Sunteți pe pagina 1din 9

The Laryngoscope

C 2013 The American Laryngological,


V

Rhinological and Otological Society, Inc.

We Can Predict Postpalatoplasty Velopharyngeal Insufficiency in


Cleft Palate Patients
Jacques E. Leclerc, MD; Audrey Godbout, MD; Isabelle Arteau-Gauthier, MD; Sophie Lacour, MOA;

Kati Abel, MOA; Elisa-Maude
McConnell, MSc
Objectives/Hypothesis: To find an anatomical measurement of the cleft palate (or a calculated parameter) that predicts
the occurrence of velopharyngeal insufficiency (VPI) after palatal cleft repair.
Study Design: Retrospective cohort study.
Methods: Charts were reviewed from cleft palate patients who underwent palatoplasty by the Von Langenbeck technique for isolated cleft palate or Bardach two-flap palatoplasty for cleft lip-palate. Seven anatomical cleft parameters were
prospectively measured during the palatoplasty procedure. Three blinded speechlanguage pathologists retrospectively scored
the clinically assessed VPI at 4 years of age. The recommendation of pharyngoplasty was also used as an indicator of VPI.
Results: From 1993 to 2008, 67 patients were enrolled in the study. The best predicting parameter was the ratio
a/(30 2 b1), in which a is defined as the posterior gap between the soft palate and the posterior pharyngeal wall and b1 is
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).
Conclusions: The width of the cleft at the hard palate level and the posterior gap between the soft palate and the posterior pharyngeal wall were found to be the most significant parameters in predicting VPI. The best correlation was obtained
with the ratio a/(30 2 b1).
Key Words: Cleft palate, hypernasality, velopharyngeal insufficiency, velopharyngeal dysfunction, palatoplasty,
pharyngoplasty.
Level of Evidence: 4
Laryngoscope, 124:561569, 2014

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

Laryngoscope 124: February 2014

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.

MATERIALS AND METHODS


In a tertiary care academic university-based medical center, we retrospectively reviewed the charts of patients who
underwent palatoplasty performed by the senior author (J.E.L.).
From 1993 to 2008, all patients born with a CP (6cleft lip) were
prospectively enrolled in a database including data on the pregnancy, birth weight, and types of associated malformations. At

Leclerc et al.: Postpalatoplasty Velopharyngeal Insufficiency

561

Fig. 1. Cleft lip/palate measurements


diagram (J.E.L.). [Color figure can be
viewed in the online issue, which is
available at wileyonlinelibrary.com.]

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.

Evaluation of Velopharyngeal Function


The SLPs were blinded to the results of the anatomical
measurements. Clinical charts were retrospectively reviewed for

Laryngoscope 124: February 2014

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

Distance between the posterior end of the soft


palate and the posterior pharyngeal wall in the
plane of the hard palate

Largest width of the cleft at the soft palate level

b1
c

Width of the cleft at the posterior end of the hard palate


Length of the soft palate, from its posterior end to
the posterior end of the hard palate

Largest width of the dental arch measured at the


top of the crests

Length of the hard palate, from its posterior end to


the top of the anterior dental arch with a rigid ruler;
the depth of the hard palate is not evaluated

Total length of the cleft, from the posterior end of


the soft palate to the anterior end of the cleft

Leclerc et al.: Postpalatoplasty Velopharyngeal Insufficiency

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.

 Perceptual speech evaluation (PSE) was considered positive


if at least one of these criteria was fulfilled: severity score of
3 on the hypernasality scale or presence of audible nasal
emission (score of 1 on the scale) or presence of nasal turbulence (score of 1 on the scale; Table II).
 Speech acceptability was assessed independently. Acceptability is defined as the degree to which speech calls attention to
itself apart from the content of the spoken language, and it
is closely related to patients quality of life. Scores of 2 and 3
were used as an indication of VPI (Table II).
 Pharyngoplasty recommendation (PR) was used as the third
indicator of significant persisting VPI. A surgical treatment
was offered to the patient when 1) speech therapy did not
improve VPI after complete SLP follow-up and 2) an obvious
anatomical defect could explain the problem (Table II). No
pharyngoplasty was recommended or performed in the
absence of VPI.

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

Fig. 2. Flowchart for patient recruitment.

Laryngoscope 124: February 2014

Leclerc et al.: Postpalatoplasty Velopharyngeal Insufficiency

563

TABLE III.
Cleft Lip/Palate Series.
Characteristic

Value

Total number

67

Gender, male %/female %


Weight at palatoplasty, kg

58.2/41.7
9.1 (61.4)

Age palatoplasty, mo

10.6 (60.9)

Age at evaluation by SPL, mo


Age at PR, mo

49.6 (616.0)
73.9 (638.4)

Isolated cleft palate

38 (57%)

Unilateral cleft lip and palate


Bilateral cleft lip and palate

23 (34%)
6 (9%)

PR 5 pharyngoplasty
pathologist.

recommendation;

SPL 5 speechlanguage

20.9%, 30.8%, and 50% for PR, speech acceptability, and


PSE. Added to our anatomical measurements, >20 combinations of the anatomical measurements were created
as other parameters. For each dependent variable (PR,
PSE, acceptability), these combinations of anatomical
measurements were tested. The 10 that best discriminated according to the dc value of the ROC curve are
presented in decreasing order in Tables V to VI.
Tables V and VI, respectively, present the data of
only 60 and 65 patients of 67. In their retrospective
chart evaluation, the SLPs felt that they could not
adequately classify the missing others according to the
available information. We did not get b1 values for all
the patients in each table. The missing values correspond to the number of patients with clefts limited to
the soft palate. We elected to assign no value for b1
instead of zero. The f parameter was our marker for the
anteroposterior extent of the cleft. There was an obvious
clerical mistake for one c value that we did not use for
any of our calculations.
For all three dependent variables, the data in these
tables show that the ratio a/(30 2 b1), in which a is
defined as the posterior gap between the soft palate and
the posterior pharyngeal wall and b1 is the width of the
cleft at the hard palate level, had a good discriminating
value and a globally better sensitivity that reached statistical significance. The cutting ratio values for PSE,

Fig. 3. Cleft palate anatomical measurements in millimeters.

Laryngoscope 124: February 2014

564

TABLE IV.
Incidence (%) of Velopharyngeal Insufficiency as Determined by
Perceptual Speech Evaluation, Speech Acceptability, and Pharyngoplasty Recommendation.
Velopharyngeal Insufficiency Evaluation

Perceptual speech evaluation

50

Hypernasality
0
1
2

31.8
16.7
33.3

3
Audible nasal emission score 1

18.2*
24.1*

Audible nasal turbulence score 1

30.9*

Speech acceptability
Pharyngoplasty recommendation

30.8
20.9

*Respective percentages for the criteria used for perceptual speech


evaluation in the study. The sum of the percentages of the individual PSE
criteria does not match the 50% total because some patients scored as
positive >1 criterion.

acceptability, and PR were respectively 0.73, 0.83, and


0.79. Table VIII shows the calculated relative risk of
developing VPI for these values. The parameter a 1 b1
was a close contender (Tables V).
b1 and a presented the highest discriminating values among the single anatomical measurements, but did
not get good rankings in all three dependent variables
(Table VIII). The measurement b1 performed well in two
of the three VPI evaluations and the depth of the nasopharynx (a) in one (PSE). They were also found within
many of the top 10 parameters (Tables V). There was no
association between age, sex and PSE, and PR and
acceptability.

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

*Number of cases above or below the cutting value.

Percentage of VPI in the patients above or below the cutting value.

v2 test.

Parameter within top 10 in all three VPI evaluation modalities (P <.05).


dc 5 discriminant capacity with associated ranking; PSE 5 perceptual speech evaluation; RR 5 relative risk; VPI 5 velopharyngeal insufficiency.

parameters (constant or inconstant nasal turbulence or


emission). They were classified as minimal VPI, but
experienced no functional or social impact on everyday
life. Of the remaining 32%, as previously mentioned, 2=3
of the cases were offered a pharyngoplasty (14 of 67
patients). We found a close correlation between the
20.9% pharyngoplasty rate and the score 3 hypernasality
rate of 18.2% (Table IV; P <.0001). We are in the process
of reviewing the nonoperated cases for a possible secondary palatal lengthening procedure such as the Furlow
operation.
The 32% rate of VPI is likely mostly related to the inherent limitations of the palatoplasty procedure. Looking
retrospectively at the charts of this group, we found other
factors that may contribute to the persisting VPI. Some
cases had minor residual fistulas for which a surgical procedure was considered but was not already done at age 4
years. Some patients had other deleterious health problems not identified by our exclusion criteria, such as significant deafness and dyspraxia. These conditions may
have interfered with the re-education of the child.
Reported rates of persistent VPI following primary
palatoplasty show a wide range in the literature, from
13% to 35%.713 A number of factors such as the size of
the cleft, age, and type of primary palatal repair performed as well as speech assessment procedures may be

responsible for these differences in VPI rates. No data


have clearly been identified to predict the occurrence of
VPI. Conflicting data about the association between age,
surgery, and classification of the cleft with VPI was noticeable in our literature review.

Laryngoscope 124: February 2014

Leclerc et al.: Postpalatoplasty Velopharyngeal Insufficiency

Top Performing Parameters


The goal of the study was to find an anatomical
measurement of the CP or a calculated parameter that
predicts the occurrence of VPI. We wanted to use linear
measurements that are readily available to the surgeon.
We were looking for a single ideal parameter to be applicable to the various forms of cleft malformations for this
combination of two surgical techniques. We selected in
our database a group of CP cases associated or not associated with a cleft lip, all treated surgically with an
identical technique. We fully acknowledge that velopharyngeal closure is a dynamic process and other neuromuscular factors are significant in the development of
VPI even in an ideal subject. We eliminated all cases
with syndromes as well as other confounding factors and
hoped this would reduce the effect of any neurologic
component in the selected cases.
The ratio a/(30 2 b1) was a top performer in all
three tests and was considered the best parameter. The

565

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 of cases above or below the cutting value.

Percentage of VPI in the patients above or below the cutting value.


2
v test.

Parameter within top 10 in all three VPI evaluation modalities (P <.05).


dc 5 discriminant capacity with associated ranking; RR 5 relative risk; VPI 5 velopharyngeal insufficiency.

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.

The dental arch width d is partly determined by the


width of the cleft b1. We think this is why both variables
were good performers when associated with a in our
study .
In the top 10 lists of predictors (Tables V), the
width of the hard palate (b1), and the depth of the
nasopharynx (a) were very prominent in the top performing parameters. As shown in Table VIII, b1 > 9
mm and a > 16 mm were the top anatomical parameters. Lam and coworkers13 studied isolated CP cases
repaired with either a Furlow double-opposing Z-plasty
or a combined Furlow palatoplasty and a V to Y pushback procedure. They also found that palatal
width > 10 mm is associated with a higher risk of VPI.
Further studies are required to determine whether our
top parameters will be similar for other types of CP
repair such as the Furlow procedure. It is likely they
will remain the best predictors, but the cutting values
of the ratios and measurements may be different.
Moreover, with a larger cohort, we may be able to separate the various types of clefts (6cleft lip, unilateral
vs. bilateral cleft lip) and get a specific ratio that
would reach statistical significance.
The length of the soft palate (c), the length of the
hard palate (e), and the length of the cleft (f) did not
show any association with VPI. Palatal length has been
studied in an attempt to predict the need for

Laryngoscope 124: February 2014

Leclerc et al.: Postpalatoplasty Velopharyngeal Insufficiency

566

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

Number of cases above or below the cutting value.


*Percentage of VPI in the patients above or below the cutting value.

Fisher exact test.

Parameter within top 10 in all three VPI evaluation modalities (P <.05).


2
v square test.
dc 5 discriminant capacity with associated ranking; PR 5 pharyngoplasty recommendation; RR 5 relative risk; VPI 5 velopharyngeal insufficiency.

pharyngoplasty. Randall et al.14 measured the distance


between the distal tips of both uvulae and the posterior
pharyngeal wall. They concluded that for a cleft patient,
if the tips of the uvulae did not reach the adenoids, a
primary pharyngoplasty must be considered. Marrinan
et al.7 and Paliobeli et al.15 suggested a working hypothesis that the vomeric muscular complex obtained by
objective measurements of palatal musculature could be
used to predict VPI.

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

a/(30 2 b1)  0.73

Perceptual Speech Evaluation

76% (0.014)

2.2

a/(30 2 b1)  0.83


a/(30 2 b1)  0.79

Acceptability
Pharyngoplasty Recommendation

72% (0.003)
81% (0.028)

3.3
5.1

Perceptual Speech Evaluation

NS

b1  9 mm
b1  9 mm

Acceptability
Pharyngoplasty Recommendation

67% (0.01)
73% (0.036)

2.7
3.8

a  16 mm

Perceptual Speech Evaluation

70% (0.01)

2.0

Acceptability
Pharyngoplasty Recommendation

NS
NS

b1

a
a

NS 5 not significant; RR 5 relative risk; VPI 5 velopharyngeal insufficiency.

Laryngoscope 124: February 2014

Leclerc et al.: Postpalatoplasty Velopharyngeal Insufficiency

567

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.

Meaning of the Findings and Future Research


The results of the study are preliminary and suggest that a surgeon undertaking a CP repair with the
Von Langenbeck technique for CP patients or the Bardach two-flap palatoplasty for CLP patients can assess
the risk of developing VPI by measuring the posterior
gap behind the soft palate (a) and the hard palate cleft
width (b1). If the ratio a/(30 2 b1) is higher than 0.7 to
0.8, the patient is more likely to develop VPI. b1 and a
values >9 mm and >16 mm, respectively, may also be
used. In the future, we will add measurements of the
thickness of the soft palate muscles and the width of the
posterior pharyngeal wall. We may be able to develop
Laryngoscope 124: February 2014

568

algorithms including the palatal area, the cleft, and the


nasopharyngeal surface, although they may not be as
easy to use on site by the surgeon. The prediction of a
suboptimal result may eventually warrant a change in
the surgical technique. Similar studies must be conducted with other surgical techniques such as the Furlow procedure.

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.

BIBLIOGRAPHY
1. McWilliams BJ, Musgrave RH. Diagnosis of speech problems in patients
with cleft palate. Br J Disord Commun 1971;6:2632.
2. Rudnick EF, Sie KC. Velopharyngeal insufficiency: current concepts in diagnosis and management. Curr Opin Otolaryngol Head Neck Surg
2008;16:530535.
3. Nyberg J, Westberg LR, Neovius E, Larson O, Henningsson G. Speech
results after one-stage palatoplasty with or without muscle reconstruction for isolated cleft palate. Cleft Palate Craniofacial J 2009;47:92103.
4. Henningsson G, Kuehn DP, Sell D, Sweeney T, Trost-Cardamone JE,
Whitehill TL. Universal parameters for reporting speech outcomes in
individuals with cleft palate. Cleft Palate Craniofac J 2008;45:117.
5. Sell D, Harding A, Grunwell P. GOS.SP.ASS.98: an assessment for speech
disorders associated with cleft palate and/or velopharyngeal dysfunction
(revised). Int J Lang Commun Disord 1999;34:1733.
6. Lohmander A, Olsson M. Methodology for perceptual assessment of speech
in patients with cleft palate: a critical review of the literature. Cleft Palate Craniofac J 2004;41:6470.
7. Marrinan EM, LaBrie RA, Mulliken JB. Velopharyngeal function in nonsyndromic cleft palate: relevance of surgical technique, age at repair,
and cleft type. Cleft Palate Craniofac J 1998;35:95100.
8. Nyberg J, Westberg LR, Neovius E, Larson O, Henningsson G. Speech
results after one-stage palatoplasty with or without muscle reconstruction for isolated cleft palate. Cleft Palate Craniofac J 2010;47:92103.
9. Phua YS, de Chalain T. Incidence of oronasal fistulae and velopharyngeal
insufficiency after cleft palate repair: an audit of 211 children born
between 1990 and 2004. Cleft Palate Craniofac J 2008;45:172178.
10. Van Lierde KM, Monstrey S, Bonte K, Van Cauwenberge P, Vinck B. The
long-term speech outcome in Flemish young adults after two different
types of palatoplasty. Int J Pediatr Otorhinolaryngol 2004;68:865875.
11. Inman DS, Thomas P, Hodgkinson PD, Reid CA. Oro-nasal fistula development and velopharyngeal insufficiency following primary cleft palate

Leclerc et al.: Postpalatoplasty Velopharyngeal Insufficiency

surgeryan audit of 148 children born between 1985 and 1997. Br J


Plast Surg 2005;58:10511054.
12. de Buys Roessingh AS, Cherpillod J, Trichet-Zbinden C, Hohlfeld J.
Speech outcome after cranial-based pharyngeal flap in children born
with total cleft, cleft palate, or primary velopharyngeal insufficiency. J
Oral Maxillofac Surg 2006;64:17361742.
13. Lam DJ, Chiu LL, Sie KC, Perkins JA. Impact of cleft width in clefts of
secondary palate on the risk of velopharyngeal insufficiency. Arch Facial
Plast Surg, 2012 Apr 16 Epub.
14. Randall P, LaRossa D, McWilliams BJ, Cohen M, Solot C, Jawad AF. Palatal length in cleft palate as a predictor of speech outcome. Plast Reconstr
Surg 2000;106:12541259; discussion 12601261.

Laryngoscope 124: February 2014

15. Paliobeli V, Psifidis A, Anagnostopoulos D. Hearing and speech assessment


of cleft palate patients after palatal closure. Long-term results. Int J
Pediatr Otorhinolaryngol 2005;69:13731381.
16. Kummer, AW, Briggs M, Lee L. The relationship between the characteristics of speech and velopharyngeal gap size. Cleft Palate Craniofac J
2003;40:590596.
17. Lam DJ, Starr JR, Perkins JA, et al. A comparison of nasendoscopy and
multiview videofluoroscopy in assessing velopharyngeal insufficiency.
Otolaryngol Head Neck Surg 2006;134:394402.
18. Kummer AW. Perceptual assessment of resonance and velopharyngeal
function. Semin Speech Lang 2011;32:159167.

Leclerc et al.: Postpalatoplasty Velopharyngeal Insufficiency

569

S-ar putea să vă placă și