Clinical Linguistics & Phonetics

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Early speech and language intervention in

Brazilian-Portuguese toddlers with cleft lip and/or
palate

Nancy J. Scherer, Renata Yamashita, Debora Natalia de Oliveira, Jennifer

DiLallo, Inge Trindade, Ana Paula Fukushiro & Kacey Richards

To cite this article: Nancy J. Scherer, Renata Yamashita, Debora Natalia de Oliveira, Jennifer
DiLallo, Inge Trindade, Ana Paula Fukushiro & Kacey Richards (2021): Early speech and language
intervention in Brazilian-Portuguese toddlers with cleft lip and/or palate, Clinical Linguistics &
Phonetics, DOI: 10.1080/02699206.2021.1912187

To link to this article: https://doi.org/10.1080/02699206.2021.1912187

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CLINICAL LINGUISTICS & PHONETICS
https://doi.org/10.1080/02699206.2021.1912187

Early speech and language intervention in Brazilian-Portuguese

toddlers with cleft lip and/or palate
Nancy J. Scherera, Renata Yamashitab, Debora Natalia de Oliveirab, Jennifer DiLalloa,
Inge Trindadeb,c, Ana Paula Fukushirob,d, and Kacey Richardsa
a
Department of Speech and Hearing Science, Arizona State University, Tempe, Arizona, USA; bLaboratory of
Physiology, Hospital for Rehabilitation of Craniofacial Anomalies, University of São Paulo, Bauru, Brazil;
c
Department of Biological Sciences, Bauru School of Dentistry, University of São Paulo, Bauru, Brazil;
d
Department of Speech-Language Pathology and Audiology, Bauru School of Dentistry, University of São Paulo,
Bauru, Brazil

ABSTRACT ARTICLE HISTORY

Young children with cleft palate with or without cleft lip (CL/P) are at Received 26 September 2020
risk for early vocabulary and speech sound production delays. Early Revised 27 March 2021
intervention studies have shown some promising findings to pro­ Accepted 30 March 2021
mote early speech and vocabulary development following palate KEYWORDS
repair; however, we know little about how these interventions can Cleft palate; early
be used in other international contexts. This study adapted an early intervention; enhanced
speech and language intervention developed in the US, Enhanced milieu teaching; Brazilian
Milieu Teaching with Phonological Emphasis (EMT+PE), to the Portuguese
Brazilian context at the Hospital for Rehabilitation of Craniofacial
Anomalies at the University of São Paulo-Bauru. The purpose of this
study was to compare the speech and language performance of 24
toddlers with CL/P randomized into an EMT+PE intervention group
and a business-as–usual (BAU) comparison group over three time
points: prior to, immediately following, and three months after
intervention. Results immediately following intervention indicate
gains in multiple measures of language. Three months following
intervention, participants showed gains in both language and
speech measures.

Introduction
Cleft lip and/or palate (CL/P) is a frequently occurring condition that presents in one
of every 500-700 births (World Health Organization [WHO], 2016). These children
have early speech and vocabulary delays that can persist into school age (Hardin-Jones
& Chapman, 2011; Scherer et al., 1999). A recent meta-analysis indicated that children
with CL/P fall behind their peers in receptive and expressive language as well as speech
through eight years of age (Lancaster et al., 2020). Early intervention is often recom­
mended by cleft palate teams; however, there are only a few published studies of their
efficacy (Kaiser et al., 2017; Lancaster et al., 2020; Scherer et al., 2008). In addition,
there are few published studies of early intervention conducted in international con­
texts (Ha, 2015; Pamplona et al., 2004).

CONTACT Nancy J. Scherer nancy.scherer@asu.edu Department of Speech and Hearing Science, Arizona State
University, 975 S Myrtle Ave., Tempe, AZ 85287.
Supplemental data for this article can be accessed on the publisher’s website.
© 2021 Taylor & Francis Group, LLC
2 N. J. SCHERER ET AL.

Speech and language development in children with CL/P

Vocabulary and speech sound development emerges simultaneously in early development
and children with CL/P show delays in both vocabulary and speech as a result of their early
structural deficits (Chapman, 2004). These early delays improve over time, but speech and
language delays can continue into school age and can impact early reading acquisition
(Chapman & Willadsen, 2011). While the focus of research has been on speech develop­
ment, language differences have been described in early vocabulary development that
persist through the preschool period (Kaiser et al., 2017; Scherer et al., 2013; Scherer,
1999). In addition to vocabulary, differences in sentence complexity and talking rate
(word per minute) have been described (Frey et al., 2018; Kaiser et al., 2017). There is
evidence to suggest that young children with CL/P are at higher risk for both receptive and
expressive language deficits (Lancaster et al., 2020). While receptive language differences
may be subclinical, they persist in the literature to early school age.
The relationship between early speech and vocabulary delays in children with clefts has
focused on the restricted consonant inventories that limit vocabulary growth (Hardin-Jones
& Chapman, 2014). However, the relationship may be bidirectional in that limited voca­
bulary reduces the opportunity to practice production of new words and sounds (Scherer
et al., 2020). Further, approximately 30% of children with clefts will have velopharyngeal
dysfunction (VPD) and require secondary surgery (Peterson-Falzone et al., 2017; Zajac &
Valino, 2017). The speech characteristics associated with VPD, including hypernasality on
vowels, audible nasal emission on pressure consonants, and compensatory articulation
errors (i.e., glottal stops, pharyngeal fricatives, nasal substitutions) are relatively easy to
assess in older children. However, identifying early indications of VPD is not as straightfor­
ward for toddlers since some of these speech characteristics persist as learned behaviors
after palate repair (Hardin-Jones et al., 2006). Further, the child’s age, maturation level, and
presence of sufficient obstruents can impact definitive instrumental assessments.
These developmental differences observed in U.S. studies are also found in the Brazilian
literature. A study conducted by the Hospital de Reabilitação em Anomalias Craniofaciais
of the Universidade de São Paulo (HRAC-USP) in Brazil compared results of the Early
Language Milestone Scale (ELMS) in children between 12 and 36 months with and without
CL/P. Findings indicated that children with CL/P performed significantly lower in areas of
receptive and expressive language when compared to typically developing peers (Lamônica
et al., 2016). A recent study also developed at HRAC-USP analyzed the performance of
gross motor, adaptive fine motor, personal-social, and language skills in 30 children with
non-syndromic cleft lip and palate, aged from 36 to 47 months, compared to a typically-
developing control group matched by age and gender. The Denver Developmental
Screening Test II and MacArthur Communicative Development Inventory (CDI) Section
D were used to analyze developmental and language skills. The findings showed
a significant difference in all tested skills between groups, except for the personal-social
area, concluding that children with CL/P are at risk for developmental disorders
(Cavalheiro et al., 2019). In older children from seven to nine years of age, Marcelino
(2009) found that language abilities (oral and written) and auditory and visual abilities, such
as memory, association, grammar and visual closure, language reception, receptive voca­
bulary, phonological processing, writing, arithmetic, reading, auditory attention, and pro­
cessing performance were poorer than expected for the age in the majority of the children
 CLINICAL LINGUISTICS & PHONETICS 3

with CL/P. The Brazilian literature suggests that early speech and language delays persist
into school age and may be associated with more general developmental concerns that could
impact social and educational outcomes.
Early speech development has been characterized by limited consonant inventories,
lower Percent Consonants Correct (PCC), persistence of developmental phonological
errors and the presence of cleft-related speech errors (i.e., compensatory articulatory
substitutions, nasal substitutions, nasal emission, hypernasality, and weak pressure con­
sonants) (Chapman & Willadsen, 2011; Jones et al., 2003; Klinto et al., 2014). Children with
clefts show developmental phonological patterns that are related to a developing or imma­
ture speech sound system and not thought to be related directly to the cleft. These errors
include patterns of phonological processes characteristic of typical early development (e.g.,
fronting (t/k in cat), stopping (t/s in sun), final consonant deletion (ha for hat), simplifica­
tion (nana for banana), assimilation (g/d in dog) (Willadsen et al., 2017). In addition to
these developmental errors, toddlers may have errors that are commonly associated with
velopharyngeal dysfunction (VPD). These errors may be a learned response to early
anatomical deficits from the cleft or VPD (Sell et al., 1999). Glottal stop and pharyngeal
fricative substitutions for stops and fricatives are one of the most common examples of the
child’s attempt to compensate for inability to implode oral air pressure prior to palate
repair. The errors may remain following palate repair as a learned substitution pattern or
may be symptom of VPD. For toddlers, it may be difficult to discern the origin of
compensatory patterns until sufficient language is present to assess the presence of VPD.
Nasal substitutions (e.g., m/b or n/d), is another error pattern that is considered a symptom
of VPD; however, it is also an error pattern observed frequently in noncleft toddlers and
may or may not be related to the presence of VPD in toddlers with CLP (Hardin-Jones &
Chapman, 2018). The presence of other signs of VPD that are related to structural deficits,
are audible nasal emission (on pressure consonants), hypernasality (on vowels or perva­
sively in connected speech) and weak pressure consonants. Toddlers present a challenge to
determine the origin of their speech errors and early intervention can provide an opportu­
nity to facilitate vocabularies that contain early developing consonants permitting more
detailed analysis of emerging patterns of sound production.

Interventions for children with CL/P

There are few intervention studies of children with CL/P and even fewer that focused on
early intervention. Bessell et al. (2013) conducted a systematic review of speech interven­
tions for individuals with cleft palate who received either a motor or linguistic approach to
intervention. Motor approaches included non-speech oral-motor approaches or articula­
tory approaches that moved from sound to syllable-, word-, and phrase-level productions.
Linguistic approaches included language approaches, such as focused stimulation, whole
word, and phonological approaches. The results of the systematic review found some
positive effects for both motor and linguistic approaches, but the limitations in methodol­
ogy for many of the studies constrained the interpretation of findings.
Enhanced Milieu Teaching with Phonological Emphasis (EMT+PE) intervention has
demonstrated positive speech and language outcomes for toddlers with clefts in the U.S.
(Kaiser et al., 2017; Scherer et al., 2008; Scherer, Kaiser et al., 2020). This approach combines
vocabulary and speech sound targets in a single intervention approach which was
4 N. J. SCHERER ET AL.

particularly well-suited to the early speech and language delays of children with CL/P. EMT
+PE utilizes naturalistic, conversationally-based strategies in the context of play and
routines to target key speech sounds, based on individualized goals, through developmen­
tally appropriate words and phrases. In a pilot study, Kaiser et al. (2017) and Scherer, Kaiser,
and Frey (2020) administered EMT+PE to English-speaking children with CL/P across 48
sessions. Compared to a business-as-usual (BAU) group, children receiving EMT+PE made
greater gains than BAU children on speech and vocabulary measures following the inter­
vention. There was a differential effect of intervention, with the children who used at least
seven words per minute (i.e., relatively higher vocabulary) making the greatest speech gains.
As a note, BAU comparison groups are children who are receiving routine services in the
community for that particular condition. So, children in the BAU comparison could
potentially be getting other interventions (e.g., speech, language, physical/occupational
therapies): however, children in this study were not receiving additional interventions but
were receiving routine follow up in their cleft palate team.

Research questions
The purpose of the present study was to compare the speech and language performance of
toddlers with CL/P randomized into an EMT+PE intervention or a business-as-usual
(BAU) comparison group over three time points: prior to, immediately following, and
three months after intervention. The following questions were investigated:

(1) Do children with CL/P that receive EMT+PE intervention demonstrate significant
post-intervention and follow-up gains in the speech measures in comparison to
a business-as-usual group?
(a) Do children with CL/P that receive EMT+PE intervention demonstrate
a significant reduction in compensatory articulation (glottal stop, pharyngeal
fricatives) and nasal substitutions in comparison to a business-as-usual group?
(2) Do children with CL/P that receive EMT+PE intervention demonstrate significant
post-intervention and follow-up gains in language measures in comparison to
a business-as-usual group?

Methods
This study was funded by NIDCD (Grant No: R03 DC013527) and approved by the Internal
Review Boards of Arizona State University and the Hospital de Reabilitação de Anomalias
Craniofaciais – Universidade de São Paulo (HRAC–USP).

Participants
A total of 29 children with CL/P participated in this study. The average age of palate repair
was 14.1 months (11–19 months). The average age at baseline for participants was
24.5 months (20-34 months). Children were randomized into groups in such a way that
the groups were optimally balanced with respect to pre-intervention age, expressive lan­
guage standard score and number of consonants in the inventory in order to control the
developmental performance within this age range. Participants varied by cleft type:
 CLINICAL LINGUISTICS & PHONETICS 5

unilateral cleft lip/palate (UCLP; n = 14), bilateral cleft lip/palate (BCLP; n = 3), and isolated
cleft palate (ICP; n = 7). Individual participant characteristics are summarized in Table 1.
Children were recruited from two sites at the time of palate repair: HRAC-USP and
a satellite clinic in São Paulo, the Fundação para o Estudo e Tratamento das Deformidades
Crânio-Faciais (FUNCRAF). Primary caregivers were provided information about the study
and Brazilian speech-language pathologists (SLPs) contacted the families at regular intervals
until 18 months to determine readiness for intervention.
Participants were selected based on the following inclusionary/exclusionary criteria: (a)
presence of a nonsyndromic cleft lip and/or palate, (b) aged 18 to 36 months at baseline, (c)
demonstrated joint attention appropriate for verbal engagement and imitation skills by
passing related items on the Avaliação do Desenvolvimento da Linguagem or Evaluation of
Language Development (ADL; Menezes, 2004) (d) produced 10 distinguishable word
approximations using Vihman criteria (Vihman, 1994), and (e) had their primary palate
repair at 18 months of age or earlier. Children were excluded from the study if they: (a) had
a sensorineural hearing loss or hearing thresholds ≥ 30-dB HL, as measured by an audiol­
ogist, (b) were multilingual or non-Portuguese speaking based on parent report, and (c) had
more than three additional dysmorphic features in addition to the cleft or a syndrome
diagnosis from a geneticist. Jones (1988) indicated that children with clefts who have three
or more other anomalies should be considered as syndromic even though they may not have
an identified syndrome.
Letters were provided to the primary caregivers describing the study. Consent was
obtained by at least one of the child’s parents or guardians.
After meeting inclusionary criteria and providing consent, participants were randomly
assigned to either the experimental intervention (n = 12) or a business-as-usual comparison
group (n = 12) using a covariate adaptive randomization procedure (Suresh, 2011; Weir &
Lees, 2003). The goal of this method is to assign subjects to treatment or control as they
enroll in the study in such a way that the groups are optimally balanced with respect to one
or more desired characteristics. For our study chronological age (within two months) and
expressive vocabulary were used as characteristics for the randomization. The Brazilian SLP
who completed the assessment collected and scored the MacArthur CDI adapted for
Brazilian Portuguese (Teixeira, 2000, 2019) to obtain a measure of expressive vocabulary
within the clinical setting. A second Brazilian SLP rescored the CDI for accuracy.
Comparison of pre–treatment vocabulary scores on the CDI showed that the groups did
have a substantial range of scores (34-425); however, the groups were not significantly
different (t = 0.59, p = .28). Additionally, three participants dropped from the intervention

Table 1. Participant characteristics by group.

Intervention group Control group
Demographics Frequency Percent Frequency Percent
Total number 12 12
Gender
Male 3 25% 6 50%
Female 9 75% 6 50%
Cleft palate type
Cleft palate only 5 42.7% 2 16.7%
Unilateral CL/P 6 50% 8 66.6%
Bilateral CL/P 1 8.3% 2 16.7%
CL/P = cleft lip and/or palate.
6 N. J. SCHERER ET AL.

group and two dropped from the BAU group prior to T1 due to the time commitment
required in the project. Figure 1 shows the screening and enrollment process for the
children in the study.

Assessor/intervention training
All children were assessed for both speech and language development at three time points:
baseline (T0), immediately following completion of the 12-week intervention (T1) and at
a three-month follow-up after T1 (T2). Assessor training of Brazilian SLPs was accom­
plished in three phases. Phase 1 consisted of a foundational training of the lead Brazilian
SLP by the primary investigator, including yearly in-person training (either in Brazil or the
U.S.) on research skills (research design, data collection, progress monitoring and fidelity,
assessment, and the EMT+PE intervention model) and five distance-learning training
modules with video examples in English, training on research skills, professional reflection,
and monthly video conferences to discuss and resolve training questions. A description of
the elements of the training is provided in the Table 2. Phase 2 consisted of a mentorship
approach to training, in which the lead SLP then trained local SLPs through a ‘coaching
cycle’ (i.e., observation, modelling, and communication) with monthly video conferences,
including the primary investigator, to discuss and resolve training questions. Phase 3
consisted of a sustainable approach to leadership development, in which the local team of
Brazilian SLPs developed their own web-based repository in Brazilian Portuguese of the

Figure 1. Participant screening and enrollment process.

 CLINICAL LINGUISTICS & PHONETICS 7

Table 2. Description of assessment and intervention training.

Training Phase Training Activities
Pre-Training 1. Virtual meeting with the US and Brazilian research teams.
2. Overview of training schedule and activities.
Phase 1: Foundational 1. Presentation of study design, methods for assessment and intervention and analysis.
Training 2. Presentation on the covariate adaptive randomization procedure by statistician
3. Presentation and discussion of data management system
4. Reviewed fidelity and reliability methods and discussed modification. Assessed understanding
of methodology on Canvas.
5. Discussed procedures for translating all training and intervention forms for the study.
6. Phonetic transcription practice and consensus training. Training presented and assessed on
Canvas.
7. Screening and standardized assessment review.
8. Presentation on language sample collection and transcription procedures. Training presented
and assessed on Canvas.
Phase 2:Intervention 1. Research team completes 10 EMT+PE modules on the strategies. Competency-based
Training knowledge assessment on Canvas to criterion of 90%.
a. Environmental arrangement
b. Responding to praise
c. Responding to teach
d. Modeling
e. Expansions
f. Speech recasting
g. Requests
h. Mirror and mapping
i. Time delay
j. Putting it all together
2. Choosing speech and language targets
3. Purchasing and organizing intervention materials
4. Parent training procedures and materials
5. Discussion and assessment of key readings on Canvas.
6. Practice EMT+PE strategies with feedback from US team.
7. Two practice videos of each strategy were submitted by
the Brazilian clinicians to the US team for fidelity measurement to criterion of 90%. Retraining
occurred if fidelity fell below criterion.

assessment techniques and intervention strategies. Criteria for completion of each training
phase included both knowledge and skills competency, as demonstrated by a score of 90%
on a competency-based assessment on the Canvas platform (knowledge) and 90% com­
pliance on a 10-component EMT+PE fidelity checklist (skills). Three licensed Brazilian
SLPs completed this training and one SLP administered speech and language assessments
and two other SLPs delivered the intervention for the purposes of this study.
Administration of each assessment was video and audio recorded.

Speech and language assessment

Speech assessment and measures
A Brazilian Portuguese adaptation of the Profiles of Early Expressive Phonological Skills
(PEEPS:BP; Scherer, Kaiser, & Frey, 2020; Scherer, Kaiser et al., 2020; Stoel–Gammon &
Williams, 2013) was administered to assess early speech development of populations with
CL/P through the production of early acquired words (see Appendix). PEEPS:BP was
constructed to represent the diversity of place, manner, voicing, and syllable and word
shapes of Portuguese consonant production for children under three years of age. The
assessment consists of 36 words, which were selected utilizing developmentally expected
vocabulary from the Brazilian Portuguese CDI and considering their consistency with
8 N. J. SCHERER ET AL.

phonological properties expected of children between 13-36 months that are monolingual
speakers of Brazilian Portuguese. To administer PEEPS:BP, assessor(s) introduced toys
representing each word in a structured, play-based manner to elicit labels from a child. If
the child did not independently label the toy after the toy was introduced and/or as
a response to the question (‘What is this?’), the assessor provided a model of the target
word embedded within a phrase (e.g., ‘Look, I have a ___.’). If the child did not produce
a label with those cues, the assessor provided a direct command (e.g., ‘Say ___.’). If the
child did not produce a label following the command, the item was scored as ‘No
Response.’ Each administration was video- and audio-recorded for transcription and
reliability.
All PEEPS:BP administrations were independently transcribed by Brazilian SLPs with
experience transcribing the speech of children with CL/P. The ASU project manager
provided ongoing feedback and training on these transcriptions. Inter- and intra-judge
transcription reliability was completed and disagreements were resolved through consen­
sus. Phonetic transcription reliability was performed on 100% of the single word samples.
The reliability was performed by the lead Brazilian clinician and a Portuguese speaking
clinician on the U.S. team who were not interventionists for the study. Twenty percent of
the transcripts contained disagreements that required consensus discussion and a third
research clinician was used when consensus was not achieved. Most often these disagree­
ments occurred when the child turned away from the camera and microphone and/or used
atypical compensatory articulation errors. Final PEEPS:BP phonetic transcriptions were
analyzed for number of words attempted; number of consonants attempted (overall and by
manner); total consonant inventory by word position; PCC by manner: stops, fricatives,
affricates, nasals, liquids, and total PCC; as well as percentage of nasal emissions, nasal
substitutions, and compensatory errors. Additionally, hypernasality was rated by three
Brazilian SLPs from spontaneous language samples. Inter-rater reliability was 95% agree­
ment. The samples were rated at T1 and T2 (only because of limited speech production at
T0) on a 0 to 3 equal-appearing interval scale (0 = absent, 1 = minimal hypernasality,
2 = moderate, and 3 = severe).
The Intelligibility in Context Scale for Brazilian Portuguese (ICS; McLeod, 2020; McLeod
et al., 2012) was administered to ascertain a measure of child’s speech intelligibility per
caregiver report. The intelligibility assessment elicits an overall judgment of intelligibility
based on the average response to seven questions on a five–point rating scale. This measure
has been used for 4-5 year olds; however, there were no similar measures for younger
children that could provide a social validity measure of intelligibility. There is evidence to
support that this tool is reliable and valid when administered in other languages and
countries (McLeod, 2020). For this study, the mothers of the children completed the ICS
at all time points.

Language assessment and measures

Multiple language measures were administered to assess children’s receptive and expressive
language development throughout the intervention, including standardized and non-
standardized language assessments in Brazilian Portuguese.
The Avaliação do Desenvolvimento da Linguagem or Evaluation of Language
Development (ADL; Menezes, 2004), ADL is a norm-referenced, clinical assessment
designed to evaluate preschool language development through play. Six scores were
 CLINICAL LINGUISTICS & PHONETICS 9

obtained from ADL administration: raw and standard receptive language scores, raw and
standard expressive language scores, and raw and standard total language scores.
In addition, language samples were collected at T0, T1, and T2 for each child. Language
samples consisted of a play-based interaction between the child and clinician with
a standard set of toys. Clinicians were blinded to study group. After approximately a 15-
minute recording was established, two native-BP-speaking SLPs transcribed the interaction
orthographically. The ASU project manager provided ongoing feedback and training on
these transcriptions. A final consensus transcription was obtained with the Brazilian SLPs
and the research assistants at ASU. Few language-specific adaptations to language sampling
transcription were made for Brazilian Portuguese (see Supplementary Materials). Language
samples were analyzed for four measures: number of different words (NDW), total words
produced, total utterances, and mean length of utterance in words (MLU-w).
The CDI adapted for Brazilian Portuguese (Teixeira, 2000, 2019) was administered to
assess children’s vocabulary size. The assessment consists of a vocabulary checklist, in which
caregivers indicate whether a child understands or understands and says a given vocabulary
item. The Brazilian Portuguese adaptation of this assessment was developed considering
early-developing words in Brazilian Portuguese. The CDI provides a measure of Total
Vocabulary Size. Further, this study considered the average length (in words) of the three
longest utterances.
A complete dataset for the speech and language measures from the current study is
available at DOI10.13140/RG.2.2.33345.38241.

EMT+PE intervention
Children in the intervention group received 30–45-minute sessions, twice weekly, for a total
of 12 weeks. The intervention sessions consisted of clinician directed intervention with
parent training on the components of the EMT+PE program presented during the sessions
to be practiced at home.
All sessions were administered by two Brazilian speech-language pathologists (SLPs),
one at each of the two sites. Both SLPs had been trained to criterion to ensure consistency of
intervention delivery before therapy administration. Training for intervention purposes
consisted of a three-phase process (see ‘Assessor/intervention training’).
Core strategies of EMT+PE were used to engage the child and facilitate target word use.
Per Kaiser et al. (2017), all EMT+PE strategies were implemented naturalistically in routines
and play across each session. There were nine component strategies of EMT+PE: (1)
environmental arrangement, in which SLPs provide environmental boundaries, provide
age-appropriate toys, and use materials that elicitation conversation and play; (2) mirroring
and mapping, in which SLPs intentionally respond to children’s play-based interests and
communication attempts; (3) responding and praising, in which SLPs praise all correct
requests, responses, and behaviors; (4) emphasis in models, in which SLP models include
and emphasize children’s vocabulary speech targets; (5) emphasis in speech recasting, in
which SLPs emphasize target speech sounds when recasting children’s speech errors; (6)
requests, in which SLPs use choice questions to elicit child speech targets; (7) time delays, in
which the SLPs pause when the child requests an item non-verbally or with minimal
verbalization (to encourage a verbal attempt); 8) expansions, in which SLP expansions
preserve as much of children’s original utterances as possible; 9) ‘proximal’ or meaningful
10 N. J. SCHERER ET AL.

verbal feedback, in which SLPs do not produce utterances that exceed the child’s average
utterance length by more than two to three words nor do SLPs ask more than five Wh-
questions in a given session.

EMT+PE implementation
Target word selection. For participants in the EMT+PE group, target words were selected
based upon an assessment of each individual child’s baseline performance in the CDI and
PEEPS:BP. The target words comprised high pressure consonants, primarily stops, and
nasals in consonant-vowel-consonant-vowel (CVCV) words. Target words were selected
from the CDI vocabulary list containing stops and nasals in word positions identified in the
speech assessment.

Procedural fidelity. Procedural fidelity of the intervention was assessed at three time points
during intervention: sessions 6, 12, and 18. Fidelity checklists were completed by the lead
Brazilian research clinician (who was not an interventionist) and verified by a Portuguese–
speaking clinician on the U.S. team. The fidelity rating maintained a criterion of 90% to
assure that the fidelity was maintained throughout the intervention. The procedural fidelity
92-100% for each of the fidelity sessions. The fidelity form is included in Appendix A.

Business-as-usual comparison group

Children assigned to the business-as-usual (BAU) group were assessed at the same three
time points as the children receiving EMT+PE intervention. None of the children in the
BAU group received speech and language services, but all received follow-up services from
the cleft lip/palate team.

Analysis
For each outcome, an analysis of covariance (ANCOVA) model compared outcomes for
intervention vs. BAU group at the target time point controlling for those outcomes
measured at the previous time point. The T0-T1 analysis estimates the immediate post­
treatment effect, while the T0-T2 analysis estimates the follow up treatment effects. Analysis
of covariance is an efficient statistical technique that produces unbiased estimates of
treatment effects when the treatment is randomly assigned, as it was in this case, and is
more powerful than alternative strategies (Van Breukelen, 2006).

Results
Post-intervention effects
The results in Table 3 shows the comparison of pre- and immediately post-treatment effects
between the EMT+PE and BAU conditions. The primary significant effects were in the
language variables including parent ratings of vocabulary and average of three longest
sentences, language sample variables (NDW, total utterances, and MLU-w) and receptive
language performance (ADL) at p < .05 with moderate and large effect sizes. Percent
Consonants Correct (PCC)–Stops and the standardized language test (ADL) total raw
score approached significance and had large effect sizes. Additionally, several variables
 CLINICAL LINGUISTICS & PHONETICS 11

Table 3. Post-intervention comparison results for ANCOVA models of change for speech and language
variables.
Measure and Outcome Est (Std Err) p d F(2, 21)
PEEPS:BP
Percent Consonants Correct (Total) 0.06 (0.06) .352 0.40 17.67
Percent Consonants Correct (Nasal) −0.19 (0.12) .132 −0.44 2.31
Percent Consonants Correct (Stop) 0.15 (0.07) .060 0.72 20.76
Percent Consonants Correct (Fricative) −0.01 (0.08) .879 −0.07 10.14
Percent Consonants Correct (Affricate) 0.03 (0.17) .867 0.06 0.69
Percent Compensatory Errors 0.00 (0.03) .931 0.03 4.01
Number of Consonants (Initial) 0.81 (1.14) .485 0.27 12.09
Number of Consonants (Medial or Final) 0.86 (1.40) .545 0.23 10.26
CDI
Vocabulary 92.19 (33.38) .012 1.18 29.29*
Average of Three Longest Utterances 0.67 (0.28) .026 0.55 6.48*
ICS Total 2.39 (1.43) .110 0.66 12.21
ADL
Raw Score for Receptive Language 3.82(1.39) .012 1.14 17.56*
Raw Score for Expressive language 3.07 (1.96) .132 0.66 11.33
Raw Score (Total) 27.68 (13.76) .057 0.71 7.00
Language Sample
Number of Different Words 24.77 (7.90) .005 1.26 17.19*
Total Words Produced 47.56 (23.34) .054 0.85 17.00
Total Utterances 31.29 (12.02) .017 1.03 13.28*
Mean Length of Utterance in Words 0.50 (0.15) .002 1.37 22.87*
PEEPS:BP = Profiles of Early Expressive Phonological Skills: Brazilian Portuguese, CDI = Communicative Development
Inventory, ICS = Intelligibility in Context Scale, ADL = Evaluation of Language Development.
*p < .05.

that did not show significant results had large effect sizes (d = 0.65-0.70). These included the
ICS and ADL expressive language scores and total words produced from the language
sample.

Follow-up effects
The results for the comparison of pre-treatment and 3-month follow-up showed
a significant difference in gains for both language and speech skills between the groups
and are displayed in Table 4. Two language variables that showed significant gains in the
post-intervention effects also continued to show these effects in the three-month follow up.
These included the language sample variables of NDW and total utterances. Three speech
variables achieved significance at the three-month follow up. These included total PCC,
PCC–stops, and the ICS score. Several speech and language variables showed nonsignificant
results but moderate effect sizes (d = 0.48-0.69). These variables included PCC-fricatives,
nasals, number of initial word position consonants, CDI vocabulary, ADL expressive
language, and total words produced and MLU-w on the language sample. Table 4 shows
the statistical analyses and effect size estimates for the three-month follow up.

Cleft-related speech errors

Speech errors of place of articulation that occur more prominently in young children with
clefts are characterised by compensatory articulatory substitutions (e.g., glottal stops,
pharyngeal fricatives), nasal substitutions (Chapman & Willadsen, 2011; Harding &
Grunwell, 1998). These speech errors are present very early in development and can
12 N. J. SCHERER ET AL.

Table 4. Follow-up results for ANCOVA models of change for speech and language variables.
Measure and Outcome Est (Std Err) p d F(2, 21)
PEEPS:BP
Percent Consonants Correct (Total) 0.14 (0.05) .011 1.17 35.07*
Percent Consonants Correct (Nasal) 0.18 (0.11) .119 0.53 5.53
Percent Consonants Correct (Stop) 0.22 (0.07) .006 1.16 27.21
Percent Consonants Correct (Fricative) 0.12 (0.07) .116 0.70 17.65
Percent Consonants Correct (Affricate) −0.06 (0.14) .685 −0.14 2.06
Percent Compensatory Errors −0.02 (0.02) .472 −0.15 4.54
Number of Consonants (Initial) 1.44 (0.96) .150 0.54 15.62
Number of Consonants (Medial or Final) 1.46 (1.30) .274 0.36 6.21
CDI
Vocabulary 92.22 (58.17) .129 0.70 5.94
Average of Three Longest Utterances 0.43 (0.29) .154 0.32 2.23
ICS Total 3.89 (1.70) .032 0.93 10.82*
ADL
Raw Score for Receptive Language 2.32 (2.28) .323 0.42 10.12
Raw Score for Expressive language 2.07 (1.85) .277 0.48 15.71
Raw Score (Total) 14.24 (13.97) .320 0.38 6.64
Language Sample
Number of Different Words 42.756 (13.85) .006 1.23 7.31*
Total Words Produced 40.47 (27.12) .151 0.62 16.26
Total Utterances 43.35 (14.84) .008 1.11 8.68*
Mean Length of Utterance in Words 0.26 (0.20) .210 0.55 5.46
Positive estimates and Cohen’s d effect sizes indicate that the intervention group mean was higher than the BAU group mean
at 3-month follow-up controlling for the pretest scores measured at pre-intervention. p-values unadjusted for multiple
comparisons. PEEPS:BP = Profiles of Early Expressive Phonological Skills: Brazilian Portuguese, CDI = Communicative
Development Inventory, ICS = Intelligibility in Context Scale, ADL = Evaluation of Language Development.
*p < .05.

undergo changes in their use along with the child’s changing phonological system and with
subsequent surgical and speech interventions (Klinto et al., 2014). Compensatory articulation
and nasal substitutions were present for most of the children in both groups, although at least
half of the children used few (i.e., three or fewer) errors. The children who used compensatory
articulation and nasal substitution showed changes in their error use during the study,
although the direction of change did not show a consistent trend in either group. Six children
(four from the EMT+PE group) showed a decline in compensatory and nasal substitutions
while the remaining seven (two from the EMT+PE group) maintained or increased their use
of these active errors. To show examples from individual children, Table 5 shows the speech
profiles across the three time points for four children selected from the intervention and BAU
groups. These profile comparisons serve to demonstrate the complex relationship between
early speech and language development and the presence of compensatory articulation and
nasal substitutions. This information has implications for assessment and treatment decisions
that will be discussed later.
Table 5 shows a profile of language and speech performance for four children (two from the
EMT+PE group and two from the BAU group) to demonstrate the differential patterns of
performance over the three timepoints in the study and their relationship to speech and
language growth. All four children began the study with relatively limited speech and language
performance (T0). Child A11 achieved substantial gains in both language and speech out­
comes during the study. However, this child showed increasing use of compensatory and nasal
substitutions during the study and child demonstrated moderate hypernasality as rated by the
Brazilian SLPs at T2, in addition to their nasal substitutions and compensatory errors,
 CLINICAL LINGUISTICS & PHONETICS 13

Table 5. Speech and language profiles for four children showing differences in language and speech
measures during the study.
A11 (EMT+PE) A12(EMT+PE) B05 (BAU) B10 (BAU)
ID T0 T1 T2 T0 T1 T2 T0 T1 T2 T0 T1 T2
Age (Months) 24 27 30 29 32 35 23 26 29 33 37 40
Language Sample*
NDW 12 45 194 29 75 97 17 68 81 82 90 92
TW 31 128 230 55 165 192 36 119 196 191 350 319
MLU 1.17 2.33 2.61 1.25 1.82 2.2 1.24 1.29 1.77 2.08 2.89 2.31
Speech**
Consonant Inventory 8 11 12 6 11 16 7 15 18 14 16 17
PCC Total 6.5 31 41 17 37 41 45 61 62 21 49 36
PCC Stops 33 100 100 0 62 67 66 79 89 10 34 24
Comp. Errors 19 33 32 29 3 2 0 0 0 14 19 18
Nasal Subs 10 16 21 3 0 2 1 3 3 6 0 1
*NDW = Number of different words, TW = Total words, MLU = Mean length of utterance, Number of Nasal Subs = Nasal
substitutions, *30-Minute Language Sample, **Speech measures were collected from single word production in the PEEP:BR.

indicating suspected presence of VPD. Child A12 had a similar profile of speech and language
performance on pre-treatment measures (as to Child A11) with relatively high compensatory
articulation use at pre-treatment. In contrast to Child A11, Child A12 decreased their use of
compensatory and nasal substitutions during the study and was not considered at risk for
VPD. Child B05 also had a similar speech and language profile to A11 at pre-treatment, but
did not use many compensatory and nasal substitutions, despite having a moderate hypernas­
ality rating; this child was also considered at risk for VPD. Child B10 (who was one of the older
children in the study) showed a significant speech delay per speech measures and presence of
compensatory and nasal substitutions. This child reduced the use of nasal substitutions, but
maintained production of compensatory errors throughout the study. This child had moder­
ate hypernasality indicating the presence of VPD. Seemingly all four children with comparable
pre-treatment speech and language profiles developed observationally different profiles in
compensatory and nasal substitution usage over the course of the study. Monitoring of the
speech errors over time did assist in team decision making for referrals for VPD.

Answering the research questions

1. Do children with CL/P that receive EMT+PE intervention demonstrate significant post
intervention and follow up gains in the speech measures in comparison to a business-as-
usual group?

The data indicates that the intervention group made significant speech gains in the
3 month follow up but less so immediately post-intervention. Post-intervention results
showed no significant group differences for speech measures, although a large effect size for
PCC-Stops (d = 0.72) was noted. However, there were significant findings for speech in the
follow-up. Total PCC, PCC-stops (the target sound class for treatment), and intelligibility
(ICS) measures all achieved statistical significance and support the positive gains over the
comparison group. Additionally, PCC-fricatives and -nasals showed large effect size differ­
ences even though the gains did not reach significance indicating that the speech effects had
14 N. J. SCHERER ET AL.

a broader impact beyond the target speech sounds. The improvement in our intelligibility
measure (ICS) occurred primarily in how well the child was understood by strangers
relative to the comparison group. This particular item was found to be predictive of
which children with CL/P had better speech intelligibility at age 8 years (Wren et al.,
2016). Additionally, overall scores on the ICS at follow up were similar for our BAU
group (M = 3.63) to those reported for a study of children clefts in the UK at 3 years of
age (M = 3.75). While our treatment group had a mean score above those comparison
groups (M = 4.45) (Seifert et al., 2021). To our knowledge this study is the first to validate
the ICS in Brazilian Portuguese as an outcome measure for intervention research.

1a. Do children with CL/P that receive EMT+PE intervention demonstrate a significant
reduction in compensatory and nasal substitutions in comparison to a business-as-usual
group?

No significant differences were observed between groups on compensatory or nasal

substitutions across time points. However, the small number of children in each group
with these errors in this pilot study likely contributed to these findings. Descriptive analysis
revealed that about half of the children in each group had no or minimal use of compensa­
tory or nasal substitutions (six Intervention, five BAU). As shown in Table 5, the relation­
ship between compensatory and nasal substitution use and VPD was not straightforward.
The monitoring of these errors over time when the child was gaining sufficient expressive
language production did inform decision-making for subsequent surgery and further
speech intervention.

2. Do children with CL/P that receive EMT+PE intervention demonstrate significant post
intervention and 3 month follow up gains in language measures in comparison to a busi­
ness-as-usual group?

The language variables showed both significant differences between groups for immedi­
ate post-intervention and follow-up gains. The EMT+PE group showed a significant dif­
ference in receptive language and expressive vocabulary and sentence combinations in the
post-intervention. EMT+PE strategies promote child engagement, model and expand
vocabulary, and increase child word and sentence attempts; such supports give the child
sufficient opportunities to practice speech production. The post-intervention language
effects pre-empt later speech-sound changes observed in the study.

Discussion
Post-intervention effects
The post-intervention effects were in the language domain including receptive language
(ADL), vocabulary (CDI, language sample different words), and number and complexity of
utterances (CDI longest sentences, total utterances in language sample). These findings
align with prior literature. A recent EMT+PE intervention study involving children with
CL/P in the USA showed a similar developmental trend in language and speech outcomes
(Philp, 2020; Scherer, Kaiser et al., 2020). The younger children in the study showed
 CLINICAL LINGUISTICS & PHONETICS 15

improvements in vocabulary use while the older children who used more than seven words
per minute showed significant speech gains. The authors concluded that a language foun­
dation is necessary to employ the speech facilitation strategies at a sufficient rate to impact
speech outcomes. The current study clearly showed the language gains during the inter­
vention when parents were trained in the matched turns, modelling, and expansions which
facilitated language development. This current study supports the language findings of
other studies with English- and Spanish-speaking non-cleft children with language delays
(Peredo et al., 2018; Roberts & Kaiser, 2015).

Three-month follow-up
The three-month follow-up showed both significant gains in language and speech measures
for the intervention group. The language gains (NDW and total utterances per the language
sample) indicate that the children receiving intervention talked more and with greater
vocabulary diversity. Speech accuracy (PCC total and PCC stops) also improved for the
intervention group. These gains are likely attributable to the intervention as stop conso­
nants were the focus of the speech intervention and embedded into the activities during the
intervention.
Why did the speech effects not appear immediately post-intervention? During the
intervention, the mothers were modeled speech facilitation strategies during the interven­
tion and as their child produced more language, they were able to use the speech recast
strategies during their interactions with their child. We hypothesize that, in part, the
children had to achieve sufficient expressive language use for the parents to use the speech
strategies. The more the children talk, the more opportunities they have to practice speech
production and receive feedback from adults in their environment (Scherer, Kaiser, & Frey,
2020).

Developmental and cleft-related speech errors

All the children in both groups used developmental speech errors that are observed in
children in the early stages of phonological development. While both groups showed
changes in their speech acquisition over time, the intervention group made significantly
more progress in acquisition of speech accuracy than the comparison group. The interven­
tion group made more speech gains than the comparison group and many of the gains
added consonants and reduced developmental substitutions to improve overall speech
accuracy. Compensatory and nasal substitutions were only used by approximately half of
the children in both groups. The presence of these errors for children with CL/P is thought
to indicate VPD, and for some children in the study their presence was associated with
a subsequent referral to the team for VPD assessment. This information did assist the team
in making databased recommendations regarding subsequent follow up for VPD. However,
the relationship between the presence of compensatory and nasal substitutions and VPD in
toddlers with clefts is not straightforward. In a recent study, Hardin-Jones and Chapman
(2018) found that 76% of toddlers with CL/P used nasal substitutions in their early words,
while only 38% of those children had associated VPD at 39 months of age. Ongoing
monitoring of compensatory and nasal substitutions prior to age 3 years, particularly in
16 N. J. SCHERER ET AL.

the presence of stop consonant use in words, will assist in recommendation for VPD
follow up.

Limitations of the study

This pilot study provided the first randomized study of early intervention effects for toddlers
with cleft palate in Brazil. However, it needs to be replicated with a larger clinical trial to
validate the results. Replication studies with a similar experimental design should be con­
ducted to evaluate the treatment efficacy of EMT+PE with a larger cohort of children. Further,
this study is limited by the lack of validation of assessments available in Brazilian Portuguese.
These non-standardized assessments, while generally clinically acceptable, limit analyses of
treatment effects among the current sample and limit possible future comparisons between
children with CL/P and typically developing children. Further standardization of relevant
speech and language assessments in Brazilian Portuguese are necessary, as well as the collec­
tion of normative data for same-aged, typically developing children on similar measures
(Albuquerque & deCunha, 2020).
Related to typical Brazilian Portuguese speech patterns, there is a need within existing
literature to explore the confluence of standard Brazilian Portuguese’s phonemic inventory
on the reliability of transcriptions pertaining to compensatory articulation. For instance,
Brazilian Portuguese features five nasalized vowels, and though this study does not directly
analyze vowel production, there is the possibility that nasalized vowels and/or errors in
nasalization may influence transcription. Similarly, Brazilian Portuguese features the uvular
fricative/χ/as part of its typical phonemic inventory, which is also conventionally consid­
ered a compensatory error for populations with CL/P. Regardless, one approach to maintain
the reliability of cross-linguistic transcriptions of CL/P speech is to rely on the transcrip­
tions of trained native speakers of the language. For instance, Yamashita et al. (2018) found
that native speakers of Swedish rated the hypernasality of Swedish-speaking children with
CL/P significantly differently from the ratings of non-native speakers. Further research
would be necessary to extend these findings to the reliability of the transcription of
compensatory errors. Regardless, recruiting trained native speakers as transcribers is
recommended for cross-linguistic CL/P speech assessments (Cordero, 2008).

Clinical implications
This study extends the evidence of language and speech benefits from the EMT+PE inter­
vention to other language contexts. EMT has been successfully applied to noncleft bilingual
Spanish-speaking families and now to Brazilian Portuguese children with cleft palate. The
simultaneous gains in both vocabulary and speech production provide evidence for clinical
application with this population. This study has led to the development of resources for
training Brazilian SLPs and parents on EMT+PE, including a website that provides instruc­
tions for getting started with the therapy and implementing the strategies as well as video
examples of the strategies (Projeto Interkids, n.d.). Proper training of SLPs is essential for this
treatment method to work. The SLP must be able to implement the strategies reliably prior to
training parents on the strategies. The resources generated in this study will be useful to
Brazilian SLPs when training parents on EMT+PE as well as SLPs based in other countries
providing services for Brazilian Portuguese-speaking clients.
 CLINICAL LINGUISTICS & PHONETICS 17

Future directions
Future research should include collecting normative speech and vocabulary data in
Brazilian Portuguese for children under age three to improve clinical comparison to
noncleft children. Additional development of the PEEPS:BP is also needed in order to
account for dialectal variation in Brazil (Scherer, Yamashita et al., 2020). In this study,
dialectal speech-sound variations were identified and participants were not penalized for
these differences, but there are likely more variations that need to be accounted for and
factored into calculations such as sound classes. These advancements would yield a more
accurate assessment and a more appropriate intervention for cleft and non-cleft Brazilian
Portuguese toddlers. Future research should also consider longer follow up at 6 or 12-
months post-intervention and adaptation of other early interventions for the Brazilian
culture and comparison of EMT+PE to these other early interventions.

Disclosure statement
No potential conflict of interest was reported by the authors.

Funding
This work was supported by the National Institute on Deafness and Other Communication Disorders
[DC013527].

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