervention components, but also by contextual factors, in addition to the interactional processes occurring between the intervention components and the context [37]. In pediatric rehabilitation, one also needs to consider a range of transactional processes: between child and parent; between the child, family and school environment; and in the community and with peers, as children rely on their environment even more than adults [38]. In the CICI study, the complex interactions that may take place between children, parents, schools and the intervention components is hard to foresee before the intervention has been tried out. Some key uncertainties include whether the complex logistics of the study are feasible, whether the families are able to maintain participation in an intensive intervention (seven sessions over 4–5 months), whether recruitment is feasible, whether it will be possible to maintain a sustainable working alliance with both children and parents through the telerehabilitation format and whether the goal-oriented approach is feasible when working with an entire family that may not have the same priorities or needs. In addition, as there have been no previous studies carried out including schools in a similar rehabilitation program, it is also of importance to assess the feasibility of this intervention component. Conducting a feasibility study prior to the future definitive RCT is therefore principal in order to evaluate the intervention manual, identify possible obstacles and be able to adjust the protocol before the RCT.

#### *Objectives*

The objectives of the CICI feasibility study were to assess the feasibility of (1) the recruitment procedures and (2) the contents and structure of the intervention, including the feasibility of treatment delivery through a videoconference solution. We also wished to evaluate the acceptability of (3) the intervention for children, parents and therapists, (4) the baseline (T1) and outcome (T2) assessment methods and finally (5) the quality of treatment delivery. In order to inform the final decisions on outcome measures in the RCT, considering preliminary indications regarding the usefulness of outcome measures was also addressed.

#### **2. Materials and Methods**

This article adheres to the CONSORT extension for the Pilot and Feasibility Trials Checklist [39].

#### *2.1. Trial Design*

This feasibility trial applied a one-group pre−post design, with a baseline (T1) and a follow-up assessment immediately after the intervention period of 4-5 months (T2). The future definitive RCT will include a two-group RCT-design. For the RCT, outcome assessments will also be performed about 9 months after baseline (T3). The T3-assessment was not included in the feasibility trial to minimize time expenditure as T3 largely matches T2.

#### *2.2. Participants and Recruitment Procedures*

The inclusion criteria were (1) school-aged children (6–16 years at inclusion) with a radiologically verified diagnosis of ABI, or loss of consciousness post-insult and verified neurological symptoms in cases where radiology could not be administered; (2) time since insult at least 1 year; (3) self- or parent-reported persistent ABI-related cognitive, emotional or behavioral challenges influencing participation in everyday life related to family, friends, school or local community, assessed through a telephone interview; (4) children attending school regularly; and (5) the family is able to participate actively in a goal-oriented study for the next 4–5 months.

Exclusion criteria were (1) severe pre- or comorbid neurological or neuropsychological disorders that would confound assessment and/or outcome measurements; (2) children with brain tumors in active treatment or at great risk of relapse; (3) children with severe psychiatric illness or with injuries so severe that they were currently in institutionalized care; (4) parental severe psychiatric illness, drug abuse or indications of a history of or risk of domestic violence; and (5) not fluent in Norwegian language, although exceptions could be made for English-speaking parents who understand and read Norwegian.

Participants were identified from the medical charts at Sunnaas Rehabilitation Hospital. The families were invited to participate through written age-appropriate information for the children and information to parents. Parents and teenagers from 16 years of age provided written informed consent. A scripted telephone interview was used to screen for inclusion and exclusion criteria and willingness to participate. Eligible and consenting families performed a baseline screening (T1) at Sunnaas Rehabilitation Hospital.

After inclusion, the children's teachers and principals at their schools received written information about the study and consent form for the teachers. They were thereafter contacted by telephone by the CICI Statped collaborator.

#### *2.3. Assessments*

The outcome measures to be evaluated in the feasibility study are listed in Table 1. Neuropsychological assessment was included at baseline to provide descriptive data regarding the children with ABI and to inform the goal-setting process. Primary outcomes were post-concussive symptom burden (HBI), parenting self-efficacy (TOPSE) and quality of life (PedsQL). Questionnaires were completed at home and returned by mail.

As part of the baseline assessments, the families were asked to name the current three most challenging areas related to the child's brain injury. These were rated on a 5-point Likert scale according to how troublesome they were perceived in everyday life. Parents agreed on three areas but scaled them separately. The child was similarly asked to name and rate his or her own three most troublesome pABI-related problem areas.

#### *2.4. Intervention*

A detailed intervention manual was developed. It was based on the manual developed for an adult population by Borgen and colleagues [35] and adapted to the pediatric and family context by experienced rehabilitation therapists (authors M.L., S.L.H., I.M.H.B., M.V.F., N. R.-B., I.L.H.). In addition to descriptions of the content of each session, the manual contained general information on the study's rationale and aims as well as detailed guidance on how to establish SMART-goals and strategies, and references to evidence-based strategies and "tools" to handle different challenges. The manual also included detailed guidelines on how to manage therapy and communication through videoconference.


**Table 1.** Measures included in the feasibility study.

The intervention included seven family sessions, four school-meetings interspersed with the family sessions (starting after the first family-session) and a one-day parent group seminar (occurring about halfway through the family sessions for all participants) over the treatment period of four to five months. Figure 1 gives an overview of the intervention components. The family sessions and the school sessions were delivered through an encrypted videoconferencing solution. The feasibility study was performed from August to December 2020. See Adaptations due to COVID-19 below.

**Figure 1.** Overview of the intervention.

Three therapists, two clinical neuropsychologists and one experienced pediatric nurse (authors N.R.-B., I.L.H. and E.J.S., respectively) with extensive rehabilitation experience delivered the intervention. Therapists received training from the therapists of the adult community-based TBI-study [35,53] concerning the goal-oriented approach and strategies related to different commonly reported ABI-related problem areas. This was discussed in repeated meetings throughout the feasibility study. Therapists delivered nearly all the family sessions in pairs to ensure adherence to the study manual and a uniform delivery across therapists. The school sessions were performed by an experienced special education counselor from Statped (author A.P.S.-S.) in close collaboration with the three therapists. The therapists conducted the parent group seminar. Weekly meetings between the therapists, the Statped counselor and the study Principal Investigator (author M.L.) were held to discuss ongoing therapies and to ensure protocol adherence. The therapists also received training in the technical solutions of telerehabilitation as well as education on important therapeutic aspects of using this format, by a researcher with relevant experience (author H.P.Ø.), and the telerehabilitation team at Sunnaas Rehabilitation Hospital.
