music in the treatment of neurological language and … 2012, 26 (1), 1–19 music in the treatment...

21
This article was downloaded by: [University of Groningen] On: 14 February 2012, At: 07:32 Publisher: Psychology Press Informa Ltd Registered in England and Wales Registered Number: 1072954 Registered office: Mortimer House, 37-41 Mortimer Street, London W1T 3JH, UK Aphasiology Publication details, including instructions for authors and subscription information: http://www.tandfonline.com/loi/paph20 Music in the treatment of neurological language and speech disorders: A systematic review Joost Hurkmans a , Madeleen de Bruijn a , Anne M. Boonstra a , Roel Jonkers b , Roelien Bastiaanse b , Hans Arendzen c & Heleen A. Reinders-Messelink a a Rehabilitation Center “Revalidatie Friesland”, Beetsterzwaag, The Netherlands b School for Behavioural and Cognitive Neuroscience (BCN), University of Groningen, Groningen, The Netherlands c Leiden University Medical Center, Department of Rehabilitation Medicine, Leiden, The Netherlands Available online: 06 Oct 2011 To cite this article: Joost Hurkmans, Madeleen de Bruijn, Anne M. Boonstra, Roel Jonkers, Roelien Bastiaanse, Hans Arendzen & Heleen A. Reinders-Messelink (2012): Music in the treatment of neurological language and speech disorders: A systematic review, Aphasiology, 26:1, 1-19 To link to this article: http://dx.doi.org/10.1080/02687038.2011.602514 PLEASE SCROLL DOWN FOR ARTICLE Full terms and conditions of use: http://www.tandfonline.com/page/terms-and- conditions This article may be used for research, teaching, and private study purposes. Any substantial or systematic reproduction, redistribution, reselling, loan, sub-licensing, systematic supply, or distribution in any form to anyone is expressly forbidden. The publisher does not give any warranty express or implied or make any representation that the contents will be complete or accurate or up to date. The accuracy of any instructions, formulae, and drug doses should be independently verified with primary sources. The publisher shall not be liable for any loss, actions, claims, proceedings, demand, or costs or damages whatsoever or howsoever caused

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This article was downloaded by: [University of Groningen]On: 14 February 2012, At: 07:32Publisher: Psychology PressInforma Ltd Registered in England and Wales Registered Number: 1072954Registered office: Mortimer House, 37-41 Mortimer Street, London W1T 3JH, UK

AphasiologyPublication details, including instructions for authors andsubscription information:http://www.tandfonline.com/loi/paph20

Music in the treatment ofneurological language and speechdisorders: A systematic reviewJoost Hurkmans a , Madeleen de Bruijn a , Anne M. Boonstraa , Roel Jonkers b , Roelien Bastiaanse b , Hans Arendzen c &Heleen A. Reinders-Messelink aa Rehabilitation Center “Revalidatie Friesland”,Beetsterzwaag, The Netherlandsb School for Behavioural and Cognitive Neuroscience (BCN),University of Groningen, Groningen, The Netherlandsc Leiden University Medical Center, Department ofRehabilitation Medicine, Leiden, The Netherlands

Available online: 06 Oct 2011

To cite this article: Joost Hurkmans, Madeleen de Bruijn, Anne M. Boonstra, Roel Jonkers,Roelien Bastiaanse, Hans Arendzen & Heleen A. Reinders-Messelink (2012): Music in thetreatment of neurological language and speech disorders: A systematic review, Aphasiology,26:1, 1-19

To link to this article: http://dx.doi.org/10.1080/02687038.2011.602514

PLEASE SCROLL DOWN FOR ARTICLE

Full terms and conditions of use: http://www.tandfonline.com/page/terms-and-conditions

This article may be used for research, teaching, and private study purposes. Anysubstantial or systematic reproduction, redistribution, reselling, loan, sub-licensing,systematic supply, or distribution in any form to anyone is expressly forbidden.

The publisher does not give any warranty express or implied or make anyrepresentation that the contents will be complete or accurate or up to date. Theaccuracy of any instructions, formulae, and drug doses should be independentlyverified with primary sources. The publisher shall not be liable for any loss, actions,claims, proceedings, demand, or costs or damages whatsoever or howsoever caused

arising directly or indirectly in connection with or arising out of the use of thismaterial.

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APHASIOLOGY, 2012, 26 (1), 1–19

Music in the treatment of neurological language and speechdisorders: A systematic review

Joost Hurkmans1, Madeleen de Bruijn1, Anne M. Boonstra1,Roel Jonkers2, Roelien Bastiaanse2, Hans Arendzen3,and Heleen A. Reinders-Messelink1

1Rehabilitation Center “Revalidatie Friesland”, Beetsterzwaag, The Netherlands2School for Behavioural and Cognitive Neuroscience (BCN), University ofGroningen, Groningen, The Netherlands3Leiden University Medical Center, Department of Rehabilitation Medicine,Leiden, The Netherlands

Background: Acquired brain injury resulting from a stroke can result in impairments in,among other things, communication. Music therapy has been used in rehabilitation tostimulate brain functions involved in speech. The use of elements of music is well knownand more often used in the treatment of aphasia and apraxia of speech.Aims: The aim of the study is to synthesise studies on the effect of music parameters inthe treatment of neurological language and speech disorders. In addition, possible mech-anisms that explain recovery are investigated.Methods & Procedures: Search terms were formulated based on the research question.A systematic search in databases was performed using these search terms. Then inclusioncriteria were formulated and articles meeting the criteria were reviewed on patient char-acteristics, interventions, and methodological quality.Outcomes & Results: A total of 1250 articles have been selected from the databases, ofwhich 15 were included in this study. The Melodic Intonation Therapy was the moststudied programme. Melody and rhythm were the music interventions that have beenapplied the most. Measurable recovery has been reported in all those reviewed studiesusing music in the treatment of neurological language and speech disorders. In threestudies research was also conducted into the mechanisms of explanation of the measuredrecovery. However, the methodological quality of the investigated studies was rated as“low”, using the ASHA level of evidence indicators for judging research.Conclusions: Although treatment outcomes were reported as positive in all of the15 reviewed studies, caution should be used relative to conclusions about the effective-ness of treatments that incorporate components of music with neurologically impairedindividuals. Methodological quality was rated as low and interpretations of mecha-nisms of recovery were contradictory. Suggestions for standardising and improvingmethodological quality drawn from the analysis are presented.

Keywords: Music; Speech disorders; Treatment.

Address correspondence to: Joost Hurkmans, MA, Rehabilitation Center “Revalidatie Friesland”,P.O. Box 1, 9244 CL Beetsterzwaag, the Netherlands. E-mail: [email protected]

A revised version of this article has been published in a Dutch journal (Stem Spraak- en Taalpathologie).

© 2012 Psychology Press, an imprint of the Taylor & Francis Group, an Informa businesshttp://www.psypress.com/aphasiology http://dx.doi.org/10.1080/02687038.2011.602514

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2 HURKMANS ET AL.

Musical structures and language structures have many similar features, which gen-erates continuous research interest. Studying the relation between these two entitiesdates back to the nineteenth century. Gamer (1892) studied animal noise and thehuman voice and transformed these sounds to musical instruments like piccoloand other flutes. More recently, sophisticated techniques like event-related poten-tial (ERP), positron emission tomography (PET), and functional magnetic resonanceimaging (fMRI) have been used to study perceptual elements of music and language inorder to gather information on the functional and neural architecture of both domains(Brown, Martinez, & Parsons, 2006; Jeffries, Fritz, & Braun, 2003; Patel, 2003).

Communication impairments resulting from neurological damage were alreadybeing studied in the nineteenth century. Broca (1861) reported language disorders inpatients who suffered from a stroke. In 1914 Déjérine, as one of the first researchers,observed a superior singing ability in aphasia. Subsequently, more researchersreported data relative to severely impaired patients who barely had the ability to speakin spontaneous speech, but were able to produce well-articulated, linguistically accu-rate words while singing familiar songs that had been learned prior to their stroke(e.g., Cohen & Ford, 1995; Gerstman, 1964; Hébert, Racette, Gagnon, & Peretz,2003; Racette, Bard, & Peretz, 2006; Straube, Schultz, Geipel, Mentzel, & Miltner,2008). Clinical applications using musical elements were then a natural consequencein aphasia intervention. Melody and rhythm have been used by non-fluent speak-ers to enhance speech production or to improve speech fluency. The most commontherapy intervention using melody and rhythm is the Melodic Intonation Therapy(MIT; Albert, Sparks, & Helm, 1973). MIT consists of speaking with a simplifiedand exaggerated prosody, characterised by a melodic component (two notes, high andlow) and a rhythmic component (two durations, long and short). Various music ther-apy approaches are aimed at verbal expression and communication as well. Therapymethods using different musical elements, like melody, rhythm, dynamics, tempo, andmetre, to regain speech production need not automatically contain music therapy. Forexample, the MIT is not a therapy of music as indicated by the original developers ofthe treatment approach.1 However, several music therapy variations have been devel-oped mostly based on MIT principles (e.g., Modified Melodic Intonation Therapy,MMIT, Baker, 2000; and Singen Intonation Prosodie Atmung RhytmusübungenImprovationen, SIPARI, Jungblut & Aldridge, 2004). Similar to MIT, the MMIT pro-gramme is also based on repetition of phrases set to musical structures. However, thephrases in MMIT are more melodic in structure and less like the “sprechgesang” styleof intonation adopted in MIT (Baker, 2000). A rather new therapy programme inwhich music performs a major role is Speech-Music Therapy for Aphasia (SMTA; deBruijn, Zielman, & Hurkmans, 2005). SMTA is a treatment programme with a com-bination of speech language pathology and music therapy. SMTA has componentssimilar to MIT; however, the most important difference is the expanding of musicalelements like dynamics, tempo, and metre.

Therapy interventions using musical elements to remediate language and speechabilities have been developed from clinical practice, including SMTA. During the past10 years positive outcomes have been experienced by patients with neurological com-munication deficits; however, evidence of the effectiveness of treatment based on thecomponents of music remains unknown. Therefore a systematic review of literature

1We refer to the appendix for a definition of music therapy and a more concrete discussion of the variousparameters.

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MUSIC IN NEUROLOGICAL SPEECH DISORDERS 3

was needed. The purpose of the study in question is a general review and meaningfulbefore studying the effect of SMTA in future research.

This article reviews the existing literature on the effect of music in the treatment ofpatients with neurological language and speech disorders. Studies were considered forthis review if published in a peer-reviewed journal prior to 2009. In addition, mech-anisms of recovery explaining positive effects of the use of music in the treatment ofpatients with neurological language and speech disorders were evaluated.

METHOD

A list of search terms was set-up in order to systematically search in the litera-ture: <language disorders>, <speech disorders>, <communication disorders>,<aphasia>, <articulation disorders>, <apraxia>, <speech>, <language>,<verbal>, <oral>, <communication>, <motor speech disorder> and <music>.These terms were linked using the combinations of: (1) <language disorders> or<speech disorders> or <communication disorders> or <aphasia> or <articulationdisorders>, (2) <apraxia> and (<speech> or <language> or <verbal> or <oral>or <communication> or <motor speech disorder>), (3) <music> and (#1 or #2).We searched in the following databases: PubMed, CINAHL, PsycINFO, andEMBASE. Reference manager was used to remove duplicates. Subsequently, inclu-sion criteria were formulated to judge whether an article contributes to the researchquestions: (1) effect controlled by measurements before and after intervention,(2) musical elements as a form of therapy of language and speech disorders causedby non-congenital neurological disorders (e.g., CVA and TBI), (3) adults, (4) any ofthe linguistic modalities, (5) language restrictions: only English, French, German,and Dutch articles were reviewed. Music was defined as follows: one or moreof the following musical elements: rhythm, melody, accent, practised in vocal orinstrumental form. Language and speech disorders were defined as follows: disordersof production as well as disorders in reception in all linguistic modalities (speech,reading, writing, and auditory language comprehension). Particular exclusion criteriawere also delineated: amusia, language acquisition disorders, stuttering, psychiatricdiseases, dementia, hearing disorders (including word deafness), voice disorders,healthy participants (including professional musicians), epilepsy, and autism.

Two authors (JH and MB) reviewed the abstracts of the selected articles indepen-dently of each other. Various articles needed to be read more extensively because it wasunclear from the abstract if they met the inclusion criteria. Both selections were thencompared. When in doubt, the two reviewers consulted with a third reviewer (HR).

A list of variables was compiled in order to describe the articles. A short pilot studywas needed to help determine this list. JH and MB independently assigned the vari-able values by reading two selected articles. Upon completion of this pilot by groupdiscussion, the list of variables was established. As a result of the completed list, thedescription of articles contains the following three variables: (1) patient characteris-tics: age, gender, education, dominance, aetiology, speech-language diagnosis, timepost onset, severity of the speech- language impairment, and musical background,(2) intervention and outcome variables: objective of the treatment, level of outcomemeasurements in terms of international classification of functioning (ICF), treatmentprogramme/method (including condition, schedule, linguistic level, musical parame-ters) and other language, speech and music therapy interventions, (3) methodologicalquality: study design, blinding, sampling, group/participant comparability, treatment

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4 HURKMANS ET AL.

fidelity, outcomes, significance, precision, and intent-to-treat. These quality indicatorsoriginate from the guidelines of the ASHA levels-of-evidence scheme. A study received1 point for each quality indicator if the highest level of quality was incorporated. In thecases of indicators with multiple possible levels, only the highest level of quality gotcredit. Table 1 outlines the indicators with a description and quality marker.

Variables had to be reported in more than 50% of the articles in order to be includedin the results of this review. When information on a variable was missing in > 50% ofthe articles, the variable was excluded from the analyses because the lack of infor-mation would be too large to make firm conclusions. All the information of patientcharacteristics, intervention and outcome variables, and methodological quality isbased on information provided by the authors of the articles. Any lack of informationis also indicated as not reported.

RESULTS

The combination of search terms yielded 1250 articles. However, 94% of the articleswere excluded since they concerned no therapy study and/or other participant groupthan patients with language and speech disorders caused by neurological disorders.A total of 50 articles were close to being included but were dropped because no effectcontrolled by measurements before and after intervention was included in the study.Two authors (JH and MB) identified a total of 18 articles that met initial inclusioncriteria, with agreement in most of the cases. During the selection of the articles threewere rejected upon review of the full text and after consultation with a third reviewer(HR). Thus 15 studies were used in the review. The results of 583 patients are describedbelow of which 82% were depicted in the study of Popovici (1995).

Patient characteristics

Table 2 provides an overview of the 15 studies and corresponding patient characteris-tics. Three variables were not reported 50% of the time, not meeting the 50% criteria.These included education, dominance, and musical background. Thus these variableswere excluded from this review. Various ages, from 18 years onwards, were representedin the studies through an adequate spreading, meaning that all age groups were equallydivided. Four articles did not report any gender information. In the other studies bothsexes were represented in group studies and case series. Notable from Popovici’s study(1995) is the high percentage of males: 77%. It has not been reported whether thishad any influence on the result of the study. In all (but one) studies, stroke was thecause of speech disorder of the treated patients (in five studies in combination withother medical diagnoses). The exception was the study of Baker (2000) who describedtwo patients with traumatic brain injury (TBI). In nine studies the location of thelesion was reported; these patients suffered from a left hemisphere stroke. The speech-language diagnosis was non-fluent aphasia (Broca’s aphasia or global aphasia) in13 studies, with an accompanying apraxia of speech in 2 studies. In two articles (Cohen& Masse, 1993; Tamplin, 2008) patients with dysarthria were also investigated. Mostpatients were treated in the chronic phase of recovery, more than 1 year post onset, forsevere language and speech disorders (not explicitly defined).

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MUSIC IN NEUROLOGICAL SPEECH DISORDERS 5

TAB

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6 HURKMANS ET AL.

TAB

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notr

epor

ted,

TB

I=tr

aum

atic

brai

nin

jury

,CVA

=ce

rebr

ovas

cula

rac

cide

nt,P

D=

Par

kins

on’s

dise

ase,

MS

=m

ulti

ple

scle

rosi

s,C

P=

cere

bral

pals

y,A

oS=

apra

xia

ofsp

eech

.

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MUSIC IN NEUROLOGICAL SPEECH DISORDERS 7

Interventions

Table 3 is a summary of information for therapeutic interventions relative to the15 studies. One variable did not meet the 50% criterion of reporting: other language,speech, and music therapy interventions. This variable was therefore not reported inthis overview.

Nine studies evaluated the effectiveness of MIT (Albert et al., 1973). MIT wastherefore the most studied treatment programme. Individual treatment (speech-language therapy as well as music therapy) was the most studied treatment condition:in 12 studies patients received individual treatment. Combinations of SLT and musictherapy have not been reported. The schedules of the treatment intervention varied.MIT prescribes an intensive schedule of twice a day, 30 minutes each, five times a week.However, this guideline was not always followed in the studies evaluating the effective-ness of MIT; generally less therapy than recommended was given. All objectives havebeen formulated at the impairment level. At this level, sentences were studied the mostat the linguistic levels, and melody and rhythm were the most frequently used musicalparameters.

Methodological quality

An overview of the quality indicators for all 15 studies is presented in Table 4. Therewas high agreement between JH and MB in classifying each article. The method-ological quality of the studies varied with scores ranges from 0–4 (on a scale of0–9). Five studies obtained a score of 0, and two studies obtained a score of 4. Thescores of the other studies were in this range. The most frequently used study design(N = 9) was case series. None of the studies involved a randomised controlled trial(RCT), and mention of blinding, the use of intention-to-treat, and precision is notreported. In eight studies information on validity and reliability of the outcome mea-sures was missing. All studies used multiple outcome measures without classificationof main study parameters. Five studies used comprehensive language tests as out-come measure like the Boston Diagnostic Aphasia Examination (BDAE; Goodglass& Kaplan, 1972) and the Aachener Aphasia Test (AAT; Huber, Poeck, & Williams,1984). No distinction has been reported in related (speech parameters) and unrelatedmeasures (non-speech parameters like reading, writing, and auditory comprehension).In eight studies no p-values were reported.

Effectiveness of intervention

An overview of the effectiveness of interventions of the 15 studies is summarised inTable 5. All studies reported positive results. It is difficult to define the exact number ofpatients that improved because the depiction of the results varied extensively. Detailedinformation on which patients improved at which outcome measures was lacking inmost studies evaluating more than one patient (group studies and case series). It is dif-ficult to state the effectiveness of the intervention because all studies included multipleoutcome measures without defining the primary study outcome measure. Cohen andMasse (1993), for instance, reported improvement at verbal intelligibility but none atspeech rate.

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8 HURKMANS ET AL.

TAB

LE3

Inte

rven

tio

nan

do

utc

om

eva

riab

les

Stu

dyO

bjec

tive

ICF

Tre

atm

ent

prog

ram

me

Con

diti

onS

ched

ule

Lin

guis

tic

leve

lM

usic

alpa

ram

eter

s

Bak

er,2

000

Sim

ilari

ties

and

diff

eren

ces

betw

een

MIT

and

MM

IT

1M

MIT

ind

(MT

)3–

4xpw

,1ho

ur,

5–23

mon

ths

Wor

dsan

dse

nten

ces

Fam

iliar

song

s,m

elod

yan

drh

ythm

Bel

inet

al.,

1996

Mec

hani

sms

ofre

cove

ry1

MIT

ind

(SLT

)1–

108

mon

ths,

freq

uenc

yan

dse

ssio

nti

me,

nr

Sent

ence

sM

elod

yan

drh

ythm

But

tet

&A

uber

t,19

80E

ffec

tive

ness

MIT

1M

ITin

d(S

LT)

4–10

pw,2

0m

in,

2–8

mon

ths

Sent

ence

sM

elod

yan

drh

ythm

Coh

en,1

992

Eff

ect

ofm

usic

onsp

eech

1G

roup

sing

ing

grou

p3x

pw,3

0m

in,

3w

eeks

Tex

tF

amili

arso

ngs,

mel

ody

and

rhyt

hmC

ohen

&M

asse

,19

93E

ffec

tof

rhyt

hman

dsi

ngin

gon

spee

ch1

Sing

ing

grou

pan

drh

ythm

grou

pgr

oup

2xpw

,30

min

,9

wee

ksP

hone

mes

,wor

ds,

sent

ence

san

dte

xtF

amili

arso

ngs,

mel

ody

and

rhyt

hmG

oldf

arb

&B

ader

,19

79E

ffec

tive

ness

MIT

1M

ITin

d(S

LT)

2xpw

,1ho

ur,

12w

eeks

+ho

me

trai

ning

Sent

ence

sM

elod

yan

drh

ythm

Jung

blut

&A

ldri

dge,

2004

Eff

ecti

vene

ssSI

PAR

I1

SIPA

RI

ind

(MT

)an

dgr

oup

1xpw

,1ho

urgr

oup,

10w

kan

d2x

pw,

1ho

urin

d+gr

oup

+ho

me

trai

ning

nrF

amili

arso

ngs,

mel

ody

and

rhyt

hm

Jung

blut

etal

.,20

06E

ffec

tive

ness

SIPA

RI

1SI

PAR

Iin

d(M

T)

and

grou

p48

wk,

1xpw

,45

min

ind

and

52w

k,2x

pin

d+gr

oup

nrF

amili

arso

ngs,

mel

ody

and

rhyt

hmK

im&

Tom

aino

,20

08T

reat

men

tpr

otoc

olw

ith

wor

king

guid

elin

es

1M

usic

ally

assi

sted

spee

chte

chni

ques

ind

(MT

)2–

3xpw

,30

min

,4

wee

ksP

hone

mes

and

sent

ence

sF

amili

arso

ngs,

rhyt

hman

ddy

nam

ics

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MUSIC IN NEUROLOGICAL SPEECH DISORDERS 9

Nae

ser

&H

elm

-Est

abro

oks,

1985

Mec

hani

sms

ofre

cove

ry1

MIT

ind

(SLT

)nr

Sent

ence

sM

elod

yan

drh

ythm

Popo

vici

,199

5E

ffec

tive

ness

MIT

1M

ITan

dse

man

tic

trai

ning

ind

(SLT

)nr

nrM

elod

yan

drh

ythm

Schl

aug

etal

.,20

08E

ffec

tive

ness

MIT

1M

ITan

dSR

Tin

d(S

LT)

5xpw

,1,5

hour

s,75

sess

ions

tota

l+ho

me

trai

ning

Sent

ence

sM

elod

yan

drh

ythm

Spar

kset

al.,

1974

Eff

ecti

vene

ssM

IT1

MIT

ind

(SLT

)an

dgr

oup

nrSe

nten

ces

Mel

ody

and

rhyt

hmT

ampl

in,2

008

Eff

ect

ofsi

ngin

gon

spee

ch1

voca

lexe

rcis

esan

dsi

ngin

gin

d(M

T)

3xpw

,30

min

,8

wee

ksP

hone

mes

and

text

Mel

ody,

tem

po,

rhyt

hman

dfa

mili

arso

ngs

Wils

onet

al.,

2006

Eff

ecti

vene

ssM

IT1

MIT

ind

(MT

)2x

pw,1

hour

,4

wee

ks+

hom

etr

aini

ng

Sent

ence

sM

elod

yan

drh

ythm

MIT

=m

elod

icin

tona

tion

ther

apy,

MM

T=

mod

ified

mel

odic

into

nati

onth

erap

y,SI

PAR

I=Si

ngen

Into

nati

onP

roso

die

Atm

ung

Rhy

tmus

übun

gen

Impr

ovis

atio

nen,

ICF,

1=

body

func

tion

/im

pair

men

t,SR

T=

spee

chre

peti

tion

ther

apy,

ind

=in

divi

dual

,gr

oup

=gr

oup

ther

apy,

MT

=m

usic

ther

apy,

SLT

=sp

eech

lang

uage

ther

apy,

nr=

not

repo

rted

.

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10 HURKMANS ET AL.

TAB

LE4

Met

ho

do

log

ical

qu

alit

y

Stu

dyS

tudy

desi

gnB

lindi

ngS

ampl

ing

Gro

up/pa

rtic

ipan

tco

mpa

rabi

lity

Tre

atm

ent

fidel

ity

Out

com

esS

igni

fican

ceP

reci

sion

Inte

ntio

nto

trea

t

Bak

er,2

000

Cas

ese

ries

nrno

nonr

nono

nrna

Bel

inet

al.,

1996

Cas

ese

ries

nrno

yes

nrye

sye

snr

nrB

utte

t&

Aub

ert,

1980

Cas

ese

ries

nrno

nonr

nono

nrnr

Coh

en,1

992

Cas

ese

ries

nrno

nonr

nono

nrnr

Coh

en&

Mas

se,

1993

Gro

upst

udy

nrye

sye

snr

noye

snr

nr

Gol

dfar

b&

Bad

er,1

979

Cas

est

udy

nano

nonr

nono

nrna

Jung

blut

&A

ldri

dge,

2004

Gro

upst

udy

nrno

yes

nrye

sye

snr

nr

Jung

blut

etal

.,20

06C

ase

stud

yna

noye

snr

yes

nonr

na

Kim

&To

mai

no,

2008

Cas

ese

ries

nrno

nonr

nono

nrnr

Nae

ser

&H

elm

-E

stab

rook

s,19

85

Cas

ese

ries

nrno

nonr

yes

nrnr

nr

Popo

vici

,199

5G

roup

stud

ynr

noye

snr

noye

snr

nrSc

hlau

get

al.,

2008

Cas

ese

ries

nrye

sye

snr

yes

nonr

nr

Spar

kset

al.,

1974

Cas

ese

ries

nrno

yes

nrye

sye

snr

nr

Tam

plin

,200

8C

ase

seri

esnr

nono

nrye

sye

snr

nrW

ilson

etal

.,20

06C

ase

stud

yna

noye

snr

noye

snr

na

nr=

not

repo

rted

,na

=no

tap

plic

able

.

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MUSIC IN NEUROLOGICAL SPEECH DISORDERS 11TA

BLE

5R

esu

lts

Stu

dyO

utco

me

mea

sure

(s)

ICF

NM

easu

red

impr

ovem

ent

Met

hodo

logi

calq

ualit

yM

echa

nism

sof

reco

very

Bak

er,2

000

Num

ber

ofw

ords

12

Num

ber

ofw

ords

incr

ease

din

both

pati

ents

0nr

Bel

inet

al.,

1996

BD

AE

,MR

I,an

dP

ET

17

Impr

ovem

ent

atit

em2,

4,5,

13,1

4,15

,and

19of

the

BD

AE

3Si

mpl

epa

ssiv

e(w

ord

hear

ing)

and

acti

ve(w

ord

repe

titi

on)

verb

alta

sks

perf

orm

edw

itho

utM

ITre

sult

sin

abno

rmal

acti

viti

esof

righ

the

mis

pher

est

ruct

ures

.W

ord

repe

titi

onpe

rfor

med

wit

hM

ITlo

aded

wor

dsre

acti

vate

sB

roca

’sar

eaan

dth

ead

jace

ntle

ftpr

efro

ntal

cort

exB

utte

t&

Aub

ert,

1980

Art

icul

atio

n,re

peti

tion

,and

audi

tory

com

preh

ensi

on1

74/

7go

odim

prov

emen

t,2/

7m

ildim

prov

emen

t,1/

7no

impr

ovem

ent

0nr

Coh

en,1

992

6as

pect

sof

voic

ean

dar

ticu

lati

on1

8Im

prov

emen

tin

spea

king

fund

amen

talf

requ

ency

vari

abili

ty,s

peec

hra

te,

and

verb

alin

telli

gibi

lity

0nr

Coh

en&

Mas

se,1

993

Spee

chra

tean

dve

rbal

inte

lligi

bilit

y1

32Si

ngin

ggr

oup:

impr

ovem

ent

atve

rbal

inte

lligi

bilit

y,no

impr

ovem

ent

atsp

eech

rate

.Rhy

thm

grou

p:no

impr

ovem

ent

4nr

Gol

dfar

b&

Bad

er,1

979

Rep

eati

ngse

nten

ces

(3co

ndit

ions

:nor

mal

,in

tone

d,an

din

tone

dw

ith

tapp

ing

11

Impr

ovem

ent

inal

lco

ndit

ions

0nr

(Con

tinu

ed)

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12 HURKMANS ET AL.

TAB

LE5

(Con

tinu

ed)

Stu

dyO

utco

me

mea

sure

(s)

ICF

NM

easu

red

impr

ovem

ent

Met

hodo

logi

calq

ualit

yM

echa

nism

sof

reco

very

Jung

blut

&A

ldri

dge,

2004

AA

T1

15Im

prov

emen

tin

arti

cula

tion

and

pros

ody

ofsp

onta

neou

ssp

eech

and

inR

EP

and

NA

M

4nr

Jung

blut

etal

.,20

06A

AT

11

Impr

ovem

ent

inal

lpar

tsof

spon

tane

ous

spee

ch,T

T,R

EP,

and

NA

M

2nr

Kim

&To

mai

no,2

008

Art

icul

atio

n,flu

ency

,pr

osod

yan

dbr

eath

supp

ort

17

At

allo

utco

me

crit

eria

vari

able

impr

ovem

ent

have

been

mea

sure

dat

all

7pa

rts

ofth

epr

otoc

ol

0nr

Nae

ser

&H

elm

-Est

abro

oks,

1985

BD

AE

and

CT

scan

18

4/8

impr

ovem

ent

atth

eB

DA

E1

Pat

ient

sw

hore

spon

dpo

siti

vely

toM

ITha

veno

ton

lyse

vere

nonfl

uent

apha

sia

wit

hsl

ow,p

oorl

yar

ticu

late

dsp

eech

and

rela

tive

lygo

odau

dito

ryco

mpr

ehen

sion

,but

also

the

lesi

ons

invo

lve

Bro

ca’s

area

,no

larg

ele

sion

inW

erni

cke’

sar

eaan

dno

lesi

onin

the

righ

the

mis

pher

ePo

povi

ci,1

995

Aud

itor

yco

mpr

ehen

sion

,re

peti

tion

and

nam

ing

148

0W

erni

cke

>B

roca

and

anom

icap

hasi

aan

dM

IT>

sem

anti

cs

3nr

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MUSIC IN NEUROLOGICAL SPEECH DISORDERS 13

Schl

aug

etal

.,20

08C

IU,n

umbe

rof

sylla

bles

,pi

ctur

ena

min

gan

dfM

RI

12

Impr

ovem

ent

inal

lout

com

em

easu

res

3F

unct

iona

lim

agin

gta

sks

targ

etin

gm

usic

alco

mpo

nent

ste

ndto

elic

itgr

eate

rac

tivi

tyin

righ

the

mis

pher

icbr

ain

regi

ons

than

inle

fthe

mis

pher

icre

gion

s.T

appi

ngth

ele

ftha

nden

gage

sa

righ

the

mis

pher

icse

nsor

imot

orne

twor

kth

atco

ordi

nate

sor

ofac

iala

rtic

ulat

ory

mov

emen

tsSp

arks

etal

.,19

74B

DA

E,r

epet

itio

nan

dun

ison

spee

ch1

8Im

prov

emen

tin

6/8

pati

ents

3nr

Tam

plin

,200

8SI

T,P

DT,

RO

S,IW

PM

and

CE

R1

4Im

prov

emen

tin

SIT,

PD

T,IW

PM

,and

CE

RN

oim

prov

emen

tin

RO

S

2nr

Wils

onet

al.,

2006

Phr

ase

leng

th1

1Im

prov

emen

tin

phra

sele

ngth

2nr

BD

AE

=B

osto

nD

iagn

osti

cA

phas

iaE

xam

inat

ion,

MR

I=m

agne

tic

reso

nanc

eim

agin

g,P

ET

=po

sitr

onem

issi

onto

mog

raph

y,A

AT

=A

ache

nA

phas

iaT

est,

CT

=co

mpu

ted

tom

ogra

phy,

CIU

=co

rrec

tin

form

atio

nun

its,

fMR

I=fu

ncti

onal

mag

neti

cre

sona

nce

imag

ing,

SIT

=Se

nten

ceIn

telli

gibi

lity

Tes

t,P

DT

=P

ictu

reD

escr

ipti

onT

ask,

RO

S=

rate

ofsp

eech

,IW

PM

=in

telli

gibl

ew

ords

per

min

ute,

CE

R=

com

mun

icat

ion

effic

ienc

yra

tios

,IC

F,1=

body

func

tion

/im

pair

men

t,R

EP

=re

peat

ing,

NA

M=

nam

ing,

TT

=To

ken

Tes

t,nr

=no

tre

port

ed.

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14 HURKMANS ET AL.

Mechanisms of recovery

In three studies (Belin et al., 1996; Naesser & Helm-Estabrooks, 1985; Schlaug,Marchina, & Norton, 2008) examinations of mechanisms of recovery by PET, CT,and fMRI were conducted in the method of the study to explain the research findings.Neural correlates focused mainly on the observed brain activities in both hemispheresduring language tasks and at the location of the lesion. The other 12 studies interprettheir research findings but are hypothetical, since mechanisms of recovery are absentfrom the method of the study and therefore not objectively identical.

Schlaug et al. (2008) described two patients: one patient received MIT and theother patient received a combination of MIT and a control treatment (SRT: speechrepetition therapy). The patient receiving only MIT showed significantly more fMRIactivities in the right hemisphere. Naeser and Helm-Estabrooks (1985) studied twogroups of patients receiving MIT: a good response group and a poor response groupof MIT. CT information characteristics of both groups have been examined. Thegood response group showed lesions in Broca’s area in the left hemisphere. The poorresponse group showed lesions in both hemispheres and/or Wernicke’s area. Belinet al. (1996) evaluated a group of seven non-fluent aphasic patients who successfullyfinished MIT intervention. They measured changes in relative cerebral blood flow withPET during listening and repetition of words and during repetition of MIT loadedwords (i.e., with melody and rhythm). Their findings revealed abnormal activationin the right hemisphere without MIT language task and, in contrast, reactivation inBroca’s area and the left prefrontal cortex by repeating MIT loaded words.

As a concluding remark of the results, we gathered extensive information aboutmusic and language in the literature. Treatment approaches using musical elementsreported measurable improvement. However, the methodological quality of the effi-cacy studies was low and mechanisms of recovery were contradictory.

DISCUSSION

The purpose of this study was to review the existing literature on the effect of treatmentusing musical elements in the treatment of patients with neurological language andspeech disorders and mechanisms of recovery explaining positive effects. This reviewshows that a certain amount of information is revealed in the literature concerningtherapies using musical elements in the treatment of neurological language and speechdisorders. In the reviewed studies frequent gaps in the descriptions of patient charac-teristics and therapy interventions have been determined. Overall, the methodologicalquality of the studies was rated as low.

All but one of the studies involved stroke patients. This is understandable becausestroke patients are a rather homogeneous group in comparison to other patientswith acquired brain injuries (ABI). Findings in these studies can also theoretically beapplied to patients with other types of ABI; however, studies on this subject still needto be done. Both males and females were included in the investigations under study.In Popovici (1995) men dominated the study population. This may be explained by alarge subgroup of patients with TBI in their study sample (Tagliaferri, Compagnone,Korsic, Servadei, & Kraus, 2006). However, selection bias cannot be ruled out.

The studies included patients who were primarily in the chronic phase of post onsetrecovery. However, therapy is also given in the subacute phase. It is therefore important

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in future research to study the effect of music elements in treatment in the subacutephase.

Education was not reported in the description of patient characteristics in onethird of the reviewed studies. Education may influence learning and is therefore animportant aspect in studying the effectiveness of treatment. Next to education, cog-nitive functioning is an important predictor of outcome since non-linguistic cognitiveimpairments may limit rehabilitation efficacy in patients with aphasia (Seniów, Litwin,& Lesniak, 2009). We will therefore study cognitive functioning next to education infuture efficacy research. Dominance was also not reported in more than half of thereviewed studies. Mainly in studies where music is a central topic of research interest,information about dominance is valuable since mechanisms of recovery focuses onbrain activities in one of the two hemispheres. Information about musical backgroundwas also lacking in patient characteristics (not specifically defined by objective crite-ria); theoretically, we assume that this variable may influence treatment outcome. Thediscrepancy is substantial between the description of language and speech functioningwhen information about the musical background is missing. This is especially the casewhen studying an intervention in which music plays such an important role. Notableis that musical elements of therapy mainly comprised melody and rhythm. The factthat MIT is the most studied programme to date may be an explanation; melody andrhythm are distinguished features of MIT. Other musical parameters like dynamics,tempo, and metre have not been applied.

In general, therapy interventions have been adequately described. MIT is an inter-nationally well-known programme (Norton, Zipse, Marchina, & Schlaug, 2009).Deviations with respect of content as well as therapy intensity of the originalmethod have been well described. Only a few times have other therapy interven-tions than the studied intervention (e.g., MIT) been reported. In clinical practiceaphasic patients receive various intervention programmes. It is therefore importantto know if the revealed improvement can be assigned to the studied programme or toco-interventions.

All the objectives of the reviewed studies were aimed at the (ICF) level of impair-ments. None of the studies conducted outcome measures at the (ICF) level of activitiesand/or participation. Therefore it is unknown whether revealed improvement at thelevel of impairments can be generalised in their application to communication in dailylife and if it has any social implications.

The power of evidence was low for the majority of the reviewed studies. A ran-domised controlled trial (RCT) with an adequate size is hardly accomplishable from apractical as well as a methodological standpoint (e.g., realising a homogeneous group).For that reason adequate alternative study designs are available: single-participantdesigns and case series (Howard, 2003). These study designs have frequently been usedin the reviewed studies. Case studies and case series offer an extraordinary opportunityto describe patient characteristics and intervention programmes in detail. The effec-tiveness of the therapy can be verified very precisely, even in a small group. Not onlyis a well-described method important to measure effectiveness of therapy but also theuse of statistics is needed to calculate p-values and to determine the likelihood thatstudy findings are results of chance. It is here that results of many articles were lim-ited: all studies report improvement but in over half the studies no statistics were used.That makes it difficult to conclude whether the measured improvement is the result ofthe studied therapy programme. For example, although a sufficient number of patients

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are included in the study of Popovici (1995), the low level of evidence (score 3) maketheir conclusions about the positive effect of the treatment doubtful.

MIT is the therapy programme that was used in the three studies that identify neu-ral correlates to explain mechanisms of recovery. The purpose of MIT is to exploit theprosodic and melodic process components of the intact right hemisphere for use withleft hemisphere brain-damaged aphasic patients. The authors of the MIT hypothesisedthat successful recovery engages expressive language areas in the undamaged righthemisphere. This hypothesis is over 30 years old. Brain plasticity is profound, and reor-ganisation processes are dynamic with recovery of language function incorporatingboth hemispheres (e.g., Saur et al., 2006). However, this premise of the original devel-opers of MIT is still appropriate, as there has been no research to date that disprovesthis hypothesis. Two out of three studies in this review (Naeser & Helm-Estabrooks,1985; Schlaug et al., 2008) support the hypothesis of Albert et al. (1973). The find-ings of Belin et al. (1996) were surprising and contrary to the hypothesis proposed bythe developers of MIT and the original interpretation of MIT successes. Belin et al.reported that the recovery process coincides with the reactivation of left prefrontalstructures with melody and rhythm tasks rather than mechanisms of compensation inright hemisphere structures.

This review shows the difficulty of proving the effectiveness of therapy using musicalelements. Research in this field is in a fairly early state and an adequate system to clas-sify and describe complex interventions is lacking. We highly recommend the develop-ment of research guidelines to standardise data-reporting parameters such as patient’scharacteristics, intervention, and methodological quality. Different models can beused relevant to rehabilitation. Wade (2005) suggests a method for describing reha-bilitation interventions derived from two models: (1) the World Health Organisation’sInternational Classification of Functioning model of illness and (2) a model describingrehabilitation interventions. Patient characteristics can be adequately reported in theICF model. Intervention and outcome variables can be adequately reported in Wade’smodel where interventions may be described in terms of the situations where theseactions are applied, the immediate goals of any action, the level at which the interven-tion acts, the actions involved, the knowledge and skills needed to give the treatment,any specific equipment used, and any concomitant actions that may be necessary. Formethodological quality, we recommend study designs using the highest level of qualityindicators in the guidelines of ASHA levels-of-evidence scheme.

CONCLUSION

The purpose of this review was to assess the effects of musical elements in the treat-ment of neurological language and speech disorders. A systematic search of theliterature yielded 15 studies that met inclusion criteria.

Measurable improvement was reported in studies where musical components wereused in the treatment of neurological language and speech disorders. However, themethodological quality of studies was rated low. Therefore no conclusions can yet bedrawn with regard to the effect of the use of musical elements in the treatment of indi-viduals with acquired neurological disorders. Mechanisms of recovery remain unclear:two of the three studies that examined mechanisms of recovery via neuroimagingtechniques supported the role of the right hemisphere, but reports are contradictory

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and exact mechanisms of recovery remain indefinable. Shortcomings in the currentresearch can be overcome by following standards as outlined by the discussion sectionin this article.

Manuscript received 26 January 2011Manuscript accepted 28 June 2011

First published online 6 October 2011

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APPENDIX

Music therapy (MT) can serve different goals in the rehabilitation of neurologicallyimpaired patients. A common approach in MT is to apply non-verbal aspects deal-ing with emotional and social problems. In addition, MT can be aimed at verbalexpression and communication. Adults suffering from neurological impairments arechallenged to “relearn” activities of daily living that were once performed with ease, aswell as to emotionally adjust to their limitations and changed life circumstances. Thesedeep emotions can negatively influence rehabilitation (Baker & Wigram 2004; Jochims1995; Magee & Davidson, 2002). Throughout the rehabilitation process clients tendto form a new identity and re-shape their future, which demands significant effortand perseverance. This process often leads to sadness, anger, and feelings of inferi-ority and insecurity. MT considers the body and mind as inseparable and thereforeMT approaches focus on the medical-physical and on the social-emotional aspectsof rehabilitation. SMTA, for example focuses primarily on speech-language exercisesset to music; however it also draws on music’s potential for relaxation and enjoyment

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so clients experience the program as being “less technical”. Patients enjoy the inter-vention and are able to sustain their participation for long periods of time (Magee,Brumfitt, Freeman, & Davidson, 2006). The joint singing of patient and therapistsemphasises the social aspect of music making, thereby acting to reduce isolation.

The MT interventions are designed to musically support the speech-language exer-cises, and as such they share the same structural linguistic levels. Using tempo, metre,rhythm, and dynamic parameters the music therapist varies the melodies, therebyincreasing the level of difficulty over the course of treatment. The different parame-ters are adapted to the individual patient’s capabilities, thereby simplifying the singingexercise for the client when needed (De Bruijn et al., 2005). MT considers music tempoto be the key for melodic adaptation. Some features of tempo are familiar in MT.A slow tempo for example creates a sense of relaxation. But a tempo that is too slowbecomes static: there is no flowing motion. This does not stimulate the client andmay negatively affect the patient’s singing. And finally, a fast/faster tempo may stim-ulate the client and increase the level of concentration required to perform the task.Variations in metre provide opportunities for the patient to practice the same materialwhile maintaining interest. Some familiar features of metre are the following: 4/4 and2/4 beats are supportive, familiar, and easy to sing; 3/4 and 3/8 beats evoke a sway-ing motion and are suitable when relaxation (decrease in tension) would enhance theclient’s performance in the exercises; and finally a 6/8 beat may be perceived as bothdouble and triple time. The movement stimulated by the 6/8 beat is relaxing, but maylead to an increase in tempo. Variations in rhythm are determined by the prosodic fea-tures of speech. Some characteristics of rhythm are important in MT: the order of longand short note values influence the degree of rhythmic complexity and therefore theexercise’s level of difficulty. For example, in 4/4 time the sequence long–short–short ismore difficult than short–short–long because it allows the patient less time to preparehimself for the repetition of the exercise. Syncopation is not part of natural speech andshould therefore be avoided in MT.

In MT dynamics ranges from mezzo-piano to mezzo-forte, which is usually the leasttaxing on the voice. However, this choice is dependent on the (emotional) content ofthe exercise. Some following features related to dynamics should be considered in MT:crescendos are useful during the repetition of sentences that are intended as exclama-tions; sometimes the use of forte or even fortissimo is necessary, for example to call orwarn someone or to express emotions.

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