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Clinical Courses of IKAROS and CTLA4 Deficiencies: A Systematic Literature Review and Retrospective Longitudinal Study.
Hoshino, Akihiro; Toyofuku, Etsushi; Mitsuiki, Noriko; Yamashita, Motoi; Okamoto, Keisuke; Yamamoto, Michio; Kanda, Kenji; Yamato, Genki; Keino, Dai; Yoshimoto-Suzuki, Yuri; Kamizono, Junji; Onoe, Yasuhiro; Ichimura, Takuya; Nagao, Mika; Yoshimura, Masaru; Tsugawa, Koji; Igarashi, Toru; Mitsui-Sekinaka, Kanako; Sekinaka, Yujin; Doi, Takehiko; Yasumi, Takahiro; Nakazawa, Yozo; Takagi, Masatoshi; Imai, Kohsuke; Nonoyama, Shigeaki; Morio, Tomohiro; Latour, Sylvain; Kanegane, Hirokazu.
Affiliation
  • Hoshino A; Department of Pediatrics and Developmental Biology, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University, Tokyo, Japan.
  • Toyofuku E; Laboratory of Lymphocyte Activation and Susceptibility to EBV Infection, INSERM UMR 1163, Imagine Institute, Paris, France.
  • Mitsuiki N; Department of Pediatrics and Developmental Biology, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University, Tokyo, Japan.
  • Yamashita M; Graduate School of Medicine and Faculty of Medicine, The University of Tokyo, Tokyo, Japan.
  • Okamoto K; Department of Pediatrics and Developmental Biology, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University, Tokyo, Japan.
  • Yamamoto M; Department of Pediatrics and Developmental Biology, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University, Tokyo, Japan.
  • Kanda K; Department of Pediatrics and Developmental Biology, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University, Tokyo, Japan.
  • Yamato G; Department of Pediatrics, Yokohama Municipal Citizen's Hospital, Yokohama, Japan.
  • Keino D; Department of Pediatrics, Hikone Municipal Hospital, Hikone, Japan.
  • Yoshimoto-Suzuki Y; Department of Hematology/Oncology, Gunma Children's Medical Center, Shibukawa, Japan.
  • Kamizono J; Division of Hematology/Oncology, Kanagawa Children's Medical Center, Yokohama, Japan.
  • Onoe Y; Department of Pediatrics, Center Hospital of the National Center for Global Health and Medicine, Tokyo, Japan.
  • Ichimura T; Department of Pediatrics, Kitakyushu City Yahata Hospital, Kitakyushu, Japan.
  • Nagao M; Department of Pediatrics, Kitakyushu Municipal Medical Center, Kitakyushu, Japan.
  • Yoshimura M; Department of Pediatrics, Yamaguchi University Graduate School of Medicine, Ube, Japan.
  • Tsugawa K; Department of Pediatrics, Ohta Nishinouchi Hospital, Koriyama, Japan.
  • Igarashi T; Department of Rheumatology, Endocrinology and Nephrology, Faculty of Medicine and Graduate School of Medicine, Hokkaido University, Sapporo, Japan.
  • Mitsui-Sekinaka K; Department of Pediatrics, Hirosaki University Graduate School of Medicine, Hirosaki, Japan.
  • Sekinaka Y; Department of Pediatrics, Nippon Medical School, Tokyo, Japan.
  • Doi T; Department of Pediatrics, National Defense Medical College, Tokorozawa, Japan.
  • Yasumi T; Department of Pediatrics, National Defense Medical College, Tokorozawa, Japan.
  • Nakazawa Y; Department of Pediatrics, Hiroshima University Graduate School of Biomedical and Health Science, Hiroshima, Japan.
  • Takagi M; Department of Pediatrics, Kyoto University Graduate School of Medicine, Kyoto, Japan.
  • Imai K; Department of Pediatrics, Shinshu University School of Medicine, Matsumoto, Japan.
  • Nonoyama S; Department of Pediatrics and Developmental Biology, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University, Tokyo, Japan.
  • Morio T; Department of Community Pediatrics, Perinatal and Maternal Medicine, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University, Tokyo, Japan.
  • Latour S; Department of Pediatrics, National Defense Medical College, Tokorozawa, Japan.
  • Kanegane H; Department of Pediatrics and Developmental Biology, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University, Tokyo, Japan.
Front Immunol ; 12: 784901, 2021.
Article in En | MEDLINE | ID: mdl-35087518
ABSTRACT
IKAROS and CTLA4 deficiencies are inborn errors of immunity and show similar clinical phenotypes, including hypogammaglobulinemia and autoimmune diseases (ADs). However, the differences in clinical features and pathogenesis of these are not fully understood. Therefore, we performed systematic literature reviews for IKAROS and CTLA4 deficiencies. The reviews suggested that patients with IKAROS deficiency develop AD earlier than hypogammaglobulinemia. However, no study assessed the detailed changes in clinical manifestations over time; this was likely due to the cross-sectional nature of the studies. Therefore, we conducted a retrospective longitudinal study on IKAROS and CTLA4 deficiencies in our cohort to evaluate the clinical course over time. In patients with IKAROS deficiency, AD and hypogammaglobulinemia often develop in that order, and AD often resolves before the onset of hypogammaglobulinemia; these observations were not found in patients with CTLA4 deficiency. Understanding this difference in the clinical course helps in the clinical management of both. Furthermore, our results suggest B- and T-cell-mediated ADs in patients with IKAROS and CTLA4 deficiencies, respectively.
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Full text: 1 Database: MEDLINE Main subject: Ikaros Transcription Factor / CTLA-4 Antigen / Metabolism, Inborn Errors Type of study: Observational_studies / Risk_factors_studies / Systematic_reviews Limits: Humans Language: En Year: 2021 Type: Article
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Full text: 1 Database: MEDLINE Main subject: Ikaros Transcription Factor / CTLA-4 Antigen / Metabolism, Inborn Errors Type of study: Observational_studies / Risk_factors_studies / Systematic_reviews Limits: Humans Language: En Year: 2021 Type: Article