Browsing by Author "Spiegel, Lynn"

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    A Canadian evaluation framework for quality improvement in childhood arthritis: key performance indicators of the process of care
    (2020-03-19) Barber, Claire E H; Twilt, Marinka; Pham, Tram; Currie, Gillian R; Benseler, Susanne; Yeung, Rae S M; Batthish, Michelle; Blanchette, Nicholas; Guzman, Jaime; Lang, Bianca; LeBlanc, Claire; Levy, Deborah M; O’Brien, Christine; Schmeling, Heinrike; Soon, Gordon; Spiegel, Lynn; Whitney, Kristi; Marshall, Deborah A
    Abstract Background The evaluation of quality of care in juvenile idiopathic arthritis (JIA) is critical for advancing patient outcomes but is not currently part of routine care across all centers in Canada. The study objective is to review the current landscape of JIA quality measures and use expert panel consensus to define key performance indicators (KPIs) that are important and feasible to collect for routine monitoring in JIA care in Canada. Methods Thirty-seven candidate KPIs identified from a systematic review were reviewed for inclusion by a working group including 3 pediatric rheumatologists. A shortlist of 14 KPIs was then assessed using a 3-round modified Delphi panel based on the RAND/UCLA Appropriateness Method. Ten panelists across Canada participated based on their expertise in JIA, quality measurement, or lived experience as a parent of a child with JIA. During rounds 1 and 3, panelists rated each KPI on a 1–9 Likert scale on themes of importance, feasibility, and priority. In round 2, panelists participated in a moderated in-person discussion that resulted in minor modifications to some KPIs. KPIs with median scores of ≥ 7 on all 3 questions without disagreement were included in the framework. Results Ten KPIs met the criteria for inclusion after round 3. Five KPIs addressed patient assessments: pain, joint count, functional status, global assessment of disease activity, and the clinical Juvenile Arthritis Disease Activity Score (cJADAS). Three KPIs examined access to care: wait times for consultation, access to pediatric rheumatologists within 1 year of diagnosis, and frequency of clinical follow-up. Safety was addressed through KPIs on tuberculous screening and laboratory monitoring. KPIs examining functional status using the Childhood Health Assessment Questionnaire (CHAQ), quality of life, uveitis, and patient satisfaction were excluded due to concerns about feasibility of measurement. Conclusions The proposed KPIs build upon existing KPIs and address important processes of care that should be measured to improve the quality of JIA care. The feasibility of capturing these measures will be tested in various data sources including the Understanding Childhood Arthritis Network (UCAN) studies. Subsequent work should focus on development of meaningful outcome KPIs to drive JIA quality improvement in Canada and beyond.
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    Development and validation of the RACER (Readiness for Adult Care in Rheumatology) transition instrument in youth with juvenile idiopathic arthritis
    (2021-06-05) Spiegel, Lynn; Tucker, Lori; Duffy, Karen W.; Lalloo, Chitra; Hundert, Amos; Bourre-Tessier, Josiane; Hazel, Elizabeth; Luca, Nadia; Mosher, Dianne; Nguyen, Cynthia; Stringer, Elizabeth; Victor, Charles; Stinson, Jennifer
    Abstract Background Current evidence suggests that many adolescents with juvenile idiopathic arthritis (JIA) do not successfully transfer to adult care, which can result in adverse health outcomes. Although a growing number of clinical programs have been designed to support healthcare transition, there is a lack of psychometrically sound instruments to evaluate their impact on development of transition-related knowledge and skills in youth with JIA. The purpose of this study was to develop and validate RACER (Readiness for Adult Care in Rheumatology), a self-administered instrument designed to measure stages of readiness for key transition-related skills in adolescents with JIA. Methods A phased approach was used to develop and evaluate the validity and reliability of RACER. Phase 1 A was a consensus conference with 19 key stakeholders to inform instrument domains and items. Phase 1B determined initial content validity using a sample of 30 adolescents with JIA and 15 clinical and research experts. Finally, Phase 2 was a prospective cohort study with repeated measures to evaluate the internal consistency, test-retest reliability, construct validity and responsiveness of the instrument within a sample of adolescents with JIA. Results In Phase 1 A, initial item generation yielded a total of 242 items across six domains from the consensus conference, which was subsequently reduced to a 32-item instrument. Phase 1B established the content validity of the instrument in adolescents with JIA. In the Phase 2 study, with a sample of 96 adolescents, the RACER instrument exhibited good internal consistency in five of its six subscales (Cronbach’s α > 0.7), and strong test-retest reliability between the first two administrations (ICC = 0.83). It also showed robust convergent validity by highly correlating with measures of self-management (SMSAG, rho = 0.73) and transition (TRANSITION-Q, rho = 0.76). The RACER was not correlated with unrelated measures (discriminant validity; PedsQL, rho = 0.14). The RACER scores increased significantly over time as expected, supporting measure responsiveness. Conclusions The RACER is a reliable and valid instrument which is sensitive to change for assessing transition readiness in adolescents with JIA.
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    Growth and weight gain in children with juvenile idiopathic arthritis: results from the ReACCh-Out cohort
    (2017-08-22) Guzman, Jaime; Kerr, Tristan; Ward, Leanne M; Ma, Jinhui; Oen, Kiem; Rosenberg, Alan M; Feldman, Brian M; Boire, Gilles; Houghton, Kristin; Dancey, Paul; Scuccimarri, Rosie; Bruns, Alessandra; Huber, Adam M; Watanabe Duffy, Karen; Shiff, Natalie J; Berard, Roberta A; Levy, Deborah M; Stringer, Elizabeth; Morishita, Kimberly; Johnson, Nicole; Cabral, David A; Larché, Maggie; Petty, Ross E; Laxer, Ronald M; Silverman, Earl; Miettunen, Paivi; Chetaille, Anne-Laure; Haddad, Elie; Spiegel, Lynn; Turvey, Stuart E; Schmeling, Heinrike; Lang, Bianca; Ellsworth, Janet; Ramsey, Suzanne E; Roth, Johannes; Campillo, Sarah; Benseler, Susanne; Chédeville, Gaëlle; Schneider, Rayfel; Tse, Shirley M L; Bolaria, Roxana; Gross, Katherine; Feldman, Debbie; Cameron, Bonnie; Jurencak, Roman; Dorval, Jean; LeBlanc, Claire; St. Cyr, Claire; Gibbon, Michele; Yeung, Rae S M; Duffy, Ciarán M; Tucker, Lori B
    Abstract Background With modern treatments, the effect of juvenile idiopathic arthritis (JIA) on growth may be less than previously reported. Our objective was to describe height, weight and body mass index (BMI) development in a contemporary JIA inception cohort. Methods Canadian children newly-diagnosed with JIA 2005–2010 had weight and height measurements every 6 months for 2 years, then yearly up to 5 years. These measurements were used to calculate mean age- and sex-standardized Z-scores, and estimate prevalence and cumulative incidence of growth impairments, and the impact of disease activity and corticosteroids on growth. Results One thousand one hundred forty seven children were followed for median 35.5 months. Mean Z-scores, and the point prevalence of short stature (height < 2.5th percentile, 2.5% to 3.4%) and obesity (BMI > 95th percentile, 15.8% to 16.4%) remained unchanged in the whole cohort. Thirty-three children (2.9%) developed new-onset short stature, while 27 (2.4%) developed tall stature (>97.5th percentile). Children with systemic arthritis (n = 77) had an estimated 3-year cumulative incidence of 9.3% (95%CI: 4.3–19.7) for new-onset short stature and 34.4% (23–49.4) for obesity. Most children (81.7%) received no systemic corticosteroids, but 1 mg/Kg/day prednisone-equivalent maintained for 6 months corresponded to a drop of 0.64 height Z-scores (0.56–0.82) and an increase of 0.74 BMI Z-scores (0.56–0.92). An increase of 1 in the 10-cm physician global assessment of disease activity maintained for 6 months corresponded to a drop of 0.01 height Z-scores (0–0.02). Conclusions Most children in this modern JIA cohort grew and gained weight as children in the general population. About 1 in 10 children who had systemic arthritis, uncontrolled disease and/or prolonged corticosteroid use, had increased risk of growth impairment.
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    Orofacial symptoms and oral health-related quality of life in juvenile idiopathic arthritis: a two-year prospective observational study
    (2018-07-13) Rahimi, Hanna; Twilt, Marinka; Herlin, Troels; Spiegel, Lynn; Pedersen, Thomas K; Küseler, Annelise; Stoustrup, Peter
    Abstract Background Little is known about the chronicity of orofacial symptoms and how this influences the oral health-related quality of life in juvenile idiopathic arthritis (JIA). Therefore, our objectives were to study the long-term changes in self-reported orofacial symptoms, and to define the impact of orofacial symptoms on oral health-related quality of life in JIA. Methods At baseline (T0), 157 consecutive JIA patients ≤20 years completed a patient pain questionnaire that incorporates domains related to the orofacial area. At the 2 year follow-up (T1), 113 patients completed the same questionnaire (response rate 72%) in addition to the Child Perception’s Questionnaire; a validated 31-item questionnaire addressing oral health-related quality of life. Results At T0, 53% (60/113) of patients reported the presence of orofacial pain, and 36% (41/113) of patients reported compromised orofacial function. At T1, 77% (46/60) of patients with pain at T0 reported persistent pain, and 66% (27/41) of patients with functional disability at T0 reported persistent disability. Patients with orofacial symptoms reported a significantly greater prevalence of negative impact of orofacial conditions on general quality of life and within the domains of emotional and social well-being compared to asymptomatic patients. Conclusion Self-reported orofacial pain and functional disability were common findings in a cohort of JIA patients followed over 2 years. These symptoms seem to persist over time in most patients, and have a significant negative impact on oral health-related quality of life.