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Music & Science
Volume 1: 1–18
Rethinking the role of music ª The Author(s) 2018
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DOI: 10.1177/2059204318769639
journals.sagepub.com/home/mns
of autism spectrum disorder

Thenille Braun Janzen and Michael H. Thaut

Abstract
Music has played a prominent role in the clinical and research literature on autism spectrum disorder (ASD) in regard to
diagnosis, therapy, and behavioral observations of exceptional artistic abilities in this population. Music as therapy for ASD
has traditionally focused on social interaction, communication skills, and social-emotional behaviors. However, recently,
there has been an increased research focus on the role of motor and attention functions as part of the hallmark features of
ASD, which may have significant implications for the role of music as an intervention for individuals with autism. The
purpose of this article is to provide a critical appraisal of new research developments for therapists and researchers to
potentially reassess the role of music as intervention to support healthy neurodevelopment in individuals with ASD and
expand the current clinical scope of practice in music therapy for autism. Our argument is based upon compelling research
evidence indicating that motor and attention deficits are deeply implicated in the healthy neurodevelopment of socio-
communication skills and may be key indicators of structural and functional brain dysfunction in ASD. In light of this
evidence, we suggest that music-based developmental training for attention and motor control may receive a critical new
functional role in the treatment of autism due to the significant effect of auditory-motor entrainment on motor and
attention functions and brain connectivity.

Keywords
Attention, autism spectrum disorder, intervention, music therapy, motor control

Submission date: 7 August 2017; Acceptance date: 19 March 2018

Autism spectrum disorders (ASD) is the term used for a new avenues for clinical research with broad implications
diverse group of developmental conditions that affect one’s for the role of music as an intervention for individuals with
ability to relate to and communicate with others (Walsh, ASD.
Elsabbagh, Bolton, & Singh, 2011). These conditions The clinical application of music to mediate the devel-
(henceforth also referred to as “autism”) are characterized opmental and therapeutic processes in autism has a long
by two cardinal clinical features: persistent deficits in history (for review, see Reschke-Hernandez, 2011). Most
social communication and social interaction, and repeti- therapy practices including music-based techniques have
tive/stereotyped behaviors, including hyper- or hypo- been traditionally centered on outcomes considered pri-
reactivity to certain sensory aspects of the environment mary features of autism: communication, social interaction,
(American Psychiatric Association, 2013). In addition to
these hallmark features, individuals with ASD also consis-
tently display several other debilitating impairments, Music and Health Science Research Collaboratory, Faculty of Music,
including motor and attention deficits (Allen & Courch- University of Toronto, Toronto, Ontario, Canada
esne, 2001; Bhat, Landa, & Galloway, 2011; Fournier,
Hass, Naik, Lodha, & Cauraugh, 2010). Indeed, recently Corresponding author:
Michael H. Thaut, Music and Health Science Research Collaboratory,
emerging research evidence demonstrates that motor Faculty of Music, University of Toronto, 80 Queens Park, Toronto,
impairments and attention deficits are deeply implicated Ontario, M5S 2C5, Canada.
in socio-communication and interaction behaviors, opening Email: michael.thaut@utoronto.ca

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2 Music & Science

and emotional skills. In this article, we propose a reassess- some sounds and loud noises has been described in this
ment of the role that music can play in interventions to population (Danesh, Lang, Kaf, Andreassen, Scott, & Esh-
support healthy neurodevelopment in ASD and suggest, raghi, 2015; Demopoulos & Lewine, 2016; Funabiki,
based on newly emerging research evidence, that music- Murai, & Toichi, 2012; O’Connor, 2012), research suggests
based developmental training for attention and motor that this abnormality is not associated with hypersensitivity
control may receive a critical new functional role in the of the auditory pathways but with difficulties in the higher
treatment of autism. To support this argument, we begin cortical processing systems (Funabiki et al., 2012).
with an overview of recent behavioral and neuroimaging Emerging evidence also emphasizes the role of music
research on preserved musical-related skills in individuals stimuli in studying emotion processing in ASD. Several
with autism. Next, we provide a critical overview of the studies have shown, for instance, that individuals with aut-
current state of clinical research into music therapy for ism are able to properly identify the positive and negative
people with autism centered on results from recent con- valence of emotional musical stimuli (Brown, 2017;
trolled clinical studies examining the effectiveness of Gebauer, Skewes, Westphael, Heaton, & Vuust, 2014; Hea-
music-based interventions to address communication, ton, Hermelin, & Pring, 1999; Quintin, Bhatara, Poissant,
social and emotional skills. In the subsequent section of Fombonne, & Levitin, 2011). These findings are consistent
the article, we present research evidence demonstrating that with neuroimaging reports indicating that cortical and sub-
motor and attention deficits have direct implications to cortical circuits underlying affect and reward processing
socio-communication and interaction skills and that these are preserved in individuals with ASD (Caria, Venuti, &
impairments may be key indicators of structural and func- De Falco, 2011). Interestingly, a recent study found that
tional brain dysfunction in ASD. To conclude, we provide a although emotional responses to music are preserved in
theoretical framework to support the application of rhythm- ASD, autistic individuals with comorbid alexithymia (a
based interventions in the treatment of autism. disorder characterized by an absence of cognitive insight
into one’s emotions) have difficulties ascribing affective
labels to these emotions (Allen, Davis, & Hill, 2013; for
Music-related skills in autism review, see Bird & Cook, 2013; Hill, Berthoz, & Frith,
There is robust research evidence that music is a domain of 2004; Molnar-Szakacs & Heaton, 2012). Therefore, these
preserved skills and interest in individuals with ASD. studies collectively indicate that, although individuals with
Empirical behavioral research has demonstrated that indi- ASD have significant deficits in processing complex emo-
viduals with autism often have intact or superior pitch and tional cues within the social context, their ability to identify
timbre processing abilities (Bonnel et al., 2003; Heaton, the emotional content of complex nonsocial affective sti-
2005, 2009; Heaton Hermelin, & Pring, 1998; Heaton, muli such as music is generally preserved.
Williams, Cummins, Happé, & Háppe, 2007; Mottron, Despite speech disabilities in autism, recent neuroima-
Peretz, Menard, & Ménard, 2000), enhanced melodic mem- ging research has demonstrated that speech-analog music
ory (Heaton, 2003), preserved abilities to understand the abilities are frequently preserved (Lai, Pantazatos, Schnei-
rules of Western musical harmony and process melodic der, & Hirsch, 2012; Sharda, Midha, Malik, Mukerji, &
complexity (Heaton et al., 2007; Thaut, 1988), and intact Singh, 2015). A recent neuroimaging study investigating
rhythm synchronization capacity (Tryfon et al., 2017). One speech and song processing in low-functioning children
particularly intriguing research finding in the ASD auditory with ASD found that the Broca’s area (left inferior frontal
research literature is that exceptionally good pitch informa- gyrus) – which is typically under-activated during speech
tion processing commonly appears in autism (for review, processing in children with autism relative to controls –
see Heaton, 2009). It has been shown, for instance, that was significantly activated during song processing, which
autistic children without musical training are often able indicates that children with ASD may be more effectively
to accurately associate musical tones to their corresponding engaged in musical stimuli (Lai et al., 2012). Another ima-
note labels – a skill known as absolute pitch, a rare ability ging study expanded this finding by showing that while
to identify or produce the pitch of a tone without reference frontotemporal connectivity is significantly disrupted dur-
to an external standard (Heaton, 2003; Heaton, Pring, & ing spoken-word perception, this network is preserved dur-
Hermelin, 2001; Heaton, Williams, Cummins, & Happé, ing sung-word processing (Sharda et al., 2015). These
2008). This enhanced ability to process simple auditory findings suggest that alternate mechanisms of speech pro-
stimuli such as musical tones contrasts with a generally cessing may be recruited in individuals with ASD through
diminished ability to process social and more complex singing, which may be due to functional and structural
sounds such as speech; a paradox that has served as a model differences between speech and song (e.g., singing being
to study atypical sensory processing in ASD (Chowdhury non-reciprocal, structured and ritualized, often metric and
et al., 2017; Just, Cherkassky, Keller, Kana, & Minshew, slower than speech, and having only associated semantic
2007; Mottron, Dawson, Soulières, Hubert, & Burack, meaning).
2006; Mottron et al., 2000; Ouimet, Foster, Tryfon, & Collectively, this growing body of research evidence
Hyde, 2012). Importantly, although hypersensitivity to corroborates anecdotal reports that music has a profound
Janzen and Thaut 3

impact on children with autism, and demonstrates that songs composed by the music therapist to include the target
music-related skills in domains such as memory, auditory words and phrases to be learned, whereas in the speech
perception, emotion, and language, are significantly pre- control condition the therapists spoke the same texts for
served in ASD. In addition, these findings suggest that the sentences and directions. Children with ASD, who were
music may be a particularly resourceful therapeutic verbal or preverbal, received both music and speech train-
approach for autism with strong potential to promote and ing for three days per week for two weeks (the order of the
facilitate functional changes in non-musical brain and intervention was counterbalanced). The results indicated
behavioral functions. that there were no differences between the treatment groups
since both music and speech training had a significant
effect on verbal operant production compared to the no-
Current state of clinical research training condition. The only measure in favor of the music
Traditionally, clinical and research practices in music ther- training condition was echoic production of speech.
apy have focused predominantly on outcomes considered Similar results were observed in other studies comparing
primary features of autism: communication, social interac- the effectiveness of infant-directed speech intervention
tion, and emotional skills. The following overview and with infant-directed song intervention on communication
critical appraisal of the current research literature focus skills. Simpson and colleagues (Simpson, Keen, & Lamb,
on randomized controlled trials and controlled clinical 2013, 2015) tested whether sung instructions embedded
trials published in peer-reviewed journals between 2000 into a computer-based communication intervention devel-
and 2017. For systematic and narrative reviews of recent oped to teach receptive labeling would facilitate engage-
clinical studies in music therapy for autism, including case ment and learning outcomes. Twenty-two children with
studies and single-group studies, see Geretsegger, Elefant, limited communication skills were randomly allocated to
Mössler, and Gold (2014) and James et al. (2015). five weeks of infant-directed song intervention or infant-
directed speech intervention (participants crossed over and
completed the other intervention after the follow-up assess-
Communication outcomes ment). Pre-recorded sung or spoken instructions directed
Social communication, language and speech deficits in aut- children to label garden creatures depicted in pictorial
ism are traditionally the primary target areas in music ther- form. Researchers found that, although children were more
apy. Speech production was the main treatment target goal engaged during the infant-directed singing condition
investigated in a few recent randomized clinical studies. (Simpson et al., 2013), there were no significant differences
Lim (2010) addressed acquisition of target words using a between the interventions regarding the improvement of
Neurologic Music Therapy technique called receptive labeling skills (Simpson et al., 2015).
“developmental speech and language training through
music (DSLM)” (Thaut & Hoemberg, 2014). The DSLM
technique is designed to use developmentally appropriate Social outcomes
musical materials and experiences to enhance speech and The main intervention approach to address social commu-
language development through singing, chanting, playing nication skills in autism is musical improvisation (Geret-
musical instruments, and combining music, speech, and segger et al., 2015; Wigram & Gold, 2006). Within this
movement. In this study, pre-recorded videos of songs therapeutic approach, music is utilized as a medium for
composed by the music therapist to include the target words self-expression, communication, and interaction, and for
were presented to children with ASD with various levels of the development or rehabilitation of appropriate socio-
impairment. The control conditions were pre-recorded emotional functioning.
videos of the speech stimuli using only the text of the songs Kim, Wigram, and Gold (2008) investigated the effects
or no treatment. The intervention was administered twice a of improvisational music therapy on joint attention beha-
day for three days, and post-tests were completed after only viors. Pre-school children with autism undertook 12 weekly
six training sessions. Results indicated that high and low- improvisational music therapy sessions and 12 weekly play
functioning participants in both music training and speech sessions with toys as the control condition (the order of
training increased verbal production in the post- interventions was counterbalanced with a one-week wash-
intervention assessment. Interestingly, low-functioning out period). The study reported improvement in initiating
children seemed to have greater gains in the music condi- joint attention bid, joint visual attention, eye contact dura-
tion. In a subsequent study, Lim and Draper (2011) added tion, and alternating eye contact during and after the treat-
music to an applied behavioral analysis verbal behavioral ment sessions in the music therapy condition but not in the
approach, a method that focuses on teaching children to control condition. In a subsequent analysis of this data,
associate a word or phrase to its functional meaning, Kim, Wigram, and Gold (2009) focused on the social-
including making requests, labeling objects, following motivational aspects of the interaction between the child
instructions, and completing sentences. The music inter- and the therapist during improvisational music therapy. For
vention consisted of singing the verbal instructions and that, behavioral microanalysis of video recordings of the
4 Music & Science

therapy sessions assessed outcomes such as frequency and musical activities. The intervention consisted of structured
duration of the participant’s emotional and motivational music activities designed to address shared attention, turn-
responsiveness (joy, emotional synchronicity, initiation of taking, response and initiation of joint attention, whereas
engagement). Results indicated that emotional and respon- the control group did not receive music therapy in addition
siveness behaviors were significantly more frequent and to standard care. Primary outcome measures were based on
longer in duration in the music therapy intervention than parent-report assessments. Results showed that parents per-
in the toy play condition. Therefore, the findings of these ceived a significant improvement in the quality of their
two studies are in favor of the use of improvisational music child’s social skills after the music therapy intervention
therapy to address joint attention and pro-social behaviors. in relation to the control condition. Assessment of video
However, replication is usually a problematic matter in recordings also suggested improvement in the level of
improvisational music therapy given the subjectivity and interpersonal engagement within music therapy sessions.
lack of protocol structure, which are characteristic of some Although encouraging, these findings must be interpreted
improvisational music therapy methods. To address this with caution given that the results were predominantly
issue, future clinical studies should provide open access based on parental assessment, which is prone to bias.
to the musical activities administered in the study, supple- Although these studies collectively provide some
mented with thorough descriptions of the intervention pro- encouraging results supporting the use of musical improvi-
tocols and justifications as to why the various musical sation in the treatment of social communication skills in
features were used to promote the desired non-musical individuals with ASD (for a systematic review, see Geret-
behavior and/or experience. segger et al., 2014), a recent large randomized controlled
Other controlled studies investigated the effect of trial cast doubt on this conclusion (Bieleninik et al., 2017).
improvisational music therapy on verbal, nonverbal and This study evaluated the effects of improvisational music
social communication skills. In Gattino, Riesgo, Longo, therapy on the generalized social communication skills of
Leite, and Faccini (2011), unstructured music improvisa- 364 children with ASD. Children allocated to the music
tion intervention was administered weekly for 16 weeks to therapy group received five months of weekly individual
a group of children diagnosed with autistic disorder, perva- therapy sessions consisting of joint musical activities aimed
sive developmental disorder not otherwise specified (PDD- to develop and enhance social skills such as affect sharing
NOS), and Asperger’s syndrome, while participants in the and joint attention. Children allocated to the control group
control group did not receive music therapy treatment in did not receive music therapy in addition to standard care.
addition to standard care. Results revealed no statistically The study found no significant differences in social com-
significant differences between groups in all outcomes munication skills among children in the improvisational
assessed. However, a subgroup analysis indicated that non- music therapy group when compared with standard care
verbal communication significantly increased after music alone. The authors acknowledged that the music therapy
intervention only for children with autism (but not PDD- intervention was not tightly controlled and perhaps not
NOS and Asperger’s). Given the small sample of children consistently implemented across multiple centers, corro-
with autism in the experimental group, limited conclusions borating our comment regarding replication of musical
can be made, warranting further investigation. In LaGasse improvisational intervention protocols.
(2014), children with autism received either music therapy
or a non-musical social skills intervention for five weeks.
The music therapy intervention consisted of structured Emotional outcomes
group musical experiences to promote cooperative play and Although social communication and interaction skills have
communication, whereas the control group participated in historically been the primary focus of music therapy inter-
similar group experiences with games and non-musical vention for ASD, self-awareness, emotional expression and
activities. Social outcome measures focused on eye gaze, understanding have also been targeted with music therapy
joint attention, and communication. Results indicated that (for review: Geretsegger et al., 2014; James et al., 2015).
children in the music therapy group significantly improved For instance, Allen and Heaton (2010) proposed a music-
in measures of joint attention with peers and eye gaze based intervention based on associative learning to help
towards other persons in relation to the control group. How- individuals with ASD and comorbid alexithymia to develop
ever, the study did not find significant differences between a vocabulary to express their experiences of music-evoked
groups in measures of communication, and the lack of emotions and to transfer this vocabulary and related socio-
standardized treatment protocols makes it difficult to inter- emotional skills to non-musical situations in everyday life.
pret and replicate the positive results. A family-centered Although promising, there is a paucity of controlled clinical
music therapy intervention was also used to assess social studies investigating the potential use of music therapy to
engagement in children with severe ASD (Thompson, facilitate emotional understanding in children with ASD.
Mcferran, & Gold, 2014). In this study, parents were Katagiri (2009) studied the effectiveness of a music-based
encouraged to actively participate in home-based music intervention to facilitate understanding of four different
therapy sessions and to collaborate with the therapist in the emotions: happiness, sadness, anger, and fear. In the music
Janzen and Thaut 5

therapy instrumental improvisational condition, structured symptoms but may be one of the earliest identifiable and
improvisations referencing the targeted emotions were pre- most prevalent features of autism (Bo, Lee, Colbert, &
recorded and presented to the participant while the therapist Shen, 2016; Dziuk et al., 2007; Hilton et al., 2007; Hilton,
gave verbal instructions about each of the emotions. In a Zhang, Whilte, Klohr, & Constantino, 2012; Ming, Brima-
second music therapy condition, the emotions were taught combe, & Wagner, 2007).
by singing a song composed by the therapist which Researchers examining home videos of infants who
included lyrics referencing the targeted emotions. The con- were later diagnosed with autism noted that these children
trol conditions included verbal instructions only or no pur- exhibited significant motor development delays and higher
poseful teaching of the targeted emotion. Results suggested frequency of unusual motor behaviors as early as in the first
that participants’ emotional understanding improved from six to nine months of life (e.g., problems in sequencing
pre-test to post-test in all conditions, with no clear indica- movements to grasp, crawl or walk) (Ozonoff, Heung,
tion that the music therapy conditions were superior to Byrd, Hansen, & Hertz-Picciotto, 2008; Teitelbaum, Tei-
control conditions in facilitating emotional expression and telbaum, Nye, Fryman, & Maurer, 1998; Weiss, Moran,
understanding. Parker, & Foley, 2013). Studies of infants at risk have also
demonstrated that early motor deficits are reliable predic-
tors of autism. For instance, manual-motor abilities such as
Expanding the clinical scope of practice grabbing and exploring objects (Gernsbacher, Sauer, Geye,
in music therapy for autism Schweigert, & Hill Goldsmith, 2008; Libertus, Sheperd,
The overview of recent controlled clinical studies provides Ross, & Landa, 2014; Sacrey et al., 2014), postural instabil-
an inconclusive scenario. While some studies support the ity and delayed posture development (Minshew, Sung,
use of music therapy to improve joint attention and general Jones, & Furman, 2004; Nickel, Thatcher, Keller, Woz-
pro-social behaviors, a recent rigorous multicenter study niak, & Iverson, 2013), poor muscle tone and motor imita-
did not arrive at the same conclusion (Bieleninik et al., tion (Brian et al., 2008; Landa, Gross, Stuart, & Bauman,
2017). Moreover, there is no clear indication that the music 2012; Rogers, 2009), and being delayed in the onset of
therapy interventions tested to date are superior to standard walking (Iverson & Wozniak, 2007), are consistently
care with respect to improving speech production, verbal/ reported in infants at risk for ASD. Furthermore, according
non-verbal communication and emotional understanding. to Sutra et al., 2007, motor skills at the age of two years are
Based on the current state of the clinical research on this the best predictors of later ASD diagnosis.
topic we propose a reassessment of the role that music can Motor development is closely intertwined with socio-
play in interventions to support healthy neurodevelopment emotional and cognitive development (Jones, Gliga, Bed-
in ASD, and suggest an expansion of the clinical scope of ford, Charman, & Johnson, 2014; Leonard & Hill, 2014).
practice in music therapy for autism. This expansion refers Recent studies have demonstrated that early motor impair-
to the need to address motor control impairments and atten- ments are strong predictors of social-communication defi-
tion deficits as additional key treatment targets. Our argu- cits developed later in childhood. Oral and manual motor
ment is based on newly emerging research evidence that impairments in infancy and toddlerhood have been linked
motor and attention deficits are directly implicated in the to communication delays (Bhat, Galloway, & Landa, 2012;
development of socio-communication and interaction skills Flanagan, Landa, Bhat, & Bauman, 2012; Lebarton & Iver-
and may be key indicators of structural and functional brain son, 2013; Libertus et al., 2014), speech fluency deficits
dysfunction in ASD. (Gernsbacher et al., 2008), and difficulties in processing
emotional facial expressions (Leonard, Bedford, Pickles,
& Hill, 2015; Leonard & Hill, 2014).
Implications of motor control deficits to socio-
There is also increasing evidence that motor control is
communication and interaction skills deeply implicated in social cognition (Cook, 2016; Mos-
Parents and clinicians often report that children with autism tofsky & Ewen, 2011; Trevarthen & Delafield-Butt, 2013).
display significant and pervasive motor impairments that Social skills depend on one’s performance of skilled and
are not associated with motor stereotypies. Motor deficits goal-oriented movements (praxis) as well as the under-
in autism include impairment in basic motor control such as standing of others’ movements and associated intentions.
clumsy gait (Fournier et al., 2010), poor balance and pos- Through observation and imitation of others’ actions, we
tural control (Bhat et al., 2011), delayed motor develop- build internal representations that are used as a template to
ment (Fatemi et al., 2012; Wang, Kloth, & Badura, 2014), interpret and predict other people’s intentions (Mostofsky
impairments to reaching and grasping (Sacrey, Germani, & Ewen, 2011). Researchers have found that dyspraxia is
Bryson, Zwaigenbaum, & Morris, 2014), and poor manual correlated with core social, communicative and behavioral
dexterity and coordination (Kindregan, Gallagher, & features of autism (Dowell, Mahone, & Mostofsky, 2009;
Gormley, 2015; Kushki, Chau, & Anagnostou, 2011). Evi- Dziuk et al., 2007). The correlation between abnormal
dence gathered in recent research suggests that motor praxis development with social/communicative impair-
impairments are not only co-occurring neurological ments in ASD suggests that “similar mechanisms may
6 Music & Science

underlie impaired development and execution of both in orienting and shifting attention can lead to severe chal-
motor skills and social/communicative skills in autism” lenges in social and communication engagement.
(Mostofsky & Ewen, 2011, p. 439). Additionally, new Individuals with ASD also have difficulties with selec-
methods of analysis have recently emerged allowing exam- tive attention, which is the ability to attend selectively to
ination of micro-movements (Torres & Denisova, 2016; meaningful sources of information while ignoring task-
Torres et al., 2013). Torres and colleagues suggested that irrelevant ones. Selective attention deficits have been
deficits in micro-movements are associated with difficul- demonstrated in tasks where the targets are defined by
ties in building internal representations of one’s own move- different shapes or colors (Allen & Courchesne, 2001,
ments, which in turn can affect the interpretation and 2003), or in tasks including competing distractors (Bel-
prediction of other people’s intentions. monte & Yurgelun-Todd, 2003; Burack, 1994). The
Therefore, this growing body of research indicates that impaired ability to filter task-irrelevant information and
the earliest observable symptoms of autism may involve control shifts of attention may be directly associated with
motor behavior and that impaired motor functions in ASD a variety of commonly observed autistic features, such as
are strong predictors of core social, communicative and stimulus overselectivity, perseveration, and narrowed inter-
behavioral features of autism. These findings collectively ests (Courchesne et al., 1994).
demonstrate that the motor system – which is known to be Studies have also found that children at risk and with
trainable – is a promising target for early intervention to ASD display significant impairments in initiating and
improve outcomes for individuals living with ASD. responding to joint attention (Presmanes, Walden, Stone,
& Yoder, 2007; Stone, McMahon, Yoder, & Walden, 2007;
Sullivan et al., 2007). Infant-initiated joint attention or
directing attention, refers to a child’s use of gestures, gaze
Implications of attention deficits to socio-
shifts, pointing and other cues to initiate a shared experi-
communication and interaction skills ence with others, whereas responding to joint attention or
Cognitive deficits are also widely observed in ASD, attention following, reflects the infant’s ability to orient
although generally regarded as secondary impairments or attention in response to a cue, such as someone’s gaze, head
comorbidities. New research evidence indicates, however, turn, point, or attention-directing utterance. Sullivan and
that attention dysfunction in ASD should be reconsidered colleagues (2007) examined responses to joint attention
as a primary feature due to its role in impairments in lan- (RJA) in children at risk of developing ASD at 14 months
guage and socio-emotional areas. Attention is critically and 24 months of age and found that 14-month-olds at risk
involved in all cognitive functions as the gateway to vol- already displayed significant deficits in responding to gaze
untary control of thoughts, emotions, and actions (Fan, shift cues and/or pointing cues. At 24 months of age, chil-
2013, p. 281). There is accumulating evidence from beha- dren at risk completely failed to respond to any RJA oppor-
vioral and neuroimaging studies that individuals with ASD tunity, indicating little developmental improvement in
display a wide range of attentional deficits across the many response behaviors in children at risk between 14 and 24
domains of attention functions (for review, see Allen & months. Moreover, the study reported that response to joint
Courchesne, 2001; Fan, 2013). Abnormal spatial orienta- attention performance predicted later receptive and expres-
tion of attention has been extensively reported in autism sive language performance, and reliably predicted later
literature. Researchers have noted, for instance, that indi- ASD diagnoses.
viduals with autism have limited capacity in disengaging Finally, comorbid attention deficit hyperactivity disor-
and shifting attention from a stimulus or task to another der (ADHD) is common and persists with age in individu-
(Landry & Bryson, 2004; Wainwright-Sharp & Bryson, als with ASD (Gargaro, Rinehart, Bradshaw, Tonge, &
1993). Orientation deficits have also been observed in tasks Sheppard, 2011; Yoshida & Uchiyama, 2004). Although
requiring rapid shifting of attention between sensory mod- previous diagnostic guidelines precluded diagnosing ASD
alities (Courchesne et al., 1994), between object features and comorbid ADHD, researchers have long reported rates
(Rinehart, Bradshaw, Moss, Brereton, & Tonge, 2001), and of comorbidity between autism and ADHD within the
between spatial locations (Townsend, Harris, & Courch- range of 14–78% (Corbett & Constantine, 2006; Johnston
esne, 1996). Orientation to human faces and social infor- et al., 2013; Sinzig, Walter, & Doepfner, 2009). A shared
mation have also been well-studied in ASD. Evidence cognitive phenotype is inattention. Sustained attention is
suggests that individuals with autism have particular diffi- consistently reported as one of the main deficits in ADHD
culty attending to and processing social stimuli (e.g., facial (Corbett & Constantine, 2006; Sinzig et al., 2009) and has
expressions, gestures) (Dawson et al., 2004; Dawson, also been reported in ASD (Allen & Courchesne, 2001;
Meltzoff, Osterling, Rinaldi, & Brown, 1998; Jones & Klin, Murphy et al., 2014; Johnson et al., 2007). A recent neu-
2013). Orientation and executive control functions of atten- roimaging study sought to elucidate commonalities and
tion are essential during the normal exchange of informa- differences in the underlying neurofunctional substrates
tion and turn-taking, which happens rapidly, frequently, of adolescents with ADHD or ASD during a vigilance task
and often unpredictably (Fan, 2013). Therefore, difficulties (Christakou et al., 2013). Results demonstrated that while
Janzen and Thaut 7

both disorders displayed the same type of underactivation associated with symptoms such as reduced exploration and
relative to controls in brain regions such as left- increased stereotyped and repetitive moments (Kaufmann
hemispheric dorsolateral prefrontal cortex (DLPFC) and et al., 2003), whereas lesions in the cerebellar lobules I–V
superior parietal cortex, ADHD patients showed a more are linked with vestibular, gross and fine motor control
severe DLPFC dysfunction associated with poorer perfor- (Fatemi et al., 2012; Gowen & Miall, 2007; Nayate, Brad-
mance measures in the sustained attention task, whereas shaw, & Rinehart, 2005). These studies collectively
patients with ASD specifically presented dysregulation of demonstrate that cerebellar lesions directly interfere with
the fronto-striato-cerebellar circuitry. a number of behaviors, including motor, cognitive and
In sum, an examination of the performance of individ- affective functions.
uals with autism across a variety of attentional operations The cerebellum has been long known for its importance
reveals a wide range of deficits, including difficulties to in motor learning, motor coordination and timing due to its
rapidly re-allocate attention to new spatial locations or tar- crucial role in prediction and adaptation mechanisms
get features, to filter task-irrelevant information, to share (Manto et al., 2012). That is, the cerebellum is key in
attention with others, and to coordinate attention between establishing sensory prediction errors by processing signal
people and objects – especially in social contexts. It is clear discrepancies between the expected sensory consequences
that deficits of attention affect one’s ability to attend, of a stimulus or movement and the actual sensory input.
engage, reciprocate, and learn, thus significantly impacting These error signals are essential for sensorimotor control
social development. and motor adaptation learning because they allow rapid
adjustments in the motor output and refinement of future
sensory predictions in order to reduce the variability of
The role of the cerebellum and dysfunctional brain
subsequent actions (Allen, Müller, & Courchesne, 2004;
connectivity Bastian, 2006; Koziol et al., 2014; Middleton & Strick,
There is strong evidence suggesting that the cerebellum is a 2000; Sokolov, Miall, & Ivry, 2017). Therefore, cerebellum
key player in the complex neural underpinning of ASD abnormalities would impair this prediction mechanism
with behavioral implications beyond the motor domain resulting in slower, more variable, inaccurate, and effortful
(Becker & Stoodley, 2013; D’Mello & Stoodley, 2015; responses (Allen & Courchesne, 2001; Courchesne et al.,
Fatemi et al., 2012; Koziol et al., 2014; Wang et al., 1994; Diamond, 2000; Sokolov et al., 2017). This assump-
2014). Histopathological changes in the cerebellum have tion has been confirmed in several studies showing, for
been observed in post-mortem studies where a decrease in instance, that patients with cerebellar lesions exhibit
the number of Purkinje cells in regions such as folia, vermis increased temporal variability in the production of periodic
and cerebellar hemispheres have been recurrently reported movements such as simple finger tapping (Diedrichsen,
(Becker & Stoodley, 2013; Fatemi et al., 2012). Structural Criscimagna-Hemminger, & Shadmehr, 2007; Ivry &
imaging studies of the cerebellum have repeatedly demon- Keele, 1989; Spencer, Ivry, & Zelaznik, 2005). Studies
strated that autism is associated with significant white mat- have also found evidence of atypical movement preparation
ter differences, abnormally lower cerebellar vermal in participants with autism characterized by a failure to
volumes, and reduced size of cerebellar hemispheres in adjust the motor preparation time in response to an
children and adults with ASD (Chukoskie, Townsend, & “expected” versus “unexpected” movement, suggesting a
Westerfield, 2013; Wang et al., 2014). Functional imaging deficit in anticipatory preparation of movement (Rinehart
research has also revealed task-dependent differences in et al., 2006; Rinehart, Bradshaw, Brereton, & Tonge,
cerebellar activation in a wide range of tasks (Anagnostou 2001).
& Taylor, 2011; Philip et al., 2012). In addition to motor behavior, current research has
There is ample evidence at multiple levels of inquiry firmly established the cerebellum’s critical role in modulat-
that abnormalities in the structure and function of the cer- ing cognitive functions (Buckner, 2013; Doya, 2000;
ebellum are associated with impaired neurobehavioral Koziol et al., 2014; Sokolov et al., 2017). Evidence of the
function. Bolduc and colleagues (2012) showed in a cohort involvement of the cerebellum in attention has emerged
of children between the ages of one and six years that from functional imaging studies demonstrating, for
reduced vermis volume was associated with impairment instance, that attention and motor performance activate
of global development, cognition, expressive language, distinct cerebellar regions – attention tasks engage areas
gross and fine motor skills, behavioral problems, and located in the superior posterior cerebellar hemispheres and
higher scores in autism spectrum screening tests. Studies those involved in motor tasks are located in the anterior
have also reported that lesions in the cerebellum hemi- cerebellar hemisphere (Allen, Buxton, Wong, & Courch-
spheres at earlier ages can result in language delay, whereas esne, 1997; Allen & Courchesne, 2003; see also Mosconi,
damage to the vermis results in withdrawn social behavior, Mohanty, Greene, Cook, Vaillancourt, & Sweeney, 2015).
impaired gaze, anxiety and stereotyped behavior (Riva & Studies have also shown that people with autism and
Giorgi, 2000; Wells, Walsh, Khademian, Keating, & patients with acquired cerebellar lesions are similarly
Packer, 2008). In autism, reduced vermal VI–VII is impaired in tasks requiring rapid voluntary shifts of
8 Music & Science

attention between stimuli, which indicates that cerebellum If this is indeed the case, then music-based developmental
abnormalities result in deficits in the timely and accurate training for attention and motor control may receive a
control of the direction of attention (Akshoomoff & critical new functional role based on evidence that the
Courchesne, 1994; Courchesne et al., 1994). Structural and cerebellum is significantly involved in virtually all
functional cerebellar abnormalities in autism have been music-related tasks, and that music increases synchronized
also linked to deficits in spatial orientation (Harris, Courch- activation of widespread networks in the brain.
esne, Townsend, Carper, & Lord, 1999), selective attention Music, like other higher cognitive tasks, requires the
(Allen & Courchesne, 2003) and sustained attention (Chris- activation of different cortical and subcortical regions in
takou et al., 2013; Murphy et al., 2014). Another important an organized and synchronized way. Research has demon-
aspect of the cerebellum, in addition to cognition, is its role strated that listening to auditory rhythmic sequences such
in affective responses. An important network loop connects as music increases connectivity in extensive reciprocal
the vermal regions in the cerebellum associated with emo- cortico-subcortical projections (Alluri et al., 2017; Bhatta-
tional functions to several cortical and subcortical systems, charya & Petsche, 2005; Ohnishi et al., 2001; Paraskevo-
among them the ventral insula, which is also highly poulos, Chalas, & Bamidis, 2017; Thaut, Trimarchi, &
involved in emotional functions. Therefore, the cerebellum Parsons, 2014) and induces cortico-cortical coherence in
may also be considered an important component in a sub- auditory and motor areas (Fujioka, Trainor, Large, & Ross,
cortical network for subjective and social emotion process- 2012; Lee & Noppeney, 2011; Nozaradan, 2014). Listening
ing in ASD (Bird et al., 2010; Reetz et al., 2012). to one’s favorite song alters connectivity between auditory
Studies often report that abnormal cerebellar activity is brain areas and the hippocampus (Wilkins, Hodges, Laur-
associated with atypical activation of cortical regions, sug- ienti, Steen, & Burdette, 2014). There is also growing evi-
gesting that cerebellar abnormalities disrupt optimization dence that auditory rhythmic stimuli processing
of both structure and function in specific cerebro-cerebellar (Nozaradan, Schwartze, Obermeier, & Kotz, 2017; Teki,
circuits (D’Angelo & Casali, 2013; D’Mello & Stoodley, Grube, & Griffiths, 2012) and auditory-motor entrainment
2015; Middleton & Strick, 2000). The cerebellum is inter- tasks rely heavily on extensive cortico-subcortical-cortical
connected with several different regions of the cerebral functional networks, including cortico-cerebellar circuits
cortex by discrete circuits or “loops” that connect a diverse (Chauvigné, Gitau, & Brown, 2014; Thaut et al., 2009).
set of cerebral cortical areas with the cerebellum and the Moreover, recent imaging research has indicated that the
basal ganglia (for review, see D’Angelo & Casali, 2013; cerebellum is not only involved in temporal perception and
D’Mello & Stoodley, 2015; Middleton & Strick, 2000). sensorimotor synchronization tasks, but that the cerebellum
This extensive connectivity provides anatomical substrates has a critical role in processing melodic properties and
by which dysfunctional cerebro-cerebellar connectivity speech information (e.g., prosody) (Callan, Kawato, Par-
could be involved in the large spectrum of symptoms that sons, & Turner, 2007; Parsons, Petacchi, Schmahmann, &
comprise ASD (D’Mello & Stoodley, 2015). Studies have Bower, 2009; Thaut et al., 2014). Putting together a new
repeatedly reported significant overconnectivity in net- framework that links the neural basis of music processing
works between the cerebellum and motor cortices (D’Mello to a new look at the behavioral and neurological deficits in
& Stoodley, 2015; Khan et al., 2015; Mostofsky & Ewen, the developing brain in ASD, some more specific directions
2011) and underconnected prefrontal-cerebellar attention towards the use of music in therapy for autism may be
networks in individuals with ASD in relation to healthy finally laid out.
controls (Belmonte, 2004; Fitzgerald et al., 2015; Keown
et al., 2013; Murphy et al., 2014). Dysfunctional connec-
tivity between the cerebellum and cerebral cortical brain
Neurobiological foundations of rhythm-
regions has been demonstrated in tasks involving finger
sequencing (Mostofsky et al., 2009), visual processing
based interventions
(Villalobos, Mizuno, Dahl, Kemmotsu, & Müller, 2005), Auditory rhythm has a profound effect on the motor sys-
language (Hodge et al., 2010; Just, Cherkassky, Keller, & tem. The auditory system is well known for its ability to
Minshew, 2004), attention (Allen et al., 1997; Belmonte & detect temporal patterns in auditory signals with extreme
Yurgelun-Todd, 2003; Murphy et al., 2014), social process- precision and rapidly construct stable temporal templates
ing (Jack & Pelphrey, 2015; Sokolov et al., 2012), and (for review, see Thaut & Kenyon, 2003). Moreover, the
executive function (Gilbert, Bird, Brindley, Frith, & Bur- auditory system has richly distributed fiber connections
gess, 2008; Koziol et al., 2014) (for review, see Stoodley & to motor centers from the spinal cord upward on to brain
Schmahmann, 2009). These findings therefore strongly stem, subcortical, and cortical levels (Felix, Fridberger,
indicate that inefficient connectivity may be a hallmark Leijon, Berrebi, & Magnusson, 2011; Koziol & Budding,
of ASD. 2009; Schmahmann & Pandya, 1995). This distributed and
In light of these findings, attention and motor deficits in reciprocal connectivity between the auditory and motor
ASD could be considered critical indicators of cerebellar systems explains why auditory rhythmic cues entrain motor
dysfunction and abnormal cortico-cerebellar connectivity. responses.
Janzen and Thaut 9

Auditory-motor entrainment occurs when the firing 1999; Jones et al., 1982), internal fluctuations in attentional
rates of auditory neurons, triggered by auditory rhythm energy entrain with the temporal patterns of external
such as music, entrain the firing patterns of motor neurons. events, generating expectations about when future events
For example, it has been demonstrated that finger and arm will occur. Consequently, if an event coincides with this
movements instantaneously entrain to the period of a rhyth- attention peak, its processing is facilitated. Indeed, people
mic stimulus (e.g., metronome beat) and maintain phase are most accurate in judging pitch changes when they occur
synchrony to the metronome even when subtle tempo on the beat of the music than at unexpected time points
changes are introduced (Thaut, Miller, & Schauer, 1998). (Honing et al., 2014; Jones, 1992; Lange, 2010; Schmuck-
The clinical importance of auditory-motor entrainment is ler & Boltz, 1994). Studies have also shown that the capac-
that the continuous time reference provided by the rhythm ity to focus attention on relevant stimuli while ignoring
of the music primes the motor system into a state of readi- distracting information is possible due to an anticipatory
ness by providing anticipatory time cues that allow move- attentional mechanism that suppresses the processing of
ment anticipation and motor planning to occur based on distracting information (Fu et al., 2001; Johnson & Zatorre,
expectations of the duration of the time cues, thus increas- 2005). It is possible, therefore, that the rhythmic structure
ing response quality (Thaut & Hoemberg, 2014; Thaut, of the music modulates these anticipatory mechanisms,
McIntosh, & Hoemberg, 2015). facilitating the process of attentionally relevant informa-
The ability to use rhythm to prime the motor system and tion. The impact of rhythmic entrainment on attention has
re-program the execution of a motor pattern has opened the been examined in cross-modal and cross-domain studies.
possibility of using complex auditory stimuli such as music Recently emerging electrophysiological and behavioral
and rhythm-based interventions for movement therapy and evidence indicates that auditory rhythmic cues can entrain
rehabilitation (Thaut, Miltner, Lange, Hurt, & Hoemberg, visual attention and facilitate processing of visual stimuli
1999). Rhythm-based techniques use rhythmic patterns to (Bolger, Trost, & Schön, 2013; Escoffier, Herrmann, &
provide a continuous time reference and generate expecta- Schirmer, 2015; Miller, Carlson, & McAuley, 2013;
tions for when an object will occur or when a movement Sacrey, Clark, & Whishaw, 2009). Indeed, when one is
needs to be performed, thus allowing preparation and listening to an auditory rhythmic sequence, processing of
increasing the quality and precision of the responses. For visual stimuli presented on the beat is facilitated, support-
instance, rhythm-based interventions such as Rhythmic ing the idea that entrainment of neuronal oscillations is a
Auditory Stimulation (RAS) involve the utilization of general mechanism through which the brain uses predictive
rhythmic cues (metronome or rhythmically accentuated cues to optimize attention and stimulus perception (Bolger
music with embedded metronome clicks) to facilitate gait et al., 2013).
and improve the kinematic stability of walking movements Evidence of the effects of rhythmic entrainment on
(Thaut & Hoemberg, 2014). There is a strong body of attention has supported the development of therapy tech-
research evidence that RAS improves movement in patients niques such as Musical Attention Control Training
with movement disorders (for review, see Thaut, 2015; (MACT), which involves perceptual and structured musical
Thaut & Abiru, 2010; Thaut et al., 2015). Specifically, it exercises built on rhythmic, melodic and harmonic patterns
has been demonstrated that rhythmic auditory cues have to train sustained, selective, divided, focused, and shifting
immediate effects on gait by increasing speed, stride attention (Thaut & Hoemberg, 2014). A recent pilot study
length, and improving symmetry and stability (Arias & tested whether this intervention would be effective to
Cudeiro, 2010; McIntosh, Brown, Rice, & Thaut, 1997; improve attention functioning in adolescents with neurode-
Thaut et al., 1996; Thaut et al., 1999; Thaut, McIntosh, & velopmental delays, including autism (Pasiali, LaGasse, &
Rice, 1997). In upper limb rehabilitation, rhythmic cueing Penn, 2014). The study found significant improvements in
is effective to improve spatiotemporal control, reduce measures of selective attention and rapid shifting of atten-
movement variability, smooth reaching trajectories, and tion after eight treatment sessions. This promising result is
reduce reliance on compensatory movements (Malcolm, based on a small sample and a relatively short intervention
Massie, & Thaut, 2009; Thaut, Kenyon, Hurt, McIntosh, period, encouraging replication in larger controlled trials.
& Hoemberg, 2002; Luft et al., 2004). The effect of auditory rhythm has also been demon-
Rhythmic entrainment extends beyond motor control. strated in the speech domain (Cason, Astésano, & Schön,
Music is inherently temporal and sequential, and the reg- 2015; Cason & Schön, 2012; Fujii & Wan, 2014; Schön &
ularity of rhythmic pulses generates temporal expectations Tillmann, 2015). In a recent study, participants listened to a
that allow us to predict when the next beat of the music will metrical sequence immediately followed by a word or a
occur and consequently direct our attention to a particular sentence and were asked to detect whether the last syllable
moment in time (Honing, Bouwer, & Háden, 2014; Large of the sentence contained the sound |a|. It was reported that
& Palmer, 2002). There is a large body of research demon- reaction times were faster when the rhythmic cueing
strating that rhythm is key in tuning and modulating atten- matched the prosody of the sentence (Cason & Schön,
tion (Drake, Jones, & Baruch, 2000; Jones, 1992). 2012). In a subsequent study, researchers used the same
According to dynamic attending theory (Large & Jones, protocol, however, one group of participants received
10 Music & Science

audio-motor training before the task, which consisted of understand the potential impact of addressing motor and
reproducing the cueing rhythmic patterned vocally using attention deficits to the healthy neurodevelopment of per-
different sounds to distinguish between strong and weak sons with ASD.
beats (e.g., “ba ba KA”). Results indicated that rhythmic
audio-motor training enhanced the priming effect of the Contributorship
rhythmic cues (Cason et al., 2015). This theoretical frame- MT and TBJ conceived the article. TBJ researched the literature
work was also applied in the development of therapeutic and wrote the first draft of the manuscript. Both authors reviewed
interventions that combine intonation (singing) and the use and edited the manuscript and approved the final version of the
of a pair of tuned drums to facilitate speech output (e.g., manuscript.
Thaut, 1984; Wan et al., 2011). For instance, in an initial
Declaration of conflicting interests
proof-of-concept study (Wan et al., 2011), six non-verbal
The authors declared no potential conflicts of interest with respect
children with autism participated in music therapy sessions
to the research, authorship, and/or publication of this article.
wherein the therapist introduced the target word or phrase
to be learned by simultaneously singing the words and Funding
tapping the drums. The child would then repeat and repro-
The authors received no financial support for the research, author-
duce the target word/phrase on their own. Results showed ship, and/or publication of this article.
that participants significantly improved in their ability to
articulate words and phrases after the intervention, suggest- Peer review
ing that auditory-motor training may be effective in facil- Pam Heaton, Goldsmiths, University of London, Department of
itating speech production, thus warranting replication in a Psychology.
larger controlled study. These studies collectively demon- Thomas Bergmann, independent scholar.
strate that auditory-motor entrainment may facilitate and One anonymous reviewer.
improve language processing and acquisition, suggesting
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