Two Cases of Cytomegalovirus Retinitis as a Manifestation of Good's Syndrome

거대세포바이러스 망막염으로 발현된 Good증후군 2예

  • Kim, Hye-In (Department of Internal Medicine, Kyungpook National University School of Medicine) ;
  • Chang, Hyun-Ha (Department of Internal Medicine, Kyungpook National University School of Medicine) ;
  • Park, Ga-Young (Department of Internal Medicine, Kyungpook National University School of Medicine) ;
  • Kwon, Eu-Gene (Department of Internal Medicine, Kyungpook National University School of Medicine) ;
  • Kim, Hyo-Hoon (Department of Internal Medicine, Kyungpook National University School of Medicine) ;
  • Kim, Shin-Woo (Department of Internal Medicine, Kyungpook National University School of Medicine) ;
  • Lee, Jong-Myung (Department of Internal Medicine, Kyungpook National University School of Medicine)
  • 김혜인 (경북대학교 의학전문대학원 내과학교실) ;
  • 장현하 (경북대학교 의학전문대학원 내과학교실) ;
  • 박가영 (경북대학교 의학전문대학원 내과학교실) ;
  • 권유진 (경북대학교 의학전문대학원 내과학교실) ;
  • 김효훈 (경북대학교 의학전문대학원 내과학교실) ;
  • 김신우 (경북대학교 의학전문대학원 내과학교실) ;
  • 이종명 (경북대학교 의학전문대학원 내과학교실)
  • Published : 2011.08.01

Abstract

Good's syndrome (GS) is a rare cause of acquired combined B- and T-cell immunodeficiency in adults. GS is also known as a rare form of paraneoplastic syndrome of thymoma that may persist after thymectomy. Some reported GS cases have been related to various diseases as a result of humoral immunodeficiency, but no report to date has related GS to cytomegalovirus (CMV) retinitis in Korea. We report two cases of CMV retinitis as a manifestation of GS with severe cellular immunodeficiency. In the first case, a 61-year-old woman was diagnosed with GS manifesting as CMV retinitis combined with coincident pulmonary tuberculosis and soft-tissue tuberculosis. In the second case, a 56-year-old man had CMV retinitis and CMV pneumonia. Both patients had a history of thymoma and had received total thymectomies, and were diagnosed with GS a few years thereafter.

Good증후군은 흉선종과 면역결핍을 나타내는 질환이다. 주로 체액성 면역결핍에 의한 세균감염이 발생하며 또한 세포성 면역결핍에 의하여 바이러스나 진균, 결핵 감염을 나타내기도 한다. Good증후군의 치료는 면역글로불린 정맥 주사이다. 시력저하를 주소로 내원한 두 환자에서 공통적으로 세포성 면역결핍에 의한 거대세포바이러스 망막염으로 발현한 Good증후군을 진단하였으며 증례 1의 환자는 폐 및 연부 조직 결핵을 동반하였고, 증례 2의 경우 거대세포바이러스 폐렴을 동반하였다. 두 환자 모두 ganciclovir 정맥주사 및 면역 글로불린 치료로 호전된 바 이에 저자들은 문헌고찰과 함께 보고하는 바이다.

Keywords

References

  1. Lee SH, Lee SM, Yang SC, et al. A case of granulomatous lung disease in a patient with Good's syndrome. Korean J Intern Med 2008;23:219-222. https://doi.org/10.3904/kjim.2008.23.4.219
  2. Kelleher P, Misbah SA. What is Good's syndrome? Immunological abnormalities in patients with thymoma. J Clin Pathol 2003;56:12-16. https://doi.org/10.1136/jcp.56.1.12
  3. Tarr PE, Sneller MC, Mechanic LJ, et al. Infections in patients with Immunedeficiency with thymoma (Good's syndrome): report of 5 cases and review of the literature. Medicine (Baltimore) 2001;80:123-133. https://doi.org/10.1097/00005792-200103000-00005
  4. Yong DS, Tsang MK, Chan EY, Tse DM. Good's syndrome in a patient with cytomegalovirus retinitis. Hong Kong Med J 2008;14:142-144.
  5. Good RA. Agammaglobulinaemia a provocative experiment of nature. Bull Univ Minn Hosp Med Found 1954;26:1-19.
  6. International union of Immunological Societies. Primary immunodeficiency disease: report of an IUIS Scientific Committee. Clin Exp Immunol 1999;118:1-28.
  7. Morgenthaler TI, Brown LR, Colby TV, Harper CM Jr, Coles DT. Thymoma. Mayo Clin Proc 1993;68:1110-1123. https://doi.org/10.1016/S0025-6196(12)60907-0
  8. Song KH, Kim KH, Kim CJ, et al. A case of Good's syndrome. Infect Chemother 2007;39:259-262.
  9. Ryoo JY. Thymoma with immunodeficiency: a case report. Korean J Thorac Cardiovasc 2006;35:85-89.
  10. Kim JY, Chin JY, Lee SS, et al. A case of Good syndrome: immunodeficiency syndrome associated with a thymoma. Korean J Med 2008;75:S935-S941.
  11. Oritani K, Medina KL, Tomiyama Y, et al. Limitin: an interferonlike cytokine that preferentially influences B-lymphocyte precursors. Nat Med 2000;6:659-666. https://doi.org/10.1038/76233
  12. Charles RJ, Sabo KM, Kidd PG, Abkowitz JL. The pathophysiology of pure red cell aplasia: implications for therapy. Blood 1996;87:4831-4838.
  13. Litwin SD, Zanjani ED. Lymphocytes suppressing both immunoglobulin production and erythroid differentiation in hypogammaglobulinaemia. Nature 1977;266:57-58. https://doi.org/10.1038/266057a0
  14. Hayward AR, Paolucci P, Webster AD, Kohler P. Pre-B cell suppression by thymoma patient lymphocytes. Clin Exp Immunol 1982;48:437-442.
  15. Ide S, Koga T, Rikimaru T, Katsuki Y, Oizumi K. Good's syndrome presenting with cytomegalovirus pneumonia. Intern Med 2000;39:1094-1096. https://doi.org/10.2169/internalmedicine.39.1094