Music Intervention, Agitation, Quality of Life and Repetitive Behaviour in People with a Form of Intellectual Disability: A Brief Pilot Report

Background: Music intervention gains increasing evidence throughout the literature. Objective: This study investigated to what extent music intervention is applied in a structured way to people with a form of intellectual disability, and whether its application is related to agitation, repetitive behaviour, and quality of life. Method: Consequently, a survey was administered analysing to what extent music is applied, and whether it is applied in the context of agitation, repetitive behaviour, and quality of life. Results: A first finding is that clients are exposed to music daily 2,5 h-3 h of music listening per day without a specific purpose, i.e. not specifically to reduce e.g. agitation. Furthermore the results show low levels of agitation and repetitive behaviour as well as high quality of life. Conclusions: Music interventions passively or actively remain an important part of a clients’ everyday life. Nonetheless, more research is needed to show more compelling and clear results.


Introduction
Evidence of the positive effects of music intervention on cognitive, communicative and social skills in persons with intellectual disabilities (ID) including Autism Spectrum Disorders (ASD) is increasing [1][2][3]. Music intervention is believed to serve as a "healing force" [4] and is used as a tool to maintain and develop these areas [5][6][7][8][9][10][11]. In addition, music intervention appears to have a beneficial influence on brain development, which in turn influences behaviour [12][13][14][15].
Challenging behaviour show the highest prevalence according to the World Health Organisation survey 2010 (54%) [16] across the lifespan of people with a form of ID. Behaviours, such as agitation and often also repetitive behaviour within a social context are often difficult to target with therapy [17]. Music, with its calming effects however, approaches these problems from an interactive and yet soothing angle [1,2,17] highlighting the underlying improvement of quality of life for each individual instead of targeting challenging behaviour at the surface of the matter [18].
Agitation is frequently mentioned in the behavioural patterns of people with a form of ID [19,20]. Studies covering this problem however, offer only pharmaceutical solutions, while a generally more riskless solution (read: music intervention) is discarded [2,21].
Against this backdrop, the results of a few studies suggest that interventions, containing music, might improve quality of life and agitation in ID; e.g. Applied Behaviour Therapy [1,22], Social Stories [23], The Nordoff-Robins method [3] and DIR/Floortime [24]. However, these positive effects are mostly based on anecdotal accounts and therefore, these interventions have difficulty to find their way into generally accepted therapeutic approaches [25]. Research investigating therapeutic effects in individuals with an ID, is so far either of an observational or qualitative  [26]. Furthermore, there is a general lack of specified music therapy approaches covering intellectual disabilities alone [26], necessitating further empirical investigation. Nonetheless, several authors have stressed the importance and superiority of music intervention as compared to for example cognitive behaviour therapy [27][28][29], underlining the beneficial effects of music intervention on reducing agitation and repetitive behaviour, and improving quality of life [27][28][29].
In line of this argument, it is important to acknowledge the form of music exposure.
On the one hand, passive exposure to music (i.e. listening to music), has shown to have positive influence on behavioral patterns in people with a form of ID, however lacks access to structured therapy strategies [29,30]. On the other hand, the use of organised sounds (read: music) allows synthesising various models of music intervention into basic therapeutic principles and furthermore supports the use of music intervention in clinical practises [31]. Further elaboration on this matter however, will exceed the purpose of this report.
The present report therefore, is meant to investigate and explore three questions: 1) general application and frequency of music intervention and music related exposure in residential community homes for people with a form of ID; 2) the relation between music intervention, quality of life and agitation levels; and 3) structural application of music intervention, if present.

Population
The population was recruited at residential community homes for persons with an ID in the Netherlands. Participants were selected by a third party, which left the researchers blind to the initial selection process. As this study set out to investigate the structural application of music intervention in first place, a control group setting was not possible. Participants were selected across all life spans regardless of receiving therapy. In the second stage of the investigation, it became apparent who has received a direct form of music therapy and who was only exposed to background music. By removing specific inclusion or exclusion criteria, a wide range of participants could be included into this study, to investigate the above-mentioned research questions. 112 participants returned the surveys, 22 participants had to be excluded, as their surveys were not complete (Response rate 81.4%). The age ranged from 11 to 78 with a mean age of M=42,91 (SD=19,92) with 43 female and 47 male participants. As the majority of family members could not provide the researchers with the exact IQ scores, participants were divided in IQ ranges (< 54 Low functioning; 55 -84 Mid functioning; > 85 High functioning). The IQ range as well as other significant medical information, was provided by the medical staff at the care home and was obtained from the participants' personal medical files. Seventeen clients were categorised as Low functioning, the majority of 63 under Mid functioning and 10 under High functioning. None of the participating clients was known to have a hearing impairment or a hypersensitivity to acoustic stimuli.
The residential community homes were situated in a more rural area of the Netherlands, where clients, under supervision, are allowed to move around freely. There are no restrictions for the clients in terms of executing their freedom.

Procedure
A Survey was distributed in a major healthcare centre for people with an ID. The survey was administered by the personal caretaker responsible for the residential community houses, where the participants reside. The caregiver, with the client's responses completed the surveys. No further restrictions were given to when and where the survey should be administered. The additional music exposure diary was administered daily over a four week period and was completed by the caregiver based on their observations on client behaviour. These observations concerned the exposure of clients to music, either as background music or active listening by the clients (clients choose and put on music themselves). The used surveys were selected, based on their validity for self-report in people with an intellectual disability.
Informed consent was obtained through family members or representatives in direct interaction with the participants, where possible.

Agitation
To assess the extent of agitation, The Cohen-Mansfield Agitation index (CMAI; [32]) was administered. The CMAI is a 29-point questionnaire with a Likert 1-7 scale, assessing aggressive behaviour from never to multiple times per day. Questions may relate to verbal aggression as well as physical aggression against others and self-harming. The 29 questions are further divided into items such as spitting, cursing, biting, exhibitionism, physical aggression such as hitting, pushing throwing things, excessive screaming, inappropriate laughing or crying, general negativity towards life as well as the misuse of furniture or objects. Additionally, verbal communication was assessed by the use of repetitive and improper sentences and phrases.

Repetitive behaviour
The Repetitive Behaviour Quotient-2, Dutch translation [33], is a 20 point questionnaire, assessing traits of repetitive behaviour in populations with autism and intellectual disabilities. A Likert scale (1-4) determines the scale of repetitive behaviour on points ranging from never to multiple times per day. The scale was developed to give an indication of repetitive behaviour in the context of the clients' surrounding.

Quality of life
Quality of Life of persons with ID was assessed by the Intellectual Disability Quality of Life-16 [34]. This instrument includes 16 questions. These questions are depicted in two drawings presented to the subject (e.g. I, my body, friends) and the action, indicated by the verb (e.g. dancing, working, activities with the family). For a full description of the pictograms used in the IDQOL-16 see [34]. The use of pictures may facilitate the communication with clients, who have difficulties with reading, or understanding verbal information. Answers are recorded with "smileys", which represent a 5-point Likert scale. Again, the use of

Music exposure and music therapy session diary
This diary was developed to get a better insight into the music related activities of the clients. The caretaker completed the diary. The diary was administered over a four week period with focus on the amount of music therapy sessions and music exposure in general. General exposure to music was further subdivided into active (the client puts on music him or herself) or passive listening (the caregiver puts on background music). Furthermore the type of music was recorded in the diary to give an indication of variety in music styles and favourites.  (Figure 1).
Informed consent was obtained prior to this research from clients, caregivers, family members or legal representatives.

Data Analysis
The received data was computed manually as a sound statistical analysis was not possible due to the unequal division of clients receiving music intervention and clients not receiving such therapy, however were generally exposed to music. Mean and standard deviation scores were computed using a standard sigma formula: score per administered questionnaire M participants The means were calculated for the three IQ ranges and yielded the here presented scores per IQ domains.
Subsequently, standard deviation was computed using a Sample Standard Deviation formula:

Level of significance
The significance of the results was manually computed with a standard significance test formula:

Results
As there was an unbalanced spread of participants across the expected groups (music therapy N=9 and general exposure to music n=81) a between groups analysis was discarded.
Following results are taken as raw scores without taking music intervention and exposure into consideration.
A Mean score was computed to give an indication of the administered CMAI, RBQ-2 and IDQOL-16.

Music exposure
The music exposure questionnaire has revealed overall exposure to music. Even though clients have not received a form of music therapy, clients regularly listen to music either by turning on the radio themselves or requesting it or by initiative of the caregiver. Analysis of the music exposure diary shows that 56 participants listen passively to music; music is played in the background without the clients requesting it. Twenty-five participants have listened actively to music; choosing the music, requesting it or putting it on themselves. The remaining nine have received a form of music therapy and have furthermore listened actively and passively to music. All three groups are exposed to music 2,5h -3h on average per day.

Overall level of agitation and perseveration
Scores on the CMAI yielded a relatively low mean (M=45,07; SD 5,94; p ≤ 0,05) with two outliers: highest score at 102 and lowest 12. However, the average participant scored between 43 and 48 on the Cohen-Manfield Agitation Index.

Overall level of repetitive behaviour
The RBQ-2 also showed a rather low mean of 26,94 (SD 6,23; p ≤ 0,05), indicating a low level of repetitive behaviour in the tested sample.

Overall quality of Life
The Mean score on the IDQOL-16 was surprisingly high (M=60,33; SD 1,58) with a significance level of p ≤ 0,05.

Agitation, repetitive behaviour and quality of life and IQ ranges
Splitting the analysis over the individual IQ-ranges revealed no compelling differences. Scores across the low functioning IQ range varied by only few points from the overall mean in the CMAI, RBQ-2 and IDQOL-16 (Table 1). Scores in the mid-functioning IQrange reflect the overall average. This was expected as this group is mostly represented in this sample (Table 1).
Here again in-between group analysis was discarded as the hereperformed analyses revealed that the samples were divided too unequally to be compared.

Active participation in music therapy
As this study has aimed at analysing the distribution as well as the effect of music intervention in populations with an intellectual disability, the amount of received music intervention was evaluated. Of note however, analysing the amount of participants receiving music therapy, has shown that only nine out of ninety participants do receive a form of music therapy (data not shown).

Discussion
The primary goal of the present study was to examine whether music was applied in a structured way, and whether its application is related to agitation, repetitive behaviour, and quality of life. The administered survey has revealed two interesting developments related to the initial question: 1) music is hardly offered in a structured manner to improve behaviour; 2) agitation and other behavioural problems were very low in the tested sample.
The question arises, if this can be accredited to the passive music exposure, which all clients received. Further research will be needed to find causality between passive music exposure and low levels of agitation.  Music exposure and intervention diary (Authors translation). Columns ask, if the client has received music intervention sessions and how often, if yes the length of the session and its influence on possible aggressive behaviour. Furthermore the diary asks, if the client has put on music, music was put on by the caretaker and which genre of music was mainly listened to. This again is related to an increase or reduction in possible aggressive behaviour.  behaviour [25]. Likewise, Koseki and colleagues have shown that an enriched environment with diverse high levels of activity has a significant effect on the development of young mice, decreasing their agitation levels in adulthood [35]: music can be seen as one of these high level enriched environment activities [35]. In the here presented sample, however, the results do not show a 'direct' need to counteract aggressive behaviour with explicit music therapy, as clients have shown very low scores on agitation and are furthermore exposed daily to music with an average time of 2,5 hours to 3 hours. Nonetheless, there were nine clients who received direct music therapy in addition to the general music exposure (data not shown). A preliminary analysis of this group however, has shown no difference as compared with clients who did not receive direct music therapy on the test scores, which makes a detailed description of the applied intervention optional. Giving the low N in the music therapy group, further investigation as well as a direct comparison was discarded for the purpose of this report.
Clients as well as caregivers recorded an interestingly high score on the administered IDQOL-16. This reflects an overall satisfaction with the facilities and surrounding the clients are living in. This finding can however, not be accredited to the general use of music alone in these facilities.
Finally, no evidence of a clear therapeutic line was evident when analysing application and needs of music intervention in a structural way within the tested sample. The here presented study has recruited the sample with the primary goal of investigating the use of the above-mentioned structural way of music intervention in first place. In the second stage of the analysis, it became apparent, which clients received direct music therapy, making an in-between group analysis not possible (data not shown).
Further research is needed as to how to explain the relation between general music exposure, direct music therapy and levels of agitation, repetitive behaviour and quality of life. Equal distribution across the participating experimental and control groups (a pre -post measurement setting) would make a direct comparison of structured music intervention against no music intervention (read: therapy) possible ( Table 2). Taking into account the heterogeneity of the participating population, a randomised controlled trial is questionable as the best research methodology, and either an N=1 or block randomisation should be taken into consideration.
This study has hardly embarked on the long journey of how music interventions are administered in practice. Missing therapeutic lines and mistrust towards the benefits of music therapy on behaviour, agitation and quality of life make this form of intervention difficult to administer in a structured way (Table 3).
After all, understanding any beneficial form of intervention supports the intention of any therapy: client well-being.