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of Pediatric Otorhinolaryngology xxx (2015) xxx–xxx Contents lists available at ScienceDirect International

Contents lists available at ScienceDirect

International Journal of Pediatric Otorhinolaryngology

j o u r n al ho m ep a g e: ww w.els evier . c om /lo cat e/ijp o r l

al ho m ep a g e: ww w.els evier . c om /lo cat e/ijp

Acoustic analysis of voice in children with cleft palate and velopharyngeal insufficiency

Rocio Villafuerte-Gonzalez a , Victor M. Valadez-Jimenez a , Xochiquetzal Hernandez-Lopez a , Pablo Antonio Ysunza b , *

a Department of Speech & Language Pathology, Instituto Nacional de Rehabilitacion, Mexico City, Mexico b Ian Jackson Craniofacial and Cleft Palate Clinic, Neuroscience Program, Beaumont Health, Royal Oak, MI, United States

A R T I C L E

I N F O

Article history:

Received 2 March 2015 Received in revised form 17 April 2015 Accepted 18 April 2015 Available online xxx

Keywords:

Cleft palate

Speech

Voice

Acoustics

A B S T R A C T

Background: Acoustic analysis of voice can provide instrumental data concerning vocal abnormalities. These findings can be used for monitoring clinical course in cases of voice disorders. Cleft palate severely affects the structure of the vocal tract. Hence, voice quality can also be also affected. Objective: To study whether the main acoustic parameters of voice, including fundamental frequency, shimmer and jitter are significantly different in patients with a repaired cleft palate, as compared with normal children without speech, language and voice disorders. Materials and methods: Fourteen patients with repaired unilateral cleft lip and palate and persistent or residual velopharyngeal insufficiency (VPI) were studied. A control group was assembled with healthy volunteer subjects matched by age and gender. Hypernasality and nasal emission were perceptually assessed in patients with VPI. Size of the gap as assessed by videonasopharyngoscopy was classified in patients with VPI. Acoustic analysis of voice including Fundamental frequency (F0), shimmer and jitter were compared between patients with VPI and control subjects. Results: F0 was significantly higher in male patients as compared with male controls. Shimmer was significantly higher in patients with VPI regardless of gender. Moreover, patients with moderate VPI showed a significantly higher shimmer perturbation, regardless of gender. Conclusion: Although future research regarding voice disorders in patients with VPI is needed, at the present time it seems reasonable to include strategies for voice therapy in the speech and language pathology intervention plan for patients with VPI.

2015 Elsevier Ireland Ltd. All rights reserved.

1. Introduction

Perceptual assessment of voice is voice is fundamental for the evaluation of patients with voice disorders. Digital acoustic analysis of voice provides instrumental data complementing perceptual assessment. Digital acoustic analysis increases the knowledge concerning specific voice characteristics and it is helpful for understanding the mechanisms of voice production. Moreover, acoustic data are useful for following the patient’s clinical course, documenting clinical improvement and providing visual biofeedback which can be used during the voice intervention [1] .

* Corresponding author. Tel.: +1 248 551 2100; fax: +1 248 551 4692. E-mail address: antonio.ysunza@beaumont.edu (P.A. Ysunza).

0165-5876/ 2015 Elsevier Ireland Ltd. All rights reserved.

Acoustic analysis includes measurement of several parameters. The most important for clinical purposes are Fundamental Frequency (F0), Jitter and Shimmer. FO is determined by the frequency at which the vocal cords vibrate during voice production and it is measured in Hz. Normal values of F0 for children have been previously reported [2] . F0 in normal female children ranges from 268 to 295 Hz. A mean F0 of 247 Hz in children under 12 years of age has also been reported, without significant differences when boys and girls were compared [3] . During consistent vocal cord vibration, F0 demonstrates a slight variation and also a variation of cycle-to-cycle amplitude, these phenomena are known as frequency perturbation or jitter and amplitude perturbation or shimmer. These variations or changes are a consequence of the mass and tension differences which characterize the vocal cords. Jitter and shimmer values can be expressed in a percentage. There are normative data for these percentages. Jitter percentage should be below 1% whereas

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shimmer percentage should be below 10%. It should be considered that shimmer and jitter are dependent of F0. Thus, in cases of abnormal voices with aperiodicity which precludes F0 measure-

ment, shimmer and jitter are not useful [1] . Systematic changes of F0 can occur as a consequence of stress or intonation, which are inherent to speech. These changes affect the acoustic assessment of F0. Thus, the clinical evaluation of F0, shimmer and jitter is usually performed during the production of

a sustained vowel sound. The most acoustically consistent vowel

sound is the vowel/a/ [4,5] . Vocal qualities are the result of individual anatomical and physiological characteristics which are strongly influenced by age, gender and physical structure [6] . Therefore, for an adequate vocal production it is necessary that all the components of the vocal tract, from the glottis to the lips and including the resonance chambers of the nasal cavities and sinuses be structurally and functionally intact. The most common craniofacial anomaly affecting resonance and speech is a palatal cleft. According to Kernahan and Stark [7] , the most common cleft is the total cleft of the primary and secondary palate. Patients with a cleft of the secondary palate show abnormal nasal resonance and nasal emission, which are a consequence of the defective or incomplete seal of the velophar- yngeal sphincter during speech. This anomaly is known as velopharyngeal insufficiency (VPI) and significantly affects speech intelligibility [8] . Besides hypernasality and nasal emission, some patients with cleft palate also develop compensatory behaviors known as compensatory articulation errors. These articulation disorders affect intelligibility even more severely than VPI [9] . Some papers have focused on the acoustic characteristics of voice in patients with cleft palate. Frequency instability, high intensity, abnormal formant distribution and abnormal spectro- grams have been reported [10–12] . In 1989, Zajac and Linvile [11] studied voice perturbations in patients with VPI. They performed acoustic analyses and found a significant relationship between abnormally high jitter values and hypernasality. The purpose of this paper is to study acoustic parameters of voice including F0, shimmer and jitter in children with persistent or residual VPI after surgical repair of a cleft palate. The acoustic parameters of these children were compared with a group of normal controls matched by age and gender, without any voice or speech disorder.

2. Materials and methods

A comparative, cross-sectional study was carried out at the Department of Speech and Language Pathology of the National Institute of Rehabilitation in Mexico City from January 2012 to December 2013. The protocol was approved by the Internal Review Board of the Institute.

2.1. Patients

Patients who met the following criteria were recruited for the study: (a) surgically corrected non-syndromic unilateral total cleft lip and palate (UCLP) with persistent or residual VPI as demonstrated by perceptual analysis of voice and videonasophar- yngoscopy. The patients should present with UCLP as an isolated malformation, not associated with a sequence or syndrome; (b) normal hearing as demonstrated by a behavioral pure – tone audiometry; (c) 6–10 years of age at the time to be recruited for the study. This age group was selected in order to assure appropriate compliance during the behavioral audiometry and the acoustic assessment, as well as to prevent voice changes as

a consequence of puberty. Patients with palatal fistula or with a history of laryngeal pathology or hoarseness were excluded.

Patients present with compensatory articulation errors including glottal stops were excluded from the study group. Patients with neurological disorders or intellectual – cognitive deficits were excluded. Patients with severe nasal emission causing nasal rustle which was being picked up during the recordings procedures were also excluded. During the recruiting period, a total of 14 patients met the criteria for being included in the study. Eleven patients were males and 3 patients were females. The age of the patients ranged from 7 to 9 years of age with a median of 8 years. The study protocol was carefully explained to the parents and/or legal guardians and all of them signed an informed consent form.

2.2. Controls

A control group was assembled, including 14 subjects matched by age and gender. These subjects were recruited at a nearby elementary school. All these subjects voluntarily participated in the study. The study protocol was carefully explained to the parents and/or legal guardians and all of them signed an informed consent form. All of these patients were examined in order to verify that there were no palatal anomalies (including submucous cleft palate), no voice disorders, no compensatory articulation errors and no perceptual signs of VPI (normal nasal resonance during speech).

2.3. Procedure

In patients with repaired cleft palate and persistent VPI, hypernasality and nasal emission were perceptually classified as previously reported [13,14] . Two experienced physicians certified

by the Board of Speech and Language Pathology of Mexico (first and second authors of this paper) were in charge of conducting the examination. The examinations were performed independently.

A Cohen’s Kappa concordance index of 85 was found between both

examiners. Regarding nasal emission a Cohen’s Kappa index of 92 was found. Whenever there was disagreement, each case was discussed until a consensus had been reached. Hypernasality was detected in all patients. Only 4 patients presented with nasal emission across plosive and fricative phonemes. It should be pointed out that none of these patients showed nasal rustle that could be picked up by the microphone during the recording procedures. Eight patients presented with mild hypernasality. Six patients presented with moderate hypernasality. All patients with repaired cleft palate and persistent VPI underwent a flexible nasoendoscopy following a protocol which has been previously reported [15] . Velopharyngeal movements during speech including velar and lateral pharyngeal motion, as well as presence of Passavant’s ridge were classified as ratios, considering the position at rest as 0 and full contact or closure as

1.0. The size of the gap during speech was estimated as a ratio considering total velopharyngeal space at rest as 1.0. Eight patients demonstrated only bubbling during speech without a discernible gap. These cases were conventionally considered as a 10% gap and

a mild VPI. Six patients demonstrated gaps with a size of 20%, that is, a moderate VPI. It should be pointed out that size of the gap as assessed by videonasopharyngoscopy is merely an estimate and not hard data. In other words, inter-observer reliability is not statistically significant. This is why the gaps are conventionally assessed in 10-point intervals (10–20–30%, and so on). It should be pointed out that the endoscope was passed further into the vocal tract in order to assess the vocal folds. All patients showed normal mobility of both vocal folds and no masses were identified in any of the cases. No stroboscopy was performed. Acoustic assessments were performed according to the protocol that was previously reported [16,17] . The patients and subjects

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Table 1 Distribution of acoustic parameters.

3

Variable

Patients

Control subjects

P ; 95% CI

Patients

Control subjects

P ; 95% CI

Males

Females

F0

270.4 Hz

SD 7.01 SD 0.86 SD 0.26

257.9 Hz

SD 6.57 SD 0.21 SD 0.09

0.000

259.1 Hz

SD 35.3 SE 3.5 SD 1.8

240.2 Hz

SD 2.1 SD 0.04 SD 0.07

0.407

Shimmer

5.2

0.78

0.000

7.7

0.6

0.024

Jitter

1.24

0.23

0.000

1.2

1.01

0.826

Table displays mean values and standard deviations (SD) of F0, shimmer and jitter of the groups of patients and control subjects. Patients and subjects were divided by gender for comparison purposes. Probability values ( P ) are also included. A 95% confidence interval was selected for considering P values as statistically significant.

were seated in a straight position on a clinical chair. They were asked to produce a sustained vowel sound (/a/) for at least 3 s at an intensity that was considered as comfortable by the patients and subjects. A Perfect Choice microphone PC-110279 LO2311027 was used at a distance of 10 cm from the lips and an angle of 30 degrees toward the patient. F0, jitter and shimmer were determined.

2.4. Statistical analysis

Conventional central tendency measures, including mean and standard deviation were used for analyzing F0, shimmer and jitter. Acoustic parameters were compared between groups (active – patients and control – subjects) through a non-paired two tailed student t test. The SPSS Statistics 20 software was used for performing the statistical tests.

3. Results

F0 was significantly higher in males from the group of patients as compared with male controls. In contrast, female patients and subjects did not demonstrate a significant difference (see Table 1 ). Shimmer was significantly higher in the group of patients as compared with controls, regardless of gender (see Table 1 ). These data suggest that patients with persistent VPI seem to show higher variability of peak-to-peak amplitude between consecutive periods. Jitter was significantly higher in males from the group of patients as compared with male controls. Female patients and subjects did not demonstrate a significant difference (see Table 1 ). These data suggest that male patients with persistent VPI seem to show higher variability in F0 signal periodicity. When patients present with VPI with nasal emission were compared with patients with VPI without nasal emission, non- significant differences in F0, shimmer or jitter were found. When the patients were stratified according to the severity of VPI (mild to moderate), a significant difference was demonstrated between shimmer values of patients with mild VPI (10% gaps) as compared with patients with moderate VPI (20% gaps). Patients with moderate VPI showed a higher shimmer perturbation. This difference was independent of gender (see Table 2 ).

Table 2 Comparison by severity of VPI.

Variable

VPI

P

Mild

Moderate

F0

263.2

SD 11.9 SD 0.38 SD 0.09

274.4

SD 19.3 SD 2.27 SD 1.2

0.204

Shimmer

4.7

7.1

0.011

Jitter

0.20

0.79

0.207

Table displays mean values and standard deviations (SD) of F0, shimmer and jitter in the group of patients with velopharyngeal insufficiency (VPI). VPI was classified according to size of the gap and hypernasality (mild to moderate). Probability (P ) values are also included. A 95% confidence interval was selected for considering P values as statistically significant.

4. Discussion

From the results of this study it seems that patients present with VPI show vocal abnormalities which were expressed by

differences in F0 and perturbations of voice frequency (jitter) and amplitude (shimmer).

It should be considered that although the group of patients was

homogeneous because of the relatively strict inclusion and exclusion criteria, the number of cases studied herein is reduced, which prevents drawing definite conclusions. The acoustic abnormalities found in patients present with VPI could be explained by the presence of VPI during speech development. VPI occurs most commonly in individuals with a history of cleft palate. Although surgeons attempt to achieve as

much velar lengthening and motion at early ages, through different surgical techniques, 20–40% of patients with cleft palate will present with persistent or residual VPI after surgical palatal repair [18,19] . Thus, if there is a significant VPI during speech development it seems plausible that several patients will show vocal compensatory behaviors for the lack of intraoral pressure.

It should be pointed out that the patients studied for this paper

did not present with compensatory articulation errors. Therefore, the vocal compensatory behaviors mentioned herein seem to be independent of compensatory articulation errors such as glottal stops and pharyngeal fricatives. Patients with VPI showed acoustic abnormalities which may be associated with vocal fold adduction and sudden release. Patients with vocal fold paresis show similar although more severe acoustic findings, but the acoustic findings in patients with VPI may be indicating a low concentration of acoustic energy at the F0 and relative to the overall energy of the signal. Patients present with VPI may be attempting to regulate active vocal tract resistances as compensation. Moreover, increased

glottal resistances during vowel production would decrease air flow and hence facilitate the regulation of subglottal pressure which is essential for continuous phonation [20] .

A possible explanation for some of the acoustic abnormalities

described herein, especially the apparent relationship between severity of VPI and shimmer perturbation, may be that the individual is attempting to regulate subglottal pressure and vocal tract aerodynamics in a condition in which there is insufficient coupling between the resonant nasal cavities and the rest of the vocal tract [21] , potentially increasing vocal noise. Another interesting finding of this study was gender dependent F0 difference. Males showed higher F0 as compared with male controls. Moreover, jitter was also higher in male patients as compared with male controls. These data suggest that male patients with persistent VPI seem to show higher variability in F0 signal periodicity. Van Lierde et al. [22] described vocal quality characteristics in children with cleft palate through a multiparameter approach. They reported that the results of their study supported the hypothesis that vocal quality disorders characterized by a more

negative DSI-value occur in male cleft palate children. These authors also described that decreased vocal quality expressed as

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acoustic abnormalities in male children with cleft palate may be caused by a direct laryngeal pathology or may be an indirect response to VPI, which supports the hypothesis that vocal hyperfunction and abnormal vocal quality in children with VPI represent the children’s attempts to compensate for the VPI. Although age does not affect mean F0, shimmer and jitter are age related. Thus, the rather broad age range of the patients studied in this paper could have affected the acoustic assessments. Moreover, in this study, nasal resonance and nasal emission were assessed perceptually using a previously published classification which has not been entirely validated concerning its reliability. It will be necessary to perform future studies using instrumental assessments including nasometry and aerodynamics measure- ments for assessing nasal resonance and emission.

5. Conclusion

The results of this study suggest that acoustic parameters of voice are significantly affected in patients with VPI. Jitter seems to be more affected in male patients and there seems to be a relationship between severity of VPI and perturbation of shimmer. The vocal abnormalities found in this study seem to be associated with compensatory behaviors related with VPI and they are independent of compensatory articulation errors such as glottal stops. Patients present with VPI may be at risk of developing vocal pathology. Future research regarding vocal disorders in patients with VPI is needed, including collection of vocal tract resonance measures (spectrograms and cepstral peak prominence measure- ments). However, at the present time, it seems reasonable to include strategies for voice therapy in the speech & language pathology intervention plan for patients with VPI.

References

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