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• W4297731321 abstract "See related article, p 164 Acute or chronic musculoskeletal pain is a common reason for referral to the pediatric rheumatologist.1McGhee J.L. Burks F.N. Sheckels J.L. Jarvis J.N. Identifying children with chronic arthritis based on chief complaints: absence of predictive value for musculoskeletal pain as an indicator of rheumatic disease in children.Pediatrics. 2002; 110: 354-359Crossref PubMed Scopus (90) Google Scholar, 2Weiss J.E. Stinson J.N. Pediatric pain syndromes and noninflammatory musculoskeletal pain.Pediatr Clin North Am. 2018; 65: 801-826Abstract Full Text Full Text PDF PubMed Scopus (20) Google Scholar, 3Cattalini M. Parissenti I. Tononcelli E. Lancini F. Cantarini L. Meini A. Developing a predictive score for chronic arthritis among a cohort of children with musculoskeletal complaints—the Chronic Arthritis Score Study.J Pediatr. 2016; 169: 188-193Abstract Full Text Full Text PDF PubMed Scopus (4) Google Scholar The differential diagnosis is extensive. Patients may be diagnosed with inflammatory joint pain (including juvenile idiopathic arthritis [JIA]), arthritis related to infection (eg, reactive arthritis, including rheumatic fever and post-strep, viral arthritis, and Lyme disease), and other autoimmune and inflammatory disorders, such as systemic lupus erythematosus. Painful noninflammatory musculoskeletal disorders include overuse injuries (eg, patellofemoral pain syndrome [PFPS]), osteochondroses (ie, Osgood–Schlatter disease), benign joint hypermobility syndromes, benign limb pain of childhood, orthopedic disorders (eg, Legg–Calvé–Perthes and osteochondritis dissecans), and chronic musculoskeletal pain syndromes (CMPS), such as juvenile fibromyalgia, complex regional pain syndrome, and localized pain (eg, back pain).1McGhee J.L. Burks F.N. Sheckels J.L. Jarvis J.N. Identifying children with chronic arthritis based on chief complaints: absence of predictive value for musculoskeletal pain as an indicator of rheumatic disease in children.Pediatrics. 2002; 110: 354-359Crossref PubMed Scopus (90) Google Scholar, 2Weiss J.E. Stinson J.N. Pediatric pain syndromes and noninflammatory musculoskeletal pain.Pediatr Clin North Am. 2018; 65: 801-826Abstract Full Text Full Text PDF PubMed Scopus (20) Google Scholar, 3Cattalini M. Parissenti I. Tononcelli E. Lancini F. Cantarini L. Meini A. Developing a predictive score for chronic arthritis among a cohort of children with musculoskeletal complaints—the Chronic Arthritis Score Study.J Pediatr. 2016; 169: 188-193Abstract Full Text Full Text PDF PubMed Scopus (4) Google Scholar Although most patients referred to pediatric rheumatology have musculoskeletal pain, many of these patients do not end up with a diagnosis of an inflammatory disease, such as JIA. Joint pain is not required for the diagnosis of JIA, and not all patients with JIA have pain. In their retrospective chart review, McGhee et al found that children ultimately diagnosed with juvenile rheumatoid arthritis more commonly present with complaints of joint swelling (positive predictive value, 0.51) and/or gait disturbance.1McGhee J.L. Burks F.N. Sheckels J.L. Jarvis J.N. Identifying children with chronic arthritis based on chief complaints: absence of predictive value for musculoskeletal pain as an indicator of rheumatic disease in children.Pediatrics. 2002; 110: 354-359Crossref PubMed Scopus (90) Google Scholar Musculoskeletal pain as a presenting complaint had a strong negative predictive value for a diagnosis of either juvenile rheumatoid arthritis (0.95) or any other chronic inflammatory disease with arthritis.1McGhee J.L. Burks F.N. Sheckels J.L. Jarvis J.N. Identifying children with chronic arthritis based on chief complaints: absence of predictive value for musculoskeletal pain as an indicator of rheumatic disease in children.Pediatrics. 2002; 110: 354-359Crossref PubMed Scopus (90) Google Scholar Similarly, Cattalini et al also found that persistent swelling and morning stiffness were important characteristics of patients with juvenile chronic arthritis, and that their absence was highly indicative of a noninflammatory condition.3Cattalini M. Parissenti I. Tononcelli E. Lancini F. Cantarini L. Meini A. Developing a predictive score for chronic arthritis among a cohort of children with musculoskeletal complaints—the Chronic Arthritis Score Study.J Pediatr. 2016; 169: 188-193Abstract Full Text Full Text PDF PubMed Scopus (4) Google Scholar There was a split in the pain distribution in the patients with juvenile chronic arthritis, with patients reporting either constant pain (56%) or absence of pain (36%), whereas patients with infection-related arthritis and noninflammatory disorder had a more heterogeneous distribution of pain qualities.3Cattalini M. Parissenti I. Tononcelli E. Lancini F. Cantarini L. Meini A. Developing a predictive score for chronic arthritis among a cohort of children with musculoskeletal complaints—the Chronic Arthritis Score Study.J Pediatr. 2016; 169: 188-193Abstract Full Text Full Text PDF PubMed Scopus (4) Google Scholar The ubiquitous nature of musculoskeletal pain, along with the lack of sensitive and specific diagnostic tests for many pediatric rheumatic diseases makes diagnosis challenging. To aid diagnosis, patient-reported outcome (PRO) measures should be used to better assess a patient's symptoms, function, and well-being.4Rothman M.L. Beltran P. Cappelleri J.C. Lipscomb J. Teschendorf B. Mayo/FDA Patient-Reported Outcomes Consensus Meeting Group. Patient-reported outcomes: conceptual issues.Value Health. 2007; 10: S66-S75Abstract Full Text PDF PubMed Scopus (143) Google Scholar This is the premise of the report published in this volume of The Journal by van Straalen et al.5van Straalen J.W. van Stigt Thans M. Wulffraat N.M. de Roock S. Swart J.F. A diagnostic prediction model for separating juvenile idiopathic arthritis and chronic musculoskeletal pain syndrome.J Pediatr. 2022; 251: 164-171.e6Abstract Full Text Full Text PDF Scopus (1) Google Scholar The use of a PRO, such as the amended Juvenile Arthritis Multidimensional Assessment Report (JAMAR), may allow pediatric rheumatologists to more easily distinguish patients with JIA from those with CMPS.5van Straalen J.W. van Stigt Thans M. Wulffraat N.M. de Roock S. Swart J.F. A diagnostic prediction model for separating juvenile idiopathic arthritis and chronic musculoskeletal pain syndrome.J Pediatr. 2022; 251: 164-171.e6Abstract Full Text Full Text PDF Scopus (1) Google Scholar The JAMAR encompasses 15 PRO measures that assess well-being, pain, functional status, health-related quality of life, morning stiffness, disease activity, disease status and course, joint disease, extra-articular symptoms, medication side effects, compliance with therapy, and satisfaction with illness outcome, which can discriminate between healthy subjects and patients with JIA, and is a validated assessment tool for children with JIA.6Filocamo G. Consolaro A. Schiappapietra B. Dalprà S. Lattanzi B. Magni-Manzoni S. et al.A new approach to clinical care of juvenile idiopathic arthritis: the Juvenile Arthritis Multidimensional Assessment Report.J Rheumatol. 2011; 38: 938-953Crossref PubMed Scopus (135) Google Scholar, 7Lovell D.J. Brunner H.I. Ringold S. Weiss P.F. Martin N. Consolaro A. et al.The American English version of the Juvenile Arthritis Multidimensional Assessment Report (JAMAR).Rheumatol Int. 2018; 38: 35-42Crossref PubMed Scopus (6) Google Scholar, 8Bovis F. Consolaro A. Pistorio A. Garrone M. Scala S. Patrone E. et al.Cross-cultural adaptation and psychometric evaluation of the Juvenile Arthritis Multidimensional Assessment Report (JAMAR) in 54 languages across 52 countries: review of the general methodology.Rheumatol Int. 2018; 38: 5-17Crossref PubMed Scopus (68) Google Scholar A heterogeneous disease of unknown etiology, JIA can be classified using the International League of Associations for Rheumatology's criteria in children aged ≤16 years with arthritis in at least 1 joint for at least 6 weeks of duration with all other causes excluded.9Petty R.E. Southwood T.R. Baum J. Bhettay E. Glass D.N. Manners P. et al.Revision of the proposed classification criteria for juvenile idiopathic arthritis: Durban, 1997.J Rheumatol. 1998; 25: 1991-1994PubMed Google Scholar JIA is the most common chronic rheumatic disease in childhood, with an estimated incidence range of 1.6-23 and a prevalence of 3.8-400 per 100 000 children.10Gäre B.A. Epidemiology.Baillieres Clin Rheumatol. 1998; 12: 191-208Abstract Full Text PDF PubMed Scopus (37) Google Scholar,11Thierry S. Fautrel B. Lemelle I. Guillemin F. Prevalence and incidence of juvenile idiopathic arthritis: a systematic review.Joint Bone Spine. 2014; 81: 112-117Crossref PubMed Scopus (232) Google Scholar Van Straalen et al diagnosed CMPS if patients had persistent musculoskeletal pain for at least 3 months in the absence of any underlying cause. Diagnosis codes for eligible patients included “arthralgia,” “myalgia,” “myofascial pain syndrome/tendinitis,” “foot osteochondrosis,” “PFPS,” “low back pain,” and “orthopedic condition–not further specified.” Patients with localized pain had one joint group involved, and those with generalized pain had two or more joint groups involved.5van Straalen J.W. van Stigt Thans M. Wulffraat N.M. de Roock S. Swart J.F. A diagnostic prediction model for separating juvenile idiopathic arthritis and chronic musculoskeletal pain syndrome.J Pediatr. 2022; 251: 164-171.e6Abstract Full Text Full Text PDF Scopus (1) Google Scholar CMPS, such as juvenile fibromyalgia, usually presents as a primary chronic pain disorder with patients reporting widespread chronic musculoskeletal pain and associated symptoms such as fatigue, sleep disturbance, dysautonomia, mood disturbance, and headaches.2Weiss J.E. Stinson J.N. Pediatric pain syndromes and noninflammatory musculoskeletal pain.Pediatr Clin North Am. 2018; 65: 801-826Abstract Full Text Full Text PDF PubMed Scopus (20) Google Scholar,12Weiss J.E. Kashikar-Zuck S. Juvenile fibromyalgia.Rheum Dis Clin North Am. 2021; 47: 725-736Abstract Full Text Full Text PDF PubMed Scopus (3) Google Scholar It also can occur as a secondary or comorbid pain syndrome in patients with rheumatic diseases such as JIA, in whom pain symptoms persist once inflammation is controlled.12Weiss J.E. Kashikar-Zuck S. Juvenile fibromyalgia.Rheum Dis Clin North Am. 2021; 47: 725-736Abstract Full Text Full Text PDF PubMed Scopus (3) Google Scholar The diagnosis of juvenile fibromyalgia is based on either the Yunus and Masi juvenile fibromyalgia classification criteria or the American College of Rheumatology's 2010 fibromyalgia syndrome criteria.13Yunus M. Masi A.T. Calabro J.J. Miller K.A. Feigenbaum S.L. Primary fibromyalgia (fibrositis): clinical study of 50 patients with matched normal controls.Semin Arthritis Rheum. 1981; 11: 151-171Crossref PubMed Scopus (785) Google Scholar,14Wolfe F. Clauw D.J. Fitzcharles M.A. Goldenberg D.L. Katz R.S. Mease P. et al.The American College of Rheumatology preliminary diagnostic criteria for fibromyalgia and measurement of symptom severity.Arthritis Care Res (Hoboken). 2010; 62: 600-610Crossref PubMed Scopus (2539) Google Scholar Patients with JIA and CMPS may have similar symptoms, and currently there is no diagnostic tool to easily differentiate between the 2 conditions. In a retrospective cohort study from The Netherlands, Van Straalen et al assessed whether the JAMAR, completed within 3 weeks of the initial visit by all patients attending a pediatric rheumatology clinic since 2012, would perform well in differentiating JIA from patients with CMPS. Patients included in the study were age <18 years, had a completed JAMAR and were diagnosed with JIA or CMPS by the pediatric rheumatology team after the first visit. A diagnostic prediction model was developed using data from 196 eligible patients (49.5% diagnosed with JIA) who completed a JAMAR before July 1, 2018. The model was subsequently validated in 91 patients (52.7% with JIA) using new data collected between July 1, 2018, and May 28, 2020. The authors predicted outcome, using the modified JAMAR, was a correct diagnosis of JIA (using International League of Associations for Rheumatology criteria). CMPS was diagnosed as defined above; no specific diagnostic criteria were used, and diagnoses ranged from arthralgia to PFPS and orthopedic conditions. Predictors included were age at the first visit, sex, and specific components of the JAMAR. Adaptation of the JAMAR, based on the authors’ hypothesis that asymmetric joint involvement would be associated with JIA rather than with CMPS, included combining items about pain and joint swelling into 3 categories: no pain or swelling, pain or swelling on the left or right side (asymmetric pain/swelling), and pain or swelling on both the left and right side (symmetric pain/swelling). Patients with JIA were more often male, had asymmetric pain/swelling, and reported more skin rash and difficulty with self-care compared with patients with CMPS. Patients with CMPS were more often female and older and more often reported symmetric joint involvement and a painful or swollen lower back and neck. They also had a significantly higher median number of self-reported painful or swollen joints than patients with JIA. Both groups had substantial pain, as demonstrated on a visual analog pain scale (median score, 5.5; IQR, 3.0-7.3).5van Straalen J.W. van Stigt Thans M. Wulffraat N.M. de Roock S. Swart J.F. A diagnostic prediction model for separating juvenile idiopathic arthritis and chronic musculoskeletal pain syndrome.J Pediatr. 2022; 251: 164-171.e6Abstract Full Text Full Text PDF Scopus (1) Google Scholar For both the model development and validation cohorts, most patients with JIA had oligoarthritis, and the majority of patients with CMPS had generalized pain. Predictions in the validation data were calculated for 83 patients, and calibration and discrimination (area under the curve, 0.83; 95% CI, 0.74-0.92) were good. At the cutoff threshold of 70% for the predicted probability of JIA, the model had a negative predictive value of 85% and a positive predictive value of 66%.5van Straalen J.W. van Stigt Thans M. Wulffraat N.M. de Roock S. Swart J.F. A diagnostic prediction model for separating juvenile idiopathic arthritis and chronic musculoskeletal pain syndrome.J Pediatr. 2022; 251: 164-171.e6Abstract Full Text Full Text PDF Scopus (1) Google Scholar The authors conclude that items from the JAMAR, which is readily available and easy to use, can distinguish patients with JIA from CMPS at the first visit, thereby facilitating earlier diagnosis, treatment, and referral. They offer a risk calculator and nomogram based on the prediction model from which predicted risks of JIA can be calculated. Because some patients with CMPS may be incorrectly diagnosed with JIA based on this PRO tool, it is important to note that patients still need to be examined by a pediatric rheumatologist to confirm the diagnosis of JIA. Strengths of this study include the large pediatric rheumatology patient registry and that approximately 300 patients with either JIA or CMPS had completed the JAMAR. Limitations include use of the JAMAR as a tool to distinguish between the different subtypes of JIA. The JAMAR also has not been validated for diagnosis or assessment of CMPS (localized or generalized) or for detection of secondary CMPSs, such as juvenile fibromyalgia in the subset of patients with JIA who suffer from widespread musculoskeletal pain as a secondary condition.15Tesher M.S. Graham T.B. Ting T. Kashikar-Zuck S. Lynch N. Wroblewski K. et al.Juvenile fibromyalgia in patients with juvenile idiopathic arthritis: utility of the Pain and Symptom Assessment Tool (PSAT).Arthritis Care Res (Hoboken). 2021; ([Epub ahead of print])https://doi.org/10.1002/acr.24739Crossref Scopus (3) Google Scholar The diagnoses used for the patients with CMPS did not include juvenile fibromyalgia or complex regional pain syndrome, making it impossible to differentiate between patients with amplified musculoskeletal pain syndromes and those with orthopedic conditions. Understandably, owing to the retrospective nature of this research, more specific diagnostic tools could not be applied to patients for all types of CMPS. The authors recognized this limitation and used different predictor variables in their secondary analyses restricted to oligoarthritis and localized pain or diffuse complaints (polyarthritis and generalized pain syndrome) with excellent discrimination in the first group and good discrimination in the second group for both the model development and validation data.5van Straalen J.W. van Stigt Thans M. Wulffraat N.M. de Roock S. Swart J.F. A diagnostic prediction model for separating juvenile idiopathic arthritis and chronic musculoskeletal pain syndrome.J Pediatr. 2022; 251: 164-171.e6Abstract Full Text Full Text PDF Scopus (1) Google Scholar They stress the point of using the modified JAMAR to make a timely diagnosis of inflammatory or noninflammatory joint pain, not to further subtype JIA and CMPS. Research assessing the range of chronic pain conditions that present to pediatric rheumatology clinics is lacking, and studies such as this add to the literature on patients with CMPS and demonstrate the frequency with which patients present to rheumatology with chronic noninflammatory joint pain. There are also additional PRO measures that could be assessed in future studies, such as the Pain and Symptom Assessment Tool, based on the American College of Rheumatology's 2010 fibromyalgia diagnostic criteria, to assess painful body locations as well as the characteristic associated symptoms mentioned earlier to more clearly identify patients who may meet criteria for a primary or secondary CMPS condition such as juvenile fibromyalgia.15Tesher M.S. Graham T.B. Ting T. Kashikar-Zuck S. Lynch N. Wroblewski K. et al.Juvenile fibromyalgia in patients with juvenile idiopathic arthritis: utility of the Pain and Symptom Assessment Tool (PSAT).Arthritis Care Res (Hoboken). 2021; ([Epub ahead of print])https://doi.org/10.1002/acr.24739Crossref Scopus (3) Google Scholar,16Ting T.V. Barnett K. Lynch-Jordan A. Whitacre C. Henrickson M. Kashikar-Zuck S. 2010 American College of Rheumatology adult fibromyalgia criteria for use in an adolescent female population with juvenile fibromyalgia.J Pediatr. 2016; 169: 181-187Abstract Full Text Full Text PDF PubMed Scopus (47) Google Scholar Improving the process of quality care delivery through timely diagnosis is a topical issue, and this study shows promising results. As of 2021, only 485 US-based pediatricians have been board-certified as pediatric rheumatologists, and some states do not have even one pediatric rheumatologist (www.abp.org).17American College of Rheumatology: Workforce study of rheumatology specialists in the United States 2015.https://www.rheumatology.org/portals/0/files/ACR-Workforce-Study-2015.pdfDate: 2016Date accessed: August 31, 2022Google Scholar, 18Argraves M. Mehta J.J. Pediatric rheumatology: Not just a little adult workforce issue.Semin Arthritis Rheum. 2021; 51: e13Crossref PubMed Scopus (2) Google Scholar, 19American Board of Pediatrics: Pediatric Physicians Workforce Data Book 2020-2021.https://www.abp.org/sites/public/files/pdf/workforcedata2020-2021.pdfDate accessed: August 31, 2022Google Scholar This PRO tool potentially can be used by general pediatricians to identify and prioritize children with possible JIA who need prompt referral to rheumatology. In addition, pediatric rheumatology specialists could use this measure prior to the visit to allow for a more productive office visit. Additional prospective studies using the modified JAMAR are needed, but physicians should consider adopting items from the JAMAR questionnaire in clinical practice to help distinguish JIA from CMPS in patients with corresponding symptoms. A Diagnostic Prediction Model for Separating Juvenile Idiopathic Arthritis and Chronic Musculoskeletal Pain SyndromeThe Journal of PediatricsVol. 251PreviewTo develop and validate a diagnostic prediction model that can distinguish between juvenile idiopathic arthritis (JIA) and chronic musculoskeletal pain syndrome (CMPS) based on patient-reported outcomes. Full-Text PDF Open Access" @default.
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• W4297731321 title "Prediction Model for Juvenile Idiopathic Arthritis: Challenges and Opportunities" @default.
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