Abstract
A 76-year-old woman with multiple coronary risk factors was admitted to our hospital because of episodes of new-onset chest pain that had begun 3 days previously. She underwent percutaneous coronary intervention (PCI) for severe stenoses in the two high lateral (HL) branches. Intravascular ultrasound (IVUS) revealed massive stenotic lesions in the HL branches and tumorous nonstenotic lesions in the left anterior descending coronary artery (LAD) and the left circumflex coronary artery (LCx). iMAP™, optical coherence tomography (OCT), and coronary computed tomography angiography (CCTA) were performed. iMAP depicted fibrosis in the vessel (green areas) and nonfibrotic tissue change suggestive of inflammation outside the vessel (yellow/red areas). OCT revealed high-intensity homogenous intimal hyperplasia with superficial calcification, and CCTA showed massive periarterial soft lesions in the HL, LAD, and LCx. The serum IgG4 level was high at 252–427 mg/dL (8 measurements) (reference range, 4.8–105.0 mg/dL). We suspected IgG4-related coronary periarteritis on the basis of the comprehensive diagnostic criteria as a possible diagnosis. The clinical course was good after initial and subsequent PCIs for both the HL stenoses and the progressing LCx stenosis, and there was no recurrence of angina pectoris thereafter. Steroids were not administered because the massive lesions did not enlarge during the 16 months of follow-up. iMAP was able to evaluate the tissue characteristics of tumorous lesions in the stenosed HL branches and the nonstenotic LAD and LCx in a patient with an elevated level of IgG4.
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References
Stone JH, Khosroshahi A, Deshpande V, Chan JK, Heathcote JG, Aalberse R, Azumi A, Bloch DB, Brugge WR, Carruthers MN, Cheuk W, Cornell L, Castillo CF, Ferry JA, Forcione D, Kloppel G, Hamilos DL, Kamisawa T, Kasashima S, Kawa S, Kawano M, Masaki Y, Notohara K, Okazaki K, Ryu JK, Saeki T, Sahani D, Sato Y, Smyrk T, Stone JR, Takahira M, Umehara H, Webster G, Yamamoto M, Yi E, Yoshino T, Zamboni G, Zen Y, Chari S (2012) Recommendations for the nomenclature of IgG4-related disease and its individual organ system manifestations. Arthritis Rheum 64:3061–3067
Hamano H, Kawa S, Horiuchi A, Unno H, Furuya N, Akamatsu T, Fukushima M, Nikaido T, Nakayama K, Usuda N, Kiyosawa K (2001) High serum IgG4 concentrations in patients with sclerosing pancreatitis. N Engl J Med 344:732–738
Kitagawa S, Zen Y, Harada K, Sasaki M, Sato Y, Minato H, Watanabe K, Kurumaya H, Katayanagi K, Masuda S, Niwa H, Tsuneyama K, Saito K, Haratake J, Takagawa K, Nakanuma Y (2005) Abundant IgG4-positive plasma cell infiltration characterizes chronic sclerosing sialadenitis (Kuttner’s tumor). Am J Surg Pathol 29:783–791
Kardar AH, Kattan S, Lindstedt E, Hanash K (2002) Steroid therapy for idiopathic retroperitoneal fibrosis: dose and duration. J Urol 168:550–555
Zen Y, Kasashima S, Inoue D (2012) Retroperitoneal and aortic manifestations of immunoglobulin G4-related disease. Semin Diagn Pathol 29:212–218
Van Moerkercke W, Verhamme M, Doubel P, Meeus G, Oyen R, Van Steenbergen W (2010) Autoimmune pancreatitis and extrapancreatic manifestations of IgG4-related sclerosing disease. Acta Gastroenterol Belg 73:239–246
Kasashima S, Zen Y, Kawashima A, Konishi K, Sasaki H, Endo M, Matsumoto Y, Kawakami K, Kasashima F, Moriya M, Kimura K, Ohtake H, Nakanuma Y (2008) Inflammatory abdominal aortic aneurysm: close relationship to IgG4-related periaortitis. Am J Surg Pathol 32:197–204
Matsumoto Y, Kasashima S, Kawashima A, Sasaki H, Endo M, Kawakami K, Zen Y, Nakanuma Y (2008) A case of multiple immunoglobulin G4-related periarteritis: a tumorous lesion of the coronary artery and abdominal aortic aneurysm. Hum Pathol 39:975–980
Kasashima S, Zen Y, Kawashima A, Endo M, Matsumoto Y, Kasashima F (2009) A new clinicopathological entity of IgG4-related inflammatory abdominal aortic aneurysm. J Vasc Surg 49:1264–1271 (discussion 1271)
Bito Y, Sasaki Y, Hirai H, Hosono M, Nakahira A, Suehiro Y, Kaku D, Kubota Y, Miyabe M, Suehiro S (2014) A surgical case of expanding bilateral coronary aneurysms regarded as immunoglobulin G4-related disease. Circulation 129:e453–e456
Ikutomi M, Matsumura T, Iwata H, Nishimura G, Ishizaka N, Hirata Y, Ono M, Nagai R (2011) Giant tumorous lesions (correction of legions) surrounding the right coronary artery associated with immunoglobulin-G4-related systemic disease. Cardiology 120:22–26
Tanigawa J, Daimon M, Murai M, Katsumata T, Tsuji M, Ishizaka N (2012) Immunoglobulin G4-related coronary periarteritis in a patient presenting with myocardial ischemia. Hum Pathol 43:1131–1134
Tanigawa J, Daimon M, Takeda Y, Katsumata T, Ishizaka N (2012) Temporal changes in serum IgG4 levels after coronary artery bypass graft surgery. Hum Pathol 43:2093–2095
Deshpande V, Zen Y, Chan JK, Yi EE, Sato Y, Yoshino T, Kloppel G, Heathcote JG, Khosroshahi A, Ferry JA, Aalberse RC, Bloch DB, Brugge WR, Bateman AC, Carruthers MN, Chari ST, Cheuk W, Cornell LD, Fernandez-Del Castillo C, Forcione DG, Hamilos DL, Kamisawa T, Kasashima S, Kawa S, Kawano M, Lauwers GY, Masaki Y, Nakanuma Y, Notohara K, Okazaki K, Ryu JK, Saeki T, Sahani DV, Smyrk TC, Stone JR, Takahira M, Webster GJ, Yamamoto M, Zamboni G, Umehara H, Stone JH (2012) Consensus statement on the pathology of IgG4-related disease. Mod Pathol 25:1181–1192
Okazaki K, Umehara H (2012) Are classification criteria for IgG4-RD now possible? The concept of IgG4-related disease and proposal of comprehensive diagnostic criteria in Japan. Int J Rheumatol 2012:357071
Umehara H, Okazaki K, Masaki Y, Kawano M, Yamamoto M, Saeki T, Matsui S, Sumida T, Mimori T, Tanaka Y, Tsubota K, Yoshino T, Kawa S, Suzuki R, Takegami T, Tomosugi N, Kurose N, Ishigaki Y, Azumi A, Kojima M, Nakamura S, Inoue D (2012) A novel clinical entity, IgG4-related disease (IgG4RD): general concept and details. Mod Rheumatol 22:1–14
Guo Y, Ansdell D, Brouha S, Yen A (2015) Coronary periarteritis in a patient with multi-organ IgG4-related disease. J Radiol Case Rep 9:1–17
Ishizaka N (2014) A suspected case of coronary periarteritis due to IgG4-related disease as a cause of ischemic heart disease. Forensic Sci Med Pathol 10:475–476
Tran MN, Langguth D, Hart G, Heiner M, Rafter A, Fleming SJ, Scalia GM (2015) IgG4-related systemic disease with coronary arteritis and aortitis, causing recurring critical coronary ischemia. Int J Cardiol 201:33–34
Treacy AD, Norita K, Ingram PJ, Sheppard MN (2015) Pseudotumour formation in atheromatous coronary arteries. J Forensic Leg Med 32:73–76
Ishizaka N (2013) IgG4-related disease underlying the pathogenesis of coronary artery disease. Clin Chim Acta 415:220–225
Urabe Y, Fujii T, Kurushima S, Tsujiyama S, Kihara Y (2012) Pigs-in-a-blanket coronary arteries: a case of immunoglobulin G4-related coronary periarteritis assessed by computed tomography coronary angiography, intravascular ultrasound, and positron emission tomography. Circ Cardiovasc Imaging 5:685–687
Gutierrez PS, Schultz T, Siqueira SA, de Figueiredo Borges L (2013) Sudden coronary death due to IgG4-related disease. Cardiovasc Pathol 22:505–507
Tanigawa J, Ishizaka N (2013) Optical coherence tomography in IgG4-related coronary periarteritis. Intern Med 52:289
Baruah D, Rubenstein J, Shahir K (2014) ‘Coronary wrap’: IgG4 related disease of coronary artery presenting as a mass lesion. Int J Cardiovasc Imaging 30:977–978
Tong AK, Tan SY, Go YY, Lam WW (2014) Cardiac structural abnormalities associated with IgG4-related coronary periarteritis and inflammation revealed by multimodality imaging. Can J Cardiol 30(956):e915–e957
Ebe H, Tsuboi H, Hagiya C, Takahashi H, Yokosawa M, Hagiwara S, Hirota T, Kurashima Y, Takai C, Miki H, Asashima H, Umeda N, Kondo Y, Ogishima H, Suzuki T, Chino Y, Matsumoto I, Sumida T (2015) Clinical features of patients with IgG4-related disease complicated with perivascular lesions. Mod Rheumatol 25:105–109
Araki T, Nakamura M, Utsunomiya M, Sugi K (2012) Visualization of coronary plaque in type 2 diabetes mellitus patients using a new 40 MHz intravascular ultrasound imaging system. J Cardiol 59:42–49
Araki T, Nakamura M, Utsunomiya M, Sugi K (2013) Visualization of coronary plaque in arterial remodeling using a new 40-MHz intravascular ultrasound imaging system. Catheter Cardiovasc Interv 81:471–480
Sathyanarayana S, Carlier S, Li W, Thomas L (2009) Characterisation of atherosclerotic plaque by spectral similarity of radiofrequency intravascular ultrasound signals. EuroIntervention 5:133–139
Raichlin E, Bae JH, Kushwaha SS, Lennon RJ, Prasad A, Rihal CS, Lerman A (2009) Inflammatory burden of cardiac allograft coronary atherosclerotic plaque is associated with early recurrent cellular rejection and predicts a higher risk of vasculopathy progression. J Am Coll Cardiol 53:1279–1286
Sakamoto A, Ishizaka N, Saito K, Imai Y, Morita H, Koike K, Kohro T, Nagai R (2012) Serum levels of IgG4 and soluble interleukin-2 receptor in patients with coronary artery disease. Clin Chim Acta 413:577–581
Kusumoto S, Kawano H, Takeno M, Kawahara F, Abe K, Hayashi H, Koide Y, Maemura K (2012) Mass lesions surrounding coronary artery associated with immunoglobulin G4-related disease. J Cardiol Cases 5:e150–e154
Nozue T, Yamamoto S, Tohyama S, Fukui K, Umezawa S, Onishi Y, Kunishima T, Sato A, Nozato T, Miyake S, Takeyama Y, Morino Y, Yamauchi T, Muramatsu T, Hibi K, Terashima M, Michishita I (2015) Comparison of the effects of pitavastatin versus pravastatin on coronary artery plaque phenotype assessed by tissue characterization using serial virtual histology intravascular ultrasound. Heart Vessels 30:36–44
Kawai H, Takakuwa Y, Naruse H, Sarai M, Motoyama S, Ito H, Iwase M, Ozaki Y (2015) Two cases with past Kawasaki disease developing acute myocardial infarction in their thirties, despite being regarded as at low risk for coronary events. Heart Vessels 30:549–553
Umehara H, Okazaki K, Masaki Y, Kawano M, Yamamoto M, Saeki T, Matsui S, Yoshino T, Nakamura S, Kawa S, Hamano H, Kamisawa T, Shimosegawa T, Shimatsu A, Ito T, Notohara K, Sumida T, Tanaka Y, Mimori T, Chiba T, Mishima M, Hibi T, Tsubouchi H, Inui K, Ohara H (2012) Comprehensive diagnostic criteria for IgG4-related disease (IgG4-RD), 2011. Mod Rheumatol 22:21–30
Yu KH, Chan TM, Tsai PH, Chen CH, Chang PY (2015) Diagnostic performance of serum IgG4 levels in patients with IgG4-Related disease. Medicine (Baltimore) 94:e1707
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Ito, S., Hasuo, T. & Nimura, Y. iMAP™ imaging of tumorous lesions surrounding the coronary arteries in a patient with an elevated serum level of immunoglobulin G4. Heart Vessels 31, 2061–2067 (2016). https://doi.org/10.1007/s00380-016-0852-8
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DOI: https://doi.org/10.1007/s00380-016-0852-8