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2. The young massive star cluster Westerlund 2 observed with MUSE. I. First results on the cluster internal motion from stellar radial velocities

Author

Zeidler, Peter, Sabbi, Elena, Nota, Antonella, Pasquali, Anna, Grebel, Eva K., McLeod, Anna Faye, Kamann, Sebastian, Tosi, Monica, Cignoni, Michele, and Ramsey, Suzanne

Subjects
Astrophysics - Solar and Stellar Astrophysics, Astrophysics - Astrophysics of Galaxies
Abstract

Westerlund 2 (Wd2) is the central ionizing star cluster of the \ion{H}{2} region RCW~49 and the second most massive young star cluster (${\rm M} = (3.6 \pm 0.3)\times 10^4\,{\rm M}_\odot$) in the Milky Way. Its young age ($\sim2\,$Myr) and close proximity to the Sun ($\sim 4\,$kpc) makes it a perfect target to study stars emerging from their parental gas cloud, the large number of OB-stars and their feedback onto the gas, and the gas dynamics. We combine high-resolution multi-band photometry obtained in the optical and near-infrared with the \textit{Hubble} Space Telescope (HST), and VLT/MUSE integral field spectroscopy to study the gas, the stars, and their interactions, simultaneously. In this paper we focus on a small, $64\times64\,{\rm arcsec}^2$ region North of the main cluster center, which we call the Northern Bubble (NB), a circular cavity carved into the gas of the cluster region. Using MUSE data, we determined the spectral types of 17 stars in the NB from G9III to O7.5. With the estimation of these spectral types we add 2 O and 5 B-type stars to the previously published census of 37 OB-stars in Wd2. To measure radial velocities we extracted 72 stellar spectra throughout Wd2, including the 17 of the NB, and show that the cluster member stars follow a bimodal velocity distribution centered around $(8.10 \pm 1.53)\,{\rm km}\,{\rm s}^{-1}$ and $(25.41 \pm 1.57)\,{\rm km}\,{\rm s}^{-1}$ with a dispersion of $(4.52 \pm 1.78)\,{\rm km}\,{\rm s}^{-1}$ and $(3.46 \pm 1.29)\,{\rm km}\,{\rm s}^{-1}$, respectively. These are in agreement with CO($J=1$-2) studies of RCW~49 leaving cloud-cloud collision as a viable option for the formation scenario of Wd2. The bimodal distribution is also detected in the Gaia DR2 proper motions., Comment: 20 pages, 11 figures, 3 tables

Published
2018
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4. 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., Rosenberg, Alan M., Kusalik, Anthony J., Maleki, Farhad, Rezaei, Elham, Baxter-Jones, Adam, Benseler, Susanne, Boire, Gilles, Cabral, David, Campillo, Sarah, Chédeville, Gaëlle, Chetaille, Anne-Laure, Dancey, Paul, Duffy, Ciaran, Duffy, Karen Watanabe, Guzman, Jaime, Houghton, Kristin, Huber, Adam M., Jurencak, Roman, Lang, Bianca, Laxer, Ron M., Morishita, Kimberly, Oen, Kiem G., Petty, Ross E., Ramsey, Suzanne E., Roth, Johannes, Schneider, Rayfel, Scuccimarri, Rosie, Stringer, Elizabeth, Tse, Shirley M.L., Tucker, Lori B., Turvey, Stuart E., Szafron, Michael, Whiting, Susan, Yeung, Rae SM, and Vatanparast, Hassan

Published
2021
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5. A decade of progress in juvenile idiopathic arthritis treatments and outcomes in Canada: results from ReACCh-Out and the CAPRI registry

Author

Nguyen, Kelly, primary, Barsalou, Julie, additional, Basodan, Daniah, additional, Batthish, Michelle, additional, Benseler, Susanne M, additional, Berard, Roberta A, additional, Blanchette, Nicholas, additional, Boire, Gilles, additional, Bolaria, Roxana, additional, Bruns, Alessandra, additional, Cabral, David A, additional, Cameron, Bonnie, additional, Campillo, Sarah, additional, Cellucci, Tania, additional, Chan, Mercedes, additional, Chédeville, Gaëlle, additional, Chetaille, Anne-Laure, additional, Chhabra, Amieleena, additional, Couture, Julie, additional, Dancey, Paul, additional, De Bruycker, Jean-Jacques, additional, Demirkaya, Erkan, additional, Dhalla, Muhammed, additional, Duffy, Ciarán M, additional, Feldman, Brian M, additional, Feldman, Debbie E, additional, Gerschman, Tommy, additional, Haddad, Elie, additional, Heale, Liane, additional, Herrington, Julie, additional, Houghton, Kristin, additional, Huber, Adam M, additional, Human, Andrea, additional, Johnson, Nicole, additional, Jurencak, Roman, additional, Lang, Bianca, additional, Larché, Maggie, additional, Laxer, Ronald M, additional, LeBlanc, Claire M, additional, Lee, Jennifer J Y, additional, Levy, Deborah M, additional, Lim, Lillian, additional, Lim, Lily S H, additional, Luca, Nadia, additional, McGrath, Tara, additional, McMillan, Tamara, additional, Miettunen, Paivi M, additional, Morishita, Kimberly A, additional, Ng, Hon Yan, additional, Oen, Kiem, additional, Park, Jonathan, additional, Petty, Ross E, additional, Proulx-Gauthier, Jean-Philippe, additional, Ramsey, Suzanne, additional, Roth, Johannes, additional, Rosenberg, Alan M, additional, Rozenblyum, Evelyn, additional, Rumsey, Dax G, additional, Schmeling, Heinrike, additional, Schneider, Rayfel, additional, Scuccimarri, Rosie, additional, Shiff, Natalie J, additional, Silverman, Earl, additional, Soon, Gordon, additional, Spiegel, Lynn, additional, Stringer, Elizabeth, additional, Tam, Herman, additional, Tse, Shirley M, additional, Tucker, Lori B, additional, Turvey, Stuart, additional, Twilt, Marinka, additional, Duffy, Karen Watanabe, additional, Yeung, Rae S M, additional, and Guzman, Jaime, additional

Published
2023
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7. Trajectories of pain severity in juvenile idiopathic arthritis: results from the Research in Arthritis in Canadian Children Emphasizing Outcomes cohort

Author

Shiff, Natalie J., Tupper, Susan, Oen, Kiem, Guzman, Jaime, Lim, Hyun, Lee, Chel Hee, Bryce, Rhonda, Huber, Adam M., Boire, Gilles, Dancey, Paul, Feldman, Brian, Laxer, Ronald, Miettunen, Paivi, Schmeling, Heinrike, Watanabe Duffy, Karen, Levy, Deborah M., Turvey, Stuart, Bolaria, Roxana, Bruns, Alessandra, Cabral, David A., Campillo, Sarah, Chédeville, Gaëlle, Feldman, Debbie Ehrmann, Haddad, Elie, Houghton, Kristin, Johnson, Nicole, Jurencak, Roman, Lang, Bianca, Larche, Maggie, Morishita, Kimberly, Ramsey, Suzanne, Roth, Johannes, Schneider, Rayfel, Scuccimarri, Rosie, Spiegel, Lynn, Stringer, Elizabeth, Tse, Shirley M., Yeung, Rae, Duffy, Ciarán M., and Tucker, Lori B.

Published
2018
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8. Differentially Expressed Inflammation-Regulating MicroRNAs in Oligoarticular Juvenile Idiopathic Arthritis

Author

McAlpine, Sarah M., primary, Roberts, Sarah E., additional, Hargreaves, Breanna K.V., additional, Bullock, Claire, additional, Ramsey, Suzanne, additional, Stringer, Elizabeth, additional, Lang, Bianca, additional, Huber, Adam, additional, György, Bence, additional, Erdélyi, Fruzsina, additional, Issekutz, Thomas B., additional, and Dérfalvi, Beáta, additional

Published
2022
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10. 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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12. 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

13. The outcomes of juvenile idiopathic arthritis in children managed with contemporary treatments: results from the ReACCh-Out cohort

Author

Guzman, Jaime, Oen, Kiem, Tucker, Lori B, Huber, Adam M, Shiff, Natalie, Boire, Gilles, Scuccimarri, Rosie, Berard, Roberta, Tse, Shirley M L, Morishita, Kimberly, Stringer, Elizabeth, Johnson, Nicole, Levy, Deborah M, Duffy, Karen Watanabe, 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, Bruns, Alessandra, Campillo, Sarah, Benseler, Susanne, Chédeville, Gaëlle, Schneider, Rayfel, Yeung, Rae, Duffy, Ciarán M, Bolaria, Roxana, Gross, Katherine, Feldman, Brian, Feldman, Debbie, Cameron, Bonnie, Jurencak, Roman, Roth, Johannes, Dorval, Jean, LeBlanc, Claire, and St. Cyr, Claire

Published
2015
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14. 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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15. 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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16. Differentially Expressed Inflammation-Regulating MicroRNAs in Oligoarticular Juvenile Idiopathic Arthritis

Author

McAlpine, Sarah M., Roberts, Sarah E., Hargreaves, Breanna K.V., Bullock, Claire, Ramsey, Suzanne, Stringer, Elizabeth, Lang, Bianca, Huber, Adam, Gyo¨rgy, Bence, Erdélyi, Fruzsina, Issekutz, Thomas B., and Dérfalvi, Beáta

Abstract

ObjectiveTo evaluate microRNA expression in synovial fluid (SF), plasma, and leukocytes from patients with juvenile idiopathic arthritis (JIA).MethodsMicroRNA expression in pooled JIA plasma and SF was assessed by absolute quantitative droplet digital PCR array. The results were validated in individual patient samples. MicroRNA content in leukocytes and extracellular vesicles was evaluated by real-time PCR in JIA blood and SF. Blood microRNA expression was compared with healthy controls (HCs). Principal component analysis was used to profile JIA plasma and SF microRNAs, and the potential biological consequences of microRNA dysregulation were investigated by pathway analysis.ResultsMiR-15a-5p and miR-409-3p levels were higher in JIA plasma than in HC plasma. JIA SF contained elevated levels of miR-21-5p, miR-27a-3p, miR-146b-5p, miR-155-5p, and miR-423-5p, and decreased miR-192-5p and miR-451a, compared to JIA plasma. Extracellular vesicle analysis demonstrated variable encapsulation among selected microRNAs, with only miR-155-5p being represented substantially in extracellular vesicles. SF leukocytes also had higher expression of miR-21-5p, miR-27a-3p, miR-146b-5p, and miR-155-5p, and lower expression of miR-409-3p and miR-451a, relative to blood. No differences were observed between JIA and HC blood leukocytes. Clusters of microRNAs were commonly altered in JIA joint fluid and leukocytes compared to JIA blood samples. In silico analysis predicted that differentially expressed microRNAs in JIA target the transforming growth factor (TGF)-β pathway.ConclusionThe expression of multiple microRNAs is dysregulated in JIA both locally and systemically, which may inhibit the TGF-β pathway. These findings advance our knowledge of JIA immunopathogenesis and may lead to the development of targeted therapies.

Published
2023
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17. 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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19. Predictors of Early Inactive Disease in a Juvenile Idiopathic Arthritis Cohort: Results of a Canadian Multicenter, Prospective Inception Cohort Study

Author

OEN, KIEM, TUCKER, LORI, HUBER, ADAM M., MIETTUNEN, PAIVI, SCUCCIMARRI, ROSIE, CAMPILLO, SARAH, CABRAL, DAVID A., FELDMAN, BRIAN M., TSE, SHIRLEY, CHéDEVILLE, GAëLLE, SPIEGEL, LYNN, SCHNEIDER, RAYFEL, LANG, BIANCA, ELLSWORTH, JANET, RAMSEY, SUZANNE, DANCEY, PAUL, SILVERMAN, EARL, CHETAILLE, ANNE-LAURE, CAMERON, BONNIE, JOHNSON, NICOLE, DORVAL, JEAN, PETTY, ROSS E., DUFFY, KAREN WATANABE, BOIRE, GILLES, HADDAD, ELIE, HOUGHTON, KRISTIN, SAINT-CYR, CLAIRE, TURVEY, STUART E., BENSELER, SUSANNE, CHEANG, MARY, YEUNG, RAE S. M., and DUFFY, CIARáN M.

Published
2009
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22. 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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23. Feasibility and safety of a 6-month exercise program to increase bone and muscle strength in children with juvenile idiopathic arthritis

Author

Houghton, Kristin M., Macdonald, Heather M., McKay, Heather A., Guzman, Jaime, Duffy, Ciarán, Tucker, Lori, Berard, Roberta, Boire, Gilles, Bruns, Alessandra, Campillo, Sarah, Chédeville, Gaëlle, Dancey, Paul, Ellsworth, Janet, Feldman, Debbie, Huber, Adam, Johnson, Nicole, Jurencak, Roman, Leblanc, Claire, Levy, Deborah, Miettunen, Paivi, Morishita, Kimberly, Ramsey, Suzanne, Rosenberg, Alan, Roth, Johannes, Rumsey, Dax, Schmeling, Heinrike, Scuccimarri, Rosie, Shiff, Natalie, and Stringer, Elizabeth

Subjects
Male, lcsh:Diseases of the musculoskeletal system, Childhood arthritis, Arthritis, Radius bone, Cohort Studies, 0302 clinical medicine, Absorptiometry, Photon, Quality of life, Bone Density, Immunology and Allergy, 030212 general & internal medicine, Longitudinal Studies, Quantitative computed tomography, Child, medicine.diagnostic_test, lcsh:RJ1-570, Exercise Therapy, medicine.anatomical_structure, Treatment Outcome, Muscle, Female, medicine.symptom, Exercise prescription, Research Article, medicine.medical_specialty, Canada, Adolescent, Juvenile arthritis, 03 medical and health sciences, Rheumatology, Internal medicine, medicine, Humans, Muscle Strength, Bone, Exercise, 030203 arthritis & rheumatology, business.industry, Physical activity, Muscle weakness, lcsh:Pediatrics, medicine.disease, Arthritis, Juvenile, Pediatrics, Perinatology and Child Health, Physical therapy, Quality of Life, Feasibility Studies, Patient Compliance, lcsh:RC925-935, business, Tomography, X-Ray Computed
Abstract

Background Arthritis in childhood can be associated with muscle weakness around affected joints, low bone mass and low bone strength. Exercise is recognized as an important part of management of children with juvenile idiopathic arthritis (JIA) but the exercise prescription to best promote bone and muscle health is unknown. We therefore aimed to: 1. assess feasibility and safety of a 6-month home- and group-based exercise program for children with JIA; 2. estimate the effect of program participation on bone mass and strength, muscle function and clinical outcomes and 3. determine if any positive changes in bone and muscle outcomes are maintained 6 months later. Methods We recruited 24 children with JIA who were part of the Linking Exercise, Physical Activity and Pathophysiology in Childhood Arthritis (LEAP) study to participate in a 6-month home-based exercise program involving jumping and handgrip exercises, resistance training and one group exercise session per month. We assessed lumbar spine bone mass (dual energy X-ray absorptiometry), distal tibia and radius bone microarchitecture and strength (high-resolution peripheral quantitative computed tomography), muscle function (jumping mechanography, dynamometry) and clinical outcomes (joint assessment, function, health-related quality of life) at baseline, 6- and 12-months. Adherence was assessed using weekly activity logs. Results Thirteen children completed the 6-month intervention. Participants reported 9 adverse events and post-exercise pain was rare (0.4%). Fatigue improved, but there were no other sustained improvements in muscle, bone or clinical outcomes. Adherence to the exercise program was low (47%) and decreased over time. Conclusion Children with JIA safely participated in a home-based exercise program designed to enhance muscle and bone strength. Fatigue improved, which may in turn facilitate physical activity participation. Prescribed exercise posed adherence challenges and efforts are needed to address facilitators and barriers to participation in and adherence to exercise programs among children with JIA. Trial registration Data of the children with JIA are from the LEAP study (Canadian Institutes of Health Research (CIHR; GRANT# 107535). http://www.leapjia.com/ Electronic supplementary material The online version of this article (10.1186/s12969-018-0283-4) contains supplementary material, which is available to authorized users.

Published
2018

25. 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

26. JAK1/2 inhibition with baricitinib in the treatment of autoinflammatory interferonopathies

Author

Sanchez, Gina A. Montealegre, primary, Reinhardt, Adam, additional, Ramsey, Suzanne, additional, Wittkowski, Helmut, additional, Hashkes, Philip J., additional, Berkun, Yackov, additional, Schalm, Susanne, additional, Murias, Sara, additional, Dare, Jason A., additional, Brown, Diane, additional, Stone, Deborah L., additional, Gao, Ling, additional, Klausmeier, Thomas, additional, Foell, Dirk, additional, de Jesus, Adriana A., additional, Chapelle, Dawn C., additional, Kim, Hanna, additional, Dill, Samantha, additional, Colbert, Robert A., additional, Failla, Laura, additional, Kost, Bahar, additional, O’Brien, Michelle, additional, Reynolds, James C., additional, Folio, Les R., additional, Calvo, Katherine R., additional, Paul, Scott M., additional, Weir, Nargues, additional, Brofferio, Alessandra, additional, Soldatos, Ariane, additional, Biancotto, Angelique, additional, Cowen, Edward W., additional, Digiovanna, John J., additional, Gadina, Massimo, additional, Lipton, Andrew J., additional, Hadigan, Colleen, additional, Holland, Steven M., additional, Fontana, Joseph, additional, Alawad, Ahmad S., additional, Brown, Rebecca J., additional, Rother, Kristina I., additional, Heller, Theo, additional, Brooks, Kristina M., additional, Kumar, Parag, additional, Brooks, Stephen R., additional, Waldman, Meryl, additional, Singh, Harsharan K., additional, Nickeleit, Volker, additional, Silk, Maria, additional, Prakash, Apurva, additional, Janes, Jonathan M., additional, Ozen, Seza, additional, Wakim, Paul G., additional, Brogan, Paul A., additional, Macias, William L., additional, and Goldbach-Mansky, Raphaela, additional

Published
2018
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27. Additional file 1: of Growth and weight gain in children with juvenile idiopathic arthritis: results from the ReACCh-Out cohort

Author

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

Abstract

Growth and Weight Gain in Juvenile Arthritis. (DOCX 2958 kb)

Published
2017
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29. Trajectories of pain severity in juvenile idiopathic arthritis: results from the Research in Arthritis in Canadian Children Emphasizing Outcomes cohort

Author

Shiff, Natalie J., primary, Tupper, Susan, additional, Oen, Kiem, additional, Guzman, Jaime, additional, Lim, Hyun, additional, Lee, Chel Hee, additional, Bryce, Rhonda, additional, Huber, Adam M., additional, Boire, Gilles, additional, Dancey, Paul, additional, Feldman, Brian, additional, Laxer, Ronald, additional, Miettunen, Paivi, additional, Schmeling, Heinrike, additional, Watanabe Duffy, Karen, additional, Levy, Deborah M., additional, Turvey, Stuart, additional, Bolaria, Roxana, additional, Bruns, Alessandra, additional, Cabral, David A., additional, Campillo, Sarah, additional, Chédeville, Gaëlle, additional, Feldman, Debbie Ehrmann, additional, Haddad, Elie, additional, Houghton, Kristin, additional, Johnson, Nicole, additional, Jurencak, Roman, additional, Lang, Bianca, additional, Larche, Maggie, additional, Morishita, Kimberly, additional, Ramsey, Suzanne, additional, Roth, Johannes, additional, Schneider, Rayfel, additional, Scuccimarri, Rosie, additional, Spiegel, Lynn, additional, Stringer, Elizabeth, additional, Tse, Shirley M., additional, Yeung, Rae, additional, Duffy, Ciarán M., additional, and Tucker, Lori B., additional

Published
2017
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30. 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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31. 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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32. 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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33. Worse Quality of Life, Function, and Pain in Children With Enthesitis, Irrespective of Their Juvenile Arthritis Category

Author

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

Abstract

To estimate the impact of enthesitis on patient‐reported outcomes in children with juvenile idiopathic arthritis (JIA), irrespective of JIAcategory. Children enrolled in the Research in Arthritis in Canadian Children Emphasizing Outcomes cohort were studied. Entheseal tenderness by physician examination in 33 defined locations, Juvenile Arthritis Quality of Life Questionnaire (JAQQ), Quality of My Life (QoML) Questionnaire, Childhood Health Assessment Questionnaire (C‐HAQ), and a pain visual analog scale were completed at enrollment, every 6 months for 2 years, and then yearly up to 5 years. Analyses consisted of descriptive statistics, linear mixed models for longitudinal data, and analysis of covariance. Among 1,371 patients followed for a median of 35.3 months (interquartile range 22.1, 49.2), 214 (16%) had enthesitis, of whom 137 (64%) were classified as having enthesitis‐related arthritis. After adjusting for JIAcategory and covariates, children with enthesitis reported higher JAQQ(mean raw score 2.71 versus 2.16, adjusted difference 0.41 points; 95% confidence interval [95% CI] 0.22, 0.59), higher C‐HAQ(0.47 versus 0.31, adjusted difference 0.14 points; 95% CI0.07, 0.22), higher pain (3.01 versus 1.68, adjusted difference 0.94 points; 95% CI0.64, 1.25), and lower QoML(7.02 versus 8.23, adjusted difference –0.80 points; 95% CI–1.09, –0.51) scores than children without enthesitis. These differences persisted up to 5 years. Children with enthesitis, regardless of JIAcategory, report worse patient‐reported outcomes than those without enthesitis. Thus, enthesitis should be assessed in all children with JIA.

Published
2020
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37. 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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39. The outcomes of juvenile idiopathic arthritis in children managed with contemporary treatments: results from the ReACCh-Out cohort

Author

Guzman, Jaime, primary, Oen, Kiem, additional, Tucker, Lori B, additional, Huber, Adam M, additional, Shiff, Natalie, additional, Boire, Gilles, additional, Scuccimarri, Rosie, additional, Berard, Roberta, additional, Tse, Shirley M L, additional, Morishita, Kimberly, additional, Stringer, Elizabeth, additional, Johnson, Nicole, additional, Levy, Deborah M, additional, Duffy, Karen Watanabe, 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, additional, Bruns, Alessandra, additional, Campillo, Sarah, additional, Benseler, Susanne, additional, Chédeville, Gaëlle, additional, Schneider, Rayfel, additional, Yeung, Rae, additional, and Duffy, Ciarán M, additional

Published
2014
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41. Tournée Images animées

Author

Amalric Mathieu, Colclasure Miranda, Craven Alexander, Lorenzo Angela de, Maracini Linda, Muz Julie Ann, Ramsey Suzanne, Amalric Mathieu, Colclasure Miranda, Craven Alexander, Lorenzo Angela de, Maracini Linda, Muz Julie Ann, and Ramsey Suzanne

Abstract

2010, Making of

Published
2010

44. Access to Biologic Therapies in Canada for Children with Juvenile Idiopathic Arthritis

Author

LEBLANC, CLAIRE M.A., primary, LANG, BIANCA, additional, BENCIVENGA, ALMA, additional, CHETAILLE, ANNE-LAURE, additional, DANCEY, PAUL, additional, DENT, PETER, additional, MIETTUNEN, PAIVI, additional, OEN, KIEM, additional, ROSENBERG, ALAN, additional, ROTH, JOHANNES, additional, SCUCCIMARRI, ROSIE, additional, TSE, SHIRLEY M.L., additional, BENSELER, SUSANNE, additional, CABRAL, DAVID A., additional, CAMPILLO, SARAH, additional, CHÉDEVILLE, GAËLLE, additional, DUFFY, CIARAN M., additional, DUFFY, KAREN WATANABE, additional, HADDAD, ELIE, additional, HUBER, ADAM M., additional, LAXER, RONALD, additional, LEVY, DEBORAH, additional, JOHNSON, NICOLE, additional, RAMSEY, SUZANNE, additional, SHIFF, NATALIE, additional, SCHMELING, HEINRIKE, additional, SCHNEIDER, RAYFEL, additional, STRINGER, ELIZABETH, additional, YEUNG, RAE S.M., additional, and TUCKER, LORI B., additional

Published
2012
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45. 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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46. 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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47. Early outcomes and improvement of patients with juvenile idiopathic arthritis enrolled in a Canadian multicenter inception cohort

Author

Oen, Kiem, primary, Duffy, Ciarán M., additional, Tse, Shirley M. L., additional, Ramsey, Suzanne, additional, Ellsworth, Janet, additional, Chédeville, Gaëlle, additional, Chetaille, Anne‐Laure, additional, Saint‐Cyr, Claire, additional, Cabral, David A., additional, Spiegel, Lynn R., additional, Schneider, Rayfel, additional, Lang, Bianca, additional, Huber, Adam M., additional, Dancey, Paul, additional, Silverman, Earl, additional, Rosenberg, Alan M., additional, Cameron, Bonnie, additional, Johnson, Nicole, additional, Dorval, Jean, additional, Scuccimarri, Rosie, additional, Campillo, Sarah, additional, Petty, Ross E., additional, Duffy, Karen N. Watanabe, additional, Boire, Gilles, additional, Haddad, Elie, additional, Houghton, Kristin, additional, Laxer, Ronald, additional, Turvey, Stuart E., additional, Miettunen, Paivi, additional, Gross, Katherine, additional, Guzman, Jaime, additional, Benseler, Susanne, additional, Feldman, Brian M., additional, Espinosa, Victor, additional, Yeung, Rae S. M., additional, and Tucker, Lori, additional

Published
2010
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48. The Biologic Basis of Clinical Heterogeneity in Juvenile Idiopathic Arthritis.

Author

Eng, Simon W. M., Duong, Trang T., Rosenberg, Alan M., Morris, Quaid, Yeung, Rae S. M., Dancey, Paul, Huber, Adam, Lang, Bianca, Ramsey, Suzanne, Stringer, Elizabeth, Chetaille, Anne‐Laure, Guillet, Chantal, Boire, Gilles, Campillo, Sarah, Chédeville, Gaëlle, Duffy, Ciáran, Duffy, Karen, Gibbon, Michele, Scuccimarri, Rosie, and Jurencak, Roman

Subjects
ARTHRITIS, EVALUATION of medical care, PEDIATRICS, JUVENILE idiopathic arthritis, RHEUMATOLOGY, SERIAL publications, DATA analysis, CROSS-sectional method, DISEASE complications, GENETICS, DIAGNOSIS
Abstract

Objective Childhood arthritis encompasses a heterogeneous family of diseases. Significant variation in clinical presentation remains despite consensus-driven diagnostic classifications. Developments in data analysis provide powerful tools for interrogating large heterogeneous data sets. We report a novel approach to integrating biologic and clinical data toward a new classification for childhood arthritis, using computational biology for data-driven pattern recognition. Methods Probabilistic principal components analysis was used to transform a large set of data into 4 interpretable indicators or composite variables on which patients were grouped by cluster analysis. Sensitivity analysis was conducted to determine key variables in determining indicators and cluster assignment. Results were validated against an independent validation cohort. Results Meaningful biologic and clinical characteristics, including levels of proinflammatory cytokines and measures of disease activity, defined axes/indicators that identified homogeneous patient subgroups by cluster analysis. The new patient classifications resolved major differences between patient subpopulations better than International League of Associations for Rheumatology subtypes. Fourteen variables were identified by sensitivity analysis to crucially determine indicators and clusters. This new schema was conserved in an independent validation cohort. Conclusion Data-driven unsupervised machine learning is a powerful approach for interrogating clinical and biologic data toward disease classification, providing insight into the biology underlying clinical heterogeneity in childhood arthritis. Our analytical framework enabled the recovery of unique patterns from small cohorts and addresses a major challenge, patient numbers, in studying rare diseases. [ABSTRACT FROM AUTHOR]

Published
2014
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49. Corticosteroid Treatment of Refractory Kawasaki Disease

Author

Lang, Bianca, Yeung, Rae, Oen, Kiem, Malleson, Peter, Huber, Adam, Riley, Mark, Ebbeson, Regan, Ramsey, Suzanne, Laxer, Ronald, Silverman, Earl, Mccrindle, Brian, Ratnapalan, Savithiri, and Feldman, Brian

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

50. 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.

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