Bangladesh Journal of Medical Science Vol. 17 No.

03 July’18

Original article
A perceptual evaluation of speech disorders in children with repaired unilateral cleft lip and palate in
Hospital UniversitiSains Malaysia
Alam MK1, Zulkipli AS 2, Percival SE3, Haque S4
Abstract
Aims and objective: The purpose of this cross sectional study was to determine the types and
severity of speech disorders in children with repaired unilateral cleft lip and palate (UCLP)
in Hospital UniversitiSains Malaysia (Hospital USM) and describe the inter- and intra-judge
reliability of perceptual evaluation of speech disorders using GOS.SP.PASS’98 and five-point
rating scale. Four children with repaired UCLP with ages ranging from 8 years old to 12 years
old were included in this study. Prior to data collection, participants were contacted through
telephone call from the list of patients with UCLP obtained Combined Cleft and Craniofacial
Deformity Clinic (Combined clinic) in Hospital USM, Kelantan from year 2013-2015 as well
as from clinical records from the database of the Record Unit of Hospital USM from year
2003-2015. Following strict inclusion criteria participant has been selected. After that, history
taking was first conducted with the participant’s parents, followed by collection of participant’s
speech sample and finally concluded with oral motor examination. Results: Fifty percent of
the speeh samples obtained from data collection were then duplicated for use in inter-rater and
intra-rater reliability investigations. Exact agreement and kappa values were used for reliability
measures. Seventy-five percent (3/4) participants exhibited speech disorders and 25% (1/4)
participant had no evidence of speech disorder. The type of speech errors that occurred the
most in the three participants were glottal articulations (39%), while the least speech errors
includedlateralizations/lateral articulations (2%), backing to velar (1%), pharyngeal articulations
(1%,) active nasal fricatives (1%) as well as nasal realizations of fricatives (1%). The severity
of speech disorder ranged from some occurrences to occurring always or almost alwaysin the
participants. Inter-judge reliability showed poor to slight agreement while intra-judge reliability
revealed almost perfect agreement. Conclusion: The results obtained from this study were more
or less similar to other studies conducted on the speech outcome of individuals with repaired
UCLP.It is important that referrals be made to speech-language pathologists (SLPs) to evaluate
the individuals’ speech outcomes even though surgical intervention has been provided to them
to ensure a comprehensive management for the individual.
Keywords: Unilateral cleft lip and palate; Speech disorder
Bangladesh Journal of Medical Science Vol. 17 No. 03 July’18. Page : 470-478
DOI: http://dx.doi.org/10.3329/bjms.v17i3.37003
Introduction our communication, which requires intact structures
Communication is made up of several components of lips, jaw, tongue, teeth and palate working
including hearing, receptive language, expressive in coordination with muscles of respiration and
language, speech, resonance, voice, and the social phonation2. The four substructures of speech consists
use of language most commonly referred to as of respiration, which is our breathing, phonation
“pragmatic skills” 1. Speech is the motor component of when sound is made by the vocal folds, articulation

1. Mohammad Khursheed Alam, Orthodontic Department, College of Dentistry, Al Jouf University, Sakaka, KSA.
2. Aimi Syahidah Zulkipli, Speech Pathology Program, School of Health Sciences, Universiti Sains
Malaysia, Kota Bharu, Kelantan, Malaysia.
3. Sandra Elizabeth Percival, Speech Pathology Program, School of Health Sciences, Universiti Sains
Malaysia, Kota Bharu, Kelantan, Malaysia.
4. Sanjida Haque Orthodontic Unit, School of Dental Sciences, Universiti Sains Malaysia, Kota Bharu,
Kelantan, Malaysia.
Correspondence to: Dr. Mohammad KhursheedAlam Associate Professor, Orthodontic Department, College of
Dentistry, Al Jouf University, Sakaka, KSA. Email: [email protected] and Aimi Syahidah Zulkipli, School of
Health Sciences, Universiti Sains Malaysia. Kubang Kerian 16150, Kota Bharu, Kelantan, Malaysia.
Email: [email protected]

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 Speech disorders in children with repaired unilateral cleft lip and palate.

which is the production of sounds using the lips, telephone to briefly explain about the research and
teeth, tongue and jaw movements and resonation or their willingness to participate. Time and date was
the quality of voice regulated by the integrity and then set to meet the participants.
the movement of the soft palate and surrounding The inclusion criterias of our study were -
structures.Among one of the populations that SLPs 1. Non-syndromic UCLP patient.
work with children with craniofacial anomalies, such 2. Individual aged 8-12 years.
as cleft lip and palate (CLP). 3. Lip surgery and palatoplasty had been performed.
CLP is one of the most common congenital 4. Speak in Bahasa Melayu
craniofacial anomalies that occurs at birth3, 4. It The exclusion criterias of our study were
affects about 1.5 per 1000 live births worldwide4). 1. Have language or developmental problems
Children with CLP do not represent a homogenous 2. Have history of hearing problems
population, and the signs and symptoms associated The participants were included in this study
with clefting depend on a variety of factors5,6,7,8. by convenience sampling, which is a form of
Common issues that children with a history of cleft nonprobability sampling. All the participants’
palate (CP) can encounter include problems related caregiver from the list were attempted to be contacted.
to feeding, dental and occlusal deviations, hearing Those who were able to be contacted, fitted the
and middle ear function, psychosocial development inclusion criteria and were willing to participate in
and in speech sound acquisition and language this study, were then recruited as participants for the
development9,10. Besides that, all of the components study. Participants were selected based on caregivers’
of communication can be affected by the presence ease in volunteering and their availability. This ease
of a cleft, particularly speech1). In individuals with was mainly due to the fact that the caregivers were
CLP, errors in speech production are noticed due provided liberty in determining the time and date
to the abnormalities in oronasal structure/function, to come in for data collection according to their
orofacial structure and growth, learned neuromotor convenience.
patterns during early infancy, and/or disturbed Sample size was not calculated for this study as the
psychosocial development11. Due to this, children sample size was taken based on those who were
with CLP often exhibit ‘cleft palate speech’ that able to participate in the research study. The total
includes atypical consonant productions, abnormal number of patients obtained from both sources was
nasal resonance, abnormal nasal airflow, altered 21 patients. Therefore, by only recruiting those who
laryngeal voice quality, and nasal or facial grimaces12. were able to participate in the research study, the
Since it is common for children born with CP or with sample size consisted of four participants. Table 1
CLP to have speech problems at some time in their describe the status of each patient from the two lists.
lives, the aim of this study to determine the types and Instruments
severity of speech disorders exhibited in children There are some instruments used during the testing
with repaired UCLP in Hospital UniversitiSains which is the perceptual speech assessment form,
Malaysia and to describe the inter-rater and intra- GOS.SP.ASS’ 98.The GOS.SP.ASS’ 98 form has
rater reliability of perceptual evaluation of speech a component for perceptual assessment of speech
disorders using GOS.SP.ASS’98 and five point rating disorders associated with CP and/or VPD under the
scale of the assessed speech parameters among SLPs. heading‘Cleft Type Characteristics’. Under Cleft
Methodology and Methods Type Characteristics (CTCs), it can be identified
This study was approved by the Ethical Committee through the transcriptions of the participants’ speech
of the Hospital Universiti Sains Malaysia (HUSM) samples. The number of the relevant characteristic/s
[USM/JEPeM/15100355]. as listed in the form is circled to show its presence
This study was designed including children with in the speech sample. CTCs can be categorized
repaired UCLP who attended Combined Cleft and into dentalization, lateralization/lateral articulation,
Craniofacial Deformity Clinic (Combined Clinic) as palatalization/palatal, double articulation, backing
patients in Hospital USM, Kelantan from year 2013- to velar, backing to uvular, pharyngeal articulation,
2015 and from clinical records from the database of glottal articulation, active nasal fricatives, weak/
the Record Unit of Hospital USM from year 2003- nasalized consonants, nasal realizations of fricatives,
2015. Only the children who fitted the age range nasal realizations of plosives, absent pressure
of 8-12 years old were taken into consideration. consonants and finally gliding of fricatives/affricates.
Patients’ parents were then contacted through Space is provided in the form of dotted lines adjacent

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 Alam MK, Zulkipli AS , Percival SE, Haque S

to the process for the transcription of speech, where speech sample recordings. This was done by the
specific examples, whole word transcription or researcher listening to all of the audio recordings of
atypical consonant cluster production may be entered speech samples and transcribing each of the speech
to provide extra information and provide a clearer samples accordingly using International Phonetic
view. Alphabet (IPA) and extra IPA (extIPA) for disordered
Another instrument used is a five-point rating scale speech (Appendix I,page 83). After transcriptions for
with description of the scale values for the assessed each of the speech samples were done (Appendix
speech parameters (Appendix C, page 77) used to J, page84) the researcher listened to the speech
determine the severity of the CTCs present. This scale samples again and classified thethe types of CTCs
ranges from 0 (no occurrence), 1 (single occurrence), based on the GOS.SP.ASS ’98 form and determined
2 (some occurrence), 3 (frequently occurring) and 5 overall severity of the speech disorder according to
(occurring always or almost always). The scales are the five-point rating scale for the assessed speech
similar to the five degrees used in Clinical Standards parameterswhile referring to the transcriptions at
Advisory Group study13 and to the four-point scale the same time for each of the participants’ speech
used by Van Lierdeetal14. These scales are used samples.
nationwide at Swedish cleft palate centers15. Once perceptual analysis was done, the total number
Besides that, validated speech stimuli in of CTCs that occurred in each of the participants’s
BahasaMelayu (Appendix D, page 78), in which speech sample was calculated. For each of the
permission was already obtained from its’ developer participant’s speech sample, the total number of
for usage in this study to obtain the speech sample CTCs was obtained by calculating the number of
of the participants (Appendix E, page 79) was also times a CTC occurred throughout the speech sample.
used, along with a history taking(Appendix F, page Percentage of each CTC for each participant was
80)and oral motor examination form (Appendix then calculated by taking the number of a particular
G, page81) and audio recorder (SONY Stereo IC CTC divided by the total number of CTCs present
Recorder, ICD-UX543F, Appendix H, page 82) to within the speech sample and multiplied by 100.
record participant’s speech sample. Overall number of CTCs from all participants was
Method then calculated by adding all of the CTCs from
On the day of data collection, history taking was each participant. Each CTC was added accordingly
first conducted to obtain information regarding the into its own classification. Total percentage of each
antenatal history, birth history, postnatal history, CTC was calculated by taking the total number of
medical history, hearing status, language development that particular CTC and divided by the total number
as well as involvement of other professionals of of CTCs that occurred throughout all of the speech
the participants.After that participants’ speech samples and multiplied by 100.
samples were then collected. Conversational speech Statistical analysis
was collected through building rapport with the The data was analyzed statistically using IBM
participants. Participants were then required to read Statistical Package for Social Sciences (SPSS)
a series of speech stimuli in BahasaMelayu and Version 22.0. The analysis of the data regarding
their speech samples were recorded using an audio the types and severity of speech disorders were
recorder and used for data analysis for the study. interpreted in the form of tables. The intra- and inter-
Once the participant’s speech sample was obtained, examiner agreements were analyzed with the kappa
oral motor examination was performed to examine statistics. According to Landis & Koch16, the kappa
the participant’s oral structures and function, so as values of the intra- and inter-examiner agreements
to identify or rule out the structural or functional were interpreted.
factors that relate to a communicative disorder. Results
A group of five listeners participated in this study. Demographic Data
Three of the listeners were certified SLPs while the Table 2 shows demographic data of each participant
other two were the researchers. The three certified in this research study including their age, gender,
SLPsare with more than five years ofexperience type of cleft, age of lip repair and palatoplasty, age at
in the field of CP speech. The researchers were which started speech therapy and duration of speech
trained through clinical exposure to cleft type therapy.
speech disorders from January 2016 till April 2016. Perceptual Evaluation
A perceptual analysis was performed from the Perceptual evaluation was carried out by first

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 Speech disorders in children with repaired unilateral cleft lip and palate.

determining the types of CTCs exhibited by the that may affect development of normal articulation
participants. Table 3 shows the CTCs as exhibited by pattern could be attributed to age of the repair or
the participants. surgery. Previous studies suggest that children who
Besides determining the different CTCs exhibited receive early palatal repair demonstrate better overall
by each participant, a rating of the severity of their speech than their peers who receive surgery at a later
overall speech disorder was given using the five- age20 and those children who receive late palatal
point rating scale of the assessed speech parameters. surgery are at greater risk for developing atypical
Therefore, by referring to the list of CTCs from articulation patterns21-25.
GOS.SP.ASS ’98 and the five-point rating scale, Clinical outcomes of primary CP repair, such as
the different CTCs and overall severity of speech articulatory deficits are related to several factors,
disorders of each participants’ are summarized in the including cleft type, the extent of innate clefting,
Table 4. surgical repair techniques, expertise of the operating
Intra- and Inter-examiner agreements surgeon, preoperative orthopedics, and timing
The exact agreement for all ratings was 100% and of primary palatal repair26-33. Thus, a possible
the measure of agreement for Kappa for all ratings explanation for the problem above could have been
was 1.00. This Kappa value suggested almost perfect one of the factors or a combination of factors that
agreement9). Table 5 shows the intra-rater reliability resulted in the presence of speech disorders even after
for all speech samples. appropriate surgical correction has been provided to
Inter-rater reliability was determined between three the participants of this study.
raters who were SLPs in Hospital USM for 50% of Glottal articulations were discovered to be the most
the speech samples from all four participants using number of CTCs produced by the participants of this
Kappa. The Kappa was -0.33 to 0.00 calculated study. Authors have hypothesized that glottal and
from the exact agreement scores which indicate poor pharyngeal articulations could develop because a
to slight agreement9). Table 5 shows the inter-rater child compensates by valving the airstream at a point
reliability between all three raters. in his/her vocal tract inferior to the velopharyngeal
Discussion port so as to normalize pressure34, 35. According
Previous studies revealed speech disorder is one of to D’Antonio& Scherer1), the most common and
the common problems associated with CLP patients. distinctive of the compensatory articulation errors
The outcome of our study is consistent with most that frequently occur in the speech of individuals
previous studies investigating speech outcomes in with CP is the glottal stop, and these misarticulations
children with CLP. According to Normasturaet al2), are often difficult to eradicate even after therapy,
there is a significant association between CLP and whichsupports the findings of this study where
speech abnormalities. The risk of CLP children for the most number of CTCs observed was glottal
having speech abnormalities is 174.5 times more articulations. Compensatory articulation errors
compared to non-cleft children, hence it is one of the such as glottal or pharyngeal articulations occur
unavoidable complication in the cleft child. Hortis- when a child with CP attempts to compensate for
Dzierzbickaetal17 investigated the speech outcomes velopharyngeal inadequacy, thus they are learned
of complete UCLP after one-stage lip and palate behaviours that typically do not resolve even after
repair in the first year of life and found articulation secondary management to repair the CP and when a
development and incidence of compensatory potentially adequate velopharyngeal mechanism has
articulations in the sample were mostly satisfactory. been achieved36. Most of the time speech therapy
In a study by Albustanjietal18 found that out of 80 will be needed to correct these compensatory
participants with CLP, 21 participants had normal articulations.
articulation and resonance, 59 of participants (74%) On the other hand, weak consonants, nasal realizations
showed speech abnormalities. Timmons etal19 found of plosives and nasal realizations of fricatives are
final speech outcomes were similar for CP and CLP passive consequence of velopharyngeal dysfunction,
patients, whereby CTCs were noted in 11 (41%) CP or due to presence of fistulae. These reduces the
and nine (53%) CLP patients. child’s ability in achieving and/or sustaining intraoral
There could be a number of reasons why speech pressure37 causing certain consonants to sound weak
disorders still occur even after the appropriate or nasalized. However during oral-motor examination
surgical management or speech therapy have been of all four participants in this study, no evidence of
conducted with the UCLP children. A possible factor fistula was found in any of the participants. Therefore,

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 Alam MK, Zulkipli AS , Percival SE, Haque S

it could be assumed that these passive CTCs are due In summary, perceptual speech assessment
to VPD such as velopharyngeal insufficiency (lack is central to the evaluation of speech outcomes
of velum tissue) or velopharyngeal incompetence associated with CP and VPD39. The primary purpose
(lack of proper movement of the lateral and posterior of the perceptual speech evaluation is to determine
pharyngeal walls as well as the velum). Both of these the characteristics and cause of the speech problem
will cause impaired velopharyngeal closure. so that appropriate treatment recommendations can
In terms of severity of the overall speech disorders, be made. It is critical to make the right diagnosis
results of this study indicated that three of the when there has been a history of CP or if there are
participants (75%) who exhibited CTCs had a characteristics of VPD because the diagnosis will
severity rating ranging from some occurrences to determine whether appropriate treatment includes
occurring always or almost always. Out of those three surgical intervention, speech therapy, or both40.
participants who exhibited CTCs, two participants Information such as the types and severity of speech
showed a severity of some occurrences while one disorders gathered through perceptual assessment
participant had a severity of occurring always or will be useful in acting as a baseline for SLPs to
almost always. This result is almost similar to a further manage a CLP case, whether in determining
study by Nyberg etal38 where in general, most of the which therapy techniques are suitable to be applied
investigated children had minor speech problems. in rehabilitating the specific CTCs as exhibited by
Similar speech outcomes were also reported by the child or in terms of frequency of speech therapy
Hortis-Dzierzbickaet al17) required.
For this study, a factor that could be attributed to the Conclusion
differences in severity rating of speech disorder is Based on the results, following conclusion can be
because of the different duration of speech therapy drawn
for each of the participants. Generally, better speech 1. Seventy five percent (75%) (3/4) participants
outcome is associated with a more frequent and exhibited speech disorders, while 25% (1/4)
longer duration of undergoing speech therapy. participant had no speech disorder
However, in this study one of the participants who 2. In terms of types of speech errors exhibited by
had undergone speech therapy for four years still children with repaired UCLP the most errors
exhibited occurring always or almost always CTCs consisted of glottal articulations, while the
while another participant who had been undergoing least were lateralization/lateral articulations,
speech therapy for one month only demonstrated double articulation, backing to velar, pharyngeal
some occurrences of CTCs. However, it should be articulation, active nasal fricatives and finally
noted that although there is a difference between the nasal realizations of fricatives. This indicated
overall duration of undergoing speech therapy and that glottal articulations are the most common
the severity rating of the participant, the frequency CTC in the CP population.
in which participants went for speech therapy was 3. Severity of the speech disorders exhibited by
not known. Therefore it is possible for a participant children with repaired UCLP, it ranged from
who frequently went for speech therapy in a some occurrences to occurring always or almost
shorter duration to have only some occurrences of always, which may be due to the different
CTCs when compared to a participant who did not duration and frequency of speech therapy
frequently attend speech therapy in a longer duration attended by each of the participants.
to exhibit occurring always or almost always CTCs. Achknowledgements
Therefore, motivation and commitment from both the None declared
parents and children is critical in ensuring successful Conflict of interest
speech therapy2). The authors have declared that no COI exists.

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 Speech disorders in children with repaired unilateral cleft lip and palate.

Table 1. Number of patients from Combined Cleft and Craniofacial Deformity Clinic HUSM (2013-2015)
and from Record Unit HUSM (2003-2015) according to status

Number of patients from Combined Number of patients from

Status Cleft and Craniofacial Deformity Record Unit HUSM (2003-
Clinic HUSM (2013-2015) 2015)
Voicemail 3 3
Could not be contacted 2 1
Refused to participate 1 2
Could be reached but did not fit the
2 0
inclusion criteria
Agreed to participate 3 1
Live in other states 0 3
Total 11 10

Table 2. Demographic data of participants

Age
(years)
Age Duration
Age Side of Age (months) of at which
Participant Gender (months) of of Speech
(years) UCLP Palatoplasty started
Lip Repair Therapy
Speech
Therapy
1 9 Female Left 3 7 3 6 years
2 12 Male Left 3 3 6 4 years
3 9 Female Right 3 11 4 1 month
4 12 Male Midline 6 12 5 1 year

Table 3. Cleft type characteristics exhibited by participants

Cleft type characteristics (CTCs) Number of participants
Lateralization/Lateral articulation 1
Double articulation 1
Backing to velar 2
Pharyngeal articulation 1
Glottal articulation 3
Active nasal fricatives 1
Weak/nasalized consonants 3
Nasal realizations of fricatives 1
Nasal realizations of plosives 2
Absent pressure consonants 2
Palatalization/Palatal 0
Backing to uvular 0
Gliding of fricatives/affricates 0

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 Alam MK, Zulkipli AS , Percival SE, Haque S

Table 4. Cleft type characteristics and overall severity of speech disorder of each participant
Overall severity of speech
Participant Cleft type characteristics
disorder
Glottal articulation
Grade 2
1 Weak consonants
(some occurrences)
Absent pressure consonants
Lateralization/Lateral articulation
Double articulation
Backing to velar
Pharyngeal articulation
Grade 4
Glottal articulation
2 (Occurring always or almost
Active nasal fricatives
always)
Weak/nasalized consonants
Nasal realizations of fricatives
Nasal realizations of plosives
Absent pressure consonant
Glottal articulation Grade 2
3
Weak consonants (Some occurrences)
Grade 0
4 None
(No occurrence)

Table 5. Kappa values of ratings for 4 speech samples

Intra-examiner agreements
Speech Samples Exact Agreement (%) Kappa value
1 100 1.00
2 100 1.00
3 100 1.00
4 100 1.00
Inter-examiner agreements
SLP Exact Agreement (%) Weighted Kappa
1 and 2 50 -0.33
2 and 3 50 0.00
1 and 3 50 0.00

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 Speech disorders in children with repaired unilateral cleft lip and palate.

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