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1. The risk and nature of flares in juvenile idiopathic arthritis: results from the ReACCh-Out cohort

Author

Guzman, Jaime, Oen, Kiem, Huber, Adam M, Watanabe Duffy, Karen, Boire, Gilles, Shiff, Natalie, Berard, Roberta A, Levy, Deborah M, Stringer, Elizabeth, Scuccimarri, Rosie, Morishita, Kimberly, Johnson, Nicole, Cabral, David A, Rosenberg, Alan M, Larché, Maggie, Dancey, Paul, Petty, Ross E, Laxer, Ronald M, Silverman, Earl, Miettunen, Paivi, Chetaille, Anne-Laure, Haddad, Elie, Houghton, Kristin, Spiegel, Lynn, Turvey, Stuart E, Schmeling, Heinrike, Lang, Bianca, Ellsworth, Janet, Ramsey, Suzanne E, Bruns, Alessandra, Roth, Johannes, Campillo, Sarah, Benseler, Susanne, Chédeville, Gaëlle, Schneider, Rayfel, Tse, Shirley M L, Bolaria, Roxana, Gross, Katherine, Feldman, Brian, Feldman, Debbie, Cameron, Bonnie, Jurencak, Roman, Dorval, Jean, LeBlanc, Claire, St. Cyr, Claire, Gibbon, Michele, Yeung, Rae S M, Duffy, Ciarán M, and Tucker, Lori B

Published
2016
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2. Supplement to: Activated STING in a vascular and pulmonary syndrome.

Author

Liu, Yin, Jesus, Adriana A., Marrero, Bernadette, Yang, Dan, Ramsey, Suzanne E., Montealegre Sanchez, Gina A., Tenbrock, Klaus, Wittkowski, Helmut, Jones, Olcay Y., Kuehn, Hye Sun, Lee, Chyi-Chia R., DiMattia, Michael A., Cowen, Edward W., Gonzalez, Benito, Palmer, Ira, DiGiovanna, John J., Biancotto, Angelique, Kim, Hanna, Tsai, Wanxia L., Trier, Anna M., Huang, Yan, Stone, Deborah L., Hill, Suvimol, Kim, Jeffery H., Hilaire, Cynthia St., Gurprasad, Shakuntala, Plass, Nicole, Chapelle, Dawn, Horkayne-Szakaly, Iren, Foell, Dirk, Barysenka, Andrei, Candotti, Fabio, Holland, Steven M., Hughes, Jason D., Mehmet, Huseyin, Issekutz, Andrew C., Raffeld, Mark, McElwee, Joshua, Fontana, Joseph R., Minniti, Caterina P., Moir, Susan, Kastner, Daniel L., Gadina, Massimo, Steven, Alasdair C., Wingfield, Paul T., Brooks, Stephen R., Rosenzweig, Sergio D., Fleisher, Thomas A., Deng, Zuoming, Boehm, Manfred, Paller, Amy S., and Goldbach-Mansky, Raphaela

Published
2014

3. Higher concentrations of vitamin D in Canadian children with juvenile idiopathic arthritis compared to healthy controls are associated with more frequent use of vitamin D supplements and season of birth

Author

Finch, Sarah L., primary, Rosenberg, Alan M., additional, Kusalik, Anthony J., additional, Maleki, Farhad, additional, Rezaei, Elham, additional, Baxter-Jones, Adam, additional, Benseler, Susanne, additional, Boire, Gilles, additional, Cabral, David, additional, Campillo, Sarah, additional, Chédeville, Gaëlle, additional, Chetaille, Anne-Laure, additional, Dancey, Paul, additional, Duffy, Ciaran, additional, Duffy, Karen Watanabe, additional, Guzman, Jaime, additional, Houghton, Kristin, additional, Huber, Adam M., additional, Jurencak, Roman, additional, Lang, Bianca, additional, Laxer, Ron M., additional, Morishita, Kimberly, additional, Oen, Kiem G., additional, Petty, Ross E., additional, Ramsey, Suzanne E., additional, Roth, Johannes, additional, Schneider, Rayfel, additional, Scuccimarri, Rosie, additional, Stringer, Elizabeth, additional, Tse, Shirley M.L., additional, Tucker, Lori B., additional, Turvey, Stuart E., additional, Szafron, Michael, additional, Whiting, Susan, additional, Yeung, Rae SM, additional, and Vatanparast, Hassan, additional

Published
2021
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4. Additional file 1 of Clinical and psychosocial stress factors are associated with decline in physical activity over time in children with juvenile idiopathic arthritis

Author

Heale, Liane D., Houghton, Kristin M., Rezaei, Elham, Baxter-Jones, Adam D. G., Tupper, Susan M., Muhajarine, Nazeem, Benseler, Susanne M., Boire, Gilles, Cabral, David A., Campillo, Sarah, Chédeville, Gaëlle, Chetaille, Anne-Laure, Dancey, Paul, Duffy, Ciaran, Duffy, Karen Watanabe, Ellsworth, Janet, Guzman, Jaime, Huber, Adam M., Jurencak, Roman, Lang, Bianca, Laxer, Ronald M., Morishita, Kimberly, Oen, Kiem G., Petty, Ross E., Ramsey, Suzanne E., Roth, Johannes, Schneider, Rayfel, Scuccimarri, Rosie, Spiegel, Lynn, Stringer, Elizabeth, Tse, Shirley M. L., Tucker, Lori B., Turvey, Stuart E., Yeung, Rae S. M., and Rosenberg, Alan M.

Subjects
Data_FILES
Abstract

Additional file 1.

Published
2021
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5. Activated STING in a Vascular and Pulmonary Syndrome

Author

Liu, Yin, Jesus, Adriana A., Marrero, Bernadette, Yang, Dan, Ramsey, Suzanne E., Montealegre Sanchez, Gina A., Tenbrock, Klaus, Wittkowski, Helmut, Jones, Olcay Y., Kuehn, Hye Sun, Lee, Chyi-Chia R., DiMattia, Michael A., Cowen, Edward W., Gonzalez, Benito, Palmer, Ira, DiGiovanna, John J., Biancotto, Angelique, Kim, Hanna, Tsai, Wanxia L., Trier, Anna M., Huang, Yan, Stone, Deborah L., Hill, Suvimol, Kim, Jeffery H., Hilaire, Cynthia St., Gurprasad, Shakuntala, Plass, Nicole, Chapelle, Dawn, Horkayne-Szakaly, Iren, Foell, Dirk, Barysenka, Andrei, Candotti, Fabio, Holland, Steven M., Hughes, Jason D., Mehmet, Huseyin, Issekutz, Andrew C., Raffeld, Mark, McElwee, Joshua, Fontana, Joseph R., Minniti, Caterina P., Moir, Susan, Kastner, Daniel L., Gadina, Massimo, Steven, Alasdair C., Wingfield, Paul T., Brooks, Stephen R., Rosenzweig, Sergio D., Fleisher, Thomas A., Deng, Zuoming, Boehm, Manfred, Paller, Amy S., and Goldbach-Mansky, Raphaela

Published
2014
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6. Clinical and Psychosocial Stress Factors are Associated with Decline in Physical Activity Over Time in Children with Juvenile Idiopathic Arthritis

Author

Heale, Liane D, primary, Houghton, Kristin, additional, Rezaei, Elham, additional, Baxter-Jones, Adam D.G., additional, Tupper, Susan M, additional, Muhajarine, Nazeem, additional, Benseler, Susanne M, additional, Boire, Gilles, additional, Cabral, David A, additional, Campillo, Sarah, additional, Chedeville, Gaelle, additional, Chetaille, Anne-Laure, additional, Dancey, Paul, additional, Duffy, Ciaran, additional, Duffy, Karen Watanabe, additional, Ellsworth, Janet, additional, Guzman, Jaime, additional, Huber, Adam M, additional, Jurencak, Roman, additional, Lang, Bianca, additional, Laxer, Ronald M, additional, Morishita, Kimberly, additional, Oen, Keim G, additional, Petty, Ross E, additional, Ramsey, Suzanne E, additional, Roth, Johannes, additional, Schneider, Rayfel, additional, Scuccimarri, Rosie, additional, Spiegel, Lynn, additional, Stringer, Elizabeth, additional, Tse, Shirley M.L., additional, Tucker, Lori B, additional, Turvey, Stuart E, additional, Yeung, Rae S.M., additional, and Rosenberg, Alan M, additional

Published
2021
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7. Clinical and associated inflammatory biomarker features predictive of short-term outcomes in non-systemic juvenile idiopathic arthritis

Author

Rezaei, Elham, primary, Hogan, Daniel, additional, Trost, Brett, additional, Kusalik, Anthony J, additional, Boire, Gilles, additional, Cabral, David A, additional, Campillo, Sarah, additional, Chédeville, Gaëlle, additional, Chetaille, Anne-Laure, additional, Dancey, Paul, additional, Duffy, Ciaran, additional, Watanabe Duffy, Karen, additional, Gordon, John, additional, Guzman, Jaime, additional, Houghton, Kristin, additional, Huber, Adam M, additional, Jurencak, Roman, additional, Lang, Bianca, additional, Morishita, Kimberly, additional, Oen, Kiem G, additional, Petty, Ross E, additional, Ramsey, Suzanne E, additional, Scuccimarri, Rosie, additional, Spiegel, Lynn, additional, Stringer, Elizabeth, additional, Taylor-Gjevre, Regina M, additional, Tse, Shirley M L, additional, Tucker, Lori B, additional, Turvey, Stuart E, additional, Tupper, Susan, additional, Yeung, Rae S M, additional, Benseler, Susanne, additional, Ellsworth, Janet, additional, Guillet, Chantal, additional, Karananayake, Chandima, additional, Muhajarine, Nazeem, additional, Roth, Johannes, additional, Schneider, Rayfel, additional, and Rosenberg, Alan M, additional

Published
2020
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8. Health-Related Quality of Life in an Inception Cohort of Children With Juvenile Idiopathic Arthritis: A Longitudinal Analysis

Author

Oen, Kiem, Guzman, Jaime, Dufault, Brenden, Tucker, Lori B, Shiff, Natalie J, Duffy, Karen Watanabe, Lee, Jennifer J Y, Feldman, Brian M, Berard, Roberta A, Dancey, Paul, Huber, Adam M, Scuccimarri, Rosie, Cabral, David A, Morishita, Kimberly A, Ramsey, Suzanne E, Rosenberg, Alan M, Boire, Gilles, Benseler, Susanne M, Lang, Bianca, Houghton, Kristin, Miettunen, Paivi M, Chédeville, Gaëlle, Levy, Deborah M, Bruns, Alessandra, Schmeling, Heinrike, Haddad, Elie, Yeung, Rae S M, Duffy, Ciarán M, and The Research in Arthritis in Canadian Children emphasizing Outcomes (ReACCh-Out) investigators

Subjects
Male, Pediatrics, Time Factors, Arthritis, Child Behavior, Disability Evaluation, 0302 clinical medicine, Child Development, Quality of life, Surveys and Questionnaires, Medicine, longitudinal studies, Longitudinal Studies, 030212 general & internal medicine, Child, Pain Measurement, Oligoarthritis, adolescent development, Age Factors, Prognosis, humanities, female, arthritis, Predictive value of tests, Child, Preschool, Cohort, Female, Polyarthritis, medicine.medical_specialty, age factors, Canada, Adolescent, disability evaluation, adolescent behavior, pain measurement, preschool, 03 medical and health sciences, Rheumatology, Predictive Value of Tests, Humans, Survival analysis, 030203 arthritis & rheumatology, business.industry, Adolescent Development, medicine.disease, Child development, Arthritis, Juvenile, juvenile, quality of life, Adolescent Behavior, Quality of Life, business
Abstract

Objective To describe changes in health-related quality of life (HRQoL) over time in children with juvenile idiopathic arthritis (JIA), relative to other outcomes, and to identify predictors of unfavorable HRQoL trajectories. Methods Children with JIA in the Research in Arthritis in Canadian Children emphasizing Outcomes (ReACCh-Out) cohort were included. The Juvenile Arthritis Quality of Life Questionnaire (JAQQ, a standardized instrument), health-related Quality of My Life (HRQoML, an instrument based on personal valuations), and JIA core variables were completed serially. Analyses included median values, Kaplan-Meier survival curves, and latent trajectory analysis. Results A total of 1,249 patients enrolled at a median of 0.5 months after diagnosis were followed for a median of 34.2 months. The degree of initial HRQoL impairment and probabilities of reaching the best possible HRQoL scores varied across JIA categories (best for oligoarthritis, worst for rheumatoid factor–positive polyarthritis). Median times to attain best possible HRQoL scores (JAQQ 59.3 months, HRQoML 34.5 months), lagged behind those for disease activity, pain, and disability measures. Most patients followed trajectories with minimal or mild impairment; however, 7.6% and 13.8% of patients, respectively, followed JAQQ and HRQoML trajectories with persistent major impairment in HRQoL. JIA category, aboriginal ethnicity, and baseline disease activity measures distinguished between membership in trajectories with major and minimal impairments. Conclusion Improvement in HRQoL is slower than for disease activity, pain, and disability. Improvement of a measure based on respondents’ preferences (HRQoML) is more rapid than that of a standardized measure (JAQQ). Higher disease activity at diagnosis heralds an unfavorable HRQoL trajectory.

Published
2018

10. Soluble Low-density Lipoprotein Receptor-related Protein 1 in Juvenile Idiopathic Arthritis

Author

Rezaei, Elham, Newkirk, Marianna M., Li, Zhenhong, Gordon, John R., Oen, Kiem G., Benseler, Susanne M., Boire, Gilles, Cabral, David A., Campillo, Sarah, Chédeville, Gae¨lle, Chetaille, Anne-Laure, Dancey, Paul, Duffy, Ciaran, Duffy, Karen Watanabe, Houghton, Kristin, Huber, Adam M., Jurencak, Roman, Lang, Bianca, Morishita, Kimberly A., Petty, Ross E., Ramsey, Suzanne E., Roth, Johannes, Schneider, Rayfel, Scuccimarri, Rosie, Spiegel, Lynn, Stringer, Elizabeth, Tse, Shirley M.L., Tucker, Lori B., Turvey, Stuart E., Yeung, Rae S.M., and Rosenberg, Alan M.

Abstract

ObjectivesThis study aimed to expand knowledge about soluble low-density lipoprotein receptor-related protein 1 (sLRP1) in juvenile idiopathic arthritis (JIA) by determining associations of sLRP1 levels in nonsystemic JIA patients with clinical and inflammatory biomarker indicators of disease activity.MethodsPlasma sLRP1 and 44 inflammation-related biomarkers were measured at enrollment and 6 months later in a cohort of 96 newly diagnosed Canadian patients with nonsystemic JIA. Relationships between sLRP1 levels and indicators of disease activity and biomarker levels were analyzed at both visits.ResultsAt enrollment, sLRP1 levels correlated negatively with age and active joint counts. Children showed significantly higher levels of sLRP1 than adolescents (mean ranks: 55.4 and 41.9, respectively; P= 0.02). Participants with 4 or fewer active joints, compared to those with 5 or more active joints, had significantly higher sLRP1 levels (mean ranks: 56.2 and 40.7, respectively; P= 0.006). At enrollment, considering the entire cohort, sLRP1 correlated negatively with the number of active joints (r = –0.235, P= 0.017). In the entire cohort, sLRP1 levels at enrollment and 6 months later correlated with 13 and 6 pro- and antiinflammatory biomarkers, respectively. In JIA categories, sLRP1 correlations with inflammatory markers were significant in rheumatoid factor–negative polyarticular JIA, oligoarticular JIA, enthesitis-related arthritis, and psoriatic arthritis at enrollment. Higher sLRP1 levels at enrollment increased the likelihood of absence of active joints 6 months later.ConclusionPlasma sLRP1 levels correlate with clinical and biomarker indicators of short-term improvement in JIA disease activity, supporting sLRP1 as an upstream biomarker of potential utility for assessing JIA disease activity and outcome prediction.

Published
2021
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11. Growth and weight gain in children with juvenile idiopathic arthritis: results from the ReACCh-Out cohort

Author

Guzman, Jaime, primary, Kerr, Tristan, additional, Ward, Leanne M., additional, Ma, Jinhui, additional, Oen, Kiem, additional, Rosenberg, Alan M., additional, Feldman, Brian M., additional, Boire, Gilles, additional, Houghton, Kristin, additional, Dancey, Paul, additional, Scuccimarri, Rosie, additional, Bruns, Alessandra, additional, Huber, Adam M., additional, Watanabe Duffy, Karen, additional, Shiff, Natalie J., additional, Berard, Roberta A., additional, Levy, Deborah M., additional, Stringer, Elizabeth, additional, Morishita, Kimberly, additional, Johnson, Nicole, additional, Cabral, David A., additional, Larché, Maggie, additional, Petty, Ross E., additional, Laxer, Ronald M., additional, Silverman, Earl, additional, Miettunen, Paivi, additional, Chetaille, Anne-Laure, additional, Haddad, Elie, additional, Spiegel, Lynn, additional, Turvey, Stuart E., additional, Schmeling, Heinrike, additional, Lang, Bianca, additional, Ellsworth, Janet, additional, Ramsey, Suzanne E., additional, Roth, Johannes, additional, Campillo, Sarah, additional, Benseler, Susanne, additional, Chédeville, Gaëlle, additional, Schneider, Rayfel, additional, Tse, Shirley M. L., additional, Bolaria, Roxana, additional, Gross, Katherine, additional, Feldman, Debbie, additional, Cameron, Bonnie, additional, Jurencak, Roman, additional, Dorval, Jean, additional, LeBlanc, Claire, additional, St. Cyr, Claire, additional, Gibbon, Michele, additional, Yeung, Rae S. M., additional, Duffy, Ciarán M., additional, and Tucker, Lori B., additional

Published
2017
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12. Real‐World Effectiveness of Common Treatment Strategies for Juvenile Idiopathic Arthritis: Results From a Canadian Cohort

Author

Chhabra, Amieleena, Oen, Kiem, Huber, Adam M., Shiff, Natalie J., Boire, Gilles, Benseler, Susanne M., Berard, Roberta A., Scuccimarri, Rosie, Feldman, Brian M., Lim, Lily Siok Hoon, Barsalou, Julie, Bruns, Alessandra, Cabral, David A., Chédeville, Gaëlle, Ellsworth, Janet, Houghton, Kristin, Lang, Bianca, Morishita, Kimberly, Rumsey, Dax G., Rosenberg, Alan M., Tse, Shirley M., Watanabe Duffy, Karen, Duffy, Ciaran M., Guzman, Jaime, Bolaria, Roxana, Gross, Katherine, Turvey, Stuart E., Chan, Mercedes, Tucker, Lori B., Petty, Ross, Johnson, Nicole, Luca, Nadia, Miettunen, Paivi, Schmeling, Heinrike, Gerhold, Kerstin, Larché, Maggie, Levy, Deborah M., Laxer, Ronald M., Feldman, Debbie, Spiegel, Lynn, Schneider, Rayfel, Silverman, Earl, Cameron, Bonnie, Yeung, Rae S. M., Roth, Johannes, Jurencak, Roman, Gibbon, Michele, Chetaille, Anne‐Laure, Dorval, Jean, Campillo, Sarah, LeBlanc, Claire, Chédeville, Gaëlle, Haddad, Elie, Cyr, Claire St., Ramsey, Suzanne E., Stringer, Elizabeth, and Dancey, Paul

Abstract

Undervaluing the effectiveness of conventional treatments may lead to overtreatment with biologic medications in children with juvenile idiopathic arthritis (JIA). Using data from a nationwide inception cohort and strict methods to control bias, the aim of our study was to estimate the real‐world effectiveness of simple JIAtreatment strategies recommended in current guidelines. Children with JIAwho were recruited at 16 Canadian centers from 2005 to 2010 were followed for up to 5 years. For each child, all observed treatment changes over time were assessed by independent physicians using prospectively collected data and published response criteria. Success was defined as attainment of inactive disease or maintenance of this state when stepping down treatment; minimally active disease was deemed acceptable for children with polyarticular JIA. Success rates were calculated for treatments tried ≥25 times, and logistic regression analysis identified features associated with success. A total of 4,429 treatment episodes were observed in 1,352 children. Nonsteroidal antiinflammatory drug (NSAID) monotherapy was attempted 697 times, mostly as initial treatment when <5 joints were involved, with a 54.4% success rate (95% confidence interval [95% CI] 50.3–58.6). NSAIDs plus joint injections had a 64.7% success rate (95% CI59.8–69.7). Adding methotrexate to NSAIDs and/or joint injections (attempted 566 times) had a 60.5% success rate (95% CI55.7–65.3). In adjusted analyses, each additional active joint reduced chances of success for treatment with NSAIDs (odds ratio [OR] 0.90 [95% CI0.85–0.94]) and for methotrexate combinations (OR0.96 [95% CI0.94–0.99]). Each additional year after disease onset reduced chances of success for treatment with methotrexate combinations (OR0.83 [95% CI0.72–0.95]). These real‐world effectiveness estimates show that conventional nonbiologic treatment strategies that are recommended in current guidelines are effective in achieving treatment targets in many children with JIA.

Published
2020
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13. The risk and nature of flares in juvenile idiopathic arthritis: results from the ReACCh-Out cohort

Author

Guzman, Jaime, primary, Oen, Kiem, additional, Huber, Adam M, additional, Watanabe Duffy, Karen, additional, Boire, Gilles, additional, Shiff, Natalie, additional, Berard, Roberta A, additional, Levy, Deborah M, additional, Stringer, Elizabeth, additional, Scuccimarri, Rosie, additional, Morishita, Kimberly, additional, Johnson, Nicole, additional, Cabral, David A, additional, Rosenberg, Alan M, additional, Larché, Maggie, additional, Dancey, Paul, additional, Petty, Ross E, additional, Laxer, Ronald M, additional, Silverman, Earl, additional, Miettunen, Paivi, additional, Chetaille, Anne-Laure, additional, Haddad, Elie, additional, Houghton, Kristin, additional, Spiegel, Lynn, additional, Turvey, Stuart E, additional, Schmeling, Heinrike, additional, Lang, Bianca, additional, Ellsworth, Janet, additional, Ramsey, Suzanne E, additional, Bruns, Alessandra, additional, Roth, Johannes, additional, Campillo, Sarah, additional, Benseler, Susanne, additional, Chédeville, Gaëlle, additional, Schneider, Rayfel, additional, Tse, Shirley M L, additional, Bolaria, Roxana, additional, Gross, Katherine, additional, Feldman, Brian, additional, Feldman, Debbie, additional, Cameron, Bonnie, additional, Jurencak, Roman, additional, Dorval, Jean, additional, LeBlanc, Claire, additional, St. Cyr, Claire, additional, Gibbon, Michele, additional, Yeung, Rae S M, additional, Duffy, Ciarán M, additional, and Tucker, Lori B, additional

Published
2015
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14. Do Adult Disease Severity Subclassifications Predict Use of Cyclophosphamide in Children with ANCA-associated Vasculitis? An Analysis of ARChiVe Study Treatment Decisions

Author

MORISHITA, KIMBERLY, primary, GUZMAN, JAIME, additional, CHIRA, PETER, additional, MUSCAL, EYAL, additional, ZEFT, ANDREW, additional, KLEIN-GITELMAN, MARISA, additional, URIBE, AMERICA G., additional, ABRAMSON, LESLIE, additional, BENSELER, SUSANNE M., additional, EBERHARD, ANNE, additional, EDE, KALEO, additional, HASHKES, PHILIP J., additional, HERSH, AIMEE O., additional, HIGGINS, GLORIA, additional, IMUNDO, LISA F., additional, JUNG, LAWRENCE, additional, KIM, SUSAN, additional, KINGSBURY, DANIEL J., additional, LAWSON, ERICA F., additional, LEE, TZIELAN, additional, LI, SUZANNE C., additional, LOVELL, DANIEL J., additional, MASON, THOMAS, additional, McCURDY, DEBORAH, additional, O’NEIL, KATHLEEN M., additional, PUNARO, MARILYNN, additional, RAMSEY, SUZANNE E., additional, REIFF, ANDREAS, additional, ROSENKRANZ, MARGALIT, additional, SCHIKLER, KENNETH N., additional, SCUCCIMARRI, ROSIE, additional, SINGER, NORA G., additional, STEVENS, ANNE M., additional, van MATER, HEATHER, additional, WAHEZI, DAWN M., additional, WHITE, ANDREW J., additional, and CABRAL, DAVID A., additional

Published
2012
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15. Assessing the Performance of the Birmingham Vasculitis Activity Score at Diagnosis for Children with Antineutrophil Cytoplasmic Antibody-associated Vasculitis in A Registry for Childhood Vasculitis (ARChiVe)

Author

MORISHITA, KIMBERLY, primary, LI, SUZANNE C., additional, MUSCAL, EYAL, additional, SPALDING, STEVEN, additional, GUZMAN, JAIME, additional, URIBE, AMERICA, additional, ABRAMSON, LESLIE, additional, BASZIS, KEVIN, additional, BENSELER, SUSANNE, additional, BOWYER, SUZANNE, additional, CAMPILLO, SARAH, additional, CHIRA, PETER, additional, HERSH, AIMEE O., additional, HIGGINS, GLORIA, additional, EBERHARD, ANNE, additional, EDE, KALEO, additional, IMUNDO, LISA, additional, JUNG, LAWRENCE, additional, KIM, SUSAN, additional, KINGSBURY, DANIEL J., additional, KLEIN-GITELMAN, MARISA, additional, LAWSON, ERICA F., additional, LOVELL, DANIEL J., additional, MASON, THOMAS, additional, McCURDY, DEBORAH, additional, NANDA, KABITA, additional, NASSI, LORIEN, additional, O’NEIL, KATHLEEN M., additional, RABINOVICH, EGLA, additional, RAMSEY, SUZANNE E., additional, REIFF, ANDREAS, additional, ROSENKRANZ, MARGALIT, additional, SCHIKLER, KENNETH, additional, STEVENS, ANNE, additional, WAHEZI, DAWN, additional, and CABRAL, DAVID A., additional

Published
2012
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17. Growth and weight gain in children with juvenile idiopathic arthritis: results from the ReACCh-Out cohort

Author

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, and Tucker, Lori B

Subjects
2. Zero hunger
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.

18. Case of the Month #144.

Author

MacDougall, Ryan F., Ramsey, Suzanne E., and Schmidt, Matthias H.

Subjects
*JUVENILE diseases, *MYALGIA, *FEVER, *DIAGNOSIS, *MAGNETIC resonance imaging, *PATIENTS
Abstract

The article invites readers to discuss the clinical case of a boy suffering from a low-grade fever, migratory myalgia, and progressive proximal muscle weakness over a period of 1 month. The patients' physical examination revealed weakness of the trunk and proximal extremities, multiple joint effusions, and a heliotrope rash over the right upper eyelid. Magnetic resonance imaging scan was performed on the patient. Images of the scan are also provided.

Published
2008

19. Clinical and psychosocial stress factors are associated with decline in physical activity over time in children with juvenile idiopathic arthritis.

Author

Heale LD, Houghton KM, Rezaei E, Baxter-Jones ADG, Tupper SM, Muhajarine N, Benseler SM, Boire G, Cabral DA, Campillo S, Chédeville G, Chetaille AL, Dancey P, Duffy C, Duffy KW, Ellsworth J, Guzman J, Huber AM, Jurencak R, Lang B, Laxer RM, Morishita K, Oen KG, Petty RE, Ramsey SE, Roth J, Schneider R, Scuccimarri R, Spiegel L, Stringer E, Tse SML, Tucker LB, Turvey SE, Yeung RSM, and Rosenberg AM

Subjects
Adolescent, Child, Cohort Studies, Female, Humans, Longitudinal Studies, Male, Time Factors, Arthritis, Juvenile complications, Arthritis, Juvenile psychology, Exercise, Stress, Psychological etiology
Abstract

Background: Physical activity (PA) patterns in children with juvenile idiopathic arthritis (JIA) over time are not well described. The aim of this study was to describe associations of physical activity (PA) with disease activity, function, pain, and psychosocial stress in the 2 years following diagnosis in an inception cohort of children with juvenile idiopathic arthritis (JIA)., Methods: In 82 children with newly diagnosed JIA, PA levels, prospectively determined at enrollment, 12 and 24 months using the Physical Activity Questionnaire for Children (PAQ-C) and Adolescents (PAQ-A) raw scores, were evaluated in relation to disease activity as reflected by arthritis activity (Juvenile Arthritis Disease Activity Score (JADAS-71)), function, pain, and psychosocial stresses using a linear mixed model approach. Results in the JIA cohort were compared to normative Pediatric Bone Mineral Accrual Study data derived from healthy children using z-scores., Results: At enrollment, PA z-score levels of study participants were lower than those in the normative population (median z-score - 0.356; p = 0.005). At enrollment, PA raw scores were negatively associated with the psychosocial domain of the Juvenile Arthritis Quality of Life Questionnaire (r = - 0.251; p = 0.023). There was a significant decline in PAQ-C/A raw scores from baseline (median and IQR: 2.6, 1.4-3.1) to 24 months (median and IQR: 2.1, 1.4-2.7; p = 0.003). The linear mixed-effect model showed that PAQ-C/A raw scores in children with JIA decreased as age, disease duration, and ESR increased. The PAQ-C/A raw scores of the participants was also negatively influenced by an increase in disease activity as measured by the JADAS-71 (p <  0.001)., Conclusion: Canadian children with newly diagnosed JIA have lower PA levels than healthy children. The decline in PA levels over time was associated with disease activity and higher disease-specific psychosocial stress.

Published
2021
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20. Associations of clinical and inflammatory biomarker clusters with juvenile idiopathic arthritis categories.

Author

Rezaei E, Hogan D, Trost B, Kusalik AJ, Boire G, Cabral DA, Campillo S, Chédeville G, Chetaille AL, Dancey P, Duffy C, Duffy KW, Eng SWM, Gordon J, Guzman J, Houghton K, Huber AM, Jurencak R, Lang B, Laxer RM, Morishita K, Oen KG, Petty RE, Ramsey SE, Scherer SW, Scuccimarri R, Spiegel L, Stringer E, Taylor-Gjevre RM, Tse SML, Tucker LB, Turvey SE, Tupper S, Wintle RF, Yeung RSM, and Rosenberg AM

Subjects
Adolescent, Age Factors, Arthritis, Juvenile epidemiology, Biomarkers blood, Canada epidemiology, Child, Cluster Analysis, Cohort Studies, Data Mining, Female, Humans, Incidence, Male, Normal Distribution, Prospective Studies, Risk Assessment, Severity of Illness Index, Sex Factors, Syndrome, Arthritis, Juvenile blood, Arthritis, Juvenile physiopathology, Inflammation Mediators blood
Abstract

Objective: To identify discrete clusters comprising clinical features and inflammatory biomarkers in children with JIA and to determine cluster alignment with JIA categories., Methods: A Canadian prospective inception cohort comprising 150 children with JIA was evaluated at baseline (visit 1) and after six months (visit 2). Data included clinical manifestations and inflammation-related biomarkers. Probabilistic principal component analysis identified sets of composite variables, or principal components, from 191 original variables. To discern new clinical-biomarker clusters (clusters), Gaussian mixture models were fit to the data. Newly-defined clusters and JIA categories were compared. Agreement between the two was assessed using Kruskal-Wallis analyses and contingency plots., Results: Three principal components recovered 35% (three clusters) and 40% (five clusters) of the variance in patient profiles in visits 1 and 2, respectively. None of the clusters aligned precisely with any of the seven JIA categories but rather spanned multiple categories. Results demonstrated that the newly defined clinical-biomarker lustres are more homogeneous than JIA categories., Conclusion: Applying unsupervised data mining to clinical and inflammatory biomarker data discerns discrete clusters that intersect multiple JIA categories. Results suggest that certain groups of patients within different JIA categories are more aligned pathobiologically than their separate clinical categorizations suggest. Applying data mining analyses to complex datasets can generate insights into JIA pathogenesis and could contribute to biologically based refinements in JIA classification., (© The Author(s) 2019. Published by Oxford University Press on behalf of the British Society for Rheumatology. All rights reserved. For permissions, please email: journals.permissions@oup.com.)

Published
2020
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21. Health-Related Quality of Life in an Inception Cohort of Children With Juvenile Idiopathic Arthritis: A Longitudinal Analysis.

Author

Oen K, Guzman J, Dufault B, Tucker LB, Shiff NJ, Duffy KW, Lee JJY, Feldman BM, Berard RA, Dancey P, Huber AM, Scuccimarri R, Cabral DA, Morishita KA, Ramsey SE, Rosenberg AM, Boire G, Benseler SM, Lang B, Houghton K, Miettunen PM, Chédeville G, Levy DM, Bruns A, Schmeling H, Haddad E, Yeung RSM, and Duffy CM

Subjects
Adolescent, Adolescent Development, Age Factors, Arthritis, Juvenile physiopathology, Arthritis, Juvenile therapy, Canada, Child, Child Development, Child, Preschool, Disability Evaluation, Female, Humans, Longitudinal Studies, Male, Pain Measurement, Predictive Value of Tests, Prognosis, Time Factors, Adolescent Behavior, Arthritis, Juvenile diagnosis, Arthritis, Juvenile psychology, Child Behavior, Quality of Life, Surveys and Questionnaires
Abstract

Objective: To describe changes in health-related quality of life (HRQoL) over time in children with juvenile idiopathic arthritis (JIA), relative to other outcomes, and to identify predictors of unfavorable HRQoL trajectories., Methods: Children with JIA in the Research in Arthritis in Canadian Children emphasizing Outcomes (ReACCh-Out) cohort were included. The Juvenile Arthritis Quality of Life Questionnaire (JAQQ, a standardized instrument), health-related Quality of My Life (HRQoML, an instrument based on personal valuations), and JIA core variables were completed serially. Analyses included median values, Kaplan-Meier survival curves, and latent trajectory analysis., Results: A total of 1,249 patients enrolled at a median of 0.5 months after diagnosis were followed for a median of 34.2 months. The degree of initial HRQoL impairment and probabilities of reaching the best possible HRQoL scores varied across JIA categories (best for oligoarthritis, worst for rheumatoid factor-positive polyarthritis). Median times to attain best possible HRQoL scores (JAQQ 59.3 months, HRQoML 34.5 months), lagged behind those for disease activity, pain, and disability measures. Most patients followed trajectories with minimal or mild impairment; however, 7.6% and 13.8% of patients, respectively, followed JAQQ and HRQoML trajectories with persistent major impairment in HRQoL. JIA category, aboriginal ethnicity, and baseline disease activity measures distinguished between membership in trajectories with major and minimal impairments., Conclusion: Improvement in HRQoL is slower than for disease activity, pain, and disability. Improvement of a measure based on respondents' preferences (HRQoML) is more rapid than that of a standardized measure (JAQQ). Higher disease activity at diagnosis heralds an unfavorable HRQoL trajectory., (© 2017, American College of Rheumatology.)

Published
2018
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22. Corticosteroid treatment of refractory Kawasaki disease.

Author

Lang BA, Yeung RS, Oen KG, Malleson PN, Huber AM, Riley M, Ebbeson R, Ramsey SE, Laxer RM, Silverman ED, McCrindle BW, Ratnapalan S, and Feldman BM

Subjects
Child, Child, Preschool, Female, Fever drug therapy, Humans, Infant, Injections, Intravenous, Male, Mucocutaneous Lymph Node Syndrome diagnosis, Mucocutaneous Lymph Node Syndrome physiopathology, Recurrence, Retrospective Studies, Treatment Failure, Treatment Outcome, Glucocorticoids therapeutic use, Methylprednisolone therapeutic use, Mucocutaneous Lymph Node Syndrome drug therapy, Pediatrics methods
Abstract

Objective: To review the indications for corticosteroids in patients with Kawasaki disease (KD) treated by pediatric rheumatologists in Canada and to determine their efficacy on fever in patients with refractory KD., Methods: All practicing pediatric rheumatologists in Canada identified KD patients treated with corticosteroids and completed a standard data form that included demographics, clinical and laboratory features, imaging studies, and therapeutic interventions, by chart review., Results: Thirty-two patients with KD (14 female; 18 male: mean age 4.6 years) were treated with corticosteroids. Corticosteroids were used in 26 patients (81%) for persistent fever despite treatment with intravenous immunoglobulin (IVIG) (refractory KD), 5 patients (19%) for congestive heart failure, and 1 patient for persistent acute phase symptoms other than fever. The 26 patients with refractory KD are the primary subject of this report. Twenty-two patients (85%) had rapid, sustained resolution of fever after corticosteroids. There were no serious reported adverse effects. Eight patients (31%) treated with corticosteroids developed coronary artery (CA) aneurysms and 9 (35%) developed CA dilatations without aneurysms. Of those who developed CA aneurysm, 4 had aneurysms detected prior to IV methylprednisolone (MP) on echocardiograms performed on days 6-27 (mean day 13) of illness. The remaining 4 patients had CA aneurysm detected after IVMP therapy, on echocardiograms performed on days 13-49 (mean day 23) of illness, 1-25 days (mean 9 days) after IVMP. In patients with one year or more of followup, 46% had resolution of CA abnormalities., Conclusion: Corticosteroids are effective in the treatment of fever in most patients with IVIG-refractory KD. A multicenter prospective study is needed to determine the effect of corticosteroids on CA outcome in patients with refractory KD.

Published
2006

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