244 results on '"Rosenberg, Alan M."'
Search Results
2. Gene Expression Deconvolution for Uncovering Molecular Signatures in Response to Therapy in Juvenile Idiopathic Arthritis
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Cui, Ang, Quon, Gerald, Rosenberg, Alan M, Yeung, Rae SM, and Morris, Quaid
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Biological Sciences ,Bioinformatics and Computational Biology ,Rare Diseases ,Pediatric ,Autoimmune Disease ,Genetics ,Arthritis ,4.1 Discovery and preclinical testing of markers and technologies ,Detection ,screening and diagnosis ,Inflammatory and immune system ,Adolescent ,Algorithms ,Antirheumatic Agents ,Arthritis ,Juvenile ,Biomarkers ,Blood Platelets ,Child ,Gene Expression ,Gene Expression Profiling ,Humans ,Lymphocyte Count ,Models ,Theoretical ,Monocytes ,Neutrophils ,Pilot Projects ,T-Lymphocytes ,Treatment Outcome ,alpha-Defensins ,BBOP Study Consortium ,General Science & Technology - Abstract
Gene expression-based signatures help identify pathways relevant to diseases and treatments, but are challenging to construct when there is a diversity of disease mechanisms and treatments in patients with complex diseases. To overcome this challenge, we present a new application of an in silico gene expression deconvolution method, ISOpure-S1, and apply it to identify a common gene expression signature corresponding to response to treatment in 33 juvenile idiopathic arthritis (JIA) patients. Using pre- and post-treatment gene expression profiles only, we found a gene expression signature that significantly correlated with a reduction in the number of joints with active arthritis, a measure of clinical outcome (Spearman rho = 0.44, p = 0.040, Bonferroni correction). This signature may be associated with a decrease in T-cells, monocytes, neutrophils and platelets. The products of most differentially expressed genes include known biomarkers for JIA such as major histocompatibility complexes and interleukins, as well as novel biomarkers including α-defensins. This method is readily applicable to expression datasets of other complex diseases to uncover shared mechanistic patterns in heterogeneous samples.
- Published
- 2016
3. Clinical presentation, immunologic features, and hematopoietic stem cell transplant outcomes for IKBKB immune deficiency
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Cuvelier, Geoffrey D.E., Rubin, Tamar S., Junker, Anne, Sinha, Roona, Rosenberg, Alan M., Wall, Donna A., and Schroeder, Marlis L.
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- 2019
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4. A decade of progress in juvenile idiopathic arthritis treatments and outcomes in Canada: results from ReACCh-Out and the CAPRI registry
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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
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- 2023
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5. Evaluation of Uveitis Induced in Rats by a Type I Collagen Peptide as a Model for Childhood Arthritis-associated Uveitis
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Osinchuk, Stephanie C, primary, Grahn, Bruce H, additional, Wilson, Tracy D, additional, Thompson, Brooke N, additional, Hart, David A, additional, Harrison, Kim D, additional, Cooper, David ML, additional, Panahifar, Arash, additional, and Rosenberg, Alan M, additional
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- 2023
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6. Cancer risk in childhood-onset systemic lupus
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Bernatsky, Sasha, Clarke, Ann E, Labrecque, Jeremy, von Scheven, Emily, Schanberg, Laura E, Silverman, Earl D, Brunner, Hermine I, Haines, Kathleen A, Cron, Randy Q, O’Neil, Kathleen M, Oen, Kiem, Rosenberg, Alan M, Duffy, Ciarán M, Joseph, Lawrence, Lee, Jennifer L, Kale, Mruganka, Turnbull, Elizabeth M, and Ramsey-Goldman, Rosalind
- Abstract
Abstract Introduction The aim of this study was to assess cancer incidence in childhood-onset systemic lupus erythematosus (SLE). Methods We ascertained cancers within SLE registries at 10 pediatric centers. Subjects were linked to cancer registries for the observational interval, spanning 1974 to 2009. The ratio of observed to expected cancers represents the standardized incidence ratio (SIR) or relative cancer risk in childhood-onset SLE, versus the general population. Results There were 1020 patients aged
- Published
- 2013
7. Teens Taking Charge: A Randomized Controlled Trial of a Web-Based Self-Management Program With Telephone Support for Adolescents With Juvenile Idiopathic Arthritis
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Stinson, Jennifer N, Lalloo, Chitra, Hundert, Amos S, Campillo, Sarah, Cellucci, Tania, Dancey, Paul, Duffy, Ciaran, Ellsworth, Janet, Feldman, Brian M, Huber, Adam M, Johnson, Nicole, Jong, Geert't, Oen, Kiem, Rosenberg, Alan M, Shiff, Natalie J, Spiegel, Lynn, Tse, Shirley M L, Tucker, Lori, and Victor, Joseph Charles
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Computer applications to medicine. Medical informatics ,R858-859.7 ,Public aspects of medicine ,RA1-1270 - Abstract
BackgroundJuvenile idiopathic arthritis (JIA) is a serious and potentially debilitating pediatric illness. Improved disease self-management may help to improve health outcomes. ObjectiveThis study aimed to evaluate the effectiveness of the Teens Taking Charge Web-based self-management intervention in reducing symptoms and improving health-related quality of life (HRQL) in adolescents with JIA compared with a Web-based education control condition. MethodsAdolescents with JIA aged 12 to 18 years were recruited from 11 Canadian pediatric rheumatology centers. Caregivers were invited to participate along with their child. In addition to standard medical care, participants were randomized to receive either (1) the Teens Taking Charge self-management intervention or (2) a Web-based education control condition for a period of 12 weeks. Adolescents in the intervention group completed website modules addressing cognitive behavioral coping skills, stress management, and other self-management topics, while also receiving monthly telephone calls from a trained health coach. Adolescents in the education control group were instructed to view a series of preselected public JIA educational websites and received monthly calls from a coach who asked about their own best efforts at managing JIA. Caregivers in the intervention group completed website modules related to promoting independence and disease self-management in their child. Caregivers in the education control group were instructed to view a series of preselected public JIA educational websites. Outcome assessment occurred at baseline, 12 weeks (posttreatment), and at 6 and 12 months postrandomization. The primary outcomes were pain intensity, pain interference, and HRQL. Secondary outcomes were emotional symptoms, adherence, coping, knowledge, and self-efficacy. ResultsIn total, 333 adolescents and 306 caregivers were enrolled. Significant overall reductions in pain intensity (P=.02) and pain interference (P=.007) were observed for intervention group participants compared with those in the education control group, after adjusting for baseline levels. There was a significant overall improvement in HRQL related to problems with pain (P=.02) and problems with daily activities (P=.01). There was also a significant difference in the intervention group over time (P=.008) for HRQL related to treatment problems, with the intervention group participants demonstrating improved HRQL by 12 months compared with education control group participants. Both groups showed nonsignificant improvements compared with baseline in other primary outcomes. There were no significant differences between the groups in any secondary outcomes or caregiver-reported outcomes. ConclusionsThe results of this randomized trial suggest that the Teens Taking Charge Web-based intervention is effective at reducing both pain intensity and pain interference, as well as improving HRQL in adolescents with JIA, compared with education control. These effects are sustained for up to 12 months following program completion. The Teens Taking Charge program is now publicly available at no cost. Trial RegistrationClinicalTrials.gov NCT01572896; https://clinicaltrials.gov/ct2/show/NCT01572896
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- 2020
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8. Vitamin D and juvenile idiopathic arthritis
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Finch, Sarah L., Rosenberg, Alan M., and Vatanparast, Hassan
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- 2018
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9. Patterns of joint involvement in juvenile idiopathic arthritis and prediction of disease course: A prospective study with multilayer non-negative matrix factorization
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Eng, Simon W. M., Aeschlimann, Florence A., van Veenendaal, Mira, Berard, Roberta A., Rosenberg, Alan M., Morris, Quaid, and Yeung, Rae S. M.
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Pediatric research ,Joints (Anatomy) -- Health aspects -- Research ,Machine learning -- Usage ,Juvenile rheumatoid arthritis -- Care and treatment -- Research ,Anti-inflammatory agents ,Inflammation ,Arthritis ,Psoriatic arthritis ,Pediatric diseases ,Medical research ,Rheumatoid factor ,Epidemiology ,Psoriasis ,Children ,Biological sciences - Abstract
Background Joint inflammation is the common feature underlying juvenile idiopathic arthritis (JIA). Clinicians recognize patterns of joint involvement currently not part of the International League of Associations for Rheumatology (ILAR) classification. Using unsupervised machine learning, we sought to uncover data-driven joint patterns that predict clinical phenotype and disease trajectories. Methods and findings We analyzed prospectively collected clinical data, including joint involvement using a standard 71-joint homunculus, for 640 discovery patients with newly diagnosed JIA enrolled in a Canada-wide study who were followed serially for five years, treatment-naïve except for nonsteroidal anti-inflammatory drugs (NSAIDs) and diagnosed within one year of symptom onset. Twenty-one patients had systemic arthritis, 300 oligoarthritis, 125 rheumatoid factor (RF)-negative polyarthritis, 16 RF-positive polyarthritis, 37 psoriatic arthritis, 78 enthesitis-related arthritis (ERA), and 63 undifferentiated arthritis. At diagnosis, we observed global hierarchical groups of co-involved joints. To characterize these patterns, we developed sparse multilayer non-negative matrix factorization (NMF). Model selection by internal bi-cross-validation identified seven joint patterns at presentation, to which all 640 discovery patients were assigned: pelvic girdle (57 patients), fingers (25), wrists (114), toes (48), ankles (106), knees (283), and indistinct (7). Patterns were distinct from clinical subtypes (P < 0.001 by X.sup.2 test) and reproducible through external data set validation on a 119-patient, prospectively collected independent validation cohort (reconstruction accuracy Q.sup.2 = 0.55 for patterns; 0.35 for groups). Some patients matched multiple patterns. To determine whether their disease outcomes differed, we further subdivided the 640 discovery patients into three subgroups by degree of localization-the percentage of their active joints aligning with their assigned pattern: localized ([greater than or equal to]90%; 359 patients), partially localized (60%-90%; 124), or extended ( We modelled time to zero joints in the discovery cohort using a multivariate Cox proportional hazards model considering joint pattern, degree of localization, and ILAR subtype. Despite receiving more intense treatment, 50% of nonlocalized patients had zero joints at one year compared to six months for localized patients. Overall, localized patients required less time to reach zero joints (partial: P = 0.0018 versus localized by log-rank test; extended: P = 0.0057). Potential limitations include the requirement for patients to be treatment naïve (except NSAIDs), which may skew the patient cohorts towards milder disease, and the validation cohort size precluded multivariate analyses of disease trajectories. Conclusions Multilayer NMF identified patterns of joint involvement that predicted disease trajectory in children with arthritis. Our hierarchical unsupervised approach identified a new clinical feature, degree of localization, which predicted outcomes in both cohorts. Detailed assessment of every joint is already part of every musculoskeletal exam for children with arthritis. Our study supports both the continued collection of detailed joint involvement and the inclusion of patterns and degrees of localization to stratify patients and inform treatment decisions. This will advance pediatric rheumatology from counting joints to realizing the potential of using data available from uncovering patterns of joint involvement., Author(s): Simon W. M. Eng 1,2,3, Florence A. Aeschlimann 1, Mira van Veenendaal 1, Roberta A. Berard 4,5,6, Alan M. Rosenberg 7, Quaid Morris 3,7,8,9,10,11,*, Rae S. M. Yeung 1,2,4,*, [...]
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- 2019
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10. Polyarticular Juvenile Idiopathic Arthritis
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Rosenberg, Alan M., primary and Oen, Kiem G., additional
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- 2016
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11. Contributors
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Akikusa, Jonathan, primary, Albani, Salvatore, additional, Allen, Roger, additional, Alsaeid, Khaled, additional, Avčin, Tadej, additional, Babyn, Paul S., additional, Bagga, Arvind, additional, Barron, Karyl S., additional, Becker, Mara L., additional, Benseler, Susanne M., additional, Beukelman, Timothy, additional, Brogan, Paul, additional, Brunner, Hermine I., additional, Burgos-Vargas, Rubèn, additional, Buyon, Jill, additional, Cabral, David A., additional, Choo, Sharon, additional, Cimaz, Rolando, additional, Colbert, Robert Allen, additional, Cole, William G., additional, Davidson, Iris, additional, Benedetti, Fabrizio De, additional, Doria, Andrea S., additional, Dressler, Frank, additional, Duffy, Ciarán M., additional, Eleftheriou, Despina, additional, Feldman, Brian M., additional, Ferguson, Polly J., additional, Fuhlbrigge, Robert, additional, Gattorno, Marco, additional, Grom, Alexei A., additional, Hashkes, Philip J., additional, Houghton, Kristin, additional, Huppertz, Hans-Iko, additional, Ilowite, Norman T., additional, Jaeggi, Edgar, additional, Kastner, Daniel L., additional, Kirton, Adam, additional, Klein-Gitelman, Marisa, additional, Kuchta, Gay, additional, Lane, Jerome Charles, additional, Laxer, Ronald M., additional, LeBlanc, Claire, additional, Leeder, Steven J., additional, Legger, G. Elizabeth, additional, Li, Suzanne C., additional, Lindsley, Carol B., additional, Lovell, Dan, additional, Makitie, Outi, additional, Martini, Alberto, additional, Miller, Frederick W., additional, Morishita, Kimberly, additional, Nigrovic, Peter A., additional, Oen, Kiem G., additional, O'Neil, Kathleen M., additional, Ozen, Seza, additional, Pepmueller, Peri H., additional, Petty, Ross E., additional, Pope, Elena, additional, Prahalad, Sampath, additional, Prakken, Berent, additional, Rapoff, Michael, additional, Rider, Lisa G., additional, Rosé, Carlos Daniel, additional, Rosenbaum, James T., additional, Rosenberg, Alan M., additional, Roth, Johannes, additional, Guillermo, Ricardo Alberto, additional, Schneider, Rayfel, additional, Scott, Christiaan, additional, Sherry, David D., additional, Silverman, Earl, additional, Son, Mary Beth, additional, Sundel, Robert P., additional, Thompson, Susan D., additional, Tiedemann, Karin, additional, Tse, Shirley M.L., additional, Tucker, Lori, additional, Uziel, Yosef, additional, van Montfrans, Joris, additional, Vazques-Mellado, Janitzia, additional, Ward, Leanne, additional, Wedderburn, Lucy R., additional, Wouters, Carine, additional, Wright, James, additional, Wulffraat, Nico M., additional, Zemel, Lawrence, additional, and Zulian, Francesco, additional
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- 2016
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12. Systemic lupus erythematosus in children
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Rosenberg, Alan M. and Miescher, Peter A., editor
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- 1995
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13. The risk and nature of flares in juvenile idiopathic arthritis: results from the ReACCh-Out cohort
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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
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- 2016
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14. 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
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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
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- 2021
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15. Predicting Which Children with Juvenile Idiopathic Arthritis Will Not Attain Early Remission with Conventional Treatment: Results from the ReACCh-Out Cohort
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Guzman, Jaime, Henrey, Andrew, Loughin, Thomas, Berard, Roberta A., Shiff, Natalie J., Jurencak, Roman, Huber, Adam M., Oen, Kiem, Gerhold, Kerstin, Feldman, Brian M., Scuccimarri, Rosie, Houghton, Kristin, Chédeville, Gaëlle, Morishita, Kimberly, Lang, Bianca, Dancey, Paul, Rosenberg, Alan M., Barsalou, Julie, Bruns, Alessandra, Duffy, Karen Watanabe, Benseler, Susanne, Duffy, Ciaran M., Tucker, Lori B., Bolaria, Roxana, Gross, Katherine, Turvey, Stuart E., Cabral, David, Petty, Ross, and Ellsworth, Janet
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Male ,Pediatrics ,medicine.medical_specialty ,Adolescent ,Immunology ,Arthritis ,Early remission ,Logistic regression ,Severity of Illness Index ,Decile ,03 medical and health sciences ,0302 clinical medicine ,Rheumatology ,medicine ,Humans ,Immunology and Allergy ,Juvenile ,Treatment Failure ,030212 general & internal medicine ,Child ,Risk stratification ,030203 arthritis & rheumatology ,Biological Products ,business.industry ,Remission Induction ,Conventional treatment ,Juvenile idiopathic arthritis ,Prognosis ,medicine.disease ,Arthritis, Juvenile ,Logistic Models ,Treatment Outcome ,Antirheumatic Agents ,Child, Preschool ,Cohort ,Cohort studies ,Female ,Prediction ,business ,Cohort study - Abstract
Objective.To estimate the probability of early remission with conventional treatment for each child with juvenile idiopathic arthritis (JIA). Children with a low chance of remission may be candidates for initial treatment with biologics or triple disease-modifying antirheumatic drugs (DMARD).Methods.We used data from 1074 subjects in the Research in Arthritis in Canadian Children emphasizing Outcomes (ReACCh-Out) cohort. The predicted outcome was clinically inactive disease for ≥ 6 months starting within 1 year of JIA diagnosis in patients who did not receive early biologic agents or triple DMARD. Models were developed in 200 random splits of 75% of the cohort and tested on the remaining 25% of subjects, calculating expected and observed frequencies of remission and c-index values.Results.Our best Cox logistic model combining 18 clinical variables a median of 2 days after diagnosis had a c-index of 0.69 (95% CI 0.67–0.71), better than using JIA category alone (0.59, 95% CI 0.56–0.63). Children in the lowest probability decile had a 20% chance of remission and 21% attained remission; children in the highest decile had a 69% chance of remission and 73% attained remission. Compared to 5% of subjects identified by JIA category alone, the model identified 14% of subjects as low chance of remission (probability < 0.25), of whom 77% failed to attain remission.Conclusion.Although the model did not meet our a priori performance threshold (c-index > 0.70), it identified 3 times more subjects with low chance of remission than did JIA category alone, and it may serve as a benchmark for assessing value added by future laboratory/imaging biomarkers.
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- 2019
16. The outcomes of juvenile idiopathic arthritis in children managed with contemporary treatments: results from the ReACCh-Out cohort
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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
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- 2015
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17. 18 - Polyarticular Juvenile Idiopathic Arthritis
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Rosenberg, Alan M., Jariwala, Mehul, and Cron, Randy Q.
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- 2021
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18. 1 - Structure and Function
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Rosenberg, Alan M. and Petty, Ross E.
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- 2021
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19. Primary Defect of Insulin Receptors in Skin Fibroblasts Cultured from an Infant with Leprechaunism and Insulin Resistance
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Schilling, Ellen E., Rechler, Matthew M., Grunfeld, Carl, and Rosenberg, Alan M.
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- 1979
20. The Biologic Basis of Clinical Heterogeneity in Juvenile Idiopathic Arthritis
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Eng, Simon W. M., Duong, Trang T., Rosenberg, Alan M., Morris, Quaid, and Yeung, Rae S. M.
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- 2014
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21. Additional file 1 of Clinical and psychosocial stress factors are associated with decline in physical activity over time in children with juvenile idiopathic arthritis
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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.
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Data_FILES - Abstract
Additional file 1.
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- 2021
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22. Clinical and Psychosocial Stress Factors are Associated with Decline in Physical Activity Over Time in Children with Juvenile Idiopathic Arthritis
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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
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- 2021
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23. Characterization of active joint count trajectories in juvenile idiopathic arthritis
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Berard Roberta A, Tomlinson George, Li Xiuying, Oen Kiem G, Rosenberg Alan M, Feldman Brian M, Yeung Rae SM, and Bombardier Claire
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Pediatrics ,RJ1-570 ,Diseases of the musculoskeletal system ,RC925-935 - Published
- 2012
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24. POLYARTHRITIS
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Rosenberg, Alan M., primary and Oen, Kiem G., additional
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- 2011
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25. CONTRIBUTORS
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Albani, Salvatore, primary, Alsaeid, Khaled, additional, Athreya, Balu H., additional, Avčin, Tadej, additional, Babyn, Paul, additional, Bagga, Arvind, additional, Barron, Karyl S., additional, Benseler, Susanne, additional, Brogan, Paul, additional, Brunner, Hermine I., additional, Burgos-Vargas, Rubén, additional, Buyon, Jill P., additional, Cabral, David A., additional, Cassidy, James T., additional, Cimaz, Rolando, additional, Colbert, Robert A., additional, Davidson, Iris L., additional, De Benedetti, Fabrizio, additional, Dillon, Michael J., additional, Doria, Andrea Schwarz, additional, Dressler, Frank, additional, Duffy, Ciarán M., additional, Eddy, Allison A., additional, Falcini, Fernanda, additional, Feldman, Brian M., additional, Ferguson, Polly J., additional, Fuhlbrigge, Robert C., additional, Gattorno, Marco, additional, Giannini, Edward H., additional, Glass, David N., additional, Grom, Alexei A., additional, Houghton, Kristin, additional, Huppertz, Hans-Iko, additional, Ilowite, Norman T., additional, Kastner, Daniel L., additional, Kuchta, Gay, additional, Kuis, Wietse, additional, Laxer, Ronald M., additional, LeBlanc, Claire, additional, Lindsley, Carol B., additional, Martini, Alberto, additional, Nigrovic, Peter A., additional, O'Neil, Kathleen M., additional, Oen, Kiem G., additional, Özen, Seza, additional, Pepmueller, Peri Hickman, additional, Prakken, Berent J., additional, Rapoff, Michael A., additional, Rider, Lisa G., additional, Rosé, Carlos D., additional, Rosenbaum, James T., additional, Rosenberg, Alan M., additional, Schneider, Rayfel, additional, Sherry, David D., additional, Silverman, Earl D., additional, Sundel, Robert P., additional, Thompson, Susan D., additional, Tucker, Lori B., additional, van Montfrans, Joris, additional, Vázquez-Mellado, Janitzia, additional, Wenkert, Deborah, additional, Wouters, Carine H., additional, Wulffraat, Nico, additional, and Zulian, Francesco, additional
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- 2011
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26. Clinical and associated inflammatory biomarker features predictive of short-term outcomes in non-systemic juvenile idiopathic arthritis
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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
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- 2020
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27. Vitamin D Deficiency in Young Children With Severe Acute Lower Respiratory Infection
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McNally, J. Dayre, Leis, Karen, Matheson, Loren A., Karuananyake, Chandima, Sankaran, Koravangattu, and Rosenberg, Alan M.
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- 2009
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28. Systemic lupus erythematosus in children
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Rosenberg, Alan M.
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- 1994
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29. Predictors of Pain in Children With Established Juvenile Rheumatoid Arthritis
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MALLESON, PETER N., OEN, KIEM, CABRAL, DAVID A., PETTY, ROSS E., ROSENBERG, ALAN M., and CHEANG, MARY
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- 2004
30. The genetic profile of RF-positive polyarticular juvenile idiopathic arthritis (JIA) resembles adult rheumatoid arthritis (RA)
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Hinks, Anne, Marion, Miranda C., Cobb, Joanna, Comeau, Mary E., Sudman, Marc, Ainsworth, Hannah C, Bowes, John, Becker, Mara L, Bohnsack, John F., Haas, Johannes-Peter, Lovell, Daniel J, Mellins, Elizabeth D, Nelson, J. Lee, Nordal, Ellen, Punaro, Marilynn, Reed, Ann M., Rosé, Carlos D., Rosenberg, Alan M, Rygg, Marite, Smith, Samantha L, Stevens, Anne M, Videm, Vibeke, Wallace, Carol A., Wedderburn, Lucy R, Yarwood, Annie, Yeung, Rae S M, Langefeld, Carl D., Thompson, Susan D., Thomson, Wendy, and Prahalad, Sampath
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musculoskeletal diseases ,genetic structures ,immune system diseases ,skin and connective tissue diseases ,eye diseases - Abstract
Objective: Juvenile idiopathic arthritis (JIA) is comprised of seven heterogeneous categories of chronic childhood arthritides. About 5% of children with JIA have rheumatoid factor (RF) positive arthritis, which phenotypically resembles adult rheumatoid arthritis (RA). Our objective was to compare and contrast the genetics of RF-positive polyarticular JIA with RA, and selected other JIA categories, to more fully understand the pathophysiological relationships of inflammatory arthropathies. Methods: RF-positive polyarticular JIA cases (n=340) and controls (n=14,412) were genotyped using the Immunochip array. Single nucleotide polymorphisms (SNPs) were tested for association using a logistic regression model adjusting for admixture proportions. Weighted genetic risk scores (wGRS) of published RA and JIA risk loci were calculated and their ability to predict RF-positive polyarticular JIA were compared. Results: As expected, the HLA region was strongly associated with RF-positive polyarticular JIA (p=5.51x10-31). Nineteen of 44 RA risk loci and 6 of 27 oligoarticular/RF-negative polyarticular JIA risk loci were associated (p70 years. Conclusions: RF-positive polyarticular JIA is genetically more similar to adult RA than to the most common JIA categories and thus appears to be a childhood-onset presentation of autoantibody positive RA. These findings suggest common disease mechanisms, which could lead to novel therapeutic targets and shared treatment strategies.
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- 2018
31. Health-Related Quality of Life in an Inception Cohort of Children With Juvenile Idiopathic Arthritis: A Longitudinal Analysis
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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
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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.
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- 2018
32. Measuring consistency among gene set analysis methods: A systematic study
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Maleki, Farhad, primary, Ovens, Katie L., additional, Hogan, Daniel J., additional, Rezaei, Elham, additional, Rosenberg, Alan M., additional, and Kusalik, Anthony J., additional
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- 2019
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33. Teens Taking Charge: A Randomized Controlled Trial of a Web-Based Self-Management Program With Telephone Support for Adolescents With Juvenile Idiopathic Arthritis (Preprint)
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Stinson, Jennifer N, primary, Lalloo, Chitra, additional, Hundert, Amos S, additional, Campillo, Sarah, additional, Cellucci, Tania, additional, Dancey, Paul, additional, Duffy, Ciaran, additional, Ellsworth, Janet, additional, Feldman, Brian M, additional, Huber, Adam M, additional, Johnson, Nicole, additional, Jong, Geert't, additional, Oen, Kiem, additional, Rosenberg, Alan M, additional, Shiff, Natalie J, additional, Spiegel, Lynn, additional, Tse, Shirley M L, additional, Tucker, Lori, additional, and Victor, Joseph Charles, additional
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- 2019
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34. Gene Set Databases
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Maleki, Farhad, primary, Ovens, Katie, additional, McQuillan, Ian, additional, Rezaei, Elham, additional, Rosenberg, Alan M., additional, and Kusalik, Anthony J., additional
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- 2019
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35. Increased T-lymphocyte dependent antibody production in female SJL/J mice following exposure to commercial grade malathion
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Johnson, Victor J, Rosenberg, Alan M, Lee, Kwang, and Blakley, Barry R
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- 2002
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36. Mycobacterium tuberculosis monoarthritis in a child
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Rosenberg Alan M and Rajakumar Derek
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Pediatrics ,RJ1-570 ,Diseases of the musculoskeletal system ,RC925-935 - Abstract
Abstract A child with isolated Mycobacterium tuberculosis monoarthritis, with features initially suggesting oligoarthritis subtype of juvenile idiopathic arthritis, is presented. This patient illustrates the need to consider the possibility of tuberculosis as the cause of oligoarthritis in high-risk pediatric populations even in the absence of a tuberculosis contact history and without evidence of overt pulmonary disease.
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- 2008
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37. Seasonal onset of systemic-onset juvenile rheumatoid arthritis
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Feldman, Brian M., Birdi, Nina, Boone, James E., Dent, Peter B., Duffy, Ciaran M., Ellsworth, Janet E., Lang, Bianca A., Laxer, Ronald M., Lewkonia, Raymond M., Malleson, Peter N., Oen, Kiem G., Paquin, Julie D., Rosenberg, Alan M., Schneider, Rayfel, and Silverman, Earl D.
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- 1996
38. Brief Report: The Genetic Profile of Rheumatoid Factor-Positive Polyarticular Juvenile Idiopathic Arthritis Resembles That of Adult Rheumatoid Arthritis
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Hinks, Anne, Marion, Miranda C., Cobb, Joanna, Comeau, Mary E., Sudman, Marc, Ainsworth, Hannah C., Bowes, John, Becker, Mara L., Bohnsack, John F., Haas, Johannes-Peter, Lovell, Daniel J., Mellins, Elizabeth D., Nelson, J. Lee, Nordal, Ellen Berit, Punaro, Marilynn, Reed, Ann M., Rose, Carlos D., Rosenberg, Alan M., Rygg, Marite, Smith, Samantha L., Stevens, Anne M., Videm, Vibeke, Wallace, Carol A., Wedderburn, Lucy R., Yarwood, Annie, Yeung, Rae S.M., Langefeld, Carl D., Thompson, Susan D., Thomson, Wendy, and Prahalad, Sampath
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musculoskeletal diseases ,Adult ,Male ,genetic structures ,Adolescent ,Genotype ,VDP::Medical disciplines: 700::Clinical medical disciplines: 750::Rheumatology: 759 ,Genetic Profile ,Polymorphism, Single Nucleotide ,Arthritis, Juvenile ,VDP::Medisinske Fag: 700::Klinisk medisinske fag: 750::Reumatologi: 759 ,Arthritis, Rheumatoid ,Logistic Models ,Phenotype ,immune system diseases ,Rheumatoid Factor ,Humans ,Female ,skin and connective tissue diseases ,Child ,Autoantibodies - Abstract
This is the peer reviewed version of the following article: Hinks, A., Marion, M.C., Cobb, J., Comeau, M.E., Sudman, M., Ainsworth, H.C., ... Prahalad, S. (2018). Brief Report: The Genetic Profile of Rheumatoid Factor? Positive Polyarticular Juvenile Idiopathic Arthritis Resembles That of Adult Rheumatoid Arthritis. Arthritis & Rheumatology, 70(6), 957-962, which has been published in final form at https://doi.org/10.1002/art.40443. This article may be used for non-commercial purposes in accordance with Wiley Terms and Conditions for Use of Self-Archived Versions. Objective: Juvenile idiopathic arthritis (JIA) comprises 7 heterogeneous categories of chronic childhood arthritides. Approximately 5% of children with JIA have rheumatoid factor (RF)–positive arthritis, which phenotypically resembles adult rheumatoid arthritis (RA). Our objective was to compare and contrast the genetics of RF‐positive polyarticular JIA with those of RA and selected other JIA categories, to more fully understand the pathophysiologic relationships of inflammatory arthropathies. Methods: Patients with RF‐positive polyarticular JIA (n = 340) and controls (n = 14,412) were genotyped using the Immunochip array. Single‐nucleotide polymorphisms were tested for association using a logistic regression model adjusting for admixture proportions. We calculated weighted genetic risk scores (wGRS) of reported RA and JIA risk loci, and we compared the ability of these wGRS to predict RF‐positive polyarticular JIA. Results: As expected, the HLA region was strongly associated with RF‐positive polyarticular JIA (P = 5.51 × 10−31). Nineteen of 44 RA risk loci and 6 of 27 oligoarticular/RF‐negative polyarticular JIA risk loci were associated with RF‐positive polyarticular JIA (P < 0.05). The RA wGRS predicted RF‐positive polyarticular JIA (area under the curve [AUC] 0.71) better than did the oligoarticular/RF‐negative polyarticular JIA wGRS (AUC 0.59). The genetic profile of patients with RF‐positive polyarticular JIA was more similar to that of RA patients with age at onset 16–29 years than to that of RA patients with age at onset ≥70 years. Conclusion: RF‐positive polyarticular JIA is genetically more similar to adult RA than to the most common JIA categories and thus appears to be a childhood‐onset presentation of autoantibody‐positive RA. These findings suggest common disease mechanisms, which could lead to novel therapeutic targets and shared treatment strategies.
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- 2017
39. Contributors
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Abinun, Mario, Aggarwal, Amita, Akikusa, Jonathan, Allen, Roger, Alsaeid, Khaled, Avčin, Tadej, Balevic, Stephen, Banwell, Brenda, Barron, Karyl, Barsalou, Julie, Becker, Mara L., Behrens, Edward M., Beresford, Michael W., Beukelman, Timothy, Bockenstedt, Linda K., Brogan, Paul, Brunner, Hermine I., Burns, Jane C., Busch, Robert, Cabral, David A., Canna, Scott W., Choo, Sharon, Cimaz, Rolando, Ciurtin, Coziana, Clinch, Jacqui, Colbert, Robert A., Consolaro, Alessandro, Cooper, Jennifer C., Cron, Randy Q., Davidson, Iris, Dusser, Perrine, Eleftheriou, Despina, Feldman, Brian Michael, Ferguson, Polly J., Foster, Helen Elisabeth, Fuhlbrigge, Robert C., Funk, Ryan S., Gattorno, Marco, Giancane, Gabriella, Griffiths, Anne, Grom, Alexei A., Harel, Liora, Hashkes, Philip J., Hedrich, Christian Michael, Horneff, Gerd, Houghton, Kristin Michelle, Jaeggi, Edgar, Jariwala, Mehul, Jones, Jordan T., Kastner, Daniel L., Kimura, Yukiko, Klein-Gitelman, Marisa S., Koné-Paut, Isabelle, Laxer, Ronald M., Anne LeBlanc, Claire Marie, Li, Suzanne C., Lindsley, Carol B., Martini, Alberto, Mellins, Elizabeth, Morishita, Kimberly A., Muscal, Eyal, Newburger, Jane W., Nigrovic, Peter A., Nott, Kerstin A., O’Neil, Kathleen M., Ombrello, Michael J., Orme, Lisa, Ozen, Seza, Petty, Ross E., Pilkington, Clarissa A., Pope, Elena, Prahalad, Sampath, Prescott, Steven A., Pääkkönen, Markus, Rabinovich, C. Egla, Ramanan, Athimalaipet V., Ravelli, Angelo, Ricciuto, Amanda, Ringold, Sarah, Rosenbaum, James Todd, Rosenberg, Alan M., Rosendahl, Karen, Rosser, Elizabeth C., Rosé, Carlos Daniel, Roth, Johannes, Ruperto, Nicolino, Schulert, Grant, Scott, Christiaan, Sen, Ethan S., Sherry, David D., Silverman, Earl D., Son, Mary Beth F., Sontichai, Watchareewan, Stevens, Anne M., Stinson, Jennifer N., Stoll, Matthew L., Sullivan, Kathleen, Takken, Tim, Torok, Kathryn S., Tse, Shirley M.L., Tucker, Lori, Unger, Sheila, Uziel, Yosef, van der Net, Janjaap, Vastert, Sebastiaan J., Ward, Leanne M., Wedderburn, Lucy R., Weiss, Pamela F., Wouters, Carine Helena, and Zemel, Lawrence
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- 2021
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40. Real-World Effectiveness of Common Treatment Strategies for Juvenile Idiopathic Arthritis: Results From a Canadian Cohort.
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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., and Rosenberg, Alan M.
- Abstract
Objective: 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 JIA treatment strategies recommended in current guidelines.Methods: Children with JIA who 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.Results: 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% CI 59.8-69.7). Adding methotrexate to NSAIDs and/or joint injections (attempted 566 times) had a 60.5% success rate (95% CI 55.7-65.3). In adjusted analyses, each additional active joint reduced chances of success for treatment with NSAIDs (odds ratio [OR] 0.90 [95% CI 0.85-0.94]) and for methotrexate combinations (OR 0.96 [95% CI 0.94-0.99]). Each additional year after disease onset reduced chances of success for treatment with methotrexate combinations (OR 0.83 [95% CI 0.72-0.95]).Conclusion: 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. [ABSTRACT FROM AUTHOR]- Published
- 2020
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41. Worse Quality of Life, Function, and Pain in Children With Enthesitis, Irrespective of Their Juvenile Arthritis Category.
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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, and Dancey, Paul
- Subjects
RESEARCH ,PAIN ,RESEARCH methodology ,JUVENILE idiopathic arthritis ,EVALUATION research ,MEDICAL cooperation ,COMPARATIVE studies ,QUALITY of life ,QUESTIONNAIRES ,LONGITUDINAL method - Abstract
Objective: To estimate the impact of enthesitis on patient-reported outcomes in children with juvenile idiopathic arthritis (JIA), irrespective of JIA category.Methods: 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.Results: 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 JIA category 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% CI 0.07, 0.22), higher pain (3.01 versus 1.68, adjusted difference 0.94 points; 95% CI 0.64, 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.Conclusion: Children with enthesitis, regardless of JIA category, report worse patient-reported outcomes than those without enthesitis. Thus, enthesitis should be assessed in all children with JIA. [ABSTRACT FROM AUTHOR]- Published
- 2020
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42. Prospective Determination of the Incidence and Risk Factors of New-Onset Uveitis in Juvenile Idiopathic Arthritis: The Research in Arthritis in Canadian Children Emphasizing Outcomes Cohort.
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Lee, Jennifer J. Y., Duffy, Ciarán M., Guzman, Jaime, Oen, Kiem, Barrowman, Nick, Rosenberg, Alan M., Shiff, Natalie J., Boire, Gilles, Stringer, Elizabeth, Spiegel, Lynn, Morishita, Kimberly A., Lang, Bianca, Reddy, Deepti, Huber, Adam M., Cabral, David A., Feldman, Brian M., Yeung, Rae S. M., Tucker, Lori B., Watanabe Duffy, Karen, and ReACCh-Out Investigators
- Subjects
AUTOANTIBODIES ,RESEARCH ,AGE distribution ,RESEARCH methodology ,JUVENILE idiopathic arthritis ,DISEASE incidence ,EVALUATION research ,MEDICAL cooperation ,UVEITIS ,COMPARATIVE studies ,KAPLAN-Meier estimator ,RESEARCH funding ,LONGITUDINAL method ,PROPORTIONAL hazards models ,DISEASE complications - Abstract
Objective: Identification of the incidence of juvenile idiopathic arthritis (JIA)-associated uveitis and its risk factors is essential to optimize early detection. Data from the Research in Arthritis in Canadian Children Emphasizing Outcomes inception cohort were used to estimate the annual incidence of new-onset uveitis following JIA diagnosis and to identify associated risk factors.Methods: Data were reported every 6 months for 2 years, then yearly to 5 years. Incidence was determined by Kaplan-Meier estimators with time of JIA diagnosis as the reference point. Univariate log-rank analysis identified risk factors and Cox regression determined independent predictors.Results: In total, 1,183 patients who enrolled within 6 months of JIA diagnosis met inclusion criteria, median age at diagnosis of 9.0 years (interquartile range [IQR] 3.8-12.9), median follow-up of 35.2 months (IQR 22.7-48.3). Of these patients, 87 developed uveitis after enrollment. The incidence of new-onset uveitis was 2.8% per year (95% confidence interval [95% CI] 2.0-3.5) in the first 5 years. The annual incidence decreased during follow-up but remained at 2.1% (95% CI 0-4.5) in the fifth year, although confidence intervals overlapped. Uveitis was associated with young age (<7 years) at JIA diagnosis (hazard ratio [HR] 8.29, P < 0.001), positive antinuclear antibody (ANA) test (HR 3.20, P < 0.001), oligoarthritis (HR 2.45, P = 0.002), polyarthritis rheumatoid factor negative (HR 1.65, P = 0.002), and female sex (HR 1.80, P = 0.02). In multivariable analysis, only young age at JIA diagnosis and ANA positivity were independent predictors of uveitis.Conclusion: Vigilant uveitis screening should continue for at least 5 years after JIA diagnosis, and priority for screening should be placed on young age (<7 years) at JIA diagnosis and a positive ANA test. [ABSTRACT FROM AUTHOR]- Published
- 2019
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43. Soluble Low-density Lipoprotein Receptor-related Protein 1 in Juvenile Idiopathic Arthritis
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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.
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- 2021
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44. Growth and weight gain in children with juvenile idiopathic arthritis: results from the ReACCh-Out cohort
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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
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- 2017
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45. Malignancy in Pediatric-onset Systemic Lupus Erythematosus
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Bernatsky, Sasha, primary, Clarke, Ann E., additional, Zahedi Niaki, Omid, additional, Labrecque, Jeremy, additional, Schanberg, Laura E., additional, Silverman, Earl D., additional, Hayward, Kristen, additional, Imundo, Lisa, additional, Brunner, Hermine I., additional, Haines, Kathleen A., additional, Cron, Randy Q., additional, Oen, Kiem, additional, Wagner-Weiner, Linda, additional, Rosenberg, Alan M., additional, O’Neil, Kathleen M., additional, Duffy, Ciarán M., additional, von Scheven, Emily, additional, Joseph, Lawrence, additional, Lee, Jennifer L., additional, and Ramsey-Goldman, Rosalind, additional
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- 2017
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46. O35. THE AUTOIMMUNE GENETIC ARCHITECTURE OF CHILDHOOD-ONSET RHEUMATOID ARTHRITIS
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Hinks, Anne, primary, Marion, Miranda C., additional, Cobb, Joanna, additional, Sudman, Marc, additional, Ainsworth, Hannah, additional, Comeau, Mary, additional, Bohnsack, John, additional, Wedderburn, Lucy R., additional, Haas, Johannes-Peter, additional, Videm, Vibeke, additional, Rygg, Marite, additional, Nordal, Ellen, additional, Yeung, Rae S. M., additional, Rosenberg, Alan M., additional, Langefeld, Carl D., additional, Thompson, Susan D., additional, Thomson, Wendy, additional, and Prahalad, Sampath, additional
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- 2017
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47. Vitamin D status and anthropometric measurements of children with Juvenile Idiopathic Arthritis compared to healthy children using the Canadian Health Measures Survey
- Author
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Finch, Sarah L, primary, Rezaei, Elham L, additional, Whiting, Susan J, additional, Rosenberg, Alan M, additional, and Vatanparast, Hassan, additional
- Published
- 2017
- Full Text
- View/download PDF
48. Identification of Novel Adenosine Deaminase 2 Gene Variants and Varied Clinical Phenotype in Pediatric Vasculitis.
- Author
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Gibson, Kristen M., Morishita, Kimberly A., Dancey, Paul, Moorehead, Paul, Drögemöller, Britt, Han, Xiaohua, Graham, Jinko, Hancock, Robert E. W., Foell, Dirk, Benseler, Susanne, Luqmani, Rashid, Yeung, Rae S. M., Shenoi, Susan, Bohm, Marek, Rosenberg, Alan M., Ross, Colin J., Cabral, David A., and Brown, Kelly L.
- Subjects
AGE factors in disease ,ALLELES ,ENZYME-linked immunosorbent assay ,GENES ,GENETICS ,HYDROLASES ,IMMUNOBLOTTING ,POLYARTERITIS nodosa ,STROKE ,VASCULITIS ,PHENOTYPES ,SEQUENCE analysis ,GENOTYPES ,ANTINEUTROPHIL cytoplasmic antibodies - Abstract
Objective: Individuals with deficiency of adenosine deaminase 2 (DADA2), a recently recognized autosomal recessive disease, present with various systemic vascular and inflammatory manifestations, often with young age at disease onset or with early onset of recurrent strokes. Their clinical features and histologic findings overlap with those of childhood‐onset polyarteritis nodosa (PAN), a primary "idiopathic" systemic vasculitis. Despite similar clinical presentation, individuals with DADA2 may respond better to biologic therapy than to traditional immunosuppression. The aim of this study was to screen an international registry of children with systemic primary vasculitis for variants in ADA2. Methods: The coding exons of ADA2 were sequenced in 60 children and adolescents with a diagnosis of PAN, cutaneous PAN, or unclassifiable vasculitis (UCV), any chronic vasculitis with onset at age 5 years or younger, or history of stroke. The functional consequences of the identified variants were assessed by ADA2 enzyme assay and immunoblotting. Results: Nine children with DADA2 (5 with PAN, 3 with UCV, and 1 with antineutrophil cytoplasmic antibody–associated vasculitis) were identified. Among them, 1 patient had no rare variants in the coding region of ADA2 and 8 had biallelic, rare variants (minor allele frequency <0.01) with a known association with DADA2 (p.Gly47Arg and p.Gly47Ala) or a novel association (p.Arg9Trp, p.Leu351Gln, and p.Ala357Thr). The clinical phenotype varied widely. Conclusion: These findings support previous observations indicating that DADA2 has extensive genotypic and phenotypic variability. Thus, screening ADA2 among children with vasculitic rash, UCV, PAN, or unexplained, early‐onset central nervous system disease with systemic inflammation may enable an earlier diagnosis of DADA2. [ABSTRACT FROM AUTHOR]
- Published
- 2019
- Full Text
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49. Complex transposition with interrupted right aortic arch and partial Di George syndrome: Successful palliation with combined medical and surgical therapy
- Author
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Duncan, Walter J., Tyrrell, Michael J., Bharadwaj, Baikunth, Rosenberg, Alan M., Schroeder, Maria-Louise, and Bingham, William T.
- Published
- 1984
- Full Text
- View/download PDF
50. IL1RN Variation Influences Both Disease Susceptibility and Response to Recombinant Human Interleukin‐1 Receptor Antagonist Therapy in Systemic Juvenile Idiopathic Arthritis.
- Author
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INCHARGE Consortium, Arthur, Victoria L., Shuldiner, Emily, Szymanski, Ann Marie, Ombrello, Michael J., Bohnsack, John F., Ilowite, Norman T., Mellins, Elizabeth D., Russo, Ricardo, Len, Claudio, Oliveira, Sheila, Yeung, Rae S. M., Rosenberg, Alan M., Wedderburn, Lucy R., Anton, Jordi, Remmers, Elaine F., Kastner, Daniel L., Haas, Johannes‐Peter, Rösen‐Wolff, Angela, and Minden, Kirsten
- Subjects
BIOMARKERS ,CELL lines ,CELL receptors ,COMPUTER simulation ,CONFIDENCE intervals ,DISEASE susceptibility ,GENE expression ,GENETIC polymorphisms ,SEQUENCE analysis ,JUVENILE idiopathic arthritis ,ODDS ratio ,GENETICS ,DISEASE risk factors - Abstract
Objective: To determine whether systemic juvenile idiopathic arthritis (JIA) susceptibility loci that were identified by candidate gene studies demonstrate association with systemic JIA in the largest study population assembled to date. Methods: Single‐nucleotide polymorphisms (SNPs) from 11 previously reported systemic JIA risk loci were examined for association in 9 populations, including 770 patients with systemic JIA and 6,947 controls. The effect of systemic JIA–associated SNPs on gene expression was evaluated in silico in paired whole genome and RNA sequencing data from the lymphoblastoid cell lines (LCLs) of 373 European subjects from the 1000 Genomes Project. Responses of systemic JIA–associated SNPs to anakinra treatment were evaluated in 38 US patients for whom treatment response data were available. Results: We found no association between the previously reported 26 SNPs and systemic JIA. Expanded analysis of the regions containing the 26 SNPs revealed only 1 significant association: the promoter region of IL1RN (P < 1 × 10
–4 ). Systemic JIA–associated SNPs correlated with IL1RN expression in LCLs, with an inverse correlation between systemic JIA risk and IL1RN expression. The presence of homozygous IL1RN high expression alleles correlated strongly with a lack of response to anakinra therapy (odds ratio 28.7 [95% confidence interval 3.2–255.8]). Conclusion: In our study, IL1RN was the only candidate locus associated with systemic JIA. The implicated SNPs are among the strongest known determinants of IL1RN and interleukin‐1 receptor antagonist levels, linking low expression with increased systemic JIA risk. Homozygous high expression alleles predicted nonresponsiveness to anakinra therapy, making them ideal candidate biomarkers to guide systemic JIA treatment. This study is an important first step toward the personalized treatment of systemic JIA. [ABSTRACT FROM AUTHOR]- Published
- 2018
- Full Text
- View/download PDF
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