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INTRODUCTION
Velopharyngeal insufficiency (VPI) can be defined
as an inadequate physiological barrier between the nasopharynx and oropharynx during speech. Nasal air
escapes during the production of various phonemes, and
affects speech intelligibility and therefore the patients
quality of life. VPI sometimes persists in cleft palate
(CP) patients after palatoplasty. Factors such as the
length and function of the soft palate, the depth and
width of the nasopharynx, and the motion of the posterior and lateral pharyngeal walls1 determine the quality
of speech. The best evaluation of this speech problem
remains the perceptual speech assessment performed by
a trained speechlanguage pathologist (SLP).2 In some
cases, speech therapy alone is insufficient to correct the
From the Quebec University Hospital Center (CHUQ) (J.E.L.), Quebec City, Quebec, Canada, Department of OtolaryngologyHead and
Neck Surgery, Laval University (A.G., I.A.-G.), Quebec City, Quebec, Canada, Department of SpeechLanguage Pathology, Quebec University
Hospital Center (S.L., K.A., E.-M.M.), Quebec City, Quebec, Canada
Editors Note: This Manuscript was accepted for publication April
22, 2013.
The authors have no other funding, financial relationships, or conflicts of interest to disclose.
Financial support was obtained from Quebec University Hospital
Center Foundation.
Send correspondence to Jacques E. Leclerc, 2705 Boul. Laurier,
Quebec, Qc. G1V 4G2 Canada. E-mail: jeleclerc@ccapcable.com
DOI: 10.1002/lary.24200
problem. A secondary surgical procedure such as pharyngoplasty is conducted after reaching a consensus
between the multidisciplinary group, the patient, and
his family. However, drawbacks such as hyponasality or
sleep apnea by overcorrection must always be taken into
consideration.
Objective
The objective of this study was to determine which
children are at risk to present VPI after palatoplasty.
Our specific objective was to find a statistically significant anatomical measurement or calculated parameter
of the palate or its cleft that can predict the occurrence
of VPI. This might help to reduce the prevalence of VPI
by the use of other surgical techniques, and could have
relevance for determining the duration of speech therapy
follow-up, the need for secondary surgery, and its inherent risks. This may also help to spare children from
the psychological and social stigmata related to this
problem.
561
the time of the palatoplasty procedure under general anesthesia, after positioning of the Dingman retractor, several anatomical parameters of the soft/hard palate and the cleft were
measured (Fig. 1, Table I). The measurements in millimeters
were obtained with an ophthalmologic caliper and a ruler and
were included in the database.
The inclusion criteria for our study were:
All the patients who had undergone palatoplasty by the senior author (J.E.L.) between 9 and 13 months of age, either by
the Von Langenbeck technique for CP patients or by Bardach two-flap palatoplasty for cleft lip-palate (CLP) patients.
In both groups, the levator palatini was freed from the posterior edge of the hard palate and retropositioned to restore
the levator sling and allow tension-free closure in the
midline.
Availability of a complete set of lip/palate measurements at
the time of the repair procedure.
A complete management and follow-up by our speech pathology team (child older than 4 years).
Patients with obvious cognitive and developmental disorders
or syndromes or other craniofacial malformations including
the Pierre Robin sequence were excluded from the study.
In addition to the previously described measurements,
>20 other parameters using two or more measurements were
created and tested in our quest to find the best possible
predictor.
562
information regarding patients speech as related to velopharyngeal function by a team of three SLPs specializing in the
evaluation of CP patients. The speech of each patient was
assessed as close as possible to the age of 4 years. Some
patients had remaining small hard palate fistulas but no soft
palate fistulas. The quality of speech and resonance is acknowledged as being the main outcome measure for evaluating postpalatoplasty patients.3 Perceptual evaluation remains the gold
standard for evaluating speech, as well as the most commonly
used method.46
TABLE I.
Description of the Palatal and Cleft Measurements.
Variable
Description
b1
c
TABLE II.
Parameters Used by the Speech Language Pathologists.
Perceptual Speech Evaluation
Hypernasality (sentences)
0 5 within normal limits
1 5 mild, indicates hypernasality resonance perceived on
vowels
2 5 moderate, hypernasality resonance perceived on vowels,
approximants/semivowels, and liquids, and presence of
weakened consonants
3 5 severe, indicates all of the above and the replacement of
voiced consonants by their nasal equivalents*
Audible nasal emission (sentences)
0 5 normal
1 5 present*
Audible nasal turbulence (sentences)
0 5 normal
1 5 present*
Speech Acceptability
0 5 normal
1 5 normal to mild
2 5 moderate (speech deviates from normal to a moderate
degree)*
3 5 severe (speech deviates from normal to a severe degree)*
Pharyngoplasty Recommendation
Failed adequate speech therapy and anatomical limitation
*Positive for velopharyngeal insufficiency.
Statistical Analysis
In regard to each outcome, the discriminating capacity
(dc) was evaluated for each variable by the c-index corresponding to the area under the receiver operating characteristic
(ROC) curve: 0 dc 1 using the SAS LOGISTIC procedure
(version 9.2.3; SAS Institute, Cary, NC). Only variables having
dc 0.6 were retained for further analysis. Using the OUTROC
option of the LOGISTIC procedure, each selected variable was
dichotomized at a corresponding level that maximized the sensitivity and the specificity of the measurement. A relative risk
ratio was calculated for each variable. All P values were determined using the v2 test or the Fisher exact test.
RESULTS
Speech Parameters and Rating Scales
The data collected underwent a mapping process, involving the conversion of our local teams evaluation protocol to the
predetermined set of chosen parameters based on Henningsson
et al.4 and Sell et al.5 For the purpose of this study, three different criteria or sets of criteria were used to classify each case as
to the presence of VPI:
The charts of 272 CLP patients who underwent palatoplasty between 1993 and 2008 were reviewed. Only
67 patients met all the inclusion criteria (Fig. 2). The
clinical features of our series are shown in Table III. The
CP measurement data are shown in Figure 3. The independent variables used in our analysis are depicted in
Table IV. Our study has shown a respective incidence of
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TABLE III.
Cleft Lip/Palate Series.
Characteristic
Value
Total number
67
58.2/41.7
9.1 (61.4)
Age palatoplasty, mo
10.6 (60.9)
49.6 (616.0)
73.9 (638.4)
38 (57%)
23 (34%)
6 (9%)
PR 5 pharyngoplasty
pathologist.
recommendation;
SPL 5 speechlanguage
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TABLE IV.
Incidence (%) of Velopharyngeal Insufficiency as Determined by
Perceptual Speech Evaluation, Speech Acceptability, and Pharyngoplasty Recommendation.
Velopharyngeal Insufficiency Evaluation
50
Hypernasality
0
1
2
31.8
16.7
33.3
3
Audible nasal emission score 1
18.2*
24.1*
30.9*
Speech acceptability
Pharyngoplasty recommendation
30.8
20.9
DISCUSSION
VPI Rates: Why Three Evaluation Modalities?
We selected to use three VPI evaluation modalities.
Within them, we defined VPI differently to be able to
pick up the subtle as well as the more severe cases and
therefore cover the widest possible range of VPI. Our
ideal anatomical parameter or combination had to perform well in all three modalities. PR was the indicator of
an obvious problem caused by a significant anatomical
deficit that needed to be corrected. The procedure was
found to be required in 20.9% of subjects in our series at
approximately 4 years old. For the second modality,
speech acceptability, we elected to use the scores 2 (moderate) and 3 (severe), which included a larger group of
children. We found that 30.5% of the 4-year-olds presented a significant difficulty. Of this group, 12% had a
moderate degree and 18.5% a severe degree of difficulty.
With the third modality, perceptual evaluation, we
opted to include all patients with even subclinical audible nasal emission or turbulence. We found that 50% of
the patients were completely normal. The residual 50%
included a wide range of VPI, from clinically normal to
severe. These figures may seem elevated. However, 12 of
67 patients (18%) scored positive in only one of the two
Leclerc et al.: Postpalatoplasty Velopharyngeal Insufficiency
TABLE V.
Top 10 Discriminating Parameters (dc Scores) for Perceptual Speech Evaluation.
PSE 5 50% (60 Patients)
Dc
Cutting Values
No.*
% of VPI
RR
Sensitivity, %
Specificity, %
Rank
a1d
0.712
52
<52
30
30
70.0
30.0
2.3
.002
70.0
70.0
a/(50 2 d)
0.709
1.12
29
72.4
2.5
.001
70.0
73.3
0.708
<1.12
560
31
32
29.0
68.8
2.4
.002
73.3
66.7
<560
28
28.6
Variables
a 3 d
a/(c 1 e)
0.687
0.33
<0.33
32
27
29.6
65.6
0.5
.006
72.4
63.3
0.676
16
32
65.6
2.0
.010
70.0
63.3
0.663
<16
0.94
28
31
32.1
64.5
2.0
.013
69.0
63.3
<0.94
28
32.1
a/c
a/(30 2 b1)
0.649
0.73
<0.73
28
19
67.9
31.6
2.2
.014
76.0
59.1
a 1 b1
0.642
25
27
66.7
1.9
.032
72.0
59.1
0.628
<25
0.79
20
31
35.0
64.5
1.9
.020
70.0
63.6
<0.79
29
34.5
126
<126
28
19
60.7
42.1
1.4
.210
68.0
50.0
10
a/(30 2 b)
a 3 b1
0.624
v2 test.
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TABLE VI.
Top 10 Discriminating Parameters (dc Scores) for Acceptability.
Acceptability, VPI 5 30.8% (65 patients)
Variables
dc
Cutting Values
No.*
% of VPI
RR
Sensitivity, %
Specificity, %
Rank
a 3 b1
0.749
154
<154
22
30
63.6
13.3
4.8
.0002
78.8
76.5
a 1 b1
0.709
27
23
56.5
3.3
.003
72.2
70.6
0.707
<27
1.06
29
23
17.2
52.2
2.7
.007
60.0
75.0
<1.06
41
19.5
a/c
a1d
0.703
52
<52
33
32
42.4
18.7
2.3
.039
70.0
57.8
a/(50 2 d)
0.698
1.12
32
43.7
2.4
.026
70.0
60.0
0.690
<1.12
0.83
33
23
18.2
56.5
3.3
.003
72.2
70.6
<0.83
29
17.2
a/(30 2 b1)
b1
0.679
9
<9
22
30
54.6
20.0
2.7
.010
66.7
70.6
a 3 d
0.678
660
19
52.6
2.4
.014
50.0
80.0
0.661
<660
16
46
51
21.7
23.5
0.4
.008
55.0
77.3
<16
13
61.5
0.24
<0.24
23
29
47.8
24.1
2.0
.075
61.1
67.6
10
c
b1/d
0.658
number 30 corresponds to the largest distance in millimeters between the inner maxillary crests in our cohort.
The subtraction 30 2 b1 represents the total width of the
insertion of the muscular band on each side of the cleft.
This finding indicates that wider clefts (with narrower
muscular bands) with a large gap between the ridge of
the soft palate and the posterior pharyngeal wall are
prone to VPI. A ratio higher than 0.7 to 0.8 is associated
with an increased risk of VPI as measured by all three
modalities: PSE, acceptability, and PR. The anatomical
measurement b and the combinations a 1 b, a 3 b, and
a/(30 2 b) did not perform quite as well as b1, a 1 b1, a
3 b1, and a/(30 2 b1). It is impossible to draw definitive
comparative conclusions, because the b group included
all the cleft patients and the b1 group only the soft palate clefts.
The combinations a 1 d, a 3 d, and a/(50 2 d) were
designed to determine whether a larger nasopharyngeal
distance (a) and a larger width of the dental arch (d)
were associated to an increased risk of VPI. The number
50 (representing millimeters) was selected so that the
subtraction 50 2 d would always produce a positive number. For example, an a/(50 2 d) ratio value 1.12 was a
very good performer in PSE. As a group, the combinations including a and d generally outperformed the isolated anatomical a and d measurements. In the PSE
evaluations, they showed high sensitivity and specificity.
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TABLE VII.
Top 10 Discriminating Parameters (dc Scores) for PR.
PR 5 20.9% (67 Patients)
Variables
dc
Cutting Values
No.
% of VPI*
RR
Sensitivity, %
Specificity, %
Rank
a 3 b1
0.723
176
<176
12
41
58.3
9.8
6.0
.001
63.6
88.1
b1/b
0.695
0.80
28
32.1
4.0
.043
81.8
54.8
0.686
<0.80
27
25
23
8.0
34.8
3.5
.028
72.7
64.3
<27
30
10.0
a 1 b1
a/(30 2 b1)
0.680
0.79
<0.79
25
28
36.0
7.1
5.1
.016
81.8
61.9
b1
0.661
9
22
36.4
3.8
.036
72.7
66.7
0.652
<9
0.94
31
35
9.7
28.6
2.2
.1428
71.4
53.8
<0.94
31
12.9
a/c
a 3 d
0.652
646
<646
22
45
36.4
13.3
2.7
.029
57.1
73.6
0.647
18
29
31.0
2.4
.074
64.3
62.3
0.646
<18
0.24
38
23
13.2
34.8
3.5
.041
72.7
66.7
<0.24
30
10.0
0.24
<0.24
41
26
12.2
34.6
0.4
.028
64.3
69.8
10
b1/d
b/d
0.642
Kummer et al.16 have looked at the possible relationship between the characteristics of speech and
velopharyngeal gap size. They found that some information about the gap size can be predicted to a certain extent if the patient has nasal rustle. Lam and
coworkers17 in 2006 developed the concept of a gap
area index. Their findings were that nasoendoscopic
evaluation correlated better with VPI than multiview
video fluoroscopy.
TABLE VIII.
Ratio a/(30 2 b1), b1, and a: Summary of Sensibility, p Value and RR of Velopharyngeal Insufficiency.
Variables
VPI Evaluation
Sensitivity (P)
RR of VPI
76% (0.014)
2.2
Acceptability
Pharyngoplasty Recommendation
72% (0.003)
81% (0.028)
3.3
5.1
NS
b1 9 mm
b1 9 mm
Acceptability
Pharyngoplasty Recommendation
67% (0.01)
73% (0.036)
2.7
3.8
a 16 mm
70% (0.01)
2.0
Acceptability
Pharyngoplasty Recommendation
NS
NS
b1
a
a
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Study Limitations
The SLPs had to classify the patients retrospectively according to the new rating systems: 1) Universal
Parameters for Reporting Speech Outcomes in Individuals With Cleft Palate4 and 2) the Great Ormond Street
Speech Assessment.5 They had to rely on the available
information in the charts. Speech recordings or instrumental assessments were not available for all the
patients and could not be used. Although the francophone speech protocol is not standardized, the same
speech material was used across time to assess all the
subjects in the study. In an attempt to facilitate crosscenter comparisons, the data collected in the present
study underwent a mapping process involving the conversion of our teams evaluation protocol to a determined
set of four parameters. The analysis carried out by three
different professionals induces a bias into the results.
Furthermore, inter-rater and intrarater reliability was
not obtained, although consensus judgment was
achieved on the first 15 charts reviewed. We cannot
determine whether our results can be extended to other
languages.
The speech of each patient was assessed at the
mean age of 4 years. In our experience, at this age, a
comprehensive evaluation of speech and language is generally possible. Furthermore, children with documented
satisfactory velopharyngeal sufficiency at this age are
unlikely to develop subsequent VPI.7 No audio/video recording or instrumental measures were obtained,
although a listening tube was systematically used.18
Instrumental measures such as nasopharyngoscopy
would have been an important addition to our study, but
were not available for all subjects.
Among the factors that affect the surgical results,
the exact surgical technique is important, and these
cases were treated with freeing and retropositioning of
the velar musculature. The majority of the cases were
operated on at the beginning of the surgeons career.
The experience of the surgeon is a significant factor, and
improvement of the results over time has not been studied. The problem of fistulas was not reviewed in the
data. With the techniques that were used, the fistulas
occurred only in the bony part of the repair (hard palate
or maxillary arch). This is a possible bias in this study.
No secondary procedure was added to lengthen the soft
palate.
568
CONCLUSION
The conclusions of this exploratory retrospective
study must be limited to CP patients undergoing palatoplasty by Von Langenbeck technique for isolated CP and
Bardach two-flap palatoplasty for CLP, between the ages
of 9 and 13 months. The speech evaluations were conducted in a French-speaking cleft population without
other malformations. Our results suggest that:
Based on all three VPI testing modalities, the best predicting
parameter was the ratio a/(30 2 b1), in which a is defined as
the distance between the posterior end of the soft palate and
the posterior pharyngeal wall and b1 is defined as the width
of the cleft at the hard palate level. An a/(30 2 b1) ratio >0.7
to 0.8 is associated with a higher risk of developing VPI (relative risk 5 2.25.1, sensitivity 5 72%81%, P <.03).
Based on two VPI testing modalities, the best anatomical parameter was a width of the cleft at the hard palate level >9
mm (relative risk 5 2.73.8, sensitivity 5 67%73%, P <.04).
A posterior gap between the soft palate and the posterior
pharyngeal wall >16 mm (relative risk 5 2.0, sensitivity 5
70%) reached statistical significance (P 5.01) with only one
VPI evaluation modality (PSE).
Acknowledgments
Members of the Committee of Orofacial Malformations of
Quebec University Hospital Center would like to acknowledge the work of Dr. Georges Demers, who has been an inspiration for all of us throughout his career.
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