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1 대한내과학회지 : 제 84 권제 1 호 후천성다발성관상동맥 - 심방실누공 1 예 고려대학교의과대학안산병원순환기내과 안젬마 이재민 황영재 서영호 김용현 안정천 송우혁 Case of cquired Coronary-Cameral Fistulae Jem Ma hn, Jae Min Lee, Young Jae Hwang, Young Ho Seo, Yong Hyun Kim, Jeong Cheon hn, and Woo Hyuk Song Division of Cardiology, Department of Internal Medicine, Korea University san Hospital, nsan, Korea We report a rare case of acquired multiple coronary-cameral fistulae. 46-year-old man presented to the cardiology department clinic complaining of recently aggravated exertional chest pain. He had been treated 10 years ago for an acute ST segment elevation myocardial infarction (STEMI) with percutaneous coronary intervention (PCI). During revascularization, diffuse multiple fistulae from the left anterior descending (LD) artery to the left ventricle (LV) had been observed. The current chest pain was evaluated by elective coronary angiography but no significant stenosis was observed. However, newly developed diffuse fistulae from the distal right coronary artery (RC) to LV were found during angiography, as well as LD-LV coronary fistulae. Multiple coronary- cameral fistulae were thought to be causing chest pain. beta-blocker was prescribed and, after 3 months of follow-up, exertional chest pain had subsided without further complication. (Korean J Med 2013;84:91-95) Keywords: Coronary vessels; Fistula; Ischemia 서론관상동맥누공 (coronary artery fistula) 은크게두가지형태로구분된다. 하나는관상동맥이심방혹은심실과직접적으로교통하는관상동맥- 심방실누공 (coronary-cameral fistula), 그리고또다른하나는관상동맥이다른혈관과직접적으로교통하는관상동정맥누공 (coronary arteriovenous fistula) 이다. 이들대부분은발생학적이상에기인하는데, 대다수의 관상동맥누공은하나의관상동맥에서기시하며 ( 우관상동맥 55%, 좌관상동맥 35%), 누공의끝 (termination) 은대개우심방이나우심실로알려져있다 [1,2]. 본증례의환자는양관상동맥에서동시에기시하고좌심실로교통하는관상동맥누공을가졌고, 이들중하나는후천적요인에의해발생된관상동맥누공이었다. 이는임상에서흔히볼수없는예로보고하는바이다. Received: Revised: ccepted: Correspondence to Jem Ma hn, M.D. Department of Internal Medicine, Korea University nam Hospital, 73 Inchon-ro, Seongbuk-gu, Seoul , Korea Tel: , Fax: , jem80@hanmail.net Copyright c 2013 The Korean ssociation of Internal Medicine This is an Open ccess article distributed under the terms of the Creative Commons ttribution Non-Commercial License ( which permits unrestricted noncommercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

- The Korean Journal of Medicine: Vol. 84, No. 1, 2013 - 증례 46세남자가 2개월전부터가벼운활동시발생하여 10여분간지속되는흉골하통증을주소로내원하였다. 내원시혈압은 110/70 mmhg 였고심박수는 76회였으며호흡곤란은호소하지않았다. 심음은규칙적이었으며심잡음은들리지않았다.

0 20 mm, Maestro, JOMED GmbH, Rangendingen, Germany) 을시행받았다. 당시좌전하행지의원위부사선분지 (LD distal diagonal branch) 와좌심실이연결된미세관상동맥누공관찰되었으나이에대해서는별다른시술없이 aspirin 100 mg, clopidogrel 75 mg, carvedilol 12.

2 - The Korean Journal of Medicine: Vol. 84, No. 1, 증례 46세남자가 2개월전부터가벼운활동시발생하여 10여분간지속되는흉골하통증을주소로내원하였다. 내원시혈압은 110/70 mmhg 였고심박수는 76회였으며호흡곤란은호소하지않았다. 심음은규칙적이었으며심잡음은들리지않았다. 과거력상고혈압이있었고 20갑년의흡연자였으며 10년전급성심근경색으로경피적관상동맥중재시술을받았었다. 과거의의무기록을참조한결과, 10년전 ST 분절상승급성심근경색이발생하여혈전용해제 (urokinase) 를투여받았으나, 흉통과 ST 분절상승이지속되어관상동맥조영술을시행, 중위부좌전하행지관상동맥의완전폐쇄 (TIMI 0) 가관찰되어경피적관상동맥중재술 ( mm, Maestro, JOMED GmbH, Rangendingen, Germany) 을시행받았다. 당시좌전하행지의원위부사선분지 (LD distal diagonal branch) 와좌심실이연결된미세관상동맥누공관찰되었으나이에대해서는별다른시술없이 aspirin 100 mg, clopidogrel 75 mg, carvedilol 12.5 mg, 하루 1회, captopril 25 mg 하루 2 회, nicorandil 5 mg 하루 3회유지하였으며, 5년전부터는개인의원에서경과관찰하며 aspirin, clopidorel, atorvastatin 만복용하였다. 이번내원시, 심전도에서 V2에서 V4전극에걸쳐서 Q파가관찰되었으나 5년전의심전도와비교, 큰차이없었으며 (Fig. 1), 검사실소견상저밀도지질단백질 / 중성지방 (LDL/ triglyceride) 185/382 mg/dl로상승된바외에이상소견관찰되지않았다. 단순흉부사진에서정상심장크기였고폐부종은 관찰되지않았으며, troponin T ng/ml으로정상이었다. 경흉부심초음파상좌심실구혈률 43% 로경도의좌심실수축기능장애를보였으며, 좌전하행지동맥영역 (whole apex, mid septum, mid anterior wall) 으로허혈손상으로인한국소벽운동장애가관찰되었으나과거의심초음파결과와비교하였을때의미있는차이는관찰되지않았다. 환자의운동능력및심근허혈의증거확보를위하여답차운동부하심전도 (treadmill test) 시행하였으며 7.0 MET 까지운동하였으나, 숨찬증상과피로감호소하여최대심박수에도달못한채검사중단하였다. 운동부하검사중흉통이나심전도상 ST 변화는보이지않았다 (Fig. 1). 새로발생한흉통의양상은다소비전형적이었으나, ST 분절상승급성심근경색의병력으로관상동맥폐쇄질환의악화를배제할수없어, 관상동맥조영술을시행하였다. 관상동맥조영술상에서중위부좌전하행지동맥 (LD) 의경도의협착소견이관찰되었으나, 그외다른관상동맥내허혈증상을유발할의미있는협착소견은관찰되지않았고 (Fig. 2), 과거관찰되던좌전하행지의원위부사선분지 (LD diagonal branch) 와좌심실간의관상동맥누공은동일한양상으로관찰되었다 (Fig. 2). 그러나과거에관찰할수없었던원위부우관상동맥의후하행지 (posterior descending branch of distal RC) 와좌심실간의관상동맥누공이새롭게관찰되었다 (Fig. 2C and 2D). 좌심실조영술 (Left ventriculargraphy) 상에서심첨부와중전벽의국소벽운동장애소견을관찰하였고, 관상동맥누공이좌심실로연결됨을확인할수있었다 (Fig. 3). Figure 1. Treadmill electrocardiogram (EKG). () aseline EKG. () t peak exercise, there was no significant ST change and the target heart rate (THR = 148/min) was not achieved

- Jem Ma hn, et al. cquired coronary-cameral fistulae - C D Figure 2. Coronary angiography.

cranial view. Moderate concentric stenosis was observed at the mid-segment of the left anterior descending artery (arrows).

(D) Diffuse fistulae from right coronary artery to left ventricular cavity (dotted line); left anterior, oblique view.

3 - Jem Ma hn, et al. cquired coronary-cameral fistulae - C D Figure 2. Coronary angiography. () Diffuse fistulae from the left anterior descending artery to left ventricular cavity (dotted line); right anterior oblique, caudal view. () Diffuse fistulae from the left anterior descending artery to left ventricular cavity (dotted line); antero-posterior, cranial view. Moderate concentric stenosis was observed at the mid-segment of the left anterior descending artery (arrows). (C) Diffuse fistulae from right coronary artery to left ventricular cavity (dotted line); right anterior, oblique view. (D) Diffuse fistulae from right coronary artery to left ventricular cavity (dotted line); left anterior, oblique view. 흉통이심하지않았고운동에의하여악화되지않았으며, 다수의크기가작은미만성관상동맥누공이어서보존적치료를하기로하고환자는퇴원하였다. 기존처방에베타차단제중하나인 carvedilol 을추가투여하며외래에서경과관찰하였으며, 3달후추적진료에서환자는흉통을더이상호소하지않았다. 고찰본증례는관상동맥이심방혹은심실과직접적으로교통하는관상동맥-심방실누공 (coronary-cameral fistula) 으로이는임상진료에서매우드물게발견된다. 진단목적으로무작위로시행했던관상동맥조영술의대략 % 에서관

- 대한내과학회지 : 제 84 권제 1 호통권제 629 호 2013 - Figure 3. Left ventriculography. () Left ventriculography, right anterior oblique view.

또한관상동맥누공의시작 (origin) 은우관상동맥 55%, 좌관상동맥 35%, 양쪽모두에서기시되는경우는 5% 이며, 끝 (termination) 은우측심방또는심실이대략 90% 로매우우세한것으로보고되고있다 [1,2].

4 - 대한내과학회지 : 제 84 권제 1 호통권제 629 호 Figure 3. Left ventriculography. () Left ventriculography, right anterior oblique view. The left ventricle is marked by arrowheads and the right coronary artery is superimposed. () Left ventriculargrophy, left anterior oblique view. 상동맥누공이보고된바있다 [3,4]. 또한관상동맥누공의시작 (origin) 은우관상동맥 55%, 좌관상동맥 35%, 양쪽모두에서기시되는경우는 5% 이며, 끝 (termination) 은우측심방또는심실이대략 90% 로매우우세한것으로보고되고있다 [1,2]. 1996년에서 2003년까지 30,829명의심장내과환자들을대상으로시행한독일의연구에서도오직 20명만이다수의관상동맥-좌심실미세누공을지니는것으로나타났다 [5]. 다발성관상동맥- 좌심실누공은드물며, 본증례처럼양측관상동맥에서기시하여좌측심실로연결된관상동맥누공은임상진료에서접하기어려운경우이다. 관상동맥누공은대부분선천적발생학적이상에의해발생하나, 후천적으로도외상이나침습적심장관련시술 ( 예 : 심근조직검사 ), 심근경색후발생할수있다 [6-8]. 본증례에서우관상동맥누공은 10년전심근경색발생당시의관상동맥조영술에서관찰되지않았던점을고려할때, 심근경색발병후후천적으로발생한경우지만, 누공부위가경색부위 ( 전벽심근경색 ) 와일치하지않았기에심근경색과의직접적인과관계는불분명하다. 본증례의관상동맥누공들은좌심실로유입되고있기때문에 thebesian vein system과관련된관상동맥누공으로는생각되지않았다. 다만과거심초음파상에서관찰할수없는심내막심근경색이새로이발생하여, 심벽의국소적미세괴사로동맥누공이발생했을것을추정할수는있을것이다 [9]. 관상동맥누공을가진대다수의환자들은증상이없으나, 소수의환자군에서협심증, 심부전, 심근경색, 심내막염, 동맥루파열, 동맥류형성, 동맥루내혈전형성, 부정맥등이발생한다. 무증상이우세한이유는대다수의관상동맥누공의크기가작고, 개수가한개이기때문이다. 다수의미만성관상동맥누공을지녔던본증례의환자는비전형적이긴하지만협심증양상의흉통을호소하였다. 이러한증상은심장혈류전환증후군 (coronary steal syndrome, 저항성이적은누관으로혈류의단락이발생하여동맥루이하의혈관이지배하는심근부위에허혈현상이발생하는것 ), 또는용적과부하 (Lt-to-Lt shunt) 에의해야기된다고알려져있다 [9]. 관상동맥누공진단은심초음파와관상동맥조영술로가능하다. 심외막에서심실내로지속적으로유입되는혈류가관찰될때심초음파로도진단이가능하나, 누공의끝이미만성의작은혈관일때는진단이어렵다. 관상동맥조영술이관상동맥누공의해부학적구조및크기를확인하는데가장좋은진단법으로알려져있다. 최근에는다배열검출기전산화단층촬영장치 (Multi-Detector Computed Tomography), 심장자기공명촬영 (Cardiac Magnetic Resonance Imaging) 도이용되고있다. 본증례는경흉부심초음파를시행했으나관상동맥누공은관찰되지않았고, 관상동맥조영술에서심실로조영제유입이확인되어진단할수있었다 [10,11]. 관상동맥누공의치료는증상유무, 누공의개수및크기

5 - 안젬마외 6 인. 후천성다발성관상동맥 - 심방실누공 - 에따라달라지며, 치료방법으로는약물치료, 수술적치료, 경피적도관시술법이있다. 증상이동반되는경우에치료를시작하며, 누공이작은크기로다수존재하는경우에는약물치료를먼저시행하고, 그에반해크기가큰누공이한개존재하는경우에는수술, 시술방법을통해치료한다. 약물치료로는베타차단제, 칼슘채널차단제가증상을호전시키는것으로보고되고있고 [12], 수술적치료법으로는해당혈관결찰이있으며, 경피적도관시술법으로코일, 풍선, 플러그등을이용한색전술이있다. 수술적혈관결찰법이사망률, 합병증발생률및장기증상완화율에서모두탁월한결과를보이지만적은비용과빠른회복률때문에최근에는경피적도관시술법이보다널리시도되고있다 [9,13-16]. 본증례의경우, 증상이있어치료가필요했지만크기가작은다수의누공이었기에시술이나수술보다는약물치료로베타차단제를투여했다. 요 본증례는급성심근경색으로시행했던응급재관류시술에서좌전하행지관상동맥- 좌심실누공 (LD-LV fistulae) 을발견했고, 10년후비전형적인흉통이재발하여시행한관상동맥조영술에서이전누공 (LD-LV fistulae) 외에우관상동맥-좌심실누공 (RC-LV fistulae) 을새롭게관찰한예이다. 대다수의관상동맥누공은선천적으로발생하고단일혈관에서기시하며대개우심방또는우심실로유입되나, 본증례의관상동맥누공은후천적으로발생하였고다혈관에서기시하였으며모두좌심실로유입되었다. 이는매우희귀한경우로문헌고찰과함께보고하는바이다. 약 중심단어 : 관상동맥 ; 누공 ; 허혈 REFERENCES 1. Stierle U, Giannitsis E, Sheikhzadeh, Potratz J. Myocardial ischemia in generalized coronary artery-left ventricular microfistulae. Int J Cardiol 1998;63: Padfield GJ. case of coronary cameral fistula. Eur J Echocardiogr 2009;10: Yamanaka O, Hobbs RE. Coronary artery anomalies in 126,595 patients undergoing coronary arteriography. Cathet Cardiovasc Diagn 1990;21: Vavuranakis M, ush C, oudoulas H. Coronary artery fistulas in adults: incidence, angiographic characteristics, natural history. Cathet Cardiovasc Diagn 1995;35: Said S, van der Werf T. Dutch survey of congenital coronary artery fistulas in adults: coronary artery-left ventricular multiple micro-fistulas multi-center observational survey in the Netherlands. Int J Cardiol 2006;110: Ryan C, Gertz EW. Fistula from coronary arteries to left ventricle after myocardial infarction. r Heart J 1977;39: Saraiva F, Matos V, Gonçalves L, ntunes M, Providência L. Coronary fistulas after cardiac transplantation. Rev Port Cardiol 2010;29: Schanzenbächer P, auersachs J. cquired right coronary artery fistula draining to the right ventricle: angiographic documentation of first appearance following reperfusion after acute myocardial infarction, with subsequent spontaneous closure. Heart 2003;89:e Sasi V, Ungi I, Forster T, Nemes. Multiple coronary fistulas originating from all major coronary arteries. cta Cardiol 2010;65: Yang Y, Li Z, Wang X. ssessment of coronary artery fistula by color Doppler echocardiography. Echocardiography 1998;15: Said S. Congenital solitary coronary artery fistulas characterized by their drainage sites. World J Cardiol 2010; 2: Wolf, Rockson SG. Myocardial ischemia and infarction due to multiple coronary-cameral fistulae: two case reports and review of the literature. Cathet Cardiovasc Diagn 1998;43: Liberthson RR, Sagar K, erkoben JP, Weintraub RM, Levine FH. Congenital coronary arteriovenous fistula: report of 13 patients, review of the literature and delineation of management. Circulation 1979;59: rmsby LR, Keane JF, Sherwood MC, Forbess JM, Perry S, Lock JE. Management of coronary artery fistulae: patient selection and results of transcatheter closure. J m Coll Cardiol 2002;39: Urrutia-S CO, Falaschi G, Ott D, Cooley D. Surgical management of 56 patients with congenital coronary artery fistulas. nn Thorac Surg 1983;35: Cheung DL, u WK, Cheung HH, Chiu CS, Lee WT. Coronary artery fistulas: long-term results of surgical correction. nn Thorac Surg 2001;71:

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ºÎÁ¤¸ÆV10N³»Áö case of brugada syndrome presented as chest pain STRCT rugada syndrome was described in 1992 as a new clinical entity characterized by electrocardiographic STsegment elevation in the right precordial leads