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      Graves disease following rabbit antithymocyte globulin treatment of severe aplastic anemia in a Korean child

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      https://www.riss.kr/link?id=A104549520

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      다국어 초록 (Multilingual Abstract) kakao i 다국어 번역

      Antithymocyte globulin (ATG) is used as an immunosuppressive treatment (IST) to deplete clonal suppressor T cells in patients with severe aplastic anemia (SAA). The depletion of suppressor T cells by ATG may affect the activation of B cells, which results in an increased risk for autoimmune conditions. A 12-year-old boy was diagnosed with idiopathic SAA. As he did not have an human leukocyte antigen-matched sibling, he was treated with rabbit ATG (3.5 mg/kg/day for 5 days) and cyclosporine. Five months later, he became transfusion independent. However, 23 months after IST, he complained of mild hand tremors, sweating, weight loss, palpitations, and goiter. Results of thyroid function tests revealed hyperthyroidism (free thyroxine, 3.42 ng/dL; thyroid stimulating hormone [TSH], <0.01 nIU/mL; triiodothyronine, 3.99 ng/mL). Results of tests for autoantibodies were positive for the antimicrosome antibody and TSH-binding inhibitory immunoglobulin, but negative for the antithyroglobulin antibody and antinuclear antibody. He was treated with methimazole, and his symptoms improved. The patient has been disease free for 39 months after IST and 9 months after methimazole treatment. This case report suggests that although rare, rabbit ATG may have implications in the pathogenesis of autoimmune hyperthyroidism. Our findings suggest that thyroid function tests should be incorporated in the routine follow-up of SAA patients treated with ATG.
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      Antithymocyte globulin (ATG) is used as an immunosuppressive treatment (IST) to deplete clonal suppressor T cells in patients with severe aplastic anemia (SAA). The depletion of suppressor T cells by ATG may affect the activation of B cells, which res...

      Antithymocyte globulin (ATG) is used as an immunosuppressive treatment (IST) to deplete clonal suppressor T cells in patients with severe aplastic anemia (SAA). The depletion of suppressor T cells by ATG may affect the activation of B cells, which results in an increased risk for autoimmune conditions. A 12-year-old boy was diagnosed with idiopathic SAA. As he did not have an human leukocyte antigen-matched sibling, he was treated with rabbit ATG (3.5 mg/kg/day for 5 days) and cyclosporine. Five months later, he became transfusion independent. However, 23 months after IST, he complained of mild hand tremors, sweating, weight loss, palpitations, and goiter. Results of thyroid function tests revealed hyperthyroidism (free thyroxine, 3.42 ng/dL; thyroid stimulating hormone [TSH], <0.01 nIU/mL; triiodothyronine, 3.99 ng/mL). Results of tests for autoantibodies were positive for the antimicrosome antibody and TSH-binding inhibitory immunoglobulin, but negative for the antithyroglobulin antibody and antinuclear antibody. He was treated with methimazole, and his symptoms improved. The patient has been disease free for 39 months after IST and 9 months after methimazole treatment. This case report suggests that although rare, rabbit ATG may have implications in the pathogenesis of autoimmune hyperthyroidism. Our findings suggest that thyroid function tests should be incorporated in the routine follow-up of SAA patients treated with ATG.

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      참고문헌 (Reference)

      1 Volpe R., "The immunoregulatory disturbance in autoimmune thyroid disease" 2 : 55-72, 1988

      2 Kumar M, "Severe aplastic anaemia and Grave's disease in a paediatric patient" 118 : 327-329, 2002

      3 Zomas A, "Rapid progression of fibrosing alveolitis and thyrotoxicosis after antithymocyte globulin therapy for aplastic anemia" 71 : 49-51, 1995

      4 Feng X, "Rabbit ATG but not horse ATG promotes expansion of functional CD4+CD25highFOXP3+ regulatory T cells in vitro" 111 : 3675-3683, 2008

      5 Teramura M, "Mechanism of action of antithymocyte globulin in the treatment of aplastic anaemia: in vitro evidence for the presence of immunosuppressive mechanism" 96 : 80-84, 1997

      6 Schroder K, "Interferon-gamma: an overview of signals, mechanisms and functions" 75 : 163-189, 2004

      7 Nishikawa T, "Interferon-gamma inhibition of human thyrotropin receptor gene expression" 77 : 1084-1089, 1993

      8 Yoshikawa N, "IFN-gamma has a protective role against thyroid-specific autoantibody production in severe combined immunodeficient(SCID)mice xenografted with Graves' thyroid tissue" 6 : 437-443, 1996

      9 Scheinberg P, "Horse versus rabbit antithymocyte globulin in acquired aplastic anemia" 365 : 430-438, 2011

      10 Todd A, "Graves' disease following successful treatment of severe aplastic anaemia with antilymphocyte globulin" 21 : 69-70, 1999

      1 Volpe R., "The immunoregulatory disturbance in autoimmune thyroid disease" 2 : 55-72, 1988

      2 Kumar M, "Severe aplastic anaemia and Grave's disease in a paediatric patient" 118 : 327-329, 2002

      3 Zomas A, "Rapid progression of fibrosing alveolitis and thyrotoxicosis after antithymocyte globulin therapy for aplastic anemia" 71 : 49-51, 1995

      4 Feng X, "Rabbit ATG but not horse ATG promotes expansion of functional CD4+CD25highFOXP3+ regulatory T cells in vitro" 111 : 3675-3683, 2008

      5 Teramura M, "Mechanism of action of antithymocyte globulin in the treatment of aplastic anaemia: in vitro evidence for the presence of immunosuppressive mechanism" 96 : 80-84, 1997

      6 Schroder K, "Interferon-gamma: an overview of signals, mechanisms and functions" 75 : 163-189, 2004

      7 Nishikawa T, "Interferon-gamma inhibition of human thyrotropin receptor gene expression" 77 : 1084-1089, 1993

      8 Yoshikawa N, "IFN-gamma has a protective role against thyroid-specific autoantibody production in severe combined immunodeficient(SCID)mice xenografted with Graves' thyroid tissue" 6 : 437-443, 1996

      9 Scheinberg P, "Horse versus rabbit antithymocyte globulin in acquired aplastic anemia" 365 : 430-438, 2011

      10 Todd A, "Graves' disease following successful treatment of severe aplastic anaemia with antilymphocyte globulin" 21 : 69-70, 1999

      11 Killick SB, "Effects of antithymocyte globulin on bone marrow CD34+ cells in aplastic anaemia and myelodysplasia" 108 : 582-591, 2000

      12 Scheinberg P, "Distinct EBV and CMV reactivation patterns following antibody-based immunosuppressive regimens in patients with severe aplastic anemia" 109 : 3219-3224, 2007

      13 Ginsberg J., "Diagnosis and management of Graves’ disease" 168 : 575-585, 2003

      14 Young NS, "Current concepts in the pathophysiology and treatment of aplastic anemia" 108 : 2509-2519, 2006

      15 Hinterberger-Fischer M, "Coincidence of severe aplastic anaemia with multiple sclerosis or thyroid disorders: report of 5 cases" 92 : 136-139, 1994

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      학술지 이력

      학술지 이력
      연월일 이력구분 이력상세 등재구분
      2023 평가 해외DB학술지평가 신청대상 (해외등재 학술지 평가)
      2020-01-15 학술지명변경 한글명 : Korean Journal of Pediatrics -> Clinical and Experimental Pediatrics
      외국어명 : Korean Journal of Pediatrics -> Clinical and Experimental Pediatrics
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      2020-01-01 등재 등재학술지 유지 (해외등재 학술지 평가) KCI등재
      2019-07-16 학회명변경 한글명 : 대한소아과학회 -> 대한소아청소년과학회 KCI등재
      2010-01-01 등재 등재학술지 유지 (등재유지) KCI등재
      2009-01-30 학술지명변경 한글명 : 소아과 -> Korean Journal of Pediatrics KCI등재
      2008-01-01 등재 등재학술지 유지 (등재유지) KCI등재
      2006-01-01 등재 등재학술지 유지 (등재유지) KCI등재
      2003-01-01 등재 등재학술지 선정 (등재후보2차) KCI등재
      2002-01-01 등재 등재후보 1차 PASS (등재후보1차) KCI등재후보
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      학술지 인용정보

      학술지 인용정보
      기준연도 WOS-KCI 통합IF(2년) KCIF(2년) KCIF(3년)
      2016 0.18 0.18 0.16
      KCIF(4년) KCIF(5년) 중심성지수(3년) 즉시성지수
      0.17 0.2 0.369 0.06
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