Fibromyalgia diagnosis and biased assessment: Sex, prevalence and bias

Abstract

Purpose: Multiple clinical and epidemiological studies have provided estimates of fibromyalgia prevalence and sex ratio, but different criteria sets and methodology, as well as bias, have led to widely varying (0.4%->11%) estimates of prevalence and female predominance (>90% to <61%). In general, studies have failed to distinguish Criteria based fibromyalgia (CritFM) from Clinical fibromyalgia (ClinFM). In the current study we compare CritFM with ClinFM to investigate gender and other biases in the diagnosis of fibromyalgia.

Methods: We used a rheumatic disease databank and 2016 fibromyalgia criteria to study prevalence and sex ratios in a selection biased sample of 1761 referred and diagnosed fibromyalgia patients and in an unbiased sample of 4342 patients with no diagnosis with respect to fibromyalgia. We compared diagnostic and clinical variables according to gender, and we reanalyzed a German population study (GPS) (n = 2435) using revised 2016 criteria for fibromyalgia.

Results: In the selection-biased sample of referred patients with fibromyalgia, more than 90% were women. However, when an unselected sample of rheumatoid arthritis (RA) patients was studied for the presence of fibromyalgia, women represented 58.7% of fibromyalgia cases. Women had slightly more symptoms than men, including generalized pain (36.8% vs. 32.4%), count of 37 symptoms (4.7 vs. 3.7) and mean polysymptomatic distress scores (10.2 vs. 8.2). We also found a linear relation between the probability of being females and fibromyalgia and fibromyalgia severity. Women in the GPS represented 59.2% of cases.

Discussion: The perception of fibromyalgia as almost exclusively (≥90%) a women's disorder is not supported by data in unbiased studies. Using validated self-report criteria and unbiased selection, the female proportion of fibromyalgia cases was ≤60% in the unbiased studies, and the observed CritFM prevalence of fibromyalgia in the GPS was ~2%. ClinFM is the public face of fibromyalgia, but is severely affected by selection and confirmation bias in the clinic and publications, underestimating men with fibromyalgia and overestimating women. We recommend the use of 2016 fibromyalgia criteria for clinical diagnosis and epidemiology because of its updated scoring and generalized pain requirement. Fibromyalgia and generalized pain positivity, widespread pain (WPI), symptom severity scale (SSS) and polysymptomatic distress (PSD) scale should always be reported.

Fig 1. Histogram of PSD scores in patients referred to NDB with diagnosis of fibromyalgia (S1 File).

A PSD ≥12 has a sensitivity of 100% for fibromyalgia diagnosis, but best cut point for correct classification is ≥16 (87.9%). Women represent 95.3% of subjects. Overall, 52.8% satisfy 2016 criteria (52.6% of women and 56.1% of men).

Fig 2. The relation of polysymptomatic distress (PSD) and its components to the sex of rheumatoid arthritis patients in the National Data Bank for Rheumatic Diseases (S2 File).

(2a, 2c and 2d) The proportion of patients who are females increases with greater PSD, WPI and SSS scores. Pr = probability. (B) The distribution of PSD scores is greater and shifted to the right in women compared with men.

Fig 3. The relation of polysymptomatic distress (PSD) and its components to the sex of rheumatoid arthritis patients in the German general population (S3 File).

(2a, 2c and 2d) The proportion of patients who are females increases with greater PSD, WPI and SSS scores. Pr = probability. (B) The distribution of PSD scores is greater and shifted to the right in women compared with men.