Helga Sanner

• Dåstøl, Vilde Øverlien; Caspersen, Ida Henriette; Haftorn, Kristine Løkås; Hestetun, Sigrid Valen; Bastakoti, Srijana & Andreassen, Ole [Vis alle 11 forfattere av denne artikkelen] (2026). Maternal weight and hypertension as risk factors for juvenile idiopathic arthritis: a prospective population-based pregnancy cohort study. RMD Open. 12(3). doi: 10.1136/rmdopen-2026-006940. Fulltekst i vitenarkiv Vis sammendrag

  Objectives: The intrauterine environment may influence childhood immune-mediated disease risk. We investigated whether maternal prepregnancy body mass index (BMI), gestational weight gain (GWG) or hypertensive disorders during pregnancy (HDP) were associated with juvenile idiopathic arthritis (JIA) in children and whether genetic susceptibility modified associations. Methods: We linked the Norwegian Mother, Father and Child Cohort Study (MoBa) to the Norwegian Patient Registry to identify children with JIA. Logistic regression estimated adjusted ORs (aORs) for associations with BMI, GWG and HDPs. Causal mediation analyses assessed whether hypertension mediated the GWG–JIA association. In a genotyped subsample, we tested interactions between exposures and a JIA Polygenic Risk Score (PRS). Results: Among 78 901 eligible children, 265 developed JIA (genotyped subsample: 192 JIA/44 053 non-JIA). By the end of follow-up, all children were ≥14 years old and 85% were ≥16 years old. Maternal prepregnancy BMI was not associated with JIA. Each 1 SD (~6 kg) increase in GWG was associated with modestly higher odds of JIA (aOR 1.12, 95% CI 1.00 to 1.26), particularly among women with prepregnancy obesity (aOR 1.40, 95% CI 1.10 to 1.77). Maternal hypertension and pre-eclampsia were associated with higher JIA risk (aOR 1.43, 95% CI 1.02 to 1.99 and aOR 1.61, 95% CI 1.10 to 2.37). Hypertension did not mediate the GWG–JIA association (p>0.05). Among children with lower PRS, maternal obesity was associated with a lower JIA risk (aOR 0.37, 95% CI 0.17 to 0.80). Conclusion: Higher GWG and HDPs were independently associated with increased JIA risk. Genetic susceptibility may modify associations with maternal BMI, supporting a role for pregnancy health in JIA development.

• Nilsson, Ulrika Ann-Charlotte; Skattør, Marianne; Sanner, Helga & Risum, Kristine (2026). Can fitness testing promote physical activity in adolescents with juvenile idiopathic arthritis? Pediatric Rheumatology. 24(1). doi: 10.1186/s12969-026-01223-6. Fulltekst i vitenarkiv Vis sammendrag

  Abstract: Background Physical activity (PA) is important for disease management and overall health in adolescents with juvenile idiopathic arthritis (JIA). In clinical practice, identifying low cardiorespiratory fitness (CRF) and encouraging higher-intensity PA are priorities. We investigated whether introducing submaximal treadmill fitness testing could increase PA, estimated peak oxygen uptake (VO 2peak ), walking distance, physical function, and health-related quality of life (HRQoL) in adolescents with JIA, and examined associations between PA, VO 2peak , and walking distance. Methods: In this exploratory pre–post study, adolescents aged 10–18 years with non-systemic JIA were consecutively recruited during routine physiotherapy visits at a rheumatology clinic. A submaximal treadmill test was used to estimate VO 2peak at baseline and follow-up. Self-reported PA was assessed with the International Physical Activity Questionnaire Short-Form (IPAQ-SF), and physical function and HRQoL with the Juvenile Arthritis Multidimensional Assessment Report (JAMAR). Within-participant changes were analysed with paired tests; associations were assessed using Spearman’s rank correlation. Results: Thirty-four participants were included; 24 completed follow-up. IPAQ-derived PA did not change significantly between assessments. Estimated VO 2peak was higher at follow-up by 2.4 (SD 4.4) mL∙kg − 1 ∙min − 1 ( P  = 0.01) and walking distance was higher by 31 (69) metres ( P  = 0.04), whereas no significant changes were observed in JAMAR physical function or HRQoL. Vigorous PA showed the strongest associations with estimated VO 2peak at baseline and follow-up (rho up to 0.51; P  < 0.05). Associations of moderate-to-vigorous and total PA with VO 2peak were weaker and evident only at follow-up (rho ~ 0.41–0.43), and vigorous PA correlated with walking distance at follow-up (rho = 0.42; P  = 0.040); other associations were low or non-significant. Conclusions: Introducing submaximal treadmill fitness testing in routine care did not change self-reported PA, but estimated VO 2peak and walking distance increased significantly. Submaximal testing may be a feasible clinical tool to identify low CRF and support counselling. Larger studies are needed to determine whether fitness testing can promote PA and to identify which adolescents benefit most, using measurement approaches that capture complementary aspects of PA (e.g., self-report for context and perceived behaviour, and device-based metrics for volume and intensity where appropriate).

• Størdal, Ketil; Almaas, Runar; Sanner, Helga; Sletner, Line & Stene, Lars Christian Mørch (2026). Incidence and co-occurrence of six autoimmune diseases in childhood: a population-based cohort study in Norway. The Lancet Child and Adolescent Health. ISSN 2352-4642. 10(6), s. 429–437. doi: 10.1016/S2352-4642(26)00008-8. Fulltekst i vitenarkiv
• Ravelli, Angelo; Rosina, Silvia; MacMahon, Jayne M.; Baird, Talia; Rebollo-Giménez, Ana Isabel & Hinze, Claas [Vis alle 41 forfattere av denne artikkelen] (2025). Treating juvenile dermatomyositis to target: Paediatric Rheumatology European Society/Childhood Arthritis and Rheumatology Research Alliance-endorsed recommendations from an international task force. Annals of the Rheumatic Diseases. ISSN 0003-4967. 84(7), s. 1055–1067. doi: 10.1016/j.ard.2025.04.024. Fulltekst i vitenarkiv
• Haftorn, Kristine Løkås; Rudsari, Hamid Khoshfekr; Jaholkowski, Piotr Pawel; Dåstøl, Vilde Øverlien; Hestetun, Sigrid Valen & Andreassen, Ole [Vis alle 8 forfattere av denne artikkelen] (2025). Nonlinearity and sex differences in the performance of a polygenic risk score for juvenile idiopathic arthritis. Frontiers in Immunology. 16. doi: 10.3389/fimmu.2025.1531390. Fulltekst i vitenarkiv Vis sammendrag

  Background: Juvenile idiopathic arthritis (JIA) is an immune-mediated pediatric disease believed to result from a complex interplay of genetic and environmental factors. Genome-wide association studies have enabled calculation of polygenic risk scores (PRS) for JIA. Understanding how the PRS associates with JIA and whether it performs similarly across sexes is essential for its utility in future studies. Methods: We studied the relationship between a PRS developed from a previously published genome-wide association study of JIA and JIA in children from the Norwegian Mother, Father and Child Cohort Study (MoBa; total n = 57,630; JIA cases = 238). Generalized linear models (GLM) and generalized additive models (GAM) were used in logistic regression to assess the association. Furthermore, we investigated whether the relationship between PRS and JIA differed by sex by applying GAM models with interaction terms. Results: PRS was significantly associated with JIA using both GLM (p< 2e-16) and GAM (p< 2e-16) models, and our results indicated a nonlinear relationship between PRS and JIA (effective degrees of freedom, EDF = 1.96). We found a significant interaction between sex and JIA PRS in relation to JIA (p = 0.017), and indications of a stronger and more logit-nonlinear relationship in females (EDF = 1.82) versus males (EDF = 1.06). Conclusion: The relationship between PRS and JIA was slightly logit-nonlinear for females and logit-linear for males. The PRS for JIA can likely be used either as a continuous or discrete variable in analyses, but sex-stratification is recommended for future studies.

• Sherman, Matthew A; Nicolai, Rebecca; Datyner, Emily K; Rosina, Silvia; Hamilton, Angela & Ardalan, Kaveh [Vis alle 20 forfattere av denne artikkelen] (2025). Approach to Janus kinase inhibition for juvenile dermatomyositis among CARRA and PReS providers. Rheumatology. ISSN 1462-0324. 64(8), s. 4732–4737. doi: 10.1093/rheumatology/keaf086. Fulltekst i vitenarkiv
• Risum, Kristine; Olarescu, Nicoleta Cristina; Godang, Kristin; Marstein, Henriette Schermacher; Bollerslev, Jens & Sanner, Helga (2025). Visceral adipose tissue is related to interleukin 6 and resistin in juvenile idiopathic arthritis - a case-control study. Rheumatology International. ISSN 0172-8172. 45(3). doi: 10.1007/s00296-025-05820-8. Fulltekst i vitenarkiv Vis sammendrag

  To compare visceral adipose tissue (VAT) mass, lipid profile, and selected adipokines/cytokines in patients with juvenile idiopathic arthritis (JIA) with controls, and to explore associations between these markers and VAT. We included 60 JIA patients (30 oligoarticular,30 polyarticular), aged 10–16 years, and 60 age-and sex-matched controls. VAT (g) was estimated by dual-energy x-ray absorptiometry. Lipid profile and selected adipokines/cytokines were analyzed by standard methods and ELISA, respectively. VAT (g) was comparable between patients and controls [median (25th-75th percentile): 64 (23–149) g vs. 66 (30–99) g, p = 0.98] and between oligoarticular and polyarticular disease courses [46 (22–123) g vs. 80 (23–167) g, p = 0.32]. Patients had lower serum levels of apolipoprotein A1 (APOA1) and elevated levels of interleukin- 6 (IL-6) and progranulin compared to controls. As compared to oligoarticular disease course, patients with polyarticular disease had lower serum levels of low-density lipoprotein cholesterol (LDL-C), lipoprotein(a) (Lp(a)), interleukin 1 receptor antagonist (IL-1RA) and progranulin, and elevated levels of interleukin-1 beta (IL-1b) and IL-1b/IL-1RA ratio. In patients (B, 95% CI), higher IL-6 (48.7, 25.1 to72.2, p < 0.001), resistin (8.5, 5.1 to 11.8, p < 0.001), and leptin (2.5, 0.9 to 4.0, p = 0.002) were associated with higher VAT. In controls, higher leptin (5.3, 3.7 to 6.9), p < 0.001) was associated with higher VAT. Despite similar VAT mass between patients and controls, VAT was related to IL-6 and resistin in patients only, suggesting an active metabolic role in JIA. Several pro-inflammatory adipokines/cytokines were increased in JIA, with differences in Lp(a) between oligoarticular and polyarticular disease courses.

• Fominykh, Vera; Shadrin, Alexey; Jaholkowski, Piotr Pawel; Fuhrer, Julian; Parker, Nadine & Wiström, Erik David [Vis alle 11 forfattere av denne artikkelen] (2025). Mapping the genetic landscape of immune-mediated disorders: potential implications for classification and therapeutic strategies. Frontiers in Immunology. 16. doi: 10.3389/fimmu.2025.1543781. Fulltekst i vitenarkiv Vis sammendrag

  Objectives: Based on clinical, biomarker, and genetic data, McGonagle and McDermott suggested that autoimmune and autoinflammatory disorders can be classified as a disease continuum from purely autoimmune to autoinflammatory with mixed diseases in between. However, the genetic architecture of this spectrum has not been systematically described. Here, we investigate the continuum of polygenic immune-mediated disorders using genome-wide association studies (GWAS) and statistical genetics methods. Methods: We mapped the genetic landscape of 15 immune-mediated disorders using GWAS summary statistics and methods including genomic structural equation modeling (genomic SEM), linkage disequilibrium score regression, Local Analysis of [co]Variant Association, and Gaussian causal mixture modeling (MiXeR). We performed enrichment analyses of tissues and biological gene sets using MAGMA. Results: Genomic SEM suggested a continuum structure with four underlying latent factors from autoimmune diseases at one end to autoinflammatory on the opposite end. Across disorders, we observed a balanced mixture of negative and positive local genetic correlations within the major histocompatibility complex, while outside this region, local genetic correlations were predominantly positive. MiXeR analysis showed large genetic overlap in accordance with the continuum landscape. MAGMA analysis implicated genes associated with known monogenic immune diseases for prominent autoimmune and autoinflammatory component. Conclusions: Our findings support a polygenic continuum across immune-mediated disorders, with four genetic clusters. The “polygenic autoimmune” and “polygenic autoinflammatory” clusters reside on margins of this continuum. These findings provide insights and lead us to hypothesize that the identified clusters could inform future therapeutical strategies, with patients in the same clusters potentially responding similarly to specific therapies.

• Dåstøl, Vilde Øverlien; Haftorn, Kristine Løkås; Rudsari, Hamid Khoshfekr; Jaholkowski, Piotr Pawel; Størdal, Ketil & Håberg, Siri Eldevik [Vis alle 12 forfattere av denne artikkelen] (2025). Maternal seafood intake, dietary contaminant exposure, and risk of juvenile idiopathic arthritis: exploring gene-environment interactions. Frontiers in Immunology. 15. doi: 10.3389/fimmu.2024.1523990. Fulltekst i vitenarkiv Vis sammendrag

  Objectives: Juvenile idiopathic arthritis (JIA) originates from a complex interplay between genetic and environmental factors. We investigated the association between seafood intake and dietary contaminant exposure during pregnancy and JIA risk, to identify sex differences and gene-environment interactions. Methods: We used the Norwegian Mother, Father, and Child Cohort Study (MoBa), a population-based prospective pregnancy cohort (1999–2008). JIA patients were identified through the Norwegian Patient Registry, with remaining mother-child pairs serving as controls. We assessed maternal seafood intake and dietary contaminants typically found in seafood using a food frequency questionnaire completed during pregnancy, mainly comparing high (≥90th percentile, P90) vs low (<P90) intake. Multivariable logistic regression calculated adjusted odds ratios (aOR), including sex-stratification analyses. A polygenic risk score (PRS) for JIA was used in a subsample to assess gene-environment interactions. Results: We identified 217 JIA patients and 71,884 controls. High vs low maternal intake of lean/semi-oily fish was associated with JIA (aOR 1.51, 95% CI 1.02-2.22), especially among boys (aOR 2.13, 95% CI 1.21-3.75). A significant gene-environment interaction was observed between total fish intake and PRS, with high fish intake associated with JIA primarily in those with low PRS (p<0.03). We found no associations between high vs low exposure to other types of seafood or environmental contaminants and JIA. Conclusions: We found a modestly increased risk of JIA associated with high intake of lean/semi-oily fish during pregnancy, not explained by estimated exposure to dietary contaminants. Our data suggest a more pronounced association in children with a lower genetic predisposition for JIA.

• Berger, Simon Girmai; Berntsen, Kristin Schjander; Risum, Kristine; Marstein, Henriette Schermacher; Sjaastad, Ivar & Stokke, Mathis Korseberg [Vis alle 7 forfattere av denne artikkelen] (2025). Predictors of persistent active disease and the longitudinal development of organ damage, patient-reported outcomes and autoantibodies in long-term juvenile dermatomyositis. RMD Open. 11(2). doi: 10.1136/rmdopen-2025-005598. Fulltekst i vitenarkiv Vis sammendrag

  Objective To quantify longitudinal development of disease activity, organ damage, patient-reported outcomes, and myositis autoantibody profiles in patients with juvenile dermatomyositis (JDM) after long-term follow-up, and to identify predictors for persistent active disease. Methods Forty patients (65% female) diagnosed with JDM were clinically examined at two different time points (visits 1 and 2). We assessed clinically inactive/active disease by the updated PRINTO criteria and the Juvenile DermatoMyositis Activity Index (JDMAI). Organ damage was evaluated by Myositis Damage Index (MDI) and physical function by Childhood Health Assessment Questionnaire (CHAQ/HAQ). Myositis autoantibodies were measured by myositis line immunoassay. Results Median disease duration from symptom onset was 15.1 (2.0–34.6) at visit 1 and 21.7 (7.6–42.7) years at visit 2. At visit 2, active disease (PRINTO) was found in 53%, impaired physical function (CHAQ/HAQ>0) in 40%, organ damage (MDItotal≥1) in 95% and myositis specific or associated antibodies in 33% of patients. Disease activity (JDMAI) was low in 24%, moderate in 8% and high in 3% of patients. There were no significant differences in organ damage and disease activity between visits. Higher disease activity and organ damage at visit 1 predicted persistent active disease at visit 2. Conclusions After a median of 21.7 years from symptom onset, the majority of JDM patients still had active disease. Higher organ damage and disease activity at the initial visit predicted persistent active disease at follow-up. These results underscore the chronic nature of JDM, emphasising the need for enhanced early and long-term management strategies to improve patient outcomes.

• Hestetun, Sigrid Valen; Rudsari, Hamid Khoshfekr; Jaholkowski, Piotr Pawel; Shadrin, Alexey; Haftorn, Kristine Løkås & Andersen, Svend [Vis alle 13 forfattere av denne artikkelen] (2024). Incidence and Genetic Risk of Juvenile Idiopathic Arthritis in Norway by Latitude. Arthritis & Rheumatology. ISSN 2326-5191. 77(4), s. 458–467. doi: 10.1002/art.43040. Fulltekst i vitenarkiv Vis sammendrag

  Objective: We aimed to investigate the incidence of juvenile idiopathic arthritis (JIA) in the three geographic regions of Norway and whether potential regional incidence differences are explained by environmental or genetic factors across regions. Methods: We conducted a register-based cohort study including all Norwegian children born from 2004 to 2019, with follow-up throughout 2020. The JIA diagnosis, defined by at least two International Classification of Diseases, Tenth Revision codes for JIA, was validated against medical records. The incidence rate (IR) and hazard ratio (HR) for JIA were estimated for all Norway and for the North, Mid, and South regions. In a subsample from the Norwegian Mother, Father, and Child Cohort Study (MoBa), the genetic risk for JIA was assessed in the three regions. Results: After median 9.1 (range 0.3-16.0) years of follow-up, we identified 1,184 patients with JIA and 910,058 controls. The IR for JIA/100,000 person-years was 14.4 in all of Norway, 25.9 in the North region, 17.9 in the Mid region, and 12.5 in the South region. The HR (95% confidence interval [CI]) of JIA in the North region was 2.07 (1.77-2.43) and in the Mid region HR 1.43 (95% CI 1.23-1.67) compared with the South region. Adjustments for perinatal factors, socioeconomic status, and early antibiotic exposure did not change our estimates substantially. In MoBa (238 patients with JIA, 57,392 controls), the association between JIA and region of birth was no longer significant when adjusting for genetic factors. Conclusion: We found a higher incidence of JIA with increasing latitude without evidence for available environmental factors explaining the observed gradient. In contrast, genetic factors modified the association, but further studies are warranted.

• Haukeland, Hilde; Moe, Sigrid Reppe; Brunborg, Cathrine; Botea, Antonela; Damjanic, Nenad & Wivestad, Gro Årthun [Vis alle 13 forfattere av denne artikkelen] (2024). Sensitivity of classification criteria from time of diagnosis in an incident systemic lupus erythematosus cohort: a population-based study from Norway. RMD Open. 10(3). doi: 10.1136/rmdopen-2024-004395. Fulltekst i vitenarkiv Vis sammendrag

  Objectives To compare the sensitivity of 2019 European Alliance of Associations for Rheumatology/American College of Rheumatology (EULAR/ACR) classification criteria against 1997 ACR criteria for systemic lupus erythematosus (SLE), for incident SLE cases in the presumably complete population-based Nor-SLE cohort from Southeast Norway (2.9 million inhabitants). Methods All cases International Statistical Classification of Diseases and Related Health Problems, Tenth Revision (ICD-10) coded as SLE during 2000–2017 were individually reviewed. Those with a confirmed SLE diagnosis by expert clinical assessment were included in the Nor-SLE cohort. Core clinical data were recorded, and the cases were classified according to 2019 EULAR/ACR and 1997 ACR criteria. Juvenile SLE was defined as <16 years at diagnosis and adult SLE was defined as ≥16 years at diagnosis. Results We included 737 incident SLE cases (701 adults, 36 juveniles). At diagnosis, 2019 EULAR/ACR criteria were more sensitive than 1997 ACR criteria for adults (91.6% vs 77.3%; p<0.001), but not for juveniles (97.2% vs 88.9%). The 2019 EULAR/ACR counts at diagnosis differed by age group and ethnicity, being higher in young cases and those originating from Asia. From time of diagnosis to study end the fulfilment rate of 2019 EULAR/ACR criteria for the adult cohort increased from 92.5% and 86.5% to 94.6% and 91.0%, respectively, for females and males (mean disease duration of 7.5 years). Conclusion Showing 92% criteria fulfilment already at time of SLE diagnosis by 2019 EULAR/ACR criteria versus 77% by 1997 ACR criteria, the results from this population-based study suggest that the 2019 EULAR/ACR criteria will achieve its goal of capturing more early-SLE cases for clinical trials.

• Bolstad, Inger Helen; Nilsson, Ulrika Ann-Charlotte; Hansbø, Silvia Elisabeth; Sanner, Helga; Krosby, Kirsti & Vaksvik, Tone [Vis alle 7 forfattere av denne artikkelen] (2024). Kunnskapsbasert fagprosedyre for ergo- og fysioterapi til barn og ungdom med utmattelse knyttet til revmatisk sykdom. Fysioterapeuten. ISSN 0016-3384. Fulltekst i vitenarkiv
• Marstein, Henriette; NOMELAND, BIRGIT; Godang, Kristin; Olarescu, Nicoleta Cristina; Schwartz, Thomas & Flatø, Berit [Vis alle 10 forfattere av denne artikkelen] (2023). Adipokine profile in long-term juvenile dermatomyositis, and associations with adipose tissue distribution and cardiac function: a cross-sectional study. RMD Open. 9:e002815(1), s. 1–9. doi: 10.1136/rmdopen-2022-002815. Fulltekst i vitenarkiv Vis sammendrag

  Objectives In long-term juvenile dermatomyositis (JDM), altered adipose tissue distribution and subclinical cardiac dysfunction have been described. Our aims were to compare adipokine levels in patients with JDM after long-term disease with controls, and explore associations between adipokines and (1) adipose tissue distribution and (2) cardiac function. Methods The study cohort included 59 patients with JDM (60% female, mean age 25.2 years, mean disease duration 16.9 years), and 59 age/sex-matched controls. Updated Pediatric Rheumatology International Trials Organization criteria for clinically inactive JDM were used to stratify patients into active (JDM-active) or inactive (JDM-inactive) disease groups. Lipodystrophy was clinically assessed in all patients. In all study participants, we measured adipose tissue distribution by dual-energy X-ray absorptiometry and cardiac function by echocardiography. Serum adipokines (adiponectin, apelin-12, lipocalin-2, leptin, visfatin and resistin) were analysed using ELISA. Results Patients with JDM had higher leptin levels compared with controls (p≤0.01). In JDM-active, apelin-12 and visfatin were higher compared with JDM-inactive (p≤0.05). In JDM-total and JDM-active, lower adiponectin correlated with lipodystrophy and total fat mass. Also, systolic dysfunction correlated with: lower adiponectin in JDM-total, JDM-inactive and JDM-active, and with lower apelin-12 in JDM-total and JDM-active and resistin in JDM-active (all p≤0.05). Lower adiponectin correlated with diastolic dysfunction in JDM-total and JDM-active. Conclusion After long-term disease, leptin levels were unfavourably regulated in patients with JDM compared with controls, and apelin-12 and visfatin in JDM-active versus JDM-inactive. We found associations between adipokines and both adipose tissue distribution and cardiac systolic function in all patients with JDM, which was most prominent in patients with active disease.

• Hestetun, Sigrid Valen; Andersen, Svend; Sanner, Helga & Størdal, Ketil (2023). Antibiotic exposure in prenatal and early life and risk of juvenile idiopathic arthritis: a nationwide register-based cohort study. RMD Open. 9(3), s. 1–8. doi: 10.1136/rmdopen-2023-003333. Fulltekst i vitenarkiv Vis sammendrag

  Objectives Early antibiotic exposure influences the gut microbiota which is believed to be involved in the pathogenesis of juvenile idiopathic arthritis (JIA). We aimed to investigate the association between systemic antibiotics in prenatal and early life and risk of JIA. Methods We conducted a register-based cohort study including all children born in Norway from 2004 through 2012. The children were followed until 31 December 2020. Main exposures were dispensed antibiotics to the mother during pregnancy and to the child during 0–24 months of age. The outcome was defined by diagnostic codes indicating JIA. Multivariate logistic regression analyses were performed to estimate the association between antibiotic exposure and JIA. Results We included 535 294 children and their mothers in the analyses; 1011 cases were identified. We found an association between exposure to systemic antibiotics during 0–24 months and JIA (adjusted OR (aOR) 1.40, 95% CI 1.24 to 1.59), with a stronger association for >1 course (aOR 1.50, 95% CI 1.29 to 1.74) vs 1 course (aOR 1.31, 95% CI 1.13 to 1.53). Subanalyses showed significant associations in all age periods except 0–6 months, and stronger association with sulfonamides/trimethoprim and broad-spectrum antibiotics. There was no association between prenatal antibiotic exposure and JIA. Conclusions The novel observation of no association with prenatal antibiotic exposure and JIA suggests that the association between antibiotics in early life and JIA is unlikely to be confounded by shared family factors. This may indicate that exposure to antibiotics in early life is an independent risk factor for JIA.

• Berger, Simon Girmai; NOMELAND, BIRGIT; Reiseter, Silje; Schwartz, Thomas; Andersson, Anna Helena & Hetlevik, Siri Opsahl [Vis alle 13 forfattere av denne artikkelen] (2023). Cardiac dysfunction in mixed connective tissue disease: a nationwide observational study. Rheumatology International. ISSN 0172-8172. s. 1055–1065. doi: 10.1007/s00296-023-05308-3. Fulltekst i vitenarkiv Vis sammendrag

  Abstract We aimed to identify cardiac function in patients with established mixed connective tissue disease (MCTD). This was a cross-sectional case–control study of well-characterised MCTD patients who had previously been included in a nationwide cohort. Assessments comprised protocol transthoracic echocardiography, electrocardiogram and blood samples. In patients only, we evaluated the findings of high-resolution pulmonary computed tomography and disease activity. We assessed 77 MCTD patients (mean age 50.5 ± 12.3 years) with a mean disease duration of 16.4 years, and 59 age- and sex-matched healthy controls (49.9 ± 11.7 years). By echocardiography, measures of left ventricular function, i.e. fractional shortening (38.1 ± 6.4% vs. 42.3 ± 6.6%, p &lt; 0.001), mitral annulus plane systolic excursion (MAPSE) (13.7 ± 2.1 mm vs. 15.3 ± 2.3 mm, p &lt; 0.001) and early diastolic velocity of the mitral annulus (e’) (0.09 ± 0.02 m/s vs. 0.11 ± 0.03 m/s, p = 0.002) were subclinical and lower in patients than controls. Right ventricular dysfunction was found in patients assessed by tricuspid annular plane systolic excursion (TAPSE) (22.7 ± 4.0 mm vs. 25.5 ± 4.0 mm, p &lt; 0.001). While cardiac dysfunction was not associated with pulmonary disease, e’ and TAPSE were found to correlate with disease activity at baseline. In this cohort of MCTD patients, echocardiographic examinations demonstrated a higher frequency of cardiac dysfunction than in matched controls. Cardiac dysfunction was associated with disease activity at baseline, but was independent of cardiovascular risk factors and pulmonary disease. Our study indicates that cardiac dysfunction is part of the multi-organ affliction seen in MCTD.

• Saevarsdottir, Saedis; Stefansdottir, Lilja; Sulem, Patrick; Thorleifsson, Gudmar; Ferkingstad, Egil & Rutsdottir, Gudrun [Vis alle 78 forfattere av denne artikkelen] (2022). Multiomics analysis of rheumatoid arthritis yields sequence variants that have large effects on risk of the seropositive subset. Annals of the Rheumatic Diseases. ISSN 0003-4967. 81(8), s. 1085–1095. doi: 10.1136/annrheumdis-2021-221754. Fulltekst i vitenarkiv Vis sammendrag

  Objectives To find causal genes for rheumatoid arthritis (RA) and its seropositive (RF and/or ACPA positive) and seronegative subsets. Methods We performed a genome-wide association study (GWAS) of 31 313 RA cases (68% seropositive) and ~1 million controls from Northwestern Europe. We searched for causal genes outside the HLA-locus through effect on coding, mRNA expression in several tissues and/or levels of plasma proteins (SomaScan) and did network analysis (Qiagen). Results We found 25 sequence variants for RA overall, 33 for seropositive and 2 for seronegative RA, altogether 37 sequence variants at 34 non-HLA loci, of which 15 are novel. Genomic, transcriptomic and proteomic analysis of these yielded 25 causal genes in seropositive RA and additional two overall. Most encode proteins in the network of interferon-alpha/beta and IL-12/23 that signal through the JAK/STAT-pathway. Highlighting those with largest effect on seropositive RA, a rare missense variant in STAT4 (rs140675301-A) that is independent of reported non-coding STAT4-variants, increases the risk of seropositive RA 2.27-fold (p=2.1×10−9), more than the rs2476601-A missense variant in PTPN22 (OR=1.59, p=1.3×10−160). STAT4 rs140675301-A replaces hydrophilic glutamic acid with hydrophobic valine (Glu128Val) in a conserved, surface-exposed loop. A stop-mutation (rs76428106-C) in FLT3 increases seropositive RA risk (OR=1.35, p=6.6×10−11). Independent missense variants in TYK2 (rs34536443-C, rs12720356-C, rs35018800-A, latter two novel) associate with decreased risk of seropositive RA (ORs=0.63–0.87, p=10−9–10−27) and decreased plasma levels of interferon-alpha/beta receptor 1 that signals through TYK2/JAK1/STAT4. Conclusion Sequence variants pointing to causal genes in the JAK/STAT pathway have largest effect on seropositive RA, while associations with seronegative RA remain scarce.

• Dijkhuizen, E. H. Pieter van; Ridella, Francesca; Naddei, Roberta; Trincianti, Chiara; Avrusin, Ilia & Mazzoni, Marta [Vis alle 20 forfattere av denne artikkelen] (2022). Validity and Reliability of Four Parent/Patient–Reported Outcome Measures for Juvenile Idiopathic Arthritis Remote Monitoring. Arthritis care & research. ISSN 2151-464X. 0(0), s. 1–10. doi: 10.1002/acr.24855. Fulltekst i vitenarkiv
• NOMELAND, BIRGIT; Schwartz, Thomas; Barth, Zoltan; Taraldsrud, Eli; Samersaw-Lund, May Brit & Aaløkken, Trond Mogens [Vis alle 9 forfattere av denne artikkelen] (2022). Associations between cardiac and pulmonary involvement in patients with juvenile dermatomyositis—a cross-sectional study. Rheumatology International. ISSN 0172-8172. 42, s. 1213–1220. doi: 10.1007/s00296-021-05071-3. Fulltekst i vitenarkiv Vis sammendrag

  Abstract This study aimed at exploring the association between detectable cardiac and pulmonary involvement in long-term juvenile dermatomyositis (JDM) and to assess if patients with cardiac and pulmonary involvement differ with regard to clinical characteristics. 57 JDM patients were examined mean 17.3 (10.5) years after disease onset; this included clinical examination, myositis specific/associated autoantibodies (immunoblot), echocardiography, pulmonary function tests and high-resolution computed tomography. Cardiac involvement was defined as diastolic and/or systolic left ventricular dysfunction and pulmonary involvement as low diffusing capacity for carbon monoxide, low total lung capacity and/or high-resolution computed tomography abnormalities. Patients were stratified into the following four groups: (i) no organ involvement, (ii) pulmonary only, (iii) cardiac only, and (iv) co-existing pulmonary and cardiac involvement. Mean age was 25.7 (12.4) years and 37% were males. One patient had coronary artery disease, seven had a history of pericarditis, seven had hypertension and three had known interstitial lung disease prior to follow-up. There was no association between cardiac (10/57;18%) and pulmonary (41/57;72%) involvement ( p  = 0.83). After stratifying by organ involvement, 21% of patients had no organ involvement; 61% had pulmonary involvement only; 7% had cardiac involvement only and 11% had co-existing pulmonary or cardiac involvement. Patients with co-existing pulmonary or cardiac involvement had higher disease burden than the remaining patients. Patients with either cardiac or pulmonary involvement only, differed in clinical and autoantibody characteristics. We found no increased risk of developing concomitant cardiac/pulmonary involvement in JDM. Our results shed light upon possible different underlying mechanisms behind pulmonary and cardiac involvement in JDM.

• Marstein, Henriette; Witczak, Birgit Nomeland; Godang, Kristin; Schwartz, Thomas; Flatø, Berit & Bollerslev, Jens [Vis alle 8 forfattere av denne artikkelen] (2022). Adipose tissue distribution is associated with cardio-metabolic alterations in adult patients with juvenile-onset dermatomyositis. Rheumatology. ISSN 1462-0324. 62(2), s. 1–9. doi: 10.1093/rheumatology/keac293. Fulltekst i vitenarkiv Vis sammendrag

  Abstract Objectives Primary aims were to compare adipose tissue distribution in adult patients with juvenile-onset DM (JDM), with matched controls. Secondary aims were to explore how adipose tissue distribution is associated with cardio-metabolic status (cardiac dysfunction and metabolic syndrome) in patients. Methods Thirty-nine JDM patients (all aged ≥18 y, mean age 31.7 y and 51% female) were examined mean 22.7 y (s.d. 8.9 y) after disease onset and compared with 39 age/sex-matched controls. In patients, disease activity and lipodystrophy were assessed by validated tools and use of prednisolone noted. In all participants, dual-energy X-ray absorptiometry (DXA) and echocardiography were used to measure visceral adipose tissue (VAT)(g) and cardiac function, respectively. Risk factors for metabolic syndrome were measured and associations with adipose tissue distribution explored. For primary and secondary aims, respectively, P-values ≤0.05 and ≤0.01 were considered significant. Results Patients exhibited a 2.4-fold increase in VAT, and reduced HDL-cholesterol values compared with controls (P-values ≤ 0.05). Metabolic syndrome was found in 25.7% of the patients and none of the controls. Cardiac dysfunction (systolic and/or diastolic) was found in 23.7% of patients and 8.1% of controls (P = 0.07). In patients, VAT levels were correlated with age, disease duration and occurrence of metabolic syndrome and cardiac dysfunction. Occurrence of lipodystrophy (P = 0.02) and male sex (P = 0.04) tended to be independently associated with cardiac dysfunction. Conclusion Adults with JDM showed more central adiposity and cardio-metabolic alterations than controls. Further, VAT was found increased with disease duration, which was associated with development of cardio-metabolic syndrome.

• Giancane, Gabriella; Papa, Riccardo; Vastert, Sebastiaan; Bagnasco, Francesca; Swart, Joost F. & Quartier, Pierre [Vis alle 24 forfattere av denne artikkelen] (2022). Anakinra in Patients With Systemic Juvenile Idiopathic Arthritis: Long-term Safety From the Pharmachild Registry. Journal of Rheumatology. ISSN 0315-162X. 49(4), s. 398–407. doi: 10.3899/jrheum.210563. Fulltekst i vitenarkiv
• Deakin, Claire T.; Bowes, John; Rider, Lisa G.; Miller, Frederick W.; Pachman, Lauren M. & Sanner, Helga [Vis alle 19 forfattere av denne artikkelen] (2022). Association with HLA-DRβ1 position 37 distinguishes juvenile dermatomyositis from adult-onset myositis. Human Molecular Genetics. ISSN 0964-6906. 31(14), s. 2471–2481. doi: 10.1093/hmg/ddac019. Fulltekst i vitenarkiv Vis sammendrag

  Juvenile dermatomyositis (JDM) is a rare, severe autoimmune disease and the most common idiopathic inflammatory myopathy of children. JDM and adult-onset dermatomyositis (DM) have similar clinical, biological and serological features, although these features differ in prevalence between childhood-onset and adult-onset disease, suggesting that age of disease onset may influence pathogenesis. Therefore, a JDM-focused genetic analysis was performed using the largest collection of JDM samples to date. Caucasian JDM samples (n = 952) obtained via international collaboration were genotyped using the Illumina HumanCoreExome chip. Additional non-assayed human leukocyte antigen (HLA) loci and genome-wide single-nucleotide polymorphisms (SNPs) were imputed. HLA-DRB1*03:01 was confirmed as the classical HLA allele most strongly associated with JDM [odds ratio (OR) 1.66; 95% confidence interval (CI) 1.46, 1.89; P = 1.4 × 10−14], with an independent association at HLA-C*02:02 (OR = 1.74; 95% CI 1.42, 2.13, P = 7.13 × 10−8). Analyses of amino acid positions within HLA-DRB1 indicated that the strongest association was at position 37 (omnibus P = 3.3 × 10−19), with suggestive evidence this association was independent of position 74 (omnibus P = 5.1 × 10−5), the position most strongly associated with adult-onset DM. Conditional analyses also suggested that the association at position 37 of HLA-DRB1 was independent of some alleles of the Caucasian HLA 8.1 ancestral haplotype (AH8.1) such as HLA-DQB1*02:01 (OR = 1.62; 95% CI 1.36, 1.93; P = 8.70 × 10−8), but not HLA-DRB1*03:01 (OR = 1.49; 95% CR 1.24, 1.80; P = 2.24 × 10−5). No associations outside the HLA region were identified. Our findings confirm previous associations with AH8.1 and HLA-DRB1*03:01, HLA-C*02:02 and identify a novel association with amino acid position 37 within HLA-DRB1, which may distinguish JDM from adult DM.

• Marstein, Henriette; Godang, Kristin; Flatø, Berit; Sjaastad, Ivar; Bollerslev, Jens & Sanner, Helga (2021). Bone mineral density and explanatory factors in children and adults with juvenile dermatomyositis at long term follow-up; a cross sectional study. Pediatric Rheumatology. 19(1), s. 1–11. doi: 10.1186/s12969-021-00543-z. Fulltekst i vitenarkiv Vis sammendrag

  Background Juvenile dermatomyositis (JDM) is the most common idiopathic inflammatory myopathy in children and adolescents. Both the disease and its treatment with glucocorticoids may negatively impact bone formation. In this study we compare BMD in patients (children/adolescence and adults) with long-standing JDM with matched controls; and in patients, explore how general/disease characteristics and bone turnover markers are associated with BMD. Methods JDM patients (n = 59) were examined median 16.8y (range 6.6–27.0y) after disease onset and compared with 59 age/sex-matched controls. Dual-energy X-ray absorptiometry (DXA) was used to measure BMD of the whole body and lumbar spine (spine) in all participants, and of ultra-distal radius, forearm and total hip in participants ≥20y only. Markers of bone turnover were analysed, and associations with outcomes explored. Results Reduced BMD Z-scores (<−1SD) were found in 19 and 29% of patients and 7 and 9% of controls in whole body and spine, respectively (p-values < 0.05). BMD and BMD Z-scores for whole body and spine were lower in all patients and for < 20y compared with their respective controls. In participants ≥20y, only BMD and BMD Z-score of forearm were lower in the patients versus controls. In patients, BMD Z-scores for whole body and/or spine were found to correlate negatively with prednisolone use at follow-up (yes/no) (age < 20y), inflammatory markers (age ≥ 20y) and levels of interferon gamma-induced protein 10 (IP-10) (both age groups). In all patients, prednisolone use at follow-up (yes/no) and age ≥ 20y were independent correlates of lower BMD Z-scores for whole body and spine, respectively. Conclusion In long-term JDM, children have more impairment of BMD than adults in spine and whole-body. Associations with BMD were found for both prednisolone and inflammatory markers, and a novel association was discovered with the biomarker of JDM activity, IP-10.

• NOMELAND, BIRGIT; Bollerslev, Jens; Godang, Kristin; Schwartz, Thomas; Flatø, Berit & Molberg, Øyvind [Vis alle 8 forfattere av denne artikkelen] (2021). Body composition in long-standing juvenile dermatomyositis: associations with disease activity, muscle strength and cardiometabolic measures. Rheumatology. ISSN 1462-0324. 61(7), s. 2959–2968. doi: 10.1093/rheumatology/keab805. Fulltekst i vitenarkiv Vis sammendrag

  Abstract Objective To (i) compare body composition parameters in patients with longstanding juvenile dermatomyositis (JDM) and controls and (ii) explore associations between body composition and disease activity/inflammation, muscle strength, health-related quality of life (HRQL) and cardiometabolic measures. Methods In a cross-sectional study, we included 59 patients (median disease duration 16.7 y; median age 21.5 y) and 59 age- and sex-matched controls. Active/inactive disease were defined by the PRINTO criteria. Body composition was assessed by total body dual-energy absorptiometry (DXA), inflammation by hs-CRP and cytokines, muscle strength by manual muscle test (MMT-8), HRQL by 36-item short form survey physical component score (SF-36 PCS) and cardiometabolic function by echocardiography (systolic and diastolic function) and serum-lipids. Results DXA analyses revealed lower appendicular lean mass index (ALMI) (reflecting limb skeletal muscle mass), higher body fat percentage (BF%) and higher android: gynoid fat ratio (A: G ratio) (reflecting central fat distribution) in patients than controls, despite similar BMI. Patients with active disease had lower ALMI and higher BF% than those with inactive disease; lower ALMI and higher BF% were associated with inflammation (elevated monocyte attractant protein-1 (MCP-1) and hs-CRP). Lower ALMI was associated with reduced muscle strength; higher BF% was associated with impaired HRQL. Central fat distribution (higher A: G ratio) was associated with impaired cardiac function and unfavorable serum-lipids. Conclusion : Despite normal BMI, patients with JDM, especially those with active disease, had unfavorable body composition, which was associated with impaired HRQL/muscle strength and cardiometabolic function. The association between central fat distribution and cardiometabolic alterations is a novel finding in JDM.

• Giancane, Gabriella; Swart, Joost Frans; Castagnola, E; Groll, Andreas; Horneff, Gerd & Huppertz, Hans-Iko [Vis alle 26 forfattere av denne artikkelen] (2020). Opportunistic infections in immunosuppressed patients with juvenile idiopathic arthritis: analysis by the Pharmachild Safety Adjudication Committee. Arthritis Research & Therapy. ISSN 1478-6354. 22(1). doi: 10.1186/s13075-020-02167-2. Fulltekst i vitenarkiv Vis sammendrag

  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.

• Barth, Zoltan; Schwartz, Thomas; Flatø, Berit; Aaløkken, Trond Mogens; Koller, Akos & Lund, May-Brith [Vis alle 8 forfattere av denne artikkelen] (2019). The association between nailfold capillary density and pulmonary and cardiac involvement in medium-to long-standing juvenile dermatomyositis. Arthritis care & research. ISSN 2151-464X. 71(4), s. 492–497. doi: 10.1002/acr.23687. Fulltekst i vitenarkiv Vis sammendrag

  Objective To explore the associations between microvascular abnormalities as assessed by nailfold capillaroscopy (NFC) and pulmonary and cardiac involvement in patients with juvenile dermatomyositis (DM) who are assessed after medium‐ to long‐term follow‐up. Methods Fifty‐eight patients with juvenile DM were examined a mean ± SD of 17.0 ± 10.6 years after symptom onset. Nailfold capillary density (NCD) and a neovascular pattern (defined as an active or late scleroderma pattern) were analyzed, with blinding to clinical data. Pulmonary involvement was assessed by pulmonary function tests including spirometry, diffusing capacity for carbon monoxide (DLco ), and body plethysmography. High‐resolution computed tomography (HRCT) was also performed. Cardiac involvement was assessed by electrocardiography, Holter monitoring (heart rate variability), and echocardiography. Results Patients with low NCD (<6 capillaries/mm) (n = 21), compared to patients with normal NCD (≥6 capillaries/mm) (n = 37) had lower forced vital capacity (89.7% versus 98.5% predicted), total lung capacity (87.8% versus 94.5% predicted), and more often had low DLco values (15 [71%] of 21 patients versus 14 [38%] of 37 controls) (all P < 0.05). Use of HRCT to assess airway disease was more frequent in the group with low NCD (6 [30%] of 20 patients versus 3 [8%] of 36 patients in the normal NCD group; P = 0.034). No associations between NCD and cardiac parameters or between neovascular pattern and pulmonary or cardiac parameters were observed. Conclusion In patients with juvenile DM, low NCD was associated with lung involvement, which was mostly subclinical. No significant associations with cardiac involvement were observed. These results shed light on possible mechanisms underlying organ involvement, but further and preferably larger studies are needed to identify NCD as a potential biomarker for lung and cardiac involvement in juvenile DM.

• Berntsen, Kristin Schjander; Edvardsen, Elisabeth; Hansen, Bjørge Hermann; Flatø, Berit; Sjaastad, Ivar & Sanner, Helga (2019). Cardiorespiratory fitness in long-term juvenile dermatomyositis: A controlled, cross-sectional study of active/inactive disease. Rheumatology. ISSN 1462-0324. 58(3), s. 492–501. doi: 10.1093/rheumatology/key342. Fulltekst i vitenarkiv Vis sammendrag

  Objectives. To compare cardiorespiratory fitness (CRF) expressed as maximal oxygen uptake (VO2max) between patients with long-term JDM and controls and between patients with active and inactive disease, as well as to explore exercise limiting factors and associations between CRF and disease variables. Methods. JDM patients (n=45) and age- and gender-matched controls (n=45) performed a cardiopulmonary exercise test (CPET) on a treadmill until exhaustion. Physical activity was measured by accelerometers. Disease activity, damage and muscle strength/function were assessed by validated tools. Clinically inactive disease was defined according to PRINTO criteria. Results. The mean disease duration was 20.8 (S.D. 11.9) years and 29/45 (64%) patients had inactive disease. A low VO2max was found in 27% of patients vs 4% of controls (P = 0.006). The mean VO2max and maximal ventilation (VEmax) were lower in patients with active and inactive disease compared with controls. Patients with active disease also had lower maximal voluntary ventilation (MVV) compared with controls and lower VEmax and MVV compared with those with inactive disease. Patients with inactive disease had lower physical activity levels compared with controls. VO2max correlated negatively with disease damage in patients with inactive disease and positively with muscle strength/function in patients with active disease. Conclusion. CRF was lower in JDM patients, both with active and inactive disease, compared with controls after a mean 20 years disease duration. Cardiopulmonary exercise test results suggested different limiting factors contributing to the reduced CRF according to disease activity, including deconditioning in inactive disease and reduced ventilatory capacity in active disease. Further research is needed to verify this.

• Risum, Kristine; Edvardsen, Elisabeth; Selvaag, Anne Marit G; Dagfinrud, Hanne & Sanner, Helga (2019). Measurement properties and performance of an eight-minute submaximal treadmill test in patients with juvenile idiopathic arthritis: A controlled study. Pediatric Rheumatology. 17. doi: 10.1186/s12969-019-0316-7. Fulltekst i vitenarkiv Vis sammendrag

  Background Poor cardiorespiratory fitness is previously reported in patients with juvenile idiopathic arthritis (JIA) measured both by maximal and submaximal exercise tests, but a submaximal exercise test with acceptable measurement properties is currently lacking for both clinical and research purposes in this patient population. The objectives of this study were to evaluate the measurement properties and performance of a submaximal treadmill test in patients with JIA, and to compare the results with those obtained in controls. Methods Fifty-nine patients (50 girls), aged 10–16 years, with oligo- (n = 30) and polyarticular (n = 29) JIA, and 59 age- and sex-matched controls performed an eight-minute submaximal treadmill test for estimating peak oxygen uptake (VO2peak) followed by a maximal treadmill test measuring VO2peak directly. During the submaximal treadmill test, the study participants walked with no inclination at a speed between 3.2–7.2 km/h for four minutes, and then continued to walk at the same speed for four minutes with five % inclination. VO2peak was directly measured during a continuous graded exercise test on treadmill until exhaustion. Thirty-seven patients participated in the evaluation of the reliability. Criterion validity and reliability were evaluated with interclass correlation coefficient (ICC); measurement errors by Bland-Altman plot, standard error of measurement and smallest detectable change. Results In patients with JIA, the ICC (95% CI) for criterion validity was acceptable at group level 0.71 (0.51, 0.82), but not at individual level. The test-retest reliability and inter-rater reliability were acceptable at individual (0.84 (0.71, 0.91) and 0.92 (0.83, 0.96), respectively) and group levels (0.91 (0.83, 0.96) and 0.96 (0.91, 0.98), respectively). The measurement errors (for test-retest reliability/inter-rater reliability) were large. Bland-Altman plots showed no systematic differences, but a large variability for both the validity and reliability. The performance of and estimated VO2peak from the submaximal test were not associated with disease variables and were comparable between patients and controls. Conclusion The submaximal treadmill test is valid for use in patients with JIA on group level, but not on individual level. The reliability is acceptable. Due to large measurement errors, the submaximal treadmill test is not optimal for use in daily clinical practice to estimate VO2peak in individual patients.

• NOMELAND, BIRGIT; Hetlevik, Siri Opsahl; Sanner, Helga; Barth, Zoltan; Schwartz, Thomas & Flatø, Berit [Vis alle 8 forfattere av denne artikkelen] (2019). Cardiac function is impaired in longstanding juvenile-onset mixed connective tissue disease - a controlled study. Journal of Rheumatology. ISSN 0315-162X. 46(7), s. 739–747. doi: 10.3899/jrheum.180526. Fulltekst i vitenarkiv Vis sammendrag

  To assess cardiac function in patients with juvenile mixed connective tissue disease (JMCTD) compared to matched controls, and to investigate possible associations between cardiac impairment and disease variables and cardiovascular risk factors.Fifty JMCTD patients (86% female) examined median 14.9 (6.6–23.0) years after disease onset were compared with 50 age- and sex-matched controls. Electrocardiogram and echocardiography [including e′ as a marker for diastolic dysfunction and long-axis strain (LAS) and left ventricular (LV) ejection fraction (EF) as markers of systolic function] were performed. LV dysfunction (LVD) was defined as low EF, low LAS, or low e′. Right ventricular function was assessed with tricuspid annular plane systolic excursion (TAPSE). Cardiovascular risk factors and disease variables were assessed.LVD was found in 16% of patients and 4% of controls (p = 0.035). EF and LAS were lower in patients compared to controls (6% lower, p &lt; 0.001, and 4% lower, p = 0.044, respectively). TAPSE was 8% lower in patients versus controls (p = 0.008). No patients had signs of pulmonary hypertension. Patients had longer corrected QT time than controls (p = 0.012). LVD was associated with higher levels of apolipoprotein B, higher disease activity measured by physician’s global assessment, longer prednisolone treatment, and more organ damage assessed with the Myositis Damage Index.Patients with JMCTD had impaired left and right ventricular function compared to matched controls after median 15 years disease duration. High disease activity and longer treatment with prednisolone were factors associated with LVD.

• Blom, Kjersti Benedicte; Bergo, Kaja Knudsen; Espe, Emil Knut Stenersen; Rosseland, Vigdis; Grøtta, Ole Jørgen & Mjøen, Geir [Vis alle 17 forfattere av denne artikkelen] (2019). Cardiovascular rEmodelling in living kidNey donorS with reduced glomerular filtration rate: rationale and design of the CENS study. Blood Pressure. ISSN 0803-7051. 29(2), s. 1–13. doi: 10.1080/08037051.2019.1684817. Fulltekst i vitenarkiv Vis sammendrag

  Purpose: Until recently, it has been believed that donating a kidney not represents any risk for development of cardiovascular disease. However, a recent Norwegian epidemiological study suggests that kidney donors have an increased long-term risk of cardiovascular mortality. The pathophysiological mechanisms linking reduced kidney function to cardiovascular disease are not known. Living kidney donors are screened for cardiovascular morbidity before unilateral nephrectomy, and are left with mildly reduced glomerular filtration rate (GFR) after donation. Therefore, they represent an unique model for investigating the pathogenesis linking reduced GFR to cardiovascular disease and cardiovascular remodelling. We present the study design of Cardiovascular rEmodelling in living kidNey donorS with reduced glomerular filtration rate (CENS), which is an investigator-initiated prospective observational study on living kidney donors. The hypothesis is that living kidney donors develop cardiovascular remodelling due to a reduction of GFR. Materials and methods: 60 living kidney donors and 60 age and sex matched healthy controls will be recruited. The controls will be evaluated to fulfil the Norwegian transplantation protocol for living kidney donors. Investigations will be performed at baseline and after 1, 3, 6 and 10 years in both groups. The investigations include cardiac magnetic resonance imaging, echocardiography, bone density scan, flow mediated dilatation, laser Doppler flowmetry, nailfold capillaroscopy, office blood pressure, 24-h ambulatory blood pressure, heart rate variability and investigation of microbiota and biomarkers for inflammation, cardiovascular risk and the calcium-phosphate metabolism. Conclusions: The present study seeks to provide new insight in the pathophysiological mechanisms linking reduced kidney function to cardiovascular disease. In addition, we aim to enlighten predictors of adverse cardiovascular outcome in living kidney donors.

• Marstein, Henriette; Schwartz, Thomas; Aaløkken, Trond M; Samersaw-Lund, May Brit; Flatø, Berit & Sjaastad, Ivar [Vis alle 7 forfattere av denne artikkelen] (2019). Novel associations between cytokines and pulmonary involvement in juvenile dermatomyositis - a cross-sectional study of long-term disease. Rheumatology. ISSN 1462-0324. 59(8), s. 1862–1870. doi: 10.1093/rheumatology/kez531. Fulltekst i vitenarkiv Vis sammendrag

  To examine associations between cytokines and pulmonary involvement in patients with medium- to long-term JDM. In a cross-sectional study, 58 patients examined median (range) 16.8 (6.6–27.0) years after symptom onset were stratified in inactive (JDM-inactive) and active (JDM-active) disease (updated PRINTO criteria); 56 age/sex matched controls were included. Twenty-nine cytokines (in serum) were analysed (Luminex technology/ELISA). Pulmonary function test included forced vital capacity, total lung capacity (TLC) and diffusing capacity for carbon monoxide reported as % of predicted and low forced vital capacity/TLC/diffusing capacity for carbon monoxide. In patients, the presence of clinical pulmonary damage was assessed and high resolution computed tomography scans were scored for interstitial lung disease, chest wall calcinosis and airways disease. Median age of patients was 21 (7–55) years, 59% were female and 36% inactive. In JDM-active and all patients, higher MCP-1, IP-10 and eotaxin correlated with high-resolution computed tomography findings (rs 0.34–0.61; P &lt; 0.05). MCP-1 and eotaxin correlated with pulmonary damage in JDM-active and all patients (rs 0.41–0.49; P &lt; 0.01). Higher TGF-β1 and PDGF (growth factors) were associated with lower lung volumes (forced vital capacity/TLC measures) in all patients; PDGF in JDM-active and TGF-β1 in JDM-inactive patients. IP-10 correlated with TLC% in JDM-active patients. No associations between cytokines and pulmonary function test were found in controls. In JDM, we found a novel association (not previously described in myositis) between eotaxin and pulmonary involvement; we have previously shown an association between eotaxin and cardiac dysfunction. The associations between IP-10/growth factors/MCP-1 and pulmonary involvement are novel in JDM and were mostly seen in JDM-active patients.

• Berntsen, Kristin Schjander; Raastad, Truls; Marstein, Henriette; Kirkhus, Eva Astrid; Merckoll, Else & Cumming, Kristoffer Toldnes [Vis alle 9 forfattere av denne artikkelen] (2019). Functional and Structural Adaptations of Skeletal Muscle in Long-Term Juvenile Dermatomyositis; a Controlled Cross-Sectional Study. Arthritis & Rheumatology. ISSN 2326-5191. doi: 10.1002/art.41174. Fulltekst i vitenarkiv Vis sammendrag

  Objective: To compare muscle strength and endurance of the knee extensors between patients with long‐term juvenile dermatomyositis (DM) and controls and between patients with active disease and those with inactive disease, and to explore associations between strength/endurance and 1) clinical parameters, 2) physical activity, and 3) humoral/structural adaptation in the skeletal muscle of patients. Methods: In a cross‐sectional study (44 patients and 44 age‐ and sex‐matched controls), we tested isometric muscle strength (peak torque, in Nm) and dynamic muscle endurance (total work, in Joules) of the knee extensors, physical activity (measured by accelerometer), and serum myokine levels (by enzyme‐linked immunosorbent assay). Patients were examined with validated tools (clinical muscle tests and measures of disease activity/damage and inactive disease) and using magnetic resonance imaging of the thigh muscles, which included evaluation of the quadriceps cross‐sectional area (CSA). Needle biopsy samples of the vastus lateralis muscle (obtained from 12 patients ages ≥18 years) were assessed by histochemistry. Results: After a mean ± SD disease duration of 21.8 ± 11.8 years, peak torque was lower in patients with juvenile DM compared to controls (mean difference 29 Nm, 95% confidence interval 13–46; P = 0.001). Similarly, total work of the knee extensors was lower in patients compared to controls (median 738J [interquartile range 565–1,155] versus 1,249J [interquartile range 815–1,665]; P < 0.001). Both peak torque and total work were lower in patients with active juvenile DM compared to those with inactive disease (both P < 0.019); in analyses controlled for quadriceps CSA, only total work remained lower in patients with active disease. Moreover, peak torque and total work correlated with findings from clinical muscle tests in patients with active disease (r = 0.57–0.84). Muscle biopsy results indicated that the fiber type composition was different, but capillary density was similar, between patients with active disease and those with inactive disease. Conclusion: In patients with long‐term juvenile DM, both muscle strength and endurance of the knee extensors were lower when compared to matched controls, and also lower in patients with active disease compared to those with inactive disease. Our results indicate a need for more sensitive muscle tests in this clinical setting. We hypothesize that impaired muscle endurance in patients with active juvenile DM may be influenced by structural/functional adaptations of muscle tissue independent of muscle size.

• Risum, Kristine; Edvardsen, Elisabeth; Godang, Kristin; Selvaag, Anne Marit G; Hansen, Bjørge Hermann & Molberg, Øyvind [Vis alle 10 forfattere av denne artikkelen] (2018). Physical Fitness in Patients With Oligoarticular and Polyarticular Juvenile Idiopathic arthritis diagnosed in the era of biologics: A controlled cross-sectional study. Arthritis care & research. ISSN 2151-464X. 71(12), s. 1611–1620. doi: 10.1002/acr.23818. Fulltekst i vitenarkiv
• Barth, Zoltan; NOMELAND, BIRGIT; Flatø, Berit; Koller, Akos; Sjaastad, Ivar & Sanner, Helga (2018). Assessment of Microvascular Abnormalities by Nailfold Capillaroscopy in Juvenile Dermatomyositis After Medium- to Long-Term Followup. Arthritis care & research. ISSN 2151-464X. 70(5), s. 768–776. doi: 10.1002/acr.23338. Fulltekst i vitenarkiv
• Risum, Kristine; Hansen, Bjørge Hermann; Selvaag, Anne Marit G; Molberg, Øyvind; Dagfinrud, Hanne & Sanner, Helga (2018). Physical activity in patients with oligo- and polyarticular juvenile idiopathic arthritis diagnosed in the era of biologics: A controlled cross-sectional study. Pediatric Rheumatology. 16(64). doi: 10.1186/s12969-018-0281-6. Fulltekst i vitenarkiv Vis sammendrag

  Background: Knowledge about objectively measured levels of physical activity (PA) and PA participation (included facilitators and barriers for PA) in patients with juvenile idiopathic arthritis (JIA) diagnosed in the era of biologics is limited. We aimed to compare objectively measured PA in patients with oligo- and polyarticular JIA diagnosed in the biologic era with controls and to examine associations between PA and disease variables; furthermore, to explore participation in PA, physical education (PE) and facilitators and barriers for PA participation in patients and controls. Methods: The study cohort included 60 patients (30 persistent oligo JIA/30 poly-articular disease) and 60 age- and sex-matched controls. Age range was 10–16 years and 83% were female. PA was measured with accelerometry for seven consecutive days. Disease activity, current treatment, disease duration, functional ability, pain and fatigue were assessed. Structured interviews were applied to explore participation in PA and PE, and PA facilitators and barriers. Results: Patients spent less time in daily vigorous PA than controls, (mean(SE) 21(2) min vs. 26(2) min, p = 0.02), while counts per minute (cpm), steps daily, sedentary time and light and moderate PA did not differ. No differences were found between JIA subgroups. The use of biologic medication was associated with higher cpm and lower sedentary time. Most patients and controls participated in organized or unorganized PA and PE, and enjoyment was the most reported facilitator for PA participation. More patients than controls reported pain as a PA barrier. Conclusion: The PA levels and participation in patients with oligo- and polyarticular JIA are mostly comparable to controls, but patients still need to be encouraged to increase vigorous PA. Enjoyment is the most important facilitator for PA participation in patients with JIA.

• Bottai, Matteo; Tjärnlund, Anna; Santoni, Giola; Werth, Victoria P.; Pilkington, Clarissa & Visser, Marianne De [Vis alle 83 forfattere av denne artikkelen] (2017). EULAR/ACR classification criteria for adult and juvenile idiopathic inflammatory myopathies and their major subgroups: A methodology report. RMD Open. 3(2). doi: 10.1136/rmdopen-2017-000507. Fulltekst i vitenarkiv
• Minoia, Francesca; Bovis, Francesca; Davì, Sergio; Insalaco, Antonella; Lehmberg, Kai & Shenoi, Susan [Vis alle 27 forfattere av denne artikkelen] (2017). Development and initial validation of the macrophage activation syndrome/primary hemophagocytic lymphohistiocytosis score, a diagnostic tool that differentiates primary hemophagocytic lymphohistiocytosis from macrophage activation syndrome. Journal of Pediatrics. ISSN 0022-3476. 189, s. 72–78.e3. doi: 10.1016/j.jpeds.2017.06.005. Fulltekst i vitenarkiv
• Lundberg, Ingrid E; Tjarnlund, Anna; Bottai, Matteo; Werth, Victoria P; Pilkington, Clarissa & Visser, Marianne De [Vis alle 37 forfattere av denne artikkelen] (2017). 2017 European League Against Rheumatism/American College of Rheumatology Classification Criteria for Adult and Juvenile Idiopathic Inflammatory Myopathies and Their Major Subgroups. Arthritis & Rheumatology. ISSN 2326-5191. 69(12), s. 2271–2282. doi: 10.1002/art.40320. Fulltekst i vitenarkiv
• Berntsen, Kristin Schjander; Tollisen, Anita; Schwartz, Thomas; Kirkhus, Eva; Aaløkken, Trond Mogens & Samersaw-Lund, May Brit [Vis alle 9 forfattere av denne artikkelen] (2017). Submaximal exercise capacity in juvenile dermatomyositis after longterm disease: The contribution of muscle, lung, and heart involvement. Journal of Rheumatology. ISSN 0315-162X. 44(6), s. 827–834. doi: 10.3899/jrheum.160997. Fulltekst i vitenarkiv
• Schwartz, Thomas; Diederichsen, Louise Pyndt; Lundberg, Ingrid E.; Sjaastad, Ivar & Sanner, Helga (2016). Cardiac involvement in adult and juvenile idiopathic inflammatory myopathies. RMD Open. 2:e000291(2), s. 1–11. doi: 10.1136/rmdopen-2016-000291. Fulltekst i vitenarkiv
• Barth, Zoltan; NOMELAND, BIRGIT; Schwartz, Thomas; Gjesdal, Knut; Flatø, Berit & Koller, Akos [Vis alle 8 forfattere av denne artikkelen] (2016). In juvenile dermatomyositis, heart rate variability is reduced, and associated with both cardiac dysfunction and markers of inflammation: a cross-sectional study median 13.5 years after symptom onset. Rheumatology. ISSN 1462-0324. 55(3), s. 535–543. doi: 10.1093/rheumatology/kev376. Fulltekst i vitenarkiv Vis sammendrag

  Objectives. Low heart rate variability (HRV) is a well-established predictor of cardiac death. The aim of this study was to investigate arrhythmias and HRV in patients with JDM, and associations between HRV and inflammatory markers, echocardiographic measurements and disease parameters. Methods. Fifty-five patients with JDM were examined 2–34 years (median 13.5 years) after disease onset, and compared with 55 age and sex matched controls. Holter ECG monitoring and echocardiography were analysed blinded to patient information. Arrhythmia and HRV (six parameters) were analysed by standard software, finally adjudicated by an experienced cardiologist. Markers of inflammation (ESR, high sensitivity (hs)CRP and cytokines) were analysed. Disease activity and organ damage were assessed by clinical examination at follow-up and retrospectively by chart review. Results. In two out of six HRV parameters, JDM patients had lower values than controls. No difference in arrhythmias was found between the groups. In patients, but not in controls, there were significant negative correlations between five out of six HRV parameters, and ESR and hsCRP (Spearman correlation coefficient, −0.306 to −0.470; P, 0.023 to <0.001). Also, in patients, negative correlations were found between three out of six HRV parameters and systolic and diastolic function. Active disease and low HRV were associated. Patients with hsCRP in the highest quartile (Q4) had lower HRV in all parameters compared with those in pooled Q1–3 (P < 0.001). Conclusion. JDM patients had reduced HRV, which was associated with elevated inflammatory markers, active disease and reduced myocardial function. This suggests reduced vagal control of the heart; further studies are needed to determine whether this is also associated with cardiac morbidity or mortality.

• Sanner, Helga; Schwartz, Thomas; Flatø, Berit; Vistnes, Maria; Christensen, Geir Arve & Sjaastad, Ivar (2014). Increased levels of eotaxin and MCP-1 in juvenile dermatomyositis median 16.8 years after disease onset; associations with disease activity, duration and organ damage. PLOS ONE. 9(3). doi: 10.1371/journal.pone.0092171. Fulltekst i vitenarkiv
• Schwartz, Thomas; Sjaastad, Ivar; Flatø, Berit; Vistnes, Maria; Christensen, Geir Arve & Sanner, Helga (2014). In active juvenile dermatomyositis, elevated eotaxin and MCP-1 and cholesterol levels in the upper normal range are associated with Cardiac dysfunction. Rheumatology (Sveits). ISSN 0080-2727. 53, s. 2214–2222. doi: 10.1093/rheumatology/keu256. Fulltekst i vitenarkiv
• Sanner, Helga; Sjaastad, Ivar & Flatø, Berit (2014). Disease activity and prognostic factors in juvenile dermatomyositis: A long-term follow-up study applying the Paediatric Rheumatology International Trials Organization criteria for inactive disease and the myositis disease activity assessment tool. Rheumatology. ISSN 1462-0324. 53(9), s. 1578–1585. doi: 10.1093/rheumatology/keu146. Fulltekst i vitenarkiv
• Minoia, Francesca; Davì, Sergio; Horne, Annacarin; Demirkaya, Erkan; Bovis, Francesca & Li, Caifeng [Vis alle 40 forfattere av denne artikkelen] (2014). Clinical features, treatment, and outcome of macrophage activation syndrome complicating systemic juvenile idiopathic arthritis: A multinational, multicenter study of 362 patients. Arthritis and Rheumatism. ISSN 0004-3591. 66(11), s. 3160–3169. doi: 10.1002/art.38802. Fulltekst i vitenarkiv
• Schwartz, Thomas; Sanner, Helga; Gjesdal, Ola; Flatø, Berit & Sjaastad, Ivar (2013). In juvenile dermatomyositis cardiac systolic dysfunction is present after long-term follow-up, and is predicted by sustained early skin activity. Annals of the Rheumatic Diseases. ISSN 0003-4967. 73, s. 1805–1810. doi: 10.1136/annrheumdis-2013-203279. Fulltekst i vitenarkiv
• Tollisen, Anita; Sanner, Helga; Flatø, Berit & Wahl, Astrid (2012). Quality of Life in Adults With Juvenile-Onset Dermatomyositis: A Case-Control Study. Arthritis Care and Research. ISSN 0893-7524. 64(7), s. 1020–1027. doi: 10.1002/acr.21637. Fulltekst i vitenarkiv
• Sanner, Helga; Aaløkken, Trond Mogens; Gran, Jan Tore; Sjaastad, Ivar; Johansen, Bjørn & Flatø, Berit (2011). Pulmonary outcome in juvenile dermatomyositis: a case-control study. Annals of the Rheumatic Diseases. ISSN 0003-4967. 70(1), s. 86–91. doi: 10.1136/ard.2010.131433. Fulltekst i vitenarkiv
• Schwartz, Thomas; Sanner, Helga; Husebye, Trygve Guttorm; Flatø, Berit & Sjaastad, Ivar (2011). Cardiac dysfunction in juvenile dermatomyositis - a case control study. Annals of the Rheumatic Diseases. ISSN 0003-4967. 70(5), s. 766–771. doi: 10.1136/ard.2010.137968. Fulltekst i vitenarkiv
• Sanner, Helga; Kirkhus, Eva; Merckoll, Else; Tollisen, Anita; Røisland, Mona & Lie, Benedicte Alexandra [Vis alle 9 forfattere av denne artikkelen] (2010). Long-Term Muscular Outcome and Predisposing and Prognostic Factors in Juvenile Dermatomyositis: A Case-Control Study. Arthritis Care and Research. ISSN 0893-7524. 62(8), s. 1103–1111. doi: 10.1002/acr.20203. Fulltekst i vitenarkiv
• Sanner, Helga; Aaløkken, Trond Mogens; Gran, Jan Tore; Sjaastad, Ivar; Johansen, Bjørn & Flatø, Berit (2010). Pulmonary outcome in juvenile dermatomyositis: a case-controll study. Annals of the Rheumatic Diseases. ISSN 0003-4967. 70, s. 86–91. doi: 10.1136/ard.2010.131433. Fulltekst i vitenarkiv
• Sanner, Helga; Gran, Jan Tore; Sjaastad, Ivar & Flatø, Berit (2009). Cumulative organ damage and prognostic factors in juvenile dermatomyositis: a cross-sectional study median 16.8 years after symptom onset. Rheumatology. ISSN 1462-0324. 48(12), s. 1541–1547. doi: 10.1093/rheumatology/kep302. Fulltekst i vitenarkiv
• Sanner, Helga (2008). Juvenile idiopatiske inflammatoriske myopatier. Norsk Epidemiologi. ISSN 0803-2491. 18(1), s. 78–82. Fulltekst i vitenarkiv

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• Solheim, Emmeline; Haftorn, Kristine Løkås; Sanner, Helga & Risum, Kristine (2026). Physical inactivity as risk factor for later developing JIA. Fulltekst i vitenarkiv
• Risum, Kristine; Stamnæs, Jorunn; Alexanderson, Helene; Molberg, Øyvind & Sanner, Helga (2026). Altered acute inflammatory response to maximal treadmill exercise in children and adolescents with juvenile idiopathic arthritis. Fulltekst i vitenarkiv
• Hestetun, Sigrid Valen; Haftorn, Kristine Løkås; Størdal, Ketil & Sanner, Helga (2025). Reply. Arthritis & Rheumatology. ISSN 2326-5191. 77(12), s. 1807–1808. doi: 10.1002/art.43239. Fulltekst i vitenarkiv
• Bøyesen, Pernille; Aga, Anna-Birgitte; Lilleby, Vibke; Pesonen, Maiju; Rygg, Marite & Nordal, Ellen Berit [Vis alle 17 forfattere av denne artikkelen] (2025). O09 Long-term treatment efficacy of intra-articular triamcinolone hexacetonide injections in juvenile idiopathic arthritis patients starting tumor necrosis factor inhibitor therapy: 48-week results from a randomized, phase 4 trial - the myjia trial. Pediatric Rheumatology. 23(2). Fulltekst i vitenarkiv
• Dahle, Merete Linden; Risum, Kristine & Sanner, Helga (2025). Disse barna kan ha økt risiko for hjerte- og karsykdom som voksne. blogg.forskning.no. doi: https:/www.forskning.no/barn-og-ungdom-partner-sykdommer/disse-barna-kan-ha-okt-risiko-for-hjerte-og-karsykdom-som-voksne/2521967. Fulltekst i vitenarkiv
• Nilsson, Ulrika Ann-Charlotte; Skattør, Marianne; Sanner, Helga & Risum, Kristine (2025). Can fitness testing promote physical activity in adolescents with juvenile idiopathic arthritis? Fulltekst i vitenarkiv
• Hestetun, Sigrid Valen; Sanner, Helga & Dahle, Merete Lindén (2024). Antibiotikabruk i de første to leveårene kan gi økt risiko for barneleddgikt. Fulltekst i vitenarkiv
• Essouma, Mickael; Araujo, Daniel Brito de; Day, Jessica; Conticini, Edoardo; Riopel, Mary Anne & Elias, Adriana Maluf [Vis alle 36 forfattere av denne artikkelen] (2024). A protocol for scoping reviews on the role of whole-body and dedicated body-part magnetic resonance imaging for assessment of adult and juvenile idiopathic inflammatory myopathies. Rheumatology International. ISSN 0172-8172. 44, s. 2403–2409. doi: 10.1007/s00296-024-05649-7. Fulltekst i vitenarkiv
• Krol, Roline; Swart, Joost Frans; Giancane, Gabriella; Roock, Sytze de; Herlin, T & Dolezalova, Pavla [Vis alle 21 forfattere av denne artikkelen] (2019). OP0058 Development of inflammatory bowel disease during treatment with etanercept in patients with juvenile idiopathic arthritis. Annals of the Rheumatic Diseases. ISSN 0003-4967. 78, s. 100–101. doi: 10.1136/annrheumdis-2019-eular.3189. Fulltekst i vitenarkiv
• Witzcak, Birgit; Schwartz, Thomas; Barth, Zoltan; Lund, M B; Sjaastad, Ivar & Sanner, Helga (2016). Cardiac dysfunction and pulmonary involvement are not associated in juvenile dermatomyositis. Fulltekst i vitenarkiv
• Witzcak, Birgit; Godang, Kristin; Schwartz, Thomas; Flatø, Berit; Bollerslev, Jens & Sjaastad, Ivar [Vis alle 7 forfattere av denne artikkelen] (2015). In adults with juvenile onset dermatomyositis, visceral adipose tissue is increased compared with controls, and is associated with serum lipids. Fulltekst i vitenarkiv
• Barth, Zoltan; Witzcak, Birgit; Schwartz, Thomas; Gjesdal, Knut; Flatø, Berit & Koller, A'kos [Vis alle 8 forfattere av denne artikkelen] (2015). Heart rate variability is related to inflammation and cardiac function in juvenile dermatomyositis. Fulltekst i vitenarkiv
• Barth, Zoltan; Witzcak, Birgit; Schwartz, Thomas; Gjesdal, Knut; Flatø, Berit & Koller, A'kos [Vis alle 8 forfattere av denne artikkelen] (2015). Heart rate variability is related to inflammation and cardiac function in juvenile dermatomyositis. Fulltekst i vitenarkiv
• Lilleby, Vibke; Haydon, Joanna; Sanner, Helga; Krossness, B.K.; Ringstad, Geir Andre & Flatø, Berit (2014). Severe macrophage activation syndrome and central nervous system involvement in juvenile dermatomyositis. Scandinavian Journal of Rheumatology. ISSN 0300-9742. 43(2), s. 171–173. doi: 10.3109/03009742.2013.863968. Fulltekst i vitenarkiv
• Sanner, Helga; Schwartz, Thomas; Flato, Berit; Vistnes, Maria; Christensen, Geir Arve & Sjaastad, Ivar (2012). Increased Levels of Eotaxin and MCP-1 in Juvenile Dermatomyositis Median 17 Years After Diagnosis; Associations with Disease Activity, Duration and Organ Damage. Arthritis and Rheumatism. ISSN 0004-3591. 64(10), s. S824–S824. Fulltekst i vitenarkiv
• Schwartz, Thomas; Sjaastad, Ivar; Flatø, Berit; Vistnes, Maria; Christensen, Geir Arve & Sanner, Helga (2012). Monocyte Chemoattractant Protein-1 and Eotaxin Are Associated with Parameters of Cardiac Dysfunction in Juvenile Dermatomyositis. Arthritis and Rheumatism. ISSN 0004-3591. 64(10), s. S381–S381. Fulltekst i vitenarkiv
• Schwartz, Thomas; Sanner, Helga; Husebye, EE; Flatø, Berit & Sjaastad, Ivar (2011). Cardiac dysfunction in juvenile dermatomyositis - a case control study. Fulltekst i vitenarkiv
• Schwartz, Thomas; Sanner, Helga; Vistnes, Maria; Christensen, Geir Arve; Flatø, Berit & Sjaastad, Ivar (2011). Increased levels of MCP-1 and eotaxin are associated with cardiac dysfunction in juvenile dermatomyositis. Fulltekst i vitenarkiv
• Tollisen, Anita; Sanner, Helga; Flatø, Berit & Wahl, Astrid Klopstad (2011). Health-related Quality of Life in Adults with Juvenile Dermatomyositis. Clinical and Experimental Rheumatology. ISSN 0392-856X. 29(2), s. 389–389. Fulltekst i vitenarkiv
• Sanner, Helga (2010). Juvenile Dermatomyositis: From child- to adulthood. Scandinavian Journal of Rheumatology. ISSN 0300-9742. 39, s. 17–17. Fulltekst i vitenarkiv
• Sanner, Helga & Flatø, Berit (2010). Outcomes and prognostic factors in Juvenile Dermatomyositis. Universitetet i Oslo. Fulltekst i vitenarkiv Vis sammendrag

  Background: Juvenile Dermatomyositis is a rare, presumably autoimmune disease of childhood, with an annual incidence of 1.9-3.2 per million children. Lately, validated measures for disease activity and damage in myositis have been developed, but scarce data about long-term outcome is available. Objectives: To investigate outcomes and predictors for outcomes in a Norwegian inception cohort of Juvenile Dermatomyositis patients, and to see how measures for muscular, pulmonary and health outcomes in JDM patients differ from that of matched controls from the general population. Methods: Patients with probable or definite JDM according to the Bohan-Peter criteria, diagnosis after 1970, disease onset < 18 years of age and disease duration > 2 years were identified from Norwegian hospital registries. The patients were clinically examined median 16.8 years (range 2.0-38.1) after symptom onset and were compared with age- and sex-matched controls (randomly selected from the Norwegian population register). Muscle strength was measured by the Manual Muscle Test (MMT) and Child Myositis Assessment Scale (CMAS); physical health by SF-36 PCS and pulmonary function by spirometry, body plethysmography and diffusion capacity for CO (DLCO). In patients, high resolution computed tomography (HRCT) and magnetic resonance imaging (MRI) of thigh muscles were carried out, and disease activity and damage was measured by Disease Activity Score (DAS), Myositis Damage Index (MDI) and Health Assessment Questionnaire (HAQ/child-HAQ). Early disease characteristics were assessed by chart review. Results: Sixty seven JDM patients were identified, which correspond to an average annual incidence of at least 1.8/million children in Norway from 1970-2005. 4 patients had died (6%); the remaining 63 could all be tracked, and 60 (95%) of those participated in the study (60% female). In patients, cumulative organ damage was found in 90% (included calcinosis in 47%), and physical disability in 40% (HAQ/child-HAQ>0). Muscle strength and physical function was lower in patients than controls (P`s < 0.001 and 0.014, respectively). Muscle weakness was found in 31% (CMAS) and 42% (MMT), but the impairment was mostly mild. MRI detected muscular damage was found in 52% (included muscular atrophy, calcinosis and fatty infiltration), and correlated with CMAS and MDI (P`s < 0.01). Patients had smaller lung volumes than controls (P=0.011). In patients, restrictive ventilatory defects and reduced diffusion capacity were found in 26% and 49%, respectively. HRCT abnormalities were found in 37%, and included interstitial lung disease (14%), calcinosis in the chest wall (14%) and airways disease (15%). High disease activity and damage 6 and 12 months post-diagnosis, predicted cumulative organ damage, calcinosis, muscle weakness and MRI detected muscle damage at follow-up. Conclusions: After median 16.8 years disease duration, a substantial portion of our JDM patients had cumulative organ damage, mild physical disability and unfavorable muscular outcomes. Although mostly subclinical, pulmonary involvement was found in the majority of patients. Our findings show that JDM patients have impaired function many years after diagnosis and that sustained early disease activity and damage predict an unfavorable outcome; also our study highlights the systemic nature of the disease. Hopefully, JDM outcomes will improve with advances in treatment.

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