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Swallowing Disorders in Sjögren's Syndrome: Prevalence, Risk Factors, and

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Dysphagia
DOI 10.1007/s00455-015-9657-7

ORIGINAL ARTICLE

Swallowing Disorders in Sjögren’s Syndrome: Prevalence, Risk

Factors, and Effects on Quality of Life
Jenny L. Pierce1 • Kristine Tanner2 • Ray M. Merrill3 • Karla L. Miller4 •

Katherine A. Kendall5 • Nelson Roy1

Received: 20 July 2015 / Accepted: 3 October 2015

Ó Springer Science+Business Media New York 2015

Abstract This epidemiological investigation examined
the prevalence, risk factors, and quality-of-life effects of
swallowing disorders in Sjögren’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 swal-
lowing 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
This work was performed at The University of Utah, Salt Lake City,
Utah, USA.
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 disor-
ders 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 dys-
phagia in this population.
Keywords Sjögren’s syndrome
Epidemiology

Deglutition disorders
Quality of life
Autoimmune
diseases

& Kristine Tanner

kristine_tanner@byu.edu Introduction
1
The Department of Communication Sciences and Disorders,
The University of Utah, Salt Lake City, UT, USA
Sjögren’s Syndrome (SS) is an autoimmune disease char-
2
acterized by salivary and lacrimal gland dysfunction
The Department of Communication Disorders, Brigham
Young University, 158 TLRB – BYU, Provo, UT 84602,
causing dryness (sicca), most notably of the mouth and
USA eyes, but with other potential effects on connective tissues,
3
The Department of Health Science, Brigham Young
internal organs, and digestion [1]. SS occurs in approxi-
University, Provo, UT, USA mately .2–4 % of the adult population and most frequently
4 in women during midlife [2–4]. SS often co-exists with
The Division of Rheumatology, Department of Internal
Medicine, The University of Utah, Salt Lake City, UT, USA other autoimmune diseases and is associated with increased
5 risk for lymphoma [2–7]. Both oropharyngeal and eso-
Division of Otolaryngology–Head and Neck Surgery, The
Department of Surgery, The University of Utah, phageal swallowing disorders have been reported in this
Salt Lake City, UT, USA population, often attributed to sicca of the mouth and

123

throat, loss of dentition, cricoarytenoid joint arthritis,
neuropathy, esophageal abnormalities, and gastroe-
sophageal reflux [8–13]. The reported prevalence of swal-
lowing disorders in SS ranges widely from 32 to 71 % [13,
14], as compared to the general adult population (12 %)
[15]. Previous studies of swallowing disorders in SS have
employed chart review [13], questionnaire [12, 16, 17], as
well as quasi-objective and subjective assessments of
swallow function [9, 12–14, 16, 17]. While each of these
studies provides important insight into the effects of SS on
swallowing, they are characterized by small numbers of
participants; lacked specific detail regarding disease his-
tory, severity, and other potential risk factors that may
contribute to dysphagia; and did not employ psychometri-
cally validated quality-of-life measures [9, 12–14, 16, 17].
Given these limitations and diversity of methodologies,
considerable uncertainty exists regarding the true preva-
lence, risk factors, and quality-of-life burden of swallowing
disorders in SS. Valid descriptive epidemiological data
regarding swallowing disorders in patients with SS are a
necessary first step to provide important information
regarding the extent of dysphagia in SS and to guide future
experimental studies. Thus, the present descriptive epi-
demiological investigation was undertaken to (1) determine
the prevalence of swallowing disorders in a large cohort of
patients diagnosed with SS, (2) identify risk factors for
these disorders, and (3) examine the functional, social,
occupational, and emotional effects of swallowing disor-
ders in this population.
Materials and Methods
Participants
Patients diagnosed with primary or secondary SS were
identified from a 3-year retrospective chart review and
invited to participate. Diagnosis was assigned by a
rheumatologist with specific expertise in SS at The
University of Utah Hospital, with diagnostic criteria
including clinical presentation of sicca symptoms, positive
SS-A or SS-B antibodies, and/or positive minor salivary
gland findings from lip biopsy. Inclusion criteria consisted
of SS diagnosis, over age 18, hearing adequate for a
lengthy telephone interview, no known cognitive deficits,
and English speaking. Sixty-three percent of eligible par-
ticipants (i.e., 101 of the 160 patients contacted who met
the inclusion criteria) agreed to participate.
Data Collection
Participants were interviewed via telephone during a
6-month period by trained students in the Department of
123
J. L. Pierce et al.: Swallowing Disorders in Sjögren’s Syndrome...
Communication Sciences and Disorders at The University
of Utah (IRB#00058438). Interviewer training included
individual instruction on questionnaire administration,
observation of initial administration of the questionnaire by
the first author (J.P.), practice on non-participants prior to
administration on participants, frequent audits of collected
data, and bi-monthly research team meetings to ensure
accuracy of administration. Additionally, if audits revealed
ambiguous or missing responses, interviewers re-contacted
participants to clarify responses. Interview questions
involved swallowing disorders and symptoms, medical
histories, occupational histories, social histories, and
additional disease and disorder-related psychometrically
validated scales, including the European League Against
Rheumatism Sjögren’s Syndrome Patient Reported Index
(ESSPRI) [18], the Sicca Symptoms Inventory-Short Form
(SSI) [19], the M.D. Anderson Dysphagia Inventory
(MDADI) [20], and the Short Form 36 Health Survey (SF-
36) [21]. A swallowing disorder was defined as ‘‘any time
an individual experiences difficulty moving food or liquid
from mouth to stomach, or experiences choking or throat
clearing during or following mealtime’’ [22]. It should be
noted that this study was undertaken in conjunction with
two other concurrent studies [23, 24] examining voice
disorders in the same large cohort of patients with SS.
Statistical Analyses
Interview data were examined using contingency tables,
summary statistics, Chi-square tests (v2), and risk ratios
(RRs). Significant main and interaction effects were eval-
uated using multiple logistic regression. To minimize the
probability of type II error, variables significant at the .2
level were retained in the regression models. Two-sided
tests of significance were based on the .05 level against a
null hypothesis of no association. Statistical analyses were
performed using SAS version 9.1 (SAS Institute Inc., Cary,
NC, USA, 2003).
Results
Participant Demographic and Disease Severity
Characteristics
The study cohort included 94 females and 7 males, with
ages ranging from 20.5 to 93.4 years (M = 59.4,
SD = 14.1). Most participants were non-Hispanic White
with at least some college education. The length of time
from when participants were diagnosed with SS ranged
from 1 to 54 years (M = 10.5 years, SD = 9.9). The
duration of medication use ranged from 1 to 53 years
(M = 9.4 years, SD = 9.8), and the majority of
J. L. Pierce et al.: Swallowing Disorders in Sjögren’s Syndrome...
participants were currently taking medication for SS
(n = 76, 75.2 %). Of all demographic variables, length of
time with SS was only significantly associated with eth-
nicity (M = 12.4 years, SD = 11.1 for Hispanic vs.
M = 10.3 years, SD = 9.8 for non-Hispanic, p = .005).
Medication use and duration of use were not significantly
associated with any demographic variable. According to
the participant-reported SS severity scales, SSI scores
ranged from 0 to 26 (M = 14.3, SD = 5.3) and ESSPRI
scores ranged from 1 to 9.7 (M = 5.9, SD = 2.0). Severity
based on these scores did not significantly differ across any
demographic variables (age, sex, race, ethnicity, income, or
education), the number of years with SS, current medica-
tion use, or the years of medication use. Possible additional
comorbid autoimmune conditions included rheumatoid
arthritis, mixed connective tissue disease, systemic lupus,
type 1 diabetes, Wegener’s granulomatosis, scleroderma,
dermatomyositis, and polymyositis. Primary SS was
reported by 56 participants (55.45 %), and secondary SS
was reported by 44 (45.6 %, SS?). The most common
comorbid autoimmune combination was SS and rheuma-
toid arthritis (n = 26, 25.7 %). SS only versus SS ? did
not significantly differ by sex, age, education, ethnicity, or
race.
Prevalence of Swallowing Disorders
Approximately two-thirds of participants (n = 65, 64.4 %)
classified themselves as having a current swallowing disor-
der. The prevalence of a current swallowing disorder did not
significantly differ with length of time with SS. Only one
demographic variable—sex—was found to significantly
differ when reporting a swallowing disorder (100 % of males
vs. 61.7 % of females, p = .041); however, males were also
underrepresented in this study (n = 7). Of the comorbid
autoimmune conditions, only mixed connective tissue dis-
ease (n = 7) was significantly associated with reporting a
current swallowing disorder (v2, p = .041). In most partic-
ipants (n = 55, 84.6 %), the swallowing disorder began
gradually and lasted for more than 4 weeks (n = 59,
90.8 %). For those with a swallowing disorder, 15 % first
noticed the disorder within the past year, 18.3 % 1–3 years
earlier, 30 % 4–9 years earlier, and 36.7 % 10 or more years
earlier. Reporting a swallowing disorder was also signifi-
cantly associated with SS severity based on mean SSI scores
[15.8 (SD = 5.2) vs. 11.5 (SD = 4.2), p \ .0001] and
approached significance based upon mean ESSPRI scores
[6.2 (SD = 1.9) vs. 5.5 (SD = 2), p = .085).
Swallowing Symptoms
Selected swallowing-related symptoms were analyzed in
the present SS cohort. Details of symptom frequency, along
with RRs to quantify how much more likely someone is to
report a swallowing disorder if they report a specific
symptom, are presented in Table 1. Virtually, all of the 101
participants reported at least one swallowing symptom
(n = 99, 98 %). Of the 19 swallowing symptoms, 5 were
reported by more than 50 % of participants and 13 were
significantly associated with reporting a swallowing dis-
order (indicated in bold in Table 1). The majority of the
selected swallowing symptoms occurred daily in most
participants [11 (57.9 %) daily, 6 (31.6 %) weekly, 2
(10.5 %) monthly, 1 (5.3 %) several times a year].
Severity of SS was also compared with swallowing
symptoms. Regression analysis showed that the SSI
severity score was simultaneously significantly associated
with three symptoms: taking smaller bites (partial
r2 = .161, p \ .0001), mucous/phlegm while eating (par-
tial r2 = .068, p = .004), and difficulty placing food in the
mouth (partial r2 = .050, p = .011). ESSPRI severity
score was simultaneously significantly associated with two
symptoms: mucous/phlegm while eating (partial r2 = .088,
p = .003) and wheezing while eating (partial r2 = .056,
p = .013).
General Health, Lifestyle, Voice Use Patterns,
and Swallowing Disorders/Symptoms
The potential influence of possible risk factors, including
participants’ general health, lifestyle, and voice use pat-
terns, on reporting a current swallowing disorder was
examined. Questions regarding voice use patterns were
considered for analyses to examine if a link between voice
and swallowing might exist. Table 2 includes each condi-
tion addressed, the number of participants with each con-
dition who also reported a current swallowing disorder with
all significant correlations bolded, p \ .05, as well as RRs
and associated CIs.
Similar to self-reported swallowing disorders, the only
demographic variable significantly associated with specific
swallowing symptoms was being male. Stepwise regression
showed that swallowing symptoms were significantly
associated with many risk factors, and polytomous logistic
and stepwise regression showed that symptom frequencies
were also significantly associated. Sixty (51 %) of the 118
risk factors analyzed were significantly associated with the
presence and frequency of swallowing symptoms. Risk
factors that were associated with the presence or frequency
of at least 3 of the 19 dysphagic symptoms included eso-
phageal reflux (associated with 7 symptoms), frequent
coughing (associated with 5 symptoms), stomach or duo-
denal ulcer (associated with 4 symptoms), frequent sinus
infections (associated with 4 symptoms), current choral
singing (associated with 4 symptoms), being male (asso-
ciated with 4 symptoms), kidney problems (associated with
123
 J. L. Pierce et al.: Swallowing Disorders in Sjögren’s Syndrome...

Table 1 Prevalence and frequency of selected swallowing-related symptoms and presence of a current swallowing disorder
Symptom Current Frequencya Current v2 RR [95 % CI]c
swallowing
disorderb
n (%) n (%) p value*

Take longer time to eat because of swallowing problem 48 (47.5) 1 44 (91.67) <.0001 2.3 [1.6, 3.3]
Difficulty swallowing liquids 21 (20.8) 2 20 (95.24) .001 1.7 [1.4, 2.1]
Difficulty swallowing solids 65 (64.4) 1 55 (84.62) <.0001 3.0 [1.8. 5.2]
Difficulty swallowing medications 47 (46.5) 1 35 (74.47) .049 1.3 [1.0, 1.8]
Gurgly or wet voice during or after eating 5 (5.0) 2–3 4 (80) .456
Coughing, throat clearing, or choking before, during, or after eating 46 (45.5) 1 39 (84.78) <.0001 1.8 [1.3, 2.4]
Inability to control food, liquid, or saliva in the mouth 28 (27.7) 1 24 (85.71) .006 1.5 [1.2, 2]
Sneezing during or after a meal 12 (11.9) 2 9 (75) .415
Pain or pressure in the throat or chest during swallowing 40 (39.6) 2 39 (97.5) <.0001 2.3 [1.7, 3.1]
Wheezing after eating 6 (5.9) 1 6 (100) .061
Food comes out of the nose while eating 3 (3.0) 4 3 (100) .193
Need to chew excessively in order to swallow safely 55 (54.5) 1 47 (85.45) <.0001 2.2 [1.5, 3.2]
Dry mouth 97 (96.0) 1 63 (64.95) .543
Difficulty placing food in mouth 3 (3.0) 2 3 (100) .193
Sensation of food sticking in throat 62 (61.4) 2 54 (87.1) <.0001 3.1 [1.9, 5.1]
Forcibly regurgitate food stuck in throat 18 (17.8) 3 18 (100) .001 1.8 [1.5, 2.1]
Avoid eating certain foods because of swallowing disorder 41 (40.6) 1 36 (87.8) <.0001 1.8 [1.4, 2.4]
Increased mucous or phlegm in throat before, during, or after eating 30 (29.7) 1 25 (83.33) .010 1.5 [1.4, 1.9]
Take smaller bites of food to swallow safely 55 (54.5) 1 48 (87.27) <.0001 2.4 [1.6, 3.5]
RR risk ratio
* Only p values significant at the \.05 level are bolded
a
Frequency of experiencing the symptom, i.e., 1 = daily, 2 = weekly, 3 = monthly, 4 = several times a year, 5 = yearly or less
b
Number and percentage of participants with a particular swallowing symptom who also reported a current swallowing disorder (defined as
‘‘any time an individual experiences difficulty moving food or liquid from mouth to stomach, or experiences choking or throat clearing during or
following mealtime’’)
c
Only significant RRs are reported indicating that the presence and frequency of a particular swallow symptom significantly elevated the
probability of reporting a current swallowing disorder

3 symptoms), bronchitis (associated with 3 symptoms),
chronic pain (associated with 3 symptoms), sleep disorder
(associated with 3 symptoms), abdomen tension (associ-
ated with 3 symptoms), past church singing (associated
with 3 symptoms), past volunteer activities (associated
with 3 symptoms), acidic foods (associated with 3 symp-
toms), and past exposure to secondary tobacco smoke
(associated with 3 symptoms).
Swallowing Disorders and Quality of Life
Scores on the MDADI swallowing-related quality-of-life
measure can range from 0 (poorest quality of life) to 100
(best quality of life). Participants’ scores ranged from 30 to
100, and scores were significantly lower for those who
indicated that they had a current swallowing disorder
(M = 75.9, SD = 15.4 vs. 90.1, SD = 11.5, p \ .0001).
MDADI was not significantly associated with age, sex,
race, ethnicity, income, or education. Mean MDADI score
was significantly lower for 15 of the 19 selected swal-
lowing-related symptoms (see Table 3). Regression anal-
ysis showed that the MDADI measure was significantly
associated with SSI severity scores (p = .001) and ESSPRI
severity scores (p = .004).
The relationship between current swallowing disorders
and specific dysphagic symptoms was assessed using the
SF-36—a general quality-of-life measure. The two sum-
mary component measures of the SF-36 are physical health
and mental health. Each summary measure is associated
with four subscales (physical health: physical function, role
physical, bodily pain, general health; mental health: vital-
ity, social functioning, role emotional, mental health) (see
Appendix for description of SF-36 scales). The relationship
between reporting a swallowing disorder and the physical
(p = .060) and mental health (p = .085) summary mea-
sures of the SF-36 approached significance. Of the eight
subscales, a swallowing disorder was significantly associ-
ated with reduced quality of life on two of the scales:

123
J. L. Pierce et al.: Swallowing Disorders in Sjögren’s Syndrome...

Table 2 Participants with current swallowing disorder and selected medical conditions
Condition n (%)a RR [95 % CI]

Medical condition
Arthritis 38 (63)
Rheumatoid arthritis 20 (59)
Heart disease 9 (82)
Hypertension 29 (64)
Circulatory problems 25 (74)
Kidney problems 15 (75)
Thyroid problems 30 (67)
Stomach or duodenal ulcer 24 (80) 1.38 [1.06, 1.81]
Esophageal reflux 49 (79) 1.93 [1.29, 2.87]
Stroke 10 (83)
Respiratory allergies 25 (69)
Pneumonia 40 (71)
Emphysema 3 (75)
COPD 1 (50)
Hearing loss 23 (74)
Bronchitis 38 (66)
Asthma 15 (68)
Severe neck, back, or head injury 19 (58)
Chronic pain 44 (66)
Cancer 15 (68)
Depression or anxiety 43 (72)
Sleep disorder 20 (77)
Stopped menstrual periods 43 (58)
Colds (3? vs. \3 year) 48 (64)
Sinus infections (3? vs. \3 year) 47 (64)
Sore throat (3? vs. \3 year) 42 (60)
Post-nasal drip (chronically vs. less) 28 (54) 1.40 [1.04, 1.89]
Medications for the following conditions
Bronchitis 12 (75)
Asthma 32 (71)
Severe neck, back, or head injury 3 (75)
Chronic pain 26 (68)
Cancer 10 (67)
Depression or anxiety 12 (75)
Sleep disorder 32 (71)
Head and neck surgery 32 (70)
Breathing assistance 11 (73)
Ever used tobacco products 15 (65)
At any year in your life, drank an average of one or more alcoholic 22 (59)
beverages a week
Ever used recreational drugs 5 (71)
Experience tension
Neck/Throat (often/constantly vs. otherwise) 26 (49) 1.66 [1.22, 2.25]
Jaw 46 (60)
Shoulders 35 (60)
Abdomen 56 (63)
Family history of any type of swallowing problem 12 (71)

123
 J. L. Pierce et al.: Swallowing Disorders in Sjögren’s Syndrome...

Table 2 continued
Condition n (%)a RR [95 % CI]

Voice activity
Talk (often/constantly vs. otherwise) 13 (65)
Talk quietly 37 (65)
Whisper 60 (63)
Talk loudly 40 (62)
Sing 56 (64)
Shout, yell, or cheer 61 (63)
Clear your throat 35 (56) 1.42 [1.08, 1.87]
Laugh 27 (64)
Cough 45 (59)
Ever in an occupation requiring you to talk a lot on a daily basis 52 (64)
Experience voice disorders with this job 25 (69)
Voice problem
Current voice problem 41 (68)
Did the problem begin suddenly? 5 (71)
Did the problem last more than 4 weeks? 38 (76) 2.00 [1.18, 3.38]
Past voice problem 19 (73)
Family history of any type of voice problem 5 (63)
Exposures
Excess dust-current 9 (53)
Excess dust-past 31 (70)
Fumes from cleaning products-current 19 (66)
Fumes from cleaning products-past 31 (67)
Secondary tobacco smoke-current 11 (100) 1.67 [1.41, 1.97]
Secondary tobacco smoke-past 42 (70)
Dry air-current 54 (64)
Dry air-past 56 (64)
Exercise (yes vs. no) 56 (62)
Personality Description
Quiet versus talkative 25 (63)
Easy-going versus worrier 35 (65)
Active versus inactive 53 (65)
Happy versus sad 58 (63)
* Only conditions with values significant at the \.05 level are bolded
a
n and % = number and percent of those participants with the condition who also reported a swallowing disorder

mental health (M = 72.5, SD = 17.9, p = .042) and gen-
eral health (M = 33.9, SD = 20.1, p = .023). Addition-
ally, as detailed in Table 4 by means of slopes indicating a
positive (?) or negative (-) effect of swallowing symptoms
on SF-36 subscales, a logistics model showed that six
swallowing symptoms were independently associated with
the SF-36 subscales. These symptoms seem to dispropor-
tionately contribute to reductions in specific aspects of
quality of life, including bodily pain and vitality. For the
model with summary physical health as the dependent
variable, only food sticking in throat (slope = -9.4,
SE = 4.8, p = .009) was significantly predictive. For the
model with summary mental health as the dependent
variable, difficulty swallowing medication (slope = -7.2,
SE = 3.5, p = .046), wheezing while eating (slope =
-31.2, SE = 7.5, p = .001), and mucous/phlegm while
eating (slope = -8.6, SE = 3.9, p = .038) were indepen-
dent predictors.
Treatment-Seeking for Swallowing Disorders
Of the 65 participants who reported a current swallowing
disorder, only 27 (42 %) had ever sought professional help
to treat their swallowing problem. Of those who sought

123
J. L. Pierce et al.: Swallowing Disorders in Sjögren’s Syndrome...

Table 3 Differences observed on swallowing-related quality of life (i.e., MDADI scores) based upon the presence or absence of specific
swallowing symptoms
Symptom MDADI scorea
No current symptom Current symptom t statistic
M (SD) M (SD) p value*

Take longer time to eat because of swallowing problem (n = 48) 88.2 (11.4) 73.0 (15.9) <.0001
Difficulty swallowing liquids (n = 21) 82.4 (15.3) 75.8 (16.0) .086
Difficulty swallowing solids (n = 65) 87.8 (11.9) 77.2 (16.2) .001
Difficulty swallowing medications (n = 47) 84.7 (14.0) 76.7 (16.5) .010
Gurgly or wet voice during or after eating (n = 5) 81.8 (15.2) 64.8 (17.3) .017
Coughing, throat clearing, or choking before, during, or after eating (n = 46) 86.3 (12.6) 74.6 (16.6) .0001
Inability to control food, liquid, or saliva in the mouth (n = 28) 83.3 (14.2) 75.0 (17.8) .017
Sneezing during or after a meal (n = 12) 81.8 (14.5) 74.8 (22.1) .142
Pain or pressure in the throat or chest during swallowing (n = 40) 85.9 (14.0) 73.5 (15.1) <.0001
Wheezing after eating (n = 6) 82.4 (14.2) 58.0 (20.6) .0001
Food comes out of nose while eating (n = 3) 81.6 (15.2) 60.0 (17.8) .018
A need to chew excessively in order to swallow safely (n = 55) 90.0 (10.3) 73.5 (15.4) <.0001
Dry mouth (n = 97) 78.5 (24.6) 81.1 (15.3) .748
Difficulty placing food in mouth (n = 3) 81.2 (15.8) 73.3 (7.4) .393
Sensation of food sticking in throat (n = 62) 88.7 (12.8) 76.1 (15.4) <.0001
Forcibly regurgitate food stuck in throat (n = 18) 83.9 (13.4) 67.7 (18.6) <.0001
Avoid eating certain foods because of swallowing disorder (n = 41) 87.8 (11.6) 71.0 (15.5) <.0001
Increased mucous or phlegm in throat before, during, or after eating (n = 30) 83.8 (13.8) 74.3 (17.8) .005
Take smaller bites of food to swallow safely (n = 55) 90.5 (9.7) 73.1 (15.3) <.0001
* Only p values significant at the \.05 level are bolded
a
Scores on the MDADI can range from 0 (poorest quality of life) to 100 (best quality of life)

help, 21 saw a physician, 5 saw a speech-language
pathologist, 3 saw a dietician, and 3 saw another provider.
Of the 27 who sought help, 15 (55.6 %) indicated that it
helped their swallowing.
Association of Swallowing and Voice Disorders
To assess the comorbidity of voice and swallowing disorders
in SS, frequency counts were undertaken. Of the 101 par-
ticipants, 41 (40.6 %) reported both voice and swallowing
disorders, 24 (23.8 %) reported a current swallowing disor-
der but not a current voice disorder, 19 (18.8 %) reported a
current voice disorder but not a current swallowing disorder,
and only 17 (16.8 %) reported having neither.
Discussion
Prevalence and Risk Factors
The current epidemiological investigation of swallowing
disorders in SS describes self-reported data from 101
participants. A large range of ages and SS severity were
observed and encompassed the spectrum of those with this
condition. The majority (64 %) of participants reported a
current swallowing disorder, which is at the upper range of
32–71 % previously reported by other studies (with far
fewer participants and different definitions of swallowing
disorders compared to the current study) [13, 14, 16]. The
prevalence rate in this cohort is higher than that reported in
the general adult population and the general geriatric
population (12–13 and 33 %, respectively) [15, 22]. Most
of the swallowing disorders in the current study had a
gradual onset (85 %) and were chronic (91 %) and long-
standing (i.e., 85 % persisted for 1? years). A current
swallowing disorder was associated with increasing SS
severity, but, interestingly, it was not associated with age.
Although results revealed a higher prevalence of swal-
lowing disorders in males, this finding must be considered
in the context of underrepresentation of males in the pre-
sent study (i.e., n = 7 males versus 94 females); future
research with greater representation of males with SS is
needed. This study did not find robust evidence to suggest
that swallowing disorders differed on the basis of primary

123
123
Table 4 Slope estimates (including means and standard errors) for SF-36 subscale scores according to current swallowing symptoms
Symptom (n) PFa RP BP GH VT SF RE MH
slope (SE) slope (SE) slope (SE) slope (SE) slope (SE) slope (SE) slope (SE) slope (SE)

Longer time to eat (48)

Difficulty swallowing liquids (21)
Difficulty swallowing solids (65)
Difficulty swallowing meds (47) 210.5 (4.6)
Gurgly/wet voice with eating (5)
Cough/throat clear/choke with eating (46)
Unable to control food/liquid/saliva in mouth (28)
Sneezing with eating (12) 29.8 (7.4)
Throat/chest pain/pressure during swallowing (40)
Wheezing after eating (6) 225.0 (9.6) 236.1 (12.9) 237.5 (12.4) 221.6 (6.7)
Food comes out of nose while eating (3)
Excessive chewing to swallow safely (55)
Dry mouth (97)
Difficulty placing food in mouth (3)
Food sticking in throat (62) 213.2 (5.9) 214.6 (4.1)
Need to regurgitate food stuck in throat (18)
Avoid eating certain foods due to poor swallowing (41)
Increased mucous/phlegm in throat with eating (30) 215.2 (5.1)
Smaller bites of food to swallow safely (55) 27.5 (5.3)
* Only significant slope estimates appear in the table, p \ .05. The magnitude and direction (- or ?) of the slope estimate indicate the extent to which the swallowing symptom is associated
with a change on a specific SF-36 subscale. All values in the table are associated with reductions on the respective subscales, with lower values corresponding to larger reductions in quality of
life. For instance, the symptom ‘‘food sticking in the throat’’ was associated with a 13.2 and 14.6 point reduction on the Physical Function (PF) and General Health (GH) scales, respectively, of
the SF-36
a
Physical Health: Physical Function (PF), Role Physical (RP), Bodily Pain (BP), General Health (GH). Mental Health: Vitality (VT), Social Functioning (SF), Role Emotional (RE), Mental
Health (MH) (See the Appendix for descriptions of each scale)
J. L. Pierce et al.: Swallowing Disorders in Sjögren’s Syndrome...
J. L. Pierce et al.: Swallowing Disorders in Sjögren’s Syndrome...
versus secondary SS, as only one comorbid autoimmune
condition, mixed connective tissue disease, was associated
with reporting a swallowing disorder, and this condition
was similarly underrepresented and present in only seven
participants. These results confirm that chronic, long-
standing swallowing disorders are common in SS and
appear to be related to worsening SS severity irrespective
of years with SS, primary versus secondary SS, or age.
Additionally, the use of SS medications, or medications for
other medical conditions included in the questionnaire, did
not appear to impact the nature and severity of swallowing
disorders.
In addition to swallowing disorders, specific swallowing
symptoms were reported by nearly all participants (98 %
reported at least one symptom), which often occurred on a
daily or weekly basis. This rate is similar to the prevalence
of these symptoms reported elsewhere at 80–100 % [12,
14, 16]. In the current study, increased likelihood of
reporting a current swallowing disorder was associated
with 13 of the 19 swallowing symptoms studied. The five
symptoms reported by more than half of all participants
were dry mouth (reported by 96 %), difficulty swallowing
solids (64 %), food sticking in the throat (61 %), need for
excessive chewing to swallow safely (55 %), and taking
smaller bites to swallow safely (55 %). Although it was the
most prominent symptom in the current study, Doig et al.
(1971) and Mandl et al. (2007) did not include dry mouth
in their listed symptoms, and their prevalence of symptoms
was 81 and 80 %, respectively. In the current study, the
presence and frequency of dysphagic symptoms were sig-
nificantly related to participant-reported SS severity on
both the SSI and ESSPRI measures, suggesting that
worsening of SS severity is associated with increased
prevalence of swallowing symptoms.
This study also examined medical conditions and
potential lifestyle or occupational factors that possibly
increased vulnerability for experiencing a swallowing dis-
order or symptoms. Based upon inspection of the RR data,
several groupings emerged. Some risk factors for reporting a
swallowing disorder, symptoms, or both were laryngeal
irritants, such as exposure to secondary tobacco smoke, post-
nasal drip, frequent sinus infections, esophageal reflux, and
acidic foods which may be related to reflux. Reflux, which
had among the highest RRs, has been reported previously as
a significant risk factor for swallowing disorders in SS [13,
16] and was the most common cause of dysphagia in the
general population seeking medical attention for dysphagia
[25]. It is also possible that reflux might be more prevalent
than the self-report data indicate due to the possibility of
silent reflux. Saliva neutralizes acid, and the lack of saliva
inherent in SS may make reflux symptoms more pro-
nounced. Reflux and other laryngeal irritants potentially
inflame the laryngeal structures within the upper
aerodigestive tract and, therefore, may contribute to swal-
lowing dysfunction. These laryngeal irritants may also
contribute to another risk factor, frequent throat clearing or
coughing, which may be a way the body attempts to clear
these irritants, or a way to compensate for dryness or
laryngeal dysfunction associated with SS. Several other risk
factors which were statistically associated with reporting a
swallowing disorder included neck/throat tension, voice
problems lasting longer than 4 weeks, current choral sing-
ing, past church singing, and past volunteer activities. These
risk factors seem to have anatomical connections to struc-
tures involved in swallowing and demonstrate a link
between voice and swallowing. Of the medical, occupa-
tional, and lifestyle risk factors considered, 51 % were
associated with at least one, and up to seven, swallowing
symptoms. Such a diversity of risk factors may indicate that
SS may be the true risk factor for swallowing symptoms,
with increased prevalence of symptoms with additional
medical and lifestyle components.
Effects of Swallowing Symptoms and Disorders
on Quality of Life in SS
Quality of life was negatively impacted by the high fre-
quency of swallowing-related problems reported by par-
ticipants. Inspection of the MDADI results revealed that
swallowing disorders in SS produced mild to moderate
reductions in swallowing-related quality of life. Lower
MDADI scores were associated with increasing severity of
SS. Our finding that SS severity was not correlated with
years with SS, but was correlated with lower MDADI
scores, is important. This indicates that a person with SS
can experience reduced swallowing-related quality of life
at any time point during their disease process.
The SF-36 general health quality-of-life measure also
revealed important findings. Those who reported a swal-
lowing disorder also had poorer quality-of-life scores on two
of the SF-36 subscales as compared to those who did not
report a swallowing disorder. Furthermore, specific symp-
toms including difficulty swallowing medications, sneezing
while eating, wheezing while eating, food stuck in the throat,
mucous/phlegm while eating, and need to take smaller bites
were significantly associated with reductions on specific
subscales of the SF-36. Of these symptoms, those indepen-
dently associated with reduced SF-36 physical and mental
health summary measures were difficulty swallowing med-
ications, wheezing with eating, food stuck in the throat, and
mucous/phlegm while eating. The origin of these specific
symptoms needs to be better understood, as they seemingly
contribute disproportionately to the quality-of-life burden
experienced by patients with SS.
Although 64 % of participants reported a swallowing
disorder which contributed to significant reductions in
123
 J. L. Pierce et al.: Swallowing Disorders in Sjögren’s Syndrome...

quality of life, only 27 % had ever sought professional help Further research is warranted to better understand the nature
to treat their swallowing disorder. One possible explanation of these disorders and their possible treatment.
for this low rate of seeking treatment may be limited edu-
cation regarding prevalence of and treatment for swallowing Acknowledgments This investigation was supported, in part, by a
David O. McKay School of Education Planning Grant, Brigham
disorders in this population. Regression analysis identified Young University. Research assistance was provided by undergrad-
that as SS severity worsens, so do swallowing disorders. On uate students in the voice laboratory in the Department of Commu-
average, the participants in this study reported moderate SS nication Sciences and Disorders at the University of Utah.
severity. Perhaps, swallowing problems and quality-of-life
Compliance with Ethical Standards
ratings would be more negatively impacted in individuals
with more severe SS. Finally, although 56 % of treatment- Conflict of Interest The authors declare that they have no conflict
seeking participants indicated improvement following of interest.
treatment, it is unclear from this study what treat-
ment(s) were associated with this improvement. Informed consent Informed consent was obtained from
participants.

Association of Swallowing and Voice Disorders

Appendix: SF-36 health survey (adopted from:
The results confirm frequent comorbidity of voice and
Ware et al. [21] comparison of methods
swallowing disorders in the SS population. With 41 % of
for the scoring and statistical analysis of SF-36
participants reporting both voice and swallowing disorders
health profile and summary measures: summary
and only 17 % reporting neither, roughly 83 % of partici-
of results from the medical outcomes study)
pants had a voice disorder, swallowing disorder, or both.
These coexisting problems logically may be linked to the
Scale Descriptions
shared anatomy underlying both swallowing and phonation
and the detrimental effects dryness can have on both of these
Physical component summary (PCS): Summary measure
functions. For detailed discussion surrounding prevalence,
assesses limitations in aspects of physical functioning, such
risk factors, and quality-of-life effects of voice disorders in
as self-care, level of activity, fatigue, pain, and participa-
SS, the interested reader is directed to the aforementioned
tion in life roles. The PCS includes the following subscales:
studies of voice disorders in SS in this cohort [23, 24].
Physical functioning (PF): ten items assess the ability to
Study Limitations perform physical tasks ranging from dressing to vigorous
activity
Although this investigation represents the largest epi- Role physical (RP): four items assess the ability to
demiological study of dysphagia in SS to date, some lim- participate in life roles, such as work or other daily
itations warrant consideration. First, it is important to activities, as a result of any physical problems
consider that this was a descriptive self-report study Bodily pain (BP): two items assess the severity and
wherein patients provided their own health history on the impact of general pain.
survey questionnaire. Additionally, while interviewers General health (GH): five items assess overall physical
received thorough and ongoing training to ensure consis- health status, susceptibility to illness, and expectations
tency of administration, formal inter-rater reliability (e.g., for future health status.
video recording of interviews) was not conducted due to
Mental component summary (MCS): Summary measure
the telephone questionnaire methodology. These concerns
assesses limitations in aspects of mental health, including
notwithstanding, the present investigation offers important
elements of psychological and emotional well-being. The
findings related to the prevalence, risks for, and impact of
MCS includes the following subscales:
swallowing problems in patients with SS.
Vitality (VT): four items assess overall level of energy.
Social functioning (SF): two items assess the ability to
Conclusion participate in normal social activities as a result of
physical or emotional problems.
Swallowing disorders in SS are frequent and associated with Role emotional (RE): three items assess the ability to
a significant quality-of-life burden. The prevalence of participate in life roles, such as time and quality applied
swallowing disorders and symptoms in SS was associated to work or other daily activities, as a result of any
with overall disease severity and coexisting voice disorders. emotional problems.

123
J. L. Pierce et al.: Swallowing Disorders in Sjögren’s Syndrome...
Mental health (MH): five items assess the frequency of
different moods, such as nervousness, depression, hap-
piness, and calmness.
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Jenny L. Pierce MS
Kristine Tanner PhD
Ray M. Merrill PhD, MPH
Karla L. Miller MD
Katherine A. Kendall MD
Nelson Roy PhD
123