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J Cardiol 2004 Oct; 44 4 : 153 159 Churg-Strauss : 1 Survivor of Cardiogenic Shock Following Acute Myocardial Infarction With Churg-Strauss Syndrome : First Angiographic Documention of Coronary Recanalization of Infarct-Related Arteries: A Case Report Takeyuki Junichi Michiaki Hidekazu Hiroshi Tetsuya Hisayuki Kenji Shinichiro Yosuke Teruhiko Masayuki Tetsushi Satoru Yuji Makoto Kamon Toshinobu KUBOTA, MD YAMAGUCHI, MD HIGASHITANI, MD MATSUSHIMA, MD SAKAMOTO, MD ISHIKAWA, MD OKADA, MD ENTA, MD TAKIZAWA, MD NAKANO, MD SUZUKI, MD MIZUNO, MD TSURUSAKI, MD ONODA, MD FUDA, MD MUTO, MD IMAI, MD, FJCC HORIE, MD, FJCC Abstract A 42-year-old man was treated under a diagnosis of Churg-Strauss syndrome with predonisolone pulse therapy. Three days later, he developed cardiogenic shock following acute myocardial infarction. Coronary angiography showed total occlusions in three peripheral coronary vessels. Intraaortic balloon pumping was used to maintain hemodynamics and predonisolone pulse therapy was repeated. However, he developed cardiogenic shock again after the second pulse therapy and needed percutaneous cardiopulmonary support and intraaortic balloon pumping. Accordingly, combination therapy of predonisolone and cyclophosphamide was given. He then recovered. Follow-up angiography showed recanalization of the infarct-related arteries. J Cardiol 2004 Oct; 44 4 : 153 159 : 360 0105 1696 Departments of Cardiology and Respiratory Medicine, Saitama Cardiovascular and Respiratory Center, Saitama Address for correspondence : KUBOTA T, MD, Department of Cardiology, Saitama Cardiovascular and Respiratory Center, Itai 1696, Konan-cho, Ohsato-gun, Saitama 360 0105 Manuscript received January 26, 2004 ; revised May 13, 2004 ; accepted May 13, 2004 153

154 Key Words Complications Churg-Strauss syndrome Myocardial infarction, treatment Coronary artery disease Angiography Assisted circulation Interventional cardiology Churg-Strauss Churg-Strauss syndrome : CSS Churg Strauss 1,2 3 CSS 4 CSS intraaortic balloon pumping : IABP percutaneous cardiopulmonary support: PCPS CSS 3 42 : : : 20 /20 : : 1995 2001 10mg/day 2002 12 2003 1 3 X 3 20 Table 1 CSS 13 164 4 17 4 5 5 11 X CSS : 178cm68 kg 35.8 C140/min70/50mmHg Table 1 Laboratory data on admission March 20, 2003 Complete blood cell count WBC 8,400/ l Neu 50.50% Eo 27.30% Lym 18.10% Mono 3.80% Baso 0.30% RBC 4.32 10 4 / l Hb 12.1 g/dl Ht 36.70% Plt 38.2 10 4 / l MCV 84.9 MCH 27.9 MCHC 32.9 ESR 102 mm/hr Coagulation system Normal Urinalysis No abnormality Blood chemistry TP 8.2 g/dl Alb 3.9 g/dl T-Bil 0.5 g/dl AST 17 IU/l ALT 17 IU/l LDH 235 IU/l -GTP 27 IU/l CK 66 IU/l AMY 78 mg/dl BUN 6 mg/dl Cr 0.6 mg/dl Na 140 meq/l K 4.4 meq/l Cl 102 meq/l TC 200 mg/dl TG 76 mg/dl HDL-C 63 mg/dl CRP 7.5 mg/dl Immunological findings ANA Rheumatoid factor C3 101 C4 40.4 CH50 33 IgG 2,333 mg/dl IgA 361 mg/dl IgM 201 mg/dl IgE 213 mg/dl C-ANCA P-ANCA Arterial blood gas room air ph 7.431 PO 2 75.5 mmhg PCO 2 40.2 mmhg BE 2.3 meq/l O 2 Sat 97.30% J Cardiol 2004 Oct; 44 4 : 153 159

Churg-Strauss 155 Fig. 1 Chest radiograph X : 55% Fig. 1 : a F QS 1 5 R 1 2 ST Fig. 2 : CK CK-MB AST LDHC C-reactive protein : CRP Table 2 Fig. 3 : 34% Killip American Heart Association Table 2 Fig. 2 Complete blood cell count WBC 15,000/ l Neu 85.5% Eo 0.2% Lym 8.1% Mono 6.00% Baso 0.20% RBC 4.53 10 4 / l Hb 12.7 g/dl Ht 36.70% Plt 30.3 10 4 / l Arterial blood gas face mask 10l ph 7.431 PO 2 75.5 mmhg PCO 2 40.2 mmhg BE 2.3 meq/l O 2 Sat 97.30% Electrocardiogram Laboratory data just before transfer to the coronary care unit April 5, 2003 Blood chemistry TP 7.4 g/dl Alb 3.2 g/dl T-Bil 0.6 g/dl AST 150 IU/l ALT 34 IU/l LDH 1,402 IU/l -GTP 27 IU/l CK 1,391 IU/l AMY 78 mg/dl BUN 13 mg/dl Cr 0.7 mg/dl Na 136 meq/l K 4.6 meq/l Cl 97 meq/l CRP 23.20 mg/dl J Cardiol 2004 Oct; 44 4 : 153 159

156 Fig. 3 Clinical course PSL predonisolone; WBC white blood cell; Eosino eosinophilic leucocyte; CRP C-reactive protein ; CAG coronary angiogram ; IABP intraaortic balloon pumping ; PCPS percutaneous cardiopulmonary support. : 8 9 13 4 Figs. 4 A B D E IABP Fig. 3 IABP CRP Table 2 Fig. 32 20 IABP 21 a F 4 6 Q 36% 22 4 11 IABP PCPS Fig. 3 3 50mg/day 23 PCPS 27 IABP 35 4 23 20 mg/day 79 CRP Fig. 3 X 91 6 18 64/20 mmhg 40 mmhg Figs. 4 C F 4 95 CRP 99 a F 4 6 Q J Cardiol 2004 Oct; 44 4 : 153 159

Churg-Strauss 157 Fig. 4 Coronary angiograms A, B: Left coronary artery B: higher magnification of white square in A on April 5, 2003. Total occlusions of segments 8, 9 and 13 were observed white arrows. C: Left coronary artery on June 8, 2003. Improved coronary flow was observed blank arrows. D, E : Right coronary artery E : higher magnification of white square in D on April 5, 2003. Total occlusion of segment 4 atrioventricular node artery was observed white arrow. F : Right coronary artery on June 8, 2003. Improved coronary flow was observed blank arrow. 46% 142 CSS 1 2 T 5 3 4 6 7,8 major basic protein eosinophil cationic protein 9,10 major basic protein eosinophil cationic protein 11 CSS CSS 12,13 J Cardiol 2004 Oct; 44 4 : 153 159

158 CRP CRP CRP CSS 4,14 IABP PCPS CSS CSS CSS IABP PCPS CRP CSS CSS 42 Churg-Strauss 3 2 Churg-Strauss J Cardiol 2004 Oct; 44 4 :153 159 1 Churg J, Strauss L : Allergic granulomatosis, allergic angiitis, and periarteritis nodosa. Am J Path 1951; 27 : 277 301 2 : 1989; 78 : 352 356 3: 1999; 15 Suppl : 179 184 4 Suzuki N, Arai Y, Miyamoto Y, Isokane N, Fukushima N, Sano : Acute myocardial injury and repeated angina pectoris-like attacks in a young patient with Churg-Strauss Syndrome. Jpn J Thorac Dis 1991; 29: 1630 1637 J Cardiol 2004 Oct; 44 4 : 153 159

Churg-Strauss 159 5: Churg-Strauss 1999; 87 : 807 811 6 : in 19821983; pp 408 419 7 : Churg- Strauss 1 Jpn Circ J 1999 ; 62 Suppl : 953 abstr 8 Kozak M, Gill EA, Green LS : The Churg-Strauss syndrome : A case report with angiographically documented coronary involvement and a review of the literature. Chest 1995; 107: 578 580 9 : 1 2000; 35 : 189 196 10 Egashira K, Tomoike H, Yamamoto Y, Yamada A, Hayashi Y, Nakamura M : Histamine-induced coronary spasm in regions of intimal thickening in miniature pigs: Roles of serum cholesterol and spontaneous or induced intimal thickening. Circulation 1986; 74 :826 837 11 Tai PC, Hayes DJ, Clark JB, Spry CJ : Toxic effects of human eosinophil products on isolated rat heart cells in vitro. Biochem J 1982; 204: 75 80 12 : Churg-Strauss 1 2002; 51 :127 133 13 Libanoff AJ, McMahon NJ: Eosinophilia and endomyocardial fibrosis. Am J Cardiol 1976; 37: 438 441 14 Shanks M, Ignaszewski AP, Chan SY, Allard MF : Churg- Strauss syndrome with myocarditis manifesting as acute myocardial infarction with cardiogenic shock : Case report and review of the literature. Can J Cardiol 2003 ; 19 : 1184 1188 J Cardiol 2004 Oct; 44 4 : 153 159