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  • 1
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    Online Resource
    Validation of the parent global assessment as a health-related quality of life measure in juvenile idiopathic arthritis: results from ReACCh-Out
    Oxford University Press (OUP) ; 2023
    In:  Rheumatology Vol. 62, No. SI2 ( 2023-02-23), p. SI152-SI162
    Details
    In: Rheumatology, Oxford University Press (OUP), Vol. 62, No. SI2 ( 2023-02-23), p. SI152-SI162
    Abstract: To (i) validate the JIA parent global assessment (parent global) as a health-related quality of life (HRQoL) instrument; (ii) evaluate measurement properties of accepted HRQoL measures relative to those of the parent global; and (iii) assess causal pathways determining parent global scores. Methods Data from the Research in Arthritis in Canadian Children emphasizing outcomes (ReACCh-Out) cohort were used. Measurement properties were assessed in 344 patients at enrolment and 6 months later. Causal pathways were tested by structural equation modelling to understand root causes and mediators leading to parent global scores. Results Construct validity was supported by Spearman correlations of 0.53–0.70 for the parent global with the Juvenile Arthritis Quality of Life Questionnaire, Quality of My Life health scale (HRQoML), Pediatric Quality of Life Inventory (PedsQL)-Parent, and Child Health Questionnaire (CHQ)-Physical. Exceptions were PedsQL-Child (0.44) and CHQ-Psychosocial (0.31). Correlations were lower (0.14–0.49) with disease activity measures (physician global assessment of disease activity, active joint count, ESR). Responsiveness of the parent global to improvement according to parent ratings (0.51) was acceptable and within the range (0.32–0.71) of that of other measures. Reliability estimates and measurement errors for all measures were unsatisfactory, likely due to the prolonged time between assessments. Causal pathways for the parent global matched those previously reported for HRQoML. Conclusions Our results offer support for the parent global as a valid measure of HRQoL for JIA. If confirmed, existing studies using the parent global may be re-interpreted, enhancing our knowledge of HRQoL in children with JIA.
    Type of Medium: Online Resource
    ISSN: 1462-0324 , 1462-0332
    URL: Article
    DOI: 10.1093/rheumatology/keac350
    Language: English
    Publisher: Oxford University Press (OUP)
    Publication Date: 2023
    detail.hit.zdb_id: 1474143-X
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  • 2
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    Reductions in Bone Mineral Density Are Apparent Early in Children With Prevalent Osteonecrosis Lesions Following Leukemia Therapy
    Wiley ; 2023
    In:  Journal of Bone and Mineral Research Vol. 38, No. 8 ( 2023-08), p. 1104-1115
    Details
    In: Journal of Bone and Mineral Research, Wiley, Vol. 38, No. 8 ( 2023-08), p. 1104-1115
    Abstract: Osteonecrosis (ON) is a serious complication of childhood acute lymphoblastic leukemia. We determined the prevalence of osteonecrotic lesions in our patient population by a one‐time multisite magnetic resonance imaging (MRI) more than 1 year following leukemia therapy. MRI findings were evaluated in relationship to clinical factors (including longitudinal changes in bone mineral density [BMD]). Eighty‐six children enrolled in the Steroid Associated Osteoporosis in the Pediatric Population (STOPP) study were evaluated for ON at 3.1 ± 1.3 years following therapy. Thirty children had a total of 150 confirmed ON lesions (35%). Lumbar spine (LS) BMD Z ‐scores (mean ± SD) were low at diagnosis and similar between patients with and without ON (−1.09 ± 1.53 versus −1.27 ± 1.25, p  = 0.549). LS BMD Z ‐scores declined from baseline to 12 months in children with ON (−0.31 ± 1.02) but not in those without (0.13 ± 0.82, p  = 0.035); the hip BMD Z ‐scores from baseline to 24 months declined in both groups, but to a greater extent in those with ON (−1.77 ± 1.22) compared to those without (−1.03 ± 1.07, p  = 0.045). At the time of the MRI, mean total hip and total body (TB) BMD Z ‐scores were lower in children with ON (hip −0.98 ± 0.95 versus −0.28 ± 1.06, p  = 0.010; TB −1.36 ± 1.10 versus −0.48 ± 1.50, p  = 0.018). Pain occurred in 11/30 (37%) with ON versus 20/56 (36%) without, p  = 0.841. In multivariable models, older age at diagnosis (odds ratio [OR] 1.57; 95% confidence interval [CI] , 1.15–2.13; p  = 0.004), and hip BMD Z ‐score at MRI (OR 2.23; 95% CI, 1.02–4.87; p  = 0.046) were independently associated with ON. Overall, one‐third of children demonstrated ON after leukemia therapy. Those with ON had greater reductions in spine and hip BMD Z ‐scores in the first 1 and 2 years of therapy, respectively. Older age and lower hip BMD Z ‐scores at MRI were significantly associated with prevalent, off‐therapy ON. These data assist in identifying children at risk of ON. © 2023 The Authors. Journal of Bone and Mineral Research published by Wiley Periodicals LLC on behalf of American Society for Bone and Mineral Research (ASBMR).
    Type of Medium: Online Resource
    ISSN: 0884-0431 , 1523-4681
    URL: Issue
    URL: Article
    DOI: 10.1002/jbmr.v38.8
    DOI: 10.1002/jbmr.4870
    RVK:
    XA 52230
    Language: English
    Publisher: Wiley
    Publication Date: 2023
    detail.hit.zdb_id: 2008867-X
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  • 3
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    Predictors of persisting pain in children with Juvenile Idiopathic Arthritis: a case control study nested in the ReACCh-Out cohort
    Springer Science and Business Media LLC ; 2023
    In:  Pediatric Rheumatology Vol. 21, No. 1 ( 2023-09-16)
    Details
    In: Pediatric Rheumatology, Springer Science and Business Media LLC, Vol. 21, No. 1 ( 2023-09-16)
    Abstract: To identify baseline predictors of persisting pain in children with Juvenile Idiopathic Arthritis (JIA), relative to patients with JIA who had similar baseline levels of pain but in whom the pain did not persist. Methods We used data from the Research in Arthritis in Canadian Children emphasizing Outcomes (ReACCh-Out) inception cohort to compare cases of ‘moderate persisting pain’ with controls of ‘moderate decreasing pain’. Moderate pain was defined as a Visual Analogue Scale (VAS) for pain measurement score of  〉  3.5 cm. Follow-up was minimum 3 years. Univariate and Multivariate logistic regression models ascertained baseline predictors of persisting pain. Results A total of 31 cases and 118 controls were included. Mean pain scores at baseline were 6.4 (SD 1.6) for cases and 5.9 (1.5) for controls. A greater proportion of cases than controls were females (77.4% vs 65.0%) with rheumatoid factor positive polyarthritis (12.9% vs 4.2%) or undifferentiated JIA (22.6% vs 8.5%). Oligoarthritis was less frequent in cases than controls (9.7% vs 33%). At baseline, cases had more active joints (mean of 11.4 vs 7.7) and more sites of enthesitis (4.6 vs 0.7) than controls. In the final multivariate regression model, enthesitis count at baseline (OR 1.40, CI 95% 1.19–1.76), female sex (4.14, 1.33–16.83), and the overall Quality of My Life (QoML) baseline score (0.82, 0.69–0.98) predicted development of persisting pain. Conclusions Among newly diagnosed children with JIA with moderate pain, female sex, lower overall quality of life, and higher enthesitis counts at baseline predicted development of persisting pain. If our findings are confirmed, patients with these characteristics may be candidates for interventions to prevent development of chronic pain.
    Type of Medium: Online Resource
    ISSN: 1546-0096
    URL: Article
    DOI: 10.1186/s12969-023-00885-w
    Language: English
    Publisher: Springer Science and Business Media LLC
    Publication Date: 2023
    detail.hit.zdb_id: 2279468-2
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  • 4
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    Management of pediatric generalized pustular psoriasis using biologics: An evidence-based review
    Elsevier BV ; 2022
    In:  Journal of the American Academy of Dermatology Vol. 87, No. 2 ( 2022-08), p. 484-486
    Details
    In: Journal of the American Academy of Dermatology, Elsevier BV, Vol. 87, No. 2 ( 2022-08), p. 484-486
    Type of Medium: Online Resource
    ISSN: 0190-9622
    URL: Article
    DOI: 10.1016/j.jaad.2021.10.028
    Language: English
    Publisher: Elsevier BV
    Publication Date: 2022
    detail.hit.zdb_id: 2001404-1
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  • 5
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    Chronic recurrent multifocal osteomyelitis with a comprehensive approach to differential diagnosis of paediatric skull pain
    BMJ ; 2023
    In:  BMJ Case Reports Vol. 16, No. 1 ( 2023-01), p. e252471-
    Details
    In: BMJ Case Reports, BMJ, Vol. 16, No. 1 ( 2023-01), p. e252471-
    Abstract: A girl in middle childhood was referred to rheumatology with a 1-month history of progressive skull pain, preceded by fleeting musculoskeletal symptoms. Apart from a scaly rash on her scalp, she was well, with moderately elevated inflammatory markers. Skull imaging (radiographs, CT and MRI) revealed osteolytic lesions, soft tissue swelling and pachymeningeal enhancement at frontal and temporal convexities. Langerhans cell histiocytosis, bone infection/inflammation or malignancy was considered. Skin and bone biopsies eventually ruled out mimicking diseases and confirmed the diagnosis of chronic recurrent multifocal osteomyelitis (CRMO). She was treated with intravenous pamidronate (IVPAM) for 9 months, with rapid resolution of pain and gradual resolution of bony abnormalities. She remains in remission at 15-month follow-up. While CRMO can affect any bone, skull involvement is extremely rare, with a broad differential diagnosis. We recommend bone biopsy to confirm skull CRMO. The patient achieved excellent clinical and radiological response to IVPAM.
    Type of Medium: Online Resource
    ISSN: 1757-790X
    URL: Article
    DOI: 10.1136/bcr-2022-252471
    Language: English
    Publisher: BMJ
    Publication Date: 2023
    detail.hit.zdb_id: 2467301-8
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  • 6
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    Acceptability of an Adolescent Self‐Management Program for Juvenile Idiopathic Arthritis
    Wiley ; 2022
    In:  ACR Open Rheumatology Vol. 4, No. 2 ( 2022-02), p. 142-151
    Details
    In: ACR Open Rheumatology, Wiley, Vol. 4, No. 2 ( 2022-02), p. 142-151
    Abstract: The study objective was to test the acceptability of a self‐management program (SMP) for adolescents with juvenile idiopathic arthritis (JIA) focused on disease information, self‐management, and social support needs. Methods This study was conducted using inductive qualitative methods to explore the acceptability of an in‐person/videoconference SMP. Two groups of four adolescents with JIA (mean age = 13.5, SD  =  0.8) and two groups of pediatric rheumatology health care professionals (n = 4, n = 5) participated in four feedback sessions each. The SMP was presented to study participants, and feedback was provided on the content, format, and structure of the program. Thematic analysis was used to analyze the data. Results Adolescents felt that the content was appropriate and would be effective in supporting self‐management of their arthritis. Participants advised that the trustworthiness of the information would be increased if a rheumatology health care provider facilitated the session. Potential barriers to participation included distance and availability (weekdays and times), but the option for videoconference‐based participation was an appropriate solution to both of these issues. Minor changes were made to content and format, and required changes were made to address participant recommendations for improvement. Conclusion This study confirmed the acceptability of an in‐person/videoconference SMP for patients with JIA. Modifications were made to the SMP based on the focus group feedback, and future directions include a pilot randomized controlled trial to assess feasibility and preliminary effectiveness of the program.
    Type of Medium: Online Resource
    ISSN: 2578-5745 , 2578-5745
    URL: Issue
    URL: Article
    DOI: 10.1002/acr2.v4.2
    DOI: 10.1002/acr2.11373
    Language: English
    Publisher: Wiley
    Publication Date: 2022
    detail.hit.zdb_id: 2971160-5
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  • 7
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    Canadian Rheumatology Association Recommendations for the Screening, Monitoring, and Treatment of Juvenile Idiopathic Arthritis-associated Uveitis
    The Journal of Rheumatology ; 2022
    In:  The Journal of Rheumatology
    Details
    In: The Journal of Rheumatology, The Journal of Rheumatology
    Abstract: To develop Canadian recommendations for the screening, monitoring, and treatment of uveitis associated with juvenile idiopathic arthritis (JIA). Methods Recommendations were developed using the Grading of Recommendations Assessment, Development, and Evaluation (GRADE)-ADOLOPMENT approach. A working group of 14 pediatric rheumatologists, 6 ophthalmologists, 2 methodologists, and 3 caregiver/patient representatives reviewed recent American College of Rheumatology (ACR)/Arthritis Foundation (AF) recommendations and worked in pairs to develop evidence-to-decision (EtD) tables. A survey to assess agreement and recommendations requiring group discussion was completed. EtD tables were presented, discussed, and voted upon at a virtual meeting, to produce the final recommendations. A health equity framework was applied to all aspects of the adolopment process including the EtD tables, survey responses, and virtual meeting discussion. Results The survey identified that 7 of the 19 recommendations required rigorous discussion. Seventy-five percent of working group members attended the virtual meeting to discuss controversial topics as they pertained to the Canadian environment, including timing to first eye exam, frequency of screening, escalation criteria for systemic and biologic therapy, and the role of nonbiologic therapies. Equity issues related to access to care and advanced therapeutics across Canadian provinces and territories were highlighted. Following the virtual meeting, 5 recommendations were adapted, 2 recommendations were removed, and 1 was developed de novo. Conclusion Recommendations for JIA-associated uveitis were adapted to the Canadian context by a working group of pediatric rheumatologists, ophthalmologists with expertise in the management of uveitis, and parent/patient input, taking into consideration cost, equity, and access.
    Type of Medium: Online Resource
    ISSN: 0315-162X , 1499-2752 , 0315-162X
    URL: Article
    DOI: 10.3899/jrheum.220261
    RVK:
    XA 10000
    Language: English
    Publisher: The Journal of Rheumatology
    Publication Date: 2022
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  • 8
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    The Accuracy of Incident Vertebral Fracture Detection in Children Using Targeted Case‐Finding Approaches
    Wiley ; 2021
    In:  Journal of Bone and Mineral Research Vol. 36, No. 7 ( 2021-07), p. 1255-1268
    Details
    In: Journal of Bone and Mineral Research, Wiley, Vol. 36, No. 7 ( 2021-07), p. 1255-1268
    Abstract: Vertebral fractures are clinically important sequelae of a wide array of pediatric diseases. In this study, we examined the accuracy of case‐finding strategies for detecting incident vertebral fractures (IVF) over 2 years in glucocorticoid‐treated children ( n  = 343) with leukemia, rheumatic disorders, or nephrotic syndrome. Two clinical situations were addressed: the prevalent vertebral fracture (PVF) scenario (when baseline PVF status was known), which assessed the utility of PVF and low lumbar spine bone mineral density (LS BMD; Z ‐score 〈 −1.4), and the non‐PVF scenario (when PVF status was unknown), which evaluated low LS BMD and back pain. LS BMD was measured by dual‐energy X‐ray absorptiometry, vertebral fractures were quantified on spine radiographs using the modified Genant semiquantitative method, and back pain was assessed by patient report. Forty‐four patients (12.8%) had IVF. In the PVF scenario, both low LS BMD and PVF were significant predictors of IVF. Using PVF to determine which patients should have radiographs, 11% would undergo radiography (95% confidence interval [CI] 8–15) with 46% of IVF (95% CI 30–61) detected. Sensitivity would be higher with a strategy of PVF or low LS BMD at baseline (73%; 95% CI 57–85) but would require radiographs in 37% of children (95% CI 32–42). In the non‐PVF scenario, the strategy of low LS BMD and back pain produced the highest specificity of any non‐PVF model at 87% (95% CI 83–91), the greatest overall accuracy at 82% (95% CI 78–86), and the lowest radiography rate at 17% (95% CI 14–22). Low LS BMD or back pain in the non‐PVF scenario produced the highest sensitivity at 82% (95% CI 67–92), but required radiographs in 65% (95% CI 60–70). These results provide guidance for targeting spine radiography in children at risk for IVF. © 2021 American Society for Bone and Mineral Research (ASBMR).
    Type of Medium: Online Resource
    ISSN: 0884-0431 , 1523-4681
    URL: Issue
    URL: Article
    DOI: 10.1002/jbmr.v36.7
    DOI: 10.1002/jbmr.4294
    RVK:
    XA 52230
    Language: English
    Publisher: Wiley
    Publication Date: 2021
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  • 9
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    Distinct phenotypes of multisystem inflammatory syndrome in children: a cohort study
    Springer Science and Business Media LLC ; 2023
    In:  Pediatric Rheumatology Vol. 21, No. 1 ( 2023-04-12)
    Details
    In: Pediatric Rheumatology, Springer Science and Business Media LLC, Vol. 21, No. 1 ( 2023-04-12)
    Abstract: Multisystem inflammatory syndrome in children (MIS-C) is a severe disease with an unpredictable course and a substantial risk of cardiogenic shock. Our objectives were to (a) compare MIS-C phenotypes across the COVID-19 pandemic, (b) identify features associated with intensive care need and treatment with biologic agents. Methods Youth aged 0–18 years, fulfilling the World Health Organization case definition of MIS-C, and admitted to the Alberta Children’s Hospital during the first four waves of the COVID-19 pandemic (May 2020-December 2021) were included in this cohort study. Demographic, clinical, biochemical, imaging, and treatment data were captured. Results Fifty-seven MIS-C patients (median age 6 years, range 0–17) were included. Thirty patients (53%) required intensive care. Patients in the third or fourth wave (indicated as phase 2 of the pandemic) presented with higher peak ferritin (µg/l, median (IQR) = 1134 (409–1806) vs. 370 (249–629), P  = 0.001), NT-proBNP (ng/l, median (IQR) = 12,217 (3013–27,161) vs. 3213 (1216–8483), P  = 0.02) and D-dimer (mg/l, median (IQR) = 4.81 (2.24–5.37) vs. 2.01 (1.27–3.34), P  = 0.004) levels, and higher prevalence of liver enzyme abnormalities (n(%) = 17 (68) vs. 11 (34), P  = 0.02), hypoalbuminemia (n(%) = 24 (100) vs. 25 (81), P  = 0.03) and thrombocytopenia (n(%) 18 (72) vs. 11 (34), P  = 0.007) compared to patients in the first two waves (phase 1). These patients had a higher need of non-invasive/mechanical ventilation (n(%) 4 (16) vs. 0 (0), P  = 0.03). Unsupervised clustering analyses classified 47% of the patients in the correct wave and 74% in the correct phase of the pandemic. NT-proBNP was the only significant contributor to the need for intensive care in all applied multivariate regression models. Treatment with biologic agents was significantly associated with peak CRP (mg/l (median, IQR = 240.9 (132.9-319.4) vs. 155.8 (101.0-200.7), P  = 0.02) and ferritin levels (µg/l, median (IQR) = 1380 (509–1753) vs. 473 (280–296)). Conclusions MIS-C patients in a later stage of the pandemic displayed a more severe phenotype, reflecting the impact of distinct SARS-CoV-2 variants. NT-proBNP emerged as the most crucial feature associated with intensive care need, underscoring the importance of monitoring.
    Type of Medium: Online Resource
    ISSN: 1546-0096
    URL: Article
    DOI: 10.1186/s12969-023-00815-w
    Language: English
    Publisher: Springer Science and Business Media LLC
    Publication Date: 2023
    detail.hit.zdb_id: 2279468-2
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  • 10
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    Patient factors associated with waiting time to pediatric rheumatologist consultation for patients with juvenile idiopathic arthritis
    Springer Science and Business Media LLC ; 2020
    In:  Pediatric Rheumatology Vol. 18, No. 1 ( 2020-12)
    Details
    In: Pediatric Rheumatology, Springer Science and Business Media LLC, Vol. 18, No. 1 ( 2020-12)
    Abstract: Early diagnosis and treatment of Juvenile Idiopathic Arthritis (JIA) is essential to optimize outcomes. Wait times (WTs) to consultation with a pediatric rheumatologist consultation is a Canadian quality measure, with benchmarks set at 7 days for systemic JIA (sJIA) and 4 weeks for other JIA categories. In this study we assess WTs for JIA at a single academic center and describe factors associated with longer WTs. Methods This was a retrospective cohort study of 164 patients enrolled in a pharmacogenetic study in Alberta between 2002 and 2018. Limited chart reviews were conducted to evaluate dates of referral and first rheumatology visit to calculate WTs for receipt of pediatric rheumatology care. Cox proportional hazard models identified factors associated with WTs considering variables at the first pediatric rheumatology visit including: JIA category, age, sex, distance to the pediatric rheumatology clinic, number of active joints, pain and C-reactive protein. Results The median age at diagnosis was 8.0 years (interquartile range, IQR 3.5, 12.0) and 46% of patients had oligoarticular JIA. Only 18 patients (11%) were from rural locations. The median WT for all patients met the national benchmark (22 days, IQR, 9, 44) with no statistically significant difference between WTs among JIA categories ( p  = 0.055). Importantly, the majority of sJIA cases met the 7-day benchmark (67%) with a median WT of 1.5 days. Older age was associated with longer WT (HR 0.94, 95% CI 0.89, 0.98, p  = 0.005). Conclusion Median benchmarks were met, however delays in older patients highlight the need for monitoring WTs.
    Type of Medium: Online Resource
    ISSN: 1546-0096
    URL: Article
    DOI: 10.1186/s12969-020-0413-7
    Language: English
    Publisher: Springer Science and Business Media LLC
    Publication Date: 2020
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