Review Journal of Autism and Developmental Disorders

https://doi.org/10.1007/s40489-020-00219-6

REVIEW PAPER

Parent-Mediated Music Interventions with Children with ASD:

a Systematic Review
Eugenia Hernandez-Ruiz 1

Received: 4 November 2019 / Accepted: 30 July 2020

# Springer Science+Business Media, LLC, part of Springer Nature 2020

Abstract
Parent-mediated interventions complement professional services and empower parents. In music therapy, this treatment modality
is emerging. A systematic review was conducted to assess the existence of parent-mediated music interventions with children
with ASD, identify the characteristics of parent coaching used, and summarize results. Thirteen studies were selected. Framework
analyses were performed on descriptions of “parent-mediated,” “parent coaching,” outcomes, measures, and findings. Quality of
the research and of the training programs varied substantially. Salient findings include emerging interest in involving parents in
treatment, limited conceptual frameworks for parent coaching, and limited development of this modality. High attrition, parental
effort, and mixed populations limit the internal and social validity of studies. Music therapists may consider investigating parent-
mediated interventions, while considering parental effort.

Keywords Parent-mediated . Music-based . Music intervention . Music therapy . Parent coaching . Systematic . ASD . Autism

Autism spectrum disorder (ASD) affects social communica- et al. 2015). Oftentimes, these models are accompanied by
tion and interactions from very early in life. Repetitive and parent coaching, which are the professional services to edu-
restrictive behaviors and difficulties in social communication, cate the parents in the use of parent-mediated interventions
the two diagnostic criteria for ASD (DSM-5 2013), can impact (Steiner et al. 2012).
not only child development but also family functioning (May Several studies examining parent-mediated interventions
et al. 2015). Early intervention improves prognosis and is show that parents can achieve treatment fidelity within rea-
recommended as best practice (Zwaigenbaum et al. 2015), sonable timeframes (2 to 12 weeks of 1- or 2-h sessions), and
but professional services are often limited due to time, cost, within different treatment approaches, such as Pivotal
or availability in the region. Parent-mediated therapies have Response Training (Coolican et al. 2010), Positive Behavior
been researched as viable options for these families (see Support (Lucyshyn et al. 2015), relationship-based video-
Strauss, Mancini, SPC Group,, and Fava 2013, for a training (Poslawsky et al. 2014), and joint attention treatment
comprehensive review). Parent-mediated, also called parent- (Ingersoll and Wainer 2013). In those studies, parents main-
led, interventions are treatment models where the parents pro- tained fidelity to procedures at follow-up, and their ability to
vide the intervention strategies, receive appropriate training provide the intervention correlated with child’s improved
and monitoring, and are involved in setting goals, locating communication skills and behavior (Coolican et al. 2010;
resources, and reinforcing children’s skills (Zwaigenbaum Ingersoll and Wainer 2013; Lucyshyn et al. 2015). Even when
professional services were also provided, parental involve-
Electronic supplementary material The online version of this article ment increased child outcomes, and parental wellbeing and
(https://doi.org/10.1007/s40489-020-00219-6) contains supplementary satisfaction (Oono et al. 2013; Zwaigenbaum et al. 2015).
material, which is available to authorized users. Music therapy for individuals with autism spectrum disor-
der has substantial research support. Preliminary scoping
* Eugenia Hernandez-Ruiz searches of systematic reviews and consultation of
[email protected]
PROSPERO revealed that recent or ongoing systematic re-
1
Music Education and Music Therapy Division, School of Music,
views of music interventions include evaluation of music ther-
Herberger Institute of Design and the Arts, Arizona State University, apy for ASD (Geretsegger et al. 2014), movement and music
P.O. Box, 870405, Tempe, AZ 85287-0405, USA therapy in ASD (Bhat and Srinivasan 2013), instruments for
 Rev J Autism Dev Disord

evaluation of outcomes in music therapy (Fusar-Poli The absence of a comparable research base for parent-
et al., ongoing), and music therapy for children with ASD mediated music interventions does not necessarily represent
(Simpson and Keen 2011). This body of aggregated research a scarcity of interest. However, its absence, along with the
indicates a strong empirical basis for the use of music inter- absence of the parent-mediated construct within the music
ventions for individuals with ASD. Further, this population is therapy literature, indicates an area of opportunity for music
one of the most frequently supported by the work of music therapists to conceptualize and develop intervention models
therapists (AMTA 2018). Given the amount of music therapy that include families as part of service delivery, thus increas-
research and clinical work in the ASD field, it would be rea- ing access to services for individuals with ASD. Such services
sonable to assume that music therapists have also explored the could support early intervention, even when professional ser-
incorporation of parents in the therapeutic milieu. vices are limited, and could increase parental self-efficacy
In fact, when performing a scoping search with the string (Oono et al. 2013). Music therapists are well-positioned to
“(parent OR caregiver) AND (mediated OR led) AND autism share effective, playful, and developmentally appropriate
AND music” during the last 10 years (2007–2016), 1069 peer- strategies for social communication of children with ASD
reviewed articles and reviews were found, pointing to consid- (Kern and Humpal 2018). A systematic review that aimed to
erable research in this area. However, two concerns arose while understand the current state of research of music interventions
screening this literature. First, many of these articles did not use with parents and children with ASD, and that aimed to criti-
music as the primary ingredient of the intervention. For the cally examine the definitions, models, and findings of those
purposes of this review, music intervention is the therapeutic studies, seemed an important first step to support the develop-
use of music where the “auditory stimuli [is used] to achieve ment of theory and future empirical studies.
desired changes in physiological and psychological function” This review aimed to explore that current state of research
(Robb et al. 2011, p. 7). In other words, music is the essential in music interventions with parents and their children with
ingredient of the intervention, without which the intervention ASD. It was the first step in a research line that included
could not be performed. Many of the articles in this scoping theory development (Hernandez-Ruiz 2020) and empirical re-
search did not comply with this definition, but rather used mu- search of a parent-mediated music intervention and corre-
sic as background or as a small, dispensable part of the inter- sponding parent coaching (Hernandez-Ruiz 2019).
vention. For example, Nunes et al. (2016) used “toys and mu- Considering this emerging process and the scarcity of the
sic” as one of the components of their intervention program, literature, efficacy and effectiveness evaluation of the inter-
which also included parent education on autism, communica- ventions were not the focus of this review. Instead, the follow-
tion stages, play, routines, adjusting language, visual aids, ing research questions guided this study:
books, and group interactions. More importantly, the only ex-
planation of the use of music was “[to] identify songs that favor 1 What parent-mediated/parent-led music interventions have
turn-taking and enhance communication” (Nunes et al. 2016, been researched with children with ASD?
Appendix 1, Table 2). A music intervention, as defined by 2 What training do researchers/interventionists have to
Robb et al. (2011), would require music to be the essential, coach parents in these interventions?
irreplaceable ingredient of the main intervention, which does 3 Do these interventionists/researchers provide systematic
not seem to be the case in this and other studies. parent coaching on the intervention?
Second, the term parent-mediated was not commonly used 4 How are “parent-mediated/parent-led” music interventions
in music therapy articles. We defined music therapy articles as described, defined, or understood? What is the definition
those where the music intervention was designed and/or of parent coaching/training in these studies?
researched by a music therapist. No studies using the term 5 What are the outcomes and measures for both parents and
parent-mediated were found during this initial search under children?
this criterion or within the Journal of Music Therapy, Nordic 6 What are the main results/contributions of these studies?
Journal of Music Therapy, and Music Therapy Perspectives,
the premiere journals of the profession. In other words, music
intervention studies conducted by professional music thera-
pists that specifically incorporate parents as providers of at Method
least parts of the services seemed to be scarce in the literature.
Contrastingly, 36 research studies of parent-mediated inter- Design
ventions without music were located across different disci-
plines, including several RCTs (Kasari et al. 2015; Siller A systematic review with a configurative approach was devel-
et al. 2014), long-term follow-up of RCTs (Pickles et al. oped. In configurative reviews, the researcher attempts to in-
2016), and a Cochrane review (Oono et al. 2013), indicating clude studies that “provide richness” of methodologies and
the importance of this mode of intervention in the ASD field. approaches (Gough et al. 2012a, p. 60). This variety allows
Rev J Autism Dev Disord

distinctions and exploration of theory in an iterative process. interventions for adolescents might be less dependent on
Different from aggregative reviews, the aim of configurative parents.
reviews is exploratory as they intend to interpret and arrange 7 Parent-mediated/led intervention: It was expected that par-
(i.e., configure) information and “to provide enlightenment ents/carers/caregivers were explicitly mentioned as re-
through new ways of understanding.” (Gough et al. 2012b, sponsible for the implementation of at least part of the
p. 3). The intention is not to identify the most effective inter- therapy outside of the sessions, and received a minimum
vention for a given condition, but rather to understand differ- of training and monitoring.
ent attributes of it, and to develop new concepts (Gough et al. 8 Music intervention: Intervention had music as its main and
2012b). This important distinction permeated the develop- critical ingredient. Bundled interventions (e.g., evaluation
ment of the method in this review. of comprehensive programs) were included as long as the
music intervention was reported as a clearly separate
component.
Inclusion Criteria

1 Articles from 2007 to 2017: this period was selected given Exclusion Criteria
that the scoping searches showed a substantial increase in
publications related to parent-mediated interventions in Further criteria that were added during the screening process
ASD in the last decade. included the following: Duplicate, Invalid citation (citations
2 Peer-reviewed articles and dissertations. This criterion was provided by the databases without readable information),
included to ensure that the studies had received appropri- Irrelevant topic (citations with unrelated topics, such as obe-
ate supervision and that the quality of the publications was sity and cancer), and No children diagnosed with ASD. The
acceptable. first three criteria were needed since some databases included
3 In English, Spanish, or French, to ensure literature from faulty citations. The last criterion was added to exclude studies
other countries was included. We selected these languages that included individuals with “developmental disabilities/dis-
given the fact that most literature in music therapy is pub- orders” (such as Down syndrome) but did not include at least
lished in them.1 some children with ASD, consistent with our stated aim.
4 Most research methodologies accepted. Since the purpose
of this review is to support the development of a concep-
tual framework, the inclusion of different methodologies Procedure
would enrich this endeavor. On the other hand, articles
without data collection (theoretical or position papers) The following procedure was designed based on recommen-
were excluded given that outcomes, measures, and find- dations by Boland and Cherry (2014), Campbell
ings were part of a research question, and such papers Collaboration (2011a, b, c, d), and Gough and collaborators
would not yield any information. Secondary research (2012a, b):
(i.e., systematic reviews, meta-analyses) were also exclud-
ed to avoid duplication. Search Strategy
5 Population studied included individuals with developmen-
tal delays, developmental disabilities, neurodevelopmental Consultation with a specialized librarian yielded the search
disorders, autism, or autism spectrum disorder. All these strategy depicted in Fig. 1. Databases included PsycInfo,
terms were included in the search since these are common PubMed, Web of Science, JSTOR, Google Scholar, ERIC,
labels for the target population. The exclusion criterion and the aggregated databases, Academic Search Complete,
(No children diagnosed with ASD, see next section) helped Music Index, Proquest Research Library, and Psychology
narrow the results during screening. and Behavioral Sciences Collections. Proquest Dissertation
6 Children receiving intervention under 12 years: It was con- and Theses Global was included later when it was observed
sidered that, even though interventions for children 0 to that very few dissertations were included in the previous
12 years of age vary substantially, this variation would be searches. Search terms included “parent/caregiver/carer,”
less than those directed for adolescents. Moreover, “music,” and “autis*/developmental disabilit*/developmental
disorder/neurodevelopmental disorder/ ASD/ developmental
1
Only one article in French and two in Spanish were found, all with English delay.” An example of the search strategy is available on
abstracts. The articles in Spanish were excluded at the Title/Abstract screening Appendix 1, p.3. A total of 2463 citations were retrieved in
level since they were theoretical/dissemination articles. The French article was the first iteration of the review. This review was updated after
included for data extraction and was translated to English by a hired certified
French-English translator, who is a French native speaker with a Master’s the dissertation document was published to include articles
degree in the USA. from 2017 (n = 324 citations) for a total of 2787 citations
 Rev J Autism Dev Disord

Fig. 1 Search strategy. Note:

Truncation (e.g., autis*), “Related Search And
searches,” “Exploding,” In individual
“Expanders,” “Related words,” individual aggregated
databases
and “Major” were used to locate terms databases
all pertinent literature in databases
that allowed them

Truncation
“Explode”,
“related
searches”,
“Major”,
“expanders”

Combine each search term

(music/autism/parent)
using “OR”

Combine all search terms

(music/autism/parent)
using “AND”

Apply FILTERS:

Publicaon date (2007-2017)

Language (English, French,
Spanish)

Type of publicaon/peer reviewed

retrieved. The numbers reported in the following section in- provides a visual representation of this process and is
clude the sum of citations from both searches. described here:

1 Title screening (2554 citations). A quick screening of title

Screening Process and keywords in the abstract, excluding a total of 1449
articles. The exclusion criteria 5 “invalid citation” and 6
The online systematic review software Covidence (Veritas “irrelevant topic” were added at this point. Also, some
Health Innovation, Melbourne, Australia. Available at www. articles missed by online databases or Covidence for type
covidence.org) was used to screen the resulting citations. The of publication (341), language (2), or duplicates (18) were
software automatically counted and extracted duplicates (233 excluded at this point. The excluded citations were
citations), but the researcher checked for accuracy. A total of downloaded to a spreadsheet and manually coded. This
2554 citations were screened. A PRISMA chart (Fig. 2) process served as a second screening and improved
Rev J Autism Dev Disord

2,554 Title screened: 1,449 Excluded

Inclusion/Exclusion
Criteria applied

1,105 Title/Abstract
screened: 969 Excluded
Inclusion/Exclusion
Criteria applied

124 Studies Excluded

Reasons:
52 Not music-based intervenon
25 Parents not leading/co-therapist
136 + 1 citaons Full-text Screen: 9 Not peer reviewed or dissertaon
Inclusion/Exclusion Criteria Applied 9 Not published 2007-2017
10 No children diagnosed with ASD
6 Children over 12 years
6 Theorecal/Posion paper
4 Irrelevant topic
1 arcle from reference
1 Invalid or incomplete citaon
list included (Nicholson
1 Duplicate
et al., 2008)
1 Not in English, Spanish or French

13 Studies Included for Data Extracon

Fig. 2 PRISMA chart

accuracy. The remainder 1105 citations were kept for the 3 Full-text review of selected articles (136 + 1). In this
following stage. step, all articles from the first search and the update were
2 Title/abstract screening (1105 citations). For the first read by two reviewers: the researcher and one of the
search (years 2007–2016), only the main researcher trained coders. Inter-rater reliability (IRR) scores were
reviewed the citations (866 citations). For the update calculated between each of the trained coders (who each
(2017), a team of three trained coders (graduate students) read a third of the articles) and the researcher (who read
and the researcher reviewed the citations (266 articles). all the articles). IRRs were 80.5%, 81.3%, and 90.6%.
The title and abstract were read, and a decision was made Discrepancies were discussed until agreement was
based on inclusion/exclusion criteria as well as the con- reached. One article cited in one of the reviewed articles
ceptual and operational definitions (see Appendix 1). was considered relevant and was included (Nicholson
When information from the title/abstract was insufficient et al. 2008). At this point, one article (Yang 2016) was
to decide, the article was transferred to the next stage (Full- excluded as a duplicate because it was the published
text review). Once again, the excluded citations (n = 969) article of a dissertation (Yang 2013), and the latter pro-
were downloaded and manually coded with the exclusion vided better information to this review. A total of 123
reason.
 Rev J Autism Dev Disord

articles were excluded at this stage. A screening tool was To answer question 1 (What parent-mediated/parent-led mu-
completed for each included article (see Appendix 2). sic interventions with children with ASD have been
researched?), question 2 (What training do researchers/
i n t e r v e n t i o n i s t s h a v e to c o a c h pa r e n t s i n t h e s e
interventions?), and question 3 (Do these interventionists/
Data Extraction (13 Articles) researchers provide systematic parent coaching on the
intervention?), data from the extraction tools were summa-
Trained coders used a data extraction tool for each article to rized in Table 3 and are explained in the “Results” section.
find and report relevant data (see Appendix 2). The resulting
data were compiled in spreadsheets for further analysis. Framework Analyses

Quality Assessment of Included Articles To answer question 4 (How are parent-mediated/parent-led

music interventions described, defined, or understood?),
Given that the purpose of this review was an exploration of the question 5 (What are the outcome measures for both parents
literature in parent-mediated music interventions, and not the and children?), and question 6 (What are the main findings/
evaluation of intervention effectiveness, no article was exclud- contributions of these studies?), framework analyses with
ed due to low quality. However, quality assessments are re- these subtopics were performed (results reported in Tables 3
ported as another feature of the existing literature. Considering to 5). Framework analysis is a tool to synthesize the concepts
that most of the music intervention literature is based on non- emerging from the review in an initial thematic framework
randomized studies, the Downs & Black Quality Assessment through an inductive approach (as described in Gough et al.
tool (Downs and Black 1998) was selected as an appropriate 2012a, b, and Ritchie and Spencer 1994). Following Ritchie
quality assessment tool (Deeks et al. 2003). Different from and Spencer’s (1994) procedure, the framework analyses in-
other quality assessment tools, which are typically intended cluded the following steps:
for randomized trials, this checklist assesses methodological
quality of both randomized and non-randomized studies. It 1 Familiarization: immersion in the data to find “key issues
has strong internal consistency (KR-20 = .90), high test- and emerging themes” (Ritchie and Spencer 1994, p. 178).
retest reliability (r = 0.88), good inter-rater reliability A review of all the materials, a selection of the pertinent
(r = 0.75), and good face and criterion validity. The tool has sources, as well as an appraisal of the ease or difficulty of
five subscales, including intervention reporting, external va- obtaining the data are considered and noted.
lidity, internal validity (bias), internal validity (confounding), 2 Identification of a thematic framework: abstraction and
and power. The instrument has a total of 27 items. Each item conceptualization of the data. The data are “sifted and
assesses whether the methodological feature is present or not sorted” to set up a thematic framework, informed by the
(e.g., “Are the characteristics of the patients included clearly research aims and extracted based on recurrence or pat-
described?”), and is scored as “1” if the feature is present, and terns observed in the data (Ritchie and Spencer 1994, p.
“0” if it is not, or the rater is unable to determine. An overall 179–180).
score is also calculated. Since not all questions were applica- 3 Indexing: the researcher conceptualizes the emerging
ble to all articles (e.g., random allocation for phenomenolog- themes from the previous steps in broader categories
ical studies), percentage scores with only relevant questions (e.g., parent outcomes, child outcomes, etc.) and creates
were calculated for each subscale (reporting, external validity, a final list (index) of concepts.
internal validity, power, and overall score). For intervention 4 Charting: the researcher creates a chart with key concepts
reporting, the point was given if the intervention, as a whole, and corresponding examples from the data.
was sufficiently explained. However, most studies did not 5 Mapping, looking for associations and explanations: the
report the musical aspect of the intervention as has been sug- researcher creates visual representations of the findings
gested (Robb et al. 2011). Music intervention reporting is and looks for explaining patterns.
analyzed as a separate rubric in the “Results” section.
Quality assessments are summarized in Addendum (p. 1).

Data Analysis Results

Descriptive Summaries Study Information

Study information was summarized using descriptive methods Data were consolidated in tables, including a PRISMA chart
and is presented in Tables 1 and 2, and in Addendum (p. 2–4). (Fig. 2), PICOT (population, intervention, comparison,
Rev J Autism Dev Disord

Table 1 Study information

summary Study information Number of studies and description

Area (country) 6 Australia

3 USA
2 Canada
1 Italy
1 New Zealand
Discipline of PI 10 music therapy
1 psychology
1 ethnomusicologist
1 curriculum & learning
Discipline of co-authors 4 no co-authors
3 music therapy
1 psychology
1 speech pathology
1 ethnomusicology
Study design 2 case study
1 ethnography
4 phenomenology
1 single-case (multiple baseline design)
1 single-group time series
3 single-group pretest-posttest
1 randomized controlled trial
Number of participants 7 (6 music therapy studies)—with less than 15 participants
2 (2 music therapy studies)—between15 and 50 participants
3 (2 music therapy studies)—more than 50 participants
1 Not clearly reported

outcomes and time) information for interventions researchers was a music therapist). Importantly, several stud-
(Addendum, p. 2), and PROGRESS information (Place of ies and programs were not exclusively created for children
residence, Race/ethnicity, Occupation, Gender, Religion, with ASD, but rather are early intervention programs that
Education, Socioeconomic status, and Social Capital; included children with other disabilities (Nicholson et al.
Addendum, p. 3),2 as recommended by Gough and collabora- 2008; Williams et al. 2012; and Yang 2013). Please see
tors (2012a, b). Table 1 provides information on geographical Table 2 for further program characteristics.
area, discipline of principal investigator, discipline of co-au-
thors, study methods, and number of participants for all stud-
ies reviewed. Most studies were conducted in two countries: Parent-Mediated Interventions, Parent Coaching, and
Australia (46%, n = 6) and USA (23%, n = 3). Ten studies Professional Training
were directed by music therapists, and the rest by a psychol-
ogist, ethnomusicologist, or teacher. Four studies were per- Regarding questions 1 and 2 (parent training programs and
formed by single researchers. Research methodologies includ- therapist training for parent coaching), it was found that inter-
ed case studies (2), ethnography (1), phenomenological inqui- vention programs with music as an added component (e.g.,
ry (5), single-case study (1), single-group designs (3), and SCERTS model) seemed to be published and well-established,
randomized controlled trial (1). Most studies had small sample but only as parent-mediated interventions, not as music inter-
sizes (10 with less than 50 participants). ventions. Contrastingly, music intervention programs (Sing &
Notably, two of the studies with more than 50 participants Grow, Nicholson et al. 2008; Musical Bonds, Yang 2013) or
were music therapy studies (i.e., where the intervention was comprehensive programs with music therapy sessions (Turtle
designed by a music therapist, and at least one of the project, DiRenzo et al. 2015) were less researched. Further, ten
of the 13 studies were directed and implemented by music
2
Most studies did not have complete information. It is included when therapists. However, in only four studies, the parent coaching/
available. training was provided by a music therapist (Ayson 2011;
 Rev J Autism Dev Disord

Table 2 Characteristics of programs included in this review

Name (author, year) Study design Population included Intervention Parent training Main outcomes
characteristics: (indicates variables
1. Group/individual targeted by intervention,
sessions not findings. See
2. Setting Appendix 3 for findings)
3. Interventionist
4. MT program or
MT sessions within
comprehensive
program

Sing & Grow Single-group, Children with 1. Group Parents included and Parental responsiveness.
(Nicholson et al. 2008; pretest-posttest developmental disabilities 2. Early education coached in sessions. Parental self-efficacy.
Williams et al. Single-group, (including ASD), families centers Manual and CD Parental mental health.
2012) pretest-posttest in disadvantaged 3. Trained music provided for home Child’s social and
conditions, teen-age therapists as practice. communication skills
mothers providers No follow-up of skill (all self-reported and
4. Music therapy acquisition. therapist observation)
program
Musical Bonds Single-group, Children with 1. Dyad Parent explicitly trained. Parent and child physical
(Yang 2013) pretest-posttest developmental disabilities 2. Home Parent fidelity and verbal positive
(6 out of 9 with ASD) 3. Music therapist measured. responses.
4. Music therapy Follow-up measure. Parent ability, perception
program and comfort with
music.
Parental use of music at
follow-up.
Perceived benefits.
Family-Centred Music Case study, Children with ASD 1. Dyad Home sessions. Parent Child social and
Therapy phenomenology- 2. Home participated but not emotional
(Thompson 2012; and randomized 3. Music Therapist trained. functioning.
Thompson 2017; controlled trial 4. Music therapy Follow-up of parent Child social engagement.
Thompson et al. program perceptions, not skill Child vocal production
2013; Thompson acquisition. & understanding.
and McFerran 2015) Child engagement in MT
Parent attitudes towards
child.
Long-term perspectives
of MT.
Turtle Project Single-group, time Children with ASD 1. Group Parent participated but Cognitive abilities as
(DiRenzo et al. 2015) series design 2. Early education not trained. measured by Leiter-R
center (Roid and Miller
3. Music therapist 2002)
4. MT sessions in ADOS scores
comprehensive
program. Results
related to all the
program.
Music Therapy and Case study Children with ASD 1. Dyad Parent participated and Child social
SCERTS 2. Home was asked to communication
(Ayson 2011) 3. Music therapist implement and take Child emotional
4. MT sessions in data. Training not regulation
comprehensive described. Parent perspective
program. Results
related to MT
sessions only.
Vaiouli 2014 Single-case study Children with ASD 1. Dyad Parent participated but Attention to faces
2. Home not trained Responding joint
3. Student music attention
therapist Initiating joint attention
4. Music therapy
program
Music therapy sessions Phenomenology 1. Dyad Parent-child interaction
Rev J Autism Dev Disord

Table 2 (continued)

Name (author, year) Study design Population included Intervention Parent training Main outcomes
characteristics: (indicates variables
1. Group/individual targeted by intervention,
sessions not findings. See
2. Setting Appendix 3 for findings)
3. Interventionist
4. MT program or
MT sessions
within
comprehensive
program

(Jacquet 2011) Children with 2. Early education Parent participated but

developmental disabilities center not trained
3. Music therapist
4. Music therapy
sessions
Music sessions with Ethnography Children with ASD 1. Group Parent participated but Child emotional
parents (Bakan 2. Music school not trained wellbeing.
et al. 2008) 3. Child self-confidence.
Ethnomusicolog- Child social and cultural
ist agency.
4. Music Child “response-ability”.
experiences.
Professional teacher Phenomenology Child with ASD 1. Dyad Parent developed and Parenting skills.
using music with 2. Home provided “Music for child
own son 3. Parent who is a “intervention” due to learning” (e.g., toilet
(Osei 2009) professional pedagogical training, training, use of words,
educator but not musically attention
4. Music in daily trained. management).
activities

Nicholson et al. 2008; Williams et al. 2012; Yang 2013). Sing to use within more comprehensive programs (Ayson 2011). See
& Grow is the only music therapy program that requires train- Table 3 for further detail.
ing for implementation (Nicholson et al. 2008). SCERTS also Related to question 3 (systematic parent coaching), the data
requires training but, as mentioned, it is not a music intervention indicated that although all 13 articles had components of par-
in its original format. Instead, music therapists have adapted it ent-mediated/parent-led interventions, only five of them

Table 3 Parent training/coaching characteristics

Characteristics Number of studies/names Citations

Formal training 5 Ayson 2011; DiRenzo et al. 2015; Nicholson et al. 2008; Williams
et al. 2012; Yang 2013
Follow-up/monitoring 5 Same as above
Specific model/program? 5 Same as above
Which model/program? SCERTS Ayson 2011
Turtle Project DiRenzo et al. 2015
Sing & Grow (2) Nicholson et al. 2008; Williams et al. 2012
Musical Bonds Yang 2013
Who provided training? 4 music therapists (RMT or Ayson 2011; Nicholson et al. 2008; Williams et al. 2012; Yang 2013.
MT-BC) DiRenzo et al. 2015
1 psychologists
Training available in this model/program? Yes, for Sing & Grow, and Ayson 2011; Nicholson et al. 2008; Williams et al. 2012
SCERTS
Model/Program mentioned, but formal SCERTS (1 study) Bakan et al. 2008;
training not provided Family-centered music therapy Thompson 2012; Thompson 2017; Thompson et al. 2013; Thompson
(4 studies) and McFerran 2015
 Rev J Autism Dev Disord

provided formal parent training or coaching (as opposed to involvement in music interventions (Table 4): support for par-
within-sessions education or coaching). Formal training ents, better child outcomes, support for parent-child interac-
ranged from structured parent training sessions to songbooks tions, parent getting to know his/her child, parent gaining
and CDs to use at home (Table 3). Only two music programs skills and knowledge, parent skills considered and involved
had all the components of parent coaching (i.e., parent training in programming.
sessions, continuous monitoring, specific accountability mea- Regarding mapping and looking for associations and
sures, and parents responsible for implementing activities at explanations (last step in the framework analysis), the rela-
home): Sing & Grow (Nicholson et al. 2008; Williams et al. tionship between reasons to implement parent-mediated in-
2012) and Musical Bonds (Yang 2013). terventions and parent coaching styles was explored. The
six reasons that support the use of parent-mediated interven-
Definition of Parent-Mediated and Parent Coaching tions seemed to have different prominence in each training
style, yielding two different approaches to parent coaching:
A significant finding of this review was that no study provided (a) behaviorally based parent training—with more struc-
a clear definition of parent-mediated/parent-led interventions tured and directive strategies—and parent participation/
nor parent coaching (question 4). However, framework anal- collaborative approach—with less direction for participa-
ysis of the studies allowed us to extract a series of concepts tion, and parents considered clients within the parent-child
that relate to these terms and that authors used to justify parent dyad (see Table 5).

Table 4 Conceptual framework extracted from parent-led/mediated definitions

Concepts Examples Studies Citations

1. Support for parent management, (parental) emotional responses, respon- 4 Nicholson et al. 2008; Thompson 2017; Thompson and
parents sive to individual differences among children and families, McFerran 2015; Thompson 2012; Williams et al. 2012
inclusion of families in these experiences, provide social
networking opportunities, finding new ways to engage
their children
2. Better child support across all daily activities, differentiated according to 7 Ayson 2011; DiRenzo et al. 2015; Nicholson et al. 2008;
outcomes age of the child, new meaning to routine activities, open a Thompson 2017; Thompson et al. 2013; Thompson and
gate to the outside world for the child, children’s McFerran 2015; Williams et al. 2012.
developing competence, support the skill development of
the child, relevance of the goals for the child, stimulate
child development; beneficial to develop communication
and relationship skills
3. Support for interpersonal supports, communication and the relationship 9 Ayson 2011; DiRenzo et al. 2015; Jacquet 2011; Nicholson
parent-child of the child, quality of parent-child relationships, et al. 2008; Thompson 2017; Thompson et al. 2013;
interactions parent-child attachment; inclusion of the families in these Thompson and McFerran 2015; Vaiouli 2014; Williams
[music] experiences; increase positive parent-child inter- et al. 2012.
actions; rare opportunities for mutual enjoyment
4. Parent father’s experience of the influence of nature and music on 3 Osei 2009; Thompson 2012, Yang 2013
knowing one autistic’s learning experience; opportunities for
child better parents to experience what motivates the child; parents
engage in a wide range of sensorimotor experiences with
their child; use of affect, behavioral and developmental
matching; shared control
5. Parents music therapist is model and teacher first, then facilitator of 7 Jacquet 2011; Nicholson et al. 2008; Osei 2009; Thompson
gaining skills the parent-child interaction; training procedures; parents’ et al. 2013; Thompson 2012; Thompson and McFerran
and behavior associated with children’s developing 2015; Yang 2013.
knowledge competence, parent’s self-confidence in parenting skills;
encourage active participation of the parent; opportunities
for parents to develop skills to enhance child’s develop-
ment; responsive parents focus primarily on supporting
and encouraging their children to participate
6. Parent families are involved in the program; parents involved in 5 Ayson 2011; DiRenzo et al. 2015; Thompson et al. 2013;
expertise establishing goals for intervention; home-therapy; value Thompson 2012; Thompson and McFerran 2015.
involved collaboration between the parent and the therapist; practi-
tioners and families striving to work together in partner-
ship; improved communication between therapist and
parents.
Rev J Autism Dev Disord

Table 5 Parent coaching styles extracted from descriptions/definitions

Examples Citations

Behaviorally based parent-log sheets; 10 h. of training over 3 days, training Ayson 2011; Nicholson et al. 2008; Williams et al. 2012; Yang
parent training material, in-class lecture, case studies and role-play; behav- 2013.
ioral parent training principles, non-didactic behavioral
strategies such as demonstration, rehearsal, feedback and
praise; workshop sessions and training; highly structured
teaching style; rapid pacing of familiar and novel developing
activities, cuing, guided assistance, and fading of assistance,
redirection, multi-sensory stimuli, and manipulatives; be-
haviorally specific verbal suggestions, use of praise,
modeling, and positive reinforcement; given a kit with the
items needed...along with written instructions, brief training;
music-based parent education program to teach parents
Parent participation parent participation (no intentional training), meetings with Bakan et al. 2008; DiRenzo et al. 2015; Jacquet 2011;
and/or collabora- parents, group or individual counseling, experiential or Thompson 2012; Thompson 2017; Thompson et al. 2013;
tive approach therapeutic groups; opportunity to create musical heritages Thompson and McFerran 2015; Yang 2013.
that the family can cherish; external support to foster healthy
relationships and socio-emotional adaptation; ongoing con-
sultation; gentle negotiation in the relationship between
parent-therapist and child; collaborative approach to working
therapeutically with children; promoting parental
responsiveness, matching their children’s interests, playing
interactively and interpreting their intentions.

Outcomes, Measures, and Findings child, responsiveness, and positive interactions), increased so-
cial interaction and receptive communication in children, bet-
A similar framework analysis was performed for outcomes, ter parent-child relationships, and generalization of music ac-
measures, and findings (Question 6). Outcomes refers to the tivities or parenting skills to daily activities. Null results were
behavioral targets of the interventions, measures to the instru- reported in some studies regarding parent-perceived warmth
ments used to collect data on the outcomes, and findings to the and self-efficacy, parental perception of child behavioral prob-
actual results of the intervention. In turn, positive findings lems outside of treatment, child social engagement outside of
refer to results congruent with expectations, negative findings treatment, and child vocabulary production and understand-
indicate outcomes in the opposite direction of the ones expect- ing. Negative results were mainly related to attrition, and pa-
ed, and null findings are outcomes showing no change after rental added effort to implement the strategies at home. In
intervention. other words, two studies (Nicholson et al. 2008; Williams
Outcome data were labeled and grouped (e.g., parental ef- et al. 2012) had high attrition, which limits the internal validity
ficacy was used for all related concepts: self-efficacy, confi- of their results. Further, high parental effort needed to imple-
dence, sense of competence, attitude towards own parenting, ment the strategies at home, despite reported satisfaction,
etc.) Four salient categories (themes) were extracted: parent limits the social validity of these interventions.
outcomes, child outcomes, parent-child outcomes, and thera-
pist outcome. An index of each outcome category was then Quality Assessments
created, and the original data were coded with these catego-
ries. Table 6 indicates all outcomes targeted in the studies. These assessments (Addendum) indicate that the strongest
Measures were listed and categorized in six groups: published area for most of these studies was the reporting score (55%
scales/measures, ad hoc scales, behavioral observation (with average score, range 14 to 80%), whereas internal validity
clear operational definitions and coding manuals), informal (specifically, selection bias) was the weakest (26% average
observations, interviews, self-reports/self-reflections score, range 0 to 83%). As mentioned, the reporting score
(Table 7). was given if the intervention as a whole was well-reported.
The findings of the reviewed studies were categorized and The specific rubrics of music intervention reporting (Robb
mapped into the outcome index, dividing them in positive, et al. 2011) are described below. Regarding research design,
null, and negative results (see Appendix 3 for a only one study used comparison conditions and was random-
comprehensive listing of findings). The main positive results ized. A study that used bundled interventions did not report
across studies refer to increased parenting skills (acceptance of the number of participants that received a music intervention
 Rev J Autism Dev Disord

Table 6 Outcome index

Group Outcome category Outcome (as reported by authors) N of Citations

studies

Parents 1. Interactions with Positive/negative interactions; responsiveness, 4 Nicholson et al. 2008; Thompson and McFerran
child parenting skills, response to child, irritable 2015; Williams et al. 2012; Yang 2013.
parenting
2. Parental efficacy Self-efficacy, confidence with music, sense of 6 Nicholson et al. 2008; Osei 2009; Thompson 2012;
competence, attitude towards parenting, Thompson et al. 2014; Vaiouli 2014; Williams
engagement in home activities et al. 2012.
3. Mental health Mental health 2 Nicholson et al. 2008; Williams et al. 2012.
4. Benefits and Satisfaction, perceived benefits, perspective, 5 Ayson 2011; Osei 2009; Thompson and McFerran
satisfaction conceptual understanding 2015; Williams et al. 2012; Yang 2013.
5. Music Understanding and relevance of music, use of music 2 Osei 2009; Yang 2013.
three months later
Children 1. Social interaction Joint attention, social play, social communication, 8 Ayson 2011; Nicholson et al. 2008; Thompson
engagement, participation, responsiveness, 2012; Thompson et al. 2014; Thompson and
interest, reciprocity, social interaction outside of McFerran 2015; Vaiouli 2014; Williams et al.
treatment 2012; Yang 2013.
2. Emotional Emotional regulation, wellbeing, confidence, 5 Ayson 2011; Bakan et al. 2008; Nicholson et al.
regulation “response-ability”, positive and negative 2008; Thompson et al. 2013; Yang 2013.
behaviors
3. Cognitive abilities Cognitive abilities 1 DiRenzo et al. 2015.
4. ASD symptoms ASD symptoms, autism classification 1 DiRenzo et al. 2015.
5. Language and Nonverbal and verbal skills, vocabulary production 1 Thompson et al. 2013.
communication and understanding, expressive and receptive
language
6. Self-help skills Toilet training 1 Osei 2009.
Parent-child 1. Parent-child inter- Synchrony, communication between family 4 Thompson 2017; Thompson et al. 2014; Vaiouli
action members, parent-child relationships, communi- 2014; Yang 2013.
cation between dyad, quality of parent-child
interactions, family quality of life
Therapist 1. Role in relation to Social support, role as therapist 2 Jacquet 2011; Thompson and McFerran 2015.
dyad
2. Appropriateness Effective strategies, generalization, practitioner’s 1 Jacquet 2011
and effectiveness understanding and relevance of music
of music
interventions
3. Social validity Compliance, parental effort, social validity, parent 2 Nicholson et al. 2008; Yang 2013.
and staff perceived benefits
4. Professional Perceptions from other professionals 1 Ayson 2011.
interactions

(DiRenzo et al. 2015). Therefore, the ability to affirm that the were least reported referred to music structure (either pub-
outcomes were due to the music intervention is compromised lished music being provided, or original/improvisational
in most studies. music being described) and music materials (n = 1, 15%
and n = 4, 31% of studies, respectively). Procedures and
Music Intervention Reporting session formats were described in several studies, but only
Yang (2013) provided enough specificity to make those
Characteristics of music interventions, when available, sessions replicable. Fidelity measures of treatment imple-
were compiled in a spreadsheet. A summary of this infor- mentation were reported in 31% (n = 4) of the studies, and
mation is reported in Addendum (p. 5). The components of only 2 studies with music therapists as principal investiga-
the music intervention that were best described by most tors reported the use of a complete, manualized procedure
studies (n = 12, 92% of the studies) refer to intervention (i.e., written set of steps, activities, and session structure) to
strategies (i.e., improvisation, songwriting, music-assisted ensure fidelity (Nicholson et al. 2008; Williams et al. 2012).
relaxation, etc.) and setting (i.e., community, clinic, and Notably, both studies referred to the same music therapy
client’s home; n = 9, 69% of the studies). The aspects that program: Sing & Grow.
Rev J Autism Dev Disord

Table 7 Measures used in studies reviewed

Citation Published scales Ad hoc scales/ Behavioral Informal Interviews/ Self-

result observation observation Survey reports

Ayson 2011. SCERTS X

Bakan et al. 2008. X X
DiRenzo et al. 2015. ADOS; Leiter-R
Jacquet 2011. X
Nicholson et al. 2008 CRQ; ECLS-BC; K-6; NEILS; PPBS. X
Osei 2009. X
Thompson 2012 X
Thompson et al. 2014 MBCDI-W&G; MTDA; PCI; X
SRS-PS; VSEEC.
Thompson 2017 X
Thompson and X X
McFerran 2015
Vaiouli 2014 X X X
Williams et al. 2012 CQR; ECLS-BC; NEILS; PPBS. Posttest X
questionnaire
Yang 2013 PUMP; feedback X X X
sheet
TOTAL STUDIES 6 3 4 3 7 4

Published scales: ADOS Autism Diagnostic Observation Schedule (Gotham et al. 2008); Leiter-R (Roid and Miller 2002), CRQ Child Rearing
Questionnaire (Paterson and Sanson 1999), ECLS-BC Early Childhood Longitudinal Study-Birth Cohort (National Center for Education Statistics
2004), K-6 (Furukawa et al. 2003), NEILS NEILS Scales of Developmental Competency (SRI International 2003), PPBS Parental Perceptions and
Behavior Scales (Institut de la Statistique du Quebec 2000), MBCDI-W&G MacArthur-Bates Communication Development Inventories-Words &
Gestures (Fenson et al. 2007), MTDA Music therapy diagnostic assessment (Oldfield 2006), PCI Parent-child inventory (Gerard 2005), SRS-PS
Social Responsiveness Scale-PreSchool (Constantino and Gruber 2005), VSEEC Vineland Social-emotional Early Childhood Scale (Sparrow et al.
1998)
Ad hoc scales: * effect size for parent-child interaction, Nicholson et al. 2008. PUMP = Parental Use of Music and Play

Discussion sessions (Nicholson et al. 2008; Williams et al. 2012; Yang

2013). However, these distinctions are not clear-cut, since all
The purpose of this systematic review was to explore the state structured programs also used tailoring of the music to family
of published research in parent-mediated music interventions needs, creating interventions based on child interests, and
with children with autism spectrum disorder (ASD). The guid- using improvisation as therapeutic strategies. Of these pro-
ing questions referred to the existence of parent-led/mediated grams, only Sing & Grow had complete, manualized fidelity
music interventions (question 1), interventionists’ training to measures of implementation and training processes (Question
coach parents (question 2), existence of systematic parent 3). Importantly, Sing & Grow and Musical Bonds were creat-
training (question 3), definitions of parent-led/mediated inter- ed as early intervention programs and were not designed for
ventions and definitions of parent coaching (question 4), out- children with ASD exclusively (although all studies included
comes and measures used in these studies (question 5) and at least 15% of children with ASD in their samples).
findings of such interventions (question 6). No study provided a clear definition of “parent-mediated
As expected after the scoping searches, an incipient body interventions” or “parent coaching” (Question 4). Notably,
of parent-mediated music interventions was evident (Question only the Family-Centred approach (Thompson 2012) present-
1). Within the music therapy field, three programs were found: ed a conceptual framework for the inclusion of parents in
Sing & Grow (Nicholson et al. 2008; Williams et al. 2012), therapy. Nonetheless, relevant quotes were found in each
Musical Bonds, (Yang 2013), and Family-Centred music ther- study’s literature review or intervention description. A frame-
apy (Thompson 2012; Thompson et al. 2013; Thompson and work analysis of these quotes showed that parent-mediated
McFerran 2015). These programs have different intervention interventions are considered important in the treatment of chil-
strategies, broadly categorized as (a) mainly improvisational dren with ASD when music is involved. The six main reasons
(Thompson 2012) and (b) mainly structured, predefined for including the parents in the sessions were (1) emotional
 Rev J Autism Dev Disord

support for parents, (2) better child outcomes, (3) support for et al. 2012). An important finding of this review relates to
parent-child interactions, (4) parents getting to know their the wide variety of outcomes measured in the studies, which
child better, (5) parents learning new skills and knowledge limits replication of results and comparisons across studies.
of ASD, and (6) inclusion of parental expertise in treatment. This finding is consistent with non-music parent-mediated
These reasons are in line with previous intervention reviews literature in ASD (Kuhaneck et al. 2015; Noyes-Grosser
and best practices in ASD that have found that parent involve- et al. 2013). Future empirical and theoretical research should
ment in therapy promotes child development and parental strive to identify the most meaningful outcomes for parent-
wellbeing and satisfaction (Oono et al. 2013; Zwaigenbaum mediated music interventions.
et al. 2015).
Regarding parent coaching (Question 4), two main styles Quality of the Evidence
were extracted from the intervention descriptions: behavioral-
ly based training and collaborative approach. Interestingly, Quality assessments were performed only to determine the
Thompson and McFerran (2015) mentioned that “in contrast state of the research, and not as an exclusion criterion for this
with parent training programmes (Vismara et al. 2009), where review. These assessments indicate that the strongest area was
professionals teach parents a prescribed set of skills, family- intervention reporting, whereas selection bias was the greatest
centred approach is a collaborative approach to working ther- threat to the internal validity. It should be noted that the
apeutically with children.” (p. 4). However, more recent pub- reporting score only includes intervention reporting as a
lications on the non-music model referenced by Thompson whole, and not music intervention reporting (Robb et al.
and McFerran (i.e., Parent-Early Start Denver Model, P- 2011). In fact, music intervention reporting seemed less than
ESDM, Estes et al. 2014) indicate that the P-ESDM does, in satisfactory: the most reported aspect was intervention strate-
fact, consider a collaborative approach for goal-setting, and gies (92% of the studies), whereas music descriptions (pub-
utilizes adult learning and coaching—where the parent’s ex- lished music provided or improvisation described) and mate-
pertise is incorporated—as principles for training. Also, as rials were significantly lacking (15% and 31% of the studies,
mentioned, all the behaviorally based music programs respectively). Fidelity measures of treatment implementation
reviewed included components of tailoring and improvisation were used in only 31% (n = 4) of the studies.
within the sessions. The underlying theoretical differences, Another limitation is the variability within the studies
therefore, might not be as stark as apparent at first sight. reviewed. Outside of the music therapy field (where the prin-
The most frequently used measures (Question 5) were cipal investigator was not a music therapist), music was
interviews/surveys (seven studies). Notably, only four music assigned different levels of importance. For example, Osei
therapy studies (where the principal investigator was a music (2009) considered music an essential component of the child’s
therapist) used published scales. This situation is unsurprising, learning, whereas DiRenzo and collaborators (2015) included
considering that most music therapy studies (n = 8 of 13) had music therapy as part of a bundled intervention. Further, rigor
single-subject or qualitative study designs, where the use of and training varied among studies. Osei (2009) conducted an
published standardized scales or parametric statistical analy- ethnography of his personal experience with his child with
ses might prove inadequate. On the other hand, the frequent autism; his spontaneous discovery of music as a teaching tool
use of behavioral observations and self-reports (n = 4 studies was uninformed by previous training. Contrastingly,
each, Table 7) seemed an appropriate alternative for feasibility Nicholson et al. (2008) and Williams et al. (2012) reported
and limited-efficacy studies, which some of these studies in- on a multisite, manualized intervention with many participants
deed are (Ayson 2011; Thompson and McFerran 2015; and trained music therapists as interventionists. However, not
Thompson 2017; Vaiouli 2014; Yang 2013). all participants had an ASD diagnosis (15%, Nicholson et al.
Findings of the reviewed articles were categorized in parent 2008; Williams et al. 2012). This finding is reasonable given
outcomes, child outcomes, parent-child outcomes, and that most of these studies were early intervention studies, not
therapist/intervention outcomes (Question 5 and 6). Similar interventions for children with ASD, specifically. However,
to reviews of non-music parent-mediated interventions mixed samples, along with the variability in the amount and
(Oono et al. 2013), this review found that positive results use of music within the studies, make comparisons and overall
referred to increased parenting skills, increased child social conclusions difficult.
interaction and receptive communication, better parent-child
relationships, and generalization of (music) activities and Limitations
skills to daily life (see Appendix 3 for a complete list of
results/findings). On the downside, some of these interven- One limitation of this review relates to the inclusion of peer-
tions seemed to require significant parental effort to imple- reviewed and dissertation literature only. As mentioned, this
ment at home and produced high attrition, despite reported choice was made to ensure a minimum quality of the research
high parental satisfaction (Nicholson et al. 2008; Williams addressed, since understanding the state of the research of
Rev J Autism Dev Disord

parent-mediated interventions (and not intervention efficacy) established for this investigation in light of current research in
was our main goal. Further, strategies to address bias within ASD that indicates that parent-mediated interventions are a
these limitations have been proposed (Boland and Cherry viable alternative when professional services are scarce. On
2014) and were included in this review: circumscribed and the other hand, these interventions might require excessive
clearly defined research questions, consultation with special- parental effort to implement at home. Interventionists might
ized librarian to define a well-constructed search strategy, a consider developing and investigating these interventions,
priori selection of databases, predefined inclusion/exclusion while being mindful of parental availability and resources.
criteria, conceptual and operational definitions of constructs,
documentation of search strategies and number of citations, Recommendations for Future Research
rationale for decision-making at each step, and rigorous data
management. The use of all these strategies allows confidence Consistent with previous reviews (Burns 2012; Geretsegger
that most relevant literature was included and handled et al. 2014; Kim and Stegemann 2016; Silva et al. 2016;
appropriately. Silverman, et al. 2016), this study indicates the need of music
intervention reporting with greater detail in procedures, music
Implications for Practice descriptions, and materials, to make the interventions replica-
ble. Additionally, including fidelity measures of treatment im-
Literature not eligible for this review (e.g., Jacobsen and plementation could support valid results and enable profes-
Thompson 2016; Oldfield and Flower 2008; Strange et al. sional training on effective models. From a family systems
2016; Warren and Nugent 2010) indicates that music thera- perspective, a report of complete demographic information
pists have included families as significant elements of therapy (such as a PROGRESS report, see Addendum, p. 3) would
for a long time. However, specific components that distin- help readers understand multiple factors that influence these
guish parent-mediated music interventions are the inclusion interventions and their potential for generalization.
of formal parent coaching/training and monitoring, account- Research of complex interventions benefits from a step-
ability measures, and parents as co-therapist, and not only as wise approach where feasibility and pilot studies of limited
participants in the sessions. Parent coaching, in turn, is the efficacy are sequenced and followed up with larger-scale ef-
formalized education that professionals provide to ensure fi- fectiveness studies (Robb 2013). Clarification of conceptual
delity of parent-mediated interventions (Steiner et al. 2012). frameworks when researching parent-mediated music inter-
Both parent-mediated interventions and parent coaching are at ventions is also needed to allow informed comparisons be-
a beginning stage in music therapy, according to this study. tween programs. Further, exploration of multi-layered models
This review adds to the existing evidence of positive results that include observed and latent measures of psychosocial
when music interventions are designed for individuals with measures (e.g., parental perceptions, parent-child bonding, so-
ASD (Bhat 2013; Fusar-Poli et al. ongoing; Geretsegger cial interaction), individual measures (e.g., child language and
et al. 2014; and Simpson and Keen 2011). This review intro- communication), and music psychological measures (e.g.,
duces a novel intervention model and concepts (parent- arousal and sensory responses to music) might prove interest-
mediated intervention and parent coaching) that seem to be ing and useful. In this case, this systematic review of parent-
an emerging area of interest and that may have critical impor- mediated music interventions is only the first step in this step-
tance when access to services is limited. Clinicians are encour- wise process; its main goal was to understand the current state
aged to consider this mode of intervention as an effective of research of parent-mediated music interventions with chil-
support for families, particularly early after diagnosis, when dren with ASD. Future steps should include theory develop-
intervention may be most impactful (Estes et al. 2014), or in ment that supports these interventions, and empirical research
situations where parents are necessary intermediaries of the to improve such theories.
therapy, such as in telehealth settings. Strategies that are dis-
tinct to parent-mediated interventions include incorporating
parents in every step of the intervention plan (particularly in Conclusions
selecting meaningful outcomes), providing coaching and
monitoring throughout the program, and evaluating not only Findings of this systematic review indicate that parent-
child outcomes, but also parental outcomes (Zwaigenbaum mediated music interventions with children with ASD are in-
et al. 2015). Considering that working with family systems creasingly researched as a valuable mode of intervention,
is more complex than working with a single child with mainly within the last decade (2007–2017). The quality of
ASD, advanced training in parent coaching is also advised. the evidence is limited due to atheoretical presentations of
It should be noted that there is no implication that parent- interventions, incomplete music reporting, scarcity of fidelity
mediated interventions are better than family music therapy measures, inclusion of non-ASD populations within the stud-
(i.e., parents as participants). Instead, a clear distinction was ies, and varied outcome measures. However, important
 Rev J Autism Dev Disord

findings include the feasibility of using music within parent- Campbell Collaboration. (2011d). Problem formulation, Jeffrey C.
Valentine. [Video file]. Retrieved from https://www.
mediated interventions, their potential to support parent and
campbellcollaboration.org/the-introductory-methods.html.
child outcomes, their alignment with best practices in ASD Constantino, J. N., & Gruber, C. P. (2005). The Social Responsiveness
treatment, and social validity (high parental satisfaction) of Scale. Torrance, CA, USA: Western Psychological Services.
music as an intervention for all family members. A next step Coolican, J., Smith, I. M., & Bryson, S. E. (2010). Brief parent training in
pivotal response treatment for preschoolers with autism. Journal of
could be the creation and evaluation of conceptual frame-
Child Psychology and Psychiatry, 51, 1321–1330.
works that explicitly state constructs and hypothesized rela- Deeks, J., Dinnes, J., D'Amico, R., Sowden, A., Sakarovitch, C., Song, F.,
tionships. This theoretical approach may allow the investiga- . . . Altman, D. (2003). Evaluating non-randomised intervention
tion of the role of music within parent-mediated interventions studies. Health Technology Assessment (Winchester, England),
7(27), iii-x, 1-173.
for children with ASD and may improve intervention design
DiRenzo, M., Di Castelbianco, F., Petrillo, M., Racinaro, L., & Rea, M.
(Burns 2012). (2015). Assessment of a long-term developmental relationship-
based approach in children with autism spectrum disorder.
Acknowledgements The present manuscript is part of the author’s dis- Psychological Reports, 117(1), 26–49.
sertation presented in partial fulfillment of a doctoral degree at the Downs, S., & Black, N. (1998). The feasibility of creating a checklist for
University of Kansas. The author wishes to thank her dissertation com- the assessment of the methodological quality both of randomised
mittee, Dr. Christopher M. Johnson, Dr. Hanson-Abromeit, Dr. Cindy and non-randomised studies of health care interventions. Journal
Colwell, Dr. Jim Daugherty, and Dr. Michael Roberts, for their support, of Epidemiology and Community Health, 52(6), 377.
as well as Stephen Chavez, Jae Young Jung, and Sophie Lott, graduate Estes, A., Vismara, L., Mercado, C., Fitzpatrick, A., Elder, L., Greenson,
students at Arizona State University, for their help in updating this J., et al. (2014). The impact of parent-delivered intervention on
review. parents of very young children with autism. Journal of Autism and
Developmental Disorders, 44(2), 353–365.
Funding Information None declared. Fenson, L., Marchman, V., Thal, D., Dale, P. S., Reznick, J. S., & Bates,
E. (2007). MacArthur-Bates Communicative Development
Inventories: User’s Guide and Technical Manual (2nd ed.).
Compliance with Ethical Standards Baltimore: Paul H. Brookes Publishing Co.
Furukawa, T. A., Kessler, R. C., Slade, T., & Andrews, G. (2003). The
Conflict of Interest The author declares no conflict of interest. performance of the K6 and K10 screening scales for psychological
distress in the Australian National Survey of Mental Health and
Well-Being. Psychological Medicine, 33, 357–362.
Fusar-Poli, L., Gattino, G.S., Carpente, J.A, & Suvini, F. (ongoing).
References Music therapy for autism spectrum disorders (ASD): A systematic
review of instruments used for evaluation of outcome. PROSPERO
American Music Therapy Association (AMTA), (2018). 2018 Member 2016: CRD42016051985. Retrieved from http://www.crd.york.ac.
Survey and Workforce Analysis. https://www.musictherapy.org/ uk/PROSPERO/display_record.asp?ID=CRD42016051985.
assets/1/7/18WorkforceAnalysis.pdf. Gerard, A. B. (2005). Parent-Child Relationship Inventory (PCRI)
Ayson, C. (2011). The use of music therapy to support the SCERTS Manual (4th ed.). Torrance, CA, USA: Western Psychological
model objectives for a three-year old boy with autism spectrum Services.
disorder in New Zealand. New Zealand Journal of Music Therapy, Geretsegger, M., Elefant, C., Messier, K., & Gold, C. (2014). Music
9, 7–31. therapy for people with autism spectrum disorder. The Cochrane
Bakan, M. B., Koen, B. D., Bakan, M., Kobylarz, F., Morgan, L., Goff, Database of Systematic Reviews, 6, CD004381.
R., & Kahn, S. (2008). Saying something else: Improvisation and Gotham, K., Risi, S., Dawson, G., Tager-Flusberg, H., Joseph, R., Carter,
music-play facilitation in a medical ethnomusicology program for A., & Sigman, M. (2008). A replication of the Autism Diagnostic
children on the autism spectrum. College Music Symposium, 48, 11– Observation Schedule (ADOS) revised algorithms. Journal of the
30. American Academy of Child & Adolescent Psychiatry, 47(6), 642–
651.
Bhat, A. N. & Srinivasan, S. M. (2013). A review of “music and move-
Gough, D., Oliver, S., & Thomas, J. (Eds.). (2012a). An introduction to
ment” therapies for children with autism: Embodied interventions
systematic reviews. Kindle edition: Sage.
for multisystem development. Frontiers in Integrative
Gough, D., Thomas, J., & Oliver, S. (2012b). Clarifying differences be-
Neuroscience, 7. https://doi.org/10.3389/fnint.2013.00022.
tween review designs and methods. Systematic Reviews, 1(28).
Boland, A., & Cherry, M. G. (Eds.). (2014). Doing a systematic review: A https://doi.org/10.1186/2046-4053-1-28.
student's guide. Kindle edition: Sage. Hernandez-Ruiz, E. (2019) Feasibility of parent coaching of music inter-
Burns, D. (2012). Theoretical rationale for music selection in oncology ventions for children with autism spectrum disorder. Music Therapy
intervention research: An integrative review. Journal of Music Perspectives. Advance online article. https://doi.org/10.1093/mtp/
Therapy, 49(1), 7–22. miz016.
Campbell Collaboration. (2011a). Coding, Sandra Wilson. [Video file]. Hernandez-Ruiz, E. (2020). Parent coaching of music interventions for
Retrieved from https://www.campbellcollaboration.org/the- children with ASD: A conceptual framework. Nordic Journal of
introductory-methods.html. Music Therapy, 29(3), 200–221.
Campbell Collaboration. (2011b). Literature search, Karianne Thune Ingersoll, B., & Wainer, A. (2013). Initial efficacy of project ImPACT: A
Hammerstrøm. [Video file]. Retrieved from https://www. parent-mediated social communication intervention for young chil-
campbellcollaboration.org/the-introductory-methods.html. dren with ASD. Journal of Autism and Developmental Disorders,
Campbell Collaboration. (2011c). Problem formulation, Aron Shlonsky. 43, 2943–2959. https://doi.org/10.1007/s10803-013-1840-9.
[Video file]. Retrieved from https://www.campbellcollaboration. Institut de la Statistique du Quebec. (2000). Longitudinal Study of Child
org/the-introductory-methods.html. Development in Quebec (ELDEQ 1998–2002): 5-month-old infants,
Rev J Autism Dev Disord

parenting and family relations, vol. 1, number 10. Quebec, Canada: Pickles, A., Le Couteur, A., Leadbitter, K., Salomone, E., Cole-Fletcher,
Institut de la Statistique du Quebec. R., Tobin, H., et al. (2016). Parent-mediated social communication
Jacobsen, S. T., & Thompson, G. (Eds.). (2016). Music therapy with therapy for young children with autism (PACT): Long-term follow-
families: Therapeutic approaches and theoretical perspectives. up of a randomised controlled trial. Lancet, 388(10059), 2501–
London: Jessica Kingsley Publishers. 2509.
Jacquet, C. (2011). Music therapy and pediatrics: Impact on the parent- Poslawsky, I. E., Naber, F. B. A., Bakermans-Kranenburg, M. J., De
child relationship. Canadian Journal of Music Therapy, 17(1), 95– Jonge, M. V., Engeland, H. V., & Van IJzendoorn, M. H. (2014).
103. Development of a video-feedback intervention to promote positive
Kasari, C., Gulsrud, A., Paparella, T., Hellemann, G., Berry, K., & Nezu, parenting for children with autism (VIPP-AUTI). Attachment &
A. M. (2015). Randomized comparative efficacy study of parent- Human Development, 16, 343–355.
mediated interventions for toddlers with autism. Journal of Ritchie, J., & Spencer, L. (1994). Qualitative data analysis for applied
Consulting and Clinical Psychology, 83(3), 554–563. policy research. In A. Bryman & R. G. Burgess (Eds.), Analyzing
Kern, P., & Humpal, M. (Eds.). (2018). Early childhood music therapy qualitative data. New York: Routledge.
and autism spectrum disorders: Developing potential in young chil- Robb, S. L. (2013). The power of the pilot. Journal of Music Therapy,
dren and their families (2nd ed.). Jessica Kingsley Publishers. 50(1), 3–5.
Kim, J., & Stegemann, T. (2016). Music listening for children and ado- Robb, S. L., Carpenter, J. S., & Burns, D. S. (2011). Reporting guidelines
lescents in health care contexts: A systematic review. The Arts in for music-based interventions. Journal of Health Psychology, 16(2),
Psychotherapy, 51, 72–85. 342–393.
Kuhaneck, H. M., Madonna, S., Novak, A., & Pearson, E. (2015). Roid, G. H., & Miller, L. J. (2002). Leiter-R: Leiter international perfor-
Effectiveness of interventions for children with autism spectrum mance scale-revised. Stoelting.
disorder and their parents: A systematic review of family outcomes. Siller, M., Swanson, M., Gerber, A., Hutman, T., & Sigman, M. (2014).
American Journal of Occupational Therapy, 69(5), 1–14. A parent-mediated intervention that targets responsive parental be-
Lucyshyn, J. M., Fosset, B., Bakeman, R., Cheremshynski, C., Miller, L., haviors increases attachment behaviors in children with ASD:
Lohrman, S., Binnendyk, L., Khan, S., Chinn, S., Kwon, S., Irvin, Results from a randomized clinical trial. Journal of Autism and
L., & K. (2015). Transforming parent-child interaction in family Developmental Disorders, 44(7), 1720–1732.
routines: Longitudinal analysis with families of children with devel- Silva, L., Baran, F., & Mercês, N. (2016). Music in the care of children
opmental disabilities. Journal of Child and Family Studies, 24, and adolescents with cancer: Integrative review. Texto & Contexto -
3526–3541. Enfermagem, 25(4), E1720015.
May, C., Fletcher, R., Dempsey, I., & Newman, L. (2015). Modeling Silverman, Letwin, & Nuehring. (2016). Patient preferred live music with
relations among co-parenting quality, autism-specific parenting adult medical patients: A systematic review to determine implica-
self-efficacy, and parenting stress in mothers and fathers of children tions for clinical practice and future research. The Arts in
with ASD. Parenting, 15(2), 119–133. Psychotherapy, 49, 1–7.
National Center for Education Statistics. (2004). Early Childhood Simpson, K., & Keen, D. (2011). Music interventions for children with
Longitudinal Study, Birth Cohort 9-Month restricted-use data files autism: Narrative review of the literature. Journal of Autism and
user's manual. NCES 2004–093. Washington, DC: NCES Developmental Disorders, 41(11), 1507–1514.
Neurodevelopmental disorder. DSM- 5 (2013). Retrieved from http:// Sparrow, S., Balla, D., & Cicchetti, D. (1998). Vineland Social-
dsm.psychiatryonline.org/doi/abs/10.1176/appi.books. Emotional Early Childhood Scales Manual. Circle Pines, MN,
9780890425596.dsm01. USA: American Guidance Service.
Nicholson, J., Berthelsen, D., Abad, V., Williams, K., & Bradley, J. Steiner, A. M., Koegel, L. K., Koegel, R. L., & Ence, W. A. (2012).
(2008). Impact of music therapy to promote positive parenting and Issues and theoretical constructs regarding parent education for au-
child development. Journal of Health Psychology, 13(2), 226–238. tism spectrum disorders. Journal of Autism and Developmental
Noyes-Grosser, D. M., Rosas, S. R., Goldman, A., Elbaum, B., Disorders, 42(6), 1218–1227.
Romanczyk, R., & Callahan, E. H. (2013). Conceptualizing child Strange, J., Odell-Miller, H., & Richards, E. (2016). Collaboration and
and family outcomes of early intervention services for children with assistance in music therapy practice: Roles, relationships,
ASD and their families. Journal of Early Intervention, 35(4), 332– challenges. London: Jessica Kingsley.
354. Strauss, K., Mancini, F., SPC Group, & Fava, L. (2013). Parent inclusion
Nunes, D. R., Araújo, E. R., Walter, E., Soares, R., & Mendonça, C. in early intensive behavior interventions for young children with
(2016). Augmenting caregiver responsiveness: An intervention pro- ASD: A synthesis of meta-analyses from 2009 to 2011. Research
posal for youngsters with autism in Brazil. Early Childhood in Developmental Disabilities, 34(9), 2967–2985.
Education Journal, 44(1), 39–49. SRI International. (2003). The National Early Intervention Longitudinal
Oldfield, A. (2006). Interactive Music Therapy in Child and Family Study (NEILS): Data collection. http://sri.com/neils/datacollect.
Psychiatry: Clinical Practice. Research and Teaching: Jessica html.
Kingsley Publishers, London, UK. Thompson, G. (2012). Family-centered music therapy in the home envi-
Oldfield, A., & Flower, C. (2008). Music therapy with children and their ronment: Promoting interpersonal engagement between children
families. Philadelphia: J. Kingsley Publisher. with autism spectrum disorder and their parents. Music Therapy
Oono, I., Honey, E., & Mcconachie, H. (2013). Parent-mediated early Perspectives, 30(20), 109–116.
intervention for young children with autism spectrum disorders Thompson, G. (2017). Long-term perspectives of family quality of life
(ASD). Cochrane Database of Systematic Reviews, (4), Cochrane following music therapy with young children on the autism spec-
Database of Systematic Reviews, 2013, Issue 4. trum: A phenomenological study. Journal of Music Therapy, 54(4),
Osei, A. (2009). Beginning with Brandon's interest: The experience of the 432–459.
influence of nature and music on one autistic student's learning. Thompson, G., & McFerran, K. S. (2015). "We've got a special connec-
(Unpublished doctoral dissertation). University of Toronto, tion": Qualitative analysis of descriptions of change in the parent-
Toronto, Ontario, Canada, 1–211. child relationship by mothers of young children with autism spec-
Paterson, G., & Sanson, A. (1999). The association of behavioural ad- trum disorder. Nordic Journal of Music Therapy, 24(1), 3–26.
justment to temperament, parenting and family characteristics Thompson, G. A., McFerran, K. S., & Gold, C. (2014). Family‐centred
among 5-year-old children. Social Development, 8(3), 293–309. music therapy to promote social engagement in young children with
 Rev J Autism Dev Disord

severe autism spectrum disorder: A randomized controlled study. Yang, Y-H. (2013). Parents and young children with disabilities: The
Child: Care, Health and Development, 40(6), 840–852. effects of home-based parent education in music on parent-child
Vaiouli, P. (2014). Joint engagement for toddlers at risk with autism: A interactions. (unpublished doctoral dissertation). Florida State
family, music-therapy intervention. Music Therapy Perspectives, University, Tallahassee, Florida, USA, pp 1–132.
32(11), 193. Yang, Y.-H. (2016). Parents and young children with disabilities: The
Vismara, L. A., Colombi, C., & Rogers, S. J. (2009). Can one hour per effects of a home-based music therapy program on parent-child
week of therapy lead to lasting changes in young children with interactions. Journal of Music Therapy, 53(1), 27–54.
autism? Autism, 13(1), 93–115. Zwaigenbaum, L., Bauman, M., Choueiri, R., Kasari, C., Carter, A.,
Warren, P., & Nugent, N. (2010). The "music connections" programme: Granpeesheh, D., et al. (2015). Early intervention for children with
Parents' perceptions of their children's involvement in music thera- autism spectrum disorder under 3 years of age: Recommendations
py. The New Zealand Journal of Music Therapy, 8, 8–33. for practice and research. Pediatrics, 136(4), s60–s81.
Williams, K. E., Berthelsen, D., Nicholson, J. M., Walker, S., & Abad, V.
(2012). The effectiveness of a short-term group music therapy inter-
Publisher’s Note Springer Nature remains neutral with regard to jurisdic-
vention for parents who have a child with a disability. Journal of
tional claims in published maps and institutional affiliations.
Music Therapy, 49(1), 23–44.