A Case of Immunoglobulin G4-Related Cardiac Tumor around the Coronary Artery

Abstract

IgG4 関連疾患はさまざまな臓器に起こりうるが，多くは腺管臓器においてであり，心血管領域での報告は 少ない．今回われわれは IgG4 関連疾患による冠動脈周囲腫瘍の 1 例を経験したので報告する．症例は 69 歳 男性．胸部 CT 上，2 年前から増大し続ける，冠動脈周囲の腫瘍性病変にたいし，確定診断目的で開胸生検 術を行った．病理組織診断では線維性結合組織内に形質細胞とリンパ球の密な増生を認め，好酸球も認めら れた．全体的に線維化が目立ち，一部で肥厚した膠原線維をともなっていた．免疫染色では形質細胞の多く は IgG4 陽性であり，生検後測定した血漿 IgG4 濃度は 1,080 mg/dl と高値を示した．以上から IgG4 関連疾 患の冠動脈周囲腫瘍と診断した．心血管領域の IgG4 関連疾患は非常に稀であり，本症例は IgG4 関連疾患 による冠動脈周囲炎が腫瘍性病変を呈したものと思われた．日心外会誌 39 巻 4 号：230-233(2010) キーワード：IgG4 関連疾患，心臓腫瘍，冠動脈周囲腫瘍

Mass lesions surrounding coronary artery associated with immunoglobulin G4-related disease

“…However, the involvement of coronary artery is very rare. To the best of our knowledge, only 5 cases of IgG4related tumorous lesions surrounding the coronary artery have been previously reported [6][7][8][9][10]. All 6 cases, including the present case, were reported in Japan.…”

“…IgG4-related disease is usually sensitive to steroid therapy. The present case and two previous cases were treated with oral prednisolone [7,8], and two cases were treated with surgical resection of the coronary aneurysm and/or abdominal aneurysm without steroid therapy [6,9], and one case was finally treated with surgical resection of the coronary aneurysm because steroid therapy was not fully effective for the coronary lesion [10].…”

“…The involvement of aorta in IgG4-related disease is relatively common and is often associated with retroperitoneal fibrosis. However, only several cases of the involvement of coronary artery have been reported so far [6][7][8][9][10]. We report a case of mass lesions surrounding coronary artery…”

A clinicopathologic study of immunoglobulin G4-related disease of the femoral and popliteal arteries in the spectrum of immunoglobulin G4-related periarteritis

“…[3][4][5][6] In contrast to the abdominal aorta, thoracic aortic lesions of IgG4-RD were observed to take different forms according to various clinicopathologic features, including inflammatory aneurysms, lymphoplasmacytic aortitis, isolated aortitis, and aortic dissection. [7][8][9][10][11] Furthermore, IgG4-related vascular lesions have been observed to spread to medium-sized arteries, such as the coronary arteries, 12,13 and to the first to second branching arteries of the aorta. 14,15 Some cases show periarterial mass formation and an absence of significant arterial dilatation.…”

“…12,15 Histopathologically, IgG4-related arterial lesions have been characterized by arterial wall thickening, corresponding to inflammation with IgG4-positive plasmacytes, and fibrosis mainly in the adventitia. [12][13][14][15] Arteriosclerosis obliterans (ASO) is the most frequent disease of the peripheral arteries (common, deep, superficial femoral, and popliteal arteries), whereas aneurysmal changes resulting in peripheral artery aneurysms (PAAs) are very rare compared with aortic lesions and are Ͻ10% as common as AAAs. 16,17 In particular, inflammatory aneurysms in the peripheral arteries are extremely rare.…”