Vestibular Infant Screening-Rehabilitation

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Correspondence to Marieke Fontaine; [email protected]

Strengths and limitations of this study

Building on the VIS-Flanders project, the Vestibular Infant Screening-Rehabilitation (VIS-REHAB) study will be conducted in a unique Flanders-based setting, leveraging a well-established multidisciplinary network and the research group’s expertise in paediatric vestibular assessment, within a compact geographical area that facilitates treatment by the peripheral physical therapist supervised by one person to ensure standardisation.
The VIS-REHAB study addresses several important limitations previously identified in paediatric Vestibular Rehabilitation Therapy (VRT) research by focusing on vestibular dysfunction (both partial and complete loss of function) rather than hearing impairment for inclusion, by implementing age stratification to account for the child’s age in relation to vestibular system maturation, and by providing a detailed description of exercise content, doses, duration and progression to enhance study replicability.
The VIS-REHAB protocol includes gaze stability exercises and employs outcome measures to assess gaze stability in vestibular-impaired children, in contrast with prior research and treatment in the paediatric field that predominantly concentrated on postural stability.
The VIS-REHAB study addresses fundamental questions about conventional VRT, establishing an essential foundation for exploring advanced and engaging alternative VRT modalities in the future.
The VIS-REHAB study involves a 9-week period during which therapy is temporarily halted (ie, CTRL protocol), raising ethical concerns due to the potential impact on the child’s motor development.

Introduction

Our balance system (figure 1) is an ingenious sensorimotor complex which relies on the peripheral vestibular, visual and somatosensory pathways as most important input systems (figure 1A). The central processing of this input information occurs at the level of the brainstem and cerebellum and allows for several output functions (figure 1B) of which the reflexive preservation of both gaze during movements through the vestibulo-ocular reflex (VOR) and posture through the vestibulospinal and vestibulocervical reflexes (VSR and VCR) are the most obvious ones. However, via neural projections to the cortex and other central structures, the balance system also has a critical impact on a variety of cognitive processes, such as perceptual/visuospatial ability, memory, attention, emotion and executive functions.¹ The various components of the balance system are fully developed at birth, but its maturation continues until adolescence.² A dysfunction in one of the input systems or central processing mechanisms, may lead to inadequate output, which can manifest in various ways. In case of a vestibular deficit in children, problems in the reflexive control of gaze and postural stabilisation may result in poor motor and balance development, delayed gross motor milestones, as well as problems with fine motor skills, because of hampered eye–hand coordination. These problems are already visible at an early age, but later in life functional abilities, practice of sports or recreational activities can also be affected.³ A vestibular dysfunction may also lead to problems in cognitive performance, emotional and social behaviour or educational performance, which often become only apparent at a later age when children start kindergarten or primary school.^{1 4–6}

View Image - Figure 1. The vestibular system and its most important input systems (A) and output functions (B) (figure adapted and translated from Dhondt et al 81 ).

Figure 1. The vestibular system and its most important input systems (A) and output functions (B) (figure adapted and translated from Dhondt et al 81 ).

Especially in case of an early-onset severe vestibular dysfunction and in situations where other sensory input systems are additionally hampered, a vestibular deficit has profound implications for the overall development of young children.⁷ Therefore, it is of utmost importance that vestibular-induced problems are diagnosed and treated as early and effectively as possible.^8–11 In adults, this is typically addressed by means of Vestibular Rehabilitation Therapy (VRT); a movement and exercise-based approach using the plasticity and neural networks of the brain, in order to improve functional activities and overall quality of life.¹² By means of ‘sensory reweighting’, the subject learns to compensate for the deficient vestibular input, by relying more on the input of the other sensory systems, or by exploiting what is left of the vestibular input.¹² Thanks to the superior neural plasticity of children compared with adults, it was presumed that their balance system would evolve spontaneously towards a mechanism with optimal function despite the absent or reduced vestibular input. Therefore, for decades, the potential impact of vestibular dysfunction in children was assumed to be negligible or temporary, and the necessity of VRT was ignored. However, more recently, an increasing number of studies indicate that vestibular dysfunctions in children can lead to various disorders ranging from delayed gross motor development to decreased cognitive performance.^{1 5 13–19} Moreover, research has already shown that without intervention, the gap in gross motor development between vestibular-impaired children and healthy peers expands with increasing age.¹⁴ These findings could be related to the maturation of the balance system, as was mentioned before.² Several studies show that children with early-onset vestibular dysfunction struggle with tasks that rely on sensory reweighting.^{14 20} This finding suggests a mutual dependence of sensory input systems. In other words, if the vestibular input is disrupted before maturation, it may hinder the integration of the complex balance system.¹⁴ This implies that vestibular dysfunctions early in life can be more disabling than adult-onset dysfunctions as they lead to poor development of the ability to compensate for the dysfunction. Consequently, VRT might be considered even more essential in children than in adults.

VRT was first described in adults in the 1940s as a form of group therapy.^{21 22} The aim of VRT is threefold; improving gaze as well as postural stability (PS) and diminishing unwanted symptoms (eg, vertigo and nausea). In order to reach these objectives, recovery mechanisms based on three different learning principles are deployed: habituation (reducing specific symptoms by repeated exposure to the stimulus that is causing this symptom), substitution (replacing a less effective strategy by another) and adaptation (causing long-term changes in the neural reaction to a specific, altered input). Because recovery mechanisms based on adaptation rely on an ‘altered’ input, these require the presence of some residual vestibular function. On the contrary, recovery mechanisms based on substitution and habituation, are always applicable; both in reduced (vestibular hypofunction) and complete loss of function (vestibular areflexia). The effectiveness of VRT has been sufficiently proven in adults. In 2015, a Cochrane review reported moderate to strong evidence that VRT is safe and effective for adults with peripheral vestibular dysfunction, based on a number of high-quality randomised controlled trials (RCTs).²³ Moreover, a recent update of the evidence-based clinical practice recommendations has been published²⁴ and various educational programmes are available. This clinical guideline recommends incorporating home exercises into the treatment plan alongside regular clinical visits, among other recommendations. Home-based exercise programmes within VRT have demonstrated effectiveness in adults.^25–32

Application of VRT in the paediatric population has also been proven effective. A recent systematic review³ delivered promising evidence that VRT programmes improve the postural control, balance and gait of (hearing-impaired) children, based on a limited number of six randomised and quasi-RCTs. The same authors have also published two other recent systematic reviews,^{33 34} focusing on motivational and playful alternatives to standard VRT aiming at improving therapy adherence. In these reviews, sports and recreational activities, as well as virtual reality-based games were found to enhance postural control, balance and gait in hearing-impaired children, supported by five and three randomised and quasi-randomised trials, respectively.

At the same time, the authors^{3 33 34} highlight the low quality of evidence of the studies due to several methodological limitations (online supplemental appendix table 1). Although several of these limitations (eg, concerning the size or calculation of the sample, the anonymous allocation, the exercises used in the intervention) are clear, others may require further explanation.

In the majority of past paediatric studies on the effectiveness of VRT, recruitment and inclusion of subjects was based on hearing impairment,^35–38 which is closely linked to vestibular impairment due to the anatomical relationship of both end organs.³⁹ The reason why vestibular (dys)function was not used as inclusion criterion or was not even evaluated at all, has probably to do with the fact that vestibular assessment in children has long been considered challenging and is therefore not yet included in common practice. Nevertheless, this is a critical limitation of past research. Adult studies show that progress with and outcome after VRT is more promising in patients with partial vestibular function compared with those with complete function loss. This is related to the extent to which certain recovery mechanisms (based on adaptation, substitution and habituation) can be deployed. Additionally, inclusion on the basis of hearing (and not vestibular) impairment does not rule out the inclusion of children without any vestibular loss. Hence, these studies do not serve as a good basis to draw reliable conclusions on the effectiveness of VRT in children with vestibular dysfunction. The limited number of studies that did set vestibular dysfunction as primary inclusion criterion, chose to include children with bilateral dysfunction only.^{20 40} These studies fail to answer the question of the effectiveness of VRT in children with unilateral dysfunction. In short, there is an urgent need for a large-scale study in children with a proven vestibular dysfunction so that the effect of and the outcome after VRT can be linked to the extent of residual vestibular function.

Details on the subject’s hearing are also important in these studies. The auditory function should be monitored closely as it (although to a lesser degree than the other input systems) covers one of the input systems that provides the balance system of sensory information. Auditory input itself can therefore be beneficial for balance.⁴¹ For that reason, VRT in hearing-impaired children should be carried out in the best aided condition (with hearing aids and/or cochlear implants (CIs)), so that the child learns to maximise its abilities in the most optimal sensory and the most realistic (as much similar to daily life as possible) conditions.

A last important factor to take into account in the study design is the impact of a child’s age. As mentioned before, the maturation of the balance system continues until adolescence.² Many questions on this issue remain, but some seem already uncovered. The critical period for the development of the intersensory organisation of the balance system, for example, is thought to end at 4–6 years of age.⁴⁰ Postural control, on the other hand, would reach adult-like level at 7–10 years of age.^{41 42} Additionally, the initiation of CIs in hearing-impaired children is defined by the critical developmental periods of the auditory cortex.⁴³ These examples illustrate that it is important to consider the fact that the benefit from vestibular rehabilitation might be age-dependent and that stratification in age categories is appropriate.

Unfortunately, the review on standard VRT³ only incorporated studies aiming to improve postural control, and did not provide information on the effectiveness of gaze stability (GS). Furthermore, the interventions examined in the clinical trials included in the systematic review on virtual reality³³ consist solely of virtual reality-based games specifically designed to enhance postural control. This reflects the current form of VRT in the paediatric population, which is primarily focused on PS. No RCTs on the effectiveness of gaze stabilisation have been performed in children to our knowledge and paediatric physical therapists are theoretically and practically limited, or even completely unfamiliar with the concepts of GS training. This might be related to the fact that GS is primarily driven by the VOR and the theoretical background on GS requires a certain knowledge of the vestibular organ and the complex balance system. This is in contrast to PS, which is the result of the VSR and VCR pathways, that are more closely related to the expertise of physical therapists. Even among adults, there is a lack of comprehensive reporting on intervention parameters and limited utilisation of outcome measures specific to gaze stabilisation in spite of the widespread incorporation of gaze stabilisation exercises.⁴⁴ Nevertheless, GS in children seems to be as important as in adults, since a preliminary study of Braswell and Rine¹⁶ suggests that exercises which have been shown to be effective in adults have a similar effect in children. Furthermore, a recent observational study evaluated the effectiveness of GS training in vestibular-impaired children, using a game with a head-mounted sensor measuring angular head movements, and revealed a significant improvement in GS.⁴⁵

The above emphasises the necessity for a high-quality RCT in order to address these shortcomings. Based on literature review and clinical practice considerations, a consensus protocol named the Vestibular Infant Screening-Rehabilitation (VIS-REHAB) protocol was developed and will be presented below. This protocol aims to elucidate the effectiveness of VRT in vestibular-impaired children of different age categories. It includes the following objectives: (1) to investigate the short-term effect of a structured, combined postural control and gaze stabilisation protocol (VIS-REHAB protocol) in a group of vestibular-impaired children, compared with receiving no therapy (CTRL protocol) and (2) to investigate the most important factors that may influence the effect of and outcome after application of the VIS-REHAB protocol in a group of vestibular-impaired children. This study aims to address lingering questions in the existing literature regarding VRT in a standardised manner, with the ultimate objective to develop comprehensive rehabilitation guidelines tailored to paediatric VRT. This information will result in an improved motor development of vestibular-impaired children, by giving them access to the most effective therapy programme, thereby limiting the impact of a vestibular dysfunction on their motor, cognitive, socioemotional and educational outcome. Eventually, our future efforts will focus on incorporating more engaging elements such as sports, recreational activities, virtual reality and games to improve adherence and feasibility of therapy, consequently aiming to provide benefits for children, parents and therapists alike. Moreover, these alternative VRT modalities can effectively facilitate the integration of a home-based exercise programme for children. The implementation of these novelties in a validated manner hinges on obtaining answers regarding the effectiveness of conventional VRT, which is currently unavailable.

Methods

Study design

The study is a two-parallel group, superiority, randomised controlled crossover trial with 1:1 allocation ratio. An RCT will be used to assess and compare the effect of the VIS-REHAB protocol with that of receiving no therapy (CTRL protocol) using a parallel design. Adding a crossover design has the advantage that all patients have access to the best possible care (figure 2). Furthermore, considering the heterogeneity of the children in the study, differences between groups unrelated to the intervention effects may arise. In such instances, using patients as their own controls allows for additional exploratory crossover analysis. Moreover, as both groups undergo the active rehabilitation protocol, subjective outcome measures assessing the overall feasibility of therapy can be evaluated in both cohorts. It has the disadvantage that the long-term effect of the therapy programme cannot be evaluated as they succeed each other. However, the assessment of long-term effects is inherently challenging, given that children cannot be withdrawn from therapy for an extended period, and they will resume their rehabilitation trajectory from before after participating in the study. In conclusion, the crossover design was considered most appropriate due to the aforementioned ethical considerations.

Figure 2. Study design. CTRL, control; IN, input evaluation; OM.I, primary outcome measures; OM.II, secondary outcome measures; VIS-REHAB, Vestibular Infant Screening-Rehabilitation.

Study setting

This study is embedded in the Paediatric Vestibular Research Group at the Ear, Nose, and Throat (ENT) department of Ghent University Hospital and the Department of Rehabilitation Sciences of Ghent University. Ghent University (Hospital) is internationally recognised as a centre of expertise in paediatric vestibular research. The VIS-REHAB study is a continuation of the Vestibular Infant Screening (VIS)- Flanders project.^{39 46–50} This project aimed to give each infant with a congenital or early-onset permanent hearing loss in Flanders since 2018 access to a basic screening of the vestibular function at the age of 6 months, as hearing-impaired children are considered a high-risk group for developing vestibular deficits. To ensure early diagnosis leading to prompt treatment, the VIS-Flanders project incorporated referral for motor evaluation and follow-up after a 'fail' on the vestibular screening. This approach aimed to minimise motor developmental delay through neurodevelopmental treatment provided by a licensed paediatric physical therapist. As the youngest children screened through the VIS-Flanders project are now 3 years, making effective VRT feasible, there is an opportunity to enhance motor and other developmental areas. This unique situation in Flanders serves as an ideal foundation for the enrolment of participants in the VIS-REHAB study. The interventions included in the VIS-REHAB study will be delivered at the peripheral physiotherapeutic practice, at the patient’s home or at Ghent University Hospital, tailored to the individual circumstances of each patient.

Eligibility criteria

Inclusion criteria

Only patients with an identified peripheral vestibular dysfunction with or without concomitant hearing loss and/or CI will be included in this study. Children will be tested and rehabilitated in the best aided condition (ie, with hearing aids and/or CI). Children between 3 and 17 years of age will be enrolled. As the critical period for the development of the intersensory organisation in children has been determined to continue up to 4–6 years of age, ideally, vestibular rehabilitation should be introduced before the age of 4.⁴⁰ The targeted approach we want to adopt in our VRT requires a certain level of cognitive maturity and working attitude. That is why the lower age limit is set at 3 years of age. The upper age limit is set at 17 years, as this is the upper age limit of the Movement Assessment Battery for Children,⁵¹ which is used as an input evaluation in this study. Although it is important to initiate therapy as early as possible to achieve the best possible outcome, there is also evidence that supports the assertion that late therapy-onset can still lead to functional improvement and a longer interval between the onset of the vestibular dysfunction and the start of the therapy does not need to be a contraindication.^{52 53} Participants will only be included after receiving a signed informed consent from the child and/or their parents after a clear and complete explanation of the study.

Exclusion criteria

For reasons mentioned above, children incapable to understand simple instructions (due to severe cognitive disorders, impaired language comprehension, etc) will not be eligible for this study. Participants with severe disorders of the other primary sensory input systems for balance function (figure 1A) will also be excluded, as the research objective is to assess the effectiveness of VRT for patients with isolated (audio)vestibular dysfunctions. These include patients with severe neuromotor disorders (incapable of independent standing and walking), severe muscle tone disorders (eg, cerebral palsy) or severe orthopaedic dysfunctions and patients with uncorrected vision problems. As CI can have a substantial impact on the vestibular function, children with planned CI surgery within 5 months before or during the study period are not eligible. The cut-off was set at 5 months based on the results of a study in our own paediatric CI population in which we did not find significant changes in the vestibular assessment scheduled on average 5 months after surgery.⁵⁴

Intervention

VIS-REHAB protocol

Content

The proposed VIS-REHAB protocol can be considered a consensus protocol comprising two essential components: PS and GS. This consensus was reached based on plentiful adult research, guidelines and therapy programmes, and previous studies on the effectiveness of paediatric VRT, as well as clinical therapeutic expertise acquired in the past 4 years of the VIS-Flanders project.^{46 48}

Within the PS component of the VIS-REHAB protocol, there are static and dynamic balance exercises, along with general gross motor activities targeting core stability, agility and bilateral coordination. The GS part includes exercises enhancing oculomotor function such as smooth pursuit and saccadic movements, as well as VOR exercises. Additionally, a general gross motor training programme emphasises GS by including exercises that improve eye–foot and eye–hand coordination (figure 3). Furthermore, each session includes counselling and background information on the exercises.

View Image - Figure 3. Overview of the two intervention protocols. CTRL, control; GS, gaze stability; PS, postural stability; VIS-REHAB, Vestibular Infant Screening-Rehabilitation.

Figure 3. Overview of the two intervention protocols. CTRL, control; GS, gaze stability; PS, postural stability; VIS-REHAB, Vestibular Infant Screening-Rehabilitation.

Both the PS and GS components entail a very specific part on the one hand (eg, static and dynamic PS for the PS component) and a general gross motor part on the other hand in which specific learning experiences can be put into practice. The general gross motor part is valuable to ensure high motivation and smooth transfer to everyday life. Moreover, as vestibular dysfunctions in children primarily affect the gross motor function (because good postural control and GS are important prerequisites for adequate gross motor development), specific attention to the gross motor skills is appropriate.

A basic exercise is defined for each exercise category within the PS and GS component (static PS, dynamic PS, oculomotor function and GS), which can be adjusted to the child’s individual abilities based on a clear-cut progression grid (online supplemental appendix tables 2 and 3). For each patient, therapists will track all applied settings per session in a patient chart. Exercises can be increased in difficulty when accurate performance of the exercises is achieved with no or limited vestibular symptoms (ie, only mild to moderate dizziness that resolves within 20 min³⁰).

Doses and duration

The VIS-REHAB protocol entails 30 min sessions conducted twice a week over a span of 9 weeks. These sessions are structured with 20 min dedicated to enhancing PS and 10 min focused on improving GS. In more detail, a minimum of 5 min is reserved for counselling and providing background information on the exercises, focused on the vestibular issue. Apart from the counselling part, each session comprises three of the following exercise categories: static PS, dynamic PS, general gross motor training (focus PS), oculomotor function, GS or general gross motor training (focus GS). Each category is conducted for 5–10 min.

The defined amount of sessions and duration of the therapy is based on scientific evidence as well as practical considerations. In the review of Melo et al ³ and Fernandes et al,⁵⁵ positive effects were reported on interventions with sessions of 30 minutes or longer and a total duration of 6 or more weeks,^{20 36–38 40} further supported by Zhou and Qi,⁵⁶ revealing that exercise interventions are more effective when extended beyond 8 weeks. Therapy sessions are divided into two 30-minute sessions per week instead of a single 60-minute session to accommodate the limited attention span of children and the often busy after-school schedules of both children and parents. Moreover, this approach ensures that exercises are distributed throughout the week, providing better opportunities for repetition and motor learning.

CTRL protocol

Content

In the control (CTRL) protocol, all forms of physical therapy in the context of motor development are ceased. Occupational therapy, speech-language therapy and physical therapy for other purposes are not covered by this commitment. Additionally, the child and parents are asked to not do any home exercises on their own. However, sports activities and other recreational hobbies will not be asked to be temporary halted, since they will be continued in the active rehabilitation programme as well.

Doses and duration

Physical therapy is temporarily suspended for a duration of 9 weeks.

Intervention delivery

The patient’s own physical therapist will administer the VIS-REHAB protocol. This approach ensures that the study does not compromise treatment opportunities for the physical therapist and maintains a familiar environment for the child. Clear rules and guidelines, encompassing therapy content, frequency, doses and duration, along with assistance and supervision from the coordinating physical therapist assigned to this project, will be provided. This ensures that the VIS-REHAB protocol is standardised and comparable across different patients and therapists. The collaborative physical therapy sessions aim to provide training in the correct application of VRT, enabling physical therapists to independently apply acquired insights in their clinical practice, separate from the scientific aspect. If the patient is not undergoing regular therapy or attends only once a week, the coordinating physical therapist will conduct additional therapy sessions to adhere to the study protocol. Depending on the preferences of the child and parents, as well as their frequently busy school and after-school schedules, these (extra) physical therapy sessions can take place either at home or at Ghent University Hospital.

Outcome measures

Input evaluation

Extensive evaluation of the vestibular and visual system will precede patient inclusion, accompanied by the collection of information pertaining to the auditory system, patient characteristics and medical history. These sensory input and processing functions of the vestibular system (figure 1A) might have an influence on the effect of or the outcome after VRT. Additionally, the MABC-2 will be used to evaluate and classify the children’s motor performance at the beginning of the study. This data will be used for inclusion, exclusion and stratifying purposes or for in-depth analysis of the results.

Vestibular assessment

Video Head Impulse Test (vHIT)

The vHIT assesses the superior and inferior vestibular nerve and the functioning of the six semicircular canals, using the VOR. vHIT measurements will be conducted using the ICS Impulse system (GN Otometrics, Taastrup, Denmark) and accompanying software ‘Otosuite’. Eye movements of the right eye will be recorded with a high-speed camera fixed on a lightweight goggle. The children will be instructed to sit on a chair and fixate an attractive visual target (ie, movie on a tablet) at 1.50 m distance. Meanwhile the examiner will perform unpredictable head movements (10°–20° amplitude) in, respectively, the horizontal, LARP (to stimulate the left anterior and right posterior canal), and RALP plane (to stimulate the right anterior and left posterior canal). Prior to interpretation of the results, the data will be thoroughly cleaned according to the following criteria: (1) head velocity between 120°/s (vertical) or 150°/s (horizontal) and 250°/s (upper limit for vertical and horizontal), and (2) head bounce below 25% of the peak head velocity. The measured gain (of the VOR) (%), the symmetry between the left and right side (%) and the presence of covert/overt saccades (n, and % of the performed HITs) will be taken as outcome measures of this test.⁵⁷

Cervical Vestibular Evoqued Myogenic Potentials (cVEMP)

The integrity of the saccule and the inferior vestibular nerve (by means of the VCR), will be investigated by a cVEMP test, using the Neuro-Audio equipment (version 2010, Neurosoft, Ivanovo, Russia) and accompanying software. For the cVEMP, air-conducted 500 Hz tone bursts of 95 dBnHL will be presented monaurally through insert earphones to elicit the responses and the response will be measured using four small self-adhesive Ag/AgCl surface electrodes (Blue Sensor, Ambu) applied on the upper one-third part of the sternocleidomastoid muscle (SCM) (active), on the sternum just beneath the interclavicular ligament (reference), and on the nasion (ground). Contraction of the SCM muscle, necessary for this examination, will be achieved by lifting and rotating the child’s head to the non-stimulus side in supine position. Outcome measures that will be included in the database are the absolute latencies of P1 and N1 (ms), rectified interpeak amplitude, asymmetry ratio (%) and absence/presence of the cVEMP response.⁵⁷

Ocular Vestibular Evoked Myogenic Potentials (oVEMP)

The oVEMP test, carried out with the same Neuro-Audio equipment, will be used to examine the functioning of the utricle and the superior vestibular nerve (by means of the VOR). To provoke this specific VOR response, a mini-shaker (500 Hz stimulus (2–2–2 ms) with an intensity of 140 dB force level) will be used. In supine position, an upward gaze of 30° will be ensured by a fixation mark on the ceiling. If necessary, a smartphone playing a movie will be attached to the wall to elicit the upward gaze. The responses will be measured using electrodes on the inferior oblique muscle just below the lateral canthus of the eye, the reference electrode next to the medial eye canthus on the nose, and the common electrode on the nasion. The absolute latencies of N1 and P1 (ms), interpeak amplitude (µV), asymmetry ratio (%) and absence/presence of the oVEMP response will be the reported outcome measures.⁵⁷

Visual assessment

Static Visual Acuity (SVA)

For the SVA test, subjects will be seated 3 m from a visual target. Subjects who wear glasses or contact lenses will be encouraged to wear them during the examination, as this is mainly a functional screening for daily life situations. SVA measurements will be conducted using the DVAstar Head Sensor (DIFRA Instrumentation, Eupen, Belgium) and accompanying software ‘DiSoft II’. The optotype (the letter ‘C’) will randomly rotate each trial by 0°, 90°, 180° or 270° and subjects will be asked to report the direction of the opening of the ‘C’ (right, left, up, down). The optotype size will decrease in steps equivalent to a visual acuity change of 0.1 LogMAR (log10X, where X=the minimum angle resolved, in arcmin, with 1 arcmin=1/60°). The purpose of SVA is to determine the smallest optotype that can be correctly identified while the head is fixed. For SVA, the optotype will decrease in size until the participant fails to correctly identify three optotypes at the same acuity level or reached the LogMAR value of 0.0.¹²

Oculomotor Function testing

Oculomotor function testing will be carried out with the ICS Impulse system (GN Otometrics, Taastrup, Denmark) and accompanying monocular oculomotor software. During smooth pursuit testing, subjects will be asked to follow a target slowly moving from left to right and from top to bottom (15° from centre). Reported response parameters include gain (%) and phase (°). For saccade testing, subjects will be instructed to move their gaze quickly back and forth, fixed on targets popping up 15° left and right, and up and down from centre. Target deviation (%), latency (ms) and maximum velocity (°/s) will be included as response parameters.¹²

Motor assessment

Movement Assessment Battery for Children, 2nd edition (MABC-2)

The MABC-2 is one of the most widely used assessment tools to evaluate a child’s motor performance. The purpose of the MABC-2 is the identification and description of impairments in motor performance of children between 3 and 17 years of age. Children have to complete a series of fine and gross motor tasks grouped into three categories: manual dexterity, aiming and catching and balance. These eight different and age appropriate tasks will yield a total score of the MABC-2, subscale scores and item scores to determine the level of motor competence of the participant.⁵¹

Audiological assessment

Pure tone audiometry will be retrieved from the attending ENT specialist. Pure tone audiometry without (unaided condition) as well as with (aided condition) listening devices (hearing aids and/or CI) will be collected.

Patient characteristics and patient history

A list of other variables that might have an impact on outcome will be questioned through a structured parent survey, such as chronological age, age of onset of the vestibular dysfunction, aetiology of the (audio)vestibular dysfunction, details on cochlear implantation (side, date), details on history of physical therapy, developmental factors (eg, motor milestones) or disabilities (eg, autism, developmental coordination disorder), physical activity (eg, sports), musculoskeletal or neuromuscular comorbidities, cognitive comorbidities, medication usage (eg, vestibular suppressants, ototoxic medications) and psychological factors (eg, sleep difficulties, anxiety, depression, fear of movement, fear of falling).

Output evaluation

The effect of VRT on the output functions of the vestibular system will be evaluated based on the selected primary and secondary outcome measures. These measures have been proven valid and reliable for the use in both normal-hearing and hearing-impaired children and have been used in previous studies on the effectiveness of VRT in children.^{16 36 58–60}

Primary outcome measures

Given the equal significance of both the postural control and GS component in the study, two primary outcome measures have been chosen.

Postural stability outcome measure

Modified Clinical Test of Sensory Integration on Balance (mCTSIB)

The mCTSIB will be used to assess static PS as well as the interaction and reliance on the most important sensory inputs for maintaining balance (ie, vision, somatosensory and vestibular information). The child stands barefoot with feet together as still as possible for 30 s. There are eight test conditions: eyes open or closed, with or without foam, and eyes open or closed while on a foam, nodding the head in yaw or pitch at 0.33 Hz. The test will be conducted on a force platform, a Wii Balance Board (Nintendo Co), using the Colorado University BrainBLoX software. The total test time (s), anteroposterior and mediolateral sway (mm), centre of pressure path length (cm), sway velocity (m/s) and 95% confidence ellipse area (cm²) are measured by a custom-made code in MATLAB (The MathWorks, Inc, Natick, Massachusetts, USA).^61–64

Gaze stability outcome measure

Dynamic Visual Acuity (DVA)

The DVA test is an evaluation of GS. The setup of the test is similar to that of the Static Visual Acuity test, the only difference being that the patient’s head will be passively moved by the examiner in the horizontal and vertical plane at a 2 Hz frequency over an amplitude of 15° from centre. The difference between the SVA and the DVA score will be included as output measure.⁵⁸

Secondary outcome measures

Postural stability outcome measures

Single Leg Sanding (SLS) Test

The SLS test is used to evaluate static PS. Subjects will be instructed to stand on one leg as long as possible. After explanation and demonstration of the procedure, children will be granted one attempt (for 10 s) to get familiar with the exercise. SLS will be carried out both with eyes open and with eyes closed and three trials will be conducted. The test will be conducted on a force platform, a Wii Balance Board (Nintendo Co), using the Colorado University BrainBloX software. The total test time (s), anteroposterior and mediolateral sway (mm), centre of pressure path length (cm), sway velocity (m/s) and 95% confidence ellipse area (cm²) are measured.^{60 65 66}

Paediatric Functional Reach Test

The Paediatric Functional Reach Test is a measure for dynamic PS in both forward and lateral directions. After explanation and demonstration of the test, children will be granted one attempt to get familiar with the exercise. Children will be instructed to reach as far forward and sideward as possible with their hand(s) and without moving the feet. Three trials will be conducted in each condition. The furthest reaching point subtracted by the reaching distance in neutral, symmetrical posture will be included for analysis (cm).^{59 67}

Timed Up and Go Test (TUG)

The TUG test assesses functional mobility and dynamic PS during walking. Subjects sitting 3 m away from a 1m × 1m square with a cone in the centre, will be asked on the ‘go’ command to get up from the chair, walk to the square, navigate around the cone, come back to the chair and have a seat. Before the test, the procedure will be explained and demonstrated. Three trials will be conducted and the best time (recorded with stopwatch) will be used for analysis. Outcome parameters include the total test duration (s), number of steps, walking speed (m/s) and mean step length (cm).^68–71

Paediatric Modified Dynamic Gait Index (DGI)

The Paediatric Modified DGI is used to evaluate dynamic PS during walking through eight tasks, including walking on a flat surface, adjusting speed and walking with horizontal and vertical head movements. It also evaluated walking over and around obstacles, turning while walking and stepping on stairs. Each task is scored on a 3-point scale, with 3 indicating normal performance and 0 indicating severe impairment. The total score will serve as the outcome parameter.^{72 73}

Gaze stability outcome measures

Video Head Impulse Test (vHIT)

See Vestibular assessment section for a detailed description.⁵⁷

Functional Head Impulse Test (fHIT)

The fHIT is a functional evaluation of the VOR. fHIT measurements will be conducted using the DVAstar Head Sensor (DIFRA Instrumentation, Eupen, Belgium) and accompanying software ‘DiSoft II’. Participants will be seated 3m away from a visual target, and those who wear glasses or contact lenses will be encouraged to use them during the examination. The child must identify the orientation of the optotype (the letter 'C') that briefly appears on a computer screen for 80 ms during unpredictable passive head movements (10°–20° amplitude) in, respectively, the horizontal, LARP (to stimulate the left anterior and right posterior canal) and RALP plane (to stimulate the right anterior and left posterior canal). In each trial, the optotype is randomly rotated by 0°, 90°, 180° or 270°. The size of the optotype corresponds to the SVA plus 0.6 LogMAR. The outcome parameter includes the percentage of correctly identified optotypes.^{74 75}

Gaze Stabilisation test (GST)

The GST is a functional evaluation of the VOR and provides velocity-dependent insights into the VOR’s role in maintaining GS. GST measurements will be conducted using the DVAstar Head Sensor (DIFRA Instrumentation, Eupen, Belgium) and accompanying software ‘DiSoft II’. Participants will be seated 3 m away from a visual target, and those who wear glasses or contact lenses will be encouraged to use them during the examination. The child must identify the orientation of the optotype (the letter ‘C’) while passively moving the head from 40°/s (30 BPM) to 200°/s (150 BPM) in steps of 40°/s (30 BPM) in the horizontal and vertical plane (20° amplitude). In each trial, the optotype is randomly rotated by 0°, 90°, 180° or 270°. The size of the optotype corresponds to the SVA plus 0.2 LogMAR. The outcome parameter represents the maximum head velocity (°/s) at which the optotype can still be correctly identified.^{76 77}

Motor performance outcome measures

Bruininks-Oseretsky Test of Motor Proficiency 2 (BOT-2)

The BOT-2 is a comprehensive assessment that evaluates both fine and gross motor skills through nine subtests. Each subtest yields an independent score, covering areas such as fine motor precision, fine motor integration, manual dexterity, upper-limb coordination, bilateral coordination, balance, running speed and agility and strength. These individual scores contribute to composite scores for fine manual skills, manual coordination, body coordination, strength and agility, fine motor skills, gross motor skills and overall motor performance, offering a comprehensive assessment of motor abilities.^{78 79}

Quality of life outcome measures

Paediatric Quality of Life Inventory (PedsQL) 4.0

The PedsQL will be used to evaluate quality of life. It is a 23-item inventory, applicable for (parents of) children from 2 to 18 year. It consists of four subdomains: (1) physical functioning (eight items), (2) emotional functioning (five items), (3) social functioning (five items) and (4) school functioning (five items). Items are scored through a 5-point rating scale.⁸⁰

Subjective outcome measures

Following each therapy session, assessment of subjective outcomes will be conducted. Two concise questionnaires will be used, incorporating Visual Analogue Scale (VAS) evaluations: one for the child (with assistance from the parents if required) and another for the treating physical therapist. These questionnaires aim to assess the overall feasibility of the exercise protocol. In more detail, the child’s questionnaire seeks to evaluate the child’s enjoyment, the perceived difficulty level for each specific exercise, and any potential symptoms (eg, dizziness, headache or neck pain) experienced during practice. The questionnaire, along with the VAS evaluations, will be adjusted to suit the child’s comprehension level, for instance, by employing emoticons instead of numerical scales. The questionnaire for the physical therapist assesses the feasibility of the exercises, as well as the motivation and task comprehension of the child during practice. In conjunction with these questionnaires, the coordinating physical therapist closely monitors subjective experiences during exercises and records reasons for halting, progressing or regressing the exercises. The data derived from the subjective outcome measures in the VIS-REHAB study will be analysed and interpreted, aiming to determine the most suitable exercises and dosage to administer based on the age of the child, as well as the severity and type of vestibular dysfunction.

Participant timeline

The first step in the trajectory entails a complete input evaluation (IN), followed by a 1-week rest period during which the patient ceases their current therapy. During this interval, the primary and secondary outcome measures (OM.I and OM.II) are assessed for the first time (figure 2). Between protocols, a 1-week rest period will be used to carry out the practical realisation of the rehabilitation trajectory for the particular patient and to assess the primary and secondary outcome measures (OM.I and OM.II). After each therapy session in the VIS-REHAB protocol, subjective outcome measures will be assessed. After having completed the two protocols, the final 1-week rest period will be used to conduct the last assessment of primary and secondary outcome measures (OM.I and OM.II). Additionally, during this period, arrangements for a potential resumption of therapy by the treating therapist will be made. At the conclusion of the entire trajectory, the input evaluation will be repeated to be able to identify possible changes in sensory input (ie, in the peripheral vestibular, visual and auditory system; cf. figure 1).

Sample size calculation

Given the novelty of the research, studies with an identical test design in children with confirmed vestibular dysfunction are lacking. The study of Rajendran et al ³⁶ is most similar and assessed the effectiveness of a combined (postural control and gaze stabilisation) VRT programme on motor skills and balance in children with hearing impairment, compared with a group of children with no therapy. This study indicated a significant improvement in all outcome measures (eg, Single Leg Standing, Bipedal Leg Standing, Paediatric Functional Reach) compared with the control group. The current study compares three age groups and two vestibular groups, which amounts to six subgroups per trajectory. Two primary outcome measures (ie, Modified Clinical Test of Sensory Integration on Balance and Dynamic Visual Acuity test) will be studied in these groups. To compensate for two primary endpoints, a Bonferroni correction will be applied, leading to an alpha level of 0.025. Aiming for a power of 90%, with the use of a two-sample t-test, the required sample sizes were calculated based on two conditions (foam—eyes open and foam—eyes closed) of the Bipedal Leg Standing Test, which are also part of the Modified Clinical Test of Sensory Integration on Balance. Table 1 illustrates the results of the Bipedal Leg Standing Test across the four outcome variables, with a minimum sample size of three and a maximum sample size of four. The latter was selected as the required sample size for each subgroup, leading to a sample of 24 per therapy trajectory. Taking into account a possible 20% dropout rate, a total of 30 children will be included in each therapy trajectory, leading to a total sample size of 60 children.

Table 1

Sample size calculation

Mean	SD	α level	Power	Sample size
Bipedal Leg Standing Test
Anteroposterior sway: eyes open
Experimental: −6.81 Controls: 0.91	1.83	0.025	90%	4
Anteroposterior sway: eyes closed
Experimental: −7.77 Controls: −0.13	1.5	0.025	90%	3
Mediolateral sway: eyes open
Experimental: −10.77 Controls: 0.004	1.83	0.025	90%	3
Mediolateral sway: eyes closed
Experimental: 35.09 Controls: 1.18	7.17	0.025	90%	3

Recruitment

Initially, the recruitment of these children will be facilitated through the different stakeholders of the VIS-Flanders project, who have given their consent to participate in the VIS-REHAB study and to assist with the recruitment. Thanks to the VIS-Flanders project, we are able to monitor the number of children diagnosed each year with a vestibular dysfunction. During a period of 3 years of screening, 35 children (13.8% refer rate out of a total of 254 children who were screened) did not pass the vestibular screening.⁴⁸ This suggests that in Flanders, about 12 hearing-impaired children are diagnosed annually with vestibular dysfunction at around 6 months of age. Among them, approximately seven present unilateral vestibular dysfunction, while five demonstrate bilateral vestibular dysfunction. Given the targeted age category in the current VIS-REHAB project (3–17 years), this suggests a total of 180 children in Flanders with vestibular impairment, comprising 105 with unilateral and 75 with bilateral vestibular dysfunction. This number is probably an underestimation as it was derived from calculations involving hearing-impaired children diagnosed at 6 months. It does not account for normal hearing children at risk for a vestibular dysfunction (eg, congenital cytomegalovirus infection) or (hearing-impaired) children with a late-onset vestibular disorder. The incidence of vestibular dysfunction in children with congenital cytomegalovirus is 17%, even surpassing the 14% observed in hearing-impaired children. Additionally, considering that six children initially had inconclusive results at 6 months but were later diagnosed with a vestibular deficit, an additional 30 children are eligible to participate. In total, a sample size of 210 children is available, making the envisioned sample size feasible. Furthermore, because of increased awareness thanks to the VIS-Flanders project, children with vestibular dysfunction but without hearing loss are now being directed to ENT departments, rehabilitation centres and physical therapists. Additionally, recruitment can also be supported via ENT departments, rehabilitation centres, and physical therapists outside the VIS-Flanders/REHAB network.

Allocation and blinding

Subjects will be randomly allocated to either trajectory A or B in a 1:1 ratio. Block randomisation with randomly varying block sizes will be employed to secure a balanced distribution of participants in each group. The randomisation process will be managed by an independent colleague (ie, PhD audiologist) who will use a computer-generated sequence. Details about the block sizes will be stored in a password-protected electronic database accessible only for personnel independent of the enrolment and assignment processes. Based on the randomisation sequence, the PhD audiologist will place the trajectory (A or B) assigned in a sequentially numbered, opaque, sealed envelope. Another independent colleague (ie, principal investigator) will open these envelopes sequentially as participants are enrolled in the study. Randomisation will be stratified by age range (3–6; 7–10; 11–17 years) and degree of vestibular dysfunction (partial vestibular function vs complete loss of vestibular function). Two categories of vestibular function will be used for stratification. Complete loss of vestibular function is defined as a gain of 0.00 on the vHIT for all horizontal and vertical semicircular canals and an absence of cervical and ocular VEMP responses in both ears. All other patients will be considered having residual vestibular function. A more precise evaluation of the degree of residual vestibular function will be considered in the statistical analysis of the results. Due to the nature of the protocols (VIS-REHAB and CTRL), participants cannot be blinded to their assigned groups, as they will be aware of the protocol received at any given time. However, the assessment of primary and secondary outcome measures will be conducted by a separate outcome assessor (ie, the physical therapist from Ghent University Hospital), who will be blinded to patient allocation. It is important to note that the data analysis procedures will not involve blinding since they will be performed by the same individual responsible for conducting the rehabilitation sessions (ie, the coordinating physical therapist).

Data collection and management

The active rehabilitation protocol will be carried out by the coordinating physical therapist, in cooperation with the peripheral physical therapists. Peripheral physical therapists will be contacted to explain all practicalities for the implementation of the intervention protocol. The data collection of the primary and secondary outcome measures will be covered by the physical therapist of Ghent University Hospital. The coordinating physical therapist will be responsible for collecting data related to the subjective outcome measures. Sensory input data will be collected by the audiologists of Ghent University Hospital. Two audiologists will conduct the input evaluation, as certain assessments necessitate the presence of two examinators, particularly in younger children. Demographic and audiological data will be requested from the patients’ ENT specialists, audiologists, physical therapists and parents. Details on therapy progression, results of primary and secondary outcome measures, as well as the sensory input information and demographic information will be collected and stored in a secure, password-protected database. The coordination and entry of data in the database will be supervised by the coordinating physical therapist. In order to streamline data collection and improve data analysis, standardised data sheets will be compiled. The coordinating physical therapist will closely monitor the obtained results and assure quality control. Personal information will be pseudonymised, with only the coordinating physical therapist and principal investigator aware of the coding system. All information obtained in this study will be treated confidentially and stored for a duration of 20 years. If approved by the editorial board of a journal and in accordance with the General Data Protection Regulation, ethical committee guidelines, and Ghent University policies, there may be the possibility of sharing the (coded) data online.

Statistical analyses

All data will be analysed using SPSS software (IBM Corp. Released 2022. IBM SPSS Statistics for Windows, V.29.0. Armonk, New York: IBM Corp). The level of significance will be set at p=0.05. The normality of the data will first be assessed using the Kolmogorov-Smirnov test, QQ plots and histograms. General characteristics of all patients will be described quantitatively. Input evaluations, primary and secondary outcome measures will be compared with normative values available in our paediatric lab or in literature to classify the obtained results as abnormal or normal. The data derived from the subjective outcome measures will be considered exploratory.

Parallel group analysis

To identify significant improvements in primary and secondary outcome measures after 9 weeks compared with the start of each protocol and to determine which protocol is significantly more effective and where improvements are more prominent, analysis of covariance (ANCOVA) will be employed. Within this analysis, the outcome measure is the postintervention measurement. The treatment (VIS-REHAB vs CTRL) and stratification factor (vestibular dysfunction) serve as categorical predictor variables. The covariate in this analysis is the preintervention measurements (baseline measurements of OM.I and OM.II), facilitating the correction for possible baseline differences between the two groups.

Crossover analysis

Given the heterogeneity and relatively small group of children in the study, an additional exploratory crossover analysis will be conducted using a linear mixed model with the child as random factor, taking into account the expected within-child correlation. The fixed factors of the model are treatment, randomisation arm (trajectory) and the interaction between these factors.

Prespecified subgroup analysis

Additional analyses will be performed to investigate the most important factors that may influence the effect and outcome after application of the VIS-REHAB protocol. For each of the following five possible moderators (chronological age, degree of residual vestibular function, baseline motor development, physical therapy history and age at onset of the vestibular dysfunction), the ANCOVA regression will be expanded with the possible moderator and the interaction between this moderator and the treatment group. Further analysis, contingent on the enrolment of specific children in the study, will be conducted within the residual vestibular function group, with additional differentiation based on unilateral or bilateral vestibular dysfunction and canal or otolith dysfunction. With the provided sample size of 60 children, overfitting is avoided.

Ethics and dissemination

For the data collection in Flanders, ethical approval has been obtained by the ethics committee of Ghent University Hospital for the vestibular screening results (EC2018/0435), and for the motor examinations (EC2018/0959). These ethical approvals are limited to children aged under 1 year. For the data collection in Ghent University Hospital, ethical approval has been obtained for the vestibular and motor test results of hearing-impaired children or children with a vestibular problem (EC2015/1441), and for typically developing ‘control’ children (EC2015/1442), and this for children under the age of 6 years. Ethical approval is also obtained for vestibular and motor examinations of vestibular-impaired children aged 6–12 years (EC2019/0668). At the start of the VIS-REHAB study, an amendment will be submitted to the above multicentre and Ghent University Hospital applications, including the following additions: a 4-year test extension; an age extension to 17 years; and the collection of all primary and secondary outcome measures, as well as all sensory input and demographic data. Following both written and verbal presentation of the study details, the parents of all participants are requested to provide written consent in alignment with the Declaration of Helsinki. All research findings will be disseminated in peer-reviewed journals and presented at vestibular, physiotherapeutic as well as multidisciplinary international conferences and meetings.

Patient and public involvement

The research question emerged from issues expressed by vestibular-impaired children, their parents and treating physical therapists (cf. community outreach). Input on the design and outcome measures was sought from motivated parents and physical therapists in Flanders. Physical therapists in Flanders will be involved in the execution of the VIS-REHAB protocol. Each participant and their parents will receive an individualised report detailing the results from the various assessment points. The comprehensive study results will be submitted to the communication departments of both Ghent University and Ghent University Hospital for a press release of the research highlights to the general public. Given the multidisciplinary nature of this research, results will not only be disseminated in specialised journals but also in broader, multidisciplinary journals, as well as in audiological and physiotherapeutic journals to ensure a wider audience reach.

Discussion

The VIS-REHAB project is a logical sequel to the VIS-Flanders project,⁴⁶ which successfully introduced vestibular screening in Flanders and ensured prompt referrals to physical therapists. It is crucial that individuals identified through the screening process, who have reached the lower age limit at which VRT becomes feasible, gain access to an effective treatment. Otherwise, the implementation of a screening programme becomes ineffective and loses its purpose. Leveraging the extensive multidisciplinary network established through VIS-Flanders, there is an opportunity to pioneer once more in the therapeutic part (VIS-REHAB, VRT), in addition to the diagnostic realm (VIS-Flanders, the vestibular screening). Currently, there is a lack of rehabilitation guidelines for VRT in the paediatric domain, despite established clinical practice guidelines for adults by Hall et al.²⁴ A survey (cf. community outreach) performed among the paediatric physical therapists involved in the VIS-Flanders project indicates that they lack insight into the duration, frequency and even the essential components of this VRT, and would greatly benefit from clear-cut rehabilitation guidelines. Furthermore, as apparent in literature and clinical practice, the current focus of rehabilitation in children with vestibular dysfunction is mainly on the improvement of PS, whereas gaze stabilisation exercises generally have no part in the therapy programme. Based on thorough and critical literature search, as well as on clinical therapeutic expertise acquired in the past 4 years of the VIS-Flanders project, a consensus VRT programme including both postural control and gaze stabilisation exercises, was set forth as the VIS-REHAB protocol. The VIS-REHAB protocol presents a consensus derived from current knowledge, and the objective is to evaluate its effectiveness in order to establish a foundation for broader application. The ultimate aim is to formulate precise guidelines for physical therapists based on insights from this study combined with findings from previous research.

Addressed shortcomings from literature

The proposed VIS-REHAB protocol aims to address the shortcomings previously identified in paediatric VRT research.^{3 33 34} To begin with, recruitment and inclusion of subjects is based on confirmed vestibular dysfunction. This involves conducting a comprehensive vestibular assessment before enrolling patients, moving beyond the sole criterion of hearing impairment used in previous paediatric VRT studies. The research group has long-time expertise in paediatric vestibular assessment, honed through involvement in the VIS-Flanders project, which is an important prerequisite for reliable realisation of this study. Moreover, the protocol broadens the scope by including children with several degrees of vestibular dysfunction. This allows for an examination of the effect and outcome after VRT in relation to the extent of residual vestibular function. Additionally, the protocol encompasses the inclusion of both children with and without concomitant hearing loss and/or CIs. Input evaluation will collect information about the participants’ hearing function. Next, stratification will be implemented based on the following age categories: 3–6, 7–10, and 11–17 years. In this matter, the protocol ensures the examination of the influence of the child’s age on the maturation of the balance system, acknowledging its potential impact on the effectiveness of VRT. Since the most critical period for motor development typically lays within the first decade of life, with balance usually fully established at 7–10 years old, it is crucial to categorise groups when including children under the age of 7.^{3 56} Furthermore, the protocol is unique in its exercises focusing on GS, along with outcome measures designed for its comprehensive assessment. Additionally, the VIS-REHAB protocol includes a thorough description of the content, doses, duration and progression of the vestibular rehabilitation exercises to enhance study replicability. Finally, input evaluation entails a structured parent survey investigating patient characteristics and medical history. In this manner, detailed information is gathered on the aetiology of (audio)vestibular dysfunction, history of physical therapy, development factors, physical activity, sports, medication usage and other important variables that could influence the outcome of VRT.

Critical considerations

Intervention protocols

VIS-REHAB protocol

Besides the shortcomings from literature addressed in the VIS-REHAB protocol, several other critical considerations on societal, clinical and scientific levels have been raised regarding the development of the study protocol. The first concern is to guarantee that the study does not displace the physical therapist’s responsibilities and patients. Hence, the intervention will be carried out by the child’s physical therapist. This organisation is feasible in our study, given that it will be conducted in Flanders, where the physical therapists are already engaged due to their participation in the VIS-Flanders project. The VIS-Flanders project has indeed successfully established an extensive multidisciplinary network. Including physical therapists in the study also aims to facilitate their understanding of the most optimal VRT tools, a topic physical therapists engaged in the VIS-Flanders project are actively seeking to explore (cf. community outreach). On resuming therapy after the child has completed the entire trajectory, the treating physical therapist has the flexibility to already incorporate certain elements from the sessions conducted under the supervision of the coordinating physical therapist. Following the analysis of study results, information sessions will be conducted to update peripheral physical therapists on the most effective VRT protocol and approach. This ensures that every physical therapist in Flanders is equipped with knowledge of the optimal VRT approach, enabling them to appropriately treat these patients in the future. As a result, children in Flanders will not only continue to benefit from the existing vestibular screening (VIS-Flanders), but will also gain access to an optimal VRT setting (VIS-REHAB), both contributing positively the motor, cognitive, socioemotional and educational development of the child.

Another concern is to ensure standardisation and a comparable therapy across different physical therapists and patients. To address this, the therapy sessions will be supervised by the coordinating physical therapist. This is again feasible due to the unique situation established in Flanders. The network of physical therapists in Flanders who are willing to collaborate, along with the compact geographical layout of the region, allows for one person to oversee therapy sessions across Flanders. This approach ensures the systematic evaluation of VRT application by one person throughout the three-year study period, ensuring unique and reliable results.

Another challenge arises when certain patients cannot visit a peripheral physical therapist (twice a week), which may be due to various reasons. In these instances, the coordinating physical therapist can conduct the additional sessions either at the patient’s home or at Ghent University Hospital, depending on the preferences of both the child and the parents. Moreover, in the event of a cancelled session, whether due to illness or unforeseen circumstances, rescheduling will be arranged within one week. This flexible approach is designed to enhance adherence to treatment.

Additionally, several concerns remain regarding the application of gaze stabilisation exercises in children, as included in the VIS-REHAB protocol. There is limited research on the effectiveness of gaze stabilisation training in children, and further research is necessary to determine the optimal dosage and the critical timeframe for administering gaze stabilisation exercises in children.^{13 40} Additionally, the execution of gaze stabilisation in children poses frequent challenges.⁴⁵ The conventional gaze stabilisation exercises are often perceived as boring by most children, potentially impacting their adherence to therapy.¹³ However, Dhondt et al’s book on gaze stabilisation in children⁸¹ outlines several options for increasing appeal to young children. Moreover, as stated before, a certain level of cognitive maturity and working attitude is needed to successfully complete the exercises. This is because the child is asked to deliberately deploy certain strategies or cues to obtain gaze as stable as possible. In contrast, postural control training proves more accessible and commonly implemented in (vestibular) rehabilitation for children.⁴⁵

CTRL protocol

Acknowledging ethical considerations, it is not evident to withhold therapy from children for a duration of 9 weeks, as proposed by the control (CTRL) protocol. However, given the crossover study design, each child will also receive the proposed VIS-REHAB protocol. Furthermore, the study aims to optimise VRT for the child, ensuring that participation in the study will ultimately result in the child engaging in a more effective VRT programme. However, precise consideration is crucial when scheduling the 9-week cessation, taking into account distinctions between vacation and school periods, as therapy often tends to take a back seat during holidays anyway. Furthermore, it is essential to take into account the child’s after-school and vacation activities, given that sports and recreational hobbies are not expected to be temporarily halted. Close monitoring and documentation of these factors are crucial, as sports and recreational activities contribute positively to the balance performance of these children.^{34 56}

Influencing factors

The study population of children with vestibular dysfunction is very diverse. Consequently, it is essential to evaluate the appropriateness of implementing a single protocol for such a heterogeneous group of children. In addressing vestibular dysfunction, it is crucial to consider both the type (congenital or acquired) and severity (partial or total, unilateral or bilateral). Additionally, it is important to consider the aetiology of (audio)vestibular dysfunction and possible progression, as observed in children with enlarged vestibular aqueduct.³ It is postulated that if the dysfunction occurred at or shortly after birth, the therapy must focus on improving the effectiveness of vision and somatosensation for balance by means of sensory reweighting. Conversely, if the dysfunction arises after the critical period of balance development, around 6 years of age, a significant component of therapy may involve exercises with focus on adaptation.¹⁴ However, in literature, the question whether the mechanisms of recovery are different and whether the interventions should vary for children with congenital versus acquired vestibular hypofunction remains.^{13 24} Additionally, children lacking residual vestibular function are less likely to derive benefits from exercises emphasising adaptation, given that such exercises necessitate some remaining vestibular capacity.⁸² In such cases, therapy should prioritise sensory substitution, and it is essential to consider that the recovery process tends to be slower.⁸³ Furthermore, it is unclear whether the optimal dosage of VRT is contingent on the type and severity of the child’s vestibular lesion.²⁴

It is important to evaluate the maturation of the different sensory input systems for balance function in children when implementing a VRT protocol. During infancy, postural control primarily relies on the visual system. As children mature, they start to use somatosensory and vestibular information more effectively.²⁰ Stabilisation of eyes and the upright position of head and body is dependent on the maturation of the vestibular system.⁴¹ In line with the critical developmental stages of the auditory cortex, defining the optimal timing for introducing CIs in hearing-impaired children,⁴³ understanding the critical developmental periods of the sensory input systems would provide valuable insights into the optimal timing for initiating VRT in vestibular-impaired children.⁴⁰ This knowledge is particularly crucial in determining the most effective timing for implementing gaze stabilisation and postural control exercises. Through the VIS-REHAB study, we aim to explore the impact of VRT on the youngest age groups, enabling us to derive specific insights into the maturation of sensory input systems. Additionally, a comprehensive literature review of the development of the various sensory input systems would be highly beneficial.

Community outreach: barriers and facilitators

Responses from surveys administered to physical therapists and parents from children with vestibular dysfunction engaged in the VIS-Flanders network have identified several barriers and facilitators influencing the research question and implementation of the VIS-REHAB project. The majority of surveyed physiotherapists reported a lack in confidence, knowledge and skills when it comes to initiating therapy for children with vestibular dysfunction in all age categories of the current study (3–17 years). As a response to these findings, the surveyed physical therapists are eager to participate in the follow-up project of the VIS-Flanders project, specifically the VIS-REHAB study, focusing on VRT for children in these age groups. Physical therapists are keen to deepen their understanding of VRT, with particular focus on the organisation and progression of therapy sessions, as well as exploring potential variations and gaining a more comprehensive understanding of the concrete implementation of VRT. They are seeking assistance in this endeavour and are interested in participating in collaborative therapy sessions with an experienced vestibular therapist, a practice that is facilitated within our study. Furthermore, a significant number of parents indicated their willingness to participate with their child in research focusing on VRT. However, only a small percentage of parents expressed the need for more tailored and specific therapeutic interventions, suggesting that this perspective might be influenced by a lack of awareness of therapy approaches that could potentially be more effective. This underscores the overall insufficient awareness regarding the importance of VRT in children.

Future perspectives

Numerous unresolved questions persist in paediatric VRT, and the VIS-REHAB protocol was specifically designed to address and resolve them. However, current research also demonstrates a significant emphasis on the implementation of home-based exercise programmes and alternative, more engaging VRT modalities.

Home exercise program

The recent clinical practice guideline designed for adult vestibular rehabilitation²⁴ recommends to combine weekly clinical visits with a home exercise programme including gaze stabilisation and balance exercises. Home exercise programmes are designed to augment therapeutic dosage.²⁶ According to literature, the implementation of home exercises is probably the primary driver of recovery, attributed to the high volume in comparison to the rehabilitation sessions with the physical therapist.⁸⁴ However, introducing a home exercise programme poses several challenges that must be addressed prior to its implementation in paediatric VRT. First, low adherence is a challenge when providing home exercise programmes in VRT, which can negatively impact efficacy.³⁰ Another obstacle arises from the suboptimal execution of home exercises,²⁶ considering that children rely on parents to implement the home exercise programme. In the recent clinical practice guideline of Hall et al,²⁴ dosage recommendations are put forward. However, many individuals, and especially children, cannot tolerate practicing vestibular rehabilitation exercises for 20–40 minutes a day, as recommended by this clinical practice guideline. Therefore, it is crucial to adapt these dose recommendations, taking into account the limited attention span of children and the capacity of both children and parents to carry out a daily home exercise programme. In addition to the parents’ role in carrying out the home-based exercise programme, it is also crucial to emphasise that a home exercise programme is meant to complement rather than replace the rehabilitation sessions with the physical therapist. According to literature, a home-based exercise programme in VRT must be simple, low-cost, customised and preferably supervised in order to obtain high adherence and success.^{24 25 30 85 86} In addition, it is crucial for the patient to comprehend the exercise procedures and adhere to the therapist’s instructions when practicing exercises at home.⁸⁵ Further research is required to explore the impact of telehealth supervision on adherence and motivation of the patient, including the identification of critical time points for supervision.²⁴ Additionally, it would be interesting to evaluate the potential and feasibility of having trained parents administer the home-based rehabilitation programme.

Alternative VRT modalities

Vestibular rehabilitation exercises are often experienced as being repetitive and monotonous,⁸⁷ particularly for children.⁸¹ Even with modification of the VRT exercises to suit the child’s interests, traditional therapeutic exercises, such as those incorporated in the VIS-REHAB protocol, may lack appeal for children. This can potentially result in decreased adherence and waning interest in treatment.³³ Adherence to therapy is particularly crucial when engaging in a home-based exercise programme.³⁰ To enhance a child’s engagement in therapy, alternative forms of rehabilitation have been proposed³³ and recent research has explored these alternative VRT modalities in the paediatric field.^88–100

Sports and recreational activities

Engaging in sports and recreational activities provides the first enjoyable alternative for conventional VRT.³⁴ A systematic review by Melo et al highlights that these activities serve as motivational and playful modalities, contributing to improvement in balance and gait in hearing-impaired children and adolescents.³⁴ An even more recent systematic review conducted by Zhou and Qi⁵⁶ similarly underscores the positive impact of exercise interventions, mainly encompassing recreational and sports activities, on balance. Therefore, it is recommended for hearing-impaired children to engage in sports activities.⁹⁷ Furthermore, to sustain the outcomes achieved through VRT, it is essential to lead an active lifestyle and engage in regular sports activities.¹⁰¹

The studies examining the effect of sports and recreational activities on balance in hearing-impaired children encompass a diverse range of exercise interventions. These interventions compromise swimming,⁸⁸ rhythmic gymnastics activities,⁸⁹ table tennis training,⁹⁰ physical exercises with music and vibration,⁹¹ tai chi,⁹² capoeira,⁹³ dance activities,⁹⁴ athletic exercises⁹⁵ and pilates.⁹⁶ Various of these sports modalities have the potential to affect balance through shifts in the centre of gravity or through head and eye movements, also referred to as key exercises in VRT.³⁴ Conversely, engaging in these activities may also result in side effects attributed to the rapid movements of the head and eyes, adversely affecting motor performance.³⁴

In conclusion, sports and recreational activities can serve as a valuable alternative or complement to standard VRT, or function as maintenance therapy for hearing-impaired children and adolescents. It is important to consider that the child’s ability to participate in sports and recreational activities is dependent on various factors such as age, the severity and type of vestibular dysfunction, and the child’s interests and motivation.

Virtual reality

Another interesting option to enhance appeal, enjoyment and motivation of VRT for children is to delve into the realms of virtual reality, augmented reality and virtual reality-based games. Drawing from adult research, these interventions have demonstrated effectiveness comparable to traditional VRT, with the added benefit of enhanced adherence due to increased motivation.⁸⁶ Moreover, the clinical practice guideline states that combining these exercises with head movements yields an extra benefit on balance and symptoms beyond increased motivation.²⁴ Research in the paediatric field reports positive effects of virtual reality-based games, using a Nintendo Wii console with Wii Balance board on postural control, balance and gait of hearing-impaired children and adolescents.³³

Technology-based interventions would offer advantages over standard VRT exercises. These include the provision of feedback, the opportunity to practice in a stimulating and enriched environment, the capacity for independent use, potentially reducing therapy costs and facilitating increased exercise dosage.^{87 102–104} Additionally, these interventions emerge as a valuable option to enhance adherence in home-based exercise programmes, underscoring its significance for the success of these interventions, as mentioned earlier.¹⁰³ Conversely, these exercise modalities may elicit adverse effects such as dizziness and cybersickness.¹⁰⁴ This occurs because of an amplified sensory mismatch, compounded by the existing disparity in sensory input systems found in children with vestibular dysfunction.¹⁴ Therefore, it is crucial to closely monitor these symptoms when administering virtual reality-based interventions.³³

A head-mounted virtual reality device (HMD) is often used in virtual reality-based VRT involving adults. It is recommended to implement the HMD in a home-based gaming programme coupled with conventional VRT exercises.^{105 106} The use of such a device allows for tracking both head and eye movements, making it a suitable tool for gaze stabilisation therapy.^{103 107} However, employing a HMD in gaze stabilisation therapy for children, particularly engaging in a game where the child must focus on a target and complete various tasks with head movements, can lead to displacement of the device, potentially inducing retinal slip. In a recent study by Ortega Solís et al,⁴⁵ gaze stabilisation exercises were performed similarly within a game setting. However, instead of employing a HMD, a laptop computer was used, and an eye- and head-mounted sensor detected the head movements of the children. This presents another challenge, as the computer screen fails to offer a full field stimulus during game play. This limitation allows the child to visually fixate elsewhere, potentially suppressing the VOR.

Virtual reality-based VRT shows promise for enhancing postural control and gaze stabilisation in children. While the temptation to immediately apply these interventions exists, caution is warranted, especially in children with vestibular dysfunction. As previously noted, virtual reality introduces a sensory mismatch, posing a challenge for children who already struggle with multisensory integration of input systems. The use of virtual reality, though a potential tool in paediatric VRT, requires careful consideration. Insufficient information, including uncertainties about potential harms, benefits and identifying suitable devices, modalities and anticipated costs for seamless integration into therapy, currently hinders the application of virtual reality in this particular patient population. Therefore, initiating its use at this point is premature.

In summary, before integrating home-based exercise programmes and alternate, more advanced VRT modalities into paediatric VRT, it is crucial to conduct thorough research as intended with the VIS-REHAB study to address the aforementioned concerns related to conventional VRT. These concerns encompass understanding the effect of VRT, the optimal age for its application, the appropriate dosage based on type and severity of a child’s vestibular lesion and the maturation of the diverse input systems and their influence on VRT administration. Integrating insights obtained from the VIS-REHAB study with findings from prior research is essential for effectively addressing these concerns and formulating comprehensive rehabilitation guidelines for paediatric VRT. This undertaking aims to bridge the existing gap in literature and provides solutions to questions that arise in clinical practice. Future initiatives can focus on integrating a child-friendly home-based exercise programme and introducing more engaging elements, such as sports, recreational activities, virtual reality and games, with the aim of enhancing adherence and feasibility of (home-based) VRT.

The author would like to thank Mieke De Bock from the Centre for Ambulatory Rehabilitation Sint-Lievenspoort Ghent and Alexandra De Kegel from Multifunctional Centre Sint-Lievenspoort Ghent for their valuable input and advice in developing the test protocol.

Ethics statements

Patient consent for publication

Not applicable.

Footnote

Contributors Each author made significant contributions to the article. MF and LM developed the test protocol, which was then subjected to critical review and refinement by ID, CD, RVH, FA, LVdB, HVH and EDL. MF drafted the initial article and enhanced subsequent versions. ID, FA and LM conducted thorough reviews and revisions of the article. All authors provided critical comments, gave their approval for the final submitted article and take responsibility for all aspects of the study. LM acted as guarantor.

Funding The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors.

Competing interests None declared.

Patient and public involvement Patients and/or the public were involved in the design, or conduct, or reporting or dissemination plans of this research. Refer to the Methods section for further details.

Provenance and peer review Not commissioned; externally peer reviewed.

Supplemental material This content has been supplied by the author(s). It has not been vetted by BMJ Publishing Group Limited (BMJ) and may not have been peer-reviewed. Any opinions or recommendations discussed are solely those of the author(s) and are not endorsed by BMJ. BMJ disclaims all liability and responsibility arising from any reliance placed on the content. Where the content includes any translated material, BMJ does not warrant the accuracy and reliability of the translations (including but not limited to local regulations, clinical guidelines, terminology, drug names and drug dosages), and is not responsible for any error and/or omissions arising from translation and adaptation or otherwise.

References

¹ Bigelow RT, Agrawal Y. Vestibular involvement in cognition: Visuospatial ability, attention, executive function, and memory. J Vestib Res 2015; 25: 73–89. doi:10.3233/VES-150544

² O’Reilly R, Grindle C, Zwicky EF, et al. Development of the vestibular system and balance function: differential diagnosis in the pediatric population. Otolaryngol Clin North Am 2011; 44: 251–71. doi:10.1016/j.otc.2011.01.001

³ Melo RS, Lemos A, Paiva GS, et al. Vestibular rehabilitation exercises programs to improve the postural control, balance and gait of children with sensorineural hearing loss: A systematic review. Int J Pediatr Otorhinolaryngol 2019; 127: 109650. doi:10.1016/j.ijporl.2019.109650

⁴ Smith PF. The vestibular system and cognition. Curr Opin Neurol 2017; 30: 84–9. doi:10.1097/WCO.0000000000000403

⁵ Hitier M, Besnard S, Smith PF. Vestibular pathways involved in cognition. Front Integr Neurosci 2014; 8: 59. doi:10.3389/fnint.2014.00059

⁶ Besnard S, Lopez C, Brandt T, et al. Editorial: The Vestibular System in Cognitive and Memory Processes in Mammalians. Front Integr Neurosci 2015; 9: 55. doi:10.3389/fnint.2015.00055

⁷ Singh A, Raynor EM, Lee JW, et al. Vestibular Dysfunction and Gross Motor Milestone Acquisition in Children With Hearing Loss: A Systematic Review. Otolaryngol--head neck surg 2021; 165: 493–506. doi:10.1177/0194599820983726

⁸ Braswell J, Rine RM. Evidence that vestibular hypofunction affects reading acuity in children. Int J Pediatr Otorhinolaryngol 2006; 70: 1957–65. doi:10.1016/j.ijporl.2006.07.013

⁹ De Kegel A, Maes L, Baetens T, et al. The influence of a vestibular dysfunction on the motor development of hearing-impaired children. Laryngosc 2012; 122: 2837–43. doi:10.1002/lary.23529

¹⁰ Popp P, Wulff M, Finke K, et al. Cognitive deficits in patients with a chronic vestibular failure. J Neurol 2017; 264: 554–63. doi:10.1007/s00415-016-8386-7

¹¹ Dhondt C, Maes L, Rombaut L, et al. Vestibular Function in Children With a Congenital Cytomegalovirus Infection: 3 Years of Follow-Up. Ear Hear 2021; 42: 76–86. doi:10.1097/AUD.0000000000000904

¹² Whitney SL, Alghwiri AA, Alghadir A. An overview of vestibular rehabilitation. Handb Clin Neurol 2016; 137: 187–205. doi:10.1016/B978-0-444-63437-5.00013-3

¹³ Christy JB. Considerations for Testing and Treating Children with Central Vestibular Impairments. Semin Hear 2018; 39: 321–33. doi:10.1055/s-0038-1666821

¹⁴ Rine RM, Wiener-Vacher S. Evaluation and treatment of vestibular dysfunction in children. NeuroRehabilitation 2013; 32: 507–18. doi:10.3233/NRE-130873

¹⁵ Dieterich M, Brandt T. The bilateral central vestibular system: its pathways, functions, and disorders. Ann N Y Acad Sci 2015; 1343: 10–26. doi:10.1111/nyas.12585

¹⁶ Braswell J, Rine RM. Preliminary evidence of improved gaze stability following exercise in two children with vestibular hypofunction. Int J Pediatr Otorhinolaryngol 2006; 70: 1967–73. doi:10.1016/j.ijporl.2006.06.010

¹⁷ Janky KL, Givens D. Vestibular, Visual Acuity, and Balance Outcomes in Children With Cochlear Implants: A Preliminary Report. E H 2015; 36: e364–72. doi:10.1097/AUD.0000000000000194

¹⁸ Janky KL, Thomas M, Al-Salim S, et al. Does vestibular loss result in cognitive deficits in children with cochlear implants? J Vestib Res 2022; 32: 245–60. doi:10.3233/VES-201556

¹⁹ Franco ES, Panhoca I. Vestibular function in children underperforming at school. Braz J Otorhinolaryngol 2008; 74: 815–25. doi:10.1016/S1808-8694(15)30141-5

²⁰ Ebrahimi AA, Jamshidi AA, Movallali G, et al. The Effect of Vestibular Rehabilitation Therapy Program on Sensory Organization of Deaf Children With Bilateral Vestibular Dysfunction. Acta Med Iran 2017; 55: 683–9.

²¹ Cawthorne T, Cawthorne D. The psychological basis for head exercises. J Chartered Soc Physiother 1944; 30: 106–7.

²² Cooksey FS. Rehabilitation in Vestibular Injuries. Proc R Soc Med 1946; 39: 273–8.

²³ McDonnell MN, Hillier SL. Vestibular rehabilitation for unilateral peripheral vestibular dysfunction. Cochrane Database Syst Rev 2015; 1: CD005397. doi:10.1002/14651858.CD005397.pub4

²⁴ Hall CD, Herdman SJ, Whitney SL, et al. Vestibular Rehabilitation for Peripheral Vestibular Hypofunction: An Updated Clinical Practice Guideline From the Academy of Neurologic Physical Therapy of the American Physical Therapy Association. J Neurol Phys Ther 2022; 46: 118–77. doi:10.1097/NPT.0000000000000382

²⁵ Maldonado-Díaz M, Vargas P, Vasquez R, et al. Teleneurorehabilitation program (virtual reality) for patients with balance disorders: descriptive study. BMC Sports Sci Med Rehabil 2021; 13: 83. doi:10.1186/s13102-021-00314-z

²⁶ Bhatti PT, Herdman SJ, Roy SD, et al. A Prototype Head-Motion Monitoring System for In-Home Vestibular Rehabilitation Therapy. J Bioeng Biomed Sci 2011; Suppl 1: 009. doi:10.4172/2155-9538.S1-009

²⁷ Aldawsary N, Almarwani M. The combined effect of gaze stability and balance exercises using telerehabilitation in individuals with vestibular disorders during the COVID-19 pandemic: A pilot study. PLoS One 2023; 18: e0282189. doi:10.1371/journal.pone.0282189

²⁸ van Vugt VA, van der Wouden JC, Essery R, et al. Internet based vestibular rehabilitation with and without physiotherapy support for adults aged 50 and older with a chronic vestibular syndrome in general practice: three armed randomised controlled trial. BMJ 2019; 367: l5922. doi:10.1136/bmj.l5922

²⁹ Geraghty AWA, Essery R, Kirby S, et al. Internet-Based Vestibular Rehabilitation for Older Adults With Chronic Dizziness: A Randomized Controlled Trial in Primary Care. Ann Fam Med 2017; 15: 209–16. doi:10.1370/afm.2070

³⁰ Tanaka R, Fushiki H, Tsunoda R, et al. Effect of Vestibular Rehabilitation Program Using a Booklet in Patients with Chronic Peripheral Vestibular Hypofunction: A Randomized Controlled Trial. Prog Rehabil Med 2023; 8. doi:10.2490/prm.20230002

³¹ Szturm T, Reimer KM, Hochman J. Home-Based Computer Gaming in Vestibular Rehabilitation of Gaze and Balance Impairment. Games Health J 2015; 4: 211–20. doi:10.1089/g4h.2014.0093

³² Kellerer S, Amberger T, Schlick C, et al. Specific and individualized instructions improve the efficacy of booklet-based vestibular rehabilitation at home - a randomized controlled trial (RCT). J Vestib Res 2023; 33: 349–61. doi:10.3233/VES-220122

³³ Melo RS, Lemos A, Delgado A, et al. Use of Virtual Reality-Based Games to Improve Balance and Gait of Children and Adolescents with Sensorineural Hearing Loss: A Systematic Review and Meta-Analysis. Sensors (Basel) 2023; 23: 6601. doi:10.3390/s23146601

³⁴ Melo RS, Tavares-Netto AR, Delgado A, et al. Does the practice of sports or recreational activities improve the balance and gait of children and adolescents with sensorineural hearing loss? A systematic review. Gait & Posture 2020; 77: 144–55. doi:10.1016/j.gaitpost.2020.02.001

³⁵ Effgen SK. Effect of an exercise program on the static balance of deaf children. Phys Ther 1981; 61: 873–7. doi:10.1093/ptj/61.6.873

³⁶ Rajendran V, Roy FG, Jeevanantham D. A preliminary randomized controlled study on the effectiveness of vestibular-specific neuromuscular training in children with hearing impairment. Clin Rehabil 2013; 27: 459–67. doi:10.1177/0269215512462909

³⁷ Soori Z, Heyrani A, Rafie F. Exercise effects on motor skills in hearing-impaired children. Sport Sci Health 2019; 15: 635–9. doi:10.1007/s11332-019-00564-y

³⁸ Demirel N. The Impact of Therapeutic Recreational Gymnastic Exercise on Basic Motor Skills of Hearing-Impaired Children Aged Between 6 and 9 Years. JETS 2018; 6: 147. doi:10.11114/jets.v6i3.3048

³⁹ Martens S, Dhooge I, Dhondt C, et al. Het VIS-Flanders-project, een uniek vestibulair screeningsproject voor kinderen met congenitaal gehoorverlies. Ned Tijdschr Keel-Neus-Oorheelk 2020; 26: 99–105.

⁴⁰ Rine RM, Braswell J, Fisher D, et al. Improvement of motor development and postural control following intervention in children with sensorineural hearing loss and vestibular impairment. Int J Pediatr Otorhinolaryngol 2004; 68: 1141–8. doi:10.1016/j.ijporl.2004.04.007

⁴¹ Rajendran V, Roy FG. An overview of motor skill performance and balance in hearing impaired children. Ital J Pediatr 2011; 37: 33. doi:10.1186/1824-7288-37-33

⁴² Shumway-Cook A, Woollacott MH. The growth of stability: postural control from a development perspective. J Mot Behav 1985; 17: 131–47. doi:10.1080/00222895.1985.10735341

⁴³ Persic D, Thomas ME, Pelekanos V, et al. Regulation of auditory plasticity during critical periods and following hearing loss. Hear Res 2020; 397: 107976. doi:10.1016/j.heares.2020.107976

⁴⁴ Cole KR, Goodman K, Volland L. Reporting of exercise dose and dosage and outcome measures for gaze stabilisation in the literature: a scoping review. BMJ Open 2022; 12: e049560. doi:10.1136/bmjopen-2021-049560

⁴⁵ Ortega Solís J, Reynard P, Spruyt K, et al. Developing a serious game for gaze stability rehabilitation in children with vestibular hypofunction. J Neuroeng Rehabil 2023; 20: 128. doi:10.1186/s12984-023-01249-x

⁴⁶ Martens S, Dhooge I, Dhondt C, et al. Vestibular Infant Screening - Flanders: The implementation of a standard vestibular screening protocol for hearing-impaired children in Flanders. Int J Pediatr Otorhinolaryngol 2019; 120: 196–201. doi:10.1016/j.ijporl.2019.02.033

⁴⁷ Martens S, Dhooge I, Dhondt C, et al. Vestibular Infant Screening (VIS)–Flanders: results after 1.5 years of vestibular screening in hearing-impaired children. Sci Rep 2020; 10: 21011. doi:10.1038/s41598-020-78049-z

⁴⁸ Martens S, Dhooge I, Dhondt C, et al. Three Years of Vestibular Infant Screening in Infants With Sensorineural Hearing Loss. Pediatrics 2022; 150: e2021055340. doi:10.1542/peds.2021-055340

⁴⁹ Martens S, Dhooge I, Dhondt C, et al. Pediatric Vestibular Assessment: Clinical Framework. Ear Hear 2023; 44: 423–36. doi:10.1097/AUD.0000000000001303

⁵⁰ Martens S, Maes L, Dhondt C, et al. Vestibular Infant Screening-Flanders: What is the Most Appropriate Vestibular Screening Tool in Hearing-Impaired Children? Ear Hear 2023; 44: 385–98. doi:10.1097/AUD.0000000000001290

⁵¹ Henderson SE, Sugden DA, Barnett AL. Movement Assessment Battery for Children (Examiner’s Manual). 2nd edn. London: Pearson Assessment, 2007.

⁵² Meldrum D, Jahn K. Gaze stabilisation exercises in vestibular rehabilitation: review of the evidence and recent clinical advances. J Neurol 2019; 266: 11–8. doi:10.1007/s00415-019-09459-x

⁵³ Herdman SJ, Hall CD, Maloney B, et al. Variables associated with outcome in patients with bilateral vestibular hypofunction: Preliminary study. J Vestib Res 2015; 25: 185–94. doi:10.3233/VES-150556

⁵⁴ Dhondt C, Maes L, Vanaudenaerde S, et al. Changes in Vestibular Function Following Pediatric Cochlear Implantation: a Prospective Study. Ear Hear 2022; 43: 620–30. doi:10.1097/AUD.0000000000001125

⁵⁵ Fernandes R, Hariprasad S, Kumar VK. Physical therapy management for balance deficits in children with hearing impairments: A systematic review. J Paediatr Child Health 2015; 51: 753–8. doi:10.1111/jpc.12867

⁵⁶ Zhou Y, Qi J. Effectiveness of Interventions on Improving Balance in Children and Adolescents With Hearing Impairment: A Systematic Review. Front Physiol 2022; 13: 13. doi:10.3389/fphys.2022.876974

⁵⁷ Dhondt C, Dhooge I, Maes L. Vestibular assessment in the pediatric population. Laryngoscope 2019; 129: 490–3. doi:10.1002/lary.27255

⁵⁸ Rine RM, Braswell J. A clinical test of dynamic visual acuity for children. Int J Pediatr Otorhinolaryngol 2003; 67: 1195–201. doi:10.1016/j.ijporl.2003.07.004

⁵⁹ Rajendran V, Roy FG, Jeevanantham D. Reliability of pediatric reach test in children with hearing impairment. Int J Pediatr Otorhinolaryngol 2012; 76: 901–5. doi:10.1016/j.ijporl.2012.02.068

⁶⁰ Condon C, Cremin K. Static balance norms in children. Physiother Res Int 2014; 19: 1–7. doi:10.1002/pri.1549

⁶¹ Friello P, Silver N, Sangi-Haghpeykar H, et al. Screening for balance in children and adults in a community science education setting: Normative data, influence of age, sex, and body mass index, and feasibility. PLoS One 2022; 17: e0268030. doi:10.1371/journal.pone.0268030

⁶² Cohen HS. A review on screening tests for vestibular disorders. J Neurophysiol 2019; 122: 81–92. doi:10.1152/jn.00819.2018

⁶³ Cohen HS, Mulavara AP, Stitz J, et al. Screening for Vestibular Disorders Using the Modified Clinical Test of Sensory Interaction and Balance and Tandem Walking With Eyes Closed. Otol Neurotol 2019; 40: 658–65. doi:10.1097/MAO.0000000000002173

⁶⁴ Van Hecke R, Deconinck FJA, Wiersema JR, et al. Balanced Growth project: a protocol of a single-centre observational study on the involvement of the vestibular system in a child’s motor and cognitive development. BMJ Open 2021; 11: e049165. doi:10.1136/bmjopen-2021-049165

⁶⁵ Atwater SW, Crowe TK, Deitz JC, et al. Interrater and test-retest reliability of two pediatric balance tests. Phys Ther 1990; 70: 79–87. doi:10.1093/ptj/70.2.79

⁶⁶ Rine RM, Lindeblad S, Donovan P, et al. Balance and Motor Skills in Young Children With Sensorineural Hearing Impairment. Pediatr Phys Ther 1996; 8: 55. doi:10.1097/00001577-199600820-00002

⁶⁷ Donahoe B, Turner D, Worrell T. The Use of Functional Reach as a Measurement of Balance in Boys and Girls Without Disabilities Ages 5 to 15 Years. Pediatr Phys Ther 1994; 6: 189. doi:10.1097/00001577-199400640-00004

⁶⁸ Nicolini-Panisson RDA, Donadio MVF. Timed “Up & Go” test in children and adolescents. Rev Paul Pediatr 2013; 31: 377–83. doi:10.1590/S0103-05822013000300016

⁶⁹ Williams EN, Carroll SG, Reddihough DS, et al. Investigation of the timed “up & go” test in children. Dev Med Child Neurol 2005; 47: 518–24. doi:10.1017/s0012162205001027

⁷⁰ Nicolini-Panisson RD, Donadio MVF. Timed “Up & Go” test in children and adolescents. Rev Paul Pediatr 2013; 31: 377–83. doi:10.1590/S0103-05822013000300016

⁷¹ Verbecque E, Schepens K, Theré J, et al. The Timed Up and Go Test in Children: Does Protocol Choice Matter? A Systematic Review. Pediatr Phys Ther 2019; 31: 22–31. doi:10.1097/PEP.0000000000000558

⁷² Lubetzky-Vilnai A, Jirikowic TL, McCoy SW. Investigation of the Dynamic Gait Index in children: a pilot study. Pediatr Phys Ther 2011; 23: 268–73. doi:10.1097/PEP.0b013e318227cd82

⁷³ Anderson DK, Cech D. Utilization of the Pediatric Modified Dynamic Gait Index: Issues Related to Child Development. Phys Occup Ther Pediatr 2019; 39: 669–78. doi:10.1080/01942638.2019.1606131

⁷⁴ Caldani S, Baghdadi M, Moscoso A, et al. Vestibular Functioning in Children with Neurodevelopmental Disorders Using the Functional Head Impulse Test. Brain Sci 2020; 10: 887. doi:10.3390/brainsci10110887

⁷⁵ Corallo G, Versino M, Mandalà M, et al. The functional head impulse test: preliminary data. J Neurol 2018; 265: 35–9. doi:10.1007/s00415-018-8910-z

⁷⁶ Thompson-Harvey A, Dutcher CE, Monroe HA, et al. Detection of VOR dysfunction during the gaze stabilization test: Does target size matter? J Vestib Res 2021; 31: 495–504. doi:10.3233/VES-201602

⁷⁷ Voelker CCJ, Lucisano A, Kallogjeri D, et al. Comparison of the gaze stabilization test and the dynamic visual acuity test in unilateral vestibular loss patients and controls. Otol Neurotol 2015; 36: 746–53. doi:10.1097/MAO.0000000000000689

⁷⁸ Griffiths A, Toovey R, Morgan PE, et al. Psychometric properties of gross motor assessment tools for children: a systematic review. BMJ Open 2018; 8: e021734. doi:10.1136/bmjopen-2018-021734

⁷⁹ Deitz JC, Kartin D, Kopp K. Review of the Bruininks-Oseretsky Test of Motor Proficiency, Second Edition (BOT-2). Phys Occup Ther Pediatr 2007; 27: 87–102.

⁸⁰ Varni JW, Burwinkle TM, Seid M, et al. The PedsQL 4.0 as a pediatric population health measure: feasibility, reliability, and validity. Ambul Pediatr 2003; 3: 329–41. doi:10.1367/1539-4409(2003)003<0329:tpaapp>2.0.co;2

⁸¹ Dhondt C, Van Hecke R, Dhooge I, et al. Vestibulaire revalidatie: blikstabilisatietraining voor kinderen. Brave New Books; 2020.

⁸² Whitney SL, Furman JM. Vestibular Rehabilitation. 2014: 754–89.

⁸³ Whitney SL, Stephen C, Furman JM. Vestibular rehabilitation. In: Vestibular Disorders. 2010: 46–52.

⁸⁴ Whitney SL, Sparto PJ. Principles of vestibular physical therapy rehabilitation. NeuroRehabilitation 2011; 29: 157–66. doi:10.3233/NRE-2011-0690

⁸⁵ Hovareshti P, Roeder S, Holt LS, et al. VestAid: A Tablet-Based Technology for Objective Exercise Monitoring in Vestibular Rehabilitation. Sensors (Basel) 2021; 21: 24: 8388. doi:10.3390/s21248388

⁸⁶ Nehrujee A, Vasanthan L, Lepcha A, et al. A Smartphone-based gaming system for vestibular rehabilitation: A usability study. J Vestib Res 2019; 29: 147–60. doi:10.3233/VES-190660

⁸⁷ Rosiak O, Krajewski K, Woszczak M, et al. Evaluation of the effectiveness of a Virtual Reality-based exercise program for Unilateral Peripheral Vestibular Deficit. J Vestib Res 2018; 28: 409–15. doi:10.3233/VES-180647

⁸⁸ Karbunarova J. Influence author methodic teaching swimming on coordination quality of children 6–10 years old with hearing disabilities. Слоб наук-спорт вісн 2016; 53: 64–8. doi:10.15391/snsv.2016-3.012

⁸⁹ Fotiadou E, Giagazoglou P, Kokaridas D, et al. Effect of rhythmic gymnastics on the dynamic balance of children with deafness. Eur J Spec Needs Educ 2002; 17: 301–9. doi:10.1080/08856250210162211

⁹⁰ Chang Y-C, et al. The Effect of Static Balance Enhance by Table Tennis Training Intervening on Deaf Children. Int J Med Health Sci 2016; 10: 356–9.

⁹¹ Borowiec J. Możliwość zastosowania muzyki i wibracji w celu poprawy koordynacyjnych zdolności motorycznych dzieci z uszkodzonym słuchem. raport z badań wstępnych / Possibilities of application of music and vibrations for improving motor coordination abilities in children with impaired hearing–pilot experiment report. Physiotherapy 2011; 19: 19. doi:10.2478/v10109-011-0009-3

⁹² Cetin SY, Erel S, Bas Aslan U. The effect of Tai Chi on balance and functional mobility in children with congenital sensorineural hearing loss. Disabil Rehabil 2020; 42: 1736–43. doi:10.1080/09638288.2018.1535629

⁹³ Lima R. Balance Assessment in Deaf Children and Teenagers Prior to and Post Capoeira Practice through the Berg Balance Scale. Int Tinnitus J 2017; 21: 77–822. doi:10.5935/0946-5448.20170016

⁹⁴ Mauerberg-deCastro E, Moraes R. Effects of dance on perception of monotonic rhythmical stimuli in deaf adolescents/A influencia da danca na percepcao de estruturas ritmicas monotonicas em adolescentes surdos. Motric 2013; 9: 69–87. doi:10.6063/motricidade.9(1).2465

⁹⁵ Ilkım M, Akyol B. The Comparison of Some Motoric Characteristics of Hearing Impaired Individuals Sports Athletic and Gymnastic. UJER 2018; 6: 2148–52. doi:10.13189/ujer.2018.061012

⁹⁶ Zarei H, Norasteh AA, Rahmanpournashrudkoli A, et al. The effects of Pilates training on static and dynamic balance of female deaf students: A randomized controlled trial. J Bodyw Mov Ther 2020; 24: 63–9. doi:10.1016/j.jbmt.2020.05.003

⁹⁷ Kaya M, Sarıtaş N. A Comparison of the Effects of Balance Training and Technological Games on Balance in Hearing-Impaired Individuals. JETS 2019; 7: 48. doi:10.11114/jets.v7i3S.4055

⁹⁸ Tzanetakos N, Papastergiou M, Vernadakis NA, et al. Utilizing physically interactive videogames for the balance training of adolescents with deafness within a physical education course. J Phys Educ Sport 2017; 17: 614–23. doi:10.7752/jpes.2017.02093

⁹⁹ Vernadakis NA, Papastergiou M, Giannousi M, et al. The effect of an exergame-based intervention on balance ability of deaf adolescents. Sport Sci 2018; 11: 36–41.

¹⁰⁰ Korkmaz C, Akın M. BOSU, KANGOO JUMP, NINTENDO-Wii BALANCE BOARD ANTRENMANLARININ İŞİTME ENGELLİ SEDANTERLERDE ÇEVİKLİK ÜZERİNE ETKİSİ. Spor ve Performans Araşt Derg 2021; 12: 91–104. doi:10.17155/omuspd.882085

¹⁰¹ Bittar RSM, Pedalini MEB, Medeiros IRY, et al. Vestibular rehabilitation in children: Preliminary study. Rev Bras Otorrinolaringol 2002; 68: 496–9. doi:10.1590/S0034-72992002000400007

¹⁰² Weber H, Barr C, Gough C, et al. How Commercially Available Virtual Reality-Based Interventions Are Delivered and Reported in Gait, Posture, and Balance Rehabilitation: A Systematic Review. Phys Ther 2020; 100: 1805–15. doi:10.1093/ptj/pzaa123

¹⁰³ Rosiak O, Jozefowicz-Korczynska M. Comment on: “Role of head-mounted displays in enhancing vestibular rehabilitation effects”. J Vestib Res 2023; 33: 229–30. doi:10.3233/VES-180665

¹⁰⁴ Hazzaa NM, Manzour AF, Yahia E, et al. Effectiveness of virtual reality-based programs as vestibular rehabilitative therapy in peripheral vestibular dysfunction: a meta-analysis. Eur Arch Otorhinolaryngol 2023; 280: 3075–86. doi:10.1007/s00405-023-07911-3

¹⁰⁵ Dunlap PM, Holmberg JM, Whitney SL. Vestibular rehabilitation: advances in peripheral and central vestibular disorders. Curr Opin Neurol 2019; 32: 137–44. doi:10.1097/WCO.0000000000000632

¹⁰⁶ Micarelli A, Viziano A, Augimeri I, et al. Three-dimensional head-mounted gaming task procedure maximizes effects of vestibular rehabilitation in unilateral vestibular hypofunction: a randomized controlled pilot trial. Int J Rehabil Res 2017; 40: 325–32. doi:10.1097/MRR.0000000000000244

¹⁰⁷ Micarelli A, Viziano A, Alessandrini M. Role of head-mounted displays in enhancing vestibular rehabilitation effects, comment on: “Evaluation of the effectiveness of a Virtual Reality-based exercise program for Unilateral Peripheral Vestibular Deficit”. J Vestib Res 2023; 33: 227–8. doi:10.3233/VES-180664

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Abstract

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Introduction

A vestibular deficit can have a substantial impact on the overall development of children. Therefore, it is of utmost importance that vestibular-impaired problems are treated early and effectively through Vestibular Rehabilitation Therapy (VRT). Although VRT is sufficiently proven and standardised in adults, there remains a lack of research examining its efficacy in children. To assess the effectiveness of VRT in vestibular-impaired children, the Vestibular Infant Screening-Rehabilitation (VIS-REHAB) protocol was developed with the following objectives: (1) to investigate the short-term effect of a combined postural control and gaze stabilisation protocol, compared with receiving no therapy and (2) to investigate the most important factors that may influence the effect of and outcome after application of the VIS-REHAB protocol in a group of vestibular-impaired children. This study aims to address lingering questions in the existing literature in a standardised manner, with the ultimate objective to establish evidence-based rehabilitation guidelines.

Methods and analysis

The VIS-REHAB study is a two-parallel group, superiority, randomised controlled crossover trial with 1:1 allocation ratio. The study includes patients aged 3–17 years old with identified peripheral vestibular dysfunction. Primary and secondary outcome measures assess gaze stability, postural stability, motor performance and quality of life. The effectiveness of the VIS-REHAB protocol will be evaluated through parallel group and crossover analyses using analysis of covariance (ANCOVA). Additionally, prespecified subgroup analyses will be conducted to assess influencing factors that may impact the outcome and effect of VIS-REHAB.

Ethics and dissemination

At the start of the VIS-REHAB study, an amendment will be submitted to the ethics committee of Ghent University Hospital for the following applications: (EC2018/0435), (EC2018/0959), (EC2015/1441) and (EC2015/1442). The trial is registered at Clinical Trials (clinicaltrials.gov) with registry name VIS-REHAB and identifier NCT06177132. All research findings will be disseminated in peer-reviewed journals.

Trial registration number

NCT06177132.

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Title

Vestibular Infant Screening-Rehabilitation (VIS-REHAB): protocol for a randomised controlled trial on Vestibular Rehabilitation Therapy (VRT) in vestibular-impaired children

Author

Fontaine, Marieke¹

; Dhooge, Ingeborg²; Dhondt, Cleo³; Ruth Van Hecke⁴

; Acke, Frederic²; Van den Bossche, Lena³; Helen Van Hoecke²; Els De Leenheer²; Maes, Leen⁵

¹ Faculty of Medicine and Health Sciences, Department of Rehabilitation Sciences, Ghent University, Gent, Belgium; Faculty of Medicine and Health Sciences, Department of Head and Skin, Ghent University, Gent, Belgium
² Faculty of Medicine and Health Sciences, Department of Head and Skin, Ghent University, Gent, Belgium; Department of Otorhinolaryngeology, Head and Neck Surgery, University Hospital Ghent, Gent, Belgium
³ Faculty of Medicine and Health Sciences, Department of Head and Skin, Ghent University, Gent, Belgium
⁴ Faculty of Medicine and Health Sciences, Department of Rehabilitation Sciences, Ghent University, Gent, Belgium
⁵ Faculty of Medicine and Health Sciences, Department of Rehabilitation Sciences, Ghent University, Gent, Belgium; Department of Otorhinolaryngeology, Head and Neck Surgery, University Hospital Ghent, Gent, Belgium

First page

e085575

Section

Ear, nose and throat/otolaryngology

Publication year

2024

Publication date

2024

Publisher

BMJ Publishing Group LTD

e-ISSN

20446055

Source type

Scholarly Journal

Language of publication

English

DOI

https://doi.org/10.1136/bmjopen-2024-085575

ProQuest document ID

3147725175

Vestibular Infant Screening-Rehabilitation (VIS-REHAB): protocol for a randomised controlled trial on Vestibular Rehabilitation Therapy (VRT) in vestibular-impaired children

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