Download as pdf or txt
Download as pdf or txt
You are on page 1of 9

Arthritis Care & Research

Vol. 65, No. 5, May 2013, pp 777–785


DOI 10.1002/acr.21931
© 2013, American College of Rheumatology
ORIGINAL ARTICLE

Fibromyalgia Prevalence, Somatic Symptom


Reporting, and the Dimensionality of
Polysymptomatic Distress: Results
From a Survey of the General Population
FREDERICK WOLFE,1 ELMAR BRÄHLER,2 ANDREAS HINZ,2 AND WINFRIED HÄUSER3

Objective. To evaluate fibromyalgia in the general population with emphasis on prevalence, dimensionality, and somatic
symptom severity.
Methods. We studied 2,445 subjects randomly selected from the German general population in 2012 using the American
College of Rheumatology 2010 preliminary diagnostic criteria for fibromyalgia, as modified for survey research, and the
polysymptomatic distress scale (PSD). Anxiety, depression, and somatic symptom severity were assessed with the Patient
Health Questionnaire (PHQ) series, and measures of symptoms and quality of life were assessed with the European
Organization for Research and Treatment of Cancer questionnaire.
Results. The prevalence of fibromyalgia was 2.1% (95% confidence interval [95% CI] 1.6, 2.7), with 2.4% (95% CI 1.5, 3.2)
in women and 1.8% (95% CI 1.1, 2.6) in men, but the difference was not statistically significant. Prevalence rose with age.
Fibromyalgia subjects had markedly abnormal scores for all covariates. We found smooth, nondisordered relationships
between PSD and all predictors, providing additional evidence against the hypothesis that fibromyalgia is a discrete
disorder and in support of a dimensional or spectrum disorder. There was a strong correlation (r ⴝ 0.790) between the
PSD and the PHQ somatic symptom severity scale; 38.5% of persons with fibromyalgia satisfied the proposed Diagnostic
and Statistical Manual of Mental Disorders, Fifth Edition criteria for a physical symptom disorder.
Conclusion. The modified 2010 diagnostic criteria do not result in high levels of fibromyalgia. PSD and fibromyalgia are
strongly related to somatic symptom severity. There is evidence in support of fibromyalgia as a dimensional or continuum
disorder. This has important ramifications for neurobiologic and epidemiology research, and for clinical diagnosis,
treatment, and ascertainment of disability.

INTRODUCTION tender point count ascertainment required by the ACR


1990 criteria (3) were eliminated (4).
The development of the 2010 American College of Soon after the publication of the criteria, it was sug-
Rheumatology (ACR) fibromyalgia criteria (1) and their gested that the 2 components of the 2010 criteria, the 0 –19
modification for survey research (2) made it possible to widespread pain index (WPI) and the 0 –12 symptom se-
conduct population-based research relating to fibromyal- verity (SS) score, could be combined by addition into a
gia because the high costs and difficulties surrounding the 0 –31 index. Originally called the “fibromyalgianess scale”
(5), a term that was a little awkward and limiting, it has
subsequently been termed the “polysymptomatic distress”
1
Frederick Wolfe, MD: National Data Bank for Rheumatic scale (PSD), a term first suggested by Wessely and Hotopf
Diseases and University of Kansas School of Medicine, (6). Patients who satisfy the 2010 criteria, defined by either
Wichita; 2Elmar Brähler, PhD, Andreas Hinz, PhD: Univer-
1) WPI ⱖ7/19 pain sites and SS score ⱖ5/12 (Type A) or
sität Leipzig, Leipzig, Germany; 3Winfried Häuser, MD:
Technische Universität München, Munich, and Klinikum 2) WPI between 3– 6/19 and SS score ⱖ9/12 (Type B), will
Saarbrücken, Saarbrücken, Germany. always have a score on the PSD scale of at least 12 (7 ⫹ 5
Dr. Häuser has received consultant fees, speaking fees, or 3 ⫹ 9). Thus, fibromyalgia can be mapped out on a
and/or honoraria (less than $10,000 each) from Daiichi
Sankyo and Abbott Germany.
dimensional or continuum scale, allowing further explo-
Address correspondence to Frederick Wolfe, MD, 1035 ration of the fibromyalgia concept (7). Fibromyalgia differs
North Emporia, Suite 288, Wichita, KS 67214. E-mail: from the frequently studied chronic widespread pain con-
fwolfe@arthritis-research.org. cept (8,9) by its inclusion of nonpain symptoms, including
Submitted for publication October 31, 2012; accepted in
revised form December 11, 2012. severity measures of fatigue, unrefreshed sleep, cognitive
problems, and somatic symptom reporting. In addition,

777
778 Wolfe et al

formed about the investigation. Subjects were presented


Significance & Innovations with self-rating questionnaires. The survey included sev-
● Using a modification for surveys of the American eral questionnaires on somatic and psychological symp-
College of Rheumatology 2010 fibromyalgia crite- toms (health survey) as well as questionnaires on eating,
ria, the prevalence of fibromyalgia in the general political attitudes, and media use. The assistant waited
German population was 2.1%. until the participants answered all questionnaires, and
● There is evidence that fibromyalgia is a continuum offered help if persons did not understand the meaning of
disorder. questions.
Data collection took place between May and June 2012.
● Forty percent of fibromyalgia cases in the general A first attempt was made at 4,448 addresses, and 2,515
population satisfy proposed Diagnostic and Statis- persons (56.7%) participated fully. Reasons for nonpar-
tical Manual of Mental Disorders, Fifth Edition
ticipation included the following: 3 unsuccessful attempts
criteria for physical symptom disorder.
to contact the household or selected household member
(12.9%), the household or selected household member
declined to participate (13.7%), or the household mem-
ber was on a holiday break (1.1%). Furthermore, 0.5% of
the participants were excluded because they were not able
to follow the interview due to illness, as were 3.3% who
fibromyalgia is a medical diagnosis that is included in the refused to finish the interview.
international list of diseases of the World Health Organi- To study the adult population, we excluded subjects
zation, in which chronic widespread pain is not. Chronic who were ⬍18 years of age. Three subjects who had miss-
widespread pain may be related to fibromyalgia by an ing data for the fibromyalgia diagnosis variables were also
increase in somatic symptoms (10) and psychological dis- excluded. After these exclusions, a total of 2,445 subjects
tress (8). were included in the analysis.
In the study that follows, we examined several issues of
importance regarding fibromyalgia in the general popula- Ethics. All participants were informed about the study
tion using data from a 2012 German epidemiology survey. procedures and signed an informed consent form. The
In the first part of the report, we studied the prevalence of study was approved by the Institutional Ethics Review
fibromyalgia, examining the effect of age and sex on prev- Board of the University of Leipzig (Az 092-12-05032012).
alence to determine whether prevalence was much greater
in women, as was the case in the clinic when tender points
Questionnaires. We assessed marital status, educa-
were in use. We also considered whether the use of self-
tional status, current professional status, and family in-
report symptom criteria would lead to an inflation of di-
come by a sociodemographic questionnaire. We used a
agnosis rates, and we considered whether the use of
slightly modified social class index that is used in rehabil-
Type B criteria would disturb the uniformity and homo-
itation care and surveys in Germany (11).
geneity of the fibromyalgia diagnosis. The second major
issue of the study was to seek evidence about the “contin-
uum hypothesis,” whether fibromyalgia was best under- Fibromyalgia Survey Questionnaire (FSQ). The modi-
stood as part of a continuum of polysymptomatic distress fied ACR 2010 diagnostic criteria were developed in a
or should be understood as a categorical disorder. Finally, longitudinal study of patients of the National Data Bank
we evaluated somatic symptom reporting and intensity as for Rheumatic Diseases by substituting a count of 3 symp-
a central part of the fibromyalgia concept. toms for the physician’s (range 0 –3) evaluation of the
extent of somatic symptom intensity by a questionnaire
assessing the number of pain sites and somatic symptom
PATIENTS AND METHODS severity (2). The FSQ included the SS score with 3 major
symptoms (fatigue, trouble thinking or remembering,
Design and subjects. A representative sample of the waking up tired [unrefreshed]), which can be coded 0 –3
German general population was selected with the assis- (0 ⫽ no problem; 1 ⫽ slight or mild problems, generally
tance of a demographic consulting company (USUMA, mild or intermittent; 2 ⫽ moderate, considerable prob-
Berlin, Germany). The random selection was based on lems, often present and/or at a moderate level; 3⫽ severe,
multistage sampling. First, 320 sample point regions were continuous, life-disturbing problems), and 3 additional
randomly drawn from the last political election register, symptoms (pain or cramps in lower abdomen, depression,
covering rural and urban areas from all regions in Ger- headache), which can be coded as present (1) or absent (0)
many. The second stage was a random selection of house- (total subscore 0 –3). These 3 items are surrogates for the
holds using the random route procedure (based on a start- somatic symptom burden item of the ACR 2010 criteria (1).
ing address). The third stage was a random selection of The SS score ranges from 0 –12. The WPI includes 19 pain
household respondents with the Kish selection grid. The sites (both jaws, both shoulders, both upper arms, both
sample was aimed to be representative in terms of age, sex, lower arms, both hips, both upper legs, both lower legs,
and education for the general German population. The neck, chest, upper back, lower back, abdomen). It is also
inclusion criteria for the study were age ⱖ14 years and the possible to determine from the WPI pain sites if a patient
ability to read and understand the German language. satisfies the ACR 1990 widespread pain criterion (3). The
All subjects were visited by a study assistant and in- sum of the WPI and the SS score constitutes the fibromy-
Prevalence, Dimensionality, and Somatic Symptom Severity of FM 779

algianess scale or PSD, a measure of physical and psycho- Table 1. The prevalence of fibromyalgia and mean
logical symptom intensity (distress) that can be applied to values of related variables in the general population*
subjects regardless of disease (12). We used the validated
German versions of the FSQ (13). Category Cases N Value (95% CI)
Subjects satisfy the fibromyalgia survey criteria if they
Fibromyalgia prevalence
meet the following 3 conditions: 1) WPI ⱖ7/19 pain sites All 52 2,445 2.1 (1.6, 2.7)
and SS score ⱖ5/12 or WPI between 3– 6/19 and SS score Women 31 1,308 2.4 (1.5, 3.2)
ⱖ9/12; 2) symptoms have been present at a similar level Men 21 1,137 1.8 (1.1, 2.6)
for at least 3 months; and 3) the patient does not have All cases
another disorder that would otherwise sufficiently explain Ages ⬍40 years 6 741 0.8 (0.2, 1.5)
Ages 40–59.9 years 22 895 2.5 (1.4, 3.5)
the pain (2). The conditions 1 and 2 can be assessed by the Ages ⱖ60 years 24 809 3.0 (1.8, 4.1)
FSQ in epidemiologic studies. Women
Ages ⬍40 years 6 392 1.5 (0.3, 2.8)
Patient Health Questionnaire (PHQ). The PHQ Somatic Ages 40–59.9 years 13 491 2.6 (1.2, 4.1)
Symptom Short Form (PHQ-SSS) is the short form of the Ages ⱖ60 years 12 425 2.8 (1.2, 4.4)
Men
PHQ-15 (14) and comprises 8 items (stomach or problems
Ages ⬍40 years 0 349 0.0 (0.0, 0.0)
going to the toilet; pain in the back; pain in the arms, legs, Ages 40–59.9 years 9 404 2.2 (0.8, 3.7)
or joints; headaches; chest pain or getting out of breath; Ages ⱖ60 years 12 384 3.1 (1.4, 4.9)
dizziness; feeling tired or having low energy; trouble PDS
sleeping), with each symptom scored from 1 (“not both- All 2,445 3.0 (2.9, 3.1)
ered at all”) to 5 (“bothered very strongly”) within the last Women 1,308 3.2 (3.0, 3.4)
Men 1,137 2.8 (2.6, 3.0)
7 days (14). The PHQ-15 is best characterized as a measure
All cases
of somatic symptom severity (14). Ages ⬍40 years 741 1.8 (1.6, 2.0)
The PHQ-SSS (not yet validated) had been proposed for Ages 40–59.9 years 895 2.9 (2.7, 3.2)
the research definition of criterion A (somatic symptoms: Ages ⱖ60 years 809 4.2 (3.9, 4.4)
one or more somatic symptoms that are distressing and/or Women
result in significant disruption in daily life) of the physical Ages ⬍40 years 392 2.1 (1.8, 2.4)
Ages 40–59.9 years 491 3.1 (2.8, 3.5)
symptom disorder of the Diagnostic and Statistical Manual Ages ⱖ60 years 425 4.3 (3.9, 4.7)
of Mental Disorders, Fifth Edition (DSM-V). Criterion A is Men
met if a person reports being bothered strongly or very Ages ⬍40 years 349 1.5 (1.2, 1.7)
strongly by a symptom (15). We assumed that criterion B Ages 40–59.9 years 404 2.7 (2.4, 3.0)
(disproportionate and persistent thoughts about the seri- Ages ⱖ60 years 384 4.0 (3.6, 4.4)
WPI
ousness of one’s symptoms) was met if a person reported at
All 2,445 1.3 (1.2, 1.4)
least one severe, continuous, life-disturbing problem in Women 1,308 1.4 (1.3, 1.5)
the 2010 criteria SS score. Men 1,137 1.2 (1.1, 1.4)
The PHQ-4 (16) is an ultra-brief self-report question- All cases
naire that consists of a 2-item depression scale (PHQ-2) Ages ⬍40 years 741 0.5 (0.4, 0.6)
and a 2-item anxiety scale (Generalized Anxiety Disorder). Ages 40–59.9 years 895 1.2 (1.1, 1.4)
Ages ⱖ60 years 809 2.2 (2.0, 2.4)
A score of 3 or greater on the depression subscale repre-
Women
sents a reasonable cut point for identifying potential cases Ages ⬍40 years 392 0.6 (0.4, 0.7)
of major depression or other depressive disorders, and a Ages 40–59.9 years 491 1.3 (1.1, 1.5)
score of 3 or greater on the anxiety subscale represents a Ages ⱖ60 years 425 2.3 (2.0, 2.6)
reasonable cut point for generalized anxiety, panic, social Men
anxiety, and posttraumatic stress disorders in primary care Ages ⬍40 years 349 0.4 (0.3, 0.5)
Ages 40–59.9 years 404 1.1 (0.9, 1.3)
settings and in the general population (17). The PHQ-4 Ages ⱖ60 years 384 2.2 (1.9, 2.4)
total score can serve as a measure for psychological dis- SS score
tress (14). We used the validated German version of the All 2,445 1.7 (1.6, 1.7)
PHQ (17). Women 1,308 1.8 (1.7, 1.9)
Health-related quality of life was assessed by the vali- Men 1,137 1.5 (1.4, 1.6)
All cases
dated German version of the European Organization for
Ages ⬍40 years 741 1.3 (1.2, 1.5)
Research and Treatment of Cancer (EORTC) QLQ-C30, Ages 40–59.9 years 895 1.7 (1.6, 1.8)
version 3.0 (18). The EORTC QLQ-C30 includes 5 func- Ages ⱖ60 years 809 1.9 (1.8, 2.0)
tioning scales, which cover the dimensions physical func- Women
tioning (5 items), role functioning (2 items), emotional Ages ⬍40 years 392 1.5 (1.4, 1.7)
functioning (4 items), cognitive functioning (6 items), and Ages 40–59.9 years 491 1.8 (1.7, 2.0)
Ages ⱖ60 years 425 2.0 (1.8, 2.2)
social functioning (2 items). In addition, the questionnaire
Men
contains a scale for global health status (2 items), and Ages ⬍40 years 349 1.1 (0.9, 1.3)
symptom scales that are relevant to cancer patients (e.g., Ages 40–59.9 years 404 1.6 (1.4, 1.8)
pain, nausea, fatigue, dyspnea). For functional and global Ages ⱖ60 years 384 1.8 (1.6, 2.0)
quality of life scales, higher scores mean a better level of
* 95% CI ⫽ 95% confidence interval; PDS ⫽ polysymptomatic distress
functioning. For symptom-oriented scales, a higher score scale; WPI ⫽ widespread pain index; SS ⫽ symptom severity.
means more severe symptoms (19).
780 Wolfe et al

Table 2. Clinical and demographic variables for full population and fibromyalgia groups*

All All FM (ⴙ) FM group A FM group B P,


(n ⴝ 2,445) (n ⴝ 52) (n ⴝ 43) (n ⴝ 9) A vs. B

Age, years (range 18–91 years) 50.2 ⫾ 17.4 59.8 ⫾ 16.4 60.7 ⫾ 16.9 55.7 ⫾ 13.7 0.410
Sex, % male 46.5 39.2 38.1 44.4 0.723
Social class, % lower 8.6 22.0 26.2 0.0 0.087
Depression score (PHQ-2) (range 0–6) 0.7 ⫾ 1.0 3.2 ⫾ 1.4 3.0 ⫾ 1.2 4.0 ⫾ 2.1 0.967
Depression (PHQ ⱖ3), % 6.0 63.5 61.9 77.8 0.366
Anxiety disorder score (GAD-2) (range 0–6) 0.5 ⫾ 0.9 2.6 ⫾ 1.5 2.4 ⫾ 1.4 3.7 ⫾ 1.7 0.015
Anxiety disorder (GAD-2 ⱖ3), % 2.4 50.0 42.9 88.9 0.012
Psychological distress (PHQ-4) (range 0–12) 1.2 ⫾ 1.7 5.8 ⫾ 2.5 5.4 ⫾ 2.1 7.7 ⫾ 3.6 0.014
Somatic symptom (PHQ-SSS) (range 0–26) 3.3 ⫾ 4.0 15.9 ⫾ 5.1 15.3 ⫾ 5.0 18.6 ⫾ 5.1 0.082
EORTC physical function (range 100–0) 92.2 ⫾ 15.0 53.1 ⫾ 25.5 56.3 ⫾ 24.0 37.8 ⫾ 27.9 0.046
EORTC cognitive function (range 100–0) 93.6 ⫾ 14.5 57.0 ⫾ 24.8 59.3 ⫾ 51.8 46.2 ⫾ 27.4 0.154
EORTC quality of life (range 100–0) 75.0 ⫾ 20.4 36.2 ⫾ 19.2 37.2 ⫾ 19.2 31.5 ⫾ 19.4 0.420
EORTC pain (range 0–100) 16.7 ⫾ 24.2 70.9 ⫾ 26.6 69.0 ⫾ 25.6 79.6 ⫾ 30.9 0.284
EORTC sleep status (range 0–100) 12.4 ⫾ 23.3 52.3 ⫾ 35.4 49.2 ⫾ 37.0 66.7 ⫾ 23.6 0.182
EORTC fatigue (range 0–100) 15.5 ⫾ 21.7 71.7 ⫾ 26.3 68.3 ⫾ 26.2 87.7 ⫾ 21.8 0.044
ACR widespread pain index (range 0–19) 1.3 ⫾ 2.3 8.9 ⫾ 3.5 9.6 ⫾ 3.0 4.3 ⫾ 1.3 0.000
ACR symptom severity score (range 0–12) 1.7 ⫾ 1.9 7.4 ⫾ 2.0 6.8 ⫾ 1.6 10.2 ⫾ 1.0 0.000
Polysymptomatic distress (range 0–27) 3.0 ⫾ 3.3 16.4 ⫾ 3.5 16.8 ⫾ 3.6 14.6 ⫾ 1.8 0.077

* Values are the mean ⫾ SD unless otherwise indicated. FM ⫽ fibromyalgia; PHQ-2 ⫽ 2-item Patient Health Questionnaire; GAD-2 ⫽ 2-item
Generalized Anxiety Disorder; PHQ-SSS ⫽ PHQ Somatic Symptom Short Form; EORTC ⫽ European Organization for Research and Treatment of
Cancer; ACR ⫽ American College of Rheumatology.

Statistical methods. We used predicted values from sec- CI 0.17, 0.72) units greater in women than in men. Overall,
ond-degree fractional polynomial (FP) regression (20) to these data show that the prevalence of fibromyalgia and its
describe the PSD and covariate relationships, after first components increases with age, but that sex has a smaller
examining each covariate pair by lowess regression and effect that is not significant for fibromyalgia or WPI, but is
scatter plots to be sure the FP was an accurate and fair significant for SS score and PSD.
representation of the underlying data. Comparisons in
Table 1 used t-tests and chi-square tests, as appropriate. Effect of Type A and B definitions. Of patients satisfy-
Data were analyzed using Stata, version 12.1. ing the modified ACR 2010 criteria, 82.7% satisfied it
using the Type A definition and 17.3% with the Type B
RESULTS definition. As mandated by the Type A and Type B defi-
nitions that required WPI to be lower and SS scores to be
higher, SS scores were significantly higher for Type B
Prevalence of fibromyalgia and component variable
subjects (mean ⫾ SD 10.2 ⫾ 1.0 versus 6.8 ⫾ 1.6) (Table 2),
levels. The study population was representative of the
while WPI scores were greater in Type A subjects (mean ⫾
German population in 2011 in terms of age and sex (21)
SD 9.6 ⫾ 3.0 versus 4.3 ⫾ 1.3). PSD scores were not
(Supplementary Appendix A, available in the online ver-
significantly different (P ⫽ 0.077). In addition, anxiety,
sion of this article at http://onlinelibrary.wiley.com/doi/
general psychological distress, physical function, and fa-
10.1002/acr.21931/abstract). The overall prevalence of fi-
tigue were worse in the Type B group. Figure 1 shows the
bromyalgia was 2.1% (95% confidence interval [95% CI]
distribution of the scores for both groups, with Type B
1.6, 2.7) (Table 1). Fibromyalgia increased with age: 0.8%
(95% CI 0.2, 1.5), 2.5% (95% CI 1.4, 3.5), and 3.0% (95% scores generally spread over the full ranges of fibromyalgia
CI 1.8, 4.1) in those ⬍40, 40 –59.9, and ⱖ60 years of age, patient scores. The 1990 widespread pain criterion was
respectively (P ⫽ 0.004). Fibromyalgia was not statistically noted in 82.7% overall, and in 93.0% of Type A and 33.3%
more common in women than men (2.4% versus 1.8%; of Type B subjects.
P ⫽ 0.372 and 59.6% versus 40.4% among those with
fibromyalgia). We examined the 3 components of fibromy- Continuum hypothesis for fibromyalgia. By definition,
algia shown in Table 1. In regression analyses, the WPI subjects with fibromyalgia (2.1%) had to have a PSD score
increased by 0.45 (95% CI 0.40, 0.50) units per 10-year of at least 12, a level met by 3.4% of all subjects when
increase in age. Adjusted for age, the WPI for women was nonfibromyalgia subjects were included (Figure 2). How-
0.17 (95% CI ⫺0.01, 0.34) units greater than in men (P ⫽ ever, a cut point of 13 better distinguished fibromyalgia
0.062). The SS score increased by 0.14 (95% CI 0.10, 0.19) from nonfibromyalgia patients, for if a score of 12 (or 13)
units per 10-year increase in age. Adjusted for age, the SS were assumed to represent fibromyalgia for research pur-
score was 0.28 (95% CI 0.13, 0.43) units greater in women poses, the respective kappa statistics for agreement would
than in men. The summary PSD score increased by 0.59 be 0.758 (95% CI 0.678, 0.839) and 0.795 (95% CI 0.715,
(95% CI 0.52, 0.67) units per 10 years and was 0.45 (95% 0.875), respectively, indicating full agreement (22) with
Prevalence, Dimensionality, and Somatic Symptom Severity of FM 781

Figure 1. The distribution of fibromyalgia syndrome (FMS) covariate scores for subjects meeting Type A and Type B FMS criteria. PHQ ⫽
Patient Health Questionnaire; GAD ⫽ Generalized Anxiety Disorder.

the ACR 2010 fibromyalgia criteria designation. As cutoff


values of 12 or 13 suggested a dimensional disorder, we
examined the relationship between various covariates and
PSD to observe for linear association or graphic disconti-
nuities. Figures 2 and 3 demonstrate continuous, generally
linear, relationships throughout the relevant ranges that
were all significant at P ⬍ 0.001. In addition, the figures
show no evidence of inflection points in the area of PSD ⫽
12 or 13. The strength of the relationships between PSD
and covariates is shown in Table 3.

Somatic symptoms and fibromyalgia variables. The


strongest correlation between PSD and non-PSD depen-
dent variables was with the PHQ-SSS (r ⫽ 0.790) (Figure 2
and Table 3); the correlation between PSD and other co-
variates is shown in the table. For diagnosis of fibromyal-
gia, the area under the receiver operating curve was 0.973 Figure 2. The distribution of polysymptomatic distress scale
(95% CI 0.958, 0.988) for PHQ-SSS and 0.996 (95% CI (PSD) scores (yellow line) and the relationship between the Pa-
tient Health Questionnaire (PHQ) Somatic Symptom Short Form
0.995, 0.998) for PSD. The difference was significant at P ⫽ and PSD in the general population. Blue points are subjects neg-
0.002. Except for the WPI and SS scores, the association ative for fibromyalgia syndrome (FMS) and red points are subjects
between the PHQ-SSS was slightly stronger for all covari- who satisfy the FMS criteria. The vertical line at 12 is the point at
which cases of FMS first appear. Scores ⱖ12 are found in 3.4% of
ates in Table 3 compared with PSD. Overall, these data
subjects and FMS criteria are met by 2.1% of the population. A
show the strong similarities as well as the differences small amount of random noise is added to the FMS-negative cases
between PSD and PHQ-SSS. to aid visualization.
782 Wolfe et al

Figure 3. The relationship between polysymptomatic distress scale (PSD) and relevant covariates. The grey area represents 95%
confidence intervals. The P values for the fractional polynomial regression models were ⬍0.001 for all models. There is no evidence of
discontinuity or important nonlinearity over the relevant ranges. BMI ⫽ body mass index.

With respect to somatic symptoms, 53.8% of fibromyal-


gia subjects had ⱖ1 severe somatic symptoms and 32.7% Table 3. Correlations between polysymptomatic distress,
had ⱖ2 such symptoms on the PHQ-SSS: criterion A of the PHQ-SSS, and study covariates*
proposed DSM-V physical symptoms disorder. Twenty-
Variable PDS PHQ-SSS
one subjects (40.4%) noted at least one severe problem in
the 2010 criteria SS score, an approximation of criterion B. PDS 1.000 0.790
Twenty subjects (38.5%) noted at least one severe problem Widespread pain index 0.880 0.635
in the 2010 criteria SS score and ⱖ1 severe somatic symp- Symptom severity score 0.810 0.714
toms on the PHQ-SSS. GAD-2 anxiety score 0.531 0.550
PHQ-SSS 0.790 1.000
EORTC pain score 0.629 0.691
DISCUSSION EORTC quality of life ⫺0.593 ⫺0.633
EORTC physical function ⫺0.575 ⫺0.629
In 2008, a similar German population study used a differ- EORTC emotional function ⫺0.556 ⫺0.621
ent version of the WPI and an earlier non-ACR version of PHQ-2 depression score 0.555 0.574
fibromyalgia criteria (23) that resulted in an estimated EORTC physical role ⫺0.541 ⫺0.579
fibromyalgia prevalence of 3.8% (24,25). In this (2012) EORTC social function ⫺0.534 ⫺0.611
nationwide population study, we used the modified 2010 EORTC financial difficulties 0.459 0.536
ACR fibromyalgia criteria and found that the prevalence of Social class ⫺0.165 ⫺0.159
Body mass index 0.130 0.146
fibromyalgia was 2.1% (95% CI 1.6, 2.7). The small num-
ber of Type B cases (17.3%) with their associated increases * PHQ-SSS ⫽ Patient Health Questionnaire Somatic Symptom
in distress covariates indicates that Type B was a minor Short Form; PDS ⫽ polysymptomatic distress scale; GAD-2 ⫽
2-item Generalized Anxiety Disorder; EORTC ⫽ European Organi-
component of the fibromyalgia definition that tapped into zation for Research and Treatment of Cancer.
the SS scores appropriately, as the criteria designers had
Prevalence, Dimensionality, and Somatic Symptom Severity of FM 783

anticipated. The Type B criterion was added to capture Definitive evidence to support or refute this view will
those persons who had all of the symptoms of fibromyalgia come from primary care or community samples, not the
but not enough painful areas. Prior to the 1990 ACR crite- study of specialist populations.” Croft et al posed the
ria, the requirement for painful regions and tender points question: “More pain, more tender points: is fibromyalgia
was less strict (26). The 2010 criteria recognized this and just one end of a continuous spectrum?” (31). In a study of
the clinical need to slightly relax requirements imposed by clinical data, Wolfe concluded that “Tender points are
the 1990 criteria. linearly related to fibromyalgia variables and distress, and
The current study noted that the mean level of WPI and there is no discrete enhancement or perturbation of fibro-
the prevalence of fibromyalgia did not differ significantly myalgia or distress variables associated with very high
by sex, although there was a nonsignificant female pre- levels of tender points” (32). The current study provides
dominance (59.6% versus 40.4%) among those with fibro- the type of evidence for fibromyalgia as the continuum that
myalgia (P ⫽ 0.372); for women, fibromyalgia prevalence Wessely and Hotopf stated was necessary.
was 2.4%, compared with 1.8% in men. Other population Although fibromyalgia in clinical practice is treated as
studies have found that rates of widespread pain for the a categorical disorder, the confirmation of fibromyalgia
sexes were similar, and that there was only a modest as a spectrum disorder has important research, clinical,
increase of fibromyalgia in women compared with men and societal connotations: “A study that takes the extreme
(25,27). By contrast, in clinical populations women con- end of the spectrum, represented by selected samples of
stitute as many as 90% of patients. Increased rates in patients referred to rheumatology or pain services, and
clinical populations have been attributed to the use of the compares them with non-fatigued controls, will produce
tender point count and sex differences in health-seeking a . . . categorical solution but for spurious reasons” (6).
behavior. George Ehrlich noted, however, that “No one has Future research, whether clinical or neurobiologic, must
FM until it is diagnosed . . . it has to be named by a doctor account for the continuum nature of fibromyalgia to be
to exist” (28). This observation underscores the difference valid.
between the number of people who satisfy criteria for The ACR 2010 criteria definition of fibromyalgia was
fibromyalgia and the number who actually seek care for constructed based on specialist physicians’ (experts) diag-
symptoms that are cast by physicians or patients as fibro- nosis of fibromyalgia using the ACR 1990 criteria. How-
myalgia. ever, there was no gold standard for patients designated as
We made use of the PSD scale to investigate whether having fibromyalgia in the 1990 criteria study, as the pres-
fibromyalgia was a categorical or dimensional disorder. ence of a fixed high number of tender points was not
The PSD scale arose after the publication of the 2010 required by most previous definitions (33–35), including
criteria, and was formed by adding together the 2 compo- the definition by Yunus et al in their 1981 criteria (26).
nents of the 2010 fibromyalgia diagnosis, the WPI and the Although one might conclude that the 1990 definition
SS score. Persons with fibromyalgia always have mini- of fibromyalgia was reasonable, it was also arbitrary.
mum levels of PSD of at least 12 regardless of whether they With that in mind, it is problematic to sustain the idea that
satisfy the Type A or Type B criterion. This allows fibro- a specific cut point accurately and validly separates dis-
myalgia to be marked as a point on the PSD continuum. ease from nondisease or illness from nonillness, particu-
The PSD scale is linearly correlated with all measures of larly in view of the continuous nature of polysymptomatic
physical and psychological distress over relevant ranges distress.
(Figures 2 and 3). There is considerable uncertainty about the nature of
In deciding whether a disorder should be considered a fibromyalgia. Virtually every type of mental illness has
categorical or a dimensional illness, Kessler indicated that been found to be more common in fibromyalgia (36). Neu-
“The critical test should be whether the predictors are robiologic abnormalities and central sensitization have
consistently related to differences in symptom severity also been suggested as causes of fibromyalgia (37). At the
across the full relevant range of the dimensional distribu- other end of the spectrum, many disbelieve in the idea of
tion. If they are, then analysis of the dimensional version fibromyalgia, citing flaws in the conceptualization of the
of the symptom scale makes most sense . . .” (29). We illness as well as the continuous nature of polysymptom-
found the following strongly supportive of dimensional atic distress (28,38 – 45). One contribution to the nature of
illness. Figure 2 shows that fibromyalgia onset (PSD ⫽ 12) fibromyalgia based on this study, in addition to the dem-
lies at the end of the PSD continuum; 96.6% of all persons onstration of the continuous nature of polysymptomatic
in the general population have values less than 12. PSD is distress, is the exploration of the relationship of somatic
also correlated with dimensional measures of distress in symptom reporting and fibromyalgia. We found that PSD
the current study and in other samples (30). Figure 3, was strongly correlated with the PHQ-SSS (r ⫽ 0.790). In
which examines Kessler’s dictum using fractional polyno- addition, 20 subjects (38.5%) noted at least one severe
mial regression that is sensitive to nonlinearity, demon- problem in the 2010 criteria SS score and ⱖ1 severe so-
strates a smooth relationship between PSD and all relevant matic symptoms on the PHQ-SSS. From these data, it
covariates. would appear that approximately 40% of persons with
In a comprehensive review of fibromyalgia, Wessely and fibromyalgia satisfy the proposed DSM-V criteria for phys-
Hotopf wrote that they were “unaware of any study” that ical symptom disorder. The idea that fibromyalgia is pri-
supported the view that fibromyalgia was a categorical marily a somatic symptom disorder is well accepted in
disorder (6), and wrote that “fatigue and myalgia syn- the pain and psychological literature (46 – 48), but not in
dromes are arbitrarily created syndromes that lie at the the rheumatology community, where the dominant idea is
extreme end of the spectrum of polysymptomatic distress. central sensitization (49).
784 Wolfe et al

An important strength of this study is the high-quality Historical and epidemiological evidence. Baillieres Best Pract
survey in which 56.7% of the requested population agreed Res Clin Rheumatol 1999;13:427–36.
7. Hauser W, Schmutzer G, Brahler E, Glaesmer H. A cluster
to participate. Participation rates that high are difficult to
within the continuum of biopsychosocial distress can be la-
get, and a recent Mayo Clinic epidemiology study that beled “fibromyalgia syndrome”: evidence from a represen-
used the 2010 criteria had 27.7% participation (16.2% in tative German population survey. J Rheumatol 2009;36:
the age group 21–39 years) (50). A potential limitation of 2806 –12.
our study was that we did not exclude subjects whose 8. Benjamin S, Morris S, McBeth J, Macfarlane GJ, Silman AJ.
The association between chronic widespread pain and mental
symptoms were less than 3 months in duration. We made disorder: a population-based study. Arthritis Rheum 2000;43:
this choice in order to be able to consider nonfibromyalgia 561–7.
subjects for whom the 3-month criterion was inappropri- 9. McBeth J, Macfarlane GJ, Hunt IM, Silman AJ. Risk factors
ate. Even so, only one fibromyalgia patient would have for persistent chronic widespread pain: a community-based
been excluded by the 3-month requirement, and the prev- study. Rheumatology (Oxford) 2001;40:95–101.
10. McBeth J, Macfarlane GJ, Benjamin S, Silman AJ. Features of
alence did not change at the first decimal point by keeping somatization predict the onset of chronic widespread pain:
this subject. Another limitation is that we did not know results of a large population-based study. Arthritis Rheum
if there were relevant clinical differences between study 2001;44:940 – 6.
participants and nonparticipants. We also made no exclu- 11. Winkler J. Measurement of social status by an index of
sions for the presence of “another disorder that would the health surveys of DHP. Hamburg: RKI-Schriften I. 1998:
69 –74.
otherwise sufficiently explain the pain.” This is a contro- 12. Wolfe F. Fibromyalgianess [editorial]. Arthritis Rheum 2009;
versial requirement because it is not certain how to define 61:715– 6.
such disorders. Even so, our epidemiology studies, as with 13. Hauser W, Jung E, Erbsloh-Moller B, Gesmann M, Kohn-
most fibromyalgia epidemiology studies, did not have suf- Becker H, Petermann F, et al. Validation of the Fibromyalgia
Survey Questionnaire within a cross-sectional survey. PLoS
ficient data to make such exclusions.
One 2012;7:e37504.
In summary, we found evidence to support the idea that 14. Kroenke K, Spitzer RL, Williams JB. The PHQ-15: validity of
fibromyalgia is a dimensional disorder, not a categorical a new measure for evaluating the severity of somatic symp-
disorder. Sex differences had limited effect on prevalence, toms. Psychosom Med 2002;64:258 – 66.
and approximately 40% of fibromyalgia patients satisfied 15. DSM-5 development. J00 somatic symptom disorder. Wash-
ington, DC: American Psychiatric Association; 2012. URL:
proposed DSM-V criteria for a physical symptom disorder. http://www.dsm5.org/ProposedRevisions/Pages/proposed
The modified fibromyalgia criteria that rely on self-report revision.aspx?rid⫽368.
can be a useful research tool for epidemiology and clinical 16. Kroenke K, Spitzer RL, Williams JB, Lowe B. An ultra-brief
research. screening scale for anxiety and depression: the PHQ-4. Psy-
chosomatics 2009;50:613–21.
17. Lowe B, Wahl I, Rose M, Spitzer C, Glaesmer H, Wingenfeld
K, et al. A 4-item measure of depression and anxiety: valida-
AUTHOR CONTRIBUTIONS tion and standardization of the Patient Health Questionnaire-
4 (PHQ-4) in the general population. J Affect Disord 2010;122:
All authors were involved in drafting the article or revising it
86 –95.
critically for important intellectual content, and all authors ap-
18. Aaronson NK, Ahmedzai S, Bergman B, Bullinger M, Cull A,
proved the final version to be published. Dr. Wolfe had full access
Duez NJ, et al. The European Organization for Research and
to all of the data in the study and takes responsibility for the
Treatment of Cancer QLQ-C30: a quality-of-life instrument for
integrity of the data and the accuracy of the data analysis.
use in international clinical trials in oncology. J Natl Cancer
Study conception and design. Brähler, Hinz, Häuser.
Inst 1993;85:365–76.
Acquisition of data. Brähler, Hinz.
19. Favers PM, Aaronson NK, Bjordal K, Groenvold M, Curran D,
Analysis and interpretation of data. Wolfe, Brähler, Häuser.
Bottomly A, et al. The EORTC QLQ-C30 scoring manual. 3rd
ed. Brussels: EORTC; 2001.
20. Royston P, Sauerbrei W. Multivariable model-building: a
REFERENCES pragmatic approach to regression analysis based on fractional
polynomials for modelling continuous variables. Chichester:
1. Wolfe F, Clauw DJ, FitzCharles MA, Goldenberg DL, Katz RS, Wiley; 2008.
Mease P, et al. The American College of Rheumatology pre- 21. Federal Statistical Office. Statistical yearbook. 2012. URL:
liminary diagnostic criteria for fibromyalgia and measurement https://www.destatis.de/DE/Publikationen/Statistisches
of symptom severity. Arthritis Care Res (Hoboken) 2010;62: Jahrbuch/StatistischesJahrbuch2012.pdf;jsessionid⫽2CD0AD
600 –10. FB63B227342A9DAD900C72E9D5.cae2?_blob⫽publicationFile.
2. Wolfe F, Clauw DJ, Fitzcharles MA, Goldenberg DL, Hauser 22. Landis JR, Koch GG. The measurement of observer agreement
W, Katz RS, et al. Fibromyalgia criteria and severity scales for for categorical data. Biometrics 1977;33:159 –74.
clinical and epidemiological studies: a modification of the 23. Katz RS, Wolfe F, Michaud K. Fibromyalgia diagnosis: a com-
ACR preliminary diagnostic criteria for fibromyalgia. J Rheu- parison of clinical, survey, and American College of Rheuma-
matol 2011;38:113–22. tology criteria. Arthritis Rheum 2006;54:169 –76.
3. Wolfe F, Smythe HA, Yunus MB, Bennett RM, Bombardier C, 24. Hauser W, Schmutzer G, Glaesmer H, Brahler E. Prevalence
Goldenberg DL, et al. The American College of Rheumatology and predictors of pain in several body regions: results of a
1990 criteria for the classification of fibromyalgia: report of representative German population survey. Schmerz 2009;23:
the Multicenter Criteria Committee. Arthritis Rheum 1990;33: 461–70. In German.
160 –72. 25. Branco JC, Bannwarth B, Failde I, Abello Carbonell J, Blotman
4. McBeth J, Mulvey MR. Fibromyalgia: mechanisms and poten- F, Spaeth M, et al. Prevalence of fibromyalgia: a survey in five
tial impact of the ACR 2010 classification criteria. Nat Rev European countries. Semin Arthritis Rheum 2010;39:448 –53.
Rheumatol 2012;8:108 –16. 26. Yunus M, Masi AT, Calabro JJ, Miller KA, Feigenbaum SL.
5. Wolfe F. New American College of Rheumatology criteria for Primary fibromyalgia (fibrositis): clinical study of 50 patients
fibromyalgia: a twenty-year journey [editorial]. Arthritis Care with matched normal controls. Semin Arthritis Rheum 1981;
Res (Hoboken) 2010;62:583– 4. 11:151–71.
6. Wessely S, Hotopf M. Is fibromyalgia a distinct clinical entity? 27. Coster L, Kendall S, Gerdle B, Henriksson C, Henriksson KG,
Prevalence, Dimensionality, and Somatic Symptom Severity of FM 785

Bengtsson A. Chronic widespread musculoskeletal pain: a 40. Hadler NM, Greenhalgh S. Labeling woefulness: the social
comparison of those who meet criteria for fibromyalgia and construction of fibromyalgia. Spine 2005;30:1– 4.
those who do not. Eur J Pain 2008;12:600 –10. 41. Cohen ML, Quintner JL. Fibromyalgia syndrome, a problem of
28. Ehrlich GE. Pain is real; fibromyalgia isn’t [editorial]. J Rheu- tautology. Lancet 1993;342:906 –9.
matol 2003;30:1666 –7. 42. Cohen ML, Quintner JL. The derailment of railway spine: a
29. Kessler RC. The categorical versus dimensional assessment timely lesson for post-traumatic fibromyalgia syndrome. Pain
controversy in the sociology of mental illness. J Health Soc Rev 1996;3:181–202.
Behav 2002;43:171– 88. 43. Cohen ML, Quintner JL. Fibromyalgia syndrome and dis-
30. Wolfe F, Rasker JJ. The Symptom Intensity Scale, fibromyal- ability: a failed construct fails those in pain. Med J Aust
gia, and the meaning of fibromyalgia-like symptoms. J Rheu- 1998;168:402– 4.
matol 2006;33:2291–9. 44. Ehrlich GE. Fibromyalgia, a virtual disease. Clin Rheumatol
31. Croft P, Burt J, Schollum J, Thomas E, Macfarlane G, Silman 2003;22:8 –11.
A. More pain, more tender points: is fibromyalgia just one end 45. Wolfe F. Fibromyalgia wars. J Rheumatol 2009;36:671– 8.
of a continuous spectrum? Ann Rheum Dis 1996;55:482–5.
46. Kroenke K. Somatoform disorders and recent diagnostic con-
32. Wolfe F. The relation between tender points and fibromyalgia
troversies. Psychiatr Clin North Am 2007;30:593– 619.
symptom variables: evidence that fibromyalgia is not a dis-
47. Fink P, Schroder A. One single diagnosis, bodily distress
crete disorder in the clinic. Ann Rheum Dis 1997;56:268 –71.
33. Freyberg RH. Non-articular rheumatism. Bull N Y Acad Med syndrome, succeeded to capture 10 diagnostic categories of
1951;27:245–58. functional somatic syndromes and somatoform disorders.
34. Graham W. The fibrositis syndrome. Bull Rheum Dis 1953;3: J Psychosom Res 2010;68:415–26.
33– 4. 48. Smith RC, Dwamena FC. Classification and diagnosis of pa-
35. Traut EF. Fibrositis. J Am Geriatr Soc 1968;16:531– 8. tients with medically unexplained symptoms. J Gen Intern
36. Fietta P, Manganelli P. Fibromyalgia and psychiatric disor- Med 2007;22:685–91.
ders. Acta Biomed 2007;78:88 –95. 49. Yunus MB. Central sensitivity syndromes: a new paradigm
37. Dadabhoy D, Clauw DJ. Therapy insight: fibromyalgia—a dif- and group nosology for fibromyalgia and overlapping condi-
ferent type of pain needing a different type of treatment. Nat tions, and the related issue of disease versus illness. Semin
Clin Pract Rheumatol 2006;2:364 –72. Arthritis Rheum 2008;37:339 –52.
38. Block SR. On the nature of rheumatism. Arthritis Care Res 50. Vincent A, Lahr BD, Wolfe F, Clauw DJ, Whipple MO, Oh TH,
1999;12:129 –38. et al. Prevalence of fibromyalgia: a population-based study in
39. Hadler NM. “Fibromyalgia” and the medicalization of misery. Olmsted County, Minnesota, utilizing the Rochester Epidemi-
J Rheumatol 2003;30:1668 –70. ology Project. Arthritis Care Res (Hoboken) 2013;65:786 –92.

You might also like