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
by guest on May 18, 2020www.aappublications.org/newsDownloaded from
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
2 by guest on May 18, 2020www.aappublications.org/newsDownloaded from
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).
3 by guest on May 18, 2020www.aappublications.org/newsDownloaded from
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
by guest on May 18, 2020www.aappublications.org/newsDownloaded from
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
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
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
ange
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,
hea
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
by guest on May 18, 2020www.aappublications.org/newsDownloaded from
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
by guest on May 18, 2020www.aappublications.org/newsDownloaded from
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
by guest on May 18, 2020www.aappublications.org/newsDownloaded from
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.
by guest on May 18, 2020www.aappublications.org/newsDownloaded from
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.
by guest on May 18, 2020www.aappublications.org/newsDownloaded from
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.
by guest on May 18, 2020www.aappublications.org/newsDownloaded from
11PEDIATRICS Volume 138 , number 3 , September 2016
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.
by guest on May 18, 2020www.aappublications.org/newsDownloaded from
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.
by guest on May 18, 2020www.aappublications.org/newsDownloaded from
13PEDIATRICS Volume 138 , number 3 , September 2016
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.
by guest on May 18, 2020www.aappublications.org/newsDownloaded from
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
important implications for future
practice. There was a strong
tendency toward facilitating parental
involvement in MT within the included
studies, either targeting mothers
as participants, 17, 26 using mother’s
recorded voice for infants, 19, 23, 24 or TABL
E 3
Ris
k of
Bia
s in
Incl
ud
ed S
tud
ies
Stu
dy
IDS
tud
y D
esig
nC
ross
-ove
r
Des
ign
App
rop
riat
e?
Adeq
uat
e
Ran
dom
izat
ion
of t
he
Ord
erin
g
of T
reat
men
ts?
Adeq
uat
e
Was
h-o
ut
Per
iod
To
Red
uce
Car
ry-o
ver
Effe
cts?
Adeq
uat
e
Seq
uen
ce
Gen
erat
ion
Allo
cati
on
Con
ceal
men
t
Blin
din
g
(Ob
ject
ive,
Ob
serv
ed
Ou
tcom
es)
Blin
din
g
(Su
bje
ctiv
e,
Sel
f-R
epor
t
Ou
tcom
es)
Inco
mp
lete
Ou
tcom
e
Dat
a
Add
ress
ed
(Sh
ort-
term
Ou
tcom
es
[2–
6 w
k])
Inco
mp
lete
Ou
tcom
e
Dat
a
Add
ress
ed
(Sh
ort-
term
outc
omes
[>6
wk]
)
Free
of
Sel
ecti
ve
Rep
orti
ng
Free
of
Oth
er
Bia
s
Arn
on 2
006
Cro
ssov
erye
su
ncl
ear
yes
n/a
un
clea
ru
ncl
ear
un
clea
rye
sn
/aye
sye
s
Arn
on 2
014
Cro
ssov
erye
sye
sye
sn
/aye
su
ncl
ear
no
yes
n/a
yes
yes
Cal
abro
200
3P
aral
lel
n/a
n/a
n/a
yes
un
clea
rye
sn
/aye
sn
/aye
sye
s
Cas
sid
y 20
09P
aral
lel
n/a
n/a
n/a
un
clea
ru
ncl
ear
un
clea
rn
/aye
sn
/an
oye
s
Cev
asco
200
8P
aral
lel
n/a
n/a
n/a
un
clea
ru
ncl
ear
un
clea
rn
oye
sn
/an
ou
ncl
ear
Ch
orn
a 20
14P
aral
lel
n/a
n/a
n/a
un
clea
ru
ncl
ear
yes
n/a
yes
n/a
yes
yes
Ette
nb
erge
r
2014
Par
alle
ln
/an
/an
/aye
sye
su
ncl
ear
no
yes
n/a
no
yes
Joh
nst
on 2
007
Cro
ssov
erye
sye
sye
sn
/au
ncl
ear
yes
n/a
yes
n/a
yes
no
Keit
h 2
009
Cro
ssov
erye
su
ncl
ear
yes
n/a
un
clea
rn
on
/aye
sn
/an
oye
s
Loew
y 20
13C
ross
over
yes
yes
yes
n/a
yes
yes
n/a
yes
n/a
yes
yes
Sch
lez
2011
Cro
ssov
erye
sye
sye
sn
/au
ncl
ear
no
no
yes
n/a
yes
yes
Sta
nd
ley
2003
Par
alle
ln
/an
/an
/au
ncl
ear
un
clea
rye
sn
/aye
sn
/aye
sye
s
Sta
nd
ley
2010
Par
alle
ln
/an
/an
/au
ncl
ear
yes
yes
n/a
yes
n/a
yes
no
Vian
na
2011
Par
alle
ln
/an
/an
/aye
sye
sye
sn
/aye
sye
sye
sye
s
Wal
wor
th 2
012
Par
alle
ln
/an
/an
/aye
su
ncl
ear
yes
n/a
no
n/a
yes
no
Wh
ipp
le 2
008
Par
alle
ln
/an
/an
/au
ncl
ear
un
clea
rn
on
/aye
sn
/aye
sye
s
n/a
, not
ap
plic
able
.
by guest on May 18, 2020www.aappublications.org/newsDownloaded from
15PEDIATRICS Volume 138 , number 3 , September 2016
REFERENCES
1. Blencowe H, Cousens S, Chou D, et al;
Born Too Soon Preterm Birth Action
Group. Born too soon: the global
epidemiology of 15 million preterm
births. Reprod Health. 2013;
10(suppl 1):S2
2. Saigal S, Doyle L. An overview of
mortality and sequelae of preterm
birth from infancy to adulthood.
Lancet. 2008;371(9608):
261–269
3. American Academy of Pediatrics.
Follow-up care of high-risk infants.
Pediatrics. 2004;114 (suppl 5):
1377–1397
4. Standley J. Music therapy research in
the NICU: an updated meta-analysis.
Neonatal Netw. 2012;31(5):311–316
5. Beth Israel Medical Center. First
Sounds: Rhythm, Breath and
Lullaby Training Roster. Available
at: http:// nicumusictherapy. com/
Nicumusictherapy/ Grandparents_
Training_ Roster. html. Accessed May 31,
2016
6. Standley JM. Premature infants:
Perspectives on NICU-MT practice.
including both parents/caregivers
when applicable. 7, 21, 29 A key policy
priority should be taken to involve both
parents in MT sessions to facilitate
mutually beneficial interactions that
support infant development, help
parents assume a primary caregiving
role, and foster healthy bonding
during the critical period of NICU
hospitalization. Greater efforts are
needed to ensure follow-up of preterm
infants after discharge.
Implications for Future Research
This review highlighted the need for
improved transparency in research
reporting and several areas that
require additional investigation.
Rigorously designed studies using
larger sample sizes, standardized
outcome measures, and interventions
implemented by music therapists
with specialized NICU training are
required. Transparent and complete
reporting of study interventions
and results is crucial, because
transparency enables replication
and transfer of research to clinical
practice settings. 43 Researchers
are strongly encouraged to follow
pertinent reporting guidelines, such as
those by Robb et al, 43 for music-based
interventions, which are evidence-
based and consistent with CONSORT
and TREND statements. Conducting
parallel group RCTs to evaluate
long-term effects of MT and extending
intervention periods past discharge
from the NICU will help assess the
long-term impact of MT, a substantial
gap in knowledge at present.
Several of the included studies had
small sample sizes, which may put
them at higher risk for underpowered
analyses. Reliable and valid outcome
measures are required, especially
in the area of assessing parental
psychological outcomes. Ideally,
parental psychological outcomes
would be measured over longer
periods of time to assess durability
of change. Because preterm parents
perceive premature birth as a stressful
and traumatic situation, research
should address parental stress,
anxiety, postpartum depression and
quality of life. Separation between
infant and parents because of intense
or prolonged hospitalization can
complicate development of healthy
parent–infant relationships. Research
assessing the long-term effects of
MT on the development of healthy
infant/parent bonding and secure
attachment is indicated.
The current systematic review
reflected a high level of clinical
heterogeneity, which may be
expected given the broad range of
outcomes that may be addressed
with MT. More research is needed
to explore the differential impact of
various MT approaches, frequencies,
durations, and interventional time
points. By systematically analyzing
the impact of clinical heterogeneity,
we may be able to determine when
and how MT is best used to promote
certain outcomes.
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.
by guest on May 18, 2020www.aappublications.org/newsDownloaded from
16 BIELENINIK et al
Voices: A World Forum for Music
Therapy. 2014:14(2). Available at:
https:// voices. no/ index. php/ voices/
article/ view/ 767.
7. Loewy J, Stewart K, Dassler AM,
Telsey A, Homel P. The effects of music
therapy on vital signs, feeding, and
sleep in premature infants. Pediatrics.
2013;131(5):902–918
8. Standley JM. Music therapy for the
neonate. Newborn Infant Nurs Rev.
2001;1(4):211–216
9. Standley JM. A meta-analysis of
the effi cacy of music therapy for
premature infants. J Pediatr Nurs.
2002;17(2):107–113
10. Hartling L, Shaik MS, Tjosvold L, Leicht
R, Liang Y, Kumar M. Music for medical
indications in the neonatal period:
a systematic review of randomised
controlled trials. Arch Dis Child Fetal
Neonatal Ed. 2009;94(5):F349–F354
11. Hodges A, Wilson L. Pretem infants’
responses to music: An intergrative
literature review. South Online J Nurs
Res. 2010;10(3). Available at: www.
resourcenter. net/ images/ snrs/ fi les/
sojnr_ articles2/ vol10num03art05. html.
12. Haslbeck FD. Music therapy for
premature infants and their parents:
an integrative review. Nord J Music
Ther. 2012;21(3):203–226
13. Moher D, Liberati A, Tetzlaff J, Altman
DG; PRISMA Group. Preferred reporting
items for systematic reviews and
meta-analyses: The PRISMA statement.
PLoS Med. 2009:6(7):e1000097
14. World Health Organization. WHO:
Recommended defi nitions, terminology
and format for statistical tables
related to the perinatal period and
use of a new certifi cate for cause
of perinatal deaths. Modifi cations
recommended by FIGO as amended
October 14, 1976. Acta Obstet Gynecol
Scand. 1977;56(3):247–253
15. Higgins JPT, Deeks JJ, Altman DG,
Cochrane Statistical Methods Group.
Special topics in statistics. In:
Higgins JPT, Green S, eds. Cochrane
Handbook for Systematic Reviews of
Interventions. Chichester, UK: Wiley-
Blackwell; 2008
16. Cohen J. Statistical power analysis
for the behavioral sciences, 2nd ed.
Hillsdale, NJ: Lawrence Erlbaum; 1988
17. Arnon S, Diamant C, Bauer S, Regev
R, Sirota G, Litmanovitz I. Maternal
singing during kangaroo care led to
autonomic stability in preterm infants
and reduced maternal anxiety. Acta
Paediatr. 2014;103(10):1039–1044
18. Arnon S, Shapsa A, Forman L, et al. Live
music is benefi cial to preterm infants
in the neonatal intensive care unit
environment. Birth. 2006;33(2):131–136
19. Johnston CC, Filion F, Nuyt AM.
Recorded maternal voice for preterm
neonates undergoing heel lance. Adv
Neonatal Care. 2007;7(5):258–266
20. Keith DR, Russell K, Weaver BS.
The effects of music listening
on inconsolable crying in
premature infants. J Music Ther.
2009;46(3):191–203
21. Schlez A, Litmanovitz I, Bauer S,
Dolfi n T, Regev R, Arnon S. Combining
kangaroo care and live harp music
therapy in the neonatal intensive
care unit setting. Isr Med Assoc J.
2011;13(6):354–358
22. Calabro J, Wolfe R, Shoemark H. The
effects of recorded sedative music
on the physiology and behaviour of
premature infants with a respiratory
disorder. Aust J Music Ther.
2003;14:3–19
23. Cevasco AM. The effects of
mothers’ singing on full-term and
preterm infants and maternal
emotional responses. J Music Ther.
2008;45(3):273–306
24. Chorna OD, Slaughter JC, Wang
L, Stark AR, Maitre NL. A pacifi er-
activated music player with mother’s
voice improves oral feeding
in preterm infants. Pediatrics.
2014;133(3):462–468
25. Standley JM. The effect of music-
reinforced nonnutritive sucking on
feeding rate of premature infants. J
Pediatr Nurs. 2003;18(3):169–173
26. Vianna MN, Barbosa AP, Carvalhaes AS,
Cunha AJ. Music therapy may increase
breastfeeding rates among mothers
of premature newborns: a randomized
controlled trial. J Pediatr (Rio J).
2011;87(3):206–212
27. Whipple J. The effect of music-
reinforced nonnutritive sucking on
state of preterm, low birthweight
infants experiencing heelstick. J Music
Ther. 2008;45(3):227–272
28. Cassidy JW. The effect of decibel level
of music stimuli and gender on head
circumference and physiological
responses of premature infants in the
NICU. J Music Ther. 2009;46(3):180–190
29. Ettenberger M, Odell-Miller H, Cardenas
CR, Serrano ST, Parker M, Camargo
Llanos SM. Music therapy with
premature infants and their caregivers
in colombia – A mixed methods pilot
study including a randomized trial.
Voices: A World Forum for Music
Therapy. 2014:14(2). Available at:
https:// voices. no/ index. php/ voices/
article/ view/ 756
30. Standley JM, Cassidy J, Grant R, et al.
The effect of music reinforcement for
non-nutritive sucking on nipple feeding
of premature infants. Pediatr Nurs.
2010;36(3):138–145
31. Walworth D, Standley JM, Robertson
A, Smith A, Swedberg O, Peyton
JJ. Effects of neurodevelopmental
stimulation on premature infants in
neonatal intensive care: Randomized
controlled trial. J Neonatal Nurs.
2012;18(6):210–216
32. Engle WA; American Academy of
Pediatrics Committee on Fetus and
Newborn. Age terminology during
the perinatal period. Pediatrics.
2004;114(5):1362–1364
33. Als H. Manual for the Naturalistic
Observation of Newborn Behavior
(Preterm and Full Term). Boston: The
Children’s Hospital; 1982
34. Als H, Lawhon G, Brown E, et al.
Individualized behavioral and
environmental care for the very low
birth weight preterm infant at high
risk for bronchopulmonary dysplasia:
neonatal intensive care unit and
developmental outcome. Pediatrics.
1986;78(6):1123–1132
35. Burns K, Cunningham N, White-Traut
R, Silvestri J, Nelson MN. Infant
stimulation: modifi cation of an
intervention based on physiologic
and behavioral cues. J Obstet Gynecol
Neonatal Nurs. 1994;23(7):581–589
36. Als H, Lester BM, Tronick EZ, Brazelton
TB. Toward a research instrument for
the Assessment of Preterm Infants’
Behavior (APIB). In: Fitzgerald BMLHE,
by guest on May 18, 2020www.aappublications.org/newsDownloaded from
17PEDIATRICS Volume 138 , number 3 , September 2016
Yogman MW, eds. Theory and Research
in Behavioral Pediatrics. NY: Plenum;
1982:65–132
37. Julian LJ. Measures of anxiety: State-
Trait Anxiety Inventory (STAI), Beck
Anxiety Inventory (BAI), and Hospital
Anxiety and Depression Scale-Anxiety
(HADS-A). Arthritis Care Res (Hoboken).
2011;63(suppl 11):S467–S472
38. Beck CT. Predictors of postpartum
depression: an update. Nurs Res.
2001;50(5):275–285
39. Zelkowitz P, Bardin C, Papageorgiou
A. Anxiety affects the relationship
between parents and their very low
birth weight infants. Infant Ment
Health J. 2007;28(3):296–313
40. Benzies, KM, Magill-Evans JE, Hayden
KA, Ballantyne M. Key components
of early intervention programs for
preterm infants and their parents:
A systematic review and meta-
analysis. BMC Pregnancy Childbirth.
2013:13(suppl 1):S10
41. Howson CP, Kinney MV, McDougall L,
Lawn JE, Born Too Soon Action Group.
Born Too Soon: Preterm birth matters.
Reprod Health, 2013:10(suppl 1):S1
42. Muraskas J, Parsi K. The cost
of saving the tiniest lives: NICUs
versus Prevention. Virtual Mentor.
2008;10(10):655–658
43. Robb SL, Burns DS, Carpenter JS.
Reporting guidelines for music-based
interventions. J Health Psychol.
2011;16(2):342–352
by guest on May 18, 2020www.aappublications.org/newsDownloaded from
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
ServicesUpdated Information &
http://pediatrics.aappublications.org/content/138/3/e20160971including high resolution figures, can be found at:
Referenceshttp://pediatrics.aappublications.org/content/138/3/e20160971#BIBLThis article cites 35 articles, 5 of which you can access for free at:
Subspecialty Collections
http://www.aappublications.org/cgi/collection/neonatology_subNeonatologysubhttp://www.aappublications.org/cgi/collection/fetus:newborn_infant_Fetus/Newborn Infantine_subhttp://www.aappublications.org/cgi/collection/evidence-based_medicEvidence-Based Medicinefollowing collection(s): This article, along with others on similar topics, appears in the
Permissions & Licensing
http://www.aappublications.org/site/misc/Permissions.xhtmlin its entirety can be found online at: Information about reproducing this article in parts (figures, tables) or
Reprintshttp://www.aappublications.org/site/misc/reprints.xhtmlInformation about ordering reprints can be found online:
by guest on May 18, 2020www.aappublications.org/newsDownloaded from
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
http://pediatrics.aappublications.org/content/138/3/e20160971located on the World Wide Web at:
The online version of this article, along with updated information and services, is
1073-0397. ISSN:60007. Copyright © 2016 by the American Academy of Pediatrics. All rights reserved. Print
the American Academy of Pediatrics, 141 Northwest Point Boulevard, Elk Grove Village, Illinois,has been published continuously since 1948. Pediatrics is owned, published, and trademarked by Pediatrics is the official journal of the American Academy of Pediatrics. A monthly publication, it
by guest on May 18, 2020www.aappublications.org/newsDownloaded from