Musical interaction with children and young people with severe or profound intellectual and multiple disabilities: a scoping review

Figures & data

Table 1. Search strategy for PubMed database.

Search ((((((((((syndrom*[Title/Abstract] OR impairment[Title/Abstract] OR disab*[Title/Abstract] OR "brain injury"[Title/Abstract] OR autism[Title/Abstract] OR "cerebral palsy"[Title/Abstract] OR "mental retardation"[Title/Abstract] OR "special needs"[Title/Abstract] OR "special need"[Title/Abstract] OR "learning disability"[Title/Abstract] OR "learning disabilities"[Title/Abstract] OR "developmental disability"[Title/Abstract] OR "developmental disabilities"[Title/Abstract])) OR brain damage, chronic[MeSH Terms]) OR disabled children[MeSH Terms]) OR intellectual disability[MeSH Terms]) OR mental retardation[MeSH Terms]) OR intellectual disabilities[MeSH Terms]) OR education, special[MeSH Terms])) AND (((((music*[Title/Abstract] OR "music therapy"[Title/Abstract] OR "interactive music"[Title/Abstract] OR "musical interaction"[Title/Abstract] OR "music interaction"[Title/Abstract] OR "music education"[Title/Abstract] OR "music activities"[Title/Abstract] OR "music activity"[Title/Abstract])) OR music therapy[MeSH Terms]) OR therapy, music[MeSH Terms]) OR music[MeSH Terms])) AND (((((child*[Title/Abstract] OR adolescen*[Title/Abstract] OR teen*[Title/Abstract] OR "young adult"[Title/Abstract] OR "young adults"[Title/Abstract] OR pupil*[Title/Abstract] OR student*[Title/Abstract] OR youth*[Title/Abstract])) OR child, preschool[MeSH Terms]) OR young adult[MeSH Terms]) OR adolescent[MeSH Terms])

Figure 1. Flowchart of study selection for scoping review.

Table 2. Overview of studies on musical interaction and children with S/PIMD included in the review.

Authors, Year, Country	Participant characteristics (n, age)	Study design/ Data collection methods	Musical-interaction activities	Abilities and behaviours in focus	Reported benefits/effectiveness, promising components	Quality appraisal
1. Yasuhara and Sugiyama 2001, Japan.	Three children with RTT (4; 5 and 6 years old). All patients had a DQ below 20.	Case-study design.Quant. data. Video observations. Extant instruments. Percentages of improvements in different abilities were presented in visual graphs.	Individual, active music therapy.	Social interaction, communication, motor abilities, musical abilities.	Reported effectiveness. No control.	Poor validity:24%
2. Ghetti 2002, USA.	Six school-aged children (7–17 years old) with profound intellectual disabilities.	Single-subject experimental design.Quant. data. Video observations. Extant instrument. Mean and SD for percentages spent in alert states. A one-way-within-subjects ANOVA was conducted to test homogeneity of subjects’ baseline measures across time. Wilks’ lambda was chosen as the test statistic. Inter-rater reliability was performed.	Individual, active music therapy, control condition.	Attention and alertness.	No effectiveness reported. Category: 2,4.	Valid: 88%
3. Rainey-Perry, 2003, Australia.	Ten children with severe and multiple disabilities (5–11 years old).	Case-study design.Qual. data. Video observations. Author-constructed instrument. Analysis focused on the balance in turn-taking (between child and therapist). Additional data involved the researcher’s field notes during data collection.	Individual, active music therapy.	Social interaction and communication.	Partial effectiveness reported. No control. Category: 1,2,3.	Valid:88%
4. Elefant and Lotan 2004, Norway/Israel.	One 9-year-old girl with RTT.	Case-study design.Qual. data. No specific instruments reported.	Individual, dual treatment: Music therapy & physiotherapy.	Communication.	Reported effectiveness. No control. Category: 1,3.	Poor validity: 23%
5. Ellis 2004, UK.	One girl with PMLD (8 years old).	Case-study design.Qual. data. Video observations. No specific instruments reported.	Individual, technology-mediated musical activities with multisensory elements.	Social interaction, communication and movement.	Reported effectiveness. No control. Category: 5.	Poor validity:12%
6. Holck 2004, Denmark.	Six children with severe functional limitations (three with a comorbid ASD).	Case-study design.Qual. data. Video observations. Author-constructed instrument. Cross-comparative analyses. Interviews with the participating music therapists were also carried out.	Individual, active music therapy.	Social interaction and communication.	Benefits of interactive music are reported. No control. Category: 1,3.	Poor validity:59%
7. Elefant and Wigram 2005, Norway, Denmark.	Seven girls with RTT (4–10 years old).	Single-subject experimental design.Quant. data. Frequencies of chosen songs were calculated and presented in tables, and trend lines in baseline, intervention and maintenance phases.	Individual, active music therapy.	Social interaction, communication and cognitive abilities.	Reported effectiveness. Control condition. Category: 2,4,6.	Poor validity: 59%
8. Wigram and Lawrence 2005, Denmark/UK.	One girl with RTT (6 years old).	Other design. Case report.Qual. data. Video observations. No specific instruments reported.	Individual, active music therapy.	Social interaction and communication, cognitive abilities and engagement.	Benefits of interactive music are reported. No control. Category: 1.	Poor validity: 47%
9. DeBedout and Worden 2006, USA.	Seventeen (5–13 years old) with severe intellectual disabilities. All but three were non-ambulatory.	Single-subject experimental design.Quant data. Within-subject analyses across conditions. Video observations. Author-constructed instrument.Analysis through placing the response totals from two conditions in rank order using Kruskal Wallis test to evaluate rank sums.	Individual, active music therapy, control condition.	Social interaction, communication, affect and movements.	Reported effectiveness. Control condition. Category: 2,6.	Valid:76%
10. Azeredo 2007, Portugal.	One 13-year-old boy with motor-cognitive disabilities.	Other design.Quant. data. Video observations. Rating scale of different abilities. Descriptive statistics and visual analysis.	Individual, technology-mediated musical activities with multisensory elements.	Musical abilities, attention, engagement.	Reported effectiveness. No control. Category: 5.	Poor validity: 29%
11. Geist et al. 2008, USA.	One four-year-old boy with global developmental delay.	Case-study design.Qual. data. Video observations. Pre- and post-musical interaction activities were analysed by blind raters in order to confirm the social validity of the results described in the qualitative treatment assessment summary.	Individual and group, dual treatment with music therapy and AAC.	Social interaction and engagement.	Reported effectiveness. No control. Category: 2.	Poor validity:71%
12. Wheeler and Stultz 2008, USA.	Four children with multiple severe disabilities (4–12 years old). The children had a variety of diagnoses. All had severe intellectual disabilities and were non-verbal.	Case-study design.Qual. data. Video observations. Analysis involved comparing the different groups in a qualitative manner within a psychosocial developmental theoretical framework. No specific instruments reported.	Individual or in dyads, active music therapy.	Social interaction, communication, engagement, affect, musical abilities.	Benefits of interactive music are reported. No control. Category: 3.	Poor validity:67%
13. Go and Mitani 2009, Japan.	Ten girls with RTT (3–17 years old).	Case-study design.Qual. data. Video observations were used to assess temporal, spatial and kinetic changes in movements in response to music. Visual/ graphic data.	Individual, active music therapy.	Movement and motor abilities.	Partial effectiveness reported. No control.	Valid: 76%
14. Welch et al. 2009, UK.	Sixty-eight participants involving children and young children with complex needs (4–19 years old).	Longitudinal observation study.Quant. data. Video observations. Author-constructed instrument. Differences in the distribution of the observed ratings were calculated using Chi-square, and similarities in the patterns of observations were calculated using Kendall’s coefficient of concordance.	Group music lessons.	Musical abilities.	Benefits of interactive music are reported. No control. Category: 4.	Valid:76%
15. Magee et al. 2011, UK/USA.	Two adolescent boys with significant cognitive challenges.	Case-study design.Qual. data. No specific instruments reported.	Individual or group, technology-mediated activities with multisensory elements.	Musical abilities, cognitive abilities, participation.	Reported effectiveness. No control. Category: 5,6.	Poor validity:13%
16. McFerran and Shanahan 2011, Australia.	Three pre-adolescent boys with profound developmental delay, physical limitations and cortical vision impairments.	Case-study design.Mixed methods. Video observations. Author-constructed instrument. Strategies used by the music therapist were recorded for frequency and duration, and calculated as a percentage of the session time. The responses from the children were recorded for frequency and categorised deductively. The results were recorded in a spreadsheet and converted to graphs for descriptive analysis.	Individual, active music therapy.	Social interaction, communication, musical abilities, therapist’s strategies.	Benefits of interactive music are reported. No control. Category: 1,2.	Valid:82%
17. Ockelford et al. 2011, UK.	Twenty children with PMLD (11–17 years old).	Longitudinal observation study.Quant. data.Observations. Author-constructed instrument. The mapped behaviours were observed, recorded in different categories and ranked as low, medium or high.	Group, music lessons.	Musical abilities.	Benefits of interactive music are reported. No control. Category: 3,4.	Valid:82%
18. Raglio et al. 2011, Italy.	One boy with severe/profound ID and ASD (seven years old).	Longitudinal observation study.Quant. data. Video observations. Author-constructed instrument. Occurrences of affect attunement were assessed between each session and the following one, and between the first and the last session using Chi-square. Behavioural sequences in interaction were analysed by sequential analysis and presented in transition diagrams.	Individual, active music therapy.	Social interaction, communication, affect.	Benefits of interactive music are reported. No control. Category: 1.	Poor validity:71%
19. Hackett et al. 2013, UK.	One girl with RTT (4 years old).	Other design.Mixed methods. Video observations. The frequency of observations in each session was counted, and plotted through trend analysis using Statistical Process Control. Parental testimonies were also collected. Interrater reliability tests were performed.	Individual, active music therapy.	Social interaction, motor abilities.	Reported effectiveness. No control. Category: 1,2,3,6.	Valid: 82%
20. McFerran and Shoemark 2013, Australia.	One boy with profound intellectual/developmental disabilities (16 years old).	Case-study design.Qual. data. Duo-ethnographic method. Video observations. A form was provided for noting observations, interpretations and questions, and was divided into 10-second intervals. Interviews were carried out with the researchers after the video reviews, as well as an interview with the participating music therapist.	Individual, active music therapy.	Engagement.	Benefits of interactive music are reported. No control. Category: 1,3,4,6.	Valid:88%
21. Stensaeth 2013, Norway.	Two children with severe/profound ID and ASD (11; 15 years old).	Case-study design.Qual. data. Video observations. No specific instruments reported.	Individual, technology-mediated activities with multisensory elements.	Social interaction and communication, engagement.	Benefits of interactive music are reported. No control. Category: 1,5.	Poor validity: 47%
22. Thompson and McFerran 2015, Australia.	Four adolescents with profound intellectual and developmental disabilities (10–15 years old).	Case-study design.Mixed methods. Video observation. Extant instrument. Session notes: the music therapist recorded her impressions of the young person’s engagement and affect, as well as her sense of connectedness after each session. In data analysis, frequencies were tallied across the range of communicative categories in the two conditions generating descriptive data.	Individual, active music therapy, control condition.	Social interaction, communication, engagement.	Reported effectiveness. Control condition. Category: 4.	Valid:82%
23. Kossyvaki and Curran 2020, UK.	Five children with autism & severe or profound intellectual disabilities (5–7 years old).	Case-study design.Mixed methods. Video observations. A self-developed checklist based on four assessment instruments was used to count occurrences of behaviours within each interval. IRR were performed. Qual. interview data were analysed thematically.	Individual, technology-mediated musical activities with multisensory elements.	Social interaction, communication, engagement.	Partial effectiveness reported. No control. Category: 1,2,5.	Valid: 88%
24. Chou et al. 2019, Taiwan.	Intervention group: 11 families with a child with RTT (mean age: 14.45 years).Control group: 12 families with a child with RTT (mean age: 12.08 years).	Experimental group design (non-randomised).Quant. data. Standardised tests pre- and post-intervention. Independent t-test to compare inter-group variability. Linear Mixed Effects Models to adjust for variations involving age or baseline severity.	Group, active music therapy, control condition.	Social interaction & communication, motor abilities, seizure frequency, parental stress.	Reported effectiveness. Control group.	Valid: 84%
25. Nielsen and Holck 2020, Denmark.	One boy with severe ASD, severe intellectual disability and ADHD (5 years old).	Case-study design.Qual. data. Video observations. Participant and music therapist’s behaviours were coded in parallel in an Excel sheet and a diagram.	Individual, active music therapy.	Musical abilities, social interaction and communication.	Benefits of interactive music are reported. No control. Category: 1,3.	Valid:88%

Note. AAC = Augmentative and alternative communication; ASD = Attention deficit hyperactivity disorder; ASD = Autism spectrum disorder; DQ = Developmental quotient; PMLD = Profound and multiple learning difficulties; RTT = Rett syndrome; SD = Standard deviation.

Figure 2. Terms used in the studies to describe the participants.

Figure 3. Abilities and behaviours in focus in the studies.

Table 3. Quality appraisal, using an adapted EBL Critical Appraisal Checklist.

Item	Yes	No	Unclear	N/A
Study population
1.1 Is the chosen study population consistent with the population about which conclusions are drawn?	23	1	1
1.2 Are inclusion and exclusion criteria definitively outlined?	21	2	2
1.3 Is the sample size large enough for sufficiently precise estimates, representative of the entire population?				25
1.4 Is the choice of population bias-free?				25
1.5 If a comparative study, were participants randomised into comparable groups?		2		23
1.6 Was informed consent obtained?	15	3	7
Data collection
2.1 Are data collection methods clearly described?	18	7
2.2 Were inter-observer and/or intra-observer bias reduced?	11	11	3
2.3 Is the instrument included in the publication or is its content clearly described?	16	8	1
2.4 Is the data collection instrument validated?	2	13	10
2.5 Does the study measure the outcome at a time appropriate for capturing the intervention’s effect?	19		6
Study design
3.1 Is the study type/methodology utilised appropriate?	19		6
3.2 Is the research methodology clearly stated at a level of detail that would allow its replication?	14	6	5
3.3 Was ethics approval obtained?	15	7	3
Results
4.1 Are the outcomes clearly stated and discussed in relation to data collection?	21		4
4.2 Are the results clearly outlined?	23	1	1
4.3 Are confounding variables accounted for?	14	9	2
4.4 Do the conclusions accurately reflect the analysis?	19		6
4.5 Does the study build upon previous research and are suggestions provided for further areas to research?	22	3
4.6 Is there external validity?		25

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