ARTICLE IN PRESS
Characteristics of the Voice Handicap Index for Patients With Unilateral Vocal Fold Paralysis Who Underwent Arytenoid Adduction D1X XKenichi WatanabeD2X X, D3X XTakeshi SatoD4X X, Yohei D5X X HonkuraD6X X, D7X XAi Kawamoto-HiranoD8X X, Kazutaka D9X X KashimaD10X X, and D1X XYukio KatoriD12X X, Sendai, Miyagi, Japan
Summary: Purpose. This study was performed to evaluate the characteristics of the Voice Handicap Index (VHI), a self-assessment measure, for patients with unilateral vocal fold paralysis (UVFP) who underwent arytenoid adduction (AA), in comparison with postoperative vocal function examinations. Methods. A retrospective chart review was conducted for patients who underwent AA at Tohoku University Hospital during the period between 2014 and 2017. VHI was compared before and after surgery; moreover, correlations were assessed between the VHI and other voice measurements, including perceptual assessment of voice, as well as aerodynamic and acoustic measures. Factors involved in the VHI score were explored by multivariate analysis. Results. Forty-three UVFP patients (28 males, age 32−81 years; 15 females, age 34−80 years) were enrolled in the study; the average age of all patients was 61.5 years (32−81 years). Among the enrolled patients, 33 (76.7%) left and 10 (23.3%) right vocal folds were impaired. After surgery, nearly all of the patients exhibited significantly improved VHI score; each of the three subscales (functional, physical, and emotional) was also improved. The postoperative VHI correlated mildly with several values of the other voice measurements, with the exception of the mean flow rate. Multivariate analysis showed that the sole variable associated with postoperative VHI score was preoperative VHI. Conclusions. The postoperative VHI likely reflects improvement in the voices of the patients with UVFP. Although there were weak correlations with other voice measures, postoperative VHI is a relatively independent measurement parameter for patients with UVFP who underwent AA. Key Words: Arytenoid adduction−Voice Handicap Index−VHI−Unilateral vocal fold paralysis−UVFP.
INTRODUCTION Unilateral vocal fold paralysis (UVFP) is a common problem encountered by the otolaryngologist. Incomplete glottal closure caused by UVFP can lead to symptoms of moderate to severe breathy hoarseness and dysphagia, which eventually influence an individual’s quality of life (QOL). Some authors have reported that patients with UVFP showed a higher voice handicap than patients with other types of organic dysphonia, such as laryngeal nodule, polyp, or cyst.1,2 Hence, several types of interventional options have been developed for the treatment of UVFP, including medialization laryngoplasty (ML), injection laryngoplasty, arytenoid adduction (AA), and laryngeal reinnervation. Among these interventions, AA is notable in that it can medialize and close the large posterior glottic gap; moreover, it can correct vocal fold height mismatch by adducting and rotating the arytenoid cartilage of the UVFP. Because the paralyzed arytenoid is immobilized through AA, the Accepted for publication December 20, 2018. Funding: This research was supported by the Japanese Foundation for Research and Promotion of Endoscopy Grant. Conflicts of interest: The authors have nothing to disclose. From the Department of Otolaryngology − Head and Neck Surgery, Tohoku University Graduate School of Medicine, Sendai, Miyagi 980-8574, Japan. Address correspondence and reprint requests to Kenichi Watanabe, Department of Otolaryngology − Head and Neck Surgery, Tohoku University Graduate School of Medicine, 1-1 Seiryo-machi, Aoba-ku, Sendai, Miyagi 980-8574, Japan. E-mail:
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vocal fold can develop strong resistance to expiration and contact from the healthy side during phonation.3 In addition, to compensate for atrophic change or bowing of the membranous portion, AA in combination with ML has recently been recommended by several clinicians.4−7 Nevertheless, because the dynamic state of the postoperative glottis during phonation is assumed to differ from the state of the healthy glottis, we consider the phonation of patients with UVFP who underwent AA to be a unique condition. Because there is no method for absolute evaluation of voice disorders, it is necessary to evaluate voice function in a multidimensional manner, including perceptual evaluation, as well as subjective and objective measurements. Historically, the emphasis has been on objective voice measurements: videolaryngostroboscopy, perceptual rating, aerodynamic measurement, and acoustic parameters. In 1997, Jacobson et al8 proposed the Voice Handicap Index (VHI) scale, a self-assessment approach, in order to evaluate the impact of voice disorders on patients’ physiological, social, and psychological function. VHI is a common questionnaire used in management of a wide range of voice disorders; importantly, it is the most widely applicable subjective self-rating questionnaire for patients who have perceived voice disability. It shows the effect of disabilities resulting from voice handicap on QOL. The VHI provides an evaluation based on a patient’s own perception of his or her disease, thus enabling treatment planning, as well as pre- and post-treatment evaluation.
ARTICLE IN PRESS 2 Previous studies examined the association between VHI score and acoustic measures; these studies revealed that acoustic measures were not predictive of the overall VHI score, despite the presence of a weak correlation between items of the VHI and some voice parameters.9−11 However, the patient populations included in these prior investigations were not homogenous and included voice disorders of different origins. Schindler et al12 reported that the patients were divided into four groups: functional dysphonia, UVFP, structural dysphonia, and nodules; they concluded that correlations between some laboratory measurements and VHI may exist in patients with dysphonia of the same origin. Conversely, another study showed a moderate correlation between perceptual examination of voice quality and subjective patients’ rating in patients with voice disorderes.13 In this study, we aimed to evaluate the characteristics of VHI scores for patients with UVFP who underwent AA in comparison with postoperative vocal function examinations, including perceptual evaluation, aerodynamic and acoustic measurements. Our results may provide further information regarding voice QOL of patients with UVFP who exhibit postoperative improvement in glottis gap insufficiency. MATERIAL AND METHODS This retrospective study was approved by the ethics committee of Tohoku University School of Medicine (20181-36). Participants Patients with UVFP who underwent AA at Tohoku University Hospital during the period between 2014 and 2017 were enrolled in this study. They received a series of voice evaluations, both preoperatively and approximately 6 months after surgery. Exclusion criteria included revision surgery and incomplete surgery (eg, due to laryngeal scarring). Voice evaluation Subjective voice quality assessment An experienced otolaryngologist and speech-languagehearing therapist, both of whom were not blinded, independently evaluated voice quality on the basis of the grade of hoarseness (G) in the GRBAS scale, which was proposed by the Japanese Society for Logopedics and Phoniatrics. Voice dysfunction severity was rated at one of four levels: 0 for normal, 1 for mild, 2 for moderate, and 3 for severe hoarseness. Patient self-assessment On the VHI scale, the impact of voice disorder on patients’ QOL is based on the assessment of functional (F), physiological (P), and emotional (E) subscale scores, as well as the total score (T) of the three subscales. A Japanese version of the VHI was used.2 Each subscale is based on 10 questions. Patients were asked to rate each question based on the
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frequency of occurrence: 0 for never, 1 for seldom, 2 for sometimes, 3 for regularly, and 4 for always. The score of each subscale ranged from 0 to 40, and the total score (T) ranged from 0 to 120. A higher total score indicated worse self-recognition of the patient with regard to voice disorder. Phonation function evaluation Aerodynamic and acoustic measures were performed on the Phonatory Aerodynamic Analyzer PA-1000 (Minato, Osaka, Japan) and CLS4500 (KayPentax, Tokyo, Japan), respectively, by an experienced speech-language-hearing therapist. All recordings were performed in a soundproof room. Among the aerodynamic data obtained by having the patient phonate /a/, mean flow rate (MFR) and maximum phonation time (MPT) were calculated. MPT was obtained by measuring the time of three trials in which the patient performed for as long as possible. Spectrography analysis was obtained of the sustained vowel /a/ at a comfortable loudness. From the acoustic measurements, Jitter, Shimmer, and noise-to-harmonic ratio (NHR) were analyzed. Statistical analysis We conducted unpaired t test, paired t test, or nonparametric Mann-Whitney rank-sum test to detect differences between groups. Correlations were evaluated using the Pearson correlation coefficient. A multivariate analysis was performed, using stepwise multiple linear regression with the variables that impacted the value of VHI. Statistical analyses were conducted using SPSS statistics version 21.0 (SPSS Inc., Chicago, Illinois). RESULTS Forty-three UVFP patients were enrolled in this study (Table 1). The average age of the patients was 61.5 years (32−81 years): 28 were men (32−81 years) and 15 were women (34−80 years). Among the patients, 33 left and 10 right vocal folds were impaired. Four surgeons were involved in these operations. Most AA surgeries were performed in combination with ML, also known as thyroplasty type 1; some AA surgeries were performed with nerve-muscle pedicle flap implantation.14 Regarding the causes of UVFP, most frequent were cardiovascular diseases (11 patients) and lung cancer (11 patients), followed thyroid diseases (nine patients). Comparison of pre- and postvoice examinations After AA surgery, nearly all of the 43 patients exhibited significant lowering of VHI; average preoperative VHI was 70.9, while average postoperative VHI was 20.6 (Table 2). Each subscale of VHI also improved. Other voice measures, including perceptual examination G, MPT, MFR, Jitter, Shimmer, and NHR, also showed statistically significant changes between pre- and postoperative values (Table 2).
ARTICLE IN PRESS Kenichi Watanabe, et al
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VHI for Patients with UVFP Who Underwent Arytenoid Adduction
There were several mild correlations between postoperative VHI score, including total or subscale, and perceptual examination G, MPT, Jitter, Shimmer, NHR, and preoperative VHI; however, there was no significant correlation between postoperative VHI and MFR. Furthermore, the VHI-E subscale did not show any correlation with other voice measures. In separate analyses of men and women, there was mild significant correlation between postoperative VHI score and MFR for men (Table 5). For women, postoperative VHI score showed no correlation with objective voice measures, whereas there was a mild correlation with preoperative VHI score (Table 6).
TABLE 1. Characteristics of Patients With UVFP Who Underwent AA Number Sex, no. (%) Male Female Years of age, average Months of follow-up before AA, median Side, no. (%) Right Left Causes of UVFP Cardiovascular diseases Lung cancer Thyroid diseases Esophagus diseases Jugular foramen syndrome Idiopathic Others Combined with TP1 NMP
43 28 (65.1%) 15 (34.9%) 61.5 (32-81) 15 (4- over 360) 10 (23.3%) 33 (76.7%) 11 (25.6%) 11 (25.6%) 9 (20.9%) 2 (4.7%) 2 (4.7%) 2 (4.7%) 6 (14.0%) 39 (90.7%) 4 (9.3%)
Abbreviations: AA, arytenoid adduction; NMP, nerve-muscle pedicle flap implantation; TP1, thyroplasty type 1; UVFP, unilateral vocal fold paralysis.
Comparison of postoperative voice examinations by sex After AA surgery, VHI scores were higher among women than among men, but this difference was not statistically significant. For other voice measures, there were no significant differences, with the exception of MFR, between sexes (Table 3). The MFR of the women was significantly lower than that of the men. Correlation between VHI and other voice measurements after AA surgery Correlations between postoperative VHI and other voice measurements plus preoperative VHI are shown in Table 4.
Multivariate analysis In order to identify which independent variables might explain postoperative VHI score, multiple regression analyses were performed. The variables analyzed were preoperative VHI, as well as postoperative objective voice evaluation involving MPT, MFR, Jitter, Shimmer, and NHR. Stepwise linear regression analysis showed that the coefficient of determination for the generated model (R2) was 0.283 (P = 0.022) with two variables: preoperative VHI and postoperative NHR. However, the sole significant variable that could explain postoperative VHI score was preoperative VHI (b = 0.435, P = 0.023), not NHR (b = 0.358, P = 0.056). DISCUSSION Patients with UVFP show a wide range of dysphonia, including both breathy and harsh; these symptoms impair QOL. To improve the glottal insufficiency with UVFP, AA, which was described by Isshiki and Tanabe4, can medialize and close the large posterior glottic gap; moreover, it can correct the physiological vocal fold height mismatch by adducting and rotating the arytenoid cartilage of the UVFP. Currently, AA in combination with ML has been recommended by several clinicians, rather than AA alone.4−7 Slavit and Maragos5 concluded that the AA procedure could
TABLE 2. Comparison of Each Voice Examination Before and After Surgery
VHI VHI-F VHI-P VHI-E Perceptual examination G MPT (s) MFR (mL/s) Jitter% Shimmer% NHR
Preoperative
Postoperative
P Value
70.9 § 22.1 25.9 § 7.3 24.6 § 7.6 20.4 § 9.0 2 [2-3] 3.6 § 1.7 713.9 § 345.0 6.235 § 4.444 14.206 § 10.944 0.447 § 0.349
20.6 § 18.9 7.1 § 6.5 8.5 § 7.2 5.0 § 6.6 1 [0-1] 15.0 § 8.1 216.7 § 114.8 2.544 § 1.961 7.148 § 4.479 0.197 § 0.117
<0.01 <0.01 <0.01 <0.01 <0.01 <0.01 <0.01 <0.01 <0.01 <0.01
Paired t test Paired t test Paired t test Paired t test Wilcoxon signed-rank test Paired t test Paired t test Paired t test Paired t test Paired t test
Abbreviations: MFR, mean flow rate; MPT, maximum phonation time; NHR, noise-to-harmonic ratio; VHI, Voice Handicap Index.
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TABLE 3. Comparison of Each Postoperative Voice Examination Between Males and Females
VHI-Total VHI-F VHI-P VHI-E G MPT MFR Jitter Shimmer NHR
Male (n = 28)
Female (n = 15)
16.8 § 17.8 5.8 § 6.1 7.1 § 7.0 4.0 § 6.0 1 [0−1] 15.1 § 8.8 256.5 § 125.1 2.327 § 1.833 6.918 § 4.048 0.178 § 0.061
25.6 § 20.1 8.8 § 7.1 10.5 § 7.2 6.3 § 7.4 1 [0−1] 14.7 § 7.4 150.3 § 49.7 2.786 § 2.181 7.303 § 5.212 0.220 § 0.172
P = 0.205 P = 0.212 P = 0.192 P = 0.327 P = 0.514 P = 0.902 P = 0.005 P = 0.488 P = 0.800 P = 0.290
Not significant Not significant Not significant Not significant Not significant Not significant Significant difference Not significant Not significant Not significant
Unpaired t test Unpaired t test Unpaired t test Unpaired t test Mann-Whitney U test Unpaired t test Unpaired t test Unpaired t test Unpaired t test Unpaired t test
Abbreviations: MFR, mean flow rate; MPT, maximum phonation time; NHR, noise-to-harmonic ratio; VHI, Voice Handicap Index.
TABLE 4. Correlation Between Total VHI as well as 3 Subscales and Voice Examination in All Subjects
G MPT MFR Jitter Shimmer NHR Pre-VHI
VHI-Total
VHI-F
VHI-P
VHI-E
0.365* ¡0.386* 0.246 0.326 0.297 0.302 0.395*
0.445* ¡0.411* 0.218 0.432* 0.426* 0.463** 0.308
0.364* ¡0.363* 0.23 0.322 0.328 0.284 0.382*
0.213 ¡0.308 0.241 0.158 0.074 0.099 0.408*
TABLE 6. Correlation Between Total VHI as well as 3 Subscales and Voice Examination in Females
G MPT MFR Jitter Shimmer NHR Pre-VHI
VHI-Total
VHI-F
VHI-P
VHI-E
0.501 ¡0.333 0.16 0.264 0.278 0.265 0.626*
0.606* ¡0.319 ¡0.027 0.424 0.504 0.504 0.509
0.538 ¡0.327 0.195 0.306 0.383 0.301 0.626*
0.257 ¡0.282 0.273 0.013 ¡0.098 ¡0.055 0.643*
Pearson correlation, significant values: *P < 0.05, **P < 0.01. Abbreviations: MFR, mean flow rate; MPT, maximum phonation time; NHR, noise-to-harmonic ratio; VHI, Voice Handicap Index.
Pearson correlation, significant values: *P < 0.05, **P < 0.01. Abbreviations: MFR, mean flow rate; MPT, maximum phonation time; NHR, noise-to-harmonic ratio; VHI, Voice Handicap Index.
correct a large posterior glottic gap; furthermore, in a patient with marked vocal cord bowing, AA combined with ML was an effective technique. McCulloch et al6 evaluated the voice outcomes of patients who underwent ML and MLAA; that study showed that the magnitude of improvement in multiple voice parameters was greater in the ML-AA patients. Mortensen et al7 reported that the AA corrected the physiology of the incompetent larynx better than ML
alone, resulting in a statistically greater degree of change of acoustic and aerodynamic parameters. Because there is no consensus regarding which voice outcome indicators should be used to evaluate the results of interventions for UVFP, voice quality assessment has been generally accepted as multidimensional by nature. A review by Daniero et al15 revealed that although most studies have measured results using objective measures alone, patient self-reported outcomes have been largely omitted: only 23% of prior studies considered patient-reported outcomes. Another review by Desuter et al16 concluded that most frequently used voice outcome indicators to evaluate the various surgical treatments for UVFP were MPT, MFR, and GRBAS; these measures were the most frequently used and most relevant. VHI showed a relatively low rate of use (11th most frequent) and had a low “percentage of significance.”16 A report by Franco and Andrus17 noted that the objective data, such as acoustic and aerodynamic measures, were effort-dependent, and that these may not be reliable tools for measuring postsurgical voice outcomes; thus, patient surveys, including VHI, may be the best instrument to measure outcomes of surgical intervention. Daniero et al15 indicated that the patient’s subjective assessment is critical; in some ways, it is the ultimate measure of success.
TABLE 5. Correlation Between Total VHI as well as 3 Subscales and Voice Examination in Males
G MPT MFR Jitter Shimmer NHR Pre-VHI
VHI-Total
VHI-F
VHI-P
VHI-E
0.265 ¡0.399 0.527* 0.334 0.283 0.35 ¡0.057
0.332 ¡0.445* 0.538* 0.398 0.331 0.424 ¡0.059
0.247 ¡0.358 0.483* 0.288 0.258 0.237 0.019
0.162 ¡0.318 0.458* 0.254 0.205 0.335 ¡0.140
Pearson correlation, significant values: *P < 0.05, **P < 0.01. Abbreviations: MFR, mean flow rate; MPT, maximum phonation time; NHR, noise-to-harmonic ratio; VHI, Voice Handicap Index.
ARTICLE IN PRESS Kenichi Watanabe, et al
VHI for Patients with UVFP Who Underwent Arytenoid Adduction
In this study, we showed that VHI score decreased in most subjects. The mean reduction between pre- and postoperative VHI was 50.3 points, which was significantly different. Similarly, for each subscale, significant changes were observed. The VHI was first developed to quantify the patients’ perception of disability related to voice disorders; however, it also has been revealed as a useful instrument to assess treatment efficacy for voice disorders.1,2 Thus, the postoperative result was almost favorable for our patients with UVFP who underwent AA. Nevertheless, it is important to note that the acquired voice function differs from normal voice function. Although the threshold between individuals with dysphonia and those with a healthy voice has not been established in a Japanese version of the VHI, some studies have suggested different cut-off values: for example, 12 points were suggested in a Polish version,18 14.5 points in a Persian version,19 and 19 points in the original version in Brazil.20 Because the average postoperative VHI score was not below the cutoff value, patients with UVFP who underwent AA continued to experience some degree of voice problems in daily life. In a comparison of postoperative voice examinations on the basis of sex, the VHI score of females was higher than that of males; VHI subscales showed a similar trend. These results were consistent with those of some previous reports, in that the VHI of dysphonic females was higher than that of dysphonic males, regardless of the treatment provided.2,21,22 There were no significant differences in other voice examinations, with the exception of MFR, on the basis of sex. The observation that the MFR of males was significantly higher than that of females could be explained as follows: a statistically significant difference between males and females, with respect to the MFR of normal subjects, has been reported23; this is reasonable, considering that males have a larger larynx and longer vocal fold length. In addition, surgical difficulty in performing AA for males, due to the larger lamina and sharper angle in males, might result in an inability to lower MFR. The relationship between VHI and other voice measurements has not yet been established; however, there was a report that a correlation may exist in patients with dysphonia of the same origin.12 Because we consider patients with UVFP after AA surgery to exhibit a unique condition, we analyzed the correlation between VHI and other voice measurements in these patients. In our study, postoperative VHI score and its subscales correlated mildly with perceptual examination G, MPT, Jitter, Shimmer, and NHR, but not with MFR. Next, when examining the correlations between VHI score and other voice examinations on the basis of sex, postoperative VHI score showed no correlation with MFR of females, but did exhibit a correlation in males. It is difficult to interpret this sex difference; however, this is not an indicator that a lower MFR is associated with a lower VHI score. Thus, a lower MFR is not necessarily associated with a higher degree of voice quality in the females. Because the average value of MFR of females was lower than that of males, and the standard deviation was
5
also smaller, the differences in MFR of females among individuals might fade; thus, other factors might have a greater influence on VHI. Additionally, in females, postoperative VHI solely correlated with preoperative VHI. There seem to be stronger individual differences in the voice evaluation of females. A stepwise multiple linear regression was completed with dependent variables for predicting the total VHI score; notably, preoperative VHI was the sole significant factor included in the final model, but it explained only 28.3% of the variability in total VHI score. Therefore, we concluded that the objective voice examinations could not adequately explain the postoperative VHI score. Postoperative VHI score is thus a relatively independent factor for voice assessment in the evaluation of patients with UVFP who underwent AA. The limitations of this study include its retrospective nature, relatively small sample size, and the unequal numbers of male and female patients. Our stratification of the data by sex, based on differences in vocal function, contributed to the limited sample size. However, our study is important in that it enhances the collective knowledge regarding UVFP after surgery. The factors that influence the perception of voice handicap can include patient personality, rate of disease progression, past experiences with vocal function, occupation, social status, and other aspects.9 Considering the established nature of VHI, which reflects the influence of voice function in an individual patient’s life, it is reasonable to expect that it will be evaluated differently from voice examinations measured in this study. Generally, AA is considered to improve the posterior glottis chink and differences in vocal fold levels during vocalization. Moreover, AA can restore the two-point support structure of the vocal fold; resistance to exhaled air will increase, thereby preventing passive dislocation of the affected arytenoid cartilage by the contralateral arytenoid.24 However, although MPT and MFR improve if the glottis narrows, this narrowing does not necessarily correlate with auditory impressions or pitch regulation.24 Because the thyroarytenoid muscle does not function to relax and shorten the vocal fold during vocalization, pitch control would be difficult for patients with UVFP who underwent AA, despite preservation of cricothyroid muscle function. In a recent article, the authors suggested that assessment of pitch range might provide an integrated view into total vocal function, given that other parameters are limited to assessment of the quality of a single tone.25 We must carefully assess sustained vowel production during the voice examination, which differs from connected speech in daily conversation. CONCLUSIONS In patients with UVFP who underwent AA, the postoperative VHI score remarkably improved, which indicated an acceptable result. Postoperative VHI showed weak correlations with other voice examinations, including perceptual examination G, MPT, Jitter, Shimmer, and NHR; moreover, it solely correlated with MFR of males. Postoperative VHI, which is an important indicator, is thus a relatively
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