DEVELOPMENTAL MEDICINE & CHILD NEUROLOGY SYSTEMATIC REVIEW

Speech and language interventions for infants aged 0 to 2 years

at high risk for cerebral palsy: a systematic review
OLENA CHORNA 1 | ELLYN HAMM 1 | CAITLIN CUMMINGS 2 | ASHLEY FETTERS2 | NATHALIE L MAITRE 1,3
1 Department of Pediatrics, Center for Perinatal Research, Nationwide Children’s Hospital, Columbus, OH; 2 Division of Clinical Therapies, Department of
Speech-Language Pathology, Nationwide Children’s Hospital, Columbus, OH; 3 Department of Hearing and Speech Sciences, Vanderbilt University Medical Center,
Nashville, TN, USA.
Correspondence to Nathalie L Maitre at Department of Pediatrics, 700 Children’s Drive, WB6225, Columbus, OH 43205, USA. E-mail: nathalie.maitre@nationwidechildrens.org

This article is commented on by Novak et al. on pages 343–344 of this issue.

PUBLICATION DATA AIM We evaluated the level of evidence of speech, language, and communication interventions
Accepted for publication 14th October for infants at high-risk for, or with a diagnosis of, cerebral palsy (CP) from 0 to 2 years old.
2016. METHOD We performed a systematic review of relevant terms. Articles were evaluated based
Published online 29th November 2016. on the level of methodological quality and evidence according to A Measurement Tool to
Assess Systematic Reviews (AMSTAR) and Grading of Recommendations Assessment,
ABBREVIATION Development and Evaluation (GRADE) guidelines.
GRADE Grading of Recommendations RESULTS The search terms provided 17 publications consisting of speech or language
Assessment, Development and interventions. There were no interventions in the high level of evidence category. The overall
Evaluation level of evidence was very low. Promising interventions included Responsivity and
Prelinguistic Milieu Teaching and other parent–infant transaction frameworks.
INTERPRETATION There are few evidence-based interventions addressing speech, language,
and communication needs of infants and toddlers at high risk for CP, and none for infants
diagnosed with CP. Recommendation guidelines include parent–infant transaction programs.

A recent emphasis on early diagnosis for cerebral palsy
(CP) in the first years of life, when brain plasticity is at its
greatest, has prompted scrutiny of therapeutic interven-
tions aimed at improving long-term neurodevelopmental
outcomes of infants at the highest risk for CP.1–4 However,
increased surveillance before the age of 2 years may high-
light the paucity of rigorously designed studies targeting
CP, and the difficulties in identifying these infants. A
growing body of research focuses instead on infants at high
risk for CP, as up to two-thirds of infants born extremely
preterm with encephalopathy of prematurity or neonates
with perinatal brain insults receive a later diagnosis of
CP.5 These infants often have abnormalities on neurologi-
cal examinations, the General Movements Assessment and/
or neuroimaging findings that together confer >90% risks
of developing CP.6–10 Some pediatric populations with
other developmental disorders can also have a higher risk
of coexisting CP, even though they show no clear perinatal
insults.11 In addition to their motor impairments, infants at
high risk for CP have frequent early speech and language
problems.12,13 By preschool age, infants born very preterm
have poorer receptive and expressive language development
than infants born at term, with delays in vocabulary size,
quality of word production, syntax, morphology, and com-
prehension.14,15 These impairments of receptive and
expressive language are present independent of
socioeconomic status, and can increase in severity from
preschool to school age.15–17 Infants born at term with
neonatal encephalopathy secondary to perinatal asphyxia,
ischemia, or other birth-related events are also at high risk
for CP with concurrent language impairments18 in the first
2 years. Similarly, infants with neonatal venous or arterial
stroke display long-term language problems despite evi-
dence of brain reorganization after injury.19,20 Collectively,
although they may not have met clinical criteria for referral
for services in infancy and early childhood,21 school-age
children with CP22–24 have extremely high rates of speech
and language impairments.
Language development from birth to 2 years presents
multiple opportunities for therapeutic interventions during
critical windows. Speech sound differentiation and voice
recognition occur in the earliest phase of language devel-
opment. Vocal learning begins in utero and continues
through infancy when infants have the ability to discrimi-
nate the phonetic constraints of language as well as
mother’s versus strangers’25–27 voices. While auditory pro-
cessing is often adversely affected by prematurity and
neonatal insults, to date no interventional studies have tar-
geted improvements of this neonatal cortical function.
In early infancy, language patterns are fundamentally
perceived as melodies and reflected in infants’ initial sound
utterances,25 with melody arc duration and amplitude

© 2016 Mac Keith Press DOI: 10.1111/dmcn.13342 355

developing in parallel, reflecting the coupled advancement
of phonation and articulation required for successful
speech production.28 Non-native language-specific vowel-
like sounds are produced at 3 months and language-speci-
fic perceptual organization of vowels begins from
6 months.29 During this period, social interactions, when
provided contingently with infant vocalizations can then
increase vocalizations and babbling.30 The next stage of
speech and language development includes phonating, with
jaw movements, and several months later, organized sylla-
bles reflecting prior exposure to speech.25 During the first
year of life, language perception (receptive) and production
(expressive) is again dependent on social interactions, along
with development of cortical areas responsible for improve-
ments of working memory and pattern recognition.31
Interventions have targeted these phases of language devel-
opment in infants at high risk for CP, particularly through
enhanced parental responsiveness. During the second year
of life, rapid language acquisition depends on multiplica-
tion of associations between different cortical processing
areas, reflected in a rapid increase in the amount of words
a child understands and produces. Interventions leveraging
the cortical associations between language and motor sys-
tems may help mediate the establishment of more complex
functions in children with CP,32 such as motor function
and behavior. Strategies addressing the early communica-
tion problems of children at high risk for CP or with CP
may be essential to long-term emotional adaptation and
social participation.33,34
The aim of this systematic review was therefore to evalu-
ate the level of evidence of current interventions targeting
the speech and language deficits of infants at high risk for,
or with, CP in the first 2 years of life. We focused on the
speech and language delays of infants with typical hearing
and without known genetic conditions that could also
affect their language development.
METHOD
This systematic review includes randomized clinical trials
along with other study designs to demonstrate the current
level of evidence. The review follows the guidelines of the
International Clinical Guideline for Cerebral Palsy search
terms and was performed in accordance with the principles
of A Measurement Tool to Assess Systematic Reviews
(AMSTAR, Table I),35 the Grading of Recommendations
Table I: AMSTAR methodological quality rating checklist
Items
1 2 3 4 5 6 7 8
63
Novak et al. Y Y Y N Y Y Y Y
Law et al. (Cochrane)55 Y Y Y Y Y Y Y Y
Pennington et al.33 Y Y Y Y Y Y Y Y
Pennington et al.64 Y Y Y Y Y Y Y Y
Pennington et al.65 Y Y Y Y Y Y Y Y
AMSTAR, A Measurement Tool to Assess Systematic Reviews; Y,
yes; N, no.
356 Developmental Medicine & Child Neurology 2017, 59: 355–360
What this paper adds
• The overall level of evidence of studies in infants at high risk for cerebral
palsy (CP) is very low.
• Several promising interventions are based on parent–infant transaction
programs.
• Recommended interventions do not specifically target infants with a diagno-
sis of CP.
Assessment, Development and Evaluation (GRADE),36 and
the Cochrane Collaboration’s tool for assessing risk of bias
in randomized trials (Table II).37 This is a review of pub-
lished literature; therefore, it did not require institutional
review board approval.
Search strategy
This review included relevant terms in online research data-
bases. Searches were performed between May and August
2014, and repeated in August 2016 to evaluate the evidence
base of speech and interventions for infants aged 0 to 2 years
with or at high risk for CP. The following databases were
included into the search: PubMED, Cochrane Library,
MEDLINE, PsycINFO, speechBITE American Speech-
Language-Hearing Association (ASHA), SCOPUS, ISI Web
of Knowledge, EBSCO (including CINAHL), and The
Communication Trust – What Works. Search terms are
included in Appendix S1 (online supporting information). In
addition, a grey literature search was performed according
to methodology described in Shpilko.38
The methodological quality of the studies was evaluated
using the AMSTAR rating checklist for systematic reviews,
GRADE level of evidence assessment, and the Cochrane
Risk of Bias checklist for randomized controlled trials.
Details of study quality are outlined in Table SI (online
supporting information).
Inclusion criteria
Published studies of interventions addressing speech and
language with infants at high risk for CP and those with a
diagnosis of CP were included in this review when any of
the study subjects were 2 years corrected age and younger.
Exclusion criteria
Articles were excluded if they were letters, editorials, notes,
books, assessment only (no intervention), protocols, had no
infant language scores/assessments, or had parent assess-
ments of dyad intervention.
Types of interventions
Interventions provided by licensed therapists/professionals
or caregivers/parents trained by therapists/professionals
were included. Individual and group interventions provided
9 10 11
in and out of the home environment were included.
Y Y Y
Y Y Y
Y Y N
Types of participants
Y Y N Studies with human participants between 0 and 24 months
Y Y N corrected age born preterm or with high risk for/diagnosis
of CP were included. High risk for CP was differentiated
from the ‘highest risk for CP’ categorization recently
Table II: The Cochrane Collaboration’s tool for assessing risk of bias table in randomized trials

Random Blinding of Blinding of Incomplete Free of

sequence Allocation participants outcome outcome data selective Free of
References generation concealment and personnel assessments addressed reporting other bias

Speech and language interventions

Kynø et al.46 H L L L L H U
Milgrom et al.47 H H L H H H U
Newnham et al.48 H L L H L H L
Goldfeld et al.66 L H H L H H H
53
Carter et al. H H L H H H U
Glogowska et al.67 H H H H H H H
Yoder and Warren51 H L L L H H U
Buschmann et al.68 L H L H L U U

H, high; L, low; U, unclear.

published.1 As this review focused on speech and language,
General Movements Assessment was not routinely per-
formed in relevant articles. Instead, we focused on perina-
tal or prenatal conditions that are strongly associated with
later CP in published literature (prematurity, encephalopa-
thy, stroke39). We then examined the participants in each
study to determine if they had any conditions associated
with CP in the developmental literature, making them at
higher risk for CP than the general population: born
preterm,5,40–42 autism spectrum disorder,42,43 or pervasive
developmental disorder.11,40
Data review and quality appraisal
Article abstracts were reviewed for inclusion to determine
applicability to the current review. Selected articles were
retained and reviewed by a second independent reviewer to
confirm inclusion/exclusion criteria. Agreement on inclu-
sion and exclusion was achieved. Included articles were
then reviewed in full-text independently by two reviewers.
The authors reviewed and achieved 95% grading agree-
ment on the GRADE level of evidence. Risk of bias was
assessed with the Cochrane criteria. Data from included
studies were extracted using a PRISMA44 data extraction
format, then summarized and assigned a level of evidence.
PRISMA guidelines were followed for compilation of
Table SI.
RESULTS
A total of 653 records were identified in the initial terms
search. After the search, duplicates, presentation materials,
and book chapters were removed. Of the screened articles,
17 were included in the final review. A flow diagram of
study selection in PRISMA format is presented in Figure S1
(online supporting information).
participants in the reviewed studies ranged from n=4 to
n=552.
The following sections discuss the types of interventions
that were included in this review. Per GRADE evaluation,
the cumulative level of evidence was very low. We found
17 studies of speech or language in infants up to
24 months with or at high risk for CP. Of these studies,
none represented high level of evidence, nine were of mod-
erate level, six were low level, and two were very low level.
DISCUSSION
The aim of this systematic review was originally to evalu-
ate the level of evidence of interventions targeting speech
or language problems of infants at high risk for, or with a
CP diagnosis in the first 2 years of life. There were no
CP-specific speech and language interventions for infants
under 24 months corrected age. There were 17 studies
focused on infants at high risk for CP. Therefore, the rec-
ommendations for guideline implementation are weak.
Summary of the evidence from level II studies
While there is a considerable body of literature about early
speech, language, and communication development, there
are few reports of evidence-based interventions for infants
at high risk for CP. Most studies identified through this
review had a small number of participants. Of the seven
randomized controlled trials included in this review the
overall level of evidence is low.
No studies focused exclusively on a population of infants
with CP, and no interventions were specifically designed
for this population. Age at intervention and heterogeneity
of the population in infants with high risk for CP were the
main methodological barriers to evaluating cumulative
effectiveness of the interventions.

Sample characteristics Interventions

Five studies were systematic reviews, eight were random-
ized controlled trials, two were quasi-experimental, and
two were case series. The start of interventions ranged
between 30 weeks’ gestation and 23 months corrected age.
Several studies also included infants enrolled above 2 years
corrected age but were included in this review if at least
one of the participants was under 2 years. The number of
Interventions varied among parent, child, or dyad as the
primary recipient, and many were not described in
sufficient detail for replication or clinical implementa-
tion. However, several studies were based on the same
framework, in particular, variants of parent–infant interac-
tion programs and can constitute a single category of inter-
ventions.

Review 357
 The Mother–Infant Transaction Program (MITP45) is a
well-established program of semi-structured sessions imple-
mented in the neonatal intensive care unit and in the home,
post discharge. The program implementation schedule spans
from the last week of hospitalization through the first
90 days after discharge. The intervention focuses on paren-
tal understanding of infant state regulation, awake and sleep
cycles, caregiver–infant interaction, and involvement into
care of the infant. Variations of the MITP program have
been reported over the last few decades, when the program
was first described in the literature.46–49
‘PremieStart’ is a modification of the MITP program.47 It
focuses on improving mother–infant dyad interactions
through an intensive eight-session training program to
recognize and minimize stress responses in infants born pre-
term. It includes touch, movement and massage, skin-to-skin
care, parental feelings affirmation, challenging dysfunctional
thoughts, and parental diary-keeping. The results of the
eight once-weekly sessions and one post-discharge home
visit included increased maternal sensitivity during infant
care, lowered infant stress, and increased responsiveness. At
6 months corrected age intervention infants had signifi-
cantly higher mean scores on the Symbolic Behavior Scales
Developmental Profile Infant–Toddler Checklist.50
Yoder and Warren51 studied the combination of
Responsivity and Prelinguistic Milieu Teaching to address
speech and language development in toddlers with devel-
opmental and cognitive delays. They reported increased
parental responsivity, and accelerated growth in frequency
of child-initiated comments and requests, and lexical den-
sity in a subgroup of participants.
The Hanen It Takes Two to Talk and More Than
Words programs train parents about the importance of
responsivity to their child’s communication attempts in a
positive, prompt, and intentional manner. More Than
Words teaches utilization of the natural environment and
repetitive, predictive structure to support communication
development. Evaluations of family progress are measured
by behavioral coding of parent–child interaction video.
Similarly, It Takes Two to Talk is a family-centered par-
ent–child interaction program. It includes six to eight par-
ent training sessions to learn about communication styles,
most effective responses and specific speech, language, and
communication techniques.52,53
Recommendations
Hanen and variations of parent–infant transaction pro-
grams had an overall level of evidence of low to moderate
for our review population; effects included improvements
in communication skills and expressive language acquisi-
tion. Based on this, the GRADE guideline recommenda-
tion54 is ‘do it’ to ‘probably do it’, or a majority of people
would implement it with children at high risk for CP
under 2 years, but a minority would not. Overall, the
intervention does more good than harm.
Although this systematic review did not find high-level
evidence for interventions in infants at high risk for CP, a
358 Developmental Medicine & Child Neurology 2017, 59: 355–360
Cochrane review reported limited effectiveness in older
children with primary speech and language delays or disor-
ders.53 To improve phonological/speech skills and expres-
sive vocabulary, parent-directed or clinician-based
interventions were equally effective.55,56 Until children
have reached a stage of brain maturation allowing lan-
guage-specific speech perception, interventions aimed at
direct and specific language acquisition may have little
impact.29 Therefore, most early studies focus on the
improvement of prelinguistic skills through auditory
speech sound exposure,57,58 and parent responsivity and
interactions with infants.47 Early childhood interventions
should provide opportunities for interaction in the form of
shared, reciprocal engagement. This provides the frame-
work for the development of reciprocal turn-taking, recep-
tive and productive functions of language, and early
communication skills. Therefore, teaching the primary
caregivers about appropriate interaction and developmental
milestones, along with specific intervention techniques
when appropriate, may empower caregivers and create an
improved and more effective environment for infant speech
and language development. The amount of parent–child
interaction,59 responsiveness to child communication,60
amount and quality of linguistic input,61,62 and the use of
language learning support strategies46 are all aspects of
parent–infant interactions that may have a positive effect
on language and communication development.56
Limitations
Additional speech and language interventions may be avail-
able for infants at high risk for, or with, CP. Some study
designs appeared in their abstracts to have a high level of
rigor but on further review were downgraded. We
attempted to include grey literature but none was available
on the topic. Expert opinion concurred with the limited
evidence available. This common challenge in reviews of
rehabilitation therapies could drive the development of
new tools allowing conversion of individual experiences
into a standardized and universal repository of knowledge.
Another potential area for improvement in design would
be the choice of widely used, validated, and quantitative
outcomes measures to allow pooling of evidence from
smaller case studies. In this review, more than 15 separate
assessments were identified; a systematic review and con-
sensus for speech and language measures in infants may
improve future interventional study design. In addition,
evidence-based strategies, randomized clinical trials, and
protocol-driven practice may reduce the gap in available
research literature and knowledge of speech and language
interventions for infants and toddlers at high risk for CP.
CONCLUSION
Several interventions were identified as potentially benefi-
cial for infants at high risk for CP, but none were specifi-
cally targeted to infants at high risk or with a diagnosis of
CP. These programs used variants of Hanen and parent–
infant transaction programs. Heterogeneity of population,
intervention approaches, age at intervention and assess- necessarily represent the official views of the National Institutes
ment, as well as methods of assessments yielded weak clini- of Health. The authors have stated that they had no interests
cal implementation recommendations. which may be perceived as posing a conflict or bias.

A CK N O W L E D G E M E N T S SUPPORTING INFORMATION
We thank Ms. Linda Demur, MLS, the director of Library Ser- The following additional material may be found online:
vices, Nationwide Children’s Hospital, for her assistance with Figure S1: PRISMA flow chart of study selection.
completing the literature search. Funding source 1 K23 Table SI: Included studies.
HD074736 and 1R01HD081120–01A1 from the National Insti- Appendix S1: Search terms.
tute of Child Health and Human Development to NLM. The Data S1: Data Extraction Form.
content is solely the responsibility of the authors and does not

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