Marquez-Garcia - 2021 - Music Therapy in Autism Spectrum Disorder, A Systematic Review

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Music Therapy in Autism Spectrum Disorder: a Systematic Review

Article  in  Review Journal of Autism and Developmental Disorders · March 2022


DOI: 10.1007/s40489-021-00246-x

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Review Journal of Autism and Developmental Disorders
https://doi.org/10.1007/s40489-021-00246-x

REVIEW PAPER

Music Therapy in Autism Spectrum Disorder: a Systematic Review


Amparo V. Marquez-Garcia 1 & Justine Magnuson 1 & James Morris 2 & Grace Iarocci 3 & Sam Doesburg 1 &
Sylvain Moreno 4

Received: 11 February 2019 / Accepted: 6 February 2021


# Crown 2021

Abstract
Individuals with autism spectrum disorder (ASD) can experience difficulties functioning in society due to social communication
deficits and restrictive and repetitive behaviors. Music therapy has been suggested as a potential intervention used to improve these
deficits in ASD. The current systematic literature review focuses on two methods of music therapy: improvisational music therapy
(IMT) and singing/listening to songs. We review the extant literature and the associated methodological limitations, and we propose a
framework to assess the effectiveness of music therapy as an intervention in ASD. We suggest the creation of a standardized framework
that should utilize neuroimaging tools as an objective marker of changes induced by music therapy as well as a combination of
functional and behaviourial outputs, rather than assessment methods addressing a broad range of functional and behavioural outputs,
rather than only the main symptoms. The methodological limitations found in the current literature prevent us from making a strong
statement about the effects of music therapy in autism. We consider treatment fidelity assessments as the key to successful future
attempts to truly understand music therapy effects in ASD.

Keywords Evidence-based practice . Focused intervention . Autism spectrum disorder . Music therapy . Review . Neuroimaging

Introduction tools, and therapies that can be selectively utilized to address the
diverse support needs of individuals with ASD.
The two main symptoms that define autism spectrum disorder Music therapy has employed a wide range of methods and
(ASD), according to the Diagnostic and Statistical Manual of techniques to support individuals with ASD (Kaplan and Steele
Mental Disorders (DSM V), are deficits in social communica- 2005; Reschke-Hernández 2011; Walworth 2007). These
tion, and repetitive behaviors/restricted interests (Edition 2013). methods are characterized by systematic procedures that specify
Hypo- or hyperactivity to sensory input or unusual interests in the particular way the technique should be executed or per-
sensory aspects of the environment are also common in the ASD formed. The effectiveness of these methods and techniques, spe-
population (Edition 2013). Although there are many behavioral cifically in improving function in certain symptom domains, is
and educational therapies that are helpful in supporting individ- not fully understood. According to the American Music Therapy
uals with ASD, no single therapy fully addresses the needs of all Association (2015), music therapy is used to address the physi-
people with ASD. Given the heterogeneity within the autism cal, emotional, cognitive, and social needs of individuals through
spectrum, it is necessary to develop a range of assistive devices, treatments such as creating, singing, moving to, or listening to
music. In this study, we performed a systematic review to inves-
tigate the empirical evidence of improvisational music therapy
* Amparo V. Marquez-Garcia (IMT) and singing/listening to songs on improving autism symp-
amarquez@sfu.ca toms. We then discuss the strengths and limitations in assessing
the effectiveness of these music therapy methods. To conclude,
1
Department of Biomedical Physiology and Kinesiology, Simon we discuss the need for further research on the effects of well-
Fraser University, Burnaby, BC, Canada defined music therapy methods in ASD.
2
School of Medicine, University College Dublin, Dublin, Ireland
3
Department of Psychology, Simon Fraser University, Music Therapy in ASD
Burnaby, Canada
4
Department of School of Interactive Art and Technology, Simon Music therapy is an evidence-based intervention wherein mu-
Fraser University, Surrey, Canada sic is utilized via a therapist-patient relationship to promote
Rev J Autism Dev Disord

individualized goals (Bruscia 1998). Music therapy, or music Table 1 Specific music therapies, their methodological descriptions,
and studies that employed these types of therapy. Table adapted from
training, does not reflect a unitary approach; it encompasses a
Geretsegger et al. (2014)
broad range of techniques and activities, and can be adminis-
tered in a clinical or home-based setting in groups or individ- Music therapy Description Studies
ually. Reschke-Hernández (2011) performed a systematic re- approach
view of music therapy as a treatment for children with ASD. Active Children were allowed to (Farmer 2003)
The history of this practice dates back to 1940, although the music-made play guitar and drums. (Arezina 2011; Gattino et
first scientific article was not officially published until 1969 in by the Interactive and relational al. 2011; Kim et al.
the British Journal of Music Therapy (Reschke-Hernández participants aspects of the music 2008; Thomas and
therapy. Hunter 2003;
2011). These early pioneers adapted music education, singing Thompson 2012)
groups, folk dancing, and rhythm activities to achieve various Music therapy Active music-making (Thompson 2012)
goals such as self-expression, socialization, rehabilitation, family-- where the family
psychological enrichment, and recreation in both individual based members are also
and group settings. Over time, these advancements in music involved.
Music therapy Listening to live or (Brownell 2002; Buday
therapy were utilized by clinicians working with children with using pre-recorded music 1995; Farmer 2003;
ASD to improve many of their symptoms, while also leverag- receptive presented by the Lim 2010; and Lim and
ing their fascination with music and their often exceptional techniques therapist. Draper 2011)
ability to reproduce musical pieces (Reschke-Hernández Singing songs Songs sung by the music (Brownell 2002; Buday
2011). The range of current music therapy methods in re- therapist that were 1995; Lim 2010; Lim
composed or chosen and Draper 2011;
search and clinical practice with ASD is described by
with specific aims like Farmer 2003)
Geretsegger et al. (2014), Kaplan and Steele (2005) and correct behavior
Kern et al. (2013). In addition, Geretsegger et al. (2014) per- problems, learn signs
formed a meta-analysis of the effects of music therapy on and words, or build a
relationship with the
ASD. In this meta-analysis, they included 10 studies (165 participants.
participants) that used a variety of approaches to music thera-
py (Table 1). Each study was a randomized controlled trial
(RCTs) or controlled clinical trial comparing (i) music thera-
py, (ii) music therapy added to standard care, (iii) “placebo” individual differences in patient populations and cultures
therapy, (iv) no treatment, and (v) standard care for individ- (Hillecke et al. 2005), and (iii) parent-child relationships.
uals with ASD. They concluded that music therapy was asso- Below, we will discuss the most commonly used forms of
ciated with improvements in the core symptoms of ASD, in- music therapy in clinical practice and present the most
cluding social interaction, verbal communication, initiating recent findings associated with those therapies.
behavior, and social emotional reciprocity. Secondary out- Kaplan and Steele (2005) carried out an analysis of the
comes included increasing social adaptation and promoting database of a music therapy clinic and found that the most
the quality of parent-child relationships. However, methodo- frequently utilized interventions were interactive instrument
logical limits in the selected articles and the reduced number playing, musical instrument instruction, interactive singing,
of RCTs (i.e., inclusion of only three RCTs) suggest the re- instrument choices, and song choices. Walworth (2007) per-
sults are encouraging but should be approached with caution. formed a national survey in the USA aimed at music therapists
In addition, the differences in methodology across the 10 ar- working with clients at risk or diagnosed with ASD. Twenty-
ticles also limit the interpretations. one therapists responded by reporting on 135 clients. They
In an effort to expend the work started by Geretsegger et al. identified 17 different music therapy methods including
(2014), TIME-A study was developed by the same group of playing instruments, singing songs, movement/dancing, and
researchers (Bieleninik et al. 2017) with a strict design and a improvisation. The main goals of music therapy for children
large sample size (N = 364). However, in this study, music with ASD were to increase eye contact, verbalizations, verbal
therapy showed no benefit to participants with ASD compared command of needs, and tolerance of routine change.
to controls. These conflicting findings create confusion mak- A more recent study by Kern et al. (2013) involved a sur-
ing further assessment of the effects of music therapy on in- vey of the members of the American Music Therapy
dividuals with ASD needed. Association (AMTA) who work with individuals with ASD.
Although the field of music therapy could provide impor- A national cross-sectional sample of 328 professional mem-
tant benefits to individuals with ASD, studying music therapy bers of AMTA were recruited. They found that 97.9% of the
through a scientific lens can be problematic due to (i) multiple therapists aimed to improve communication skills, 90.6%
theoretical approaches and techniques, including variations in aimed to improve social skills, and 41.3% aimed to improve
the administration of music therapy by practitioners, (ii) emotional skills. Therapists employed singing and
Rev J Autism Dev Disord

vocalization therapy (98.6%), instrument play (98.6%), move- Coding Categories


ment and dance therapy (84%), free and thematic music im-
provisation (75.3%), and song writing and composition Each article was coded for (a) participant diagnoses, (b) age of
(55.7%). participants, (c) method of music therapy, (d) tool to measure
the improvement, and (e) result (areas of improvement or
areas with no significant differences).
The Present Study
Quality Assessment:
Several studies since the review by Reschke-Hernández
(2011) have attempted to better understand the effectiveness
We used the assessment methods in the Cochrane
of music therapy for children with ASD. The current work
Collaboration Risk of Bias Tool (CCRBT) to evaluate the risk
builds on previous research by taking a critical stance to ana-
of bias in each of the reported randomized controlled trials.
lyze (a) the reported empirical evidence of music therapy for
This tool evaluates (a) adequacy of random sequence genera-
individuals with ASD, (b) the strengths and weaknesses of the
tion, (b) allocation concealment, (c) blinding of participants
studies, and (c) the extent to which methodological variables
and study personnel, (d) incomplete outcome data, (e)
influenced treatment outcomes. Furthermore, this study pre-
reporting bias due to selective outcome reporting, and (f) other
sents future directions for further analysis of the effectiveness
forms of bias including reliable outcome assessment and base-
of music therapy in ASD.
line measures. High, low, and unclear risks of bias are indi-
cated in red, green, and yellow respectively. A study was
denoted as an unclear risk of bias if not enough information
Methods was provided, or if the information presented was unclear, for
at least one of these subcategories. Two researchers indepen-
Search Strategy dently coded the risk of bias for each article presented in this
review. Where disagreement was present, responses from the
The electronic databases PubMed and Ebsco were searched first reviewer were reported.
using keywords {(music therapy OR music OR songs OR To assess quality and risk of bias of the non-randomized
sound therapy) AND autism*} to identify studies that were studies, we used the Downs and Black scoring system (Downs
published in peer-reviewed journals between 2008 and 2018. and Black 1998). This tool consists of a 27-point checklist,
which is used to examine reporting bias, external validity,
bias, selection bias, and power of observation studies.
Selection Criteria
Reporting bias reflects whether adequate information was pro-
vided in the article in order to allow the reader to make an
Articles retrieved from the initial search were included in the
unbiased assessment of the findings. External validity refers to
present review based on the following inclusion criteria: (a)
the degree to which the results are reflective of the general
participants were diagnosed with ASD, including Asperger’s
population under analysis. Bias and confounding categories
syndrome; (b) the study was published in a peer reviewed
are associated with subjective measurement of intervention
journal; (c) participants were exposed to a method of music
and outcome, and bias in the selection of study subjects
therapy (singing/listening songs or improvisational music
specifically.
therapy), as selected based on the most prominent methods
in literature; (d) family interaction improvement, communica-
tion, psychological or physiological change in response to the
Results
music therapy were measured; and (e) the study was published
between 2008 and 2018. Studies were excluded if they pre-
The initial literature search yielded 458 articles after dupli-
sented the same data analyzed in a separate published article
cates were removed. All articles were published in the period
or if they were not written in English.
of 2008–2018. Titles and abstracts were examined in order to
remove irrelevant and review articles. A total of 36 articles
Data Extraction were included in the current review after applying inclusion
and exclusion criteria (Fig. 1).
Empirical data and methodologies described in each study
were recorded in a data-extraction form and entered into Improvisational Music Therapy (IMT)
NVivo for reporting and analysis in the current study. Each
study was coded for a list of information pertinent to the cur- One of the most researched music therapy methods for chil-
rent review’s aims. dren with ASD is IMT. In this type of therapy, the child and
Rev J Autism Dev Disord

Fig. 1 Flow diagram of article


eligibility for inclusion in the Records identified Additional records

Identification
present review through PubMed identified through Ebsco
searching (n= 267) searching (n= 205)

Records after duplicates removed (n= 458)

Screening
Records screened Records excluded
(n= 458) (n= 402)

Eligibility
Full-text articles Full-text articles
assessed for excluded, with
eligibility (n= 56) reasons (n=20)
Included

Studies included in qualitative


synthesis (n=36)

the therapist play tuned and untuned musical instruments ex- should be given more weight when evaluating the effects of
pressing rhythms, melodic patterns, and timbre. During the music therapy on ASD symptoms (Table 2).
child’s musical play, the therapist introduces synchronization One specific IMT method, the Orff-Based music therapy
and communication with the child by mirroring or method, includes musical exploration, imitation, improvization
complimenting the child’s musical expressions. The child is and creation, as well as guiding the patient through several
also encouraged to practice imitation, joint attention, turn- phases of musical development. Orff-based therapeutic music
taking and affect sharing, with the aim of improving language has shown a decrease in repetitive behaviors and improvements
skills and social competency (Crawford et al. 2017). Studies in social interaction and verbal communication in children with
implementing IMT have shown positive impact on individuals ASD (Dezfoolian et al. 2013). Effective teaching of specific
with ASD including improvements in joint attention, actions concepts (e.g., names of colors) has also occurred through the
of social engagement (Vaiouli et al. 2015; Kim et al. 2008), use of this therapy (Eren et al. 2013). When Orff-based music
more and longer events of “joy,” “emotional synchronicity,” therapy incorporates dance music therapy, individuals with
and “initiation of engagement: behaviors (Kim et al. 2009). ASD showed improvements in regulation/behavioral variabili-
Studies using IMT also show decreased stress levels, ty, imitation, disorders pertaining to various aspects of instinct,
(Poquérusse et al. 2018), increased self-esteem, reduced anx- emotional disorders, and interaction disorders (Mateos-Moreno
iety, more positive attitudes toward peers (Hillier et al. 2012), and Atencia-Doña 2013).
and improvements in verbal (Ghasemtabar et al. 2015) and Although IMT has shown positive effects in individuals
non-verbal social communication skills (Kim et al. 2008). with ASD, behavioral characteristics of the client-therapist
However, negative results have also been published. dyad seem to influence the effect of IMT. Geretsegger et al.
Bieleninik et al. (2017) did not find a significant difference (2012) showed that as the social interaction involving the
in symptom severity based on the ADOS social effect domain client and the therapist improved (the therapist became more
with 5 months of IMT intervention completed. It is important attuned to the child’s expressions musically and emotionally),
to emphasize that Bieleninik’s and collaborators study (2017) the participants showed consequent improvements in social
has a larger sample size and the highest methodological qual- skill development. A dyadic relationship while playing drums
ity of all the studies included in this review and, therefore, has shown greater improvement in motor coordination,
Table 2 This table includes the N, diagnosis (distribution), age range, study design, descriptive statistics, indexes of comparison, and outcomes. ATEC, Autism Treatment Evaluation Checklist; AQR,
Applied Behavior, Assessment of Quality of Relationship; ADOS, Autism Diagnostic Observation Schedule; ADI-R, Autism Diagnostic Interview-Revised; CG, control group; ESC, enhanced standard
care; ESCS, Early Social, Communication Scales; FEAS, Functional Emotional Assessment Scale; FCMT, family center music therapy; HF, high frequency; IPR, Index of Peer Relations; LF, low
frequency; MBCT, Melodic Based Communication Therapy; ECA-R, Revised Clinical Scale for the Evaluation of Autistic Behavior; SSRS, Social Skill Rating System Scale; TEA-Ch Test of Everyday
Attention for Children; CCC–2, Children’s Communication Checklist–Second Edition; CARS2-HF, CARS-2 High Functioning Version Rating Booklet; PPVT-4, Peabody Picture Vocabulary Test Fourth
Edition; SRS, Social Responsiveness Scale; STAI, State-Trait Anxiety Inventory state version; rsfMRI, resting state fMRI; PDDBI PDD Behavior Inventory
Rev J Autism Dev Disord

Improvisational music therapy

Author and year N, Diagnosis (distribution) Age range Study design Comparator Measure of improvement Sessions duration Outcome (significant
frequency improvements)

Bieleninik et al. 364, ASD (182 ESC vs 4 to 7 Randomized clinical IMT vs ESC ADOS HF:60 sessions, No significant
(2017) 182 ESC plus IMT) trial 60 min, 3 per week improvements
LF:20 sessions,
60 min, 1 per week
CG: 45 sessions, 60 min
Carpente (2017) 4, ASD 4 to 8 Multiple baseline Developmental FEAS 26 sessions, 15–30 min, Social communication
design relationship-based 2 per week skills
IMT
Crawford et al. 364, ASD (90 to HF 4 to 7 Randomized IMT vs ESC ADOS HF:60 sessions, 60 min, No significant
(2017) sessions, 92 to LF controlled trial 3 per week improvements
sessions and 182 on the LF:20 sessions, 60 min,
ESC group) 1 per week
Dezfoolian et al. 8, parent of children with 3 to 5 Clinical trial study ADI-R ADI-R 60 sessions, 20 min, 3 Children’s social
(2013) ASD per week interaction and verbal
communication
Eren et al. 3, ASD 3 to 6 Pre-test and post-test Orff-based music Data collection Form for 25 sessions, 60 min, Subjects are able to learn
(2013) activities probe sessions, training 2 per week the concept of color
sessions procedural yellow
reliability data collection
form, probe sessions
Pprocedural reliability and
interobserver reliability data
collection forms, social
validity data collection form
(measured before, during
and 1, 2, and 4 weeks after
completion of practice)
Ghasemtabar 27, ASD (randomly 7 to 12 Clinical trial study Music therapy SSRS 12 sessions, 60 min, Children’s social skills
et al. (2015) assigned to each Orff-schulwerk method 2 per week
condition) vs No intervention
Hillier et al. 22 ASD 13 to 29 Multiple baseline Hands-on music making Self-report questionnaire: 8 sessions, 90 min, Self-esteem, attitudes
(2012) design activities SoundScape Questionnaire, weekly toward peers, lowered
IPR and STAI trait anxiety
Kim et al. 3 to 5 Randomized controlled IMT vs playing with toys PDDBI, ESCS 12 sessions, 30 min, Joint attention behaviors
(2008) study with repeated weekly
Table 2 (continued)

Improvisational music therapy

Author and year N, Diagnosis (distribution) Age range Study design Comparator Measure of improvement Sessions duration Outcome (significant
frequency improvements)

10, ASD (randomly measures comparison


assigned to one of the design
groups)
Kim et al. 10, ASD (all in both 3 to 5 Randomized controlled IMT vs playing with toys Observation of behavior 12 sessions, 30 min, Emotional and
(2009) groups) trial weekly motivational
responsiveness and
interpersonal
responsiveness
LaGasse 17, ASD (randomly 6 to 9 Randomize controlled Musical version of the social SRS, ATEC, and behavioral 10 sessions, 50 min, Joint attention
(2014) assigned to each trial skills therapy training vs observation 2 per week Eye gaze
condition) no-music social skills Time in SRS scores
therapy
Mateos-Moreno 16, ASD (8 per group) 25.5 Controlled study Orff method and instruments ECA-R 36 sessions, 60 min, Regulation/behavioral
and + dance/movement 2 per week variability, imitation
Atencia-Doña activities vs no intervention disorder, instinct
(2013) disorder, and emotional
disorder
Mössler et al. 48, ASD (randomly 4 to 7 Observational Low-intensity IMT vs AQR, ADOS (with main HI: 32.8 sessions, 30 This study found that the
(2019) assigned to each longitudinal design high-intensity IMT focus on social affect min, 3 per week therapeutic relationship
condition) domain), SRS LI: 16.2 sessions, 30 predicts generalized
min, 1 per week clinical changes of
symptom severity in
children with autism
spectrum disorder
Pasiali et al. 9, (7 with ASD) 13 to 20 Single group pre and Group music therapy TEA-Ch, CARS2-HF, 9 8 sessions, 45 min Control of attention, ability
(2014) posttest intervention: musical behavioral tasks to switch attention,
attention control training selective attention
(with adapted Orff-based
intervention)
Poquérusse et al. 15, ASD (all in both 8 to 14 Multiple baseline Group discussion related to Levels of salivary a-amylase 5 sessions No significant
(2018) conditions) design emotions upon listening levels (marker of stress) eachtreatment, 50 improvements
music and group-based min, daily
musical improvisation vs
occupational therapy
sessions (independent
studies)
Sharda et al. 51, ASD (randomly 6 to 12 Accessor-blinded parallel Music intervention vs CCC-2, SRS-II, PPVT-4, 8–12 sessions, 45 min, Social communication;
(2018) assigned to each group randomized non-music play-based rsfMRI weekly Greater resting-state
condition) control design intervention functional brain
connectivity.
Rev J Autism Dev Disord
Table 2 (continued)

Improvisational music therapy

Author and year N, Diagnosis (distribution) Age range Study design Comparator Measure of improvement Sessions duration Outcome (significant
frequency improvements)
Rev J Autism Dev Disord

Spiro and 4, ASD and 1 4 to 7 Assessment of reliability Early sessions vs later sessions Analysis of video 10 sessions, 30 min Any change that is seen in
Himberg non-specified and validity of improvisational music recordings the client is dependent
(2016) communication therapies on the relationship with
disorder the therapist. Therapist
must promote mutual
facing and shared pulse
in order to obtain
improvements in these
two activities.
Thompson 8, mothers of children with 31–46 Follow-up interview FCMT A descriptive 16 sessions, 30 to Mothers' confidence to
(2017) ASD (mother) phenomenological analysis 40 min, weekly engage their child
3 to 5 of interviews with mothers Child social
(child) of children with ASD who communication
received FCMT Quality of life
Thompson and 11, mothers of children 3 to 6 Follow-up interview FCMT A descriptive 16 sessions, 15 to Positive change in parent
McFerran with ASD (child) phenomenological analysis 40 min, weekly child relationship
(2015) of interviews with mothers
of children with ASD who
received FCMT
Thompson et al. 23, ASD (randomly 3 to 6 Parallel randomized FCMT vs family-centered Parents-rated standardized 16 sessions, 30 to Social interactions in the
(2014) assigned to each controlled trial early childhood measures, clinician 40 min, weekly home and community
condition) intervention observation and structured Parent-child relationship
parent interview
Vaiouli et al. 3, ASD 5 to 7 Multiple baseline IMT Teacher and parent interviews 21–22 sessions, Levels of focusing on faces
(2015) design and weekly classroom 10 min, weekly Responding to joint
observations attention, initiating joint
attention. Parents and the
teachers reported gains
of the intervention
across settings and
across trainers.
Yoo and Kim 15, ASD 11 to 16 Multiple baseline Instructed drum K-WISC-IV, Korean-Social Once, duration depend Cooperation and
(2018) design tapping task Skills Rating System, on individual self-control, engagement
ability to mimic gestures, participants in joint action, increased
ability to recognize facial occurrence of target
expressions behaviors
Rev J Autism Dev Disord

sequence of engagement, interpersonal coordination, and based therapy. The results revealed improvements in both
adaptive adjustment (Yoo and Kim 2018). According to groups, with greater improvements in the music therapy group
Spiro and Himberg (2016), IMT is a mutual process (thera- (MTG). MTG increased occurrences of joint attention and eye
pist-client), and any change seen in the client is dependent on gaze. Children in the social skills group (SSG) maintained
the relationship with the therapist. interactions with adults but decreased peer-to-peer interac-
In a study performed to assess the neurobehavioral out- tions. Initiating communication showed no improvements
comes of a music intervention in ASD, fifty-one children with across groups. Eye gaze towards other people decreased in
ASD aged 6–12 years were randomized to receive music in- the SSG and increased slightly in the MTG group. Musical
tervention or intervention without music. The music group structure provided in the MTG appeared to be more successful
showed higher communication scores and increased resting- at maintaining the children’s attention to their peers. However,
state functional connectivity between auditory and subcortical no improvements were reflected in the Autism Treatment
regions, and auditory and fronto-motor regions post- Evaluation Checklist (ATEC).
intervention compared with the group that did not receive In the category of IMT that includes FCMT and MTG, we
the IMT. In addition, in areas typically over-connected in in- included 21 articles (Table 3). In this group, we found one study
dividuals with ASD, the music group showed lower connec- that includes a large sample size with 364 subjects (Bieleninik
tivity (between auditory and visual regions), suggesting ben- et al. 2017 & Crawford et al. 2017); nine articles with sample
eficial effects of music therapy in children with ASD (Sharda sizes below ten (Carpente 2017; Dezfoolian et al. 2013; Eren
et al. 2018). et al. 2013; Kim et al. 2008; Kim et al. 2009; Pasiali et al. 2014;
Spiro and Himberg 2016; Thompson 2017; Vaiouli et al. 2015);
Family Centered Music Therapy (FCMT) and ten articles with a moderate sample that ranges between 11
and 51 subjects (Ghasemtabar et al. 2015; Hillier et al. 2012;
In an effort to increase the engagement of children with ASD LaGasse 2014; Mateos-Moreno and Atencia-Doña 2013;
with members of their family, one important variant of IMT Mössler et al. 2019; Poquérusse et al. 2018; Sharda et al.
was developed, FCMT. This therapy shares the same princi- 2018; Thompson and McFerran 2015; Thompson et al. 2014;
ples as IMT; however, the FCMT technique is implemented Yoo and Kim 2018). The study with a large sample size found
within a family-centered framework. Each FCMT session no significant improvements in the patients with ASD. The
usually includes a greeting song, specific music activities, free studies with a small sample size had only positive outcomes,
exploration time that gives the participants the opportunity to and the studies with a moderate sample size found had mostly
improvise with the instruments, and a good-bye song. Within positive results. Underpowered studies are more likely to miss
this framework, interventions included instrument exercises, genuine effects, and as a group they are more likely to include a
movement to music, rhythm-based activities, singing, impro- higher proportion of false positives. Estimating the influence of
visational music, simple folk dances, and music games. The session duration and frequency is challenging due to broad
activities were specifically chosen to promote joint attention, variations (e.g., 60 sessions of 60 min, 5 sessions of 50 min,
interaction among group members, self-expression, and coop- or 22 sessions of 10 min).
erative group experiences (Allgood 2005).
Thompson et al. (2014) followed up on the work done by Singing/Listening Songs
Allgood (2005) by designing a replication with a larger sam-
ple size (n = 23). They found that FCMT improved social Although a large percentage of individuals with ASD (25%)
interactions in the home, community, and the parent-child are non-verbal, few interventions are able to produce signifi-
relationship; however, no improvements in language skills cant improvements in communication in these individuals
or general social responsiveness were found. During the inter- (Wan et al. 2011). Educational experiences through songs
views, every mother expressed how musical play activities suggest that singing might be useful due to their high-pitch
provided opportunities to experience a sense of closeness perception and their ability to vocalize musically (Scott 2015).
and connection with their child at a time when they felt uncer- Greater improvements in verbal production in low-
tain about how to do so. functioning individuals with ASD have been found compared
to high-functioning individuals with ASD (Lim 2010).
Group-Based Improvisational Music Therapy Studies also suggest that therapies involving singing/
listening to songs increase on-task behaviors, social commu-
Based on the premise that group-based therapy can improve nicative responsiveness, and decrease negative self-
overall social competence in individuals with ASD (Reichow stimulatory behaviors (Dieringer et al. 2017; Paul et al.
et al. 2012), the main principles of IMT were extended to a 2015; Gee et al. 2014; Finnigan and Starr 2010). This type
group format by LaGasse (2014) to explore whether group- of therapy also improves parent-child relationships through
based musical therapy differed from a non-musical group- imp ro ved communication, especia lly non-verbal
Table 3 This table includes the N, diagnosis (distribution), age range, study design, descriptive statistics, indexes of comparison, and outcomes. ADOS, Autism Diagnostic Observation Schedule; SB4,
The Stanford-Binet Intelligence Scale-Fourth Edition; PPVT-III, The Peabody Picture Vocabulary Test-Third Edition; EOWVT, The Expressive One Word Vocabulary Test; ALT-PE, Academic Learning
Time in Physical Activity; SOR, Sensory over-responsivity; ESCS, Early Social Communication Scales; ASSP, Autism Social Skills Profile; CCs comprehension checks; SBIs, sound-based interventions;
ABA-VB, Applied Behavior Analysis Verbal Behavior; ABA. Applied Behavior Analysis; MBCT, Melodic Based Communication Therapy

Singing/listening songs
Rev J Autism Dev Disord

Author and year N, diagnosis Age range Study design Comparator Measure of improvement Sessions duration Outcome (significant
(distribution) frequency improvements)

Corbett et al. 11, ASD (all in both 4 to 8 Crossover randomized SBIs vs no intervention ADOS (generic), SB4, 30 sessions, 120 min, No significant improvements
(2008) conditions) controlled trial PPVT-III, daily, 10 days
and EOWVT.
Dieringer et al. 5, ASD (all in both 5 to 6 Multiple baseline Music with lyrics ALT-PE 10 to 13 sessions, 10 No significant improvements
(2017) conditions) design + singing instructions min, 4 per week
vs music with lyrics
Fees et al. 11–15, ASD, Down 3 to 5 Clinical trial design Musical interaction Open ended questions 12 sessions, 6 h, Increase in self-prompting
(2014) syndrome, reactive for teachers 4 per week behavior
attachment disorder,
and speech delay
Finnigan and Starr 1, ASD 3 Single-subject Musical interaction vs Visual inspection. Frequency 29 sessions, 15 min, Eye contact
(2010) alternating treatment non-musical of social responsive 4 per week Higher percentages of
design interaction behaviors and social imitation and turn-taking
avoidance behaviors skills
Gattino et al. 24 7 to 12 Parallel randomized Relational music therapy CARS-BR 16 sessions, 30 min, 1 No significant improvements
(2011) controlled trial interventions plus per week
clinical routine activities
vs clinical routine
activities
Gee et al. (2014) 1, ASD 7 Single-subject case SBIs SOR and observation of 100 sessions, 15 min, Decrease in the number of
controlled design related behaviors 5 per week/twice negative (avoidant, verbal,
per day and physical negative) and
self-stimulatory behaviors
Kalas (2012) 30, ASD (assigned into 4 to 6 2 × 2 repeated Simple music ESCS 6 sessions, 10 min, Simple music: elicit RJA for
severe group and measures factorial vs complex music 2 per week severe ASD group
mild-moderate multiple baseline Complex music: elicit RJA for
group) design mild/moderate ASD group
Katagiri (2009) 12, ASD (randomly 9 to 15 Multiple baseline No intervention vs verbal Decoding and encoding 8 sessions, 30 min, Improved understanding of
assigned to one of the design teaching of emotions vs assessment 2 per week emotions with background
groups) improvised of 4 emotions music condition
emotion-related
background music vs
using songs to teach
about emotions
Lim and Draper 22, ASD (randomly 3 to 5 Randomized Music incorporated with Measurement of levels of 6 sessions, 3 per week Most effective in echoic
(2011) assigned to each controlled trial ABA VB method vs verbal production and production
condition) echolalic production of
Table 3 (continued)

Singing/listening songs

Author and year N, diagnosis Age range Study design Comparator Measure of improvement Sessions duration Outcome (significant
(distribution) frequency improvements)

ABA VB method vs target words using coded


no-training analysis of video tapes
Lim (2010) 50, ASD (randomly 3 to 5 Randomized controlled Music training, speech Experimenter designed verbal 6 sessions, twice per day No significant improvements
assigned to each trial training and no training production evaluation scale
condition) (a form fill-in-the-blank that
includes the target words of
the treatment)
Paul et al. (2015) 3, ASD (all in both 3 and 4 Adapted single-subject Singing instructions Post-hoc observational coding 9 session per condition Performance, social gesture,
conditions) design vs spoken directives of video tapes assessing (18 sessions total), eye contact
frequency of social gesture 3–4 min per condition,
and eye contact weekly
Preis et al. (2016) 5, ASD (all in both 4.3 to 6.0 Single-subject Background music during Frequency of spontaneous 28 sessions, 15 min, 1 No significant improvements
conditions) alternating normal routine of guided verbalization to a peer or an per week (increased utterances and
treatments design structured play vs no adult, social disengagements)
music responsiveness, or
engagement as measured by
frequency of
disengagement
Sandiford et al. 12, ASD (randomly 5 to 7 Randomized control MBCT vs traditional Number of verbal attempts, 20 sessions, 45 min, 4 No significant improvements
(2013) assigned to each design speech and language number of correct words, per week
condition) therapy number of words reported
by the parent, and number
of imitative attempts
Schwartzberg and 30, ASD 9 to 21 Randomized control Music-based social story ASSP and CCs 7 sessions, 50 min, daily No significant improvements
Silverman (2013) design vs reading a social story
Wang and Oldfield 4 ASD and Asperger 3 to 11 Multiple baseline Regular music therapy Analysis of non-verbal 3 sessions, 30–45 min, Parent-child relationship
(2018) syndrome design communication, facial weekly
expression, and tone of
voice of subjects
Rev J Autism Dev Disord
Rev J Autism Dev Disord

communication, and increasing feelings of closeness and un- progress. The groups did not show significant differences
derstanding (Wang and Oldfield 2018). in the number of correct words, verbal attempts, or number
In addition to improvements in communication, other of imitative attempts following treatment. Only parents of
studies involving singing/listening to songs suggest that it the MBCT group report significant improvement in the
improves behavior in individuals with ASD. Teachers per- number of new words heard in the home and other environ-
ceived the use of social story songs adapted for pre- ments (Sandiford et al. 2013). Kalas (2012) examined the
schoolers to be effective in supporting positive behavioral effects of simple versus complex music in joint attention
change (Fees et al. 2014). Furthermore, Gee et al. (2014) responses of children with ASD. He observed a statistically
investigated the efficacy of sound-based interventions significant interaction between music modality and function
(SBIs), where psycho-acoustically modified classical mu- level, showing that playing simple music was more effec-
sic was delivered using specialized headphones and ampli- tive in eliciting responses to joint attention for children with
fiers and a standard CD player. The results showed a de- severe ASD, whereas playing complex music was more ef-
crease in the number of negative and self-stimulatory be- fective in eliciting a response to joint attention in children
haviors. Finnigan and Starr (2010) explored the effects of with mild to moderate ASD (Kalas 2012).
music and non-music interventions using familiar melodies There were 15 studies in this group (Table 3), six of which
verses-spoken scripts similar to the melodies. They had fewer than ten participants; from these, two presented
assessed the effect on the social responsiveness and negative results. In addition, we included nine articles with a
avoidant behaviors (behaviors that reduced the amount of moderate sample size ranging from 11 to 50 participants.
interaction between the child and the therapist). Results From these, five did not present significant results in their
indicated that the music intervention was more effective investigations. Similarly to the IMT group, it is problematic
than the non-music intervention in increasing social to identify the effect of the sessions’ duration and frequency
responsiveness and avoidant behaviors in the child with due to variability that ranges from 100 sessions of 15 min to 3
autism. Dieringer et al. (2017) investigated the effect of sessions of 30–45 min.
music with lyrics versus music with lyrics and instruction.
The author reported that music with lyrics plus instruction
increased on-task behaviors to a greater extent than did Risk of Bias in Included Studies
music with lyrics only.
Despite these positive effects of music therapy, there are For the RCTs in this meta-analysis, most of the domains
also negative and contradictory results. The two studies were evaluated as having a low risk of bias (allocation se-
with the largest sample sizes in this category indicated that quence concealment, incomplete outcome data, and selec-
both singing/listening songs and speech therapy are effec- tive outcome reporting of domains). Within the “allocation
tive in inducing verbal production, including new vocabu- concealment” category, an unclear risk of bias was assigned
lary, semantics, phonology, pragmatics and prosody; how- to many of the reported studies due to unclear or absent
ever, Lim and colleagues reported no statistically signifi- descriptions regarding the allocation concealment methods.
cant differences between the results obtained from speech Nine of the studies were assigned moderate or high risk of
training and music training in terms of speech production bias in the blinding category due to knowledge of the allo-
(Lim and Draper 2011; Lim 2010). A similar study with cated interventions by participants and/or personnel during
only three participants compared song instruction with spo- the study. An unclear risk of bias was also largely identified
ken directives in the elicitation of socio-communicative be- across studies in the “free of other bias” risk category due to
haviors in children with ASD. They showed no significant factors including low number of participants, low number of
differences between strategies in improving social commu- therapy sessions, lack of a follow-up period, and lack of
nicative responsiveness (Paul et al. 2015). Schwartzberg standardized/objective analysis methods (Fig. 2).
and Silverman (2013) found music-based social stories to To assess quality and risk of bias of the non-
be ineffective for comprehension and generalization of randomized studies in the current review, we used the
targeted social skills in children with ASD. In addition, Downs and Black scoring system (Downs and Black
Corbett et al. (2008) were not able to prove the effectiveness 1998), where a high score indicates a low overall risk of
of Tomatis sound therapy (a therapy that utilizes high- and bias. The majority of the studies reported in Table 4 met
low-frequency-filtered music) for expressive and receptive less than or close to 50% of the checklist investigating
language. When melodic based communication therapy risk of bias, suggesting that relatively high methodologi-
(MBCT), a music-based therapy that utilizes a standard cal weaknesses were present in the reported studies.
melody for each target word, was compared with traditional Although we included high-quality with low risk of bias
speech and language therapy for eliciting speech, the main papers, it is essential to mention that many present a mod-
differences between group outcomes were the timing of erated risk of bias and severe limitations.
Rev J Autism Dev Disord

Free of selective reporting


(Short-term outcomes 2-6
Incomplete outcome data
Allocation concealment?

addressed (Mortality)
Blinding (Subjective
Adequate sequence

Free of other bias?


generation?

outcomes)

wks)
Bieleninik et al. (2017)

+ + + + + +
Corbett et al. (2008)

+ + + + + +
Crawford et al. (2017)

+ ? ? + + +
Dieringer et al. (2017)

+ ? + + + ?
Finnigan & Starr (2010)

+ ? + + + -
Schulz et al. (2011)

+ + + + + +
Ghasemtabar et al. (2018)

? ? ? + + +
Kalas, A. (2012)

+ + - + + +
Katagiri, J. (2009)

+ ? - + + ?
Kim et al. (2008)

+ - + + + ?
Kim et al. (2009)

+ - + + + ?
LaGasse (2014)

+ + ? + + ?
Lim (2010)

+ ? + + + ?
Lim & Draper (2011)

+ ? + + + ?
Fig. 2 Breakdown of each type of risk of bias identified in the included studies

Discussion strengths and weaknesses of the studies reporting this therapy


effectiveness, and investigate the extent to which methodolog-
This research aimed to review empirical evidence of the ben- ical variables influenced treatment outcomes. To archive this,
efits of music therapy for individuals with ASD, identify we searched in two databases (PubMed and Ebsco) to
Rev J Autism Dev Disord

Mateos-Moreno et al. (2013)

- - - + + ?
Mossler et al. (2017)

+ ? + + + +
Paul et al. (2015)

+ - + + + ?
Poquérusse et al. (2017)

+ + + + + ?
Preis et al. (2015)

+ - - + + ?
Sandiford et al. (2013)

+ ? + + + +
Schwartzberg & Silverman (2013)

+ ? ? + + ?
Sharda et al. (2018)

+ + + + + +
Thompson et al. (2013)

+ + - + + +
Yoo & Kim (2016)

+ - + + + ?
Fig. 2 continued.

examine all studies published between 2008 and 2018 that can have a positive impact on children with ASD, particularly
explored the effects of music therapy in ASD. We assessed in social interaction and communication (verbal and non-ver-
the risk of bias, coded, and analyzed all selected papers. bal). On the other hand, other studies with a large sample size
In summary, we found that research to date indicates mixed and methodological quality did not find a significant differ-
results; on the one hand, evidence suggests that music therapy ence in symptom severity. Interestingly, one study that used

Table 4 Risk of bias and


methodological quality for non- Author Reporting External validity Bias Confounding Total
randomized studies, using the (10) (3) (7) (6)
Downs and Black tool for
retrospective cohort studies (n = Carpente (2018) 10 1 1 4 16
11 studies) Dezfoolian et al. (2013) 10 0 5 4 20
Eren et al. (2013) 5 1 2 4 12
Fees et al. (2014) 6 2 0 4 12
Gee et al. (2014) 7 1 4 3 15
Hillier et al. (2012) 9 1 4 3 17
Pasiali et al. (2014) 10 1 3 4 18
Spiro and Himberg (2016) 4 0 3 2 9
Thompson (2017) 4 1 2 3 10
Thompson and McFerran 7 0 3 3 13
(2015)
Vaiouli et al. (2015) 6 1 4 3 14
Wang and Oldfield (2018) 6 1 2 4 13
Rev J Autism Dev Disord

neuroimaging to assess music therapy’s impact on ASD found challenge because of the broad spectrum of symptoms which
increased resting-state functional connectivity. There was in- requires a variety of approaches to address. The extant litera-
sufficient information to clearly understand the relationship ture on music therapy in ASD is insufficient to understand the
between methodological variables such as duration and fre- impact of these variables, largely due to the lack of specificity
quency of the training, age, the therapists abilities, and the on the reported effectiveness of a given music therapy method
severity of the symptoms and treatment outcome. Finally, on improving symptoms of individuals with ASD at varying
several methodological weaknesses in the current literature levels of severity.
were observed in many of the articles. For future recommen-
dations, we suggest creating high-quality studies with larger Strengths and Weaknesses of the Studies Reporting
sample sizes that include neuroimaging techniques to gain a This Therapy Effectiveness
more objective understanding of the effect of music therapy
on individuals with ASD. The common methodological weaknesses presented in these
studies include small sample sizes, lack of blinded participants
Reported Empirical Evidence of Benefits of Music and/or researchers, limitations in study design, lack of ade-
Therapy for Individuals with ASD quate randomization, and lack of follow-up. One additional
significant weakness of these therapeutic studies lies in the
We investigated two very commonly reported methods of evaluation methods of treatment effectiveness. Many studies
music therapy, IMT and listening/singing songs, highlighting utilize parent-based or self-reports to measure treatment effec-
the main outcomes and the contradictory/negative results of tiveness, while others use diagnostic measures such as the
each method. There are several IMT techniques; however, Autism Diagnostic Observation Schedule (ADOS) and
most of the studies have small samples and were not random- Childhood Autism Rating Scale (CARS) to analyze social/
ized. They mainly show improvement in engaging with the behavioral differences before and after therapy. It is important
people who participate in the therapy with them (family, par- to point out that diagnostic measures might not be the best
ents, therapist). One study investigated IMT in ASD with a option to assess treatment improvements due to their resis-
large multicenter randomized clinical trial (Bieleninik et al. tance to change and the lack of sensitivity to the effects of
2017; Crawford et al. 2017). The results of this study were the given therapy on specific participants. For this reason, it
consistent with the previous findings, indicating that this type is important to consider a combination of assessment methods,
of therapy does not affect ASD symptoms as indexed by such as parent/self-reports, psychometric measures, coded
ADOS scores. The results in this IMT also emphasized the analysis of video recordings, and even neurophysiological as-
importance of the therapeutic relationship (the social interac- sessments prior to, and after, therapy. The focus should be to
tion between the client and the therapist) in improving the address a broad range of dimensions. The need for measures
social skills of the ASD participants. Most studies conducted on treatment fidelity may be added, such as the Assessment of
included moderate-to-high functioning (IQ > 70) participants the Quality of Relationship (AQR) which is used to measure
with ASD, and accordingly they fail to demonstrate effective- the appropriateness of interventions in a subsample of the
ness across the spectrum. TIME-A study and shows interventions results by develop-
In the singing/listening to songs therapy method, it was mental level of the client (Mössler et al. 2019). Treatment
suggested that singing and listening to songs is as good as or fidelity assessments could also be improved with measures
better than language therapy. This therapy showed greater such as the social responsiveness and Vineland scales. In fact,
impact on low functioning individuals with ASD, who are the field of music therapy and its effectiveness in ASD would
non-verbal as compared to high functioning individuals with also benefit from a more standardized assessment model, such
ASD. These studies mainly targeted children between 3 and 7 that multiple assessments as described above should be orga-
years old. One of the main problems in these studies was small nized and grouped so that results across studies become more
sample sizes. This may be due to difficulty recruiting individ- meaningful and comparable.
uals with ASD and intellectual disability. Furthermore, as reflected in the literature, it is not common
to find studies on the effects of music therapy in both high-
Extent to Which Methodological Variables Influenced functioning and low-functioning individuals with ASD.
Music Therapy Treatment Outcomes Children on the low level of functioning might be assigned
to music therapy due to its nonverbal and playful low-demand
The level of effectiveness of music therapy, however, depends quality. This group has a lower prognosis in improvement of
on several factors. We must consider the factors that impact ASD-related symptoms compared to those with high function-
many behavioral treatments such as the duration and frequen- ing autism. It is imperative to address the differences between
cy of the training, age, the therapist’s abilities, and the severity these subgroups when comparing the outcome variables. We
of the symptoms. In particular, ASD presents a significant should also question the comparability of studies assessing
Rev J Autism Dev Disord

cognitive-based behavioral interventions in contrast to identify specific neural changes as a result of music.
musical-bodily interventions in these subgroups of ASD. These types of assessments do not depend solely on the
Addressing these recommendations would provide a more parent, participant, or therapists’ interpretations of the
reliable, standardized, and sensitive assessment of the effect therapy effectiveness and thus avoid potential biases in-
of therapy on individuals with ASD. cluding lack of blinding. Furthermore, neuroimaging
In addition to the lack of objectivity in treatment effec- methods are easy to repeat in a precise manner across
tiveness measures, the types of therapies that are common- various research groups, allowing for consistency between
ly used are rarely directed at the individual’s specific skill studies, a necessary component to accurately compare the
set and developmental goals. Such therapies would foster effectiveness of therapies across studies.
improved functional gains and quality of life in these indi- A music therapy designed to specifically address social
viduals. One treatment model that encourages this type of and/or behavioral deficits in ASD, in childhood, when the
individualized intervention is the Developmental, brain is most sensitive to this therapy (de Villers-Sidani
Individual Differences, Relationship-based model (DIR) et al. 2007; Hyde et al. 2009), could elicit important brain
Carpente 2017). This therapeutic model is relationship- changes that are identifiable through neuroimaging assess-
based, client-centered, and stresses the importance of spon- ment methods, as previously shown (Moreno 2009).
taneous interactions, engagement, and creativity in order to Altogether, there is reason to believe that music therapy
improve learning, socialization, communication, and be- may be able to improve brain function in ASD if it is
havior in the individual (Carpente 2017). This is achieved specifically targeted to known impaired functions and tai-
by providing developmentally appropriate relational expe- lored to the individual (Sharda et al. 2018). Further stud-
riences to each child through child-led interventions. A ies are necessary to identify these possible brain changes
spontaneous and individualized therapy, such as the one over time with neuroimaging methods in order to gain a
suggested by Greenspan, provokes an even greater need more objective understanding of the effect of music ther-
for transparency in testing and assessment methods. With apy on individuals with ASD.
current technology it is also possible, and highly benefi-
cial, to include audio or video excerpts within studies.
Overall, transparent reports of the individual skills, needs,
and goals of the child, rather than ASD symptom reports,
Conclusion
would be beneficial for guiding more individualized, and
likely more effective, treatment plans. Subsequently, treat- We identify a total of 36 studies investigating the use of music
ment plans targeted at these individual needs would sup- therapy in ASD. After the analysis of these studies, the current
port a greater quality of life for the individual receiving review found methodological limitations in several of them.
treatment. These limitations prevent us from making a strong statement
about the effects of music therapy on autism. Thus, we con-
clude that more studies are needed that utilize large sample
sizes, appropriate experimental methodology, and objective
Future Directions
measurements of treatment effectiveness. We encourage the
inclusion of neuroimaging modalities in more studies address-
Neuroimaging in ASD: a Potential Objective Marker of
ing music therapy effects. In addition, more knowledge is
Changes Induced by Music Therapy
needed about the specific effects of particular methods and
their effects on individuals with ASD with and without intel-
Given the heterogeneity of symptoms across individuals
lectual disability. Only in light of such comprehensive evi-
with ASD, including intellectual disabilities, paired with
dence can we truly determine the benefits of music therapy
inconsistent results across studies that assess the effective-
on individuals with ASD. Lastly, it is essential to employ
ness of music therapy on changing ASD symptomatology,
therapies aimed at engagement and creativity and outcome
more, objective research might be necessary. More specif-
measures that evaluate functional gains and quality of life.
ically, the TIME-A study, which had a large sample size
and sound methodology, did not support IMT as an effec-
tive therapy for individuals with ASD. It is possible that
studies up to date are too focused on changes in overall
Declarations
symptom severity, behavioral and social ability, rather
Ethics Approval This study does not involve human participants.
than more specific, objective assessments. Behavioral
changes induced by music therapy might not be evident Consent to Participate This study does not involve human participants.
right away to the experimenter, whereas more objective
assessments such as neuroimaging recordings, could Conflict of Interest The authors declare no competing interests.
Rev J Autism Dev Disord

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