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      Fibromyalgia diagnosis and biased assessment: Sex, prevalence and bias

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          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.

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          Most cited references33

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          The dimensions of health outcomes: the health assessment questionnaire, disability and pain scales.

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            Primary fibromyalgia (fibrositis): clinical study of 50 patients with matched normal controls.

            Detailed clinical study of 50 patients with primary fibromyalgia and 50 normal matched controls has shown a characteristic syndrome. Primary fibromyalgia patients are usually females, aged 25-40 yr, who complain of diffuse musculoskeletal aches, pains or stiffness associated with tiredness, anxiety, poor sleep, headaches, irritable bowel syndrome, subjective swelling in the articular and periarticular areas and numbness. Physical examination is characterized by presence of multiple tender points at specific sites and absence of joint swelling. Symptoms are influenced by weather and activities, as well as by time of day(worse in the morning and the evening). In contrast, symptoms of psychogenic rheumatism patients have little fluctuation, if any, and are modulated by emotional rather than physical factors. In psychogenic rheumatism, there is diffuse tenderness rather than tender points at specific sites. Laboratory tests and roentgenologic findings in primary fibromyalgia are normal or negative. Primary fibromyalgia should be suspected by the presence of its own characteristic features, and not diagnosed just by the absence of other recognizable conditions. This study has also shown that primary fibromyalgia is a poorly recognized condition. Patients were usually seen by many physicians who failed to provide a definite diagnosis despite frequent unnecessary investigations. A guideline for diagnosis of primary fibromyalgia, based upon our observations, is suggested. Management is usually gratifying in these frustrated patients. The most important aspects are a definite diagnosis, explanation of the various possible mechanisms responsible for the symptoms, and reassurance regarding the benign nature of this condition. A combination of reassurance, nonsteroidal antiinflammatory drugs, good sleep, local tender point injections, and various modes of physical therapy is successful in most cases.
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              Prevalence of fibromyalgia: literature review update

              ABSTRACT The present study aimed to update the literature review on the prevalence of fibromyalgia published in 2006. A bibliographical survey was carried out from 2005 to 2014 in the MEDLINE, Web of Science, Embase, LILACS and SciELO databases and 3274 records were identified. Five researchers selected the studies, following the inclusion criteria: studies that obtained the prevalence of fibromyalgia. Fibromyalgia studies in associated diseases were excluded. When screening by title and abstract, 2073 irrelevant articles were excluded. The full texts of 210 articles were evaluated for eligibility and this review included 39 studies, described in 41 articles. The selected studies were grouped into four categories: (A) prevalence of fibromyalgia in the general population; (B) prevalence of fibromyalgia in women; (C) prevalence of fibromyalgia in rural and urban areas; (D) prevalence of fibromyalgia in special populations. The literature shows values of fibromyalgia prevalence in the general population between 0.2 and 6.6%, in women between 2.4 and 6.8%, in urban areas between 0.7 and 11.4%, in rural areas between 0.1 and 5.2%, and in special populations values between 0.6 and 15%. This literature review update shows a significant increase in fibromyalgia prevalence studies in the world. The new 2010 American College of Rheumatology criteria have not been widely used yet and the COPCORD (Community-oriented program for control of Rheumatic Diseases) methodology has increased the quality of studies on the prevalence of rheumatic diseases in general.
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                Author and article information

                Contributors
                Role: ConceptualizationRole: Data curationRole: Formal analysisRole: MethodologyRole: Writing – original draft
                Role: ConceptualizationRole: Formal analysisRole: InvestigationRole: Writing – review & editing
                Role: ConceptualizationRole: Writing – review & editing
                Role: ConceptualizationRole: Writing – review & editing
                Role: ConceptualizationRole: Writing – review & editing
                Role: Editor
                Journal
                PLoS One
                PLoS ONE
                plos
                plosone
                PLoS ONE
                Public Library of Science (San Francisco, CA USA )
                1932-6203
                13 September 2018
                2018
                : 13
                : 9
                : e0203755
                Affiliations
                [1 ] National Data Bank for Rheumatic Diseases, Wichita, Kansas, United States of America
                [2 ] University of Kansas School of Medicine, Wichita, Kansas, United States of America
                [3 ] Georgetown University, Washington, DC, United States of America
                [4 ] Pain Clinic, Cochin-Hôtel Dieu Hospital, Paris Descartes University, Paris, France
                [5 ] Faculty of Behavioral Management & Social Sciences, Psychology, Health & Technology, University of Twente, Enschede, the Netherlands
                [6 ] Department Internal Medicine 1, Klinikum Saarbrücken, Saarbrücken, Germany
                [7 ] Department Psychosomatic Medicine and Psychotherapy, Technische Universität München, München, Germany
                University of Würzburg, GERMANY
                Author notes

                Competing Interests: The authors have declared that no competing interests exist.

                Author information
                http://orcid.org/0000-0002-2801-6413
                Article
                PONE-D-17-31720
                10.1371/journal.pone.0203755
                6136749
                30212526
                72049d76-677f-4319-af50-7f23e96910e4

                This is an open access article, free of all copyright, and may be freely reproduced, distributed, transmitted, modified, built upon, or otherwise used by anyone for any lawful purpose. The work is made available under the Creative Commons CC0 public domain dedication.

                History
                : 29 August 2017
                : 27 August 2018
                Page count
                Figures: 3, Tables: 2, Pages: 14
                Funding
                The authors received no specific funding for this work.
                Categories
                Research Article
                Medicine and Health Sciences
                Neurology
                Neuromuscular Diseases
                Fibromyalgia
                Medicine and Health Sciences
                Rheumatology
                Fibromyalgia
                People and Places
                Population Groupings
                Professions
                Medical Personnel
                Medical Doctors
                Physicians
                Medicine and Health Sciences
                Health Care
                Health Care Providers
                Medical Doctors
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                Medicine and Health Sciences
                Rheumatology
                Arthritis
                Rheumatoid Arthritis
                Medicine and Health Sciences
                Clinical Medicine
                Clinical Immunology
                Autoimmune Diseases
                Rheumatoid Arthritis
                Biology and Life Sciences
                Immunology
                Clinical Immunology
                Autoimmune Diseases
                Rheumatoid Arthritis
                Medicine and Health Sciences
                Immunology
                Clinical Immunology
                Autoimmune Diseases
                Rheumatoid Arthritis
                Medicine and Health Sciences
                Diagnostic Medicine
                Medicine and Health Sciences
                Diagnostic Medicine
                Signs and Symptoms
                Pain
                Abdominal Pain
                Medicine and Health Sciences
                Pathology and Laboratory Medicine
                Signs and Symptoms
                Pain
                Abdominal Pain
                Medicine and Health Sciences
                Rheumatology
                Medicine and Health Sciences
                Diagnostic Medicine
                Signs and Symptoms
                Pain
                Lower Back Pain
                Medicine and Health Sciences
                Pathology and Laboratory Medicine
                Signs and Symptoms
                Pain
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