Music Therapy for Preterm Infants and Their Parents: A Meta … · Music Therapy for Preterm Infants and Their Parents: A Meta-analysis Łucja Bieleninik, PhD, a Claire Ghetti, PhD,

REVIEW ARTICLEPEDIATRICS Volume 138 , number 3 , September 2016 :e 20160971

Music Therapy for Preterm Infants and Their Parents: A Meta-analysisŁucja Bieleninik, PhD, a Claire Ghetti, PhD, b Christian Gold, PhDa

abstractCONTEXT: Given the recent expansion of research in the area of music therapy (MT) for preterm

infants, there is a need for an up-to-date meta-analysis of rigorously designed studies that

focus exclusively on MT.

OBJECTIVE: To systematically review and meta-analyze the effect of MT on preterm infants and

their parents during NICU hospitalization and after discharge from the hospital.

DATA SOURCES: PubMed/Medline, PsycINFO, Embase, Cochrane Database of Systematic Reviews,

CINAHL, ERIC, Web of Science, RILM.

STUDY SELECTION: Only parallel or crossover randomized controlled trials of MT versus standard

care, comparison therapy, or placebo were included.

DATA EXTRACTION: Independent extraction by 2 reviewers, including risk of bias indicators.

RESULTS: From 1803 relevant records, 16 met inclusion criteria, of which 14 contained

appropriate data for meta-analysis involving 964 infant participants and 266 parent

participants. Overall, random-effects meta-analyses suggested significant large effects

favoring MT for infant respiratory rate (mean difference, –3.91/min, 95% confidence

interval, −7.8 to −0.03) and maternal anxiety (standardized mean difference, –1.82, 95%

confidence interval, −2.42 to −1.22). There was not enough evidence to confirm or refute

any effects of MT on other physiologic and behavioral outcomes or on short-term infant

and service-level outcomes. There was considerable heterogeneity between studies for the

majority of outcomes.

LIMITATIONS: This review is limited by a lack of studies assessing long-term outcomes.

CONCLUSIONS: There is sufficient evidence to confirm a large, favorable effect of MT on infant

respiratory rate and maternal anxiety. More rigorous research on short-term and long-term

infant and parent outcomes is required.

aThe Grieg Academy Music Therapy Research Centre, Uni Research Health, Bergen, Norway; and bThe Grieg Academy Department of Music, University of Bergen, Bergen, Norway

Dr Bieleninik conceived of the review, prepared the protocol, searched databases, handsearched, screened titles/abstracts, assessed eligibility of studies, extracted

data, and followed-up for missing data; Dr Ghetti conceived of the review, prepared the protocol, assessed eligibility of studies, extracted data, and followed-up for

missing data; Dr Gold conceived of the review, resolved confl icts of agreement for eligibility, and conducted data analysis; and all authors prepared, revised, and

approved the fi nal manuscript as submitted and agree to be accountable for all aspects of the work.

DOI: 10.1542/peds.2016-0971

Accepted for publication Jun 14, 2016

Address correspondence to Christian Gold, The Grieg Academy Music Therapy Research Centre, Uni Research Health, Uni Research, Lars Hilles gate 3, 5015 Bergen,

Norway. E-mail: [email protected]

PEDIATRICS (ISSN Numbers: Print, 0031-4005; Online, 1098-4275).

Copyright © 2016 by the American Academy of Pediatrics

To cite: Bieleninik Ł, Ghetti C, Gold C. Music Therapy for Preterm Infants and Their Parents: A Meta-analysis. Pediatrics. 2016;138(3):e20160971

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BIELENINIK et al

Preterm birth and prematurity is

a major medical, psychological,

and socioeconomic problem

worldwide. More than 1 in 10 of

the world’s newborns are born

prematurely, corresponding to 14.9

million premature infants each

year. 1 Children born prematurely

are vulnerable to mortality,

morbidity, and various forms of

disability, neurodevelopmental

disorders, developmental delays,

and long-lasting sequelae. 2

Perinatal interventions may

significantly impact long-term

growth and development in high-

risk preterm infants. 3 Cautious,

infant-specific, nonpharmacological

early intervention methods, such

as kangaroo care (KC) and music

therapy (MT), have been initiated

in NICUs to minimize adverse

short- and long-term consequences

of prematurity. The evidence base

supporting the use of MT in the

NICU is gradually accumulating,

with positive immediate and short-

term outcomes demonstrated for a

variety of MT approaches. 4 More than

400 professional music therapists

worldwide have obtained specialized

training in MT in the NICU, 5, 6 and

∼50% of the top 25 US children’s

hospitals offer MT in their NICUs. 6

MT within the setting of prematurity

relates to the informed use of

music and aspects of a therapeutic

relationship to promote optimal

infant development and facilitate

secure attachment with primary

caregivers. When used within a NICU

environment, MT may facilitate infant

sensory regulation 7 and promote

ongoing neurologic development.

Music therapists tailor MT to the

developmental readiness of the

neonate and may include live or

prerecorded music with a focus on

the infant or on the caregiver/infant

dyad. Music therapists can help

caregivers recognize engagement/

disengagement cues and support

caregivers in using infant-led

musical interactions to facilitate

developmental progress while

promoting bonding.

Six systematic reviews of MT

and music-based interventions

for premature infants have been

published. 4, 8 – 12 Three of these

reviews 4, 9, 10 followed reporting

guidelines outlined in the

PRISMA statement.13 None of the

aforementioned 6 reviews required

music therapist involvement

(in either development of or

implementation of the MT protocol)

as a condition of inclusion, and a

majority of these reviews included

studies with designs that are less

rigorous than randomized controlled

trials (RCTs). 4, 8, 9, 11, 12 The single

systematic review that restricted

inclusion to RCTs included music-

based interventions without music

therapist involvement. 10 The results

of Hartling et al 10 demonstrate

preliminary evidence that music

may have beneficial effects on

physiologic parameters, behavioral

states, oral feeding rates, and pain

among preterm infants, although

the authors did not complete meta-

analysis because of heterogeneity

in outcomes, interventions, and

populations. 10 The motivation to

conduct the current systematic

review was to provide an updated

analysis of RCTs specific to MT as

implemented by or in consultation

with a trained music therapist. The

requirement for music therapist

involvement helps assure that at

least an entry-level understanding

of the theory, practice training, and

research related to the professional

use of MT for premature infants has

been achieved.

The objective was to review RCTs

to examine the effects of MT versus

standard care or standard care

combined with other therapies

for preterm infants and their

parents/caregivers during NICU

hospitalization and after discharge

from the hospital.

METHODS

Search Strategy

We searched electronic databases

for eligible studies and hand-

searched reference lists from existing

review papers. We screened the

following databases: PubMed/

Medline, PsycINFO, Embase,

Cochrane Database of Systematic

Reviews, CINAHL (Cumulative

Index to Nursing and Allied Health

Literature), ERIC (Education

Resources Information Center), Web

of Science, and RILM. Databases

were searched using the following

terms: (prematur* OR preterm

OR neonat* OR low birth weight

OR LBW OR parent OR caregiver

OR NICU) AND (music* OR music

therap* OR auditory stimulation)

AND (randomized controlled OR

randomised controlled OR RCT).

Searching was not restricted to

any language, reference type, or

year of publication. Unpublished

studies were included, but no

additional steps were taken to locate

unpublished material.

Study Selection

One reviewer screened database

search results to identify relevant

titles and abstracts. All potentially

relevant records were extracted to

EndNote reference management

software. Duplicates were detected

and deleted, and 2 reviewers

independently inspected titles and

abstracts of potentially relevant

records to exclude irrelevant reports.

Studies were included if they met the

following criteria:

1. Participants: children born

prematurely, defined by the

World Health Organization as

birth before 37 completed weeks

of gestation, or fewer than 259

days since the first day of the

woman’s last menstrual period, 14

and their parents/caregivers.

We aimed to include children up

to 3 years of age who were born

preterm, to assess longer-term

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PEDIATRICS Volume 138 , number 3 , September 2016

neurodevelopmental outcomes

of this high-risk group. 3 There

were no restrictions as to gender,

ethnicity, or type of setting.

2. Intervention: all forms of MT

carried out by, or in consultation

with, a trained music therapist,

conducted in hospital, community,

or home settings.

3. Comparison: trials in which

MT combined with standard

treatment is compared with:

standard care alone; standard care

combined with other therapies;

or standard care with placebo.

Placebo treatment could involve

the use of headphones to deliver

silence or nonmusic auditory

stimuli (eg, white noise).

4. Outcomes: reported at least 1

outcome of the following domains:

psychodevelopmental, behavioral,

physiologic, anthropometric,

socioemotional development,

parental functioning,

adverse effects, and length of

hospitalization.

5. Study design: RCT studies (parallel

and crossover).

Two reviewers reviewed and

independently assessed full texts

to determine eligible studies. Any

disagreements were resolved

through discussion with a third

reviewer. When information about

music therapist involvement was not

reported or unclear in the paper, we

contacted authors for clarification.

Data Collection and Extraction Process

Two reviewers independently

extracted data and confirmed

accuracy using a shared, pilot-tested

data extraction sheet for information

on participants, interventions,

control conditions, outcomes, and

results. We contacted study authors

via e-mail to request missing data or

for clarification and provided each

with an individualized data table for

reporting the requested data. Risk

of bias was assessed by 2 reviewers

independently, using the Cochrane

risk of bias tool. 15 Any discrepancies

were resolved through consultation

with the third reviewer.

Data Analyses

Relevant outcomes for which data

were available from >1 study were

aggregated using meta-analysis;

outcomes where data were available

for only 1 study were described

narratively. In the meta-analyses,

we analyzed means and SDs of

end-point data for intervention

and control conditions. When

>1 treatment condition included

music, the intervention that used

live music was selected for analysis

instead of the recorded music

group. The preference for live

music interventions and use of sung

voice is consistent with active MT

approaches that are individually

tailored to infant and infant/

parent dyadic responses. When

>1 treatment condition used live

music, we selected the intervention

that most strongly reflected the

authors’ theoretical rationale for

the study (frequently the first-listed

MT intervention). When there

were multiple MT conditions with

equivalent intervention that varied

by frequency of intervention, we

selected the MT condition with

maximal frequency for analysis.

When MT was compared with a

nonmusic comparison condition

and a standard care condition, we

selected the standard care condition

as a basis for comparison. We

selected end-point data immediately

postintervention for treatment

and control groups. When studies

included >1 postintervention end

point, the data point directly after

completion of intervention was

selected for analysis. When data

were reported for clinical subgroups

(eg, based on gender or gestational

age [GA]) but were not available for

the whole sample, we calculated

weighted means and pooled SDs to

obtain a single result for each study

outcome to avoid artificially inflated

heterogeneity estimates between

studies.

Meta-analyses for each outcome were

performed using weighted mean

differences on the original metric

when possible (ie, when the outcome

was measured on the same scale,

or could be transferred to the same

scale, in all included studies assessing

that outcome). When different scales

were used for the same outcome,

we used effect sizes (standardized

mean differences [SMD]; Hedges’ g).

We interpreted effect sizes in line

with common guidelines (ie, 0.2,

small; 0.5, medium; 0.8, large 16;). For

crossover studies, SEs depend on the

correlation between measurements,

which is often not reported. We

assumed a correlation of 0; that is,

we analyzed data from crossover

trials as if they were from parallel

trials. This approach is conservative

because crossover trials are

underweighted. 15 We calculated

fixed-effects and random-effects

meta-analyses and inspected I2 as a

measure of heterogeneity. Because

heterogeneity was high for most

outcomes, we only present random-

effects meta-analyses. We inspected

study design (parallel versus

crossover) and clinical characteristics

(treatment frequency, postmenstrual

age at study start, birth weight) as

potential sources of heterogeneity.

Although we had planned to address

these sources of heterogeneity

quantitatively by subgroup analyses

or meta-regression, we refrained

from such advanced analyses because

of the limited number of studies per

outcome, and relied instead on visual

examination of the figures (except

for study design). Meta-analyses

were performed using R (Version

3.2.3, GNU project, Boston, MA) and

R package meta. We analyzed all

subjects who were assigned to the

treatment intervention regardless

of whether they received the full

treatment or not (intention-to-treat).

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4 BIELENINIK et al 4

TABL

E 1

Stu

dy

Ch

arac

teri

stic

s

Firs

t Au

thor

Stu

dy

Su

bje

cts

Inte

rven

tion

Gro

up

(s)

Con

trol

Gro

up

(s)

Ou

tcom

es

Year

Cou

ntr

yD

esig

nN

Infa

nt

Par

tici

pan

ts

Ran

dom

ized

(An

alyz

ed)

N P

aren

t

Par

tici

pan

ts

Ran

dom

ized

(An

alyz

ed)

GA

at

Bir

th

(wk)

Pos

tmen

stru

al

Age

at S

tud

y

Sta

rt (

wk)

BW

(g)

Sex

, n

Fem

ale

(%)

Trea

tmen

t To

tal

Ses

sion

s

(Tim

e)

Du

rati

on

(d)

1) In

clu

ded

in

Met

a–an

alys

is;

2)S

um

mar

ized

Nar

rati

vely

; 3)N

ot

Incl

ud

ed in

Th

is

Rev

iew

Arn

on20

06Is

rael

Cro

ssov

er31

(31

)n

/aM

dn

, 29

(IQ

R,

25–

34)

Md

n, 3

4 (I

QR

,

32–

40)

Md

n, 1

175

(IQ

R,

650–

1737

)

17 (5

5%)

Gro

up

1a :

live

mu

sic;

Gro

up

2: r

ecor

ded

mu

sic

2 (3

0 m

in)

3N

o M

T1)

HR

, RR

, O2

SAT

,

beh

avio

r st

ate;

3) S

elf-

rep

ort

of

effe

ct o

f th

erap

y

Arn

on20

14Is

rael

Cro

ssov

er86

(86

)86

Md

n, 3

1

(IQ

R,

25–

33)

Md

n, 3

5 (I

QR

,

27–

42)

Md

n, 1

411

(IQ

R,

640–

2512

)

47 (5

5%)

Live

mat

ern

al

sin

gin

g +

KC

2 (4

0 m

in)

2KC

1) H

R, R

R, O

2

SAT

, beh

avio

r

stat

e, m

ater

nal

anxi

ety

(STA

I);

3) H

R v

aria

bili

ty,

mot

her

's H

R,

mot

her

’s R

R,

mot

her

’s O

2 S

AT

Cal

abro

2003

Aust

ralia

Par

alle

l22

(17

)n

/a—

M, 3

4—

10 (5

9%)

Rec

ord

ed

sed

ativ

e

mu

sic

4 (2

0 m

in)

4N

o M

T2)

HR

, RR

, O2

SAT

,

beh

avio

ral

dis

tres

sb

Cas

sid

y20

09U

SP

aral

lel

63 (

63)

n/a

(ran

ge,

28–

33)

——

32 (5

0%)

Gro

up

1a

+ 2

a :

reco

rded

lulla

by

mu

sic

and

cla

ssic

mu

sic

4 (2

0 m

in)

4S

tan

dar

d

NIC

U

care

1) H

R, R

R, O

2

SAT

; 2)

Hea

d

circ

um

fere

nce

.

Cev

asco

2008

US

Par

alle

l25

(20

)21

(16

)M

, 32.

1

(SD

,

1.7,

ran

ge,

28–

34)

—M

, 165

7.25

(SD

,

365.

8,

ran

ge,

954–

2535

)

8 (4

0%)

Rec

ord

ed

mu

sic

wit

h

mot

her

's

sin

gin

g

3–5

per

wk

(20

min

)

un

til

dis

char

ge

Sta

nd

ard

NIC

U

care

1) P

ostm

enst

rual

age

at d

isch

arge

;

2) B

ond

ing,

dis

char

ge

wei

ght,

par

enta

l

adju

stm

ent;

3)

Valu

e of

mu

sic

inte

ract

ion

, CD

imp

orta

nce

scor

e, a

fter

-bir

th

com

plic

atio

ns,

freq

uen

cy o

f

mu

sic

inte

ract

ion

pos

t d

isch

arge


5PEDIATRICS Volume 138 , number 3 , September 2016 5

Firs

t Au

thor

Stu

dy

Su

bje

cts

Inte

rven

tion

Gro

up

(s)

Con

trol

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Year

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N P

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GA

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Sta

rt (

wk)

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Sex

, n

Fem

ale

(%)

Trea

tmen

t To

tal

Ses

sion

s

(Tim

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rati

on

(d)

1) In

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ded

in

Met

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2)S

um

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ized

Nar

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; 3)N

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Incl

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Th

is

Rev

iew

Ch

orn

a20

14U

SP

aral

lel

100

(94)

n/a

Md

n, 3

0

(IQ

R,

28–

32)

(ran

ge, 3

4–35

)M

dn

, 145

0

(IQ

R,

1089

–

1713

)

47 (5

0%)

PAL

wit

h

mot

her

’s

reco

rded

voic

e

5 (1

5 m

in)

5S

tan

dar

d

NIC

U

care

1) L

engt

h o

f

hos

pit

aliz

atio

n;

2) O

ral f

eed

ing

volu

me,

oral

fee

din

g

freq

uen

cy,

nu

mb

er o

f

day

s to

fu

ll

oral

fee

din

g,

dis

char

ge

wt;

3)

Gro

wth

rate

, ch

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in

saliv

ary

cort

isol

leve

ls

Ette

nb

erge

r20

14C

olom

bia

Par

alle

l30

(19

)26

(18

)M

, 32.

4

(SD

,

1.5,

ran

ge,

30–

37)

M, 3

4.16

M, 1

710.

4

(SD

,

310.

2)

8 (4

2%)

Gro

up

1a :

MT

wit

h

care

give

rs

du

rin

g KC

;

Gro

up

2:

MT

wit

h t

he

bab

ies

alon

e

4 (8

–25

min

)

14KC

1) H

R, O

2 S

AT, w

eigh

t

gain

, len

gth

of

hos

pit

aliz

atio

n,

mat

ern

al a

nxi

ety

(STA

I–C

); 2

)

size

, dis

char

ge

wei

ght,

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d

circ

um

fere

nce

,

neur

odev

elop

men

t

(BSI

D–I

I), b

ondi

ng

(MIB

S)

Joh

nst

on20

07C

anad

aC

ross

over

39 (

20)

n/a

M, 3

3.1

(SD

,

0.89

)

M, 3

4.1

M, 1

985

(SD

,

312)

9 (4

3%)

Rec

ord

ed

mot

her

’s

voic

e

6 pr

epar

ator

y

+ 1

proc

edur

e

(∼10

min

)

4N

o Ac

oust

ic

stim

ula

tion

1) H

R, O

2 S

AT,

beh

avio

ral

dis

tres

s (P

IPP

),

beh

avio

r st

ate;

3) F

acia

l act

ion

(NFC

S)

Keit

h20

09U

SC

ross

over

24 (

22)

n/a

—M

, 33.

12 (

SD

,

2.45

, ran

ge,

32–

40)

—7

(32%

)R

ecor

ded

lulla

by

mu

sic

2 (∼

18

min

)

4S

tan

dar

d

NIC

U

care

1) H

R, R

R, O

2 S

AT; 2

)

Blo

od p

ress

ure

;

3) L

engt

h o

f

inco

nso

lab

le

epis

ode,

freq

uen

cy

inco

nso

lab

le

epis

ode

TABL

E 1

Con

tin

ued


6 BIELENINIK et al 6

Firs

t Au

thor

Stu

dy

Su

bje

cts

Inte

rven

tion

Gro

up

(s)

Con

trol

Gro

up

(s)

Ou

tcom

es

Year

Cou

ntr

yD

esig

nN

Infa

nt

Par

tici

pan

ts

Ran

dom

ized

(An

alyz

ed)

N P

aren

t

Par

tici

pan

ts

Ran

dom

ized

(An

alyz

ed)

GA

at

Bir

th

(wk)

Pos

tmen

stru

al

Age

at S

tud

y

Sta

rt (

wk)

BW

(g)

Sex

, n

Fem

ale

(%)

Trea

tmen

t To

tal

Ses

sion

s

(Tim

e)

Du

rati

on

(d)

1) In

clu

ded

in

Met

a–an

alys

is;

2)S

um

mar

ized

Nar

rati

vely

; 3)N

ot

Incl

ud

ed in

Th

is

Rev

iew

Loew

y20

13U

SC

ross

over

272

(272

)—

M, 2

9.57

(SD

,

2.89

)

—M

, 132

1.22

(SD

,

495.

32)

152 (5

6%)

Gro

up

1a :

live

,

son

g of

kin

or p

aren

t–

pre

ferr

ed

lulla

by;

Gro

up

2:

hea

rtb

eat

sou

nd

s vi

a

Gat

o b

ox;

Gro

up

3:

bre

ath

ing

sou

nd

s vi

a

Oce

an d

isc

6 (∼

10

min

)

14N

o ex

plic

it

aura

l

stim

ula

tion

1) H

R, R

R, O

2 S

AT;

2) A

ctiv

ity

leve

ls,

suck

ing

beh

avio

r,

beh

avio

r st

ate,

calo

ric

inta

ke,

par

enta

l str

ess

Sch

lez

2011

Isra

elC

ross

over

52 (

52)

52 (

52)

Md

n, 3

2

(IQ

R,

26–

36)

Md

n, 3

7 (I

QR

,

28–

47)

Md

n, 1

641

(IQ

R,

760–

2715

)

28 (5

4%)

Live

har

p m

usi

c

ther

apy

+ K

C

1 (3

0 m

in)

1KC

1) H

R, R

R, O

2

SAT

, beh

avio

r

stat

e, m

ater

nal

anxi

ety

(STA

I);

3) M

oth

er’s

HR

,

mot

her

’s R

R,

mot

her

’s O

2 S

AT

Sta

nd

ley

2003

US

Par

alle

l32

(32

)n

/aM

, 31.

7

(ran

ge,

24–

40)

M, 3

6.1

(ran

ge,

33–

41)

M, 1

561.

2

(ran

ge,

620–

2640

)

16 (5

0%)

PAL

1 (1

5–20

min

)

1S

tan

dar

d

NIC

U

care

2) N

ipp

le f

eed

ing

rate

s

Sta

nd

ley

2010

US

Par

alle

l68

(68

)n

/aM

, 29.

2(r

ange

, 32–

36)

M, 1

179.

835

(51%

)

PAL

Gro

up

1:

1 (1

5

min

);

Gro

up

2a : 3

(15

min

)

5; 5

No

PAL

1) W

eigh

t ga

in; 2

)

Dis

char

ge w

eigh

t,

d b

efor

e n

ipp

le

feed

ing,

d o

f

nip

ple

fee

din

g

Vian

na

2011

Bra

zil

Par

alle

l—

101

(94)

M, 3

0.14

(SD

,

2.71

)

—M

, 127

1

(SD

,

308)

—M

oth

er-

cen

tere

d

mu

sic

ther

apy

wit

h

or w

ith

out

KC

∼9 (

60

min

)

Un

til

dis

char

ge

Sta

nd

ard

NIC

U c

are

and

usu

al

care

aft

er

dis

char

ge

1) L

engt

h o

f

hos

pit

aliz

atio

n;

2) B

reas

tfee

din

g

freq

uen

cy

TABL

E 1

Con

tin

ued


7

RESULTS

Study Characteristics

We identified a total of 1803 records

from electronic searching and 20

studies from hand-searching. The last

date of searching was April 29, 2015.

After excluding duplicates and clearly

irrelevant references, we obtained

74 studies to assess for eligibility.

Of these, 16 met inclusion criteria

and were included in the systematic

review ( Table 1); most of these

(14) also had usable data for meta-

analyses ( Fig 1). The final sample

included 6 crossover RCTs, 7, 17 – 21 6

RCTs with 2 parallel arms, 22 – 27

and 4 RCTs with 3 parallel arms.28 – 31

Sample size ranged from 22 to

272 participants with a median

of 52. Table 1 displays study

characteristics.

Participant Characteristics

Selected trials included a total

of 1071 infant participants (496

female, 46%) and 286 parent

participants. Six trials included

parents/caregivers, 7, 17, 21, 23, 26, 29 with

1 trial specially targeting mothers. 26

Fathers participated in at least 2

studies, 7, 29 but were not specifically

targeted in any study. Recruitment

settings included NICU level I, level II

(intermediate care to grow and gain

feeding skills), and level III (high-risk

care for infants requiring advanced

treatment to sustain life), and no

included studies provided MT after

discharge from the NICU. GA, defined

as the time elapsed between the first

day of the last menstrual period and

the day of delivery, 32 varied from

24 to 37 weeks with a median of 32

weeks. Birth weight varied from 620

g to 2715 g. Infants’ postmenstrual

age at study start (the sum of GA,

as defined above, and chronological

age [the time elapsed since birth 32])

varied from 27 to 47 weeks. All

trials included medically and

clinically stable preterm neonates.

Some studies included infants

with all grades of periventricular

leukomalacia or/and intraventricular

PEDIATRICS Volume 138 , number 3 , September 2016 7

Firs

t Au

thor

Stu

dy

Su

bje

cts

Inte

rven

tion

Gro

up

(s)

Con

trol

Gro

up

(s)

Ou

tcom

es

Year

Cou

ntr

yD

esig

nN

Infa

nt

Par

tici

pan

ts

Ran

dom

ized

(An

alyz

ed)

N P

aren

t

Par

tici

pan

ts

Ran

dom

ized

(An

alyz

ed)

GA

at

Bir

th

(wk)

Pos

tmen

stru

al

Age

at S

tud

y

Sta

rt (

wk)

BW

(g)

Sex

, n

Fem

ale

(%)

Trea

tmen

t To

tal

Ses

sion

s

(Tim

e)

Du

rati

on

(d)

1) In

clu

ded

in

Met

a–an

alys

is;

2)S

um

mar

ized

Nar

rati

vely

; 3)N

ot

Incl

ud

ed in

Th

is

Rev

iew

Wal

wor

th20

12U

SP

aral

lel

167

(108

)n

/a(r

ange

,

32–

36)

—<

2500

50 (

46%

)G

rou

p 1

a : D

MS

wit

h

unac

com

pani

ed

live

lulla

by

sin

gin

g;

Gro

up

2a :

DM

S

wit

h g

uit

ar

acco

mp

anie

d

live

lulla

by

sin

gin

g

1 p

er w

k

(20

min

)

Un

til

dis

char

ge

Sta

nd

ard

NIC

U

care

1) L

engt

h o

f

hos

pit

aliz

atio

n,

wei

ght

gain

,

pos

tmen

stru

al

age

at d

isch

arge

,

tim

e to

fu

ll or

al

feed

s; 2

) Ti

me

of r

ecei

vin

g

intr

aven

ous

nu

trit

ion

Wh

ipp

le20

08U

SP

aral

lel

60 (

60)

n/a

(ran

ge,

32–

37)

—<

2500

30 (5

0%)

PAL

1 (1

5 m

in)

11:

Pac

ifi er

only

; 2a :

Sta

nd

ard

NIC

U c

are

1) H

R, R

R, O

2 S

AT,

beh

avio

r st

ate,

beh

avio

ral

dis

tres

s

BS

ID-II

, Bay

ley

Sca

les

of In

fan

t D

evel

opm

ent-

Sec

ond

Ed

itio

n; B

W, b

irth

wei

ght;

DM

S, d

evel

opm

enta

l m

ult

imod

al s

tim

ula

tion

; IQ

R, i

nte

rqu

arti

le r

ange

; M, m

ean

; Md

n, m

edia

n; M

IBS

, Mot

her

-To-

Infa

nt

Bon

din

g S

cale

; n/a

, not

ap

plic

able

; NFC

S, N

eon

atal

Faci

al C

odin

g S

yste

m; P

AL, P

acifi

er A

ctiv

ated

Lu

llab

y d

evic

e; P

IPP,

Pre

mat

ure

Infa

nt

Pai

n P

rofi

le; S

TAI-C

, STA

I for

Ch

ildre

n; —

, dat

a n

ot a

vaila

ble

.a

The

trea

tmen

t gr

oup

sel

ecte

d f

or a

nal

ysis

.b R

evie

wed

nar

rati

vely

bec

ause

no

usa

ble

en

d-p

oin

t d

ata

for

met

a-an

alys

is w

ere

avai

lab

le.

TABL

E 1

Con

tin

ued


8 BIELENINIK et al

hemorrhage 24, 31; respiratory distress

syndrome, clinical sepsis and small

size for GA 7; and chronic lung

disorders or oxygen dependency.20, 22

Intervention Characteristics

There was marked variation across

studies for type of MT approach

used, with lullabies being the

most common, often with parents

providing live, infant-directed

lullabies. 7, 17, 18, 23 Use of recorded

lullaby music20, 22 or exposure to

recorded maternal voice 19 were less

common. Other variations included

use of the Pacifier Activated Lullaby

system to promote nonnutritive

sucking, 25, 27, 30 Pacifier Activated

Lullaby combined with mother’s

recorded voice, 24 parent-focused

interactive MT, 26 and developmental

multimodal stimulation either with

accompanied or unaccompanied

live lullaby singing. 31 The majority

of studies (n = 13) compared MT

combined with NICU standard care to

standard care alone, 7, 18 – 20, 22 – 28, 30, 31

and 3 studies compared KC combined

with MT to KC alone. 17, 21, 29 In all

studies, trained music therapists

were involved in at least 1 aspect of

study design or implementation.

Outcome Characteristics

Of 45 different outcomes in the

included studies, 39 were deemed

directly related to our outcome

inclusion criteria. We consulted with

a panel of neonatal researchers to

determine which of these outcomes

were most clinically relevant and

included those outcomes in meta-

analyses if they were available from

>1 study. Other relevant outcomes

were summarized narratively (see

Table 1).

We present outcomes of the meta-

analyses first, followed by outcomes

described narratively. Within those 2

sections, we organize presentation of

the results by time span: immediate

(during or directly after a single

intervention session), short-

term (after the completion of the

intervention period), and long-term

outcomes (posthospitalization); and

by level (infant, parent, service-level),

as data permits.

Effects of MT Versus Standard Care: Results of Meta-analyses

An overview of the meta-analysis

results is given in Table 2.

Immediate Effects of MT on Infant Well-Being: Physiologic

Immediate physiologic outcomes

that were meta-analyzed included

heart rate (HR), respiratory rate

(RR), and oxygen saturation (O2

SAT; Fig 2, Table 2). Overall, we

found significant effects favoring MT

for RR (P = .048), a nonsignificant

trend favoring MT for HR (P = .058),

and no effect on O2 SAT (P = .431;

Table 2). Heterogeneity was high for

HR and RR but not for O2 SAT; this

heterogeneity was not explained

by study design ( Table 2). Visual

inspection of results for HR (Fig

2A) suggested Johnston et al 19 as

a potential outlier. The authors of

this study noted that the volume of

the recorded mother’s voice used as

a sound stimulus might have been

too high. For RR, although there

was no obvious outlier ( Fig 2B), the

study showing the least beneficial

effect 27 provided MT offered during a

painful procedure. Overall, the results

suggested that MT reduced infants’ RR

by 3.91 breaths per minute ( Fig 2B).

Immediate Effects of MT on Infant Well-Being: Behavioral

Immediate behavioral outcomes

included measures of behavior state

and behavioral distress. Behavior

state was assessed with slight

variations. Three studies 17, 18, 21 used

a 7-point behavior state numeric rating

scale (adapted from refs 33, 34), 1 27

used continuous recording of 6

behavior states (adapted from ref

33), and 1 7 evaluated the percentage

of time in an active sleep state. All

except 1 7 measured this outcome on

a scale where low scores represent

more time spent in sleep states;

therefore, we reversed the data for

FIGURE 1Flow of studies through the systematic review process.


9PEDIATRICS Volume 138 , number 3 , September 2016

this study. Behavioral distress was

also examined in different ways.

One study20 evaluated the duration

of inconsolable (crying) episodes

in minutes, whereas another 27

assessed stress behaviors and signs

of overstimulation using continuous

recording of 9 categories of minimal

to maximal stress behaviors (adapted

from refs 35, 36). Meta-analyses

of these outcomes suggested

high heterogeneity and overall

nonsignificant results ( Fig 3, Table

2). For behavior state, heterogeneity

was not explained by study design

( Table 2). The study showing the

most beneficial effects of MT 18 was

clinically similar to the other studies

( Fig 3A, Table 1). For behavioral

distress, study design may have

explained the heterogeneity (Table

2). The crossover study 20 showed

a large, significant effect in favor of

MT, whereas the parallel study 27

showed no significant effect ( Fig

3B). However, other factors may also

be responsible for this difference.

More positive results were found

with 2 MT sessions 20 than with 1.27

Less favorable effects were found

for MT during a painful heel stick

procedure 27 than during periods of

inconsolable crying. 20 Overall, there

was no conclusive evidence that

MT improves immediate behavioral

outcomes, in spite of a large overall

effect size for behavioral distress

(SMD –1.47; Fig 3B).

Short-term Effects of MT on Infant and Parent

Effects on infant and parent in the

short term included infant weight

gain, time to full oral feeds, and

maternal anxiety. Weight gain was

analyzed as average daily weight

gain 29, 31 or as total weight gain across

the intervention period 30 (which we

transformed to average daily weight

gain). Heterogeneity was high ( Table

2) and could not be due to study

design because all 3 studies were

parallel. One small study, 29 suggested

as a potential outlier, reported

stronger effects in favor of MT than TABL

E 2

Ove

rvie

w o

f O

utc

omes

Ou

tcom

eU

nit

N o

f S

tud

iesa

N o

f P

arti

cip

ants

aD

iffe

ren

ce B

etw

een

Par

alle

l an

d C

ross

over

Stu

die

s

Ove

rall

Effe

ct (

Ran

dom

Eff

ects

) M

(95%

CI)

PH

eter

ogen

eity

% (

I2 )

Imm

edia

te e

ffec

ts

P

hys

iolo

gic

HR

1/m

in8

(2 P

, 6 C

)57

8 (9

0 P,

488

C)

n.s

.−2

.99

(–6.

08 t

o 0.

11)

.058

69

RR

1/m

in6

(1 P

, 5 C

)50

4 (2

7 P,

477

C)

n.s

.−3

.91

(–7.

8 to

–0.

03)

.048

*79

O2

SAT

Per

cen

t7

(1 P

, 6 C

)51

5 (2

7 P,

488

C)

n.s

.0.

18 (

–0.

26 t

o 0.

62)

.431

0

B

ehav

iora

l

Beh

avio

r st

ate

SM

D5

(1 P

, 4 C

)48

1 (4

0 P,

441

C)

n.s

.−0

.32

(–0.

89 t

o 0.

25)

.275

92

Beh

avio

ral d

istr

ess

SM

D2

(1 P

, 1 C

)62

(40

P, 2

2 C

)**

*−1

.47

(–3.

32 t

o 0.

38)

.120

92

Sh

ort-

term

eff

ects

In

fan

t

Wei

ght

gain

g/d

3 (a

ll P

)16

6 (a

ll P

)—

3.34

(–

3.14

to

9.82

).3

1274

Tim

e to

fu

ll or

al

feed

s

d2

(all

P)

138

(all

P)

—−4

(-1

1.02

to

3.02

).2

640

P

aren

t

Mat

ern

al a

nxi

ety

SM

D3

(1 P

, 2 C

)15

1 (1

3 P,

138

C)

*−1

.82

(–2.

42 t

o –

1.22

)<

.001

***

69

S

ervi

ce-le

vel

Pos

tmen

stru

al a

ge

at d

isch

arge

d2

(all

P)

122

(all

P)

—−2

.5 (

–13

.89

to 8

.9)

.668

41

Len

gth

of

hos

pit

aliz

atio

n

d5

(all

P)

354

(all

P)

—−3

.27

(–10

.7 t

o 4.

16)

0.38

829

C, c

ross

over

RC

T; C

I, 95

% c

onfi

den

ce in

terv

al; M

, mea

n; n

.s, n

ot s

ign

ifi ca

nt;

P, p

aral

lel R

CT;

—, n

ot a

pp

licab

le.

a N

um

ber

s an

alyz

ed.

*** P

< .0

01.

* P <

.05.


10 BIELENINIK et al

the remaining studies ( Fig 4A), and

may have been affected by KC, which

was a part of the intervention. Time

to full oral feeds was defined as days

before nipple feeding, computed

as days from birth to date of last

nasogastric/orogastric tube feed.

Heterogeneity was low ( Table 2). The

average effect of 4 days fewer until

full oral feeds for those receiving MT

was not significant ( Fig 4B). Maternal

anxiety was analyzed using the State-

Trait Anxiety Inventory (STAI) 17, 21

or its Colombian adaptation. 29 We

selected state anxiety (STAI Factors

1 and 2 in ref 29) because it may

be a more sensitive indicator of

caregivers’ current distress than

trait anxiety. Heterogeneity was high

( Table 2), with the parallel study 29

showing smaller effects than the

2 crossover studies (Fig 4C). The

overall effect was significant, with a

large effect size in favor of MT (SMD,

–1.82; Fig 4C). In summary, we found

evidence of a beneficial effect of MT

on maternal anxiety, whereas there

FIGURE 2Immediate effects of MT on infant well-being: Physiologic. CI, 95% confi dence interval.



was not enough evidence to confirm

or refute any effects on other short-

term outcomes.

Short-term Effects of MT on Service-Level Outcomes

Postmenstrual age at discharge

and length of hospitalization

were assessed in a number of

parallel RCTs. We found moderate

heterogeneity ( Table 2). Confidence

intervals were wide ( Fig 5), so

there was too little evidence to

confirm or refute any effects on

these outcomes.

Effects of MT: Narrative Summary of Additional Outcomes

An overview of the outcomes

included in the narrative summary is

given in Table 1.

Immediate Effects of MT on Infant Well-Being: Physiologic/Behavioral

Blood pressure was assessed in 1

study and showed no statistically

significant difference for

inconsolable/crying infants receiving

recorded lullaby music. 20 End-point

data were not available from Calabro

et al, 22 which precluded inclusion of

the study in meta-analyses, but no

significant effects were found for HR,

RR, or O2 SAT.

One study assessed the percentage

of time in a quiet alert behavior

state, 7 demonstrating a statistically

significant increase during

presentation of live lullaby music

followed by a decrease after

intervention. Another study 22

assessing behavioral distress

using 11 categories of negative

disorganized states (adapted

from ref 34), but without usable

end-point data for meta-analysis

reported, found no significant

effects.

Short-term Effects of MT on Infant: Physiologic/Behavioral

There was no statistically significant

difference between MT and standard

care in studies assessing head

circumference, 28, 29 infant size, 29 or

discharge weight. 24, 29, 30

Use of the Pacifier Activated Lullaby

significantly improved oral feeding

rates, 24, 25 oral feeding volume, 24

and oral feeding frequency. 24

Live, parent-preferred, culturally-

specific lullabies were associated

with higher levels of caloric intake

and sucking behavior than a well-

known lullaby.7 Infants receiving

developmental multimodal

stimulation took less time to

integrate feeding behaviors than

control infants as demonstrated by a

positive trend for decreased number

of days receiving intravenous

nutrition and decreased number of

days to full oral feeds. 31

Short-term Effects of MT on Parent

Mother–infant bond was assessed in

2 studies ( Table 1), but data from 1

study 23 were reported incompletely

and could not be included in a

meta-analysis. Results from both

trials were nonsignificant. 23, 29

FIGURE 3Immediate effects of MT on infant well-being: Behavioral. Note: Means for Loewy 2013 were reversed to match the directionality of the outcome with the other studies. CI, 95% confi dence interval.


12 BIELENINIK et al

Effects of mothers’ singing on

their adjustment to their preterm

infants was evaluated in 1 study,

again with nonsignificant results.23

Another study 7 found a statistically

significant decrease in parental

perception of stress, which

could, however, not be compared

between conditions because of the

design.

Long-term Effects of MT on Infant and Parent

One study evaluated long-term

outcomes in mothers 26 and another

study in infants. 29 Mother-focused

interactive MT with or without

KC led to statistically significant

increases in breastfeeding rates

at first follow-up visit (7–15 days

postdischarge), and nonsignificant

trends toward increased

breastfeeding rates at point of

discharge and 30 and 60 days after

discharge. 26 One study 29 aimed to

assess infant development, but did

not analyze these data because of

high attrition.

Risk of Bias of Included Studies

The use of cross-over designs and

wash-out periods were generally

judged to be adequate. Lack of clarity

was relatively common for details of

randomization procedures (sequence

generation, allocation concealment)

and whether outcome assessors

were blinded ( Table 3). None of the

included studies tested the success of

blinding.

DISCUSSION

This systematic review and meta-

analysis examined the effect of MT on

preterm infants and their parents/

caregivers during NICU hospitalization

and after discharge to home. Although

the impact of MT on preterm infants

and caregivers has long been of

interest, to our knowledge this study

is the first comprehensive meta-

analytic review restricted to RCTs

with music therapist involvement.

Our meta-analysis showed significant

positive effects of MT on the

clinically important outcomes of

RR and maternal anxiety. Narrative

synthesis of less common outcomes

also suggested some benefits of MT

during and immediately after NICU

hospitalization.

FIGURE 4Short-term effects of MT: Infant and parent. CI, 95% confi dence interval.



MT reduced infants’ RR by 3.91

breaths per minute, an effect that

probably carries clinical significance

as an indicator of a relaxation

response. Although the effect on

reduced HR with MT intervention did

not achieve statistical significance, an

observed trend toward lowered HR

is also consistent with a relaxation

response. Taken together, these

findings suggest that MT lowers

stress and contributes to clinical

stability.

The significant positive, short-term

effect of MT on maternal anxiety

was large according to Cohen’s

guidelines for interpreting effect

sizes. 16 The observed reduction

in mean anxiety scores also

corresponded to a shift from clinical

to subclinical levels of anxiety 37 in

2 of the 3 included studies. 17, 21

Elevated maternal anxiety is

associated with postpartum

depression38 and impaired

parenting 39 in mothers of preterm

infants, whereas reduction in

maternal anxiety is associated with

improvements in child development

during the first 2 years of life. 40

The 3 studies included in the meta-

analysis of maternal anxiety all

used live music in conjunction

with KC, demonstrating greater

improvements than with KC alone.

These results support the beneficial

impact of interventions that instate

the parent in a nurturing and

caregiving role (provider of KC) that

includes engagement in live music.

Prematurity is associated with

significant public health costs. The

high prevalence and costs demand

attention in many high-income

countries. Preterm infants are

hospitalized longer than full-term

infants (13 vs 1.5 days 41), and

daily costs of NICU care per infant

exceed $3500 in the United States. 42

The 3-day reduction in length of

hospitalization in our meta-analysis

failed to reach statistical significance,

but if confirmed in a larger study,

would have important implications

for service costs.

Limitations

One of the major limitations of

this review is the lack of long-term

observations. We intentionally

included preterm infants through

the first 3 years of life to assess

the long-term impact of MT on

preterm infants and their parents/

caregivers. Despite an exhaustive

search, we were only able to

identify 2 studies that assessed

long-term outcomes for infants or

parents. 26, 29 Our restriction to RCTs

with music therapist involvement

undoubtedly reduced the number

of eligible studies that assess long-

term outcomes for preterm infants.

Six of the 16 studies included in the

systematic review were crossover

studies, precluding the ability to

assess long-term effects, and 11

studies had intervention protocols

lasting ≤5 days.

The comprehensiveness of our

review is also impacted by the

limited sample size in areas of long-

term and service-level outcomes.

Because it is likely that various other

FIGURE 5Short-term effects of MT: Service-level. CI, 95% confi dence interval.


14 BIELENINIK et al

influences impact service-level and

long-term outcomes due to the distal

nature of these outcomes, larger

sample sizes could better enable

the detection of treatment effects

over time. To address the limitations

of previous systematic papers, we

undertook comprehensive searching

following the strict guidelines of the

PRISMA statement. 13 We believe

this systematic review contains

all relevant studies that have been

conducted in this field, but it is

possible that there are unpublished

studies of which we were not aware.

Studies included in the review

demonstrated a fairly high level of

clinical heterogeneity, especially

variations in the type, duration,

and frequency of MT. This clinical

heterogeneity may have contributed

to heterogeneity in observed effects,

and more research is needed

to systematically explore such

variations and learn from such

comparisons.

Our meta-analysis was limited by

data reporting issues in the included

studies. We attempted to contact

the authors of 11 of the 16 included

studies to request missing data, and

successfully obtained requested data

in 6 cases. Our systematic review

was also limited by the lack of

transparent intervention reporting

among included studies in relation to

total number of sessions and detailed

protocols for intervention and

control groups. However, a majority

of the included studies specified

type of music used and duration of

intervention.

Implications for Clinical Practice

The findings of this review have

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practice. There was a strong

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involvement in MT within the included

studies, either targeting mothers

as participants, 17, 26 using mother’s

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ACKNOWLEDGMENTS

We would like to thank Elise Marie

Angeltveit, MT graduate student, who

assisted with database searching

and record screening, and Trond

Jacob Markestad, Bente Vederhus,

and Hallvard Martin Reigstad for

consultation regarding the clinical

relevance of outcome measures and

meta-analysis results.

ABBREVIATIONS

GA: gestational age

HR: heart rate

KC: kangaroo care

MT: music therapy

O2 SAT: oxygen saturation

RCT: randomized controlled

trial

RR: respiratory rate

SMD: standardized mean

difference

STAI: State-Trait Anxiety

Inventory

FINANCIAL DISCLOSURE: The authors have indicated they have no fi nancial relationships relevant to this article to disclose.

FUNDING: The study was funded through the University of Bergen, POLYFON Kunnskapsklynge for musikkterapi, and the Research Council of Norway (grant

213844, the Clinical Research and the Mental Health Programmes).

POTENTIAL CONFLICT OF INTEREST: Dr Bieleninik has indicated she has no potential confl icts of interest to disclose. Claire Ghetti and Christian Gold are trained

music therapists.


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DOI: 10.1542/peds.2016-0971 originally published online August 25, 2016; 2016;138;Pediatrics

Lucja Bieleninik, Claire Ghetti and Christian GoldMusic Therapy for Preterm Infants and Their Parents: A Meta-analysis

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Music Therapy for Preterm Infants and Their Parents: A Meta … · Music Therapy for Preterm Infants and Their Parents: A Meta-analysis Łucja Bieleninik, PhD, a Claire Ghetti, PhD,