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Voice, Speech, and Laryngeal Features of Primary Sjögren's Syndrome Amanda Heller, Kristine Tanner, Nelson Roy, Shawn L. Nissen, Ray M. Merrill, Karla L. Miller, Daniel R. Houtz, Julia Ellerston and Katherine Kendall Ann Otol Rhinol Laryngol published online 18 June 2014 DOI: 10.1177/0003489414538762 The online version of this article can be found at: http://aor.sagepub.com/content/early/2014/06/16/0003489414538762
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AORXXX10.1177/0003489414538762Annals of Otology, Rhinology & LaryngologyHeller et al
Article
Voice, Speech, and Laryngeal Features of Primary Sjögren’s Syndrome
Annals of Otology, Rhinology & Laryngology 1–8 © The Author(s) 2014 Reprints and permissions: sagepub.com/journalsPermissions.nav DOI: 10.1177/0003489414538762 aor.sagepub.com
Amanda Heller, MS1,2, Kristine Tanner, PhD2,3, Nelson Roy, PhD1,2, Shawn L. Nissen, PhD3, Ray M. Merrill, PhD, MPH4, Karla L. Miller, MD5, Daniel R. Houtz, MA2, Julia Ellerston, MA2, and Katherine Kendall, MD2,6
Abstract Objective: This study examined voice, speech, and laryngeal characteristics in primary Sjögren’s syndrome (pSS). Methods: Eleven patients (10 female, 1 male; mean [SD] age = 57 [14] years) from The University of Utah Division of Rheumatology provided connected speech and sustained vowel samples. Analyses included the Multi-Dimensional Voice Profile, the Analysis of Dysphonia in Speech and Voice, and dysphonia severity, speech clarity, and videolaryngostroboscopy ratings. Results: Shimmer, amplitude perturbation quotient, and average fundamental frequency differed significantly from normative values (P < .01). Cepstral Spectral Index of Dysphonia values indicated mild-to-moderate dysphonia in connected speech (mean [SD] = 20.26 [8.36]) and sustained vowels (mean [SD] = 16.91 [11.08]). Ratings of dysphonia severity and speech clarity using 10-cm visual analog scales suggested mild-to-moderate dysphonia in connected speech (mean [SD] = 2.11 [1.72]) and sustained vowels (mean [SD] = 3.13 [2.20]) and mildly reduced speech clarity (mean [SD] = 1.46 [1.36]). Videolaryngostroboscopic ratings indicated mild-to-moderate dryness and mild reductions in overall laryngeal function. Voice Handicap Index scores indicated mild-to-moderate voice symptoms (mean [SD] = 43 [23]). Conclusion: Individuals with pSS may experience dysphonia and articulatory imprecision, typically in the mild-to-moderate range. These findings have implications for diagnostic and referral practices in pSS. Keywords primary Sjögren’s syndrome (pSS), voice disorders, speech clarity, autoimmune disease
Introduction Primary Sjögren’s syndrome (pSS) is an autoimmune disease characterized by reduced exocrine gland secretion, causing chronic dryness—or sicca—symptoms. The most common features of pSS include xerostomia and keratoconjunctivitis sicca due to poor function of the salivary and lacrimal glands.1 Dryness of the nose, pharynx, larynx, trachea, bronchi, vagina, and skin is common. Additional systemic effects may include peripheral and cranial neuropathy, renal dysfunction, pancreatitis, vasculitis, glandular infections, parotid tumors, and lymphoma. Other symptoms include focal or generalized pain, stiffness, swelling, and fatigue.2-4 Recent epidemiologic reports indicate that pSS occurs in approximately 0.5% to 1% of the population. Risk rates are 9 to 1 female to male, occurring most frequently in mid-life, although pediatric cases also exist. In contrast to secondary SS, pSS occurs independently of other inflammatory autoimmune diseases.5 Individuals with SS can have swallowing, voice, and speech problems. Although the dysphagia literature is more
developed in this population, only infrequent reports of voice and speech problems exist or are inferred from associated mouth and throat symptoms. Because secondary SS is more prevalent, these studies include either a diverse group 1
Department of Communication Sciences & Disorders, The University of Utah, Salt Lake City, Utah, USA 2 Voice Disorders Center, The University of Utah, Salt Lake City, Utah, USA 3 Department of Communication Disorders, Brigham Young University, Provo, Utah, USA 4 Department of Health Science, Brigham Young University, Provo, Utah, USA 5 Division of Rheumatology, The University of Utah School of Medicine, Salt Lake City, Utah, USA 6 Division of Otolaryngology–Head & Neck Surgery, The University of Utah School of Medicine, Salt Lake City, Utah, USA Corresponding Author: Kristine Tanner, PhD, Department of Communication Disorders, Brigham Young University, 1190 N 900 E, 158 TLRB, Provo, UT 84602, USA. Email: [email protected]
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of both primary and secondary SS or exclusively secondary SS cohorts.6-11 Among these studies, SS has been associated with hoarseness, roughness, strain, and articulatory imprecision. Videolaryngostroboscopic findings are diverse and include supraglottic adduction, vocal fold lesions, weakness, edema, pseudosulcus, inflammation, dryness, and thick mucus. Lingual weakness, tongue deviation, labial asymmetry, lingual adhesion to the palate, and palate- pharyngeal adhesion as well as severe oropharyngeal mucosal dryness and loss of dentition have been observed. Voice- and speech-related patient symptoms include increased vocal effort, throat dryness, cough, pitch breaks, hypernasality, and decreased loudness. Atypical voice and speech acoustic patterns have also been observed. Unfortunately, the relative rarity of pSS has prevented systematic investigations of voice and speech problems specific to this population. The purpose of this study was to characterize voice, speech, and laryngeal problems in patients with pSS. To our knowledge, no studies exist that have examined voice and speech production exclusive to this population. This next step is essential, because secondary SS, by definition, is the result of another primary autoimmune disease, such as rheumatoid arthritis or lupus erythematosus. Due to the overlapping manifestations of these autoimmune diseases (eg, bamboo nodules or inflammation), it is difficult in cases of secondary SS to know which symptoms are uniquely associated with each disease process(es). The pure effects of pSS on voice and speech production warrant further investigation. To that end, this study examined acoustic, auditory-perceptual, and laryngeal characteristics of pSS.
Methods Participants Eleven individuals with pSS (10 female, 1 male; mean [SD] age = 57 [14] years) were recruited from The University of Utah Division of Rheumatology treatment-seeking population. Inclusion criteria were based on the presence of sicca symptoms, antinuclear antibody testing, and/or lip biopsy. Participants were included only if SS was the primary causative factor attributed to their sicca symptoms. All participants were receiving medical management of SS, including disease-related medications. The male participant reported a history of smoking 1 pack of cigarettes per day for more than 30 years. Participant mean total score on the EULAR (European League Against Rheumatism) Sjögren’s Syndrome Patient Reported Index (ESSPRI)12—a 3-domain patient-based symptom severity scale including dryness, pain, and fatigue—was 5.4 out of 10 (SD = 2.1). Audio recordings were collected using a Canon VIXIA HD Camcorder (HF S21) and Shure (SM 48) cardioid
microphone with a mouth-to-mic distance of 4 inches. Samples consisted of the second and third sentences of “The Rainbow Passage”13 and the central 3 seconds of a sustained vowel /ɑ/ production collected on 2 consecutive weeks. Samples were extracted using Adobe Audition (version 3.0; Adobe, San Jose, California, USA) at a sample rate of 44 100 Hz. Numeric data for acoustic variables were averaged across weeks for repeated samples.14 Nine of the 11 participants tolerated rigid videolaryngostroboscopy without topical anesthesia.
Acoustic Analyses Sustained vowel recordings were analyzed using the MultiDimensional Voice Program (MDVP Advanced for MultiSpeech, Model 5105; KayPentax, Montvale, New Jersey, USA). Voice parameters sampled included those previously shown or theorized to be sensitive to voice problems in SS,9 including jitter (%), shimmer (%), pitch perturbation quotient (PPQ), amplitude perturbation quotient (APQ), and average fundamental frequency (AFF). Jitter and PPQ are sensitive to cycle-to-cycle variations in frequency, whereas shimmer and APQ are sensitive to cycle-to-cycle variations in amplitude of the voice signal. Sustained vowel and connected speech recordings were analyzed using the Analysis of Dysphonia in Speech and Voice (ADSV, version 5109; KayPentax, Montvale, New Jersey, USA), which subsequently generates the Cepstral Spectral Index of Dysphonia (CSID). The CSID is an acoustic measure of dysphonia severity, derived from spectral and cepstral measurements. The program automatically generates a CSID value for sustained vowels but requires a manual input of the cepstral peak prominence (CPP), the low/high (L/H) spectral ratio, and the L/H spectral ratio standard deviation to compute a value for connected speech. This measure has been established to be highly associated with listener ratings of dysphonia severity and is sensitive and specific to heterogeneous voice qualities and severities.15
Auditory-Perceptual Ratings Eight speech-language pathologists with extensive experience in voice disorder assessment and treatment completed auditory-perceptual ratings of dysphonia severity (sustained vowel and connected speech) and speech clarity (connected speech). The identical samples selected for acoustic analysis were also used for auditory-perceptual ratings but were normalized for loudness in the listening task. Samples were presented via Sennheiser HD 558 headphones. Three samples were presented prior to beginning the ratings to orient the listener to the range and variety of samples. Ratings were accomplished using a 10-cm visual analog scale (VAS). The extreme left (ie, 0 cm) represented normal voice
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Heller et al and extreme right (ie, 10 cm) represented profoundly abnormal voice. Following the dysphonia severity ratings, listeners rated speech clarity using a 10-cm VAS. Speech clarity was defined as how clear, or distinct, the listener perceived the words in the connected speech sample to be.16 The extreme left represented typical speech clarity (all the words sounded distinct, clear, or pure) and the extreme right represented profoundly abnormal speech clarity (all of the words sounded muffled or blurred). Listeners also rated 20% of samples a second time to estimate intrajudge reliability.
Videolaryngostroboscopy One expert laryngologist (K.K.) with more than 20 years of experience in assessment and management of voice disorders assessed the 9 rigid videolaryngostroboscopy exams. The ratings evaluated structural, vibratory, and mucosal laryngeal features. Parameters assessed included vocal fold edges (straight vs bowed), vocal fold texture (normal vs rough), vertical level (on plane vs off plane), vascular markings (none, capillary ectasia, microvarices, hemorrhage), mobility (normal, reduced, absent), length of vocal folds at normal pitch normal volume (NPNV; symmetrical, left shorter, right shorter), length of vocal folds at high pitch normal volume (HPNV; symmetrical, left shorter, right shorter), glottic closure (complete, anterior chink, posterior chink, spindle, hourglass, incomplete), phase symmetry (symmetrical, asymmetrical), vibratory behavior including mucosal wave and amplitude of vibration (normal, reduced, absent), supraglottic activity (none, mild, moderate, or severe mediolateral and/or anterior-posterior compression), mucus of the pharynx (thin, thick, foamy, frothy, transient, white, yellow, clear, tacky), and mucus of the vocal folds (thin, thick, foamy, frothy, transient, white, yellow, clear, tacky). An overall rating of laryngeal appearance was made using a 10-cm VAS, with the extreme left of the scale indicating a normal laryngeal appearance and the extreme right representing a profoundly abnormal laryngeal appearance. The same method of rating was completed to evaluate overall dryness of the pharynx, larynx, and vocal folds; the extreme left indicated normal appearance and extreme right indicated severe dryness.
Statistical Analysis Summary statistics, including means, standard deviations, and ranges, were used to explore all variables. Relationships among variables were evaluated using Pearson correlations. Differences between acoustic variables and normative values were evaluated using 1-sample t tests with a Bonferroni correction for multiple comparisons. Agreement among raters for auditory-perceptual ratings was evaluated using intraclass correlation coefficients (ICCs); intrarater agreement was evaluated using multiple regression analysis.
Analyses were performed with the Statistical Analysis System (SAS) software, version 9.4 (2013; SAS Institute Inc, Cary, North Carolina, USA).
Results Acoustic Analysis Statistically significant differences in shimmer (%; P = .004), APQ (P = .003), and AFF (P = .001) were observed between pSS participants and normative values based on 1-sample t tests (Bonferroni correction = 0.050/5). Differences in jitter (%) approached statistical significance (P = .015). Differences in PPQ were not statistically significant. CSID values ranged from mild to mild-to-moderate dysphonia for sustained vowels and connected speech samples. Summary data are provided in Table 1. Pearson correlations ranged from 0.970 to 1.000 for 10% repeated acoustic analyses (P = .001-.022), indicating acceptable measurement reliability.
Auditory-Perceptual Ratings Assessment of reliability of listener ratings of dysphonia severity (P < .050) revealed ICCs of 0.939 (95% confidence interval [CI], 0.864-0.981) for sustained vowels and 0.821 (95% CI, 0.599-0.944) for connected speech, indicating acceptable interjudge reliability. For speech clarity, the ICC was 0.962 (95% CI, 0.915-0.988), also indicating acceptable reliability. Regression analysis showed a strong correlation between repeated ratings of voice severity on vowels (slope = 0.85, P = .001) and speech clarity (slope = 0.90, P = .001); an adequate correlation was observed for voice severity during connected speech (slope = 0.42, P = .001). These results confirm acceptable inter- and intra-listener reliability and provide confidence in the reported results that follow. For sustained vowel VAS ratings, mean voice severity was 3.13 cm (SD = 1.51 cm; range, 0-9.75 cm); for connected speech VAS ratings, mean voice severity was 2.11 cm (SD = 0.61 cm; range, 0-8.55 cm). Mean speech clarity based on the VAS was 1.47 cm (SD = 1.22 cm; range, 0-8.75 cm). Auditory-perceptual ratings for voice severity and speech clarity are illustrated in Figure 1.
Videolaryngostroboscopic Ratings Videolaryngostroboscopic ratings based on structural, vibratory, and mucosal parameters are presented in Table 2. The most common laryngeal findings were the presence of supraglottic adduction (ie, compression), thick mucus, and vascular markings. Vocal fold vibratory and mobility parameters were essentially normal. Visual analog scale ratings of pharyngeal, laryngeal, and vocal fold dryness are illustrated in Figure 2. Ratings ranged from 1 to 6.25 cm for
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Table 1. Select Multi-Dimensional Voice Program and Analysis of Dysphonia in Speech and Voice Acoustic Variables, Including Means, Standard Deviations, and Comparison With Normative Values. Acoustic Variable
Mean (SD)
Jitter, % Female 1.48 (0.90) Male 0.93 Shimmer, % Female 4.5 (2.07) Male 5.28 Pitch perturbation quotient Female 2.75 (5.91) Male 0.51 Amplitude perturbation quotient Female 3.00 (1.24) Male 4.28 Average fundamental frequency Female 200.87 (25.13) Male 124.94 Cepstral Spectral Index of Dysphonia (vowel) Female 16.84 (11.68) Male 17.58 Cepstral Spectral Index of Dysphonia (rainbow) Female 18.60 (6.63) Male 36.84
Min
Max
Normative Mean (SD)
P Value
0.42 —
3.23 —
0.63 (0.35) 0.59 (0.54)
.015 —
2.44 —
8.48 —
2.00 (0.79) 2.52 (1.00)
.004a —
0.25 —
19.51 —
0.37 (0.21) 0.34 (0.29)
.235 —
1.79 —
5.42 —
1.40 (0.53) 1.99 (0.81)
.003a —
156.94 —
234.18 —
243.97 (27.46) 145.22 (23.41)
.001a —
0.44 —
36.09 —
— —
— —
6.02 —
27.49 —
— —
— —
a
Statistically significant at alpha < .01.
pharyngeal dryness, 1.25 to 7 cm for laryngeal dryness, and 1.45 to 7 cm for vocal fold dryness. On average, dryness ratings were in the mild to mild-to-moderate range. On a VAS of normal to profoundly abnormal, the mean rating of overall laryngeal function—a composite global score of laryngeal form and function—was 2.13 cm (SD = 1.45 cm), with a range of 0.5 to 4.75 cm.
Patient-Based Severity and Correlations Among Variables VHI and ESSPRI. The Voice Handicap Index (VHI), a 30-item, psychometrically validated questionnaire of voicerelated disablement,17 was used to measure each individual’s perception of the overall impairment of his or her voice. Mean total score on the VHI was 43 (SD = 23; range, 14-79). Voice Handicap Index and ESSPRI disease severity scores were significantly correlated (r = 0.690, P = .019). Voice Handicap Index and ESSPRI scores were not significantly associated with age, VAS ratings of dysphonia severity or speech clarity, or VAS ratings of pharyngeal, laryngeal, or vocal fold dryness. Acoustic, auditory-perceptual, and laryngeal relationships. Jitter (%) was significantly correlated with auditory-perceptual ratings of dysphonia severity on vowels (r = 0.853, P = .001) and connected speech (r = 0.804, P = .003). Cepstral
Spectral Index of Dysphonia values for sustained vowels were significantly correlated with auditory-perceptual ratings of dysphonia severity on vowels (r = 0.624, P = .040). Videolaryngostroboscopic ratings demonstrated significant correlations between pharyngeal and laryngeal (r = 0.996, P = .001), pharyngeal and vocal fold (r = 0.871, P = .002), and laryngeal and vocal fold (r = 0.900, P = .001) dryness.
Discussion The purpose of this study was to examine voice, speech, and laryngeal characteristics of individuals with pSS. Specifically, this study examined acoustic measures of sustained vowel and connected speech, auditory-perceptual ratings of dysphonia severity and speech clarity, and videolaryngostroboscopic features of individuals with pSS. Analysis of the results indicated that individuals with pSS have voice and speech patterns that distinguish them from typical speakers. In general, voice and speech disorder severity fell within the mild to mild-to-moderate range. Patients with pSS also tended to rate themselves as mildlyto-moderately handicapped by their voice symptoms, based on their VHI scores. These findings shed light on the nature and extent of voice and speech problems in pSS and have implications for assessment and management in this population.
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Heller et al Table 2. Videolaryngostroboscopic Ratings, Including Structural, Vibratory, and Mucosal Parameters.a Parameter Vocal fold edges Straight Bowed Vocal fold texture Normal Rough Vertical level On plane Vascular markings None Capillary ectasia Microvarices Hemorrhage Mobility Normal Length of vocal folds (NPNV) Symmetrical Length of vocal folds (HPNV) Symmetrical Not sampled Glottic closure Complete Anterior chink Posterior chink Phase symmetry Symmetrical Asymmetrical Not sampled Mucosal wave/amplitude Normal Not sampled Supraglottic activity (NPNV) None Mild Moderate Mucus (pharynx) Thin Thick Foamy Tacky Mucus (vocal folds) Thin Thick Foamy Tacky
Frequency 8 1 8 1 9 6 2 2 2 9 9
Figure 1. Auditory-perceptual ratings of voice severity and speech clarity using a 10-cm visual analog scale, where 0 represents normality and 10 represents extreme severity.
8 1 4 2 3 7 1 1 7 2 3 5 1 6 2 1 0 4 5 0 0
Abbreviations: HPNV, high pitch, normal volume; NPNV, normal pitch, normal volume. a Numbers in bold indicate possible non-normal findings for each parameter.
Acoustic analyses indicated that individuals with pSS differ significantly from normative values for voice production during sustained vowels. Of the 5 MDVP acoustic
Figure 2. Visual-perceptual ratings of pharyngeal, laryngeal, and vocal fold dryness using a 10-cm visual analog scale, where 0 represents no dryness and 10 represents extreme dryness.
parameters selected for acoustic analysis (jitter %, PPQ, shimmer %, APQ, and AFF), shimmer, APQ, and AFF were significantly different from normative values; jitter approached statistical significance. Among the MDVP measures, jitter appears to be the most significantly correlated with prediction of voice severity ratings for sustained vowel and connected speech. These acoustic parameters were originally selected for analysis for 2 reasons. First, these measures represent traditional time-based measures of
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aperiodicity routinely employed during clinical measurement of voice. Second, several of these measures were found to be sensitive to voice changes in a heterogeneous group of pSS and secondary SS patients in a prior investigation.9 The results from this study indicated that individuals with pSS demonstrated differences in the periodicity of the voice signal as compared with typical speakers. In general, these differences related to cycle-to-cycle variations in amplitude of the voice signal, as well as differences in fundamental frequency. These findings indicate that individuals with pSS experience mild phonatory instability during sustained vowel production and fundamental frequency is significantly lower than typical speakers. In addition to differences observed on the MDVP, ADSV and CSID values for sustained vowels and connected speech also indicated that individuals with pSS experience voice problems that are in the mild to mild-to-moderate severity range. The CSID was selected as another measure used in clinical voice evaluation, as it can accurately quantify voice severity in significantly aperiodic voice signals. The significant correlation between MDVP and CSID analyses is confirmatory, indicating that the voice changes experienced by individuals with pSS could be reliably and consistently detected during routine clinical voice acoustic evaluation. CSID analyses also offer additional evidence of voice changes apparent not only in sustained vowels but also during connected speech. Auditory-perceptual ratings of voice severity were generally consistent with acoustic analyses. Although the VAS ratings for sustained vowels were not significantly correlated with ratings for connected speech, both indicated abnormal voice quality in this group of individuals with pSS. Voice severity ratings for sustained vowels were higher than those for connected speech, perhaps partially explaining the lack of significant correlation. Collectively, the auditory-perceptual ratings indicate a mild to mild-to-moderate voice severity for these individuals. These results are fairly consistent with the only other study that reported auditory-perceptual ratings of voice severity in (mainly secondary) SS patients; however, that study reported dysphonia in only 70% of patients examined.10 In the present study, all participants were judged to have some level of dysphonia. Given that listeners in both studies were experienced raters, these results would suggest that the present sample of individuals with pSS experienced voice problems with a greater frequency or severity than those individuals with SS in the previous study. It might also suggest that individuals with pSS are more likely to have voice problems, or more severe voice problems, than individuals with secondary SS. Future research is needed to clarify these results in quantifying the nature and severity as well as the variability of voice problems in pSS. The speech clarity ratings in this study were based on work by Eisenberg et al.16 The purpose of these ratings was
to quantify articulatory imprecision in this population. Individuals with pSS are at risk for articulatory problems due to a number of factors, including but not limited to xerostomia, neuropathy, and poor dentition. No data exist regarding the incidence and prevalence of articulatory problems in pSS. Speech clarity ratings in this study indicated mild articulatory imprecision in connected speech. Certainly, articulatory disturbances can have a significant effect on quality of life and activities of daily living. Depending on the severity of the disturbance, articulatory imprecision can affect functional communication. More research is needed on articulatory problems in this population, to improve referral and treatment practices in these individuals. The videolaryngostroboscopic findings in this study are particularly interesting in light of previous reports of laryngeal changes in individuals with SS.6-11 This group of individuals presented with mild to mild-to-moderate pharyngeal, laryngeal, and vocal fold dryness. These findings are consistent with the sicca symptoms associated with pSS; however, it is difficult to determine the sensitivity and specificity of visual-perceptual dryness ratings in identifying laryngeal sicca. Similarly, it is difficult to know the clinical significance of laryngeal dryness as a causative factor for dysphonia. Perhaps most interesting was the lack of significant structural changes in this sample of individuals with pSS; the most common laryngeal findings were supraglottic compression, thick mucus, and vascular markings. It is possible that some of the structural changes reported in the literature previously (eg, bamboo nodules, edema, reflux) are related to other causative factors and not to SS. Given the relatively common presentation of primary rheumatoid arthritis and secondary SS, the attribution of bamboo nodules to the primary disease process would be a logical conclusion. Similarly, the frequency of vascular markings in this study (ie, 3 of 11 participants) might be related to additional factors, such as frequent use of nonsteroidal antiinflammatory drugs for pain. The lack of obvious structural changes might also indicate that the voice problems experienced by individuals with pSS could be related to inflammation, dryness, or muscle tension dysregulation. Future research should explore the relationships among these laryngeal features and the nature and severity of voice problems in larger groups of individuals with primary and secondary SS. The patient-based reports of voice and pSS disease severity also warrant attention. The mean VHI score in this group of individuals with pSS was in the mild-to-moderate range.18 Disease severity scores are in the moderate range based on the ESSPRI.12 In general, VHI scores indicated more severe voice symptoms than indicated by acoustic and auditory-perceptual ratings. This finding is consistent with new research examining relationships between patientbased and acoustic measures of dysphonia.19-23 Because
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Heller et al even expert listeners may tend to underestimate the effect of voice problems on voice handicap based on auditory-perceptual judgments alone, multidisciplinary voice assessment and management appears to be important in this population. Several limitations should be considered when interpreting the results from this investigation. Although this study represents the largest investigation of voice problems in pSS—and the first to examine speech problems in this population—it still represents a relatively small sample size. In addition, raters were aware of the patients’ diagnosis of pSS in this study, perhaps influencing the auditory-perceptual and videolaryngostroboscopic results. Future work should incorporate the use of blinding and similar experimental controls when evaluating these parameters. Even so, health care providers employ similar methods for evaluating voice and speech clinically. Therefore, this study employed methods that will often be used to quantify voice and speech problems in these individuals during routine clinical practice. In light of the present findings, visual identification of laryngeal dryness, thick mucus, vascular markings, or supraglottic compression—combined with unexplained oral and ocular sicca symptoms—should prompt referral for pSS evaluation. Similarly, individuals with primary or secondary SS should be asked about voice problems to determine the need for voice evaluation. Future research is needed to explore the incidence and prevalence of these problems in pSS, including treatment-seeking behaviors and assessment and management clinical practices. Additional studies should also explore chronicity of symptoms and identify appropriate treatment and symptom management plans for this population.
Conclusion Individuals with pSS may experience dysphonia and articulatory imprecision. When present, dysphonia and speech clarity are typically mildly affected based on acoustic analysis and expert perceptual ratings. In contrast, individuals with pSS rate their own overall voice severity as mild-tomoderate. These findings have implications for diagnostic and referral practices in pSS. Acknowledgments The authors thank The University of Utah Voice Disorders Center and Division of Otolaryngology–Head and Neck Surgery for their assistance during this study. They would like to give special thanks to Christopher Dromey, PhD, CCC-SLP, Heather Elwell, MS, CCC-SLP, Kim Korbin-Lewis, PhD, CCC-SLP, Cara Sauder, MA, CCC-SLP, and Anna Siciliano, MS, CCC-SLP, for serving as expert raters. The authors thank Callie Payne for her assistance with data entry and Jenny Pierce for assistance with participant scheduling.
Declaration of Conflicting Interests The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.
Funding The author(s) disclosed receipt of the following financial support for the research, authorship, and/or publication of this article: This work was supported by a University of Utah Rehabilitation Services Clinical Research Grant and a Brigham Young University McKay School of Education faculty planning grant.
References 1. Kruszka P, O’Brian RJ. Diagnosis and management of Sjögren syndrome. Am Fam Physician. 2009;79:465-470, 472. 2. Huang YF, Cheng Q, Jiang CM, et al. The immune factors involved in the pathogenesis, diagnosis, and treatment of Sjogren’s syndrome. Clin Dev Immunol. doi:10.1155/2013/160491. 3. Malladi AS, Sack KE, Shiboski SC, et al. Primary Sjögren’s syndrome as a systemic disease: a study of participants enrolled in an international Sjögren’s syndrome registry. Arthritis Care Res. 2012;64:911-918. 4. Palm O, Garen T, Berge Enger T, et al. Clinical pulmonary involvement in primary Sjogren’s syndrome: prevalence, quality of life and mortality—a retrospective study based on registry data. Rheumatology. 2013;52:173-179. 5. Reksten TR, Jonsson MV. Sjogren’s syndrome: an update on epidemiology and current insights on pathophysiology. Oral Maxillofac Surg Clin North Am. 2014;26:1-12. 6. Doig JA, Whaley K, Dick WC, Nuki G, Williamson J, Buchanan WW. Otolaryngological aspects of Sjögren’s syndrome. Br Med J. 1971;20:460-463. 7. Murano E, Hosako-Naito T, Oka T, Miyaji M, Kumada M, Niimi S. Bamboo node: primary vocal fold lesion as evidence of autoimmune disease. J Voice. 2001;15:441-450. 8. Belafsky PC, Postma GN. The laryngeal and esophageal manifestations of Sjogren’s syndrome. Curr Rheumatol Rep. 2003;5:297-303. 9. Ogut F, Midilli R, Oder G, Engin EZ, Karci B, Kabasakal Y. Laryngeal findings and voice quality in Sjögren’s syndrome. Aurus Nasus Larynx. 2005;32:375-380. 10. Ruiz Allec LD, Hernández López X, Arreguín Porras JB, Velasco Ramos R, Pacheco del Valle JC, Pérez Garcia AI. Alterations in voice, speech, and swallowing in patients with Sjögren’s syndrome. Acta Otorrinolaringol Esp. 2011;62:255-264. 11. Sanz L, Sistiaga JA, Lara AJ, Cuende E, García-Alcántara F, Rivera T. The prevalence of dysphonia, its association with immunomediated diseases and correlation with biochemical markers. J Voice. 2011;26:148-153. 12. Seror R, Bootsma H, Bowman SJ, et al. Outcome measures for primary Sjögren’s syndrome. J Autoimmun. 2012;39:97102. 13. Fairbanks G. Voice and Articulation Drillbook. 2nd ed. New York: Harper & Row; 1960.
Downloaded from aor.sagepub.com at EMORY UNIV on June 21, 2014
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14. Shrivastav R, Sapienza C, Nandur V. Application of psychometric theory to the measurement of voice quality using rating scales. J Speech Lang Hear Res. 2005;48:323-335. 15. Peterson EA, Roy N, Awan SN, Merrill RM, Banks R, Tanner K. Toward validation of the Cepstral Spectral Index of Dysphonia (CSID) as an objective treatment outcomes measure. J Voice. 2013;27:401-410. 16. Eisenberg L, Dirks D, Takayanagi S, Martinez A. Subjective judgement of clarity and intelligibility for filtered stimuli with equivalent speech intelligibility index predictions. J Speech Lang Hear Res. 1998;41:327-339. 17. Jacobson BH, Johnson A, Grywalski C, Silbergleit A, Jacobson G, Benninger MS. The Voice Handicap Index (VHI). Am J Speech Lang Pathol. 1997;6:66-70. 18. Behrman A, Rutledge J, Hembree A, Sheridan S. Vocal hygiene education, voice production therapy, and the role of patient adherence: a treatment effectiveness study in women with phonotrauma. J Speech Lang Hear Res. 2008;51:350366.
19. Eadie TL, Kapsner M, Rosenzweig J, Waugh P, Hillel A, Merati A. The role of experience on judgments of dysphonia. J Voice. 2010;24:564-573. 20. Eadie TL, Nicolici C, Baylor C, Almand K, Waugh P, Maronian N. Effect of experience on judgments of adductor spasmodic dysphonia. Ann Otol Rhinol Laryngol. 2007;116:695-701. 21. Lee M, Drinnan M, Carding P. The reliability and validity of patient self-rating of their own voice quality. Clin Otolaryngol. 2005;30:357-361. 22. Lenderking WR, Hillson E, Crawley JA, Moore D, Berzon R, Pashos CL. The clinical characteristics and impact of Iaryngopharyngeal reflux disease on health-related quality of life. Value Health. 2003;6:560-565. 23. Awan S, Roy N, Cohen S. Exploring the relationship between spectral and cepstral measures of voice and the Voice Handicap Index (VHI) [published online March 31, 2014]. J Voice. doi:10.1016/j.jvoice.2013.12.008.
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Voice, Speech, and Laryngeal Features of Primary Sjögren's Syndrome Amanda Heller, Kristine Tanner, Nelson Roy, Shawn L. Nissen, Ray M. Merrill, Karla L. Miller, Daniel R. Houtz, Julia Ellerston and Katherine Kendall Ann Otol Rhinol Laryngol published online 18 June 2014 DOI: 10.1177/0003489414538762 The online version of this article can be found at: http://aor.sagepub.com/content/early/2014/06/16/0003489414538762
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AORXXX10.1177/0003489414538762Annals of Otology, Rhinology & LaryngologyHeller et al
Article
Voice, Speech, and Laryngeal Features of Primary Sjögren’s Syndrome
Annals of Otology, Rhinology & Laryngology 1–8 © The Author(s) 2014 Reprints and permissions: sagepub.com/journalsPermissions.nav DOI: 10.1177/0003489414538762 aor.sagepub.com
Amanda Heller, MS1,2, Kristine Tanner, PhD2,3, Nelson Roy, PhD1,2, Shawn L. Nissen, PhD3, Ray M. Merrill, PhD, MPH4, Karla L. Miller, MD5, Daniel R. Houtz, MA2, Julia Ellerston, MA2, and Katherine Kendall, MD2,6
Abstract Objective: This study examined voice, speech, and laryngeal characteristics in primary Sjögren’s syndrome (pSS). Methods: Eleven patients (10 female, 1 male; mean [SD] age = 57 [14] years) from The University of Utah Division of Rheumatology provided connected speech and sustained vowel samples. Analyses included the Multi-Dimensional Voice Profile, the Analysis of Dysphonia in Speech and Voice, and dysphonia severity, speech clarity, and videolaryngostroboscopy ratings. Results: Shimmer, amplitude perturbation quotient, and average fundamental frequency differed significantly from normative values (P < .01). Cepstral Spectral Index of Dysphonia values indicated mild-to-moderate dysphonia in connected speech (mean [SD] = 20.26 [8.36]) and sustained vowels (mean [SD] = 16.91 [11.08]). Ratings of dysphonia severity and speech clarity using 10-cm visual analog scales suggested mild-to-moderate dysphonia in connected speech (mean [SD] = 2.11 [1.72]) and sustained vowels (mean [SD] = 3.13 [2.20]) and mildly reduced speech clarity (mean [SD] = 1.46 [1.36]). Videolaryngostroboscopic ratings indicated mild-to-moderate dryness and mild reductions in overall laryngeal function. Voice Handicap Index scores indicated mild-to-moderate voice symptoms (mean [SD] = 43 [23]). Conclusion: Individuals with pSS may experience dysphonia and articulatory imprecision, typically in the mild-to-moderate range. These findings have implications for diagnostic and referral practices in pSS. Keywords primary Sjögren’s syndrome (pSS), voice disorders, speech clarity, autoimmune disease
Introduction Primary Sjögren’s syndrome (pSS) is an autoimmune disease characterized by reduced exocrine gland secretion, causing chronic dryness—or sicca—symptoms. The most common features of pSS include xerostomia and keratoconjunctivitis sicca due to poor function of the salivary and lacrimal glands.1 Dryness of the nose, pharynx, larynx, trachea, bronchi, vagina, and skin is common. Additional systemic effects may include peripheral and cranial neuropathy, renal dysfunction, pancreatitis, vasculitis, glandular infections, parotid tumors, and lymphoma. Other symptoms include focal or generalized pain, stiffness, swelling, and fatigue.2-4 Recent epidemiologic reports indicate that pSS occurs in approximately 0.5% to 1% of the population. Risk rates are 9 to 1 female to male, occurring most frequently in mid-life, although pediatric cases also exist. In contrast to secondary SS, pSS occurs independently of other inflammatory autoimmune diseases.5 Individuals with SS can have swallowing, voice, and speech problems. Although the dysphagia literature is more
developed in this population, only infrequent reports of voice and speech problems exist or are inferred from associated mouth and throat symptoms. Because secondary SS is more prevalent, these studies include either a diverse group 1
Department of Communication Sciences & Disorders, The University of Utah, Salt Lake City, Utah, USA 2 Voice Disorders Center, The University of Utah, Salt Lake City, Utah, USA 3 Department of Communication Disorders, Brigham Young University, Provo, Utah, USA 4 Department of Health Science, Brigham Young University, Provo, Utah, USA 5 Division of Rheumatology, The University of Utah School of Medicine, Salt Lake City, Utah, USA 6 Division of Otolaryngology–Head & Neck Surgery, The University of Utah School of Medicine, Salt Lake City, Utah, USA Corresponding Author: Kristine Tanner, PhD, Department of Communication Disorders, Brigham Young University, 1190 N 900 E, 158 TLRB, Provo, UT 84602, USA. Email: [email protected]
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of both primary and secondary SS or exclusively secondary SS cohorts.6-11 Among these studies, SS has been associated with hoarseness, roughness, strain, and articulatory imprecision. Videolaryngostroboscopic findings are diverse and include supraglottic adduction, vocal fold lesions, weakness, edema, pseudosulcus, inflammation, dryness, and thick mucus. Lingual weakness, tongue deviation, labial asymmetry, lingual adhesion to the palate, and palate- pharyngeal adhesion as well as severe oropharyngeal mucosal dryness and loss of dentition have been observed. Voice- and speech-related patient symptoms include increased vocal effort, throat dryness, cough, pitch breaks, hypernasality, and decreased loudness. Atypical voice and speech acoustic patterns have also been observed. Unfortunately, the relative rarity of pSS has prevented systematic investigations of voice and speech problems specific to this population. The purpose of this study was to characterize voice, speech, and laryngeal problems in patients with pSS. To our knowledge, no studies exist that have examined voice and speech production exclusive to this population. This next step is essential, because secondary SS, by definition, is the result of another primary autoimmune disease, such as rheumatoid arthritis or lupus erythematosus. Due to the overlapping manifestations of these autoimmune diseases (eg, bamboo nodules or inflammation), it is difficult in cases of secondary SS to know which symptoms are uniquely associated with each disease process(es). The pure effects of pSS on voice and speech production warrant further investigation. To that end, this study examined acoustic, auditory-perceptual, and laryngeal characteristics of pSS.
Methods Participants Eleven individuals with pSS (10 female, 1 male; mean [SD] age = 57 [14] years) were recruited from The University of Utah Division of Rheumatology treatment-seeking population. Inclusion criteria were based on the presence of sicca symptoms, antinuclear antibody testing, and/or lip biopsy. Participants were included only if SS was the primary causative factor attributed to their sicca symptoms. All participants were receiving medical management of SS, including disease-related medications. The male participant reported a history of smoking 1 pack of cigarettes per day for more than 30 years. Participant mean total score on the EULAR (European League Against Rheumatism) Sjögren’s Syndrome Patient Reported Index (ESSPRI)12—a 3-domain patient-based symptom severity scale including dryness, pain, and fatigue—was 5.4 out of 10 (SD = 2.1). Audio recordings were collected using a Canon VIXIA HD Camcorder (HF S21) and Shure (SM 48) cardioid
microphone with a mouth-to-mic distance of 4 inches. Samples consisted of the second and third sentences of “The Rainbow Passage”13 and the central 3 seconds of a sustained vowel /ɑ/ production collected on 2 consecutive weeks. Samples were extracted using Adobe Audition (version 3.0; Adobe, San Jose, California, USA) at a sample rate of 44 100 Hz. Numeric data for acoustic variables were averaged across weeks for repeated samples.14 Nine of the 11 participants tolerated rigid videolaryngostroboscopy without topical anesthesia.
Acoustic Analyses Sustained vowel recordings were analyzed using the MultiDimensional Voice Program (MDVP Advanced for MultiSpeech, Model 5105; KayPentax, Montvale, New Jersey, USA). Voice parameters sampled included those previously shown or theorized to be sensitive to voice problems in SS,9 including jitter (%), shimmer (%), pitch perturbation quotient (PPQ), amplitude perturbation quotient (APQ), and average fundamental frequency (AFF). Jitter and PPQ are sensitive to cycle-to-cycle variations in frequency, whereas shimmer and APQ are sensitive to cycle-to-cycle variations in amplitude of the voice signal. Sustained vowel and connected speech recordings were analyzed using the Analysis of Dysphonia in Speech and Voice (ADSV, version 5109; KayPentax, Montvale, New Jersey, USA), which subsequently generates the Cepstral Spectral Index of Dysphonia (CSID). The CSID is an acoustic measure of dysphonia severity, derived from spectral and cepstral measurements. The program automatically generates a CSID value for sustained vowels but requires a manual input of the cepstral peak prominence (CPP), the low/high (L/H) spectral ratio, and the L/H spectral ratio standard deviation to compute a value for connected speech. This measure has been established to be highly associated with listener ratings of dysphonia severity and is sensitive and specific to heterogeneous voice qualities and severities.15
Auditory-Perceptual Ratings Eight speech-language pathologists with extensive experience in voice disorder assessment and treatment completed auditory-perceptual ratings of dysphonia severity (sustained vowel and connected speech) and speech clarity (connected speech). The identical samples selected for acoustic analysis were also used for auditory-perceptual ratings but were normalized for loudness in the listening task. Samples were presented via Sennheiser HD 558 headphones. Three samples were presented prior to beginning the ratings to orient the listener to the range and variety of samples. Ratings were accomplished using a 10-cm visual analog scale (VAS). The extreme left (ie, 0 cm) represented normal voice
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Heller et al and extreme right (ie, 10 cm) represented profoundly abnormal voice. Following the dysphonia severity ratings, listeners rated speech clarity using a 10-cm VAS. Speech clarity was defined as how clear, or distinct, the listener perceived the words in the connected speech sample to be.16 The extreme left represented typical speech clarity (all the words sounded distinct, clear, or pure) and the extreme right represented profoundly abnormal speech clarity (all of the words sounded muffled or blurred). Listeners also rated 20% of samples a second time to estimate intrajudge reliability.
Videolaryngostroboscopy One expert laryngologist (K.K.) with more than 20 years of experience in assessment and management of voice disorders assessed the 9 rigid videolaryngostroboscopy exams. The ratings evaluated structural, vibratory, and mucosal laryngeal features. Parameters assessed included vocal fold edges (straight vs bowed), vocal fold texture (normal vs rough), vertical level (on plane vs off plane), vascular markings (none, capillary ectasia, microvarices, hemorrhage), mobility (normal, reduced, absent), length of vocal folds at normal pitch normal volume (NPNV; symmetrical, left shorter, right shorter), length of vocal folds at high pitch normal volume (HPNV; symmetrical, left shorter, right shorter), glottic closure (complete, anterior chink, posterior chink, spindle, hourglass, incomplete), phase symmetry (symmetrical, asymmetrical), vibratory behavior including mucosal wave and amplitude of vibration (normal, reduced, absent), supraglottic activity (none, mild, moderate, or severe mediolateral and/or anterior-posterior compression), mucus of the pharynx (thin, thick, foamy, frothy, transient, white, yellow, clear, tacky), and mucus of the vocal folds (thin, thick, foamy, frothy, transient, white, yellow, clear, tacky). An overall rating of laryngeal appearance was made using a 10-cm VAS, with the extreme left of the scale indicating a normal laryngeal appearance and the extreme right representing a profoundly abnormal laryngeal appearance. The same method of rating was completed to evaluate overall dryness of the pharynx, larynx, and vocal folds; the extreme left indicated normal appearance and extreme right indicated severe dryness.
Statistical Analysis Summary statistics, including means, standard deviations, and ranges, were used to explore all variables. Relationships among variables were evaluated using Pearson correlations. Differences between acoustic variables and normative values were evaluated using 1-sample t tests with a Bonferroni correction for multiple comparisons. Agreement among raters for auditory-perceptual ratings was evaluated using intraclass correlation coefficients (ICCs); intrarater agreement was evaluated using multiple regression analysis.
Analyses were performed with the Statistical Analysis System (SAS) software, version 9.4 (2013; SAS Institute Inc, Cary, North Carolina, USA).
Results Acoustic Analysis Statistically significant differences in shimmer (%; P = .004), APQ (P = .003), and AFF (P = .001) were observed between pSS participants and normative values based on 1-sample t tests (Bonferroni correction = 0.050/5). Differences in jitter (%) approached statistical significance (P = .015). Differences in PPQ were not statistically significant. CSID values ranged from mild to mild-to-moderate dysphonia for sustained vowels and connected speech samples. Summary data are provided in Table 1. Pearson correlations ranged from 0.970 to 1.000 for 10% repeated acoustic analyses (P = .001-.022), indicating acceptable measurement reliability.
Auditory-Perceptual Ratings Assessment of reliability of listener ratings of dysphonia severity (P < .050) revealed ICCs of 0.939 (95% confidence interval [CI], 0.864-0.981) for sustained vowels and 0.821 (95% CI, 0.599-0.944) for connected speech, indicating acceptable interjudge reliability. For speech clarity, the ICC was 0.962 (95% CI, 0.915-0.988), also indicating acceptable reliability. Regression analysis showed a strong correlation between repeated ratings of voice severity on vowels (slope = 0.85, P = .001) and speech clarity (slope = 0.90, P = .001); an adequate correlation was observed for voice severity during connected speech (slope = 0.42, P = .001). These results confirm acceptable inter- and intra-listener reliability and provide confidence in the reported results that follow. For sustained vowel VAS ratings, mean voice severity was 3.13 cm (SD = 1.51 cm; range, 0-9.75 cm); for connected speech VAS ratings, mean voice severity was 2.11 cm (SD = 0.61 cm; range, 0-8.55 cm). Mean speech clarity based on the VAS was 1.47 cm (SD = 1.22 cm; range, 0-8.75 cm). Auditory-perceptual ratings for voice severity and speech clarity are illustrated in Figure 1.
Videolaryngostroboscopic Ratings Videolaryngostroboscopic ratings based on structural, vibratory, and mucosal parameters are presented in Table 2. The most common laryngeal findings were the presence of supraglottic adduction (ie, compression), thick mucus, and vascular markings. Vocal fold vibratory and mobility parameters were essentially normal. Visual analog scale ratings of pharyngeal, laryngeal, and vocal fold dryness are illustrated in Figure 2. Ratings ranged from 1 to 6.25 cm for
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Table 1. Select Multi-Dimensional Voice Program and Analysis of Dysphonia in Speech and Voice Acoustic Variables, Including Means, Standard Deviations, and Comparison With Normative Values. Acoustic Variable
Mean (SD)
Jitter, % Female 1.48 (0.90) Male 0.93 Shimmer, % Female 4.5 (2.07) Male 5.28 Pitch perturbation quotient Female 2.75 (5.91) Male 0.51 Amplitude perturbation quotient Female 3.00 (1.24) Male 4.28 Average fundamental frequency Female 200.87 (25.13) Male 124.94 Cepstral Spectral Index of Dysphonia (vowel) Female 16.84 (11.68) Male 17.58 Cepstral Spectral Index of Dysphonia (rainbow) Female 18.60 (6.63) Male 36.84
Min
Max
Normative Mean (SD)
P Value
0.42 —
3.23 —
0.63 (0.35) 0.59 (0.54)
.015 —
2.44 —
8.48 —
2.00 (0.79) 2.52 (1.00)
.004a —
0.25 —
19.51 —
0.37 (0.21) 0.34 (0.29)
.235 —
1.79 —
5.42 —
1.40 (0.53) 1.99 (0.81)
.003a —
156.94 —
234.18 —
243.97 (27.46) 145.22 (23.41)
.001a —
0.44 —
36.09 —
— —
— —
6.02 —
27.49 —
— —
— —
a
Statistically significant at alpha < .01.
pharyngeal dryness, 1.25 to 7 cm for laryngeal dryness, and 1.45 to 7 cm for vocal fold dryness. On average, dryness ratings were in the mild to mild-to-moderate range. On a VAS of normal to profoundly abnormal, the mean rating of overall laryngeal function—a composite global score of laryngeal form and function—was 2.13 cm (SD = 1.45 cm), with a range of 0.5 to 4.75 cm.
Patient-Based Severity and Correlations Among Variables VHI and ESSPRI. The Voice Handicap Index (VHI), a 30-item, psychometrically validated questionnaire of voicerelated disablement,17 was used to measure each individual’s perception of the overall impairment of his or her voice. Mean total score on the VHI was 43 (SD = 23; range, 14-79). Voice Handicap Index and ESSPRI disease severity scores were significantly correlated (r = 0.690, P = .019). Voice Handicap Index and ESSPRI scores were not significantly associated with age, VAS ratings of dysphonia severity or speech clarity, or VAS ratings of pharyngeal, laryngeal, or vocal fold dryness. Acoustic, auditory-perceptual, and laryngeal relationships. Jitter (%) was significantly correlated with auditory-perceptual ratings of dysphonia severity on vowels (r = 0.853, P = .001) and connected speech (r = 0.804, P = .003). Cepstral
Spectral Index of Dysphonia values for sustained vowels were significantly correlated with auditory-perceptual ratings of dysphonia severity on vowels (r = 0.624, P = .040). Videolaryngostroboscopic ratings demonstrated significant correlations between pharyngeal and laryngeal (r = 0.996, P = .001), pharyngeal and vocal fold (r = 0.871, P = .002), and laryngeal and vocal fold (r = 0.900, P = .001) dryness.
Discussion The purpose of this study was to examine voice, speech, and laryngeal characteristics of individuals with pSS. Specifically, this study examined acoustic measures of sustained vowel and connected speech, auditory-perceptual ratings of dysphonia severity and speech clarity, and videolaryngostroboscopic features of individuals with pSS. Analysis of the results indicated that individuals with pSS have voice and speech patterns that distinguish them from typical speakers. In general, voice and speech disorder severity fell within the mild to mild-to-moderate range. Patients with pSS also tended to rate themselves as mildlyto-moderately handicapped by their voice symptoms, based on their VHI scores. These findings shed light on the nature and extent of voice and speech problems in pSS and have implications for assessment and management in this population.
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Heller et al Table 2. Videolaryngostroboscopic Ratings, Including Structural, Vibratory, and Mucosal Parameters.a Parameter Vocal fold edges Straight Bowed Vocal fold texture Normal Rough Vertical level On plane Vascular markings None Capillary ectasia Microvarices Hemorrhage Mobility Normal Length of vocal folds (NPNV) Symmetrical Length of vocal folds (HPNV) Symmetrical Not sampled Glottic closure Complete Anterior chink Posterior chink Phase symmetry Symmetrical Asymmetrical Not sampled Mucosal wave/amplitude Normal Not sampled Supraglottic activity (NPNV) None Mild Moderate Mucus (pharynx) Thin Thick Foamy Tacky Mucus (vocal folds) Thin Thick Foamy Tacky
Frequency 8 1 8 1 9 6 2 2 2 9 9
Figure 1. Auditory-perceptual ratings of voice severity and speech clarity using a 10-cm visual analog scale, where 0 represents normality and 10 represents extreme severity.
8 1 4 2 3 7 1 1 7 2 3 5 1 6 2 1 0 4 5 0 0
Abbreviations: HPNV, high pitch, normal volume; NPNV, normal pitch, normal volume. a Numbers in bold indicate possible non-normal findings for each parameter.
Acoustic analyses indicated that individuals with pSS differ significantly from normative values for voice production during sustained vowels. Of the 5 MDVP acoustic
Figure 2. Visual-perceptual ratings of pharyngeal, laryngeal, and vocal fold dryness using a 10-cm visual analog scale, where 0 represents no dryness and 10 represents extreme dryness.
parameters selected for acoustic analysis (jitter %, PPQ, shimmer %, APQ, and AFF), shimmer, APQ, and AFF were significantly different from normative values; jitter approached statistical significance. Among the MDVP measures, jitter appears to be the most significantly correlated with prediction of voice severity ratings for sustained vowel and connected speech. These acoustic parameters were originally selected for analysis for 2 reasons. First, these measures represent traditional time-based measures of
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aperiodicity routinely employed during clinical measurement of voice. Second, several of these measures were found to be sensitive to voice changes in a heterogeneous group of pSS and secondary SS patients in a prior investigation.9 The results from this study indicated that individuals with pSS demonstrated differences in the periodicity of the voice signal as compared with typical speakers. In general, these differences related to cycle-to-cycle variations in amplitude of the voice signal, as well as differences in fundamental frequency. These findings indicate that individuals with pSS experience mild phonatory instability during sustained vowel production and fundamental frequency is significantly lower than typical speakers. In addition to differences observed on the MDVP, ADSV and CSID values for sustained vowels and connected speech also indicated that individuals with pSS experience voice problems that are in the mild to mild-to-moderate severity range. The CSID was selected as another measure used in clinical voice evaluation, as it can accurately quantify voice severity in significantly aperiodic voice signals. The significant correlation between MDVP and CSID analyses is confirmatory, indicating that the voice changes experienced by individuals with pSS could be reliably and consistently detected during routine clinical voice acoustic evaluation. CSID analyses also offer additional evidence of voice changes apparent not only in sustained vowels but also during connected speech. Auditory-perceptual ratings of voice severity were generally consistent with acoustic analyses. Although the VAS ratings for sustained vowels were not significantly correlated with ratings for connected speech, both indicated abnormal voice quality in this group of individuals with pSS. Voice severity ratings for sustained vowels were higher than those for connected speech, perhaps partially explaining the lack of significant correlation. Collectively, the auditory-perceptual ratings indicate a mild to mild-to-moderate voice severity for these individuals. These results are fairly consistent with the only other study that reported auditory-perceptual ratings of voice severity in (mainly secondary) SS patients; however, that study reported dysphonia in only 70% of patients examined.10 In the present study, all participants were judged to have some level of dysphonia. Given that listeners in both studies were experienced raters, these results would suggest that the present sample of individuals with pSS experienced voice problems with a greater frequency or severity than those individuals with SS in the previous study. It might also suggest that individuals with pSS are more likely to have voice problems, or more severe voice problems, than individuals with secondary SS. Future research is needed to clarify these results in quantifying the nature and severity as well as the variability of voice problems in pSS. The speech clarity ratings in this study were based on work by Eisenberg et al.16 The purpose of these ratings was
to quantify articulatory imprecision in this population. Individuals with pSS are at risk for articulatory problems due to a number of factors, including but not limited to xerostomia, neuropathy, and poor dentition. No data exist regarding the incidence and prevalence of articulatory problems in pSS. Speech clarity ratings in this study indicated mild articulatory imprecision in connected speech. Certainly, articulatory disturbances can have a significant effect on quality of life and activities of daily living. Depending on the severity of the disturbance, articulatory imprecision can affect functional communication. More research is needed on articulatory problems in this population, to improve referral and treatment practices in these individuals. The videolaryngostroboscopic findings in this study are particularly interesting in light of previous reports of laryngeal changes in individuals with SS.6-11 This group of individuals presented with mild to mild-to-moderate pharyngeal, laryngeal, and vocal fold dryness. These findings are consistent with the sicca symptoms associated with pSS; however, it is difficult to determine the sensitivity and specificity of visual-perceptual dryness ratings in identifying laryngeal sicca. Similarly, it is difficult to know the clinical significance of laryngeal dryness as a causative factor for dysphonia. Perhaps most interesting was the lack of significant structural changes in this sample of individuals with pSS; the most common laryngeal findings were supraglottic compression, thick mucus, and vascular markings. It is possible that some of the structural changes reported in the literature previously (eg, bamboo nodules, edema, reflux) are related to other causative factors and not to SS. Given the relatively common presentation of primary rheumatoid arthritis and secondary SS, the attribution of bamboo nodules to the primary disease process would be a logical conclusion. Similarly, the frequency of vascular markings in this study (ie, 3 of 11 participants) might be related to additional factors, such as frequent use of nonsteroidal antiinflammatory drugs for pain. The lack of obvious structural changes might also indicate that the voice problems experienced by individuals with pSS could be related to inflammation, dryness, or muscle tension dysregulation. Future research should explore the relationships among these laryngeal features and the nature and severity of voice problems in larger groups of individuals with primary and secondary SS. The patient-based reports of voice and pSS disease severity also warrant attention. The mean VHI score in this group of individuals with pSS was in the mild-to-moderate range.18 Disease severity scores are in the moderate range based on the ESSPRI.12 In general, VHI scores indicated more severe voice symptoms than indicated by acoustic and auditory-perceptual ratings. This finding is consistent with new research examining relationships between patientbased and acoustic measures of dysphonia.19-23 Because
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Heller et al even expert listeners may tend to underestimate the effect of voice problems on voice handicap based on auditory-perceptual judgments alone, multidisciplinary voice assessment and management appears to be important in this population. Several limitations should be considered when interpreting the results from this investigation. Although this study represents the largest investigation of voice problems in pSS—and the first to examine speech problems in this population—it still represents a relatively small sample size. In addition, raters were aware of the patients’ diagnosis of pSS in this study, perhaps influencing the auditory-perceptual and videolaryngostroboscopic results. Future work should incorporate the use of blinding and similar experimental controls when evaluating these parameters. Even so, health care providers employ similar methods for evaluating voice and speech clinically. Therefore, this study employed methods that will often be used to quantify voice and speech problems in these individuals during routine clinical practice. In light of the present findings, visual identification of laryngeal dryness, thick mucus, vascular markings, or supraglottic compression—combined with unexplained oral and ocular sicca symptoms—should prompt referral for pSS evaluation. Similarly, individuals with primary or secondary SS should be asked about voice problems to determine the need for voice evaluation. Future research is needed to explore the incidence and prevalence of these problems in pSS, including treatment-seeking behaviors and assessment and management clinical practices. Additional studies should also explore chronicity of symptoms and identify appropriate treatment and symptom management plans for this population.
Conclusion Individuals with pSS may experience dysphonia and articulatory imprecision. When present, dysphonia and speech clarity are typically mildly affected based on acoustic analysis and expert perceptual ratings. In contrast, individuals with pSS rate their own overall voice severity as mild-tomoderate. These findings have implications for diagnostic and referral practices in pSS. Acknowledgments The authors thank The University of Utah Voice Disorders Center and Division of Otolaryngology–Head and Neck Surgery for their assistance during this study. They would like to give special thanks to Christopher Dromey, PhD, CCC-SLP, Heather Elwell, MS, CCC-SLP, Kim Korbin-Lewis, PhD, CCC-SLP, Cara Sauder, MA, CCC-SLP, and Anna Siciliano, MS, CCC-SLP, for serving as expert raters. The authors thank Callie Payne for her assistance with data entry and Jenny Pierce for assistance with participant scheduling.
Declaration of Conflicting Interests The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.
Funding The author(s) disclosed receipt of the following financial support for the research, authorship, and/or publication of this article: This work was supported by a University of Utah Rehabilitation Services Clinical Research Grant and a Brigham Young University McKay School of Education faculty planning grant.
References 1. Kruszka P, O’Brian RJ. Diagnosis and management of Sjögren syndrome. Am Fam Physician. 2009;79:465-470, 472. 2. Huang YF, Cheng Q, Jiang CM, et al. The immune factors involved in the pathogenesis, diagnosis, and treatment of Sjogren’s syndrome. Clin Dev Immunol. doi:10.1155/2013/160491. 3. Malladi AS, Sack KE, Shiboski SC, et al. Primary Sjögren’s syndrome as a systemic disease: a study of participants enrolled in an international Sjögren’s syndrome registry. Arthritis Care Res. 2012;64:911-918. 4. Palm O, Garen T, Berge Enger T, et al. Clinical pulmonary involvement in primary Sjogren’s syndrome: prevalence, quality of life and mortality—a retrospective study based on registry data. Rheumatology. 2013;52:173-179. 5. Reksten TR, Jonsson MV. Sjogren’s syndrome: an update on epidemiology and current insights on pathophysiology. Oral Maxillofac Surg Clin North Am. 2014;26:1-12. 6. Doig JA, Whaley K, Dick WC, Nuki G, Williamson J, Buchanan WW. Otolaryngological aspects of Sjögren’s syndrome. Br Med J. 1971;20:460-463. 7. Murano E, Hosako-Naito T, Oka T, Miyaji M, Kumada M, Niimi S. Bamboo node: primary vocal fold lesion as evidence of autoimmune disease. J Voice. 2001;15:441-450. 8. Belafsky PC, Postma GN. The laryngeal and esophageal manifestations of Sjogren’s syndrome. Curr Rheumatol Rep. 2003;5:297-303. 9. Ogut F, Midilli R, Oder G, Engin EZ, Karci B, Kabasakal Y. Laryngeal findings and voice quality in Sjögren’s syndrome. Aurus Nasus Larynx. 2005;32:375-380. 10. Ruiz Allec LD, Hernández López X, Arreguín Porras JB, Velasco Ramos R, Pacheco del Valle JC, Pérez Garcia AI. Alterations in voice, speech, and swallowing in patients with Sjögren’s syndrome. Acta Otorrinolaringol Esp. 2011;62:255-264. 11. Sanz L, Sistiaga JA, Lara AJ, Cuende E, García-Alcántara F, Rivera T. The prevalence of dysphonia, its association with immunomediated diseases and correlation with biochemical markers. J Voice. 2011;26:148-153. 12. Seror R, Bootsma H, Bowman SJ, et al. Outcome measures for primary Sjögren’s syndrome. J Autoimmun. 2012;39:97102. 13. Fairbanks G. Voice and Articulation Drillbook. 2nd ed. New York: Harper & Row; 1960.
Downloaded from aor.sagepub.com at EMORY UNIV on June 21, 2014
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14. Shrivastav R, Sapienza C, Nandur V. Application of psychometric theory to the measurement of voice quality using rating scales. J Speech Lang Hear Res. 2005;48:323-335. 15. Peterson EA, Roy N, Awan SN, Merrill RM, Banks R, Tanner K. Toward validation of the Cepstral Spectral Index of Dysphonia (CSID) as an objective treatment outcomes measure. J Voice. 2013;27:401-410. 16. Eisenberg L, Dirks D, Takayanagi S, Martinez A. Subjective judgement of clarity and intelligibility for filtered stimuli with equivalent speech intelligibility index predictions. J Speech Lang Hear Res. 1998;41:327-339. 17. Jacobson BH, Johnson A, Grywalski C, Silbergleit A, Jacobson G, Benninger MS. The Voice Handicap Index (VHI). Am J Speech Lang Pathol. 1997;6:66-70. 18. Behrman A, Rutledge J, Hembree A, Sheridan S. Vocal hygiene education, voice production therapy, and the role of patient adherence: a treatment effectiveness study in women with phonotrauma. J Speech Lang Hear Res. 2008;51:350366.
19. Eadie TL, Kapsner M, Rosenzweig J, Waugh P, Hillel A, Merati A. The role of experience on judgments of dysphonia. J Voice. 2010;24:564-573. 20. Eadie TL, Nicolici C, Baylor C, Almand K, Waugh P, Maronian N. Effect of experience on judgments of adductor spasmodic dysphonia. Ann Otol Rhinol Laryngol. 2007;116:695-701. 21. Lee M, Drinnan M, Carding P. The reliability and validity of patient self-rating of their own voice quality. Clin Otolaryngol. 2005;30:357-361. 22. Lenderking WR, Hillson E, Crawley JA, Moore D, Berzon R, Pashos CL. The clinical characteristics and impact of Iaryngopharyngeal reflux disease on health-related quality of life. Value Health. 2003;6:560-565. 23. Awan S, Roy N, Cohen S. Exploring the relationship between spectral and cepstral measures of voice and the Voice Handicap Index (VHI) [published online March 31, 2014]. J Voice. doi:10.1016/j.jvoice.2013.12.008.
Downloaded from aor.sagepub.com at EMORY UNIV on June 21, 2014
