A case of giant right coronary artery aneurysm due to IgG4-related disease
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CASE REPORT
A case of giant right coronary artery aneurysm due to IgG4‑related disease Sho Matsuyama1 · Takehiro Kishigami1 · Masato Sakamoto1 Received: 3 October 2019 / Accepted: 10 December 2019 © The Japanese Association for Thoracic Surgery 2019
Abstract A man diagnosed with immunoglobulin G4 (IgG4)-related disease at the age of 65 years underwent abdominal aortic replacement due to an abdominal aortic aneurysm. In the same hospitalization period, a small coronary artery aneurysm was noticed. He was treated with corticosteroids and his serum IgG levels returned to normal. After experiencing sudden chest pain at age 74 years, coronary angiography showed that the size of the aneurysm had increased dramatically. He underwent coronary artery bypass graft and coronary artery resection without using cardiopulmonary bypass. Thus, we conclude that observation of aneurysms in patients with IgG4-related disease is important, even under corticosteroid therapy. Keywords Coronary artery aneurysm · IgG4-related disease · Off-pump CABG
Introduction In 2001, the patient considered in this case study was diagnosed with immunoglobulin G4-related disease (IgG4-RD) after manifestation of autoimmune pancreatitis [1, 2]. IgG4RD can cause inflammation and sclerotic lesions in various organs due to infiltration of IgG4-positive lymphoplasmacytes. Furthermore, inflammatory abdominal aortic aneurysm (IAAA) is one of the most common lesions associated with IgG4-RD [2, 3]. This is due to IgG4-positive cells infiltrating the adventitia causing inflammation and sclerotic lesions, resulting in IAAA. In addition, pseudotumor lesions often develop around the coronary artery in IgG4-RD and coronary artery aneurysmal (CAA) changes can occur at the same site. Because lymphoplasmacytic infiltration and fibrous proliferation were found in the pseudotumor lesion, the cause of the CAA in the patient was thought to be similar to the development of IAAA. We previously reported a case of IgG4-RD that was complicated by IAAA and right CAA [4]. In the present case, corticosteroid therapy was started after surgery for IAAA and the CAA temporarily improved. However, the CAA * Sho Matsuyama [email protected]‑u.ac.jp 1
Department of Cardiovascular Surgery, Kitakyushu Municipal Medical Center, 2‑1‑1 Bashaku, Kokurakita‑ku, Fukuoka 802‑0077, Japan
gradually became larger despite normalization of serum IgG level and reduction in the level of IgG4. We report this case, because little is known about the relapse of cardiovascular lesions of IgG4-RD after steroid therapy.
Case A 65-year-old man was diagnosed with IgG4-RD and was being treated with 5 mg/day of prednisolone at our hospital. A right coronary artery (RCA) aneurysm (RCAA) measuring 2 × 2 cm at the distal segment and a stenotic region of the atrioventricular (AV) branch was detected by coronary angiography (Fig. 1a). The patient also had an IAAA and undergone open surgery during the same hospitalization period. His serum IgG level improved from 2988 to 962 mg/ dL after receiving cortico
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