A rare case of immunoglobulin G4-related disease presenting with coronary artery pseudotumor and aneurysm
  • Sukrisd Koowattanatianchai
    Division of Cardiology, Department of Medicine, Faculty of Medicine, Burapha University, Chonburi, Thailand
  • Nichakarn Teawprasert
    Department of Medicine, Faculty of Medicine, Burapha University, Chonburi, Thailand
  • Sornsupha Limchareon
    Department of Radiology and Nuclear Medicine, Faculty of Medicine, Burapha University, Chonburi, Thailand
  • Yodying Kaolawanich
    Division of Cardiology, Department of Medicine, Faculty of Medicine Siriraj Hospital, Mahidol University, Bangkok, Thailand

Keywords

Coronary aneurysm, coronary artery disease, immunoglobulin G4-related disease, pseudotumor

Abstract

Immunoglobulin G4-related disease (IgG4-RD) is an autoimmune disease characterized by elevated serum IgG4 levels. It has the potential to affect multiple organs. Despite the diverse manifestations of IgG4-RD, the association with coronary artery disease (CAD) remains poorly understood due to limited evidence. We report the case of a 52-year-old male patient who exhibited typical angina upon exertion, accompanied by elevated serum IgG4 levels. Coronary computed tomographic angiography (CCTA) revealed the presence of pseudotumor formations surrounding and aneurysm changes affecting all coronary arteries, consistent with IgG4-RD. The patient was treated with prednisolone and azathioprine, with the possibility of additional rituximab therapy if symptomatology failed to improve. This case sheds light on the rare occurrence of IgG4-RD with coronary artery involvement and underscores the importance of recognizing this unique clinical entity for appropriate management and further research.

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References

  • Pourafkari M, Pal P, Luk A, Ennis D, Pakkal M, Rogalla P. New cardiac manifestation of IgG4-related disease: a case report. Br J Cardiol 2020;27:28.
  • Tajima M, Nagai R, Hiroi Y. IgG4-related cardiovascular disorders. Int Heart J 2014;55:287-295.
  • Matsumoto Y, Kasashima S, Kawashima A, Sasaki H, Endo M, Kawakami K, et al. A case of multiple immunoglobulin G4-related periarteritis: a tumorous lesion of the coronary artery and abdominal aortic aneurysm. Hum Pathol 2008;39:975-980.
  • Kamisawa T, Zen Y, Pillai S, Stone JH. IgG4-related disease. Lancet 2015;385:1460-1471.
  • Guo Y, Ansdell D, Brouha S, Yen A. Coronary periarteritis in a patient with multi-organ IgG4-related disease. J Radiol Case Rep 2015;9:1-17.
  • Okuyama T, Tanaka TD, Nagoshi T, Yoshimura M. Coronary artery disease concomitant with immunoglobulin G4-related disease: a case report and literature review. Eur Heart J Case Rep 2019;3:ytz013.
  • Umehara H, Okazaki K, Kawa S, Takahashi H, Goto H, Matsui S, et al. The 2020 revised comprehensive diagnostic (RCD) criteria for IgG4-RD. Mod Rheumatol 2021;31:529-533.
  • Kusunose K, Hotchi J, Takagawa Y, Nishio S, Ise T, Tobiume T, et al. Serial imaging changes during treatment of immunoglobulin G4-related disease with multiple pseudotumors. Circulation 2015;131:1882-1883.
  • Khosroshahi A, Wallace ZS, Crowe JL, Akamizu T, Azumi A, Carruthers MN, et al. International Consensus Guidance Statement on the Management and Treatment of IgG4-Related Disease. Arthritis Rheumatol 2015;67:1688-1699.
  • Perugino CA, Mattoo H, Mahajan VS, Maehara T, Wallace ZS, Pillai S, et al. Emerging Treatment Models in Rheumatology: IgG4-Related Disease: Insights Into Human Immunology and Targeted Therapies. Arthritis Rheumatol 2017;69:1722-1732.
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    Published: 2023-12-06
    Issue: 2024: Vol 11 No 1 (view)


    How to cite:
    1.
    Koowattanatianchai S, Teawprasert N, Limchareon S, Kaolawanich Y. A rare case of immunoglobulin G4-related disease presenting with coronary artery pseudotumor and aneurysm. EJCRIM 2023;11 doi:10.12890/2023_004215.