On The Benefits of Speech-Language Therapy For

Review Article
On the Benefits of Speech-Language Therapy for

Individuals Born With Cleft Palate: A Systematic
Review and Meta-Analysis of Individual
Participant Data
Anders Sand,a Emilie Hagberg,a,b and Anette Lohmandera,b
a
Division of Speech and Language Pathology, Department of Clinical Science, Intervention and Technology, Karolinska Institutet, Stockholm,
Sweden b Medical Unit Speech and Language Pathology and Stockholm Craniofacial Team, Karolinska University Hospital, Stockholm, Sweden
ARTICLE INFO ABSTRACT

Article History: Purpose: Cleft lip and/or palate (CLP) is a common birth defect, and after reconstructive
Received July 1, 2021 surgery, about 50% of children at 5 years of age have speech deviations and are referred
Revision received September 2, 2021 to speech-language therapy (SLT). The peer-reviewed evidence for the benefit of
Accepted September 23, 2021 SLT has been uncertain. Our objective was to systematically review and meta-
analytically summarize the benefit of SLT for individuals born with CLP.
Editor-in-Chief: Bharath Chandrasekaran Method: A systematic search was conducted (last search on February 19, 2021)
Editor: Kate Bunton on studies evaluating SLT with pre and post measures on speech production,
language ability, intelligibility, and/or patient-reported outcomes. We sought
https://doi.org/10.1044/2021_JSLHR-21-00367 individual participant data (IPD) and evaluated on an individual level if the outcome
measure had improved to a clinically relevant degree during SLT and if the outcome
measure was on a level with peers or not after SLT. Meta-analyses and meta-
regressions were applied to synthesize IPD across studies.
Results: Thirty-four eligible studies were found. Nineteen studies provided IPD
(n = 343) for the main analysis on speech production. The synthesized information
suggests that, during SLT, speech production improved to a clinically relevant
degree for many individuals (95% CI [61%, 87%]) and that speech production
was on a level with peers for some individuals after SLT (95% CI [10%, 34%]).
Conclusions: The main strength of this meta-analysis is that we evaluated on an
individual level pre- and post-intervention data based on considerations of clinical
relevance. This approach allowed us to conclude that many individuals benefit from
SLT and that further work on evaluating SLT in this patient group is meaningful.
Supplemental Material: https://doi.org/10.23641/asha.17700992
Cleft lip and/or palate (CLP) has an incidence rate of reconstructive surgery, a large number of children have no such
approximately one to two per 1,000 births (Mossey et al., problems and develop in line with peers. However, approxi-
2009; Mossey & Modell, 2012). It can affect a person’s facial mately 50% of children with repaired CLP have persisting
appearance, speech development, hearing, and well-being. speech deviations at 5 years of age (Britton et al., 2014;
Children born with CLP are at an increased risk of demonstrat- Lohmander, Persson, et al., 2017), perhaps influenced by age
ing speech and language problems, including phonological/ at surgery, surgical technique, or learned speech behaviors,
articulation delay or disorder, resonance disorders, and expres- and are often referred to speech-language therapy (SLT;
sive language delay or disorder (Kuehn & Moller, 2000). After Hardin-Jones et al., 2009). Thus, the incidence of speech de-
viations is high among children born with CLP, in compari-
son to peers of the same age. Typically developing children
Correspondence to Anders Sand: Anders.Sand@ki.se. Disclosure: The
show a gradual progress in speech sound production with
authors have declared that no competing financial or nonfinancial interests age, and after 4 years of age, most speech sounds has been
existed at the time of publication. acquired correctly (Stoel-Gammon & Sosa, 2007). However,
Journal of Speech, Language, and Hearing Research • Vol. 65 • 555–573 • February 2022 • Copyright © 2022 The Authors 555
This work is licensed under a Creative Commons Attribution 4.0 International License.
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sibilant fricatives and the /r/ sound are typically established et al., 2017; Sell, 2005). However, we also believe that the
during a wide time period, and distortions may occur until unclear literature stems from how interventions have been
around 6 years of age. This general description has been veri- evaluated on a fundamental level and that intervention
fied in a study of 684 English-speaking typical children aged outcomes mostly have been evaluated based on statistical
3;0–6;11 (years;months; Dodd et al., 2003) and in 473 significance (e.g., Alighieri et al., 2020; Hardin-Jones &
Swedish typical children aged 3–6 years (Blumenthal & Chapman, 2008; Pamplona et al., 2005, 2014), rather than
Lundeborg Hammarström, 2014). clinical relevance.
In the group of children with repaired CLP, some
persisting speech problems require secondary surgery, such
as problems related to velopharyngeal incompetence, Clinical Versus Statistical Evaluation
where the normal balance between the oral and nasal cavity
is compromised, resulting in passive speech errors such as In many intervention studies on SLT, null hypothe-
hypernasal resonance, audible nasal emission, weak pressure sis significance tests were applied to group aggregated
on consonants. These are the unavoidable consequence of an data (e.g., t tests, analyses of variance, and Wilcoxon
unwanted oral–nasal coupling on speech when no effort is signed-ranks tests), and a statistically significant pre–post
made by the speaker to change it. Active speech errors, difference in the outcome measure was interpreted as sup-
“compensatory errors,” occur when consonants are realized porting that the intervention was beneficial for the individ-
via a change in place and/or manner of articulation in order uals. Although this analytical approach has been wide-
to compensate for the lack of adequate intraoral air pressure spread, a statistically significant pre–post difference is ac-
required for production of stop and fricative consonants tually theoretically and practically unrelated to the re-
(Harding & Grunwell, 1998). Active speech errors are one of search interests many clinical researchers have (Angst
the contributors to reduced intelligibility and should be et al., 2017; Wasserstein & Lazar, 2016). There are two re-
treated with SLT. Problems with expressive language may search issues. The first issue is about which, if any, of the
further compromise intelligibility, and children might de- individuals enrolled in the study benefited from the inter-
velop less positive attitudes to speech and communication vention to a clinically relevant degree. The second issue is
compared to those without a cleft (Havstam & Lohmander, about what proportion of future individuals, based on the
2011; Whitehill et al., 2011). study at hand, are estimated to benefit from similar
Two major SLT approaches have been adopted to interventions.
treat active speech errors: a motor-phonetic–based ap- The first issue is not a statistical query. It requires
proach grounded in the theory that a motor skill is learned us to define what pre–post changes in a particular out-
through repeated actions of that specific skill using a hierar- come measure should be considered clinically relevant.
chical structured therapy (Albery & Enderby, 1984; Ruscello This is a research aspect that has been somewhat oversha-
& Vallino, 2014) and a language (linguistic)–phonological dowed in both SLT research and clinical research at large
approach that focuses on the organization and representa- (Ioannidis, 2016; McShane et al., 2019; Revicki et al.,
tion of the sound system of a particular language (Chapman, 2008). It is a complex issue because what improvement
1993). In 1965, Prins and Bloomer observed that the majority constitutes a clinically relevant benefit must be sensitive
of individuals with CLP who receive SLT do not achieve to, for example, the patient group in question, the particu-
a speech production at the level of their peers. They lar outcome of interest, and the dosage of the interven-
stated, “The burden of proof to demonstrate the effective- tion. In the specific context of individuals born with CLP,
ness of therapy for oral cleft patients is increasing, and a it is also important to consider, for example, the type of
prerequisite for this is the development of meaningful pro- cleft, the surgical technique, and the age at intervention.
cedures for studying changes in speech behavior” (Prins & Interpreting clinical benefit based only on a statistical
Bloomer, 1965, p. 357). Unfortunately, the peer-reviewed analysis does not take these considerations into account
evidence for the benefit of SLT for individuals born with (Wasserstein et al., 2019). Furthermore, the focus of statis-
CLP is still uncertain (Vallino-Napoli, 2011). In 2013, tical analysis on aggregated data often obfuscates the fact
Bessell et al. conducted a systematic review of the pub- of large interindividual variability in treatment outcomes
lished intervention studies, and despite evaluating 17 (see Box 1). Thus, to evaluate SLT, we cannot only statis-
studies, they found little evidence to support the effective- tically evaluate treatment results but also need to evaluate
ness of SLT in children with CLP. Bessell et al. (2013) di- clinical benefit on an individual level. Finally, in SLT con-
agnosed this uncertainty as stemming from very heteroge- texts, we are interested not only in whether a pre–post
neous measuring and analytical procedures and argued change is clinically relevant or not but also in whether the
that more rigorous methodology is needed. We certainly outcome measure (e.g., speech production) is on a level
agree with calls for more rigorous and standardized with peers or not after SLT. This is again a complex issue
methods for studying changes in speech behavior (Allori that requires us to consider prior knowledge of typical
556 Journal of Speech, Language, and Hearing Research • Vol. 65 • 555–573 • February 2022

speech development and the individual in question, not Purpose
only statistical concerns.
The second issue is a statistical query, but one quite Our primary goal in this systematic review was to
easily answered once the more complex first issue has been obtain individual participant data (IPD) to evaluate the
dealt with: Simply count the proportion of individuals that treatment effectiveness of SLT in individuals born with
benefitted from the intervention. To get a statistic that CLP. Our first aim was to meta-analytically estimate what
summarizes what should be generalized from the study, proportion of individuals receiving SLT improved to a
calculate a confidence interval (CI) around this proportion. clinically relevant degree during treatment. Our second
The CI should be interpreted as a “compatibility interval” aim was to meta-analytically estimate what proportion of
illustrating what parameter values are most compatible individuals had outcome measures on a level with peers
with the data (Amrhein et al., 2019; Greenland, 2019). In after SLT. We also asked if the dispersion in treatment effects
this context, the CI can be interpreted as summarizing could be related to individual factors or intervention details.
what possible proportions of future individuals might benefit Using clinical considerations to evaluate IPD is novel in this
from similar interventions. area because previous studies and systematic reviews have
focused either on inferential group-level statistics or on
abstract effect size measures. Our secondary goal of this
review was to narratively summarize the intervention
Box 1. The difference between statistically
studies IPD could not be obtained from. Our outcomes of
significant and clinically relevant.
interest were speech production, language ability, intelligi-
bility, and patient-reported outcomes (PROs).
PCC before PCC after
Participant SLT SLT Difference
A 20% 26% 6%-points Method

B 25% 29% 4%-points
C 30% 35% 5%-points
D 35% 41% 6%-points A protocol of our systematic review was registered in
E 40% 45% 5%-points 2019 at PROSPERO (https://www.crd.york.ac.uk/PROSPERO/
M (SD) 30% 35% 5%-points display_record.php?RecordID=165754). See Supplemental
(8%-points) (8%-points) (0.8%-points)
Material S1 for the PRISMA IPD checklist.
In the table above, we have illustrated five individ- Inclusion Criteria

uals’ pre- and post-intervention speech production
measured as percentage of consonants correct The inclusion criteria for this systematic review was
[PCC] in a picture-naming task. The pre–post differ- that a study had to (a) evaluate a conventional SLT inter-
ence is consistent between individuals and, thus, vention; (b) measure an outcome that related to speech
statistically significant (t4 = 13.9, p < .001). However, production, language ability, intelligibility, or PROs; and
none of the individuals improved to a clinically rele- (c) be readable in English.
vant degree. As conventional SLT, we considered therapy aiming at
In the table below, we have illustrated a similar the correct production of deviant consonant sounds using either
set of data. Here, the pre–post difference is inconsis- a motor-phonetic approach (Ruscello & Vallino, 2014) or a
tent between individuals and, thus, not statistically language (linguistic)–phonological approach (Chapman,
significant (t4 = 1.6, p = .18). Some individuals have, 1993). We did not include studies evaluating the use of
however, improved to a clinically relevant degree; an speech bulbs, continuous airway pressure, and aerodynamic
increase from 25% or 35% to 95% should be impor- treatment, for example.
tant in everyday settings. Regarding eligible outcomes, as measures of speech
production, we included measures of articulation and
PCC before PCC after consonant proficiency, consonant errors, and compensatory
Participant SLT SLT Difference articulation. We did not include acoustical measures of
A 20% 20% 0%-points
speech production or physiological measures of tongue
B 25% 95% 70%-points position, lateral pharyngeal wall movements, or measures
C 30% 30% 0%-points of oral pressure. As measures of ability in different aspects
D 35% 95% 60%-points
E 40% 40% 0%-points
of language, we included all expressive aspects, such as
M (SD) 30% 56% 26%-points consonant inventory and other measures of phonology,
(8%-points) (36%-points) (36%-points) lexicon or vocabulary measures, sentence complexity, and
measures of grammar. As measures of intelligibility, we
Sand et al.: On the Benefits of Speech-Language Therapy 557

included caregiver or clinician ratings or other measures however, through these means identify any further
of intelligibility or understandability. As measures of studies.
PROs, we included self- or caregiver-reported indications
of communicative participation or quality of life. Study Selection
Study design (e.g., randomized controlled trial
[RCT]) was not an inclusion criterion, instead we included We conducted two rounds of screening using Rayyan
evidence from all types of studies from RCT to case reports. (Ouzzani et al., 2016). In the first round, we excluded,
This was the case because our IPD-focused analysis often based on title and abstract information, articles that did
implied a comparison other than the original study compari- not meet our inclusion criteria. One author (A.S.) screened
son anyway. all articles, and another author (E.H.) independently
On a participant level, the inclusion criteria for the screened a randomly selected subset of 433 articles (10%
IPD-focused analysis were that participants had to (a) have of the initial search). The two authors strongly agreed in
been born with a CLP, (b) have outcome measures below this first round of screening; they agreed on 431 out of
the level of their peers before intervention, and (c) not have 433 articles (Cohen’s kappa = .94).
additional malformations or syndromes. We included indi- In the second round, we excluded, based on full-text
viduals born with unilateral or bilateral cleft lip and palate information, articles that did not meet our inclusion criteria.
and cleft palate but excluded individuals with noncleft velo- Two authors (A.S. and E.H.) independently reviewed these
pharyngeal dysfunction, for example. We excluded individ- articles. When there was disagreement, the third author (A.L.)
uals with syndromes, for example, 22Q11.2 deletion or helped settle this. See Figure 1 for a flowchart of the reviewed
CHARGE. After obtaining IPD, we concluded that most in- studies. For more information on why we excluded some
cluded participants were young (below 20 years old) and studies, including some studies included in the review by
therefore excluded one participant because of his age Bessell et al. (2013), please see Supplemental Material S3.
(39 years old).
Search Strategy
Data Collection
To perform our electronic search, we engaged the
Karolinska Institute University Library search consulta- We found 34 eligible studies that are listed in Supplemen-
tion group. Our search targeted Medline, Embase, tal Material S4. Twenty-one of these studies contributed with
Cochrane, Web of Science, PsycInfo, and CINAHL. The IPD. From 19 studies, IPD could be tabulated from the article
specific search strategies for each database can be found and from two studies; IPD was received upon request from the
in Supplemental Material S2, but they generally con- author. These 19 studies contributed with IPD on one or more
sisted of two “blocks”: one describing the population of the outcomes of interest (see Figure 1 and Supplemental
group (e.g., “cleft palate,” “cleft lip,” and “velopharyngeal Material S4). Thirteen studies did not contribute with IPD: In
insufficiency”) and the other one describing the intervention/ seven cases, we contacted the authors but were not able to ob-
outcomes of interest (e.g., “speech therap*” and “language tain IPD, and in six cases, where the studies were published be-
training”). The MeSH terms identified for searching Med- tween 13 and 50 years ago, we did not request IPD because we
line (OVID) were adapted in accordance to corresponding judged it unlikely that we would obtain it.
vocabularies in Embase, PsychInfo, and CINAHL. Each On a study level, we tabulated type of therapy (motor-
search concept was also complemented with relevant free- phonetic vs. phonological/language), the delivery of therapy
text terms, and these were, if appropriate, truncated and/ (directly by a speech-language pathologist or by a speech as-
or combined with proximity operators. We had no restric- sistant or caregiver, and if the therapy was delivered in a
tion regarding year of publishing, language, or type of ar- clinic or some other setting), and the dosage of the interven-
ticle (dissertations, theses, etc.) in our search strategy as tion. Because therapy details were heterogeneous between
including studies reported in all types of publication will studies, we were not able to systematically tabulate more de-
reduce potential bias (Boutron et al., 2021). Our last tailed information on dosage (Allen, 2013) than the total in-
search was on February 19, 2021. This search yielded tervention duration.
4,728 results. On the IPD level, we tabulated demographic data
Our electronic search was supplemented by reviewing regarding age, type of cleft, gender, syndrome, and the out-
the reference lists from included articles and published re- come measures outlined above pre- and post-intervention.
views and by an expert’s insights. To gain access to poten- When there were uncertainties in the reporting, we contacted
tially unpublished data (Boutron et al., 2021), we also authors to verify that we correctly interpreted the information.
contacted several researchers who had described their The main aspect of the IPD-focused analysis was that
studies as “preliminary” or “pilot studies.” We did not, we evaluated on an individual level separately (a) if the

Figure 1. Flow chart of study identification, inclusion, and organization of studies based on the data obtained and outcome measure. A total of 34
studies were included in this systematic review. We obtained individual participant data (IPD) from 21 studies. Several of these contributed with
IPD on more than one outcome; 19 contributed IPD to the main analysis on speech production. We were not able to obtain IPD from 13 studies,
only the aggregated data presented in the article. Wrong outcome = the study did not measure one of our targeted outcomes; Wrong treatment =
the study did not investigate conventional SLT intervention; PROs = patient-reported outcomes. See inclusion criteria for more details.
individual improved to a clinically relevant degree during completely on the same level as peers without a history of
treatment and (b) if the individual’s outcome measure was clefts or other speech deviations. However, one speech pro-
on a level with peers or not after SLT. These evaluations duction error was accepted as being on peer level at all ages
were based on the second and third authors’ (E.H. and A.L.) as one error could be an error of measurement.
clinical expertise, knowledge of the studied therapies and The prior information we used to evaluate the IPD
measures, and knowledge on typical speech and language de- was, in short, that after 4 years of age the phonemic in-
velopment, complemented with reference data for specific ventory is completed in most children, who thereafter
measures where available. In evaluating (a), the IPD infor- gradually reach adultlike precision of consonant sounds
mation we considered was the age of the individual, the indi- (Stoel-Gammon & Sosa, 2007). Although many conso-
vidual’s pre–post change, and the dosage of the intervention nants are similar across languages, there are differences re-
(in relation to the prior information described below). In garding voicing, aspiration, and subtle differences in the
evaluating (b), the IPD information we considered was the place and manner of articulation (Hutters & Henningsson,
age of the individual and the outcome measure after SLT (in 2004). Such differences could not be fully considered in all
relation to the prior information described below), disregard- included studies. Reference values on PCC and consonant
ing the dosage of the intervention and any idiosyncratic errors among typically developed children between 3 and
speech deviations of the patient before intervention. We were 19 years of age (Dodd et al., 2003; Lohmander,
strict in evaluating (b), requiring the outcome to be Lundeborg, & Persson, 2017) were used to interpret the

included IPD and to grossly evaluate relevant improve- Risk of Bias Assessment
ment during treatment and if speech production was on a
level with peers or not after SLT. Typically, a strong We were inspired by the ROBINS-I tool (Sterne
progress in consonant production between 3 and 5 years et al., 2016) in assessing the risk of bias in the reviewed
of age is seen and again from 7 years of age where the studies. ROBINS-I mainly targets studies in which two
median number of consonant errors in this group is zero. groups of patients are compared, often one intervention
As mentioned, speech production was measured in group versus a no-intervention group. The comparison
different ways in the included studies. Below, we exemplify that we tabulated from the included studies, however, was
our evaluation for three types of measures and studies. uncontrolled before–after comparisons; in other words, it
Prathanee et al. (2014) reported the number of consonant only contained an intervention group. Thus, some do-
errors for their patients. For example, they reported on a 6- mains of the ROBINS-I tool were not relevant in this review.
year-old child who had 13 consonant errors before and one Instead, we focused on two domains of the ROBINS-I tool:
error after SLT. We evaluated this child’s pre–post change bias due to confounding factors (Question 1.1 in the
as being a clinically relevant improvement and the child’s ROBINS-I tool) and bias in measurement of outcomes. We
speech production as being on a level with peers after SLT. used the categories for risk of bias judgments in line with the
In comparison, for a 7-year-old child who had 15 errors be- ROBINS-I tool (Sterne et al., 2016). We did not include the
fore and three errors after SLT, we evaluated this child’s risk of bias judgments in the quantitative analysis by, for ex-
pre–post change as being a clinically relevant improvement, ample, weighing down studies with greater risk of bias.
but the child’s speech production as not being on a level with We identified two probable confounding factors in
peers after SLT. No consideration was taken to type of con- the included studies. (a) We assessed whether maturation
sonant error. (i.e., that the cleft-related speech behaviors or language
Van Demark and Hardin (1986) reported PCC. For delay resolved spontaneously) could explain any improve-
example, they reported on an 8-year-old child who had a ment occurring during treatment. We judged this to be
PCC of 77% before and 96% after SLT. We evaluated this more likely if the patients were very young and/or if the
child’s improvement as being clinically relevant and her total intervention duration was long. (b) We assessed
speech production as being on a level with peers after whether familiarity with the testing procedure or material
SLT. In comparison, for an almost 9-year-old child who had could explain any improvement occurring during treat-
71% before and 87% after SLT, we evaluated the improvement. We judged this to be more likely if the patient was
ment as being clinically relevant, but the child’s speech pro- repeatedly tested with short intervals between the tests.
duction as not being on a level with peers after SLT. We also assessed whether the outcome measure could
Pamplona et al. (2014) reported a rating of the qual- have been influenced by the assessors not being blinded
ity of consonant articulation in their studies. We evaluated toward the patient’s treatment or time point of measure-
a change from constant compensatory articulation/a single ment (Questions 6.1–6.2 in the ROBINS-I tool).
phoneme correct (Scale Steps 0 and 1) before SLT to cor-
rect consonant production in syllables, words, or short Analytical Procedure
phrases in a closed context (Scale Steps 2 or 2–4 in differ-
ent scales used) after SLT, as an improvement at all ages. For each study that we were able to obtain IPD (see
For peer-level evaluation, however, this level was only ac- Supplemental Material S4), we calculated the proportion
cepted in 3-year-olds, whereas correct production in an of individuals who improved to a clinically relevant degree
open context (Scale Steps 3–5 in the different scales) was during treatment and the proportion of individuals whose
required at 4 years of age, and appropriate production or outcome measures were on a level with peers after SLT.
with only a single/inconsistent error (Scale Steps 5–7) was For the outcomes other than speech production, we found
accepted from 5 years of age as being on a level with too few studies to carry out any formal quantitative syn-
peers. thesizing and instead only present the result of our evalua-
In summary, we could establish more formal proce- tions in the Results section. For the studies on speech pro-
dures for our clinical evaluations in some studies, but for duction for which we were not able to obtain IPD, we
many studies, the evaluation was made in an informal man- only narratively summarized the aggregated results pre-
ner. In a few studies in which the primary researchers made, sented in the articles.
formal or informational, evaluations of clinical improve- We performed a proper meta-analysis on the IPD
ment, we based our evaluation on their evaluation. We also for the outcome speech production. All statistical analyses
tried to contact the primary researchers to understand were conducted in R (R Core Team, 2013) and are avail-
how they evaluated their data from a clinical perspective. able as Supplemental Material S6. We carried out two
IPD tabulated from the included studies, and all our clini- parallel analytical procedures on (a) what proportion of
cal evaluations can be found in Supplemental Material S5. individuals improved to a clinically relevant degree during

treatment and (b) what proportion of individuals whose studies (Alighieri et al., 2019; Lindeborg et al., 2020; Luyten
speech production was on a level with peers after SLT. In et al., 2016). See Table 1 for a general overview of these
both analyses, we first calculated a meta-analytical aver- studies, and see Table 2 for risk of bias assessment of these
age across studies and the dispersion (prediction intervals) studies. In all studies, possible biases favored treatment.
around these estimates (Borenstein, 2019). Briefly, the Five, eight, and one studies contributed with IPD
meta-analytical average proportion was estimated by for the outcomes language ability, intelligibility, and
weighting each individual study by its inverse variance PROs, respectively. We elected to not perform a quantita-
(for more information on meta-analyses, please see tive synthesis of these studies and instead only present the
Borenstein, 2019; Viechtbauer, 2010; Wang, 2018). Be- study results in Table 3.
cause the studied interventions differed in type, delivery, Twelve studies measured speech production but did
and dosage of intervention and the studied patient not contribute with IPD. We elected to not quantitatively
groups were heterogeneous, we assumed there to be synthesize the aggregated results of these studies and in-
large heterogeneity in intervention effects and used a stead narratively describe them in Supplemental Material
random-effects meta-analytical model. Because sample S7. One study measured language ability, but we were not
sizes were often small and extreme proportions were in- able to obtain IPD (Pamplona et al., 2015).
cluded (e.g., 0.0 and 1.0), we applied a double arcsine
transformation on the proportions (Viechtbauer, 2010) but Aim 1: To Estimate the Proportion of
present the back-transformed results. We fitted the models Individuals Who Improved to a Clinically
using restricted maximum-likelihood estimation (Laplace Relevant Degree During Intervention
approximation). The meta-analyses were conducted using
the “metafor” package in R (Viechtbauer, 2010; Wang, Figure 2 summarizes the meta-analysis on the pro-
2018). portion of individuals who improved to a clinically rele-
In both analyses, our second step was to investigate vant degree during intervention. On average, across inter-
to what extent individual factors and intervention details vention details and individual demographics, 75% of indi-
could explain the dispersion in the intervention effect. For viduals improved during treatment (95% CI [61%, 87%]).
individual-level factors (e.g., age at intervention), we ap- However, the dispersion around this meta-analytical aver-
plied mixed-effects logistic regression models that allow age was large; the prediction interval indicates that, for
studies to have random intercepts rather than meta- some interventions and demographic groups studied, the
regression, because of the large within-study heterogeneity proportion could be as low as 20%, and for other cases, it
of these factors. For study-level factors, we applied a could be as high as 100%. Our second step was to address
meta-regression model. The mixed-effects logistic regres- factors potentially able to explain the variation in inter-
sions were conducted using the “lme4” package and the vention effect. No individual study was deemed an outlier
meta-regression using the “metafor” package in R (Bates in the sense that it influenced the meta-analytical estimate
et al., 2015; Viechtbauer, 2010). to a disproportionate degree.
Figure 3 summarizes the mixed-effects logistic re-
gression we used to assess how age at intervention was re-
Results lated to the probability to improve during intervention.
In summary, age at intervention was strongly related to
Overview of the Included Studies the probability that an individual would improve during
intervention: For children 6 years of age or younger, the
Nineteen studies contributed with IPD on speech model estimated on average roughly 80% probability that
production. As a rough overview of these studies, the therapy they would improve during SLT. For individuals older
was implemented in the form of intensive summer camps in than 14 years of age, the predicted probability was only
three studies (Pamplona et al., 2014, 2017; Van Demark & about 40%. This model was based on 343 individuals from
Hardin, 1986), the SLT was implemented in the form of con- 19 studies. The intercept in this model (in log odds) was
ventional clinical care in four studies (Derakhshandeh et al., 1.99 (95% CI [1.02, 3.07]), and the regression coefficient
2016; Pamplona & Ysunza, 2018; Roxburgh et al., 2016; (in log odds) was −0.011 (95% CI [−0.021, −0.002]) per
Sweeney et al., 2020), the therapy was implemented through month of age. As illustrated in Figure 3, there was consider-
an initial short workshop led by SLTs and then carried out able between-studies variation; the standard deviation be-
by parents or speech assistants over a long duration in nine tween the random intercepts was (in log odds) 1.12.
studies (Dobbelsteyn et al., 2014; Hanchanlert et al., 2015; Table 4 summarizes the mixed-effects logistic regression
Makarabhirom et al., 2015; Prathanee, 2011; Prathanee we used to assess how cleft type was related to the probabil-
et al., 2014, 2020; Pumnum et al., 2015; Scherer et al., 2008; ity of improving during intervention. In this analysis, we
Sritacha et al., 2016), and the therapy was very brief in three dummy-coded types of cleft into unilateral CLP, bilateral

562
Table 1. Study level information on the studies included in the quantitative synthesis.
Journal of Speech, Language, and Hearing Research • Vol. 65 • 555–573 • February 2022
Age
Therapy Intervention range
Study type Delivery duration N a
(months) Speech measure Intervention description
Alighieri et al. Motor-phonetic SLP (clinic) 3 days 2 137–140 PCC based on 15 sentencesb in Six 1-hr sessions
(2019) the SNAP test SLT in patients’ secondary language
Derakhshandeh Motor-phonetic SLP (clinic) 10 weeks 5 54–108 PCC in 14 oral and two nasal Four sessions of 45-min per week for
et al. (2016) sentencesb (by picture naming) 10 weeks
Dobbelsteyn Motor-phonetic Parent (home) 4 months 7 51–192 PCC in SAF containing 11 oral Parents completed a 4-hr workshop
et al. (2014) phrasesb and then gave daily 10-min sessions
for 4 months.
Hanchanlert Motor-phonetic Speech assistant 9 months 6 47–124 Number of articulation errors on One- to 3-day speech camps with three
et al. (2015) and parent sentencesb on the TUPS to four 45-min sessions daily with SLP
(home) Speech assistants and caregivers gave
weekly or biweekly 30-min sessions for
9 months.
Lindeborg Motor-phonetic SLP and speech 1 week 38 36–216 Number of articulation errors on One week speech camp with daily sessions
et al. (2020) assistant repeated single words of 30–60 min administered by speech
(camp) assistants assisted by SLPs
Luyten et al. Motor-phonetic SLP (clinic) 3 days 5 88–235 PCC based on 15 sentencesb in Six 1-hr sessions over 3–4 days
(2016) the SNAP test SLT in patients’ secondary language
Makarabhirom Motor-phonetic Speech assistant 9 months 16 40–147 Number of articulation errors on Three-day speech camps with three to
et al. (2015) and parent sentencesb on the TUPS four 45-min sessions daily with SLP
(home) Speech assistants and caregivers then
gave weekly 30- to 45-min sessions
for 9 months.
Pamplona & Language/ SLP (clinic) No information 32 43–81 20 min of spontaneous speech Fifteen sessions of 45 min (unclear interval)
Ysunza (2018) phonological rated according to severity of
compensatory articulation
Pamplona et al. Language/ SLP (camp) 4 weeks 90 36–80 20 min of spontaneous speech Summer camp with 4-hr therapy per day,
(2014) phonological rated according to severity 5 days a week for 4 weeks
of compensatory articulation
Pamplona et al. Language/ SLP (camp) 3 weeks 41 36–77 20 min of spontaneous speech Summer camp with 4-hr therapy per day,
(2017) phonological rated according to severity of 5 days a week for 3 weeks
compensatory articulation
Prathanee et al. Motor-phonetic Speech assistant 9 months 16 49–96 Number of articulation errors on Three-day speech camps with SLP
(2014) (home) sentencesb on the TUPS Speech assistants then gave six 1-day
sessions at home over 9 months.
Prathanee et al. Motor-phonetic Speech assistant 1 year 12 43–192 Number of articulation errors on Three-day speech camps with six sessions
(2020) and parent words on the TUPS of 45 min per day with SLP
(home) Speech assistants then gave six 1-day
sessions at home over 9 months.
Three 1-day follow-up speech camps with
SLP
Prathanee (2011) Motor-phonetic Speech assistant 6 months 9 54–150 Number of articulation errors in Four-day speech camps with 18 hr of
and parent an articulation test (by picture treatment with SLP
(home) naming) Caregivers and health providers were
responsible for children’s speech
program for 6 months.
(table continues)

Table 1. (Continued).
Age
Therapy Intervention range
Study type Delivery duration N a
(months) Speech measure Intervention description
Pumnum et al. Motor-phonetic Speech assistant 9 months 4 68–104 Number of articulation errors on One- to 3-day speech camps with three to
(2015) and parent sentencesb on the TUPS four 45-min sessions daily with SLP
(home) Speech assistants and caregivers then gave
weekly or biweekly 30-min sessions for
9 months.
Roxburgh et al. Motor-phonetic SLP (clinic) 2 weeks 2 74–110 PCC on 36 untrained words selected Eight sessions of 60 min over 2 weeks
(2016) specifically for the patient’s
articulation errors
Scherer et al. Motor-phonetic Parent (home) 3 months 10 18–35 PCC-R measured from 30 min of Parents completed two to four training
(2008) recorded language sample from sessions and implemented this for
parent–child interaction 3 months.
Sritacha et al. Motor-phonetic Speech assistant 9 months 6 49–72 Number of articulation errors on One- to 3-day speech camps with three
(2016) and parent sentencesb on the TUPS to four 45-min sessions daily with SLP
(home) Speech assistants and caregivers then
gave weekly or biweekly 30-min sessions
Sand et al.: On the Benefits of Speech-Language Therapy
for 9 months.
Sweeney et al. Motor-phonetic SLP (clinic) vs. 3 months 29 35–85 PCC calculated for sentencesb in One group (n = 12a) received six therapy
(2020) parent (home) CAPS-A sessions over 12 weeks.
In the other group (n = 17a), parents attended
2 days of training and then carried out the
intervention for 12 weeks.
Van Demark & Motor-phonetic SLP (camp) 6 weeks 13 80–144 PCC articulation on ICPAT Summer camp with 4-hr therapy per day for
Hardin (1986) sentences. 6 weeks
Note. SLP = speech-language pathologist; PCC = percentage of consonants correct; PCC-R = percentage of consonants correct–revised; SNAP = simplified nasometric assessment proce-
dures; SLT = speech-language therapy; SAF = speech assessment form; TUPS = Thai universal parameters of speech outcomes for people with cleft palate; CAPS-A = Cleft Audit Protocol for
Speech–Augmented; ICPAT = Iowa Cleft Palate Articulation Test.
a
Number of individuals included in our synthesis; we excluded individuals because of syndromes, because pretherapy speech production was already on a level with peers (accord-
ing to the presented information), because of missing values, or because of patient overlap between studies. See Supplemental Material S5 for more information. bWhen both were
available, we selected speech production on sentence rather than word level, because we believe this to have more ecological validity.
563

564
Table 2. Risk of bias assessment for the studies included in the quantitative synthesis.
Bias in
Journal of Speech, Language, and Hearing Research • Vol. 65 • 555–573 • February 2022
measurement
Judgment
Bias due to confounding of outcomes
across
Study Maturation Familiarity Judgment Blind Judgment domains Note
Alighieri et al. N Y Serious Y Low Serious Possible familiarity from using same speech material at pre and
(2019) post. The short revision before assessment might enhance this.
(Contacted authors; no reply.)
Derakhshandeh N N Low N Serious Serious
et al. (2016)
Dobbelsteyn Y N Moderate N Serious Serious Possible maturation over the 4 months of intervention, especially for
et al. (2014) the younger children.
Hanchanlert Y N Serious N Serious Serious Possible maturation over the 9 months of intervention, especially for
Lindeborg et al. N N Low Y Low Low
(2020)
Luyten et al. N Y Serious N Serious Serious Possible familiarity from using the same speech material at pre and
(2016) post. One of three assessors was blinded.
Makarabhirom Y N Serious N Serious Serious Possible maturation over the 9 months of intervention, especially for
Pamplona & No information N No information N Serious Serious Unclear interval of treatment, so we cannot judge risk of maturation
Ysunza effects.
(2018) Unclear if and how assessors were blinded.
Pamplona et al. N N Low N Serious Serious Unclear if and how assessors were blinded.
(2014)
Pamplona et al. N N Low N Serious Serious Unclear if and how assessors were blinded.
(2017)
Prathanee et al. Y N Serious N Serious Serious Possible maturation over the 6 months of intervention, especially for
(2014) the younger children.
Prathanee et al. Y N Serious N Serious Serious Possible maturation over the 9 months of intervention, especially for
Prathanee Y N Serious N Serious Serious Possible maturation over the 1 year of intervention, especially for
Pumnum et al. Y N Serious N Serious Serious Possible maturation over the 9 months of intervention, especially for
Roxburgh et al. N N Low N Moderate Moderate Two of three assessors were blinded.
(2016)
Scherer et al. Y N Serious Y Low Serious Possible maturation over the 3 months of intervention, especially for
Sritacha et al. Y N Serious N Serious Serious Possible maturation over the 9 months of intervention, especially for
Sweeney et al. N N Low Y Low Low
(2020)
Van Demark & N N Low N Serious Serious
Hardin (1986)
Note. Y = yes; N = no.

Table 3. Summary of our evaluations on outcomes, other than speech production, regarding improvement during therapy and outcome measures
on a level with peers after speech-language therapy (SLT).
Improved during therapy At peer level after SLT

Outcome Total
measure Study Na n Proportion (95% CI ) b
n Proportion (95% CIb)
Language aspects Hanchanlert et al. (2015) 1 0 0.00 [0.00, 0.98] 0 0.00 [0.00, 0.98]
Pamplona & Ysunza (2000) 41 28 0.68 [0.52, 0.82] 7c 0.17 [0.07, 0.32]
Prathanee et al. (2020) 7 3 0.43 [0.10, 0.82] 3 0.43 [0.10, 0.82]
Pumnum et al. (2015) 1 1 1.00 [0.03, 1.00] 1 1.00 [0.03, 1.00]
Sritacha et al. (2016) 3 2 0.67 [0.09, 0.99] 2 0.67 [0.09, 0.99]
Intelligibility Alighieri et al. (2019) 2 0 0.00 [0.00, 0.84] 0 0.00 [0.00, 0.84]
Hanchanlert et al. (2015) 0 — —
Luyten et al. (2016) 5 2 0.40 [0.05, 0.85] 2 0.40 [0.05, 0.85]
Prathanee et al. (2020) 8 3 0.38 [0.09, 0.76] 3 0.38 [0.09, 0.76]
Prins & Bloomer (1965) 10 5 0.50 [0.19, 0.81] 0 0.00 [0.00, 0.31]
Pumnum et al. (2015) 2 0 0.00 [0.00, 0.84] 0 0.00 [0.00, 0.84]
Sritacha et al. (2016) 1 1 1.00 [0.03, 1.00] 1 1.00 [0.03, 1.00]
Sweeney et al. (2020) 24 10 0.42 [0.22, 0.63] 8 0.33 [0.16, 0.55]
PROs Sweeney et al. (2020) 15 10 0.67 [0.38, 0.88] 8 0.53 [0.27, 0.79]
Note. Em dashes signify that no participant was included from Hanchanlert et al. (2015), as all participants either were included in other
studies or had outcome measures before SLT already on reference level.
a
We excluded participants whose outcome measures before SLT already were on reference level. See Supplemental Material S5 for more in-
formation. bClopper–Pearson “exact intervals.” cA conservative estimate because of lack of information on the individual children’s age.
CLP, and cleft palate, and we used unilateral CLP as refer- log odds. This model was based on 303 individuals from 18
ence and included studies as random intercepts. In summary, studies; Lindeborg et al. (2020) did not include information
the model suggests that individuals with a bilateral CLP on type of cleft and could thus not be included in this analy-
were slightly less likely to improve during therapy; however, sis. The intercept in this model (in log odds) was 1.35 (95% CI
note that there is considerable uncertainty in how strong this [0.63, 2.16]), and the regression coefficients (in log odds) were
effect is; the 95% CI around the coefficient of having a bilat- −0.61 (95% CI [−1.67, 0.44]) and −0.19 (95% CI [−1.19, 0.86])
eral CLP, compared to unilateral CLP, was [−1.67, 0.44] in for bilateral CLP or cleft palate, as compared to unilateral
Figure 2. Overview of the meta-analysis on proportion of individuals who improved to a clinically relevant degree during intervention (number
of improved participants divided by sample size). In the forest plot, the point estimate of each study is illustrated as gray boxes, and the
lines illustrate 95% confidence intervals (CIs; Clopper–Pearson “exact method”). The sizes of the gray boxes are relative to the (random-
effects) weights of the studies (their relative “precision” or inverse variance, compared to full data set). The studies are roughly grouped and
delimited by horizontal lines based on study design and intervention implementation. On the bottom, the meta-analytical CI is illustrated as a
diamond, and the prediction interval is illustrated as a thick line.

Figure 3. Illustration of the mixed-effects logistic regression model in production on a level with peers after SLT (95% CI [10%,
which age at intervention was related to the probability that an indi-
vidual improved to a clinically relevant degree during the intervention. 34%]). However, the dispersion around this meta-analytical
The logistic regression function has been back-transformed to the average was large; the prediction interval was between 0%
probability scale (y-axis). The solid black line illustrates the logistic and 71%.
function over the whole age range using the average of the ran-
dom intercepts. The gray lines illustrate each included study. Note Figure 5 summarizes the mixed-effects logistic regres-
that the studies differ in intercepts (studies are random effects) sion we used to assess how age at intervention was related to
and age range of studied individuals, but not the coefficient (age the probability to have speech production on a level with
was fitted as a fixed effect).
peers after SLT. In summary, age at intervention was weakly
related to this probability. For example, a 6-year-old child
was not that much more probable to achieve this than a 14-
year-old teen (note that they are compared to peers of their re-
spective age). Figure 5 illustrates that there is more variation
in the studies intercepts than there is along the age range, indi-
cating that most of the between-studies heterogeneity is unac-
counted for. This model was based on 343 individuals from
19 studies. The intercept in this model (in log odds) was −0.77
(95% CI [−1.94, 0.34]), and the regression coefficient (in log
odds) was −0.01 (95% CI [−0.020, 0.003]) per month of age.
As mentioned, there was considerable between-studies varia-
tion; the standard deviation between the random intercepts
was (in log odds) 1.24.
Table 4 summarizes the mixed-effects logistic regres-
sion we used to assess how cleft type was related to the
probability to have a speech production on a level with
peers after SLT. In summary, individuals with cleft palate
only seem to have a greater chance of achieving this. This
model was based on 303 individuals from 18 studies;
Lindeborg et al. (2020) did not include information on
type of cleft and could thus not be included in this analysis.
CLP. The standard deviation between the random intercepts The intercept in this model (in log odds) was −1.59 (95% CI
was (in log odds) 1.06. [−2.65, −0.78]), and the regression coefficients (in log odds)
were −0.06 (95% CI [−1.18, 0.99]) and 1.01 (95% CI [0.03,
Aim 2: To Estimate the Proportion of 2.03]) for having a bilateral CLP or cleft palate, as compared
Individuals Whose Speech Production Was to unilateral CLP. The standard deviation between the ran-
on a Level With Peers dom intercepts was (in log odds) 1.33.
Figure 6 summarizes the meta-regression we used to
Figure 4 summarizes the meta-analysis on the propor- assess how intervention duration was related to the probability
tion of individuals whose speech production was on a level to have a speech production on a level with peers after SLT.
with peers after SLT. On average, across intervention details In summary, longer intervention durations were related to a
and individual demographics, 21% of individuals had speech larger proportion of patients achieving this. This model was
Table 4. Probability to improve during therapy and to have speech production on a level with peers after
speech-language therapy as a function of type of cleft.
Improved Peer level

Total
Type of cleft Na n (%) Predictedb n (%) Predictedb
Unilateral CLP 240 193 (80) 79% 52 (22) 17%

Bilateral CLP 28 18 (64) 68% 8 (29) 16%
Cleft palate 35 25 (71) 76% 14 (40) 36%
Note. CLP = cleft lip and/or palate.

a
One individual with submucous cleft palate and one individual with soft palate cleft were coded as having a
cleft palate; one individual with a cleft lip was excluded. bThe probabilities reported under “Predicted” are the
model predictions (back-transformed to the probability scale) that take between-studies variability into account
(as random intercepts).

Figure 4. Overview of the meta-analysis on proportion of individuals who had speech production on a level with peers after speech-
language therapy (SLT). In the forest plot, the point estimate of each study is illustrated as gray boxes, and the lines illustrate 95% confidence intervals
(CIs; Clopper–Pearson “exact method”). The sizes of the gray boxes are relative to the (random effects) weights of the studies (their relative “precision”
or inverse variance compared to full data set). The studies are roughly grouped and delimited by horizontal lines based on study design and interven-
tion implementation. On the bottom, the meta-analytical CI is illustrated as a diamond, and the prediction interval is illustrated as a thick line.
Figure 5. Illustration of the mixed-effects logistic regression model

in which age at intervention was related to the probability that an
individual had speech production on a level with peers after SLT.
The logistic regression function has been back-transformed to the Figure 6. Illustration of the meta-regression model in which intervention
probability scale (y-axis). The solid black line illustrates the logistic duration was related to the proportion of individuals who had speech
function over the whole age range using the average of the ran- production on a level with peers after speech-language therapy. Circles
dom intercepts. The gray lines illustrate each included study. Note illustrate each included study; the sizes of the circles are relative to the
that the studies differ in intercepts (studies are random effects) (random effects) weights of the studies. The solid black line illustrates
and age range of studied individuals, but not the coefficient (age the regression line. The gray and dashed lines illustrate the upper and
was fitted as a fixed effect). lower limits of a 95% confidence interval around the regression line.

based on 18 studies; Pamplona and Ysunza (2018) did not helpful, about 20% (lower limit of the prediction interval)
include information on intervention duration and could thus of individuals are predicted to improve to a clinically rele-
not be included in this analysis. The intercept in this model vant degree. On average, across intervention details and
(in percentage points) was 0.33 (95% CI [0.19, 0.47]), and the study designs, about 61%–87% (95% CI) of individuals are
regression coefficient was 0.01 (95% CI [0.006, 0.018]) per predicted to improve during SLT. Thus, although we can-
week of intervention. In conclusion, intervention duration not specify the parameters of SLT that provide the best
was able to explain parts of the heterogeneity in the interven- results for the patients, SLT does seem to have positive ef-
tion effect (Q1 = 14.75, p = .0001). fects for many or most individuals who receive it.
The main factor explaining the dispersion in the esti-
mated proportion of individuals who improved from inter-
Discussion vention was the individual’s age at intervention: Young
children (e.g., younger than 6 years old) had a greater
Although our original intent was to treat the four probability to improve than teens receiving intervention
outcomes (speech production, language ability, intelligibility, (e.g., older than 14 years old). Note, however, that the re-
and PROs) in a similar manner, we found that we could only lationship between age and beneficial outcome could be
meta-analytically analyze data on speech production. Thus, biased by (a) the fact that our evaluation of clinical im-
large parts of the Results section and our inferences concern provement considered age at intervention, (b) a greater
only this outcome. risk of bias in studying younger children who receive long
intervention durations (see the Limitations section below),
Speech Production and (c) fewer studies included teens, and these studies
differed from the other studies in other important de-
We found 34 eligible studies on SLT for individuals tails. For example, two studies with the oldest individ-
born with CLP. The studies were heterogeneous with regard uals (Alighieri et al., 2019; Luyten et al., 2016) used a
to intervention details, demographics of the studied individ- relatively low intervention dosage (six 1-hr sessions over
uals, specific outcome measures, and statistical reporting. We 3 days), and the intervention was conducted in the pa-
decided that transformations of the various reported effect tients’ secondary language. In other studies with older
sizes to a standardized mean difference scale would be impos- individuals (Dobbelsteyn et al., 2014; Makarabhirom
sible to interpret in relation to clinical relevance. Instead, we et al., 2015; Prathanee, 2011), speech assistants or par-
evaluated IPD from 19 studies regarding improvement during ents were the persons who delivered most of the SLT. In
treatment and speech production after SLT based on clinical comparison, many of the studies with younger individ-
considerations and meta-analytically summarized these re- uals (Pamplona & Ysunza, 2018; Pamplona et al., 2014,
sults. We obtained only aggregated data in 12 studies (see 2017) used intensive and relatively high-dosage therapies
Supplemental Material S7), and although the authors inter- delivered by speech-language pathologists. Still, it seems
preted their results as being supportive of SLT benefiting pa- quite reasonable that younger individuals are more
tients to some degree, we were not able to infer how many (if likely to benefit from SLT (Peterson-Falzone et al.,
any) individuals benefited to a clinically relevant degree in 2010).
those studies.
Our main conclusion from the quantitative synthesis Speech Production After SLT
of the IPD is that SLT does benefit the speech production
of many individuals born with CLP. We do, however, rate SLT should not only improve the speech of patients
the overall quality of evidence for this conclusion to be but also bring their speech production up to the level of their
low due to (a) serious risks of bias in the studies, (b) im- peers. Less positive, then, is our estimate that, on average,
precision in many of the studies, and (c) serious inconsis- around 10%–34% (95% CI) of individuals are predicted to
tency in the implementation of the therapies between the have speech production on a level with peers after SLT. To
included studies. understand this somewhat negative result, we first want to
note that none of the studied interventions were designed to
Clinically Relevant Improvement in Speech produce satisfactory speech production but were, rather,
Production planned to end in a specified time frame and with a predeter-
mined intervention dosage. Thus, it is not unreasonable that
Because the between-studies heterogeneity was sub- the reviewed studies underestimate the proportion of individ-
stantial, we cannot deliver a definitive estimate of the ben- uals who would reach peer-level speech after SLT if the dos-
efit of SLT, but we have provided some reasonable limits age of therapy were to be increased. However, this is far
in the intervention effect. Based on our meta-analysis, from demonstrated: In the Scandcleft Project, many of the
even in cases in which SLT is estimated to be the least children who had the largest amount of speech therapy visits

were those with persistent speech problems (Persson et al., We do not, however, want to devalue the great im-
2020; Willadsen et al., 2017). pact that many of the interventions in the reviewed studies
The prediction interval for the proportion of individ- have had on the quality of life of many participants, espe-
uals who have speech production on a level with peers cially in areas where SLT or cleft-related interventions are
was very large (0%–71%), indicating that the intervention otherwise rare (e.g., Lindeborg et al., 2020; Luyten et al.,
effect was very inconsistent between studies. On a study 2016; Prathanee, 2011). We only would prefer a scenario
level, intervention duration was able to explain some of where we have more systematized data on these effects.
the dispersion in speech production following SLT (see
Figure 6). This is reasonable both because (a) longer inter- Limitations
vention duration should be able to produce better out-
comes (but see the Discussion section above) and (b) lon- The main aspect of this project was our evaluations
ger intervention durations were related to a greater risk of of improvement during treatment and outcome measures
bias from maturation (see the Limitations section). On an immediately after SLT on IPD, and the certainty of our
individual level, type of cleft was somewhat related to results is totally founded on these evaluations. We based
speech production after SLT: Individuals being born with these evaluations on the reported data in the included
cleft palate only, as compared to unilateral or bilateral studies. This is necessarily a flawed approach. As re-
CLP, perhaps have a slightly better prognosis (see viewers of published studies, we have no contact with the
Table 4). We must point out, however, that this result is individuals enrolled in the study, little insight into the par-
based on very few individuals with cleft palate (n = 35) ticulars of the intervention, and imperfect information on
and therefore is far from certain. the actual outcome instruments. Furthermore, we could
We were thus not able to fully explain the inconsis- not operationalize any formal criteria for “improvement
tency in intervention outcomes between studies, but this to a clinically relevant degree” or “outcome measures at
should not be too surprising. The between-studies hetero- peer level” across studies, or indeed within some studies.
geneity regarding context, intervention details, and patient Thus, we believe our IPD evaluations should be seen as a
groups was very large. For example, in some studies, individ- “proof of concept,” rather than as a “gold standard,” and
uals were specifically included because of their severe articu- as highlighting the need for standardized ways to evaluate
latory deviations (Pamplona & Ysunza, 2018; Pamplona improvement on an individual level. We encourage all sec-
et al., 2014, 2017) and would accordingly a priori be less ond opinions on the set of IPD we analyzed here (which
likely to reach peer-level speech production than patients in can be found as Supplemental Material S5) and invite any
other studies. Such study idiosyncrasies were too particular discussion on this important topic. A stronger methodol-
for us to be able to adequately model. Thus, because of the ogy in the future, clearly, would be if primary researchers
large heterogeneity in the reviewed studies, we do not believe evaluated which, if any, of their participants improved to a
that we have produced a stable estimate of the probability to clinically relevant degree and could do so based on standard-
reach peer-level speech production after SLT but have at ized criteria (see the Recommendations for Future Research
least summarized the information in the literature. section). Nevertheless, in lieu of this, our evaluations, we be-
lieve, have allowed us to distill that the published literature
Other Outcomes demonstrates that SLT and further work on evaluating SLT
in this patient group are meaningful endeavors.
We found very little information in the published litera- We must acknowledge that the results in many of
ture on the effects of SLT on language ability, intelligibility, the primary studies included in the quantitative synthesis
and PROs. The take-home message from Table 3, we believe, were at a serious risk of bias (see Table 2). In our individ-
is that the published literature, so far, has very low evidential ual evaluations, we could not, nor did we try to, establish
value regarding these outcomes. whether any clinically relevant improvement during ther-
This is a problematic situation. First, the end goal of apy or peer-level outcome measures following SLT were
SLT is to increase the patient’s ability to communicate and caused by the SLT. For example, patients may have ma-
participate positively in real-world settings (Havstam & tured spontaneously over long intervention durations
Lohmander, 2011). Ecologically valid measures of intelligibil- (Lohmander et al., 2006). We have only evaluated the
ity, attitudes toward communication, and communicative par- magnitude of improvement that occurred during treatment
ticipation are necessary to evaluate whether SLT leads to and the outcome measures at a particular time point after
these effects. Second, from a methodological perspective, eco- SLT. Thus, we must acknowledge that the benefits of
logically valid measures of intelligibility in everyday settings SLT, especially for younger age groups, may be overesti-
are necessary to validate the different measures of speech pro- mated in this synthesis. However, it is not clear exactly
duction that are used in clinical research today (Neumann & what study design could be implemented in this research
Romonath, 2012). context to control for this confound as the intervention

duration can be much longer than what is reasonable for a may perfectly fit any particular study setting. One suggested
waitlist control group to wait for treatment, for example, standard outcome set is the ICHOM Standard Set for Cleft
and possibly biased estimates may be the only estimates we Lip & Palate, which contains measures of speech production,
can hope for (Sterne et al., 2016). intelligibility, and PRO (Allori et al., 2017; https://www.
In most of the primary studies, we also judged the ichom.org/portfolio/cleft-lip-palate/). In this set, articulation is
risk of bias to be serious because assessors were not measured via the modified PCC based on single words
blinded regarding the patients’ treatment status. This is (Klintö et al., 2011; Shriberg et al., 1997), velopharyngeal
something that can and needs to be addressed in future competence is measured with the Velopharyngeal Compe-
studies. tence Scale (e.g., Lohmander, Hagberg, et al., 2017;
Lohmander et al., 2009), intelligibility is measured via the In-
Recommendations for Future Research telligibility in Context Scale (McLeod et al., 2012), and com-
municative attitude is measured via the Cleft Q Speech Dis-
Our primary recommendation for future intervention tress and Speech Function Scales (Klassen et al., 2018;
studies is that primary researchers use their expert and Stiernman et al., 2021).
clinical insight to evaluate which, if any, of their patients
benefitted to a clinically relevant degree from the studied
intervention. We cannot rely solely on “context blind” statis- Conclusions
tical approaches in evaluating intervention outcomes (Angst
et al., 2017; McShane et al., 2019; Wasserstein et al., 2019). Based on our clinical evaluations on individual in-
As we noted above, what pre–post changes should tervention outcomes, the peer-reviewed studies included in
constitute clinically relevant benefit is a complex issue— this meta-analysis indicate that SLT benefits the speech
that should take individual factors, intervention details, production of many individuals born with CLP. Especially
and PROs into account—but it is an issue that clinical re- young children (e.g., below 6 years of age) are likely to
search on SLT must confront if we are to adequately eval- benefit from SLT to a clinically relevant degree. It is less
uate SLT. Several approaches to delineate clinically rele- certain, from the published literature, what proportion of
vant change in other medical fields have been proposed individuals can reach speech production on a level with
and discussed and could form baselines for discussions in peers after SLT. The imprecision in the estimates and
SLT research (Angst et al., 2017; Crosby et al., 2003; overall quality of evidence in the literature should encourage
Guyatt et al., 2002; Jayadevappa et al., 2017; Revicki more research on this important topic.
et al., 2008). Although the reliability of the outcome mea-
sures must be considered, we hope that discussions on this
important topic will not overly rely on statistical defini-
tions of benefit, for example, minimal differences based
Acknowledgments
on measurement error or statistical change compared to
We thank the Karolinska Institute University Library
reference data (Jacobson & Truax, 1991; Weir, 2005). In-
search consultation group and Sabina Gillsund, specifically,
stead, our preliminary recommendation would be that
for formulating and conducting our literature search. We
clinically relevant benefit should be context sensitive to a
also want to thank all primary researchers who were helpful
particular patient group and established in relation to pa-
in our correspondence.
tient reports, where possible, supplemented by caregiver or
clinician insight, where reasonable (Angst et al., 2017;
Guyatt et al., 2002). We also believe that absolute out-
come levels after SLT should also be reported and related References
to reference standards to note which, if any, patients
reached peer levels. Albery, L., & Enderby, P. (1984). Intensive speech therapy for cleft
Second, we should strive to make intervention stud- palate children. British Journal of Disorders of Communication,
ies on SLT less heterogeneous and to evaluate the global 19(2), 115–124. https://doi.org/10.3109/13682828409007182
Alighieri, C., Bettens, K., Bruneel, L., Sseremba, D., Musasizi, D.,
benefits of SLT, not only related to speech production but Ojok, I., & Van Lierde, K. (2020). Comparison of motor-
also regarding intelligibility and communicative participation phonetic versus phonetic-phonological speech therapy ap-
in everyday settings. A standard outcome set to measure proaches in patients with a cleft (lip and) palate: A study in
speech production, intelligibility, and PROs and standardized Uganda. International Journal of Pediatric Otorhinolaryngol-
criteria for evaluating these outcomes are necessary for future ogy, 131, 109849. https://doi.org/10.1016/j.ijporl.2019.109849
Alighieri, C., Bettens, K., Bruneel, L., Vandormael, C., Musasizi, D.,
systematic reviews and meta-analyses of SLT. The ability to Ojok, I., D’haeseleer, E., & Van Lierde, K. (2019). Intensive
stringently compare and systematize intervention outcomes speech therapy in Ugandan patients with cleft (lip and) palate: A
across studies outweighs the drawback that no standard set pilot-study assessing long-term effectiveness. International

Journal of Pediatric Otorhinolaryngology, 123, 156–167. https:// Journal of Pediatric Otorhinolaryngology, 86, 104–113. https://
doi.org/10.1016/j.ijporl.2019.05.007 doi.org/10.1016/j.ijporl.2016.04.025
Allen, M. M. (2013). Intervention efficacy and intensity for children Dobbelsteyn, C., Bird, E. K.-R., Parker, J., Griffiths, C., Budden,
with speech sound disorder. Journal of Speech, Language, and A., Flood, K., & Stilson, A. (2014). Effectiveness of the correc-
Hearing Research, 56(3), 865–877. https://doi.org/10.1044/1092- tive babbling speech treatment program for children with a
4388(2012/11-0076) history of cleft palate or velopharyngeal dysfunction. The
Allori, A. C., Kelley, T., Meara, J. G., Albert, A., Bonanthaya, K., Cleft Palate–Craniofacial Journal, 51(2), 129–144. https://doi.
Chapman, K., Cunningham, M., Daskalogiannakis, J., De Gier, org/10.1597/12-188
H., Heggie, A. A., Hernandez, C., Jackson, O., Jones, Y., Dodd, B., Holm, A., Hua, Z., & Crosbie, S. (2003). Phonological
Kangesu, L., Koudstaal, M. J., Kuchhal, R., Lohmander, A., Long, development: A normative study of British English-speaking
R. E., Magee, L., . . . Wong, K. W. (2017). A standard set of out- children. Clinical Linguistics & Phonetics, 17(8), 617–643.
come measures for the comprehensive appraisal of cleft care. https://doi.org/10.1080/0269920031000111348
The Cleft Palate–Craniofacial Journal, 54(5), 540–554. https:// Greenland, S. (2019). Valid P-values behave exactly as they
doi.org/10.1597/15-292 should: Some misleading criticisms of P-values and their reso-
Amrhein, V., Trafimow, D., & Greenland, S. (2019). Inferential lution with S-values. The American Statistician, 73(Suppl. 1),
statistics as descriptive statistics: There is no replication crisis if we 106–114. https://doi.org/10.1080/00031305.2018.1529625
don’t expect replication. The American Statistician, 73(Suppl. 1), Guyatt, G. H., Osoba, D., Wu, A. W., Wyrwich, K. W., Norman,
262–270. https://doi.org/10.1080/00031305.2018.1543137 G. R., & Clinical Significance Consensus Meeting Group.
Angst, F., Aeschlimann, A., & Angst, J. (2017). The minimal clin- (2002). Methods to explain the clinical significance of health
ically important difference raised the significance of outcome status measures. Mayo Clinic Proceedings, 77(4), 371–383.
effects above the statistical level, with methodological implica- https://doi.org/10.4065/77.4.371
tions for future studies. Journal of Clinical Epidemiology, 82, Hanchanlert, Y., Pramakhatay, W., Pradubwong, S., & Prathanee, B.
128–136. https://doi.org/10.1016/j.jclinepi.2016.11.016 (2015). Speech correction for children with cleft lip and palate by
Bates, D., Mächler, M., Bolker, B., & Walker, S. (2015). Fitting networking of community-based care. Journal of the Medical
linear mixed-effects models using lme4. Journal of Statistical Association of Thailand, 98, S132–S139.
Software, 67(1), 1–48. https://doi.org/10.18637/jss.v067.i01 Harding, A., & Grunwell, P. (1998). Active versus passive cleft-
Bessell, A., Sell, D., Whiting, P., Roulstone, S., Albery, L., type speech characteristics. International Journal of Language
Persson, M., Verhoeven, A., Burke, M., & Ness, A. R. (2013). & Communication Disorders, 33(3), 329–352. https://doi.org/
Speech and language therapy interventions for children with cleft 10.1080/136828298247776
palate: A systematic review. The Cleft Palate–Craniofacial Jour- Hardin-Jones, M. A., & Chapman, K. L. (2008). The impact of
nal, 50(1), 1–17. https://doi.org/10.1597/11-202 early intervention on speech and lexical development for tod-
Blumenthal, C., & Lundeborg Hammarström, I. (2014). LINUS dlers with cleft palate: A retrospective look at outcome. Lan-
LINköpingsUnderSökningen Ett fonologiskt testmaterial för guage, Speech, and Hearing Services in Schools, 39(1), 89–96.
barn från 3 år [LINUS LINköpingsUnderSökningen A phonologi- https://doi.org/10.1044/0161-1461(2008/009)
cal test material for children from 3 years of age.] [Master thesis Hardin-Jones, M. A., Karnell, M. P., & Peterson-Falzone, S. J.
in Swedish, Linköping University Electronic Press]. http://www. (2009). Cleft palate speech (4th ed.). Mosby.
diva-portal.org/smash/get/diva2:737467/FULLTEXT01.pdf Havstam, C., & Lohmander, A. (2011). Communicative participation.
Borenstein, M. (2019). Common mistakes in meta-analysis and In Cleft palate speech: Assessment and intervention (pp. 305–315).
how to avoid them. Biostat. Wiley. https://doi.org/10.1002/9781118785065.ch17
Boutron, I., Page, M., Higgins, J., Altman, D., Lind, A., & Hutters, B., & Henningsson, G. (2004). Speech outcome following
Hróbjartsson, A. (2021). Chapter 7: Considering bias and con- treatment in cross-linguistic cleft palate studies: Methodologi-
flicts of interest among the included studies. In Cochrane cal implications. The Cleft Palate–Craniofacial Journal, 41(5),
handbook for systematic reviews of interventions Version 6.2 544–549. https://doi.org/10.1597/02-164.1
(updated February 2021). Cochrane. http://www.training. Ioannidis, J. P. A. (2016). Why most clinical research is not use-
cochrane.org/handbook ful. PLOS Medicine, 13(6), e1002049. https://doi.org/10.1371/
Britton, L., Albery, L., Bowden, M., Harding-Bell, A., Phippen, journal.pmed.1002049
G., & Sell, D. (2014). A cross-sectional cohort study of speech Jacobson, N. S., & Truax, P. (1991). Clinical significance: A sta-
in five-year-olds with cleft palate ± lip to support develop- tistical approach to defining meaningful change in psychother-
ment of national audit standards: Benchmarking speech stan- apy research. Journal of Consulting and Clinical Psychology,
dards in the United Kingdom. The Cleft Palate–Craniofacial 59(1), 12–19. https://doi.org/10.1037//0022-006x.59.1.12
Journal, 51(4), 431–451. https://doi.org/10.1597/13-121 Jayadevappa, R., Cook, R., & Chhatre, S. (2017). Minimal im-
Chapman, K. L. (1993). Phonologic processes in children with portant difference to infer changes in health-related quality of
cleft palate. The Cleft Palate–Craniofacial Journal, 30(1), 64–72. life—A systematic review. Journal of Clinical Epidemiology,
https://doi.org/10.1597/1545-1569_1993_030_0064_ppicwc_2. 89, 188–198. https://doi.org/10.1016/j.jclinepi.2017.06.009
3.co_2 Klassen, A. F., Riff, K. W. Y. W., Longmire, N. M., Albert, A.,
Crosby, R. D., Kolotkin, R. L., & Williams, G. R. (2003). Defin- Allen, G. C., Aydin, M. A., Baker, S. B., Cano, S. J., Chan,
ing clinically meaningful change in health-related quality of A. J., Courtemanche, D. J., Dreise, M. M., Goldstein, J. A.,
life. Journal of Clinical Epidemiology, 56(5), 395–407. https:// Goodacre, T. E. E., Harman, K. E., Munill, M., Mahony,
doi.org/10.1016/S0895-4356(03)00044-1 A. O., Aguilera, M. P., Peterson, P., Pusic, A. L., . . . Forrest,
Derakhshandeh, F., Nikmaram, M., Hosseinabad, H. H., C. R. (2018). Psychometric findings and normative values for
Memarzadeh, M., Taheri, M., Omrani, M., Jalaie, S., the CLEFT-Q based on 2434 children and young adult pa-
Bijankhan, M., & Sell, D. (2016). Speech characteristics after tients with cleft lip and/or palate from 12 countries. Canadian
articulation therapy in children with cleft palate and velopharyn- Medical Association Journal, 190(15), E455–E462. https://doi.
geal dysfunction—A single case experimental design. International org/10.1503/cmaj.170289

Klintö, K., Salameh, E.-K., Svensson, H., & Lohmander, A. McLeod, S., Harrison, L. J., & McCormack, J. (2012). The intel-
(2011). The impact of speech material on speech judgement in ligibility in context scale: Validity and reliability of a subjective
children with and without cleft palate. International Journal of rating measure. Journal of Speech, Language, and Hearing Re-
Language & Communication Disorders, 46(3), 348–360. https:// search, 55(2), 648–656. https://doi.org/10.1044/1092-4388(2011/
doi.org/10.3109/13682822.2010.507615 10-0130)
Kuehn, D. P., & Moller, K. T. (2000). Speech and language issues McShane, B. B., Gal, D., Gelman, A., Robert, C., & Tackett, J. L.
in the cleft palate population: The state of the art. The Cleft (2019). Abandon statistical significance. The American Statis-
Palate–Craniofacial Journal, 37(4), 1–35. https://doi.org/10. tician, 73(Supp_1), 235–245. https://doi.org/10.1080/00031305.
1597/1545-1569_2000_037_0348_saliit_2.3.co_2 2018.1527253
Lindeborg, M. M., Shakya, P., Pradhan, B., Rai, S. K., Gurung, Mossey, P. A., Little, J., Munger, R. G., Dixon, M. J., & Shaw,
K. B., Niroula, S., Rayamajhi, B., Chaudhary, H., Gaire, B., W. C. (2009). Cleft lip and palate. The Lancet, 374, 1773–1785.
Mahato, N., Rana, L., Rokaya, P., Shrestha, N., Shrestha, R., https://doi.org/10.1016/S0140-6736(09)60695-4
Tamang, J., Joshi, H. D., Gaha, P., Khorja, D. K., Nakarmi, Mossey, P. A., & Modell, B. (2012). Epidemiology of oral clefts
K. K., . . . Shaye, D. A. (2020). A task-shifted speech therapy 2012: An international perspective. Frontiers of Oral Biology,
program for cleft palate patients in rural Nepal: Evaluating 16, 1–18. https://doi.org/10.1159/000337464
impact and associated healthcare barriers. International Journal Neumann, S., & Romonath, R. (2012). Application of the Interna-
of Pediatric Otorhinolaryngology, 134, 110026. https://doi.org/ tional Classification of Functioning, Disability and Health–
10.1016/j.ijporl.2020.110026 Children and Youth Version (ICF-CY) to cleft lip and palate.
Lohmander, A., Friede, H., Elander, A., Persson, C., & Lilja, J. The Cleft Palate–Craniofacial Journal, 49(3), 325–346. https://
(2006). Speech development in patients with unilateral cleft lip doi.org/10.1597/10-145
and palate treated with different delays in closure of the hard Ouzzani, M., Hammady, H., Fedorowicz, Z., & Elmagarmid, A.
palate after early velar repair: A longitudinal perspective. (2016). Rayyan—A web and mobile app for systematic re-
Scandinavian Journal of Plastic and Reconstructive Surgery views. Systematic Reviews, 5(1), Article 210. https://doi.org/10.
and Hand Surgery, 40(5), 267–274. https://doi.org/10.1080/ 1186/s13643-016-0384-4
02844310600973621 Pamplona, C., Silis, S. C., Ysunza, P. A., & Morales, S. (2015).
Lohmander, A., Hagberg, E., Persson, C., Willadsen, E., Metacognitive strategies for enhancing language development
Lundeborg, I., Davies, J., Havstam, C., Boers, M., Kisling- in children with cleft palate. European Journal of Plastic Surgery,
Møller, M., Alaluusua, S., Aukner, R., Pedersen, N. H., 38(5), 377–384. https://doi.org/10.1007/s00238-015-1094-1
Turunen, L., & Nyberg, J. (2017). Validity of auditory percep- Pamplona, C., & Ysunza, A. (2000). Active participation of
tual assessment of velopharyngeal function and dysfunction—The mothers during speech therapy improved language develop-
VPC-Sum and the VPC-Rate. Clinical Linguistics & Phonetics, ment of children with cleft palate. Scandinavian Journal of
31(7–9), 589–597. https://doi.org/10.1080/02699206.2017.1302510 Plastic and Reconstructive Surgery and Hand Surgery, 34(3),
Lohmander, A., Lundeborg, I., & Persson, C. (2017). SVANTE— 231–236. https://doi.org/10.1080/02844310050159800
The Swedish Articulation and Nasality Test—Normative data Pamplona, C., & Ysunza, A. (2018). Deliberate practice: Preliminary
and a minimum standard set for cross-linguistic comparison. results of a useful strategy for correcting articulation in children
Clinical Linguistics & Phonetics, 31(2), 137–154. https://doi. with cleft palate. Journal of Craniofacial Surgery, 29(6),
org/10.1080/02699206.2016.1205666 1490–1494. https://doi.org/10.1097/SCS.0000000000004707
Lohmander, A., Persson, C., Willadsen, E., Lundeborg, I., Pamplona, C., Ysunza, A., & Morales, S. (2014). Strategies for
Alaluusua, S., Aukner, R., Bau, A., Boers, M., Bowden, M., treating compensatory articulation in patients with cleft pal-
Davies, J., Emborg, B., Havstam, C., Hayden, C., ate. International Journal of Biomedical Sciences, 10(1), 43–51.
Henningsson, G., Holmefjord, A., Hölttä, E., Kisling-Møller, M., Pamplona, C., Ysunza, A., & Morales, S. (2017). Audiovisual ma-
Kjøll, L., Lundberg, M., . . . Semb, G. (2017). Scandcleft rando- terials are effective for enhancing the correction of articula-
mised trials of primary surgery for unilateral cleft lip and pal- tion disorders in children with cleft palate. International Jour-
ate: 4. Speech outcomes in 5-year-olds—Velopharyngeal com- nal of Pediatric Otorhinolaryngology, 93, 17–23. https://doi.
petency and hypernasality. Journal of Plastic Surgery and org/10.1016/j.ijporl.2016.12.011
Hand Surgery, 51(1), 27–37. https://doi.org/10.1080/2000656X. Pamplona, C., Ysunza, A., Patiño, C., Ramírez, E., Drucker, M.,
2016.1254645 & Mazón, J. J. (2005). Speech summer camp for treating ar-
Lohmander, A., Willadsen, E., Persson, C., Henningsson, G., ticulation disorders in cleft palate patients. International Jour-
Bowden, M., & Hutters, B. (2009). Methodology for speech nal of Pediatric Otorhinolaryngology, 69(3), 351–359. https://
assessment in the scandcleft project—An international ran- doi.org/0.1016/j.ijporl.2004.10.012
domized clinical trial on palatal surgery: Experiences from a Persson, C., Pedersen, N.-H., Hayden, C., Bowden, M., Aukner,
pilot study. The Cleft Palate–Craniofacial Journal, 46(4), 347–362. R., Vindenes, H. A., Abyholm, F., Withby, D., Willadsen, E.,
https://doi.org/10.1597/08-039.1 & Lohmander, A. (2020). Scandcleft Project Trial 3: Compari-
Luyten, A., Bettens, K., D’Haeseleer, E., Hodges, A., Galiwango, G., son of speech outcomes in relation to sequence in 2-stage pal-
Vermeersch, H., & Van Lierde, K. (2016). Short-term effect of atal repair procedures in 5-year-olds with unilateral cleft lip
short, intensive speech therapy on articulation and resonance in and palate. The Cleft Palate–Craniofacial Journal, 57(3), 352–363.
Ugandan patients with cleft (lip and) palate. Journal of Commu- https://doi.org/10.1177/1055665619896637
nication Disorders, 61, 71–82. https://doi.org/10.1016/j.jcomdis. Peterson-Falzone, S. J., Hardin-Jones, M. A., & Karnell, M. P.
2016.03.006 (2010). Cleft palate speech (4th ed.). Mosby/Elsevier.
Makarabhirom, K., Prathanee, B., Suphawatjariyakul, R., & Prathanee, B. (2011). Cost effectiveness of speech camps for chil-
Yoodee, P. (2015). Speech therapy for children with cleft lip dren with cleft palate in Thailand. Journal of the Medical As-
and palate using a community-based speech therapy model sociation of Thailand, 94, S33–S39.
with speech assistants. Journal of the Medical Association of Prathanee, B., Makarabhirom, K., Pummnum, T., Seepuaham, C.,
Thailand, 98, S140–S150. Jaiyong, P., & Pradubwong, S. (2014). Khon Kaen: A

community-based speech therapy model for an area lacking in studies of interventions. BMJ, 355. https://doi.org/10.1136/bmj.
speech services for clefts. Southeast Asian Journal of Tropical i4919
Medicine and Public Health, 45(5), 1182–1195. Stiernman, M., Klintö, K., Persson, M., & Becker, M. (2021).
Prathanee, B., Pumnum, T., Yoodee, P., & Makarabhirom, K. Comparison of corresponding scores from the Cleft Hearing Ap-
(2020). Speech therapy model for patients with cleft palate in pearance and Speech Questionnaire (CHASQ) and CLEFT-Q in
Lao People’s Democratic Republic: Lack of speech services. In- Swedish patients with cleft lip and/or palate. The Cleft Palate–
ternational Journal of Pediatric Otorhinolaryngology, 138, Craniofacial Journal, 58(6), 736–745. https://doi.org/10.1177/
110366. https://doi.org/10.1016/j.ijporl.2020.110366 1055665620964124
Prins, D., & Bloomer, H. H. (1965). A word intelligibility ap- Stoel-Gammon, C., & Sosa, A. V. (2007). Phonological develop-
proach to the study of speech change in oral cleft patients. ment. In E. Hoff & M. Shatz (Eds.), Blackwell handbook of
The Cleft Palate Journal, 2, 357–368. language development (pp. 238–256). Blackwell. https://doi.
Pumnum, T., Kum-ud, W., & Prathanee, B. (2015). A networking of org/10.1002/9780470757833.ch12
community-based speech therapy: Borabue District, Maha Sweeney, T., Hegarty, F., Powell, K., Deasy, L., Regan, M. O., &
Sarakham. Journal of the Medical Association of Thailand, 98, Sell, D. (2020). Randomized controlled trial comparing Parent
S120–S127. Led Therapist Supervised Articulation Therapy (PLAT) with
R Core Team. (2013). R: A language and environment for statistical routine intervention for children with speech disorders associ-
computing. R Foundation for Statistical Computing. http://www. ated with cleft palate. International Journal of Language &
R-project.org/ Communication Disorders, 55(5), 639–660. https://doi.org/10.
Revicki, D., Hays, R. D., Cella, D., & Sloan, J. (2008). Recom- 1111/1460-6984.12542
mended methods for determining responsiveness and mini- Vallino-Napoli, L. D. (2011). Evaluation and evidence-based
mally important differences for patient-reported outcomes. practice. In A. Lohmander & S. Howard (Eds.), Cleft palate
Journal of Clinical Epidemiology, 61(2), 102–109. https://doi. speech—Assessment and intervention (pp. 317–358). Wiley-
org/10.1016/j.jclinepi.2007.03.012 Blackwell.
Roxburgh, Z., Cleland, J., & Scobbie, J. M. (2016). Multiple Van Demark, D. R., & Hardin, M. A. (1986). Effectiveness of in-
phonetically trained-listener comparisons of speech before and tensive articulation therapy for children with cleft palate. The
after articulatory intervention in two children with repaired Cleft Palate Journal, 23(3), 215–224.
submucous cleft palate. Clinical Linguistics & Phonetics, 30(3), Viechtbauer, W. (2010). Conducting meta-analyses in R with the
398–415. https://doi.org/10.3109/02699206.2015.1135477 metafor package. Journal of Statistical Software, 36(3), 1–48.
Ruscello, D., & Vallino, L. (2014). The application of motor https://doi.org/10.18637/jss.v036.i03
learning concepts to the treatment of children with compensatory Wang, N. (2018). How to conduct a meta-analysis of proportions
speech sound errors. SIG 5 Perspectives on Speech Science and Oro- in R: A comprehensive tutorial. John Jay College of Criminal
facial Disorders, 24(2), 39–47. https://doi.org/10.1044/ssod24.2.39 Justice. https://doi.org/10.13140/RG.2.2.27199.00161
Scherer, N. J., D’Antonio, L. L., & McGahey, H. (2008). Early Wasserstein, R. L., & Lazar, N. A. (2016). The ASA statement
intervention for speech impairment in children with cleft palate. on p-values: Context, process, and purpose. The American
The Cleft Palate–Craniofacial Journal, 45(1), 18–31. https://doi. Statistician, 70(2), 129–133. https://doi.org/10.1080/00031305.
org/10.1597/06-085.1 2016.1154108
Sell, D. (2005). Issues in perceptual speech analysis in cleft palate Wasserstein, R. L., Schirm, A. L., & Lazar, N. A. (2019). Moving to
and related disorders: A review. International Journal of Lan- a world beyond “p < 0.05.” The American Statistician, 73(Suppl. 1),
guage & Communication Disorders, 40(2), 103–121. https://doi. 1–19. https://doi.org/10.1080/00031305.2019.1583913
org/10.1080/13682820400016522 Weir, J. P. (2005). Quantifying test–retest reliability using the
Shriberg, L. D., Austin, D., Lewis, B. A., McSweeny, J. L., & intraclass correlation coefficient and the SEM. Journal of
Wilson, D. L. (1997). The percentage of consonants correct Strength and Conditioning Research, 19(1), 231–240. https://
(PCC) metric: Extensions and reliability data. Journal of Speech, doi.org/10.1519/15184.1
Language, and Hearing Research, 40(4), 708–722. https://doi.org/ Whitehill, T. L., Gotzke, C. L., & Hodge, M. (2011). Speech in-
10.1044/jslhr.4004.708 telligibility. In Cleft palate speech: Assessment and intervention
Sritacha, P., Pumnum, T., & Prathanee, B. (2016). Speech correc- (pp. 293–304). Wiley. https://doi.org/10.1002/9781118785065.ch16
tion for children with cleft lip and palate in community: Willadsen, E., Lohmander, A., Persson, C., Lundeborg, I.,
Kantharawichai networking. Journal of the Medical Associa- Alaluusua, S., Aukner, R., Bau, A., Boers, M., Bowden, M.,
tion of Thailand, 99, S1–S8. Davies, J., Emborg, B., Havstam, C., Hayden, C.,
Sterne, J. A. C., Hernán, M. A., Reeves, B. C., Savović, J., Henningsson, G., Holmefjord, A., Hölttä, E., Kisling-Møller,
Berkman, N. D., Viswanathan, M., Henry, D., Altman, D. G., M., Kjøll, L., Lundberg, M., . . . Semb, G. (2017). Scandcleft
Ansari, M. T., Boutron, I., Carpenter, J. R., Chan, A.-W., randomised trials of primary surgery for unilateral cleft lip and
Churchill, R., Deeks, J. J., Hróbjartsson, A., Kirkham, J., Jüni, P., palate: 5. Speech outcomes in 5-year-olds—Consonant profi-
Loke, Y. K., Pigott, T. D., . . . Higgins, J. P. T. (2016). ciency and errors. Journal of Plastic Surgery and Hand Surgery,
ROBINS-I: A tool for assessing risk of bias in non-randomised 51(1), 38–51. https://doi.org/10.1080/2000656X.2016.1254647


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A 20% 26% 6%-points Method

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Sand et al.: On the Benefits of Speech-Language Therapy 557

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Sand et al.: On the Benefits of Speech-Language Therapy 559

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Note. Y = yes; N = no.

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Improved during therapy At peer level after SLT

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Improved Peer level

Unilateral CLP 240 193 (80) 79% 52 (22) 17%

Note. CLP = cleft lip and/or palate.

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Figure 5. Illustration of the mixed-effects logistic regression model

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Sand et al.: On the Benefits of Speech-Language Therapy 569

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Sand et al.: On the Benefits of Speech-Language Therapy 573

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