Publications by authors named "Marite Rygg"

45 Publications

Pain sensitivity in young adults with juvenile idiopathic arthritis: a quantitative sensory testing study.

Arthritis Res Ther 2020 11 5;22(1):262. Epub 2020 Nov 5.

Department of Clinical and Molecular Medicine, NTNU-Norwegian University of Science and Technology, Trondheim, Norway.

Background: To study for the first-time, pain perception, pain sensitivity, and self-reported pain in young adults with long disease duration of juvenile idiopathic arthritis (JIA) compared with controls.

Methods: Children from Central Norway diagnosed with JIA between 1997 and 2004 were included consecutively in a population-based prospective study. Children with onset 1997-2000 were part of the Nordic JIA cohort. Controls were age- and sex-matched. In 2015-2017, study visits with investigator-blinded quantitative sensory testing (QST) comprising cold and warm detection thresholds (CDT/WDT), cold and heat pain thresholds (CPT/HPT), pressure pain threshold (PPT), and a suprathreshold heat pain test were performed. We constructed separate multilevel models for each variable of detection and pain thresholds with interaction between groups and site adjusted for the effect of age and sex.

Results: Among 96 young adults with JIA, 71% were female, median age was 22.7 years, disease duration was 16.1 years, and 47% had oligoarticular disease. Among 109 controls, 71% were female, and median age was 23.5 years. Participants with JIA had lower pressure pain thresholds (PPTs) (95% CI) compared to controls, upper limb 888 (846,930) versus 1029 (999,1059) kPa and lower limb 702 (670,734) versus 760 (726,794) kPa. Participants with inactive disease had the lowest PPTs and cold pain thresholds (CPTs), compared to those in remission off medication and those with active disease. Minor differences were found regarding CDT/WDT and CPT/HPT in JIA compared to controls. The median (IQR) temperature needed to evoke pain = 6 on a 0-10 numeric rating scale (NRS) in the suprathreshold heat pain tests were lower in JIA than in controls (46 °C (45-47 °C) versus 47 °C (46-48 °C)). We found no associations between self-reported pain and pain thresholds.

Conclusions: Our results indicate for the first time that young adults with long disease duration of JIA may have altered pain perception and sensitivity compared to controls. A clinical implication may be the importance of early treatment to quickly achieve pain-free remission and avoid long-term pain sensitization.
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http://dx.doi.org/10.1186/s13075-020-02345-2DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7643261PMC
November 2020

Uveitis in Juvenile Idiopathic Arthritis: 18-Year Outcome in the Population-based Nordic Cohort Study.

Ophthalmology 2020 Aug 29. Epub 2020 Aug 29.

Department of Pediatrics and Adolescent Medicine, University Hospital of North Norway, Tromsø, Norway; Department of Clinical Medicine, UiT - The Arctic University of Norway, Tromsø, Norway.

Purpose: To assess the long-term outcome of uveitis in juvenile idiopathic arthritis (JIA).

Design: Population-based, multicenter, prospective JIA cohort, with a cross-sectional assessment of JIA-associated uveitis (JIA-U) 18 years after the onset of JIA.

Participants: A total of 434 patients with JIA, of whom 96 had uveitis, from defined geographic areas of Denmark, Finland, Norway, and Sweden.

Methods: Patients with onset of JIA between January 1997 and June 2000 were prospectively followed for 18 years. Pediatric rheumatologists and ophthalmologists collected clinical and laboratory data.

Main Outcome Measures: Cumulative incidence of uveitis and clinical characteristics, JIA and uveitis disease activity, ocular complications, visual outcome, and risk factors associated with the development of uveitis-related complications.

Results: Uveitis developed in 96 (22.1%) of 434 patients with JIA. In 12 patients (2.8%), uveitis was diagnosed between 8 and 18 years of follow-up. Systemic immunosuppressive medication was more common among patients with uveitis (47/96 [49.0%]) compared with patients without uveitis (78/338 [23.1%]). Active uveitis was present in 19 of 78 patients (24.4%) at the 18-year visit. Ocular complications occurred in 31 of 80 patients (38.8%). Short duration between the onset of JIA and the diagnosis of uveitis was a risk factor for developing ocular complications (odds ratio [OR], 1.4; 95% confidence interval [CI], 1.1-1.8). Patients with a diagnosis of uveitis before the onset of JIA all developed cataract and had an OR for development of glaucoma of 31.5 (95% CI, 3.6-274). Presence of antinuclear antibodies (ANAs) was also a risk factor for developing 1 or more ocular complications (OR, 3.0; 95% CI, 1.2-7.7). Decreased visual acuity (VA) <6/12 was found in 12 of 135 eyes (8.9%) with uveitis, and 4 of 80 patients (5.0%) with JIA-U had binocular decreased VA <6/12.

Conclusions: Our results suggest that uveitis screening should start immediately when the diagnosis of JIA is suspected or confirmed and be continued for more than 8 years after the diagnosis of JIA. Timely systemic immunosuppressive treatment in patients with a high risk of developing ocular complications must be considered early in the disease course to gain rapid control of ocular inflammation.
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http://dx.doi.org/10.1016/j.ophtha.2020.08.024DOI Listing
August 2020

Psychiatric morbidity, somatic comorbidity and substance use in an adolescent psychiatric population at 3-year follow-up.

Eur Child Adolesc Psychiatry 2020 Jul 15. Epub 2020 Jul 15.

Department of Clinical and Molecular Medicine (IKOM), Faculty of Medicine and Health Sciences, Norwegian University of Science and Technology, Trondheim, Norway.

Knowledge is scarce on the course of psychiatric disorders in adolescence. We aimed to assess changes in the frequency of psychiatric disorders, somatic disorders, pain, and substance use in a clinical psychiatric cohort from adolescence to young adulthood. This study is part of the Health Survey in Department of Children and Youth, St. Olavs Hospital, Norway. At age 13-18 years, 717 (43.5% of eligible) participated in the first study visit (T) in 2009-2011, 549 were reassessed 3 years later with telephone interview (T), and 464 had diagnostic evaluation at both time points. Data included: ICD-10 diagnoses (T), DSM-IV diagnoses (T), self-reported pain and substance use (T and T). The overall rate of psychiatric disorders decreased (T vs. T: 94.8% vs. 72.2%, p < 0.001); while, an increased rate of anxiety disorders was marked among girls (37.5% vs. 55.9%, p < 0.001), with accompanying raised frequencies of psychiatric comorbidity (14.1% vs. 42.6%, p < 0.001), somatic comorbidity (9.4% vs. 19.5%, p = 0.001), chronic pain (31.6% vs. 49.4%, p < 0.001), smoking, alcohol use and trying illicit drugs. Chronic pain, smoking and trying illicit drugs were associated with persisting psychiatric disorders, with highest risk differences for girls (RD = 25.4%, p = 0.002, RD = 15.6%, p = 0.008, RD = 18.0%, p = 0.001, respectively). Three out of four adolescents still had a psychiatric disorder after 3 years. Unlike boys, girls had an increasing rate of anxiety disorders and comorbidities. Chronic pain, smoking and trying illicit drugs were associated with persisting psychiatric disorders. Despite methodological limitations, these findings emphasize the importance of early targeted intervention for adolescents with psychiatric disorders.
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http://dx.doi.org/10.1007/s00787-020-01602-8DOI Listing
July 2020

Opportunistic infections in immunosuppressed patients with juvenile idiopathic arthritis: analysis by the Pharmachild Safety Adjudication Committee.

Arthritis Res Ther 2020 04 7;22(1):71. Epub 2020 Apr 7.

IRCCS Istituto Giannina Gaslini, Clinica Pediatrica e Reumatologia, PRINTO, Genoa, Italy.

Background: To derive a list of opportunistic infections (OI) through the analysis of the juvenile idiopathic arthritis (JIA) patients in the Pharmachild registry by an independent Safety Adjudication Committee (SAC).

Methods: The SAC (3 pediatric rheumatologists and 2 pediatric infectious disease specialists) elaborated and approved by consensus a provisional list of OI for use in JIA. Through a 5 step-procedure, all the severe and serious infections, classified as per MedDRA dictionary and retrieved in the Pharmachild registry, were evaluated by the SAC by answering six questions and adjudicated with the agreement of 3/5 specialists. A final evidence-based list of OI resulted by matching the adjudicated infections with the provisional list of OI.

Results: A total of 772 infectious events in 572 eligible patients, of which 335 serious/severe/very severe non-OI and 437 OI (any intensity/severity), according to the provisional list, were retrieved. Six hundred eighty-two of 772 (88.3%) were adjudicated as infections, of them 603/682 (88.4%) as common and 119/682 (17.4%) as OI by the SAC. Matching these 119 opportunistic events with the provisional list, 106 were confirmed by the SAC as OI, and among them infections by herpes viruses were the most frequent (68%), followed by tuberculosis (27.4%). The remaining events were divided in the groups of non-OI and possible/patient and/or pathogen-related OI.

Conclusions: We found a significant number of OI in JIA patients on immunosuppressive therapy. The proposed list of OI, created by consensus and validated in the Pharmachild cohort, could facilitate comparison among future pharmacovigilance studies.

Trial Registration: Clinicaltrials.gov NCT01399281; ENCePP seal: awarded on 25 November 2011.
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http://dx.doi.org/10.1186/s13075-020-02167-2DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7136994PMC
April 2020

Distinct interferon signatures and cytokine patterns define additional systemic autoinflammatory diseases.

J Clin Invest 2020 04;130(4):1669-1682

Translational Autoinflammatory Diseases Section (TADS), NIAID/NIH, Bethesda, Maryland, USA.

BACKGROUNDUndifferentiated systemic autoinflammatory diseases (USAIDs) present diagnostic and therapeutic challenges. Chronic interferon (IFN) signaling and cytokine dysregulation may identify diseases with available targeted treatments.METHODSSixty-six consecutively referred USAID patients underwent underwent screening for the presence of an interferon signature using a standardized type-I IFN-response-gene score (IRG-S), cytokine profiling, and genetic evaluation by next-generation sequencing.RESULTSThirty-six USAID patients (55%) had elevated IRG-S. Neutrophilic panniculitis (40% vs. 0%), basal ganglia calcifications (46% vs. 0%), interstitial lung disease (47% vs. 5%), and myositis (60% vs. 10%) were more prevalent in patients with elevated IRG-S. Moderate IRG-S elevation and highly elevated serum IL-18 distinguished 8 patients with pulmonary alveolar proteinosis (PAP) and recurrent macrophage activation syndrome (MAS). Among patients with panniculitis and progressive cytopenias, 2 patients were compound heterozygous for potentially novel LRBA mutations, 4 patients harbored potentially novel splice variants in IKBKG (which encodes NF-κB essential modulator [NEMO]), and 6 patients had de novo frameshift mutations in SAMD9L. Of additional 12 patients with elevated IRG-S and CANDLE-, SAVI- or Aicardi-Goutières syndrome-like (AGS-like) phenotypes, 5 patients carried mutations in either SAMHD1, TREX1, PSMB8, or PSMG2. Two patients had anti-MDA5 autoantibody-positive juvenile dermatomyositis, and 7 could not be classified. Patients with LRBA, IKBKG, and SAMD9L mutations showed a pattern of IRG elevation that suggests prominent NF-κB activation different from the canonical interferonopathies CANDLE, SAVI, and AGS.CONCLUSIONSIn patients with elevated IRG-S, we identified characteristic clinical features and 3 additional autoinflammatory diseases: IL-18-mediated PAP and recurrent MAS (IL-18PAP-MAS), NEMO deleted exon 5-autoinflammatory syndrome (NEMO-NDAS), and SAMD9L-associated autoinflammatory disease (SAMD9L-SAAD). The IRG-S expands the diagnostic armamentarium in evaluating USAIDs and points to different pathways regulating IRG expression.TRIAL REGISTRATIONClinicalTrials.gov NCT02974595.FUNDINGThe Intramural Research Program of the NIH, NIAID, NIAMS, and the Clinical Center.
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http://dx.doi.org/10.1172/JCI129301DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7108905PMC
April 2020

Oral health in children and adolescents with juvenile idiopathic arthritis - a systematic review and meta-analysis.

BMC Oral Health 2019 12 19;19(1):285. Epub 2019 Dec 19.

Department of Clinical Dentistry, Pediatric Dentistry, The Faculty of Medicine, University of Bergen, Norway, Årstadveien 19, N-5009, Bergen, Norway.

Background: Observational studies examining the association between oral health and juvenile idiopathic arthritis (JIA) among children and adolescents have reported inconsistent findings. The aims of this systematic review and meta-analysis were to ascertain a potential difference in oral health and oral health-related quality of life (OHRQoL) among children and adolescents with JIA and healthy peers, and to assess the association of prevalence of oral diseases/conditions, temporomandibular disorders (TMD), including temporomandibular joint (TMJ) diseases, in relation to activity and severity of JIA.

Method: Medline Ovid, Embase, CINAHL, SweMed+ and Cochrane Library were searched up to 25 November 2018. All articles published in English, German and Scandinavian languages focusing on children and adolescents with JIA and without JIA in relation to oral health measures, were considered. Two authors independently evaluated observational studies for inclusion. The study quality was assessed using modified Newcastle Ottawa Scale. Meta-analysis was performed for studies focusing on dental caries as an outcome.

Results: Nineteen articles met the inclusion criteria, covering a range of oral diseases/conditions and OHRQoL. Eighteen studies had cross-sectional design. No mean difference of dmft/DMFT indices (decayed/missed/filled teeth) was observed between the JIA - and healthy group. None of the oral health measures including dental erosive wear, enamel defects, dental maturation and OHRQoL, indicated better oral health among children and adolescents with JIA compared to healthy group. However, periodontal conditions and TMD were more predominant among children and adolescents with JIA compared to healthy peers.

Conclusions: Based on the cross-sectional studies, periodontal diseases and TMD were found to be more frequent in children and adolescents with JIA compared to healthy peers. Furthermore, more high-quality studies with large sample size are needed before we infer any concrete conclusion regarding the association between the prevalence of oral and TMJ diseases or oral conditions in relation to activity and severity of JIA.
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http://dx.doi.org/10.1186/s12903-019-0965-4DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6921440PMC
December 2019

Validation of prediction models of severe disease course and non-achievement of remission in juvenile idiopathic arthritis: part 1-results of the Canadian model in the Nordic cohort.

Arthritis Res Ther 2019 12 5;21(1):270. Epub 2019 Dec 5.

Department of Pediatrics, University Hospital of North Norway, Tromsø, Norway.

Background: Models to predict disease course and long-term outcome based on clinical characteristics at disease onset may guide early treatment strategies in juvenile idiopathic arthritis (JIA). Before a prediction model can be recommended for use in clinical practice, it needs to be validated in a different cohort than the one used for building the model. The aim of the current study was to validate the predictive performance of the Canadian prediction model developed by Guzman et al. and the Nordic model derived from Rypdal et al. to predict severe disease course and non-achievement of remission in Nordic patients with JIA.

Methods: The Canadian and Nordic multivariable logistic regression models were evaluated in the Nordic JIA cohort for prediction of non-achievement of remission, and the data-driven outcome denoted severe disease course. A total of 440 patients in the Nordic cohort with a baseline visit and an 8-year visit were included. The Canadian prediction model was first externally validated exactly as published. Both the Nordic and Canadian models were subsequently evaluated with repeated fine-tuning of model coefficients in training sets and testing in disjoint validation sets. The predictive performances of the models were assessed with receiver operating characteristic curves and C-indices. A model with a C-index above 0.7 was considered useful for clinical prediction.

Results: The Canadian prediction model had excellent predictive ability and was comparable in performance to the Nordic model in predicting severe disease course in the Nordic JIA cohort. The Canadian model yielded a C-index of 0.85 (IQR 0.83-0.87) for prediction of severe disease course and a C-index of 0.66 (0.63-0.68) for prediction of non-achievement of remission when applied directly. The median C-indices after fine-tuning were 0.85 (0.80-0.89) and 0.69 (0.65-0.73), respectively. Internal validation of the Nordic model for prediction of severe disease course resulted in a median C-index of 0.90 (0.86-0.92).

Conclusions: External validation of the Canadian model and internal validation of the Nordic model with severe disease course as outcome confirm their predictive abilities. Our findings suggest that predicting long-term remission is more challenging than predicting severe disease course.
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http://dx.doi.org/10.1186/s13075-019-2060-2DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6896283PMC
December 2019

Longterm Outcomes of Temporomandibular Joints in Juvenile Idiopathic Arthritis: 17 Years of Followup of a Nordic Juvenile Idiopathic Arthritis Cohort.

J Rheumatol 2020 05 15;47(5):730-738. Epub 2019 Sep 15.

From the Department of Pediatrics, Aarhus University Hospital; Section of Orthodontics, Aarhus University; Section of Oral Radiology, Department of Dentistry and Oral Health, Aarhus University, Aarhus, Denmark; Department of Pediatrics, University Hospital of North Norway, Tromsø; Department of Clinical Medicine, Faculty of Health Sciences, UiT The Arctic University of Norway, Tromsø; Department of Otorhinolaryngology and Department and Division of Oral and Maxillofacial Surgery, University Hospital North Norway and Public Dental Service Competence Center of North Norway, Tromsø; Department of Clinical and Molecular Medicine, Faculty of Medicine and Health Sciences, Norwegian University of Science and Technology (NTNU), Trondheim; Department of Pediatrics, Levanger Hospital, Nord-Trøndelag Hospital Trust, Levanger; Department of Pediatrics, St. Olavs Hospital, Trondheim; Department of Oral and Craniomaxillofacial Surgery, St. Olavs Hospital, Trondheim, Norway; Department of Pediatrics, Ryhov County Hospital, Jönköping; Department of Women's and Children's Health, Uppsala University, Uppsala; Department of Pediatrics, Sahlgrenska Academy, University of Gothenburg, Gothenburg; Department of Oral and Maxillofacial Surgery/Stomatognathic Physiology, The Institute for Postgraduate Dental Education, Jönköping, Sweden; Hospital for Children and Adolescents, University of Helsinki, Helsinki; Orthodontics, Oral and Maxillofacial Diseases, University of Helsinki and Helsinki University Hospital, Helsinki, Finland; Department of Pediatrics, Copenhagen University Hospital, Copenhagen; Department of Pediatric Dentistry and Clinical Genetics, University of Copenhagen, Copenhagen; Department of Oral and Maxillofacial Surgery, Aarhus University Hospital, Aarhus, Denmark.

Objective: To determine the prevalence of orofacial symptoms, dysfunctions, and deformities of the temporomandibular joint (TMJ) in juvenile idiopathic arthritis (JIA) 17 years after disease onset.

Methods: Drawn from a prospective, population-based Nordic JIA cohort with disease onset from 1997 to 2000, 420 consecutive cases were eligible for orofacial evaluation of TMJ involvement. The followup visit included demographic data, a standardized clinical orofacial examination, and full-face cone-beam computed tomography (CBCT). For comparison, 200 age-matched healthy controls were used.

Results: Of 420 eligible participants with JIA, 265 (63%) were included (mean age 23.5 ± 4.2 yrs) and completed a standardized clinical orofacial examination. Of these, 245 had a full-face CBCT performed. At least 1 orofacial symptom was reported by 33%. Compared to controls, the JIA group significantly more often reported TMJ pain, TMJ morning stiffness, and limitation on chewing. Further, among participants reporting complaints, the number of symptoms was also higher in JIA. The mean maximal incisal opening was lower in the JIA group (p < 0.001), and TMJ pain on palpation was more frequent. Condylar deformities and/or erosions were observed in 61% as assessed by CBCT, showing bilateral changes in about 70%. Risk factors of condylar deformities were orofacial dysfunction or biologic treatment; enthesitis-related arthritis was protective.

Conclusion: This study of the longterm consequences of TMJ involvement in a population-based JIA cohort reports persistence of comprehensive symptoms, dysfunctions, and damage of the TMJ into adulthood. We suggest interdisciplinary followup of JIA patients also in adulthood.
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http://dx.doi.org/10.3899/jrheum.190231DOI Listing
May 2020

Growth and Puberty in Juvenile Dermatomyositis: A Longitudinal Cohort Study.

Arthritis Care Res (Hoboken) 2020 Feb;72(2):265-273

IRCCS Istituto Giannina Gaslini, Paediatric Rheumatology International Trials Organisation, Genoa, Italy.

Objective: To study growth and puberty in a multinational longitudinal prospective cohort of children with juvenile dermatomyositis (DM).

Methods: Children from 31 countries who were ages <18 years and had juvenile DM in active phase were studied, and analyses of height, weight, and pubertal development were conducted in those who had follow-up visits during a 2-year period and for whom anthropometric data was available.

Results: A total of 196 of 275 children (71%) were included. We found a significant reduction in parent-adjusted height Z score over time in female patients (P < 0.0001) and male patients (P = 0.001), but with catch-up growth at the final study visit. Median body mass index Z score peaked at 6 months (P < 0.0001) and was still significantly above baseline at the final study visit, which was at a median of 26 months after baseline (P = 0.007), with no difference between sexes. Female patients with a disease duration ≥12 months after onset had significantly lower parent-adjusted height Z score (P = 0.002) and no 2-year catch-up growth. At the final study visit, growth failure was seen in 20 of 97 female patients (21%) and in 11 of 73 male patients (15%). Height deflection (∆height Z score less than -0.25/year) was observed in 29 of 116 female patients (25%) and 25 of 80 male patients (31.3%). Delayed puberty was seen in 20 of 55 female patients (36.4%) and in 11 of 31 male patients (35.5%). Children in early pubertal stage at baseline had the highest risk of growth failure.

Conclusion: Juvenile DM in the active phase and/or its treatment has a significant impact on growth and puberty in affected children. Children with recent onset of puberty or previous growth failure have the highest risk of delayed pubertal development and further growth retardation.
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http://dx.doi.org/10.1002/acr.24065DOI Listing
February 2020

Complement lectin pathway protein levels reflect disease activity in juvenile idiopathic arthritis: a longitudinal study of the Nordic JIA cohort.

Pediatr Rheumatol Online J 2019 Sep 9;17(1):63. Epub 2019 Sep 9.

Department of Pediatrics, Aarhus University Hospital, Palle Juul-Jensens Blvd. 99, 8200, Aarhus N, Denmark.

Background: To determine the serum levels of the lectin pathway proteins early in the disease course and 17 years after disease onset and to correlate the protein levels to markers of disease activity in participants from a population-based Nordic juvenile idiopathic arthritis (JIA) cohort. Additionally, to assess the predictive value of lectin pathway proteins with respect to remission status.

Methods: A population-based cohort study of consecutive cases of JIA with a disease onset from 1997 to 2000 from defined geographical areas of Finland, Sweden, Norway and Denmark with 17 years of follow-up was performed. Clinical characteristics were registered and H-ficolin, M-ficolin, MASP-1, MASP-3, MBL and CL-K1 levels in serum were analyzed.

Results: In total, 293 patients with JIA were included (mean age 23.7 ± 4.4 years; mean follow-up 17.2 ± 1.7 years). Concentrations of the lectin protein levels in serum were higher at baseline compared to the levels 17 years after disease onset (p ≤ 0.006, n = 164). At baseline, the highest level of M-ficolin was observed in systemic JIA. Further, high M-ficolin levels at baseline and at 17-year follow-up were correlated to high levels of ESR. In contrast, high MASP-1 and MASP-3 tended to correlate to low ESR. CL-K1 showed a negative correlation to JADAS71 at baseline. None of the protein levels had prognostic abilities for remission status 17 years after disease onset.

Conclusion: We hypothesize that increased serum M-ficolin levels are associated with higher disease activity in JIA and further, the results indicate that MASP-1, MASP-3 and CL-K1 are markers of inflammation.
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http://dx.doi.org/10.1186/s12969-019-0367-9DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6734250PMC
September 2019

Participation in school and physical education in juvenile idiopathic arthritis in a Nordic long-term cohort study.

Pediatr Rheumatol Online J 2019 Jul 15;17(1):44. Epub 2019 Jul 15.

Department of Clinical and Molecular Medicine, NTNU - Norwegian University of Science and Technology, Trondheim, Norway.

Background: The aim of the study was to describe school attendance and participation in physical education in school among children with juvenile idiopathic arthritis (JIA).

Methods: Consecutive cases of JIA from defined geographical areas of Finland, Sweden and Norway with disease onset in 1997 to 2000 were followed for 8 years in a multi-center cohort study, aimed to be as close to population-based as possible. Clinical characteristics and information on school attendance and participation in physical education (PE) were registered.

Results: Participation in school and in PE was lowest initially and increased during the disease course. Eight years after disease onset 228/274 (83.2%) of the children reported no school absence due to JIA, while 16.8% reported absence during the last 2 months due to JIA. Full participation in PE was reported by 194/242 (80.2%), partly by 16.9%, and none by 2.9%. Lowest participation in PE was found among children with ERA and the undifferentiated categories. Absence in school and PE was associated with higher disease activity measures at the 8-year visit. School absence > 1 day at baseline predicted use of disease-modifying anti-rheumatic drugs, including biologics (DMARDs) (OR 1.2 (1.1-1.5)), and non-remission off medication (OR 1.4 (1.1-1.7) 8 years after disease onset.

Conclusion: School absence at baseline predicted adverse long-term outcome. In children and adolescents with JIA participation in school activities is mostly high after 8 years of disease. For the minority with low participation, special attention is warranted to promote their full potential of social interaction and improve long-term outcome.
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http://dx.doi.org/10.1186/s12969-019-0341-6DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6631827PMC
July 2019

Long-Term Outcomes in Juvenile Idiopathic Arthritis: Eighteen Years of Follow-Up in the Population-Based Nordic Juvenile Idiopathic Arthritis Cohort.

Arthritis Care Res (Hoboken) 2020 04;72(4):507-516

Aarhus University Hospital, Aarhus, Denmark.

Objective: The present study was undertaken to assess the long-term course, remission rate, and disease burden in juvenile idiopathic arthritis (JIA) 18 years after disease onset in a population-based setting from the early biologic era.

Methods: A total of 510 consecutive cases of JIA with disease onset between 1997 and 2000 from defined geographic regions in Denmark, Norway, Sweden, and Finland were prospectively included in this 18-year cohort study. At the follow-up visit, patient-reported demographic and clinical data were collected.

Results: The study included 434 (85%) of the 510 eligible JIA participants. The mean ± SD age was 24.0 ± 4.4 years. The median juvenile arthritis disease activity score in 71 joints (JADAS-71) was 1.5 (interquartile range [IQR] 0-5), with the enthesitis-related arthritis (ERA) category of JIA having the highest median score (4.5 [IQR 1.5-8.5], P = 0.003). In this cohort, 46% of patients still had active disease, and 66 (15%) were treated with synthetic disease-modifying antirheumatic drugs and 84 (19%) with biologics. Inactive disease indicated by a JADAS-71 score of <1 was seen in 48% of participants. Clinical remission off medication (CR) was documented in 33% of the participants with high variability among the JIA categories. CR was most often seen in persistent oligoarticular and systemic arthritis and least often in ERA (P < 0.001).

Conclusion: A substantial proportion of the JIA cohort did not achieve CR despite new treatment options during the study period. The ERA category showed the worst outcomes, and in general there is still a high burden of disease in adulthood for JIA.
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http://dx.doi.org/10.1002/acr.23853DOI Listing
April 2020

Pulmonary Manifestations and Progression of Lung Disease in Juvenile-onset Mixed Connective Tissue Disease.

J Rheumatol 2019 01 1;46(1):93-100. Epub 2018 Aug 1.

From the Department of Rheumatology, the Department of Radiology and Nuclear Medicine, and the Department of Respiratory Medicine, Oslo University Hospital, Rikshospitalet; Faculty of Medicine, Institute of Clinical Medicine, University of Oslo, Oslo; Department of Pediatrics, University Hospital of North Norway; Institute of Clinical Medicine, University of Tromsø, Tromsø; Department of Pediatrics, St. Olavs Hospital; Institute of Laboratory Medicine, Children's and Women's Health, Faculty of Medicine, Norwegian University of Science and Technology, Trondheim, Norway.

Objective: To assess the occurrence and extent of interstitial lung disease (ILD) in patients with juvenile mixed connective tissue disease (JMCTD), compare pulmonary function in patients and matched controls, study associations between ILD and disease-related variables, and examine progression of pulmonary manifestations over time.

Methods: A cohort of 52 patients with JMCTD were examined in a cross-sectional study after a mean 16.2 (SD 10.3) years of disease duration with high-resolution computed tomography (HRCT) and pulmonary function tests (PFT) comprising spirometry, DLCO, and total lung capacity (TLC). Matched controls were examined with PFT. Previous HRCT and PFT were available in 37 and 38 patients (mean 8.8 and 10.3 yrs before study inclusion), respectively.

Results: Compared to controls, patients with JMCTD had lower forced vital capacity (FVC), DLCO, and TLC (p < 0.01). The most frequent abnormal PFT was DLCO in 67% of patients versus 17% of controls (p < 0.001). Fourteen patients (27%) had ILD on HRCT. Most had ILD in < 10% of their lungs. ILD was associated with low values for FVC and TLC, but not with DLCO. HRCT findings did not progress significantly over time, but FVC declined (p < 0.01).

Conclusion: Compared to controls, patients with JMCTD had impaired pulmonary function. ILD was present in 27% of patients after a mean 16 years of disease duration, mostly as mild disease, and did not progress. ILD seems to be less common in juvenile-onset than in adult-onset MCTD, and ILD in JMCTD seems mostly mild and stable over time.
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http://dx.doi.org/10.3899/jrheum.180019DOI Listing
January 2019

Early Self-Reported Pain in Juvenile Idiopathic Arthritis as Related to Long-Term Outcomes: Results From the Nordic Juvenile Idiopathic Arthritis Cohort Study.

Arthritis Care Res (Hoboken) 2019 07 12;71(7):961-969. Epub 2019 Jun 12.

Norwegian University of Science and Technology and St. Olavs Hospital, Trondheim, Norway.

Objective: To study self-reported pain early in the disease course of juvenile idiopathic arthritis (JIA) as a predictor of long-term disease outcomes.

Methods: Consecutive cases of JIA with disease onset from 1997 to 2000 from defined geographical areas of Norway, Sweden, Finland, and Denmark were prospectively enrolled in this population-based cohort study. Self-reported, disease-related pain was measured on a 10-cm visual analog scale (VAS pain). Inclusion criteria were a baseline visit with a pain score 6 months after disease onset, followed by an 8-year study visit. Remission was defined according to Wallace et al (2004) preliminary criteria. Functional disability was measured by the Childhood Health Assessment Questionnaire and the Child Health Questionnaire Parent Form if the child was age <18 years and by the Health Assessment Questionnaire if age ≥18 years. Damage was scored using the Juvenile Arthritis Damage Index.

Results: The final study cohort consisted of 243 participants, and 120 participants (49%) had oligoarticular onset. At baseline, 76% reported a VAS pain score >0 compared to 57% reporting at 8 years. Half of those who reported baseline pain also reported pain at 8 years but at a lower intensity. Compared to no pain, higher pain intensity at baseline predicted more pain at 8 years, more functional disability, more damage, and less remission without medication. Baseline pain predicted more use of disease-modifying antirheumatic drugs/biologics during the disease course. Participants with oligoarticular JIA reporting pain at baseline were more likely to develop extended oligoarticular JIA or other JIA categories with an unfavorable prognosis.

Conclusion: Early self-reported, disease-related pain among children and adolescents with JIA is common and seems to predict persistent pain and unfavorable long-term disease outcomes.
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http://dx.doi.org/10.1002/acr.23715DOI Listing
July 2019

Predicting unfavorable long-term outcome in juvenile idiopathic arthritis: results from the Nordic cohort study.

Arthritis Res Ther 2018 05 3;20(1):91. Epub 2018 May 3.

Department of Pediatrics, University Hospital of North Norway, Tromsø, Norway.

Background: The aim was to develop prediction rules that may guide early treatment decisions based on baseline clinical predictors of long-term unfavorable outcome in juvenile idiopathic arthritis (JIA).

Methods: In the Nordic JIA cohort, we assessed baseline disease characteristics as predictors of the following outcomes 8 years after disease onset. Non-achievement of remission off medication according to the preliminary Wallace criteria, functional disability assessed by Childhood Health Assessment Questionnaire (CHAQ) and Physical Summary Score (PhS) of the Child Health Questionnaire, and articular damage assessed by the Juvenile Arthritis Damage Index-Articular (JADI-A). Multivariable models were constructed, and cross-validations were performed by repeated partitioning of the cohort into training sets for developing prediction models and validation sets to test predictive ability.

Results: The total cohort constituted 423 children. Remission status was available in 410 children: 244 (59.5%) of these did not achieve remission off medication at the final study visit. Functional disability was present in 111/340 (32.7%) children assessed by CHAQ and 40/199 (20.1%) by PhS, and joint damage was found in 29/216 (13.4%). Model performance was acceptable for making predictions of long-term outcome. In validation sets, the area under the curves (AUCs) in the receiver operating characteristic (ROC) curves were 0.78 (IQR 0.72-0.82) for non-achievement of remission off medication, 0.73 (IQR 0.67-0.76) for functional disability assessed by CHAQ, 0.74 (IQR 0.65-0.80) for functional disability assessed by PhS, and 0.73 (IQR 0.63-0.76) for joint damage using JADI-A.

Conclusion: The feasibility of making long-term predictions of JIA outcome based on early clinical assessment is demonstrated. The prediction models have acceptable precision and require only readily available baseline variables. Further testing in other cohorts is warranted.
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http://dx.doi.org/10.1186/s13075-018-1571-6DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5934822PMC
May 2018

The Norwegian version of the Juvenile Arthritis Multidimensional Assessment Report (JAMAR).

Rheumatol Int 2018 Apr 7;38(Suppl 1):291-298. Epub 2018 Apr 7.

Clinica Pediatrica e Reumatologia, Paediatric Rheumatology International Trials Organisation (PRINTO), Istituto Giannina Gaslini, Via Gaslini 5, 16147, Genoa, Italy.

The Juvenile Arthritis Multidimensional Assessment Report (JAMAR) is a new parent/patient-reported outcome measure that enables a thorough assessment of the disease status in children with juvenile idiopathic arthritis (JIA). We report the results of the cross-cultural adaptation and validation of the parent and patient versions of the JAMAR in the Norwegian language. The reading comprehension of the questionnaire was tested in 10 JIA parents and patients. Each participating centre was asked to collect demographic and clinical data and the JAMAR in 100 consecutive JIA patients or all consecutive patients seen in a 6-month period and to administer the JAMAR to 100 healthy children and their parents. The statistical validation phase explored descriptive statistics and the psychometric issues of the JAMAR: the 3 Likert assumptions, floor/ceiling effects, internal consistency, Cronbach's alpha, interscale correlations, test-retest reliability, and construct validity (convergent and discriminant validity). A total of 301 JIA patients (3.3% systemic, 41.2% oligoarticular, 25.9% RF negative polyarthritis, and 29.6% other categories) and 74 healthy children were enrolled in three centres. The JAMAR components discriminated well healthy subjects from JIA patients. All JAMAR components revealed good psychometric performances. In conclusion, the Norwegian version of the JAMAR is a valid tool for the assessment of children with JIA and is suitable for use both in routine clinical practice and clinical research.
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http://dx.doi.org/10.1007/s00296-018-3963-yDOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5893693PMC
April 2018

Pain Sensitivity and Thermal Detection Thresholds in Young Adults Born Preterm With Very Low Birth Weight or Small for Gestational Age at Term Compared With Controls.

J Pain 2018 08 21;19(8):873-884. Epub 2018 Mar 21.

Department of Clinical and Molecular Medicine, Faculty of Medicine and Health Sciences, NTNU-Norwegian University of Science and Technology, Trondheim, Norway; Department of Pediatrics, St. Olavs Hospital, Trondheim University Hospital, Trondheim, Norway.

The objective of this prospective long-term follow-up study was to investigate whether somatosensory function is altered among young adults born preterm with very low birth weight (VLBW; ≤1,500 g) or small for gestational age (SGA; <10th percentile) at term. In a blinded quantitative sensory testing protocol, we determined thermal detection, thermal pain, and pressure pain thresholds and the response to prolonged supra-threshold heat among 51 VLBW, 66 term SGA, and 86 term-born controls (birth weight ≥10th percentile) at 28 years. Self-reported chronic pain was also investigated. Except for increased sensitivity to cool in the term SGA group versus controls, we found no significant group differences regarding thermal or pain thresholds. Overall, male participants had higher pain thresholds, and no significant interactions of group and sex were observed (P > .14). Within the VLBW group, neonatal mechanical ventilation was associated with reduced sensitivity to cool, and length of mechanical ventilation correlated with lower pressure pain thresholds. The response to prolonged supra-threshold heat was similar between the groups, and the prevalence of self-reported chronic pain was not reliably different. In conclusion, low birth weight young adults were as sensitive to thermal and pain stimuli as term-born, normal birth weight controls, with the same sex differences.

Perspective: To our knowledge, this is the first report on thermal and pain sensitivity among young adults born preterm with VLBW or SGA at term. The negative results from a comprehensive quantitative sensory testing protocol oppose previous findings of altered sensory perception among children and adolescents born preterm.
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http://dx.doi.org/10.1016/j.jpain.2018.03.001DOI Listing
August 2018

Brief Report: The Genetic Profile of Rheumatoid Factor-Positive Polyarticular Juvenile Idiopathic Arthritis Resembles That of Adult Rheumatoid Arthritis.

Arthritis Rheumatol 2018 06 21;70(6):957-962. Epub 2018 Apr 21.

Emory University School of Medicine and Children's Healthcare of Atlanta, Atlanta, Georgia.

Objective: Juvenile idiopathic arthritis (JIA) comprises 7 heterogeneous categories of chronic childhood arthritides. Approximately 5% of children with JIA have rheumatoid factor (RF)-positive arthritis, which phenotypically resembles adult rheumatoid arthritis (RA). Our objective was to compare and contrast the genetics of RF-positive polyarticular JIA with those of RA and selected other JIA categories, to more fully understand the pathophysiologic relationships of inflammatory arthropathies.

Methods: Patients with RF-positive polyarticular JIA (n = 340) and controls (n = 14,412) were genotyped using the Immunochip array. Single-nucleotide polymorphisms were tested for association using a logistic regression model adjusting for admixture proportions. We calculated weighted genetic risk scores (wGRS) of reported RA and JIA risk loci, and we compared the ability of these wGRS to predict RF-positive polyarticular JIA.

Results: As expected, the HLA region was strongly associated with RF-positive polyarticular JIA (P = 5.51 × 10 ). Nineteen of 44 RA risk loci and 6 of 27 oligoarticular/RF-negative polyarticular JIA risk loci were associated with RF-positive polyarticular JIA (P < 0.05). The RA wGRS predicted RF-positive polyarticular JIA (area under the curve [AUC] 0.71) better than did the oligoarticular/RF-negative polyarticular JIA wGRS (AUC 0.59). The genetic profile of patients with RF-positive polyarticular JIA was more similar to that of RA patients with age at onset 16-29 years than to that of RA patients with age at onset ≥70 years.

Conclusion: RF-positive polyarticular JIA is genetically more similar to adult RA than to the most common JIA categories and thus appears to be a childhood-onset presentation of autoantibody-positive RA. These findings suggest common disease mechanisms, which could lead to novel therapeutic targets and shared treatment strategies.
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http://dx.doi.org/10.1002/art.40443DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5984672PMC
June 2018

Normal magnetic resonance appearances of the temporomandibular joints in children and young adults aged 2-18 years.

Pediatr Radiol 2018 03 12;48(3):341-349. Epub 2017 Dec 12.

Department of Clinical Medicine, University of Bergen, Bergen, Norway.

Background: Knowledge of normal appearances of the temporomandibular joint (TMJ) is paramount when assessing the joint for disease in juvenile idiopathic arthritis. Reliable features defining normal TMJs in children are limited.

Objective: To establish reliable normal standards for the TMJ at magnetic resonance imaging (MRI).

Materials And Methods: We included children and young adults aged 2-18 years undergoing a head MRI for reasons not believed to affect the TMJs. We assessed TMJ anatomy and contrast enhancement using a high-resolution 3-D T1-weighted sequence. We noted joint fluid and bone marrow oedema based on a T2-weighted sequence. Three experienced radiologists read all examinations twice in consensus and defined intraobserver consensus agreement.

Results: We evaluated the TMJs in 101 children and young adults (45 female), mean age 10.7 years (range 2-18 years). The intraobserver consensus agreement for the assessment of anterior condylar inclination in the sagittal/oblique plane was moderate to good (Cohen κ=0.7 for the right side). Cohen κ for intraobserver consensus agreement for condylar shape in the coronal plane on a 0-2 scale was 0.4 for the right and 0.6 for the left. Intraobserver agreement for measurement of joint space height and assessment of bone marrow oedema was poor. There was a statistically significant increase in anterior inclination by age in the sagittal plane on a 0-2 scale (P<0.0001). Eighty percent of the condyles showed a rounded shape in the coronal plane while 20% showed mild flattening. Thirty-five of 36 right TMJs showed contrast enhancement (mild enhancement in 32 joints, moderate in 3 joints).

Conclusion: Subjective assessment of the anterior condylar inclination in the sagittal/oblique plane and condylar flattening in the coronal plane can be considered precise features for describing TMJ anatomy in healthy children. There is an increasing anterior inclination by age. Mild contrast enhancement of the TMJs should be considered a normal finding.
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http://dx.doi.org/10.1007/s00247-017-4048-xDOI Listing
March 2018

Incidence and predictors of Uveitis in juvenile idiopathic arthritis in a Nordic long-term cohort study.

Pediatr Rheumatol Online J 2017 Aug 18;15(1):66. Epub 2017 Aug 18.

Department of Clinical and Molecular Medicine, NTNU - Norwegian University of Science and Technology, and Department of Pediatrics, St. Olavs Hospital, Trondheim, Norway.

Background: The incidence of uveitis associated with juvenile idiopathic arthritis (JIA) varies around the world. Our aim was to investigate the incidence and predictors of uveitis in a Nordic population-based cohort.

Methods: Consecutive JIA cases from defined geographical areas in Denmark, Finland, Sweden and Norway with disease onset between January 1997 to June 2000 were followed for median 98 months in this prospective longitudinal cohort study. Potential clinical and immunological predictors of uveitis were identified with logistic regression analysis.

Results: Uveitis occurred in 89 (20.5%) of the 435 children with regular ophtalmologic follow-up among the 500 included. Chronic asymptomatic uveitis developed in 80 and acute symptomatic uveitis in 9 children. Uveitis developed at a median interval of 0.8 (range - 4.7 to 9.4) years after onset of arthritis. Predictors of uveitis were age < 7 years at JIA onset (Odds ratio (OR) 2.1, 95% confidence interval (CI) 1.3 to 3.5), presence of antihistone antibodies (AHA) > 15 U/ml (OR 4.8 (1.8 to 13.4)) and antinuclear antibodies (ANA) (OR 2.4 (1.5 to 4.0)). Mean combined IgM/IgG AHA was significantly higher in the uveitis group (19.2 U/ml) than in the non-uveitis group (10.2 U/ml) (p = 0.002). Young age at JIA onset predicted uveitis in girls (p < 0.001), but not in boys (p = 0.390).

Conclusion: Early-onset arthritis and presence of AHA in girls, as well as presence of ANA in both genders, were significant predictors of chronic uveitis. The high incidence of uveitis in this long-term Nordic JIA cohort may have severe implications in a lifelong perspective.
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http://dx.doi.org/10.1186/s12969-017-0195-8DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5562983PMC
August 2017

Relationship between pubertal timing and chronic nonspecific pain in adolescent girls: the Young-HUNT3 study (2006-2008).

Pain 2017 08;158(8):1554-1560

Department of Laboratory Medicine, Children's and Women's Health, Faculty of Medicine and Health Sciences, NTNU - Norwegian University of Science and Technology, Trondheim, Norway.

The aim of this study was to examine a possible relationship between early puberty and chronic nonspecific pain in 13- to 18-year-old girls. All adolescents in Nord-Trøndelag County, Norway, were invited to participate in the Young-HUNT3 study (2006-2008). Of the invited girls, 81% answered the questionnaire and of these 3982 were 13 to 18 years of age. Menarche and perceived physical maturation were used as exposure measures. Early menarche was defined as <12 years, normal menarche as ≥12 and <14 years, and late menarche as ≥14 years. Perceived physical maturation was divided into maturing earlier, the same or later than others of their own age. The main outcome measure was chronic nonspecific pain, defined as pain in at least one location not related to any known disease or injury, for at least once a week during the last 3 months. The median age at menarche was 13.2 years. Chronic nonspecific pain was more prevalent among girls with early menarche (68%, 95% CI: 64%-72%) compared to girls with either normal (55%, 95% CI: 53%-57%), late (50%, 95% CI: 46%-54%), or no menarche (35%, 95% CI: 29%-40%). The association persisted after adjusting for age, body mass index, socioeconomic factors, and anxiety and depression. A similar association was found between girls that perceived themselves as earlier physically matured than their peers and chronic nonspecific pain. Headache/migraine was the most common type of chronic nonspecific pain regardless of menarcheal age. In all reported locations, pain was more prevalent in the group with early menarche compared to normal or late menarche.
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http://dx.doi.org/10.1097/j.pain.0000000000000950DOI Listing
August 2017

High-sensitive CRP as a predictive marker of long-term outcome in juvenile idiopathic arthritis.

Rheumatol Int 2017 May 10;37(5):695-703. Epub 2017 Mar 10.

Department of Pediatrics and Adolescent Medicine, Copenhagen University Hospital, Rigshospitalet, Denmark.

To evaluate whether C-reactive protein (CRP), including variation within the normal range, is predictive of long-term disease outcome in Juvenile Idiopathic Arthritis (JIA). Consecutive patients with newly diagnosed JIA were included prospectively from defined geographic areas of the Nordic countries from 1997 to 2000. Inclusion criteria were availability of a baseline serum sample within 12 months after disease onset and 8-year clinical assessment data. Systemic onset JIA was not included. CRP was measured by high-sensitive ELISA (detection limit of 0.2 mg/l). One hundred and thirty participants with a median follow-up time of 97 months (range 95-100) were included. At follow-up, 38% of the patients were in remission off medication. Absence of remission was associated with elevated level of CRP at baseline (odds ratio (OR) 1.33, confidence interval (CI) 1.08-1.63, p = 0.007). By applying a cutoff at the normal upper limit (>10 mg/l), the risk of not achieving remission was increased to an OR of 8.60 (CI 2.98-24.81, p < 0.001). Variations of CRP within the normal range had no predictive impact on disease activity at follow-up. Baseline levels of ESR were available in 80 patients (61%) and elevated ESR was associated with absence of remission in a multivariable logistic regression analysis (OR 2.32, CI 1.35-4.00, p = 0.002). This results of this study indicate that baseline CRP concentrations above 10 mg/l are predictive of a poor outcome at 8-year follow-up. We could not demonstrate any predictive value of CRP variations within the normal range.
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http://dx.doi.org/10.1007/s00296-017-3657-xDOI Listing
May 2017

Psoriasis and associated variables in classification and outcome of juvenile idiopathic arthritis - an eight-year follow-up study.

Pediatr Rheumatol Online J 2017 Feb 22;15(1):13. Epub 2017 Feb 22.

Department of Women's and Children's Health, Uppsala University, Uppsala, Sweden.

Background: To study the impact of psoriasis and features associated with psoriasis on classification and outcome in a population-based follow-up cohort of children with juvenile idiopathic arthritis (JIA).

Methods: In all, 440 children with JIA were followed for a median of 8 years in a prospective Nordic population-based cohort study. Data for remission was available for 427 of these children. The presence of psoriasis, psoriasis-like rash, dactylitis, nail pitting, enthesitis, tenosynovitis and heredity was assessed in relation to ILAR classification and remission.

Results: Clinical findings associated with psoriasis developed consecutively during the 8-year period. Six of 14 children with psoriasis were not classified as juvenile psoriatic arthritis according to the ILAR criteria at 8 year follow-up. Dactylitis was more common in children with early onset of JIA. After 8 years we found a cumulative median number of eleven arthritic joints in children with psoriasis or psoriasis-like rash compared with six in the rest of the cohort (p = 0.02). Also, the chance for not being in remission after 8 years increased significantly in patients with psoriasis, psoriasis-like rash or at least two of: 1) first-degree heredity for psoriasis or psoriatic arthritis, 2) dactylitis or 3) nail pitting, compared with the rest of the group (OR 3.32, p = 0.010).

Conclusions: Our results indicate a more severe disease over time in psoriasis-associated JIA, as features of psoriasis develop during the disease course. This group is a major challenge to encompass in a future JIA classification in order to facilitate early tailored treatment.
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http://dx.doi.org/10.1186/s12969-017-0145-5DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5320636PMC
February 2017

Temporomandibular Joint Involvement in Association With Quality of Life, Disability, and High Disease Activity in Juvenile Idiopathic Arthritis.

Arthritis Care Res (Hoboken) 2017 05;69(5):677-686

Istituto Giannina Gaslini and PRINTO Coordinating Center, Genoa, Italy.

Objective: To evaluate the demographic, disease activity, disability, and health-related quality of life (HRQOL) differences between children with juvenile idiopathic arthritis (JIA) and their healthy peers, and between children with JIA with and without clinical temporomandibular joint (TMJ) involvement and its determinants.

Methods: This study is based on a cross-sectional cohort of 3,343 children with JIA and 3,409 healthy peers, enrolled in the Pediatric Rheumatology International Trials Organisation HRQOL study or in the methotrexate trial. Potential determinants of TMJ involvement included demographic, disease activity, disability, and HRQOL measures selected through univariate and multivariable logistic regression.

Results: Clinical TMJ involvement was observed in 387 of 3,343 children with JIA (11.6%). Children with TMJ involvement, compared to those without, more often had polyarticular disease course (95% versus 70%), higher Juvenile Arthritis Disease Activity Score (odds ratio [OR] 4.6), more disability, and lower HRQOL. Children with TMJ involvement experienced clearly more disability and lower HRQOL compared to their healthy peers. The multivariable analysis showed that cervical spine involvement (OR 4.6), disease duration >4.4 years (OR 2.8), and having more disability (Childhood Health Assessment Questionnaire Disability Index >0.625) (OR 1.6) were the most important determinants for TMJ involvement.

Conclusion: Clinical TMJ involvement in JIA is associated with higher disease activity, higher disability, and impaired HRQOL. Our findings indicate the need for dedicated clinical and imaging evaluation of TMJ arthritis, especially in children with cervical spine involvement, polyarticular course, and longer disease duration.
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http://dx.doi.org/10.1002/acr.23003DOI Listing
May 2017

Self-reported Chronic Pain in Young Adults With a Low Birth Weight.

Clin J Pain 2017 04;33(4):348-355

Departments of *Laboratory Medicine, Children's and Women's Health §Public Health and General Practice, Faculty of Medicine †Regional Centre for Child and Youth Mental Health and Child Welfare, Norwegian University of Science and Technology ‡Department of Child and Adolescent Psychiatry, St. Olavs Hospital, Trondheim University Hospital ∥Department of Physiotherapy, Trondheim Municipality ¶Department of Pediatrics, St. Olavs Hospital, Trondheim University Hospital, Trondheim, Norway.

Objective: To investigate self-reported pain in young adults with a low birth weight.

Materials And Methods: This study was a part of a long-term follow-up study of preterm very low birth weight (VLBW; birth weight ≤1500 g), term small for gestational age (SGA; birth weight <10th percentile adjusted for sex and parity), and control young adults born during 1986 to 1988. Of the 300 individuals invited, 216 (62 VLBW, 67 term SGA, and 87 controls) completed a pain questionnaire. Of these, 151 (70%) had answered a pain severity question at 19 years. Chronic pain was defined as pain lasting for >6 months and being moderate, severe, or very severe during the past 4 weeks.

Results: The prevalence of chronic pain at 26 years was 16% in the VLBW group, 21% in the term SGA group, and 7% in the control group. The VLBW and the term SGA groups had higher odds ratios for chronic pain (crude OR, 2.6; 95% CI, 0.9-7.6 for the VLBW group and crude OR, 3.6; 95% CI, 1.3-9.9 for the term SGA group vs. controls). The main results remained after adjusting for potential confounding factors. Some attenuation was observed when adjusting for anxiety and depressive problems. Moderate to very severe pain increased from 16% to 41% in the term SGA group from 19 to 26 years, whereas less changes were seen in the VLBW and the control groups.

Discussion: Results of our study imply that pain should be in focus when conducting long-term follow-up programs of individuals with a low birth weight.
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http://dx.doi.org/10.1097/AJP.0000000000000399DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5348108PMC
April 2017

Long-term outcome in juvenile-onset mixed connective tissue disease: a nationwide Norwegian study.

Ann Rheum Dis 2017 Jan 9;76(1):159-165. Epub 2016 Jun 9.

Department of Rheumatology, Oslo University Hospital, Rikshospitalet, Oslo, Norway.

Objectives: To describe the characteristics, outcome and predictive factors of juvenile mixed connective tissue disease (JMCTD) in a nationwide cohort of patients.

Methods: We examined 55 patients with JMCTD after a mean disease duration of 16.2 years (SD 10.0). Patients were registered according to Kasukawa's criteria. Remission criteria were defined according to those for juvenile idiopathic arthritis, plus absence of cytopenia, myositis, progressive sclerodactyly, lung and oesophageal manifestations. Organ damage was assessed with the Systemic Lupus International Collaborating Clinics (SLICC)/American College of Rheumatology (ACR) Damage Index and the Juvenile Arthritis Damage Index (JADI). Medical records were reviewed for early predictors for outcome, which were assessed by multivariate logistic regression analyses.

Results: Three patients developed systemic lupus erythematosus (SLE). Fifty-two patients had continuous JMCTD; the most common manifestations were: Raynaud (100%), arthritis (94%), puffy hands (77%) and pulmonary manifestations (58%). SLE-like, systemic sclerosis (SSc)-like and polymyositis (PM)-like findings were found in 98%, 77% and 48%, respectively. Over time, SLE-like and PM-like manifestations decreased, and SSc-like findings increased. At follow-up, 35 patients (67%) had active disease and 17 (33%) were in remission. In 34 patients (65%), SLICC or JADI≥1 assessments indicated organ damage. Active disease was associated with higher anti-ribonucleoprotein antibody titres at follow-up and positive rheumatoid factor (RF) at diagnosis and follow-up.

Conclusions: Most patients with JMCTD had active disease and organ damage after a mean follow-up of 16.2 years. Active disease was associated with higher anti-ribonucleoprotein antibody levels and positive RF. The presence of RF at diagnosis predicted persistent disease activity.
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http://dx.doi.org/10.1136/annrheumdis-2016-209522DOI Listing
January 2017

Ankle arthritis predicts polyarticular disease course and unfavourable outcome in children with juvenile idiopathic arthritis.

Clin Exp Rheumatol 2015 Sep-Oct;33(5):751-7. Epub 2015 Jul 23.

Department of Women's and Children's Health, Uppsala University, Uppsala, Sweden.

Objectives: To evaluate the occurrence, clinical characteristics and prognostic factors associated with ankle arthritis in children with juvenile idiopathic arthritis (JIA).

Methods: 440 children with JIA were followed for eight years in a prospective Nordic population-based cohort study. Data on remission was available for 427 of these children. Occurrence of clinically assessed ankle arthritis was analysed in relation to JIA category, clinical characteristics and remission data eight years after disease onset.

Results: In 440 children with JIA, 251 (57%) experienced ankle arthritis during the first eight years of disease. Ankle arthritis was least common in the persistent oligoarticular category (25%) and most common in children with extended oligoarticular (83%) and polyarticular RF-negative (85%) JIA. Children who developed ankle arthritis during the first year of disease were younger at disease onset (median age 4.9 (IQR 2.1-8.8) vs. 6.6 (IQR 2.8-10.1) years, p<0.003) and had more cumulative affected joints at 8-year follow-up (median involved joints 10 (IQR 6-16) vs. 3 (IQR 2-9), p<0.001). The odds ratio for not achieving remission eight years after disease onset, if the ankle joint was involved during the first year of disease was 2.0 (95 % CI:1.3-3.0, p<0.001). Hind-, mid- and forefoot involvements were more common compared to patients without ankle arthritis.

Conclusions: In this Nordic population-based 8-year follow-up study, occurrence of ankle arthritis during the first year was associated with an unfavourable disease outcome. We suggest that ankle arthritis should be recognised in the assessment of prognosis and choice of treatment strategy in JIA.
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December 2015

Anti-type II collagen antibodies, anti-CCP, IgA RF and IgM RF are associated with joint damage, assessed eight years after onset of juvenile idiopathic arthritis (JIA).

Pediatr Rheumatol Online J 2014 11;12:22. Epub 2014 Jun 11.

Department of Immunology, Genetics and Pathology, Uppsala University, Uppsala, Sweden.

Background: Early appearance of antibodies specific for native human type II collagen (anti-CII) characterizes an early inflammatory and destructive phenotype in adults with rheumatoid arthritis (RA). The objective of this study was to investigate the occurrence of anti-CII, IgM RF, IgA RF and anti-CCP in serum samples obtained early after diagnosis, and to relate the occurrence of autoantibodies to outcome after eight years of disease in children with juvenile idiopathic arthritis (JIA).

Methods: The Nordic JIA database prospectively included JIA patients followed for eight years with data on remission and joint damage. From this database, serum samples collected from 192 patients, at a median of four months after disease onset, were analysed for IgG anti-CII, IgM RF, IgA RF and IgG anti-CCP. Joint damage was assessed based on Juvenile Arthritis Damage Index for Articular damage (JADI-A), a validated clinical instrument for joint damage.

Results: Elevated serum levels of anti-CII occurred in 3.1%, IgM RF in 3.6%, IgA RF in 3.1% and anti-CCP in 2.6% of the patients. Occurrence of RF and anti-CCP did to some extent overlap, but rarely with anti-CII. The polyarticular and oligoarticular extended categories were overrepresented in patients with two or more autoantibodies. Anti-CII occurred in younger children, usually without overlap with the other autoantibodies and was associated with high levels of C-reactive protein (CRP) early in the disease course. All four autoantibodies were significantly associated with joint damage, but not with active disease at the eight-year follow up.

Conclusions: Anti-CII, anti-CCP, IgA RF and IgM RF detected early in the disease course predicted joint damage when assessed after eight years of disease. The role of anti-CII in JIA should be further studied.
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http://dx.doi.org/10.1186/1546-0096-12-22DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC4061516PMC
November 2015

Amyloid arthropathy associated with multiple myeloma: a systematic analysis of 101 reported cases.

Semin Arthritis Rheum 2013 Dec 28;43(3):405-12. Epub 2013 Aug 28.

Department of Rheumatology and Rehabilitation, Sohag University, Sohag, Egypt.

Objective: Amyloid deposition in multiple myeloma (MM) may lead to an arthropathy resembling rheumatoid arthritis (RA). Since a systematic description of its natural history is lacking, we have performed a systematic analysis of all published cases.

Methods: Literature review featuring backward and forward database searches and direct inspection of reference lists. Inclusion criteria were as follows: publication between 1931 and 2012, diagnosis of multiple myeloma, and demonstration of light chain amyloid (AL) in any organ or in synovial fluid, arthritis, or synovitis.

Results: Overall, 101 cases were identified. Median age was 59 years and the male-to-female ratio was 1:1. A systemic manifestation of MM was reported in 88 cases. In 53 of these, characteristic physical findings (carpal tunnel syndrome, macroglossia, shoulder pad, and soft tissue swelling/masses) were present. Arthritis manifested before the diagnosis of MM in 63 cases, with 33 cases initially misdiagnosed as RA. There were 72 cases of poly-, 17 of oligo-, and three of monoarthritis. The shoulder joint was most commonly affected, followed by knees and small hand joints. Median synovial fluid leukocyte count was 2460 cells/mm(3), and was normal in seven cases. Synovial histopathology often featured mild synovitis without plasma cell infiltration. Imaging revealed articular or periarticular inflammation in many cases and bone lesions near 22% of affected joints. Treatments varied but led to some improvement in the majority of cases.

Conclusions: These results solidify previous experience that MM arthropathy tends to feature a symmetric RF-negative nonerosive polyarthritis. However, the results also highlight the diversity of its presentations and stress the importance of arthropathy as a potentially under-recognized presenting manifestation of MM.
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http://dx.doi.org/10.1016/j.semarthrit.2013.07.004DOI Listing
December 2013