TY - JOUR
T1 - Familial autoimmunity in the childhood arthritis and rheumatology research alliance registry
AU - Prahalad, Sampath
AU - McCracken, Courtney E.
AU - Ponder, Lori A.
AU - Angeles-Han, Sheila T.
AU - Rouster Stevens, Kelly A.
AU - Vogler, Larry B.
AU - Langefeld, Carl D.
AU - Thompson, Susan D.
AU - Abramson, L.
AU - Anderson, E.
AU - Andrew, M.
AU - Battle, N.
AU - Becker, M.
AU - Benham, H.
AU - Beukelman, T.
AU - Birmingham, J.
AU - Blier, P.
AU - Brown, A.
AU - Brunner, H.
AU - Cabrera, A.
AU - Canter, D.
AU - Carlton, D.
AU - Caruso, B.
AU - Ceracchio, L.
AU - Chalom, E.
AU - Chang, J.
AU - Charpentier, P.
AU - Clark, K.
AU - Dean, J.
AU - Dedeoglu, F.
AU - Feldman, B.
AU - Ferguson, P.
AU - Fox, M.
AU - Francis, K.
AU - Gervasini, M.
AU - Goldsmith, D.
AU - Gorton, G.
AU - Gottlieb, B.
AU - Graham, T.
AU - Griffin, T.
AU - Grosbein, H.
AU - Guppy, S.
AU - Haftel, H.
AU - Helfrich, D.
AU - Higgins, G.
AU - Hillard, A.
AU - Hollister, J. R.
AU - Hsu, J.
AU - Hudgins, A.
AU - Klein-Gitelman, M.
AU - and for The CARRA Registry Investigators
N1 - Funding Information:
Dr. Prahalad is supported by grants from The National Institute of Arthritis and Musculoskeletal and Skin Diseases (NIAMS) (01-AR060893), Arthritis Foundation and The Marcus Foundation Inc. CARRA Registry is supported by grants from NIAMS (RC2-AR058934), Friends of CARRA, the Arthritis Foundation, and the Duke Clinical Research Institute. Dr. Thompson is supported by grants from NIAMS (P01-AR048929 and P30-AR0470363), The Arthritis Foundation and Cincinnati Children’s Hospital Research Foundation. The content is solely the responsibility of the authors and does not necessarily represent the official views of the NIAMS or the National Institutes of Health. The authors would like to thank all participants and hospital sites that recruited patients for the CARRA Registry. The authors thank the following CARRA Registry site principal investigators and research coordinators: L. Abramson, E. Anderson, M. Andrew, N. Battle, M. Becker, H. Benham, T. Beukelman, J. Birmingham, P. Blier, A. Brown, H. Brunner, A. Cabrera, D. Canter, D. Carlton, B. Caruso, L. Ceracchio, E. Chalom, J. Chang, P. Charpentier, K. Clark, J. Dean, F. Dedeoglu, B. Feldman, P. Ferguson, M. Fox, K. Francis, M. Gervasini, D. Goldsmith, G. Gorton, B. Gottlieb, T. Graham, T. Griffin, H. Grosbein, S. Guppy, H. Haftel, D. Helfrich, G. Higgins, A. Hillard, J.R. Hollister, J. Hsu, A. Hudgins, C. Hung, A. Huttenlocher, N. Ilowite, A. Imlay, L. Imundo, C.J. Inman, J. Jaqith, R. Jerath, L. Jung, P. Kahn, A. Kapedani, D. Kingsbury, K. Klein, M. Klein-Gitelman, A. Kunkel, S. Lapidus, S. Layburn, T. Lehman, C. Lindsley, M. Macgregor-Hannah, M. Malloy, C. Mawhorter, D. McCurdy, K. Mims, N. Moorthy, D. Morus, E. Muscal, M. Natter, J. Olson, K. O’Neil, K. Onel, M. Orlando, J. Palmquist, M. Phillips, L. Ponder, S. Prahalad, M. Punaro, D. Puplava, S. Quinn, A. Quintero, C. Rabinovich, A. Reed, C. Reed, S. Ringold, M. Riordan, S. Roberson, A. Robinson, J. Rossette, D. Rothman, D. Russo, N. Ruth, K. Schikler, A. Sestak, B. Shaham, Y. Sherman, M. Simmons, N. Singer, S. Spalding, H. Stapp, R. Syed, E. Thomas, K. Torok, D. Trejo, J. Tress, W. Upton, R. Vehe, E. von Scheven, L. Walters, J. Weiss, P. Weiss, N. Welnick, A. White, J. Woo, J. Wootton, A. Yalcindag, C. Zapp, L. Zemel, and A. Zhu.
Publisher Copyright:
© 2016 Prahalad et al.
PY - 2016/3/10
Y1 - 2016/3/10
N2 - Background: Clinically distinct autoimmune phenotypes share genetic susceptibility factors. We investigated the prevalence of familial autoimmunity among subjects with juvenile idiopathic arthritis (JIA), childhood systemic lupus erythematosus (cSLE) and juvenile dermatomyositis (JDM) in the CARRA Registry, the largest multicenter observational Registry for pediatric rheumatic disease. Methods: Children with JIA, cSLE and JDM enrolled in the CARRA Registry between May 2010 and May 2012 were investigated for differences in proportion of subjects who had first-degree relatives (FDR) with autoimmunity. If a significant difference was detected, pairwise comparisons, adjusted for multiple comparisons, were made. Results: There were 4677 JIA, 639 cSLE and 440 JDM subjects. The proportion of subjects having FDR with any autoimmune disease in the JDM group (20.5%) was less compared to subjects with JIA (31.8%, p < 0.001) or SLE (31.9%; p < 0.001). Significantly greater proportion of JIA cases had FDR with inflammatory arthritis (13%) compared to cSLE (9.2%, p = 0.007) or JDM (4.3%, p <0.001). Significantly greater proportion of cSLE cases had FDR with SLE (11.1% vs. 1.7% for JIA and 1.1% for JDM p < 0.001) or type-I diabetes (7.4% for cSLE vs. 3.1% for JIA and 3.0% for JDM p < 0.001). Conclusion: Higher proportions of subjects with JIA and cSLE have FDR with autoimmunity compared to those of JDM. Relatives of cSLE cases had an increased prevalence of SLE, and relatives of JIA cases were enriched for inflammatory arthropathies demonstrating distinct patterns of familial autoimmunity among these phenotypes.
AB - Background: Clinically distinct autoimmune phenotypes share genetic susceptibility factors. We investigated the prevalence of familial autoimmunity among subjects with juvenile idiopathic arthritis (JIA), childhood systemic lupus erythematosus (cSLE) and juvenile dermatomyositis (JDM) in the CARRA Registry, the largest multicenter observational Registry for pediatric rheumatic disease. Methods: Children with JIA, cSLE and JDM enrolled in the CARRA Registry between May 2010 and May 2012 were investigated for differences in proportion of subjects who had first-degree relatives (FDR) with autoimmunity. If a significant difference was detected, pairwise comparisons, adjusted for multiple comparisons, were made. Results: There were 4677 JIA, 639 cSLE and 440 JDM subjects. The proportion of subjects having FDR with any autoimmune disease in the JDM group (20.5%) was less compared to subjects with JIA (31.8%, p < 0.001) or SLE (31.9%; p < 0.001). Significantly greater proportion of JIA cases had FDR with inflammatory arthritis (13%) compared to cSLE (9.2%, p = 0.007) or JDM (4.3%, p <0.001). Significantly greater proportion of cSLE cases had FDR with SLE (11.1% vs. 1.7% for JIA and 1.1% for JDM p < 0.001) or type-I diabetes (7.4% for cSLE vs. 3.1% for JIA and 3.0% for JDM p < 0.001). Conclusion: Higher proportions of subjects with JIA and cSLE have FDR with autoimmunity compared to those of JDM. Relatives of cSLE cases had an increased prevalence of SLE, and relatives of JIA cases were enriched for inflammatory arthropathies demonstrating distinct patterns of familial autoimmunity among these phenotypes.
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U2 - 10.1186/s12969-016-0075-7
DO - 10.1186/s12969-016-0075-7
M3 - Article
C2 - 26965173
AN - SCOPUS:84960427338
SN - 1546-0096
VL - 14
JO - Pediatric Rheumatology
JF - Pediatric Rheumatology
IS - 1
M1 - 14
ER -