ORIGINAL RESEARCH
published: 18 November 2021
doi: 10.3389/fmed.2021.770422
The Experiences of Functioning and
Health of Patients With Primary
Sjögren’s Syndrome: A Multicenter
Qualitative European Study
Julia Unger 1,2 , Malin Mattsson 3,4 , Răzvan G. Drăgoi 5 , Claudiu Avram 6 , Carina Boström 3 ,
Frank Buttgereit 7 , Angelika Lackner 1 , Torsten Witte 8 , Bernd Raffeiner 9 , Peter Peichl 10 ,
Martina Durechova 11 , Josef Hermann 1 , Tanja A. Stamm 12* and Christian Dejaco 1,13
1
Edited by:
Rosaria Talarico,
University of Pisa, Italy
Reviewed by:
Alen Zabotti,
Università degli Studi di Udine, Italy
Daniela Costa,
Universidade NOVA de
Lisboa, Portugal
*Correspondence:
Tanja A. Stamm
tanja.stamm@meduniwien.ac.at
orcid.org/0000-0003-3073-7284
Specialty section:
This article was submitted to
Rheumatology,
a section of the journal
Frontiers in Medicine
Received: 03 September 2021
Accepted: 05 October 2021
Published: 18 November 2021
Citation:
Unger J, Mattsson M, Drăgoi RG,
Avram C, Boström C, Buttgereit F,
Lackner A, Witte T, Raffeiner B,
Peichl P, Durechova M, Hermann J,
Stamm TA and Dejaco C (2021) The
Experiences of Functioning and Health
of Patients With Primary Sjögren’s
Syndrome: A Multicenter Qualitative
European Study.
Front. Med. 8:770422.
doi: 10.3389/fmed.2021.770422
Frontiers in Medicine | www.frontiersin.org
Department of Rheumatology and Immunology, Medical University of Graz, Graz, Austria, 2 Department of Health Studies,
Institute of Occupational Therapy, University of Applied Sciences FH JOANNEUM, Bad Gleichenberg, Austria, 3 Division of
Physiotherapy, Department of Neurobiology, Care Sciences and Society, Karolinska Institutet, and Affiliated to Women’s
Health and Allied Health Professionals Theme, Medical Unit Occupational Therapy and Physical Therapy, Karolinska
University Hospital, Stockholm, Sweden, 4 Department of Physiotherapy, Sunderby Hospital, Luleå, Sweden, 5 Department of
Balneology, Rehabilitation Medicine and Rheumatology, “Victor Babeş” University of Medicine and Pharmacy Timisoara,
Timisoara, Romania, 6 Department of Physical Therapy and Special Motility, West University of Timisoara, Timisoara,
Romania, 7 Department of Rheumatology and Clinical Immunology, Charitè University Medicine, Berlin, Germany,
8
Department of Rheumatology and Immunology, Medical University of Hanover, Hanover, Germany, 9 Department of
Rheumatology, Hospital of Bolzano (SABES-ASDAA), Bolzano, Italy, 10 Evangelic Hospital of Vienna, Vienna, Austria,
11
Division of Rheumatology and Immunology, Department of Internal Medicine, Medical University Vienna, Vienna, Austria,
12
Section for Outcomes Research, Centre for Medical Statistics, Informatics, and Intelligent Systems, Medical University
Vienna, Vienna, Austria, 13 Department of Rheumatology, Hospital of Brunico (SABES-ASDAA), Brunico, Italy
Objective: To identify a spectrum of perspectives on functioning and health of patients
with primary Sjögren’s syndrome (pSS) from the five European countries in order to reveal
commonalities and insights in their experiences.
Methods: A multicenter focus group study on the patients with pSS about their
perspectives of functioning and health was performed. Focus groups were chaired by
trained moderators based on an interview guide, audiotaped, and transcribed. After
conducting a meaning condensation analysis of each focus group, we subsequently
combined the extracted concepts from each country and mapped them to the
International Classification of Functioning, Disability and Health (ICF).
Results: Fifty-one patients with pSS participated in 12 focus groups. We identified
a total of 82 concepts meaningful to people with pSS. Of these, 55 (67%) were
mentioned by the patients with pSS in at least four of five countries and 36
(44%) emerged in all the five countries. Most concepts were assigned to the ICF
components activities and participation (n = 25, 30%), followed by 22 concepts
(27%) that were considered to be not definable or not covered by the ICF; 15
concepts (18%) linked to body structures and functions. Participants reported several
limitations in the daily life due to a mismatch between the capabilities of the
person, the demands of the environment and the requirements of the activities.
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Patient Perspectives in pSS
Conclusion: Concepts that emerged in all the five non-English speaking countries
may be used to guide the development and adaption of the patient-reported outcome
measures and to enhance the provision of treatment options based on the aspects
meaningful to patients with pSS in clinical routine.
Keywords: Sjögren’s syndrome, quality of life, PROMs, focus group technique, psychological impact, social
impact, physical impact, ICF
INTRODUCTION
descriptive way can facilitate more patient-centered decisions
(31) ultimately improving the quality of care.
Due to the increased relevance of the patient perspective (3),
there is a growing body of qualitative evidence on the experiences
of patients with pSS (7, 32–36). Thereby, the general impact of
pSS on (health related) quality of life (33) or daily life (32, 36) was
studied and specific aspects of the disease like physical, mental or
ocular fatigue (34), fatigue, sleep, and pain (35), sleep disruption
(7) or treatment experiences (32) were evaluated. However, all of
these previous studies were conducted on a national level. None
of them examined the perspectives of patients with pSS from
different countries in one study so far, focusing on a consistent
data collection and data analysis and taking potential cultural,
geographical, and social variations into account.
In this study, the main objective was to identify a spectrum of
perspectives on the functioning and health of patients with pSS
in the five European countries in order to reveal commonalities
and insights in their experiences.
Primary Sjögren’s syndrome (pSS) is an autoimmune disease
of unknown etiology with female predominance that is
characterized by an inflammation of exocrine glands, particularly
salivary and lachrymal glands as well as variable extra-glandular
manifestations such as musculoskeletal, gastrointestinal,
and/or neurological symptoms (1–3). Patients with pSS can
experience multiple facets of the mucosal dryness, pain,
fatigue, and other complaints resulting in an impairment
in everyday life and an altered health-related quality of life
(HRQoL) (4–9).
Perceptions of the disease may not only differ between the
patients and health professionals (10, 11), but also between
individuals suffering from the same condition (12). This is
majorly due to that the perception is built upon several mental
sources that are implicit and specific to the individuals (13).
A recent study in the psoriatic arthritis (PsA) highlighted
the influence of cultural backgrounds on differences in the
perception of illness (14). The illness perception is dependent on
the disease activity, and negative views of the illness are linked to
the worse future health outcomes (15–17) whereas positive beliefs
are associated with the better health outcomes (18–21).
Patient-reported outcomes (PROs) represent the “voice of
the patient” and are increasingly applied in the research studies
and clinical practice to ensure a patient-centered care (22, 23).
PRO Measurements (PROMs) are the instruments to measure
PROs. Several PROMs are available for pSS, however, only a
few of them have been validated (24). Besides, the patients
from non-English countries rarely participate in the studies
aimed at the development or validation of PROMs leading to
the underrepresentation of the specific cultural context of these
countries in the final PROM (25). PROMs are therefore often
cross-culturally adapted only later after their development (25,
26). However, the cross-cultural adaption of PROMs is linked
with challenges such as not being familiar with different cultures
or not considering geographical variations and differences in the
physical and social infrastructure. Thus, cultural equivalence is
not always achieved (25, 27–29).
An approach to truly capture what is important to the patients
is qualitative methodology. It offers the opportunity to explore
the perspectives, motivations, values, beliefs, and needs of the
patients in a scientific and systematic manner. A qualitative
approach supports the development of a deeper understanding of
the patient perspective and enables the examination of complex
issues that cannot be measured using strictly defined variables
(30). The aggregation of views of the patients in a non-numeric,
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PATIENTS AND METHODS
Study Design
We conducted a qualitative multicenter study using the focus
groups (37) in seven rheumatology centers in five European
countries, namely, Austria, Germany, Italy, Romania, and
Sweden. The “COnsolidated criteria for REporting QUalitative
research” (COREQ)—guideline provided by the EQUATOR
Network was used for the reporting of this study (38).
Patients and Sampling
Patients with pSS had to meet the American–European
Consensus Group Classification criteria, (6) and were recruited
via telephone or face-to-face by the local investigators (BR, FB,
TW, MD, MM, RD, CA, and PP) of the participating centers.
Patients who already participated in another study at the same
time or who had severe mental problems were excluded. Patients
had to be fluent in the local language. We aimed to follow
purposeful sampling by selecting patients of different ages and
gender. However, pSS is predominantly affecting women (1–
3, 39). According to the other qualitative studies in rheumatology
(30, 40) and to balance the data quality and data quantity, we
recruited two to four focus groups per country in order to gain
data of enough richness.
Data Collection
All focus groups were chaired by the trained moderators
at the local rheumatology centers: JU (female, PhD student,
occupational therapist) for Austria, Germany, and Italy, RD
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Patient Perspectives in pSS
and CA (males, PhD, medical doctors) for Romania and MM
(female, Ph.D., physiotherapist) for Sweden. We developed
a common interview guide, which was translated into local
languages, back translated, pilot tested, and refined. Interview
questions used in this study are depicted in the supplement files
(Supplementary Table S1). If necessary, field notes were taken
during the focus groups by the moderators. Each focus group
was audiotaped and transcribed in the local language. Ethical
approval was obtained in each center, and all the participants
provided oral and written informed consent.
the focus groups (JU, MM, and RD) was held. An experienced
researcher (TAS) moderated this meeting, at which concepts
from the five countries were compared and grouped together
according to their meaning. Thereby, concepts representing
the perspectives of the patients with pSS on functioning and
health were identified. In order to describe these concepts in
a standardized way with a common language, we used the
International Classification of Functioning, Disability and Health
(ICF) (43). This integrative biopsychosocial model, created by the
WHO, is a highly recognized framework for classification that
enables the description of problems of the patient, the selection
of important outcome domains or the comparison of health
information (43, 44). Hereinafter, two investigators (JU and AL)
independently linked the concepts identified in the focus groups
to the most precise ICF category according to the published
linking rules (45). In case of any disagreement, the consensus was
sought by discussion among the investigators.
Data Analysis
The data analysis was carried out independently by the local
researchers (JU for Austria, Germany, and Italy; RD for Romania;
MM and CB for Sweden), using the method of meaning
condensation (41). This involved the following four steps: first,
local researchers read through all the focus groups transcripts
and potential field notes of his/her center in order to become
familiar with the data material. Second, specific units of a text,
a few words, or a few sentences with a common meaning were
identified in the data and defined as “meaning units”. Third,
subconcepts contained in the “meaning units” were identified. A
“meaning unit” could obtain more than one subconcept. Fourth,
these subconcepts were grouped together to more comprehensive
“higher-level” concepts, which were formulated by the local
researchers in English language. A professional qualitative data
analysis and research software known as Atlas.ti was used for the
management of the data material (42).
After the focus groups were analyzed in each country, a oneday meeting with all researchers who conducted and analyzed
Rigor and Accuracy of the Qualitative Data
Collection and Analysis
In order to ensure rigor and accuracy of the study, we followed
several approaches. A detailed draft of the study protocol was
available for all the study members prior to the beginning of the
study. We ensured that the local researchers had the required
knowledge and conducted an extensive training and debriefing
session before data collection. Furthermore, we established
a detailed track record for the data collection process that
determined important conditions for conducting the focus
groups. After data retrieval, a pilot analysis was done by each
TABLE 1 | Demographic data of the participants and characteristics of the focus groups per country.
Demographic data and focus group characteristics
Number of participants
Women, n (%)
Age (years), mean (±SD)
Disease duration (years), mean
Austria
Germany
Italy
Romania
Sweden
Total
8
16
6
11
10
51
8 (100)
15 (94)
6 (100)
11 (100)
10 (100)
50 (98)
59 (±10.3)
57 (±14.9)
52 (±18.7)
65 (±10.1)
61 (± 9.2)
59 (± 13)
9
6
8
7
16
9
23 (45.1)
Marital status, n (%)
Married
2 (25)
8 (50)
3 (50)
3 (27.3)
7 (70)
Cohabitant
4 (50)
1 (6.3)
2 (33.3)
0 (0)
1 (10)
8 (15.7)
Single
0 (0)
4 (25)
1 (16.7)
0 (0)
2 (20)
7 (13.7)
Divorced
1 (12.5)
1 (6.3)
0 (0)
3 (27.3)
0
5 (9.8)
Widowed
1 (12.5)
2 (12.5)
0 (0)
5 (45.5)
0
8 (15.7)
Employed
4
11
3
1
4
23 (45.1)
Self-Employed
0
1
0
0
0
1 (2)
Student
0
1
1
0
0
2 (3.9)
Unemployed
1
1
0
0
0
2 (3.9)
Retired
3
2
2
10
6
23 (45.1)
Occupation, n (%)
Number of focus groups
Number of participants per focus group, mean (range)
Interview duration (min), mean
2
4
2
2
2
12
4 (3–5)
4 (3–6)
3 (3–3)
5.5 (5–6)
5 (5–5)
4.3 (3–6)
112
94
73
63
81
86
Because of the small sample size in the qualitative research and the focus on qualitative analysis in this study, no comparative statistics were calculated. We aimed to follow purposeful
sampling by selecting patients with different age and gender. However, pSS is predominantly affecting women (1–3, 39).
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TABLE 2 | Activities and participation.
Concepts named by the people with pSS
ICF codes
A
Difficulty taking care of the own body and
hygiene, such as washing and brushing the
hair, using deodorant, applying the body lotion
all over the body
d510 Washing oneself
d520 Caring for body parts
X
Difficulty getting dressed properly, including
putting on pants, socks and shoes
d540 Dressing
X
Reduced fine motor skills
d440 Fine hand use
X
Difficulty doing activities with hands and
fingers, such as opening a bottle, opening a
jar, peeling vegetables, cutting with a knife,
using the mobile phone or television remote
control, needlework, manipulating jewelry,
handling children’s skiing boots or playing an
instrument
d440 Fine hand use
d445 Hand and arm use
d9203 Crafts
d550 Eating
d630 Preparing meals
d660 Assisting others
d9202 Arts and culture
d430 Lifting and carrying objects
X
Difficulty managing the household,
including complex household work, preparing
meals, cleaning the house or doing the
shopping
d640 Doing housework
d630 Preparing meals
d650 Caring for household objects
d6200 Shopping
d6402 Cleaning living area
Needing more time for the performance of
activities, the difficulty of organizing job,
daily life and therapy
d2301 Managing daily routine
d2303 Managing one’s own activity level
d299 General tasks and demands, other
specified—needing more time for the
performance of activities
Difficulty performing (paid)
work/education activities, such as speaking,
studying, writing, using the computer, working
in front of a computer screen and following a
discussion
G
I
R
S
X
X
X
X
X
X
X
X
X
X
X
X
X
X
X
X
X
X
d830 Higher education
d850 Remunerative employment
d8451 Maintaining a job
d330 Speaking
d170 Writing
d3601 Using writing machines
d355 Discussion
X
X
X
X
X
Changed or lost employment due to
disease, including not able to work full
time
d8509 Remunerative employment,
unspecified–changed or lost employment due
to disease
d8501 Part-time employment
X
X
X
X
X
Mobility problems and difficulty engaging
in activities for leisure and recreation, such
as walking (the dog), hiking, driving a car, riding
the motorbike, doing sports, dancing, traveling,
gardening, going to the theater, watching TV or
reading
d455 Moving around
d450 Walking
d6506 Taking care of animals, other
specified—walking (the dog)
d475 Driving
d920 Recreation and leisure
d9201 Sports
d6505 Taking care of plants, indoors
and outdoors
d9202 Arts and culture
d110 Watching
d166 Reading
X
X
X
X
X
Difficulty in keeping and changing the
body position, such as getting up in the
morning, climbing the stairs, sitting on the floor
for a long period of time, getting in and out of
the car
d410 Changing basic body position
d415 Maintaining a body position
d4551 Climbing
X
X
X
X
X
Decreased opportunity to join social
activities, such as going to a party, inviting
friends at home, joining invitations for dinner
d9205 Socializing
d9208 Recreation and leisure, other
specified—going to a party d799 Interpersonal
interactions and relationships,
unspecified—joining invitations for dinner
X
X
X
X
(Continued)
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TABLE 2 | Continued
Concepts named by the people with pSS
ICF codes
A
G
I
R
S
Caring for others is demanding, including
caring for children, partners, grandchildren or
pets
d660 Assisting others
d6506 Taking care of animals
d660 Assisting others, unspecified—caring for
others is demanding
X
X
X
Impaired sex life
d7702 sexual relationships
X
Occupational loss, such as “stopped
hunting,” “stopped playing in the theater,” “not
lying in the sun” or “skipped political work”
nd
d910 Community life
d9204 Hobbies, unspecified—lying in the sun
d9204 Hobbies, unspecified—stopped hunting
X
X
Some activities can make symptoms
worse, such as reading, watching TV, carrying
heavy bags, going to work or doing intensive
sport
d Activities and Participation, other
specified—some activities make
symptoms worse
d110 Watching
d166 Reading
d850 Remunerative employment
d430 Lifting and carrying objects
d9201 Sports, unspecified—intensive sports
X
X
X
X
Negative impact on relationships, such as
end of partnership/relationship or arguments
with family members and friends
d720 Complex interpersonal interactions
d770 Intimate relationships
d750 Informal social relationships
d760 Family relationships
X
X
X
X
X
Balanced diet influences well-being, such as
eating no gluten, sugar, tomatoes and drinking
no milk
d5701 Managing diet and fitness
e1100 Food, unspecified—no gluten, sugar,
tomatoes and milk
X
X
Regular meaningful exercises in order to
feel well and strengthen the body, such as
yoga, singing, going for a walk
d Activities and Participation, other
specified—regular meaningful exercises in
order to feel well and strengthen the body
d570 Looking after one’s health
d9201 Sports, unspecified—Yoga
d332 Singing
d450 Walking
X
X
X
X
X
Leisure activities experienced as
meaningful, such as reading, traveling,
listening to music, surfing in the internet, doing
crafts
d Activities and Participation, other
specified—leisure activities experiences
as meaningful
d920 Recreation and leisure,
unspecified—leisure activities experiences
as meaningful
d9204 Hobbies
d920 Recreation and leisure,
unspecified—surfing in the internet
d115 Listening
d9230 Crafts
X
X
X
X
X
Everyday and self-care activities
experienced as meaningful, such as baking,
cooking, doing the household and drinking
morning coffee
d5 Self-care, other specified—everyday and
self-care activities experienced as meaningful
d6 Domestic life, other specified—everyday
and self-care activities experienced
as meaningful
d630 Preparing meals
d630 Preparing meals, unspecified—baking
d640 Doing housework
d560 Drinking, unspecified—drinking
morning coffee
X
X
X
X
X
(Paid) work and lifelong learning
experienced as meaningful
d8508 Remunerative employment, other
specified—(paid) work experiences
as meaningful
d838 Education other specified—lifelong
learning experiences as meaningful
X
X
X
X
X
X
X
X
(Continued)
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TABLE 2 | Continued
Concepts named by the people with pSS
ICF codes
A
G
I
R
S
Co-existence and activities with others are
experienced as meaningful, such as
engaging in gatherings with others, spending
time with partner and friends, being a member
of an association or joining a society
d9 Community, social and civic life—other
specified, Co-existence and activities with
others are experienced as meaningful
d9205 Socializing
d9100 Informal associations
d9101 Formal associations
X
X
X
X
X
Importance of rest, sleep and making
breaks to manage daily life
d2303 Managing one’s own activity level
X
X
X
X
X
Self-management strategies to reduce
symptoms
d570 Looking after one’s health
X
X
X
Looking for information and getting
knowledge about the disease and drugs,
including reading books, obtaining information
d138 Acquiring information
X
X
X
X
This table shows the commonalities and insights in the lifeworld providing concepts meaningful to people with pSS as well as the corresponding ICF codes. ICF, International Classification
of functioning, disability and health; nd, not defineable.
Countries in which we identified the concept were marked with X. Countries: A, Austria; G, Germany; I, Italy; R, Romania; S, Sweden.
Commonalities and Insights in the
Lifeworld of People With pSS
local researcher (CB, MM, JU, and RD) before the full proper
analysis was conducted. The whole process was supervised by
a researcher with extensive experience in the field of qualitative
research (TAS). Both investigators (JU and AL) who conducted
the ICF-linking process were trained in linking concepts to
the ICF.
Out of 82 concepts, 55 (67%) were mentioned by the patients
with pSS in at least four of five countries and 36 concepts (44%)
emerged in all the five countries. Tables 2–6 gives an overview of
the 82 concepts meaningful to the patients with pSS with their
specific ICF codes.
Participants from all the countries reported several limitations
in daily life, including self-care, productivity, and leisure. The
reasons for these limitations are characterized by a mismatch
between the capabilities of the person, the demands of the
physical, social, and cultural environment, and the requirements
to conduct the activity. A participant in Austria explained the
impact of her decreased fine motor skills on the performance
of activities as follows: “. . . in the morning, opening milk bottles
or juice, that was impossible (. . . ), but for example I cannot do
anything with buttons, I cannot make the buttons, jewelry. I
cannot do the little things. (. . . ) you have to look what you wear, I
want to put on a nice blouse, but I cannot do this if he (husband)
is not at home because of the buttons.” Another example is given
by a participant in Italy who described her experience with her
decreased ability to remember things: “I am no longer receptive,
I was a great student in high school, but when I started at the
university (. . . ), I was not able to remember things. I do not
know what was happening, you know, from 1 day to the other,
I just did not have the ability to concentrate anymore. When I
attended lectures for 4 h in the morning, I did not know what
was going on.”
How the environment can lead to participation restrictions
was mentioned in all countries. One participant in Sweden clearly
described: “Everybody else can go to town to go shopping, I
cannot go inside (in the store) because of those big fans at
the entrance, it does not work, my eyes do not manage it (the
flowing air) at the entrance. So I am left outside waiting in
the car.”
The importance of social support and understanding from
other people was highlighted by the participants from all the five
RESULTS
Participants and Focus Groups
Demographic information from 51 patients with pSS who
participated in 12 focus groups is provided in Table 1.
Concepts Identified in the Focus Groups
and Their ICF Linkage
The local investigators (JU, MM/CB, and RD) identified 184
subconcepts in Austria, 156 in Germany, 151 in Italy, 98 in
Romania, and 103 in Sweden. After the one-day-meeting of the
local investigators, the analysis of all focus groups resulted in
82 concepts meaningful to the patients with pSS. Out of these
82 concepts, the largest number of concepts (n = 25, 30%)
was linked to the ICF component activities and participation.
The second largest number of concepts (n = 22, 27%) was
considered to be not definable or not covered by the ICF. These
concepts contained general facets of pSS such as the difficulty
obtaining a diagnosis, the uncertainty of the origin of the disease
or that being ill is patience and energy demanding. Fifteen (18%)
concepts were linked to the ICF component body functions and
structures, another 13 (16%) concepts expressed environmental
factors. The remaining seven (9%) concepts revealed insights
in the personal thoughts, coping styles and behavior patterns
of patients with pSS and were therefore considered as personal
factors. The ICF components of the concepts are shown in
Tables 2–6 (column 2).
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TABLE 3 | Not covered, not defineable.
Concepts named by the people with pSS
ICF codes
A
G
I
R
S
Uncertainty about the origin of the disease, the
symptoms and discomforts
nd-dis
X
X
X
X
X
Being ill is patience- and energy demanding, including
insecurity due to having a chronic disease
nd-dis
X
X
X
X
Factors that intensify emotional problems, such as being
alone, extreme fatigue, unpredictability of the disease and
other environmental factors
nd- mh
X
X
X
X
Side effects of drugs or intolerance toward drugs,
including loss of hair, hearing problems, nausea, diarrhea,
fatigue, feeling dizzy
nd
b850 Functions of hair b240 Sensations
associated with hearing and vestibular function
b5350 Sensation of nausea
b525 Defecation functions
b4552 Fatiguability
b2401 Dizziness
X
X
X
X
X
Factors that increase the dosage of medication, such as
“changing weather,” “certain activities,” and “severity of
disease”
nd
X
Financial burden of the disease, including a missing
financial support from insurance system
nc
e570 Social security services, systems
and policies
e5802 Health policies
X
X
X
X
X
Time (and economy) limit for recreational activities
nc d230 Carrying out daily routine, other
specified—time limit e1650 financial assets, other
specified—economy limit
X
X
X
X
Co-morbidities and susceptible to other disease like
pneumonia, fungal infection or skin problems
nc-hc
X
X
X
X
X
Ups and downs of symptoms (swelling, stiffness, fatigue
and dryness), including changes over the day and the general
unpredictability of the disease
nc
X
X
X
X
X
Lack of user-friendly information with the risk for
misjudging the available information
nc
X
X
X
Insufficient time with health personnel at the doctoral
visits
nc
X
X
X
Frequent doctoral visits cause stress and remind on
being sick
nc
X
X
Long travel distances and waiting times for a doctoral
appointment inhibit regular doctoral visits or joining
support groups
nc
X
X
X
Invisibility of disease is either a relief or a curse
nc
X
X
Explaining the disease is important, but challenging
nc
X
Influenced roles within relationship, partnership and
family life
nc
Difficulty obtaining a diagnosis, including a great variability
of the way until diagnosis
nc
X
X
X
Nothing to worry, symptoms are in relation to age
nc
X
X
X
Presenteeism due to fear of losing the job or sense of duty
nc
Knowing other people with disease causes a
comfortable feeling
nc
X
X
X
Adaption of activities to own abilities, such as changing
the way of performing the activities or using other items
nc
X
X
X
X
Having knowledge about the own abilities, the disease
and its consequences for life cause a comfortable
feeling and is necessary for adaptions in daily life
nc
X
X
X
X
X
X
X
X
X
X
X
X
X
X
X
This table shows the commonalities and insights in the lifeworld, providing concepts meaningful to people with pSS as well as the corresponding ICF codes. ICF, International
Classification of functioning, disability and health; nc, not covered, nc-hc, not covered—health condition; nd, not defineable; nd–dis, not defineable—disability in general; nd–mh,
not defineable-mental health.
Countries in which we identified the concept were marked with X. Countries: A, Austria; G, Germany; I, Italy; R, Romania; S, Sweden.
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TABLE 4 | Body functions and structures.
Concepts named by the people with pSS
ICF codes
A
G
I
R
S
Disturbed sleep, either due to disease-related
symptoms or due to side effects of coping
strategies against dryness (e.g. frequent toilet
visits as consequence of drinking a lot)
b134 Sleep functions
X
X
X
X
X
Dryness- or disease related impact on
body parts, such as
red/inflamed/sensitive/watery eyes, mouth,
lymph nodes, blocked nose, nose bleeding,
sicca, affected joints, skin/dental/vaginal
problems
s220 Structure of eyeball
s230 Structures around eye
b210 Seeing functions
b215 Functions of structures adjoining the eye
b220 Sensations associated with the eye and
adjoining structures
s320 Structure of mouth
s510 Structure of salivary glands
s3200 Teeth
b5104 Salivation
b2153 Functions of lachrymal glands
s4201 Lymphatic nodes
b4353 Functions of lymph nodes
s310 Structure of nose
b255 Smell function
b898 functions of the skin and
related structures
b710 Mobility in joints
b28016 Pain in joints
s6303 Structure of vagina and
external genitalia
b6709 Sensations associated with genital and
reproductive functions,
unspecified—vaginal problems
X
X
X
X
X
Dryness-or disease related impact on
functions, such as an impaired ability to cry,
ability to swallow, ability to dose salivary flow,
ability to speak and sing, hearing and seeing
problems
b340 Alternative vocalization functions
b2153 functions of lachrymal glands
b5105 Swallowing
b5104 Salivation
b310 Voice functions
d330 Speaking
b230 Hearing functions
b210 Seeing functions
X
X
X
X
X
Experiences of dryness and pain, such
“having vinegar in the yes,” “crackling voice,”
“pelvis is like a wooden frame” or “my body
bursts”
b280 Sensation of pain
b220 Sensations associated with the eye and
adjoining structures
b3108 Voice functions, other
specified—crackling voice
b7808 Sensations related to muscles and
movement functions, unspecified—pelvis is
like a wooden frame
X
X
X
X
X
Pain and aches in the whole body and/or
specific body parts
b280 Sensation of pain
X
X
X
X
X
Fatigue and feeling exhausted and their
quantity and quality, such as
“extreme/enormous fatigue”
b4552 Fatiguability
X
X
X
X
X
Headache and heavy body due to fatigue
b28010 Pain in head and neck
b780 Sensations related to muscles and
movement functions b4552 Fatiguability
Lack of energy and endurance
b130 Energy and drive functions
b4550 General physical endurance
Experiences with other sensations like
cramps or paraesthesia, such as “like 1,000
ants running over the body”
b780 Sensations related to muscles and
movement functions
b735 Muscle tone functions
b2702 Sensitivity to pressure
X
X
X
X
X
X
X
X
X
X
(Continued)
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TABLE 4 | Continued
Concepts named by the people with pSS
ICF codes
A
G
I
R
S
Swelling and stiffness affecting the whole
body or specific body parts
b780 Sensations related to muscles and
movement functions
b7800 Sensation of muscle stiffness
b545 Water, mineral and electrolyte
balance functions
X
X
X
X
X
Decreased memory and attention
functions, such as forgetfulness, troubles with
finding the words and poor concentration
b140 Attention functions
b144 Memory functions
X
X
X
X
X
Emotional problems like sadness,
depression, worries, anger, anxiety,
embarrassment, guilty feelings guilty feelings,
fear and/or having suicidality thoughts and
self-injury behaviors
b152 Emotional functions
b1263 Psychic stability
X
X
X
X
X
Reduced self-esteem, feeling odd and
bored
b1800 Experience of self b1801 Body image
X
X
Hands become white and blue when it is
cold (indoor and outdoor)
b415 Blood vessel functions
Thoughts about getting pregnant or
thoughts during pregnancy
b6601 Functions related to pregnancy
X
X
X
X
X
This table shows the commonalities and insights in the lifeworld, providing concepts meaningful to people with pSS as well as the corresponding ICF codes. ICF, International Classification
of functioning, disability and health. Countries in which we identified the concept were marked with X. Countries: A, Austria; G, Germany; I, Italy; R, Romania; S, Sweden.
DISCUSSION
countries. In addition, the participants mentioned comfortable
feeling that is created by knowing other people with the same
disease. A participant in Austria explained it in the following
way: “I think you meet so many friends and gossip with
them about something. And you can’t deal with your own
problems with others, because they have no idea. So you can
meet people who are concerned, and if you just have a coffee
and chat. . . ”.
However, it was not always possible to join support
groups due to long travel distances. Some patients with pSS
described the negative impact of long travel distances that
inhibit them from visiting a healthcare provider or joining
the support groups. Similar, but different is the concept about
the negative impact of having no access to the internet or
living on the countryside for the development of knowledge
about disease.
Patients with pSS explained that they not always felt
taken seriously and understood by the health personnel. One
participant in Austria described it as follows: “. . . when you go to
the dentist, he doesn’t care (. . . ), your whole mouth hurts anyway,
because you don’t have any saliva, and then he gives you the
cotton wool, which is not ideal if you don’t have any saliva. If
he takes it out, he’ll rip half of your skin with it. And then you
say to him already five times ‘Listen’. And if he is not aware
of Sjögren and I say, ’Listen, I have a dry mouth, please, watch
out!’, but he doesn’t care.” At the same time the participants from
four countries mentioned that the time with health personnel
at routine visits is not sufficient. Furthermore, they specified
the absence of user-friendly information and pointed the risk
of misjudging. However, the importance of having knowledge
about the own abilities, the disease and its consequences for life
was found to be meaningful by the patients with pSS of the
four countries.
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To our knowledge, this is the first qualitative study on the
European level that examined the perspectives of patients with
pSS. We gained insights into the experiences of the patients
with pSS from the Northern (Sweden), Central (Austria and
Germany), and Southern/Eastern Europe (Italy, Romania). The
results of our study highlight the importance of multinational
data collection and go beyond previous small samples sized
national studies, showing that the patients with pSS from
different countries seem to experience similar challenges
regarding their functioning and health. Concepts that expressed
body functions and structures as well as concepts that were
linked to activities and participation may be universal across
the different European countries and might be used to guide
the development and the cross-cultural adaption of the PROMs
in pSS.
Our study led to the identification of 82 concepts; however,
the largest number of concepts (n = 25, 30%) expressed aspects
of an everyday living. This is in contrast to a study that
aimed at exploring the perspectives of the patients with pSS
on a national level (33), in which the concepts most often
reported expressed the physical dimension of the disease. These
differences may be explained by the multidimensional impact
of pSS and the dynamic interaction between pSS, the affected
body structures and functions and also the limitations in activities
and restrictions in participation in relation to the personal and
environmental factors.
Among the concepts found, the majority (67%) were
mentioned by the people with pSS in at least four of five countries,
and nearly half of the concepts (44%) were common in all
the five countries. A similar observation was made in a recent
study in which people with PsA from two different countries
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TABLE 5 | Environmental factors.
Concepts named by the people with pSS
ICF codes
A
G
I
R
S
Hypersensitivity to environmental factors,
including spicy and sour food, alcohol,
sparking beverages, coffee, bad air, sun (light),
and water in the eyes, rooms with low humidity
or air conditioning, extreme weather, make up,
stress, stressful surroundings and noise
nc
e1100 Food, unspecified—spicy and sour food e1100
Food, unspecified—alcohol
e1100 Food, unspecified—sparkling beverages
e1100 Food, unspecified—coffee
e260 Air quality
e225 Climate
b21020 Light sensitivity
e240 Light
e1150 General products and technology for personal
use in daily living, unspecified—make up
e250 Sound
X
X
X
X
X
Efficacy of drugs, including good, limited or
no effect
e1101 drugs, other specified—efficacy of drugs
X
X
X
Not taken seriously and not understood by
health personnel, including a lack of
counseling and understanding about what is
important to patients
e450 Individual attitudes of health professionals
e355 Health professionals
e455 Individual attitudes of other professionals
X
X
X
No access to wlan or living on the
countryside is negative for developing
knowledge about the disease
e5608 Media services, systems and policies, other
specified—No access to wlan or living on the
countryside is negative for developing knowledge about
the disease
X
Attitudes of employer, including the
employers (missing) duty of care toward
employees
e430 Individual attitudes of people in positions of
authority
X
Being forced to paid work because of the
social insurance system or the physicians
interpretation of it
e5708 Social security services, systems and policies,
other specified—Being forced to paid work because of
the social insurance system
Adaptions in or of the environment,
including an ergonomic workplace, having a
high bed or wearing certain kind of shoes
e1351 Assistive products and technology
for employment
e1401 Assistive products and technology for culture,
recreation and sport
e1151 Assistive products and technology for personal
use in daily living d175 Solving problems d163 Thinking
X
(Missing) Support and understanding
other people, including family, partner,
children, relatives, colleagues and lecturers
e310 immediate family
e410 Individual attitudes of immediate family members
e315 Extended family
e415 Individual attitudes of extended family members
e320 Friends
e420 Individual attitudes of friends
e325 Acquaintances, peers, colleagues, neighbors and
community members
e425 Individual attitudes of acquaintances, peers,
colleagues, neighbors and community members
X
X
X
X
X
Family, children, grandchildren and friends
enrich life and increase well-being
e399 Support and relationships, unspecified—Family,
children, grandchildren and friends enrich life and
increase well-being e310 immediate family
X
X
X
X
X
Experiences with non-medical treatment,
alternative medicine and products, such as
physical therapy, ayurveda, salt water bath,
sauna, thermal springs, massage, pedicure
and taking homeopathic drugs
e5, other specified—Experiences with non-medical
treatment, alternative medicine and products, such as
physical therapy, ayurveda, salt water bath, sauna,
thermal springs, massage, pedicure and taking
homeopathic drugs
X
X
X
X
Animals increase well-being
e2201 Animals e2208 Fauna and flora, other
specified—Animals increase well-being
X
X
Nature as an important factor for health,
including being active or passive in the
nature
e2200 Plants
e2208 Fauna and flora, other specified—Nature as an
important factor for health
X
X
X
X
X
X
X
X
X
X
X
X
X
(Continued)
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TABLE 5 | Continued
Concepts named by the people with pSS
ICF codes
A
G
I
Looking for assistive devices and
products that help
d5702 Maintaining one’s health, unspecified—looking
for assistive devices and products that help
e1151 Assistive products and technology for personal
use in daily living
e1401 Assistive products and technology for culture,
recreation and sport e1351 Assistive products and
technology for employment
X
X
X
R
S
X
This table shows the commonalities and insights in the lifeworld, providing concepts meaningful to people with pSS as well as the corresponding ICF codes. ICF, International Classification
of functioning, disability and health; nc, not covered.
Countries in which we identified the concept were marked with X. Countries: A, Austria; G, Germany; I, Italy; R, Romania; S, Sweden.
TABLE 6 | Personal factors.
Concepts named by the people with pSS
ICF codes
A
G
I
R
S
Own attitudes toward drugs and drug
intake, either “accepting them”, “feeling
dependent”, “some drugs are more dangerous
than others” or “having the desire to stop
taking them”
pf
X
X
X
X
Difficulty to accept the disease and
deterioration, including a negative description
pf
d130 Copying, unspecified—difficulty to
accept the disease and deterioration
X
X
X
(No) Openess with which people deal with
their disease and their limitations, including
“telling others about the disease” or “not telling
others and ”concealing the disease”
pf
X
X
X
X
Being selfish and taking time for myself
pf
X
X
X
X
X
Prioritizing and performing activities that
are meaningful and despite inner
resistance
pf
X
X
X
X
X
Learned to live with the disease and try to
live a normal life, including accepting the
disease and being positive
pf
d130 Copying, unspecified—learned to
live with the disease and try to live a
normal life
X
X
X
X
X
Personal beliefs and spirituality
pf
d9301 Spirituality
X
X
This table shows the cross-cultural similarities and differences of concepts meaningful to people with pSS as well as the corresponding ICF codes. ICF, International Classification of
functioning, disability and health; pf, personal factor.
Countries in which we identified the concept were marked with X. Countries: A, Austria; G, Germany; I, Italy; R, Romania; S, Sweden.
had two thirds of categories in common (14). In our study,
two thirds of concepts assigned to body functions and structures
(67%) and nearly half of the concepts related to the activities
and participation (52%) were mentioned in all the five countries,
indicating the importance of those two domains.
Some findings of our study are in line compared with other
qualitative studies in pSS. We already know that the patients with
pSS are confronted with a long way until diagnosis and thoughts
about the origin of the disease and also the consequences of the
disease on the physical, emotional, and social level (7, 33, 35, 36,
46, 47). We also know from other studies in pSS (33) and other
rheumatic conditions that the patients sometimes feel that they
are not taken seriously or that their complaints are dismissed by
the health professionals (33, 46). The participants of our study
specifically felt that the time the health personnel spent with
them was insufficient. This is a notable result and should be seen
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in the context of ongoing debates about the future workforce
requirements in rheumatology (48, 49). Current length of visits
per patient and the time spent on the clinical care seem to be too
short from point of view of the patients. Given that we expect
an increasing lag between workforce supply and demand in the
rheumatology in future, new concepts of care of pSS and other
rheumatic patients might be needed.
Furthermore, attitudes toward drugs varied substantially
between the participants of this study, either accepting them or
feeling independent and having the desire to stop taking them.
We do know from other studies that the attitudes toward taking
drugs influence the adherence of the patients (50). Therefore,
the health professionals should pay more attention to this
point in order to ensure patient-centered care for the patients
with pSS, also addressing non-pharmacological aspects and selfmanagement strategies.
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ETHICS STATEMENT
Our study has several limitations. First, we included a few
European countries only. Considering the influence of social,
cultural, and physical contexts into account, results might have
been somewhat different if people from the other continents
had participated in the study. On the other hand, we observed
only few variations between the five countries (belonging to
the Northern, Central, and Southern/Eastern Europe) studied,
supporting the generalizability of our data.
Second, some concepts could have been influenced by the
age, comorbidities, and other factors. Stratification of the focus
groups according to these variables could have helped to measure
the influence of these factors on the importance of the individual
concepts. On the other hand, such an approach bore the risk
that concepts emerged that are relevant for a small subgroup
of the patients only. Our goal was to collect data representing
the opinion and concerns of patients with pSS of everyday
clinical practice and decided therefore against selecting patients
according to the prespecified characteristics.
Third, the individual sample size in each country except
Germany could have been bigger, however, saturation within
the countries was reached that strengthens our results. We
want to emphasize that the transferability of the identified lived
experiences and perspectives is still limited, but a first attempt to
investigate pSS with a cross-cultural understanding in order to
inform practice and policy.
A specific strength of our study is that the health professionals
with different occupations (occupational therapists, physicians,
and physiotherapists) were involved in setting up the interview
questions, analyzing and interpreting the data.
In conclusion, concepts meaningful to the patients with pSS
identified in the five European countries might be used to
guide the development and the adaption of PROMs. Concepts
identified in this study enhance the clinical routine of the health
professionals in order to provide support and treatment options
based on the aspects relevant to the patients with pSS. However,
the results of our study have to be considered preliminary and
need to be confirmed by the future research.
The studies involving human participants were reviewed
and approved by the Ethic Committees of each
center: [EK 1653/2015, (Vienna, 8 September 2015),
EudraCT/EOMpss/Sjögren Syndrom (Vienna, 24 February
2016), EA1/014/16 (Berlin, 28 April 2016), 2961-2015 (Hanover,
1 December 2015), 93-2015 (Italy, 16 September 2015),
27/16.12.2015 (Romania, 16 December 2015), and 2016/3531Ö (Sweden, 15 March 2016)]. The patients/participants
provided their written informed consent to participate in
this study.
AUTHOR CONTRIBUTIONS
TS, CD, and JU were involved in the study conception. BR,
CB, FB, JU, MD, MM, PP, RD, TS, and TW were involved in
the recruitment of the patients. CA, JU, MM, and RD collected
the data. AL, CA, CB, CD, JU, MM, RD, and TS contributed
substantially to analysis and interpretation of the data. CB, CD,
JU, MM, RD, and TS drafted the manuscript. All authors read
and approved the final manuscript.
FUNDING
This work was supported by a grant of the Austrian Association
of Rheumatology and the Region Norrbotten. None of them were
involved in the data collection, nor in the analysis or preparation
of the manuscript.
ACKNOWLEDGMENTS
The authors are indebted to the patients with pSS who kindly
participated in this study and shared their perspectives, beliefs,
and needs. We thank the Austrian Association of Rheumatology
and the Region Norrbotten for partly funding this study. Parts of
this work were presented at the conferences of EULAR and the
Austrian Association of Rheumatology (51, 52).
DATA AVAILABILITY STATEMENT
SUPPLEMENTARY MATERIAL
The datasets presented in this article are not available because
participants of this study did not provide consent for the sharing
of the transcripts of the focus groups. For further information
please contact Julia Unger, julia.unger@fh-joanneum.at.
The Supplementary Material for this article can be found
online at: https://www.frontiersin.org/articles/10.3389/fmed.
2021.770422/full#supplementary-material
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Conflict of Interest: The authors declare that the research was conducted in the
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