This epidemiological investigation examined the prevalence, risk factors, and quality-of-life effects of swallowing disorders in Sjogren's syndrome (SS). One hundred and one individuals with primary or secondary SS (94 females, 7 males; mean age 59.4, SD = 14.1) were interviewed regarding the presence, nature, and impact of swallowing disorders and symptoms. Associations among swallowing disorders and symptoms, select medical and social history factors, SS disease severity, and the M.D. Anderson Dysphagia Inventory (MDADI) and Short Form 36 Health Survey (SF-36) were examined. The prevalence of a current self-reported swallowing disorder was 64.4 %. SS disease severity was the strongest predictor of swallowing disorders, including significant associations with the following swallow symptoms: taking smaller bites, thick mucus in the throat, difficulty placing food in the mouth, and wheezing while eating (p < .05). Additional swallowing disorder risk factors included the presence of a self-reported voice disorder, esophageal reflux, current exposure to secondary tobacco smoke, frequent neck or throat tension, frequent throat clearing, chronic post-nasal drip, and stomach or duodenal ulcers. Swallowing disorders did not differ on the basis of primary or secondary SS. Swallowing disorders and specific swallowing symptoms were uniquely associated with reduced quality of life. Among those with swallowing disorders, 42 % sought treatment, with approximately half reporting improvement. Patient-perceived swallowing disorders are relatively common in SS and increase with disease severity. Specific swallowing symptoms uniquely and significantly reduce swallow and health-related quality of life, indicating the need for increased identification and management of dysphagia in this population.

Swallowing Disorders in Sjogren's Syndrome: Prevalence, Risk Factors, and Effects on Quality of Life

Objectives/Hypothesis

This study examined the effects of a laryngeal desiccation challenge and two nebulized hydration treatments on phonation threshold pressure (PTP), vocal effort, and throat dryness in patients with chronic airway dryness.



Study Design

Double-blind, within-subjects crossover design.



Methods

Eleven individuals with Primary Sjogren's Syndrome received a 15-minute laryngeal desiccation challenge (breathing dry air–<1% relative humidity–transorally), followed by nebulized isotonic saline or nebulized water treatments (3 mL) on 2 consecutive weeks. PTP, as well as self-perceived vocal effort, mouth, and throat dryness were assessed before and after the desiccation challenge, and at 5, 35, and 65 minutes after the nebulized treatment.



Results

The laryngeal desiccation challenge produced statistically significant increases in PTP, vocal effort, and mouth and throat dryness (P < 0.05). Nebulized saline produced greater—but not statistically significant—treatment effects than water. PTP was more correlated with throat dryness than vocal effort.



Conclusion

Patients with chronic airway dryness experienced phonatory changes following dry air exposure. Nebulized isotonic saline may offset this effect. Future research should explore dose-response relationships among dry air exposure, nebulized treatments, voice change, and self-perceived throat dryness.



Level of Evidence

1b. Laryngoscope, 123:2787–2792, 2013

Comparing nebulized water versus saline after laryngeal desiccation challenge in Sjögren's Syndrome

Objective

This study examined the effects of a topical vocal fold hydration treatment on voice production over time.



Study Design

Prospective, longitudinal, within-subjects A (baseline), B (treatment), A (withdrawal/reversal), B (treatment) experimental design.



Methods

Eight individuals with primary Sjogren's syndrome (SS), an autoimmune disease causing laryngeal dryness, completed an 8-week A-B-A-B experiment. Participants performed twice-daily audio recordings of connected speech and sustained vowels and then rated vocal effort, mouth dryness, and throat dryness. Two-week treatment phases introduced twice-daily 9-mL doses of nebulized isotonic saline (0.9% Na+Cl−). Voice handicap and patient-based measures of SS disease severity were collected before and after each 2-week phase. Connected speech and sustained vowels were analyzed using the Cepstral Spectral Index of Dysphonia (CSID). Acoustic and patient-based ratings during each baseline and treatment phase were analyzed and compared.



Results

Baseline CSID and patient-based ratings were in the mild-to-moderate range. CSID measures of voice severity improved by approximately 20% with nebulized saline treatment and worsened during treatment withdrawal. Posttreatment CSID values fell within the normal-to-mild range. Similar patterns were observed in patient-based ratings of vocal effort and dryness. CSID values and patient-based ratings correlated significantly (P < .05).



Conclusion

Nebulized isotonic saline improves voice production based on acoustic and patient-based ratings of voice severity. Future work should optimize topical vocal fold hydration treatment formulations, dose, and delivery methodologies for various patient populations. This study lays the groundwork for future topical vocal fold hydration treatment development to manage and possibly prevent dehydration-related voice disorders.



Level of Evidence

2b. Laryngoscope, 125:2333–2340, 2015

Nebulized isotonic saline improves voice production in Sjögren's syndrome.

Descriptive Epidemiology of Voice Disorders in Rheumatoid Arthritis: Prevalence, Risk Factors, and Quality of Life Burden.

Objective:This study examined voice, speech, and laryngeal characteristics in primary Sjogren’s syndrome (pSS).Methods:Eleven patients (10 female, 1 male; mean [SD] age = 57 [14] years) from The Un...

Voice, speech, and laryngeal features of primary Sjögren's syndrome.

Objective: To identify and describe voice, speech and swallowing abnormalities in patients with Sjogren’s syndrome (SS). Materials and methods: This was a prospective cross-sectional descriptive observational study. Patients with SS were interviewed and physically explored. Nasolaryngeal endoscopy, video laryngeal stroboscopy, fiberoptic endoscopic evaluation of swallowing and computerized voice spectrographic analysis (PRAAT ® software) of voice and speech were also performed. Results: We included 31 patients (96.7% women). Average time of evolution was 5 years; mean age was 48.4 years. Of these SS cases, 87% were secondary and 13% primary. Symptomatology: 70.9% dysphagia, 41.9% dysphonia, 35.4% dysglossia, 3.2% dysarthria. We found abnormalities principally in: one or more cranial nerves (V, VII, IX, X, XII) (67.7%), nasopharyngolaryngeal mucosa (77.4%), mucosal wave of vocal cords (90%), swallowing mechanism (90.3%), spectrogram of the vowels /e/ (58.06%) and /i/ (51.61%), and rhythm of the trisyllable ‘‘pataka’’ (35.48%). Conclusions: Patients with SS have voice, speech and swallowing abnormalities, not only associated to xerosis but perhaps also to neurological abnormalities, probably secondary to the syndrome.

Alterations in Voice, Speech and Swallowing in Patients With Sjögren's Syndrome

Resumen Objetivo Identificar y describir alteraciones de voz, habla y deglucion en pacientes con sindrome de Sjogren (SS). Material y metodos Estudio prospectivo, descriptivo, transversal, observacional. Pacientes con SS fueron interrogados y explorados fisicamente, realizamos ademas nasofibrolaringoendoscopia, videolaringoestroboscopia, evaluacion fibroendoscopica de la deglucion y analisis espectrografico computarizado de voz y habla (software PRAAT®). Resultados Incluimos 31 pacientes (96,7% mujeres), tiempo de evolucion promedio 5 anos 8 meses, edad promedio 48,4 anos. El SS en 87% secundario, en 13% primario. Sintomatologia: 70,9% disfagia, 41,9% disfonia, 35,4% disglosias, 3,2% disartria. Encontramos principalmente alteraciones en: uno o mas pares craneales (V, VII, IX, X, XII) (67,7%), mucosa nasofaringolaringea (77,4%), ondulacion de la mucosa alterada en cuerdas vocales (90%), mecanismo de la deglucion (90,3%), espectrograma de vocales /e/ (58,06%), /i/ (51,61%) y ritmo del trisilabo «pataka» (35,48%). Conclusiones El SS presenta alteraciones en voz, habla y deglucion, quiza no solo asociadas a xerosis, sino tambien a trastornos neurologicos probablemente secundarios al sindrome.

Ángel Israel Pérez García

Papers

Alterations in Voice, Speech and Swallowing in Patients With Sjögren's Syndrome

Alteraciones de la voz, el habla y la deglución en pacientes con síndrome de Sjögren