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Thongprayoon et al.
Sunisa ongprayoon, M.D.*, Kanokwan Liadprathom, B.Sc.**, Apirag Chuangsuwanich, M.D., FRCST*,
Mark H. Moore, MBChB, FRACS***, Sarut Chaisrisawadisuk, M.D., FRCST*,***
*Division of Plastic Surgery, Department of Surgery, **Division of Speech erapy, Department of Rehabilitation Medicine, Faculty of Medicine
Siriraj Hospital, Mahidol University, Bangkok 10700, ailand., ***Cle and Craniofacial South Australia, Women’s and Children’s Hospital, North
Adelaide, South Australia, Australia.
Speech Outcome Analysis after Primary Cleft
Palate Repair: Interim Siriraj Hospital Audit
ABSTRACT
Objective: To evaluate the speech outcomes aer primary cle palate repair in a single tertiary medical institution
of ailand.
Materials and Methods: A prospective cohort study was performed. Patients who had cle palate with/without cle
lip and underwent primary cle palate repair were included. Speech assessment was performed using the Pittsburgh
weighted speech score (PWSS) by a speech-language pathologist.
Results: Forty patients (21 males and 19 females) who underwent primary cle palate repair at Siriraj Hospital
were included. e median age at the time of speech evaluation was 7 years. e median age at primary cle palate
surgery was 12 months. e predominant cle palate type was Veau 3 (47.5%). Oronasal stula occurred 40%.
Two-ap palatoplasty and intravelar veloplasty were the most common procedures. Median PWSS was 7, in which
the competence velopharyngeal mechanism was found 5%, borderline competence 10%, borderline incompetence
32.5%, and incompetence velopharyngeal mechanism 52.5%. Among the velopharyngeal incompetence group,
articulation disorder was the most common disorder with median score of 3. Besides, the median scores for nasality,
nasal emission, phonation, and facial grimace disorder were 1, 2, 0 and 0, respectively. ere was no statistically
signicant association between velopharyngeal incompetence and cle types, age at primary surgery, type of operation,
the width of cle palate and prevalence of postoperative oronasal stula or otitis media eusion.
Conclusion: Velopharyngeal incompetence has been commonly identied aer cle palate repair in our institute.
e articulation disorder is the most common characteristic.
Keywords: Cle palate; speech outcome; velopharyngeal insuciency; craniofacial abnormalities (Siriraj Med J
2021; 73: 744-751)
Corresponding author: Sarut Chaisrisawadisuk
E-mail: sarut.cha@mahidol.ac.th; drsarut@gmail.com
Received 28 May 2021 Revised 3 May 2021 Accepted 12 September 2021
ORCID ID: https://orcid.org/0000-0001-6040-0784
http://dx.doi.org/10.33192/Smj.2021.96
INTRODUCTION
Cle lip and/or cle palate (CLP) are common
craniofacial anomalies in ailand. Chuangsuwanich
et al., in 1998, reported that the incidence of patients
with CLP was around 1 in 600 per live births at Siriraj
Hospital.
1
Recently, Chowchuen et al. also reported a
prevalence of CLP of 1.93 per 1000 live births in the
northeast region of ailand.
2
Patients with CLP have
cosmetic and functional concerns, which typically require
comprehensive multidisciplinary team (MDT) care
and long-term follow-up, starting with cle lip repair
at around 3-6 months and cle palate repair between
6-18 months of age.
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Cle palate (CP) repair aims to create an anatomically
intact and functional palate to improve feeding, achieve
normal speech, and minimize maxillary growth restriction
and middle ear infection.
3
In order to produce normal
speech, a child must have velopharyngeal competence,
dened as the ability to completely close the velopharyngeal
sphincter, which separates the oro- and nasopharynx.
e absence of this ability is termed velopharyngeal
insuciency.
4
e primary eects of velopharyngeal
insuciency are nasal air escape and hypernasality. A
wide range of postoperative velopharyngeal insuciency
cases requiring secondary operation have been reported
(15%–50%).
4-5
e phonemic system in the ai language is dierent
from English. A ai syllable consists of an initial, a
vocalic nucleus, a nal, and a tone.
6
e ai language
is a tonal language with sentences typically comprising a
subject, verb, and object in order. e subject is usually not
obviously stated but contextually assumed, similar to the
case with the object. e verb has no declensions, tense,
or conjugations.
7
According to this, if cle palate patients
have a decreased intelligibility of speech expression or
are especially prone to articulation errors, it may disturb
their communication skills, resulting in dicult social
circumstances and communications.
Perceptual evaluation of speech by an experienced
speech-language pathologist remains a widely accepted
standard tool.
8-9
e Pittsburgh weighted speech score
(PWSS), originally described by McWilliams and
Phillips
10
, is one of the key standardized methods used
for a perceptual speech assessment. is tool rates the
severity of velopharyngeal incompetency (VPI) by
evaluating ve speech components: nasality/resonance,
nasal air emission, facial grimace, phonation/voice, and
articulation.
Due to the lack of postoperative speech outcomes
aer CP repair in our centre, we conducted this study
using PWSS to evaluate the postoperative speech outcomes
aer primary palatal repair.
MATERIALS AND METHODS
Patient enrollment
is research involved a prospective cohort study.
Ethical approval was granted by the Institutional Review
Board committee, Faculty of Medicine Siriraj Hospital,
Mahidol University (Si 382/2018(EC2)). Informed consent
was obtained. Patients who were diagnosed with CP
with/without CL and underwent primary CP repair at
Siriraj Hospital were enrolled. Exclusion criteria were
syndromic patients with associated anomalies and patients
who had follow-up time less than 3 years (co-operable
assessment issue). Aer reviewing medical records, the
eligible patients were contacted via telephone and invited
for study participation.
Speech outcome assessment
The speech evaluation was conducted between
October 2019 and 2020. Perceptual speech outcome
assessments using PWSS were conducted face-to-face
by a qualied speech-language pathologist (K.L.) (Fig 1).
Five components were investigated: nasal air emission,
facial grimace, nasality/resonance, phonation/voice,
and articulation. e sum of scores equal to 0 indicated
velopharyngeal competency, 1-2 indicated borderline
velopharyngeal competency, 3-6 indicated borderline
VPI, and ≥7 indicated VPI.
Data collection
e patients’ medical records were reviewed to
collect the following data: age and weight at primary
palatoplasty, gender, cle palate type based on Veau
classication
11
, cle gap, techniques used in hard/so
palate procedures, the use of Vomerine ap, operation
time, estimated blood loss, hospital length of stay, and
the incidence of postoperative oronasal stula (ONF) and
otitis media eusion (OME). e Veau classication
11
categorizes cle palate into four groups: Veau 1 (defects
involving the so palate only), Veau 2 (defects involving
the hard and so palate), Veau 3 (defects involving the
so palate to the alveolus, usually with lip involvement),
and Veau 4 (complete bilateral cle palate). Intraoral
examination and photography were performed to identify
postoperative ONF, dened as an abnormal connection or
hole between the oral and nasal cavities, while intentionally
unrepaired anterior hard palate and lingual–alveolar or
labial–alveolar stulas were not dened as this particular
condition.
Statistical analysis
As appropriate, continuous variables were summarized
using Mean ± standard deviation (SD) or Median (range).
Categorical variables were summarized using counts
with the percentage. e clinical and peri-operative
characteristics between patients with and without VPI
were compared using the Student’s t-test or Mann-
Whitney U test for continuous variables, while Pearson
chi-square, Yates’ continuity correction or Fisher’s exact
test was used for categorical variables, as appropriate.
P-value of ≤0.05 was considered statistically signicant.
Statistical analyses were performed using PASW Statistics
for Windows, Version 18.0. Chicago: SPSS Inc.
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Thongprayoon et al.
Fig 1. Perceptual speech evaluation form adapted from the Weighted Values for Speech Symptoms Associated with Velopharyngeal
Incompetence of the Cle Palate Center of the University of Pittsburgh
10
Nasal Emission: Facial Grimace: ____2
Not present _____0
_____1
Nasality:
Inconsistent, Visible Normal ____0
Consistent, Visible _____2 Mild Hypernasality ____1
Nasal escape on nasals appropriate _____0
_____0
_____0
Moderate Hypernasality ____2-3
Reduced Severe Hypernasality ____4
Absent Hypo-Hypernasality ____2
Audible _____3 Cul de sac resonance ____2
Nasal Turbulence _____3 Hyponasality ____ 0
******Summary: Nasal emission scores: _____* ******Summary: Facial grimace
and nasality scores: ____*
Phonation: Articulation:
Normal ____0 Normal ____0
Hoareseness or Breathiness Development errors ____0
Mild ____1 Errors from other causes not related to VPI ____0
Moderate ____2 Errors related to anterior dentition ____0
Severe ____3 Reduced intraoral pressure for sibila nts ____1
Reduced Loudness ____2 Reduced intraoral pressure for other fricatives ____2
Tension in system ____3 Reduced intraoral pressure for plosives ____3
Other__________________ Omission of fricatives or plosives ____2
******Summary: Phonation scores: ____* Omission of fricatives or plosives plus
hard glottal attacks for vowels ____3
Lingual-palatal sibilants ____2
Pharyngeal fricatives, snorts,inhalation or
exhalation substitutions ____3
Glottal stops ____3
Nasal substitutions for pressure sounds ____4
******Summary: Articulation scores: ____*
******TOTAL SCORE (from all sections above): ____
Speech Indicates:
____0 Competent Velopharyngeal Mechanism
____1-2 Competent to Borderline Competent
____3-6 Borderline to Boderline Incompetent
____7 Incompetent Velopharyngeal Mechanism
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RESULTS
Forty patients (21 males and 19 females) with CP
between 3-15 years of age who underwent primary cle
palate repair in Siriraj Hospital were included in the study.
e median age at primary palatoplasty was 12 months
old (range 8-40). Veau type 3 was found in 47.5% of
patients, while Veau type 4, type 2, and type 1 were found
in 30%, 15%, and 7.5%, respectively. e mean cle gap
width was 12.3 mm. e Bardach two-ap palatoplasty
technique was performed in 94.6% of patients, while 5.4%
underwent Veau-Wardill-Kilner palatoplasty for hard
palate surgery. Intravelar veloplasty was performed in
82.5% of patients, while 2.5% underwent radical intravelar
veloplasty, and 12.5% underwent Furlow z-plasty for so
palate surgery. One patient (2.5%) has unknown detail
of the so palate procedure. e vomerine ap was used
in 42.5% of patients. e mean operation time was 127
min. Table 1 summarizes the patients’ demographic
data.
At the time of speech evaluation, the median age
was 7 years (range 3-15 years). Among 40 patients, 21
(52.5%) had VPI, while 13 (32.5%) had borderline VPI,
and 4 (10%) had borderline velopharyngeal competency,
and 2 (5%) had velopharyngeal competency. e median
total PWSS score was 7.0 (0-18). Median score for nasal
emission was 2 (0-3), phonation was 0 (0-5), facial grimace
was 0 (0-2), nasality was 1 (0-4), and articulation was
3 (0-10) (Table 2). ere was no signicant dierence
in age at primary palatoplasty, or for the type and width
of cle palate, type of surgery for hard and so palate,
or incidence of postoperative ONF and OME between
patients with or without VPI (data shown in Table 3).
Overall, 16 patients (40%) had postoperative ONF. All
of those underwent stula closure operations. However,
8 patients had recurrence stula at the time of speech
evaluation. e median interval between primary CP
surgery and ONF occurrence was 22 months (range
3-40 months). OME was found in 30 patients (75%).
DISCUSSION
We present interim data on the perceptual speech
outcomes aer primary cle palate repair from a tertiary
referral centre in ailand. We found over half of our
patients with CP had postoperative VPI aer primary
CP repair at around 7 years of age. Moreover, making
articulation errors was the most common characteristic
aecting achieving a higher PWSS.
Normal speech achievement is one of the most
important goals of CP surgery. Perceptual evaluation
(i.e., listening) is the “gold standard” clinical assessment
method for speech and voice disorders in the cle palate
population, and has been used in several previous studies.
8,12-16
In this study, VPI was identied by perceptual speech
evaluation using PWSS in 53% of patients. is rate is
higher than those reported in prior studies, ranging
from 15% to 45%.
4-5
Articulation errors were found to
be the most common speech distortions in our series.
Pratanee et al., in 2016, found that articulation errors
were the most common speech and language defects in
ai cle palate patients.
17
Oopanasak et al. organized
a case-control study of ai children aged 6-13 years
old and found that patients with CLP had signicantly
higher articulation defects than normal children, with
velar and trill errors the most common articulation
patterns.
18
Another study in Saudi Arabic-speaking
children aged between 6-15 years old by Albustanji et
al. found speech abnormalities, including articulation,
hypernasality, and resonance, in 74% of patients aer
CP repair.
19
A study from Korea reported that 20% of
patients had postoperative VPI and 50% demonstrated
articulation decits.
20
Recently, a study of Arabic-speaking
Egyptian children between 3-9 years old demonstrated
that articulation disorders, especially substitution, were
the most common errors in CP patients with VPI.
21
ere are many factors involved in articulation.
Every element of the speech apparatus, including the lips,
teeth, palate, tongue, velum, and larynx, are engaged in
producing intelligible sounds.
22
Patients in our study were
evaluated at 7 years old, which is in the mixed dentition
phase. Abnormal dental alignment (e.g., severe crowding),
including transverse maxillary collapse during this time,
maybe a causative factor in articulatory disorders in cle
palate patients. Another factor, including the prevalence
of remaining alveolar cle during this particular phase,
may interfere with the incidence of articulation errors.
Signicantly, most children with CP in our study did
not receive any regular long-term speech therapy aer
surgery. Although speech therapy in ailand is oered
to cle palate patients free of charge due to our universal
health care program, regular long-term speech therapy
can be burdensome. Ideally, patients are required to
attend 30-minute speech therapy sessions at least every
2-3 months for several years. Further, the speech therapy
service is only available in a restricted number of tertiary
referral hospitals. In addition, as the patients are children,
their parents need to accompany them to the hospital
for the service. is would cost them transportation
expenses and a need to miss work, resulting in reduced
income. erefore, access to and take-up speech therapy
in our patients is limited, especially when considering the
long distance to the service and socioeconomic status of
many of our patients’ families.
23-24
Unsurprisingly, many
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TABLE 1. Demographic data of 40 patients with cle palate who underwent primary cle palate repair.
Characteristics (Total=40)
Genders, number (%)
Male 21 (52.5%)
Female 19 (47.5%)
Surgeons
Attending staff 33 (82.5%)
Trainees 7 (17.5%)
Age at primary palatoplasty (months), Median (range) 12 (8-40)
Age at speech assessment (years), Median (range) 7 (3-15)
Cleft types, number (%)
Veau type I 3 (7.5%)
Veau type II 6 (15%)
Veau type III 19 (47.5%)
Veau type IV 12 (30%)
Cleft gap width (mm.), Mean ± SD 12.3 ± 4.1
Type of hard palate procedure, number (%) Total 37
Two-ap palatoplasty 35 (94.6%)
Veau–Wardill–Kilner palatoplasty 2 (5.4%)
Type of soft palate procedure, number (%) Total 40
Intravelar veloplasty 33 (82.5%)
Radical intravelar veloplasty 1 (2.5%)
Furlow Z-plasty 5 (12.5%)
Unknown detail of the procedure 1 (2.5%)
Vomerine ap use, number (%) 17 (42.5%)
Blood loss (ml.), Median (range) 20 (1-150)
Surgery duration (minute), Mean ± SD 127 ± 49
Hospital stay (days), Mean ± SD 4 ± 1
Presence of oronasal stula, number (%) 16 (40%)
Presence of otitis media effusion, number (%) 30 (75%)
Speech indicates, number (%)
Velopharyngeal competency 2 (5%)
Borderline velopharyngeal competency 4 (10%)
Borderline VPI 13 (32.5%)
VPI 21 (52.5%)
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TABLE 2. Modied weighted values for speech symptoms associated with velopharyngeal incompetence of the Cle
Palate Center of the University of Pittsburgh by perceptual evaluation.
TABLE 3. Various parameters and the association with VPI.
Modality Score
Total : Median (range) 7 (0-18)
Articulation : Median (range) 3 (0-10)
Nasality : Median (range) 1 (0-4)
Nasal emission : Median (range) 2 (0-3)
Phonation : Median (range) 0 (0-5)
Facial grimace : Median (range) 0 (0-2)
Parameter
PWSS < 7 PWSS ≥ 7
(n= 19) (n= 21)
P-value
Age at primary palatoplasty, Median (range) (months) 12 (8-40) 12 (9-39) 0.539
Cleft types, number (%)
Veau type I 3 (15.8) 0 (0) 0.238
Veau type II 3 (15.8) 3 (14.3)
Veau type III 7 (36.8) 12 (57.1)
Veau type IV 6 (31.6) 6 (28.6)
Cleft gap width, Mean ± SD (mm.) 12.2 ± 3.4 12.3 ± 4.7 0.909
Type of hard palate procedure, number (%)
Two-ap palatoplasty 16 (84.2) 19 (90.5) 0.495
Veau–Wardill–Kilner palatoplasty 0 (0) 2 (9.5)
Type of soft palate procedure, number (%)
Intravelar veloplasty 13 (68.4) 20 (95.2) 0.130
Radical intravelar veloplasty 1 (5.3) 0 (0)
Furlow Z-plasty 4 (21.1) 1 (4.8)
Vomerine ap use, number (%) 7 (36.8) 10 (47.6) 0.713
Presence of oronasal stula, number (%) 6 (31.6) 10 (47.6) 0.477
Presence of otitis media effusion, number (%) 15 (78.9) 15 (71.4) 1.000
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factors, not only velopharyngeal anatomical deciencies,
are associated with VPI in our patients in this centre.
Our study revealed no signicant association between
age at primary surgery, types and width of cle palate,
type of CP surgery, or incidence of postoperative ONF,
OME, and severity of VPI. ese data are consistent with
a previous study from a tertiary university hospital in
Northeast ailand.
25
Postoperative ONFs in cle palate have been reported
in 9%-50% of cases.
26-27
e ONF rate in the present study
was 40%, which is consistent with previous literature.
28-31
A large case series from Khon Kaen University, ailand,
revealed that ONF formation was signicantly associated
with higher types of Veau classication, syndromic cle
patients, and a cle width more than 11.5 mm.
32
In our
series, we found that the majority of patients had Veau type
3 or 4 (77%), and the mean cle gap was 12.3 mm. From
our study, 48% of patients with VPI had postoperative
ONF. Although an ONF closure procedure was performed
in all these patients, 50% still had residual stula at the
time of the study. In the borderline VPI group, 38% of
patients had ONF. Also, 60% of those still had residual
ONF at the time of the study despite a stula closure
procedure having been performed. ere were no ONF
cases in the velopharyngeal competency and borderline
velopharyngeal competency group. ONF might be the
cause for this, producing a higher hypernasality score
in the PWSS.
OME is common in CLP, oen resulting in hearing
loss, speech delay, and learning disabilities. If untreated,
chronic otitis media or cholesteatoma may occur, leading
to permanent middle ear damage. In our hospital, the
prevalence of OME was reported by Ungkanont et al.
to be around 50%–80%.
33
ey also found a signicant
improvement in the audiograms aer palatoplasty. In
this series, we found 75% of patients had OME. is
condition might be another confounding factor that
aected the poor speech outcomes in this study, despite
the statistical analysis suggesting no signicant association
between OME and speech outcomes in this study. Further
subgroup analysis should be performed to conrm this
outcome.
We realize that one of the signicant limitations
of our study was the small population sample. We had
previously planned to enrol a study population of 200
by calculating the predicted sample size using nQuery
Advisor (San Diego, CA) and by assuming the rate of
velopharyngeal incompetency as 37.5% with condence
interval of 95%. However, due to the COVID-19 pandemic
in ailand during 2020, we, unfortunately, could only
enrol 40 patients to take part in face-to-face speech
evaluation. Ongoing speech evaluations are continuing
for a complete long-term speech outcome assessment.
is interim study should, however, be of benet
for our hospital and can reinforce the need for MDT
care, including cle surgeon, speech pathologist, ear nose
throat surgeon, and dental team, which is mandatory for
all patients with CLP. Objective investigations, such as
nasendoscopy and/or video-uoroscopy, should be further
used in patients who have VPI from initial perceptual
speech evaluation as the diagnostic tools and preoperative
planning before any subsequent correction procedure.
CONCLUSION
Postoperative VPI is common aer cle palate
repair in our hospital. e articulation disorder is the
most common characteristic aecting speech outcomes.
We encourage establishing a cle and craniofacial centre
to deliver MDT care in our hospital to achieve the best
benets in CLP care.
ACKNOWLEDGEMENTS
The authors gratefully thank all the attending
sta in the Division of Plastic Surgery, Department of
Surgery, Faculty of Medicine Siriraj Hospital for their
great contribution to this study and patient management.
Funding: e authors received no nancial support for
this article’s research, authorship, and/or publication.
Conicts of interest: e authors declare no conicts
of interest concerning this article’s research, authorship,
and/or publication.
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