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1 대한내과학회지 : 제 87 권제 2 호 이상성오른쪽빗장밑동맥이있는흉부대동맥류의스텐트이식편치료 부산대학교의학전문대학원부산대학교병원 1 순환기내과, 2 흉부외과 최종현 1 장혜윤 1 전무송 1 이혜원 1 박진섭 1 김상필 2 이한철 1 Hybrid Treatment of an Aortic Arch Aneurysm with an Aberrant Right Subclavian Artery Jong Hyun Choi 1, Hye Yoon Jang 1, Moo Song Jeon 1, Hye-Won Lee 1, Jin Sup Park 1, Sang-pil Kim 2, and Han Cheol Lee 1 1 Division of Cardiology, Departments of Internal Medicine and 2 Thoracic and Cardiovascular Surgery, Pusan National University Hospital, Pusan National University School of Medicine, Busan, Korea Aberrant right subclavian artery (ARSA) is a rare congenital anomaly but is the most common of the congenital vascular anomalies of the aortic arch. We report the case of a 68-year-old female undergoing chemotherapy for multiple myeloma who had a large thoracic aortic aneurysm (7.4 cm) with ARSA. She was treated with a hybrid procedure that combined a left common carotid-to-subclavian artery bypass with a "thoracic endovascular aortic repair (TEVAR)" because of the risk associated with a thoracotomy. A stent graft was deployed in the proximal part of the descending aorta to cover the thoracic aortic aneurysm after a left common carotid-to-subclavian bypass was made to restore blood flow in the left arm. There was no endoleak on digital subtraction angiography. Hybrid therapy can be performed successfully for the treatment of thoracic aortic aneurysm with ARSA. (Korean J Med 2014;87: ) Keywords: Aorta; Aortic aneurysm; Stents; Aberrant subclavian artery 서론대동맥궁과흉부대동맥을침범하는흉부대동맥류 (thoracic aortic aneurysm) 는크기가증가할수록파열의위험성이커지 는위험한질환이다. 발생빈도는매년 10만명당약 6-10명이발생하는드문질환이나대동맥류의직경이 5-6 cm 이상증가하는경우는 5년동안파열위험률이 20% 에달하는것으로알려져있다 [1]. 이러한흉부대동맥류환자에서참을 Received: Revised: Accepted: Correspondence to Han Cheol Lee, M.D. Division of Cardiology, Department of Internal Medicine, Pusan National University Hospital, Pusan National University School of Medicine, 179 Gudeok-ro, Seo-gu, Busan , Korea Tel: , Fax: , glaraone@hanmail.net * This study was supported by a grant of the Korea Healthcare Technology R&D Project, Ministry of Health & Welfare (A084177). Copyright c 2014 The Korean Association of Internal Medicine This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License ( which permits unrestricted noncommercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

2 - The Korean Journal of Medicine: Vol. 87, No. 2, 수없는통증이반복되거나크기가지속적으로증가하는경우와발견당시대동맥류의직경이큰경우는파열의위험성이커지므로수술적치료또는스텐트이식편 (stent graft) 삽입술이필요하다 [2]. 개흉술 (thoracotomy) 로대동맥궁과흉부대동맥의대동맥류를제거하고인조혈관으로대치후경동맥 (carotid artery), 완두동맥 (brachiocephalic artery) 을인조혈관에연결하는것이고식적인수술법이고이러한전대동맥궁치환수술 (total aortic arch replacement) 은심폐체외순환기 (cardiopulmonary bypass) 의보조와저체온순환정지법 (hypothermic circulatory arrest) 을사용해야하므로초기환자의사망률과이환율이높고수술시간도오래걸리는단점이있다 [3]. 이러한이유로최근들어고령, 당뇨, 중풍, 고혈압등의많은위험인자를가지는수술고위험군환자에서는흉부대동맥혈관내스텐트이식편삽입술 (thoracic endovascular aortic repair, TEVAR) 이수술을대체하여널리시행되고있다. 이에저자들은고령및다발성골수종의위험인자를가진수술고위험군환자에서이상성오른쪽빗장밑동맥 (aberrant right subclavian artery) 이동반된대동맥궁침범대동맥류에대해왼쪽온목동맥 (common carotid artery)-왼쪽빗장밑동맥우회로술과흉부대동맥혈관내스텐트이식편삽입술을같이시행하여 Hybrid 방법으로치료한 1예를문헌고찰과함께보고하는바이다. 증례 68세여자환자가호흡곤란을주소로내원하였다. 환자는고혈압이나당뇨는없었으나다발성골수종 (multiple myeloma) 으로 10년전에자가골수이식을시행한후본원에서항암치료중이었다. 3년전에호흡곤란으로시행한흉부전산화단층촬영에서폐렴소견과함께대동맥궁에서하행대동맥까지대동맥벽내혈종 (intramural hematoma) 또는죽종 (atheroma) 이의심되는병변이관찰되었고특별한증상이없어정기적으로외래에서경과관찰중이었다. 이후외래에서시행한흉부 X-선촬영에서대동맥음영의너비가커지는것이관찰되어흉부전산화단층촬영을다시시행하였고이전에왼쪽빗장밑동맥과같은위치직하부에서기시하는대동맥벽내혈종이의심되었던병변이 7.4 cm로확장된흉부대동맥류가관찰되었다. 특히정상적인위치보다아래쪽에위치하는이상성오른쪽빗장밑동맥이왼쪽빗장밑동맥 에서약 3 cm 하방하행대동맥의흉부대동맥류안에서기시하고식도뒤쪽을지나서종격동 (mediastinum) 을따라오른쪽팔부위로주행하는것이관찰되었다 (Fig. 1). 심장초음파및관상동맥조영술에서는특별한이상소견은보이지않았으며환자의입원당시말초혈액검사소견은백혈구 4,710/mm 3, 혈색소 10.7g/dL, 혈소판 163,000/mm 3, 혈청생화학검사에서 AST 12 IU/L, ALT 15 IU/L, 총빌리루빈 0.46 mg/dl, creatinine 0.64 mg/dl로경미한빈혈소견이외에특이소견은관찰되지않았다. 이환자는고령및다발성골수종의위험인자가있었고이상성오른쪽빗장밑동맥이있어서전대동맥궁치환수술과함께상행대동맥에각각의동맥을연결하기에는수술전후의위험성이컸다. 또한대동맥류가왼쪽빗장밑동맥직하방에서시작하여서스텐트이식편의근위부안착지점 (proximal landing zone of stent graft) 을확보하기가어려웠다. 왼쪽빗장밑동맥의우회로술을실시할경우에는 25 mm 정도의스텐트이식편의안착지점을확보할수있었다. 자기공명영상에서왼쪽척추동맥 (left vertebral artery) 이우세 (dominant) 한소견을보였기에왼쪽온목동맥- 왼쪽빗장밑동맥우회로술을먼저시행한후왼쪽온목동맥직전까지흉부대동맥혈관내스텐트이식편삽입술을시행하기로계획하였다. 우측상지에허혈이나쇄골하동맥도류증후군등이있으면시술후에이차적으로오른쪽온목동맥 오른쪽빗장밑동맥우회로술을시행하기로하였다. 환자를전신마취한후에왼쪽빗장밑동맥을 side biting clamp로잡고 8 mm tube graft를연결한뒤내목정맥 (internal jugular vein) 뒤쪽으로 graft를진행시켜온목동맥의원위부와끝옆연결술 (end to side anastomosis) 을하여왼쪽온목동맥에서왼쪽빗장밑동맥을연결하는수술을시행하였다. 수술은 hybrid room에서시행되었고대동맥에서오는혈류와왼쪽온목동맥에서오는혈류의상호교란을막기위하여우회로술직후바로흉부대동맥혈관내스텐트이식편삽입술을실시하였다. 오른쪽대퇴동맥 (left femoral artery) 에 2 개의 Perclose (Abbott Vascular Devices, Redwood City, CA, USA) 를미리시행하였고 Marked pigtail catheter를왼쪽대퇴동맥을통하여상행대동맥에위치시킨후대동맥조영술을시행하면서스텐트이식편의위치를조절하였다. 5 French multipurpose catheter와 inch Terumo guidewire (Terumo Corp, Tyoko, Japan) 를상행대동맥으로진행시킨후 inch

- Jong Hyun Choi, et al. Aortic aneurysm with aberrant right subclavian artery - A B C D Figure 1. Thoracic aortic aneurysm with an aberrant right subclavian artery.

(C) Three-dimensional reconstructed CT images of the aberrant right subclavian artery.

Lunderquist extrastiff guidewire (Cook Inc, Bloomington, IN, USA) 로바꾸었다.

3 - Jong Hyun Choi, et al. Aortic aneurysm with aberrant right subclavian artery - A B C D Figure 1. Thoracic aortic aneurysm with an aberrant right subclavian artery. (A) Computed tomography image shows a 7.3-cm aortic arch aneurysm (arrowhead). (B) Aberrant right subclavian artery behind the esophagus and trachea (arrow). (C) Three-dimensional reconstructed CT images of the aberrant right subclavian artery. (D) Magnetic resonance angiogram shows the aberrant right subclavian artery (arrow) and dominant left vertebral artery (arrowhead). Lunderquist extrastiff guidewire (Cook Inc, Bloomington, IN, USA) 로바꾸었다. 대퇴동맥을단계적으로확장후 18 French sheath로확장하였고 32 mm 190 mm Seal stent graft (S&G biotech, Seongnam, Korea) 를왼쪽온목동맥직하방까지넣었다. 이환자의경우왼쪽빗장밑동맥은동맥류와바로붙어있어서안정된안착지점 (landing zone) 의확보를위하여그혈류를막았고대신에왼쪽온목동맥에서의우회로를통하여왼쪽팔로혈류가공급되었다. 시술후의척추동맥을통 하여양쪽빗장밑동맥으로혈류가역류하여서생기는 type II endoleak 을막기위하여먼저왼쪽위팔동맥을통하여왼쪽빗장밑동맥에코일색전술 (coil embolization) 을시행하였다. 왼쪽빗장밑동맥은온목동맥에서우회로를통하여혈류가공급되었다. 오른쪽빗장밑동맥도오른쪽척추동맥을통하여혈류가대동맥류내로역류하는 type II endoleak를막기위하여코일색전술을시행하였고오른쪽팔로의혈류는오른쪽척추동맥을통하여우회하여공급되었다. 시술을마친

대한내과학회지: 제 87 권 제 2 호 통권 제 648 호 2014 A B C D Figure 2.

(B) Thoracic aorta angiography shows both common carotid arteries (arrowhead) and left subclavian

Shunt blood flow, from the left carotid artery to the left subclavian artery, was intact.

right vertebral artery (arrowhead) and the left common carotid-to-subclavian artery bypass after

(A) Chest X-ray, performed immediately after thoracic endovascular aneurysm repair (TEVAR).

4 대한내과학회지: 제 87 권 제 2 호 통권 제 648 호 2014 A B C D Figure 2. (A) Aortogram of the thoracic aorta before coil embolization. (B) Thoracic aorta angiography shows both common carotid arteries (arrowhead) and left subclavian artery (arrow). Shunt blood flow, from the left carotid artery to the left subclavian artery, was intact. (C) Endovascular coil embolization of the aberrant right subclavian artery and the left subclavian artery. (D) Digital subtraction angiography shows intact blood flow in both subclavian arteries, from the right vertebral artery (arrowhead) and the left common carotid-to-subclavian artery bypass after coil embolization. A B Figure 3. (A) Chest X-ray, performed immediately after thoracic endovascular aneurysm repair (TEVAR). (B) One-year follow-up chest X-ray showing no significant interval change in the aortic aneurysm. 후 시행한 대동맥 조영술에서 스텐트이식편 밖으로의 endoleak 치료 유지하며 경과관찰 중이며 1년 뒤 시행한 chest X-ray에 은 보이지 않았고 왼쪽 온목동맥에서 우회로를 통한 왼쪽 서 변화된 소견은 관찰되지 않았다(Fig. 3). 빗장밑동맥의 혈류는 원활하였으며 오른쪽 빗장밑동맥의 고 혈류 역시 오른쪽 척추동맥으로부터 원활하게 공급되었다 찰 (Fig. 2). 환자는 시술 후 쇄골하 동맥 도류 증후군이나 오른 1994년 Dake 등[4]이 처음으로 흉부 대동맥류에 대해 흉 쪽 상지의 통증은 관찰되지 않았다. 환자는 퇴원 후 1년 동안 특이한 임상증상 없었고 항암 부 대동맥 혈관 내 스텐트이식편 삽입술을 시행한 이후, 최

5 - 최종현외 6 인. 이상성빗장밑동맥가진대동맥류 - 근에는임상에서의적용이점점증가하는추세에있다. 이러한흉부대동맥혈관내스텐트이식편삽입술은고식적수술에비해덜침습적이며대동맥병변의범위가넓고수술의위험도가높은고위험군에서도비교적안전하게적용이가능하다는장점이있다. Jonker 등 [5] 이시행한다변량분석에따르면파열된하행흉부대동맥류의치료에있어서흉부대동맥혈관내스텐트이식편삽입술또는개흉술을통한고식적인수술을시행한총 224명의환자를분석하여보았을때 30일째사망률에있어서흉부대동맥혈관내스텐트이식편삽입술을시행한군에서 19%, 고식적수술을시행한군에서 33% 로통계적으로유의하게흉부대동맥혈관내스텐트이식편삽입술을시행한군의사망률이감소한다는결과가있었다 (p = 0.016). Makaroun 등 [6] 은 Gore TAG device (Gore comp, Flagstaff, AZ, USA) 를이용하여흉부대동맥혈관내스텐트이식편삽입술을시행한환자군과고식적개흉술을시행한환자군을전향적으로분석한 2상연구를시행하였으며그결과 5년째총사망률 (all-cause mortality) 은각각 68%, 67% 로양군간의통계적으로유의한차이를관찰할수없었으나 5년째동맥류와연관된사망률의경우에는흉부대동맥혈관내스텐트이식편삽입술을시행한환자군에서 2.8%, 고식적개흉술을시행한환자군에서 11.7% 로통계적으로더양호한결과를얻을수있었다. 스텐트이식편의안착지점이원위대동맥궁에위치할경우에는최소 mm의스텐트안착부위확보가필요하고따라서대동맥궁에서분지하는완두동맥 (brachiocephalic artery), 왼쪽온목동맥 (left common carotid), 왼쪽빗장밑동맥등의분지혈관이스텐트이식편에의하여막히게된다. 이러한경우예전에는흉부대동맥혈관내스텐트이식편삽입술을시행하기어려웠으나최근에는이러한한계점을극복하기위하여다양한우회로술이흉부대동맥혈관내스텐트이식편삽입술과같이시행되고있으며이러한시술및수술을위해서는대동맥궁및하행대동맥의병변범위및위치에따라그방법을결정하여야한다. Mitchell 등 [7] 은스텐트이식편의근위부의안착지점을기준으로완두동맥, 왼쪽온목동맥, 왼쪽빗장밑동맥에위치한경우각각 Zone 0, Zone 1, Zone 2로분류하였으며 Zone 0은상행대동맥과대동맥궁에서기시하는모든혈관에대하여우회로술이필요하고, Zone 1의경우오른쪽온목동맥에서왼쪽온목동맥으로 의우회로술이필요하며 Zone 2의경우는왼쪽온목동맥- 왼쪽빗장밑동맥우회로술을시행하여야수술후신경학적합병증을예방하고스텐트이식편의안착지점을확보할수있다고알려져있다. 이상성오른쪽빗장밑동맥은대동맥혈관기형중에는가장흔하며인구의 0.5% 에서존재하는것으로알려져있다. 대부분의사람들에서는무증상이나약 60% 의환자에서 Kommerell s diverticulum이존재하고이것이커지게되면동맥류가형성되어식도와기관지를누르면서연하곤란또는상부기도압박증세가나타나는것으로알려져있다 [8]. 또한파열의위험성때문에 3 cm 이상의이상성오른쪽빗장밑동맥에의한동맥류를가지는경우는수술적치료를고려해야하나최근에는이러한환자의치료에있어개흉술에비하여덜침습적인우회로술과흉부대동맥혈관내스텐트이식편삽입술을단계적으로함께시행하는 hybrid 방법에대한여러증례가보고되고있다. Lacroix 등 [9] 은이상성오른쪽빗장밑동맥동맥류환자의치료에있어서개흉술의위험도가높은환자에서오른쪽온목동맥에서이상성오른쪽빗장밑동맥으로의우회로술과흉부대동맥혈관내스텐트이식편삽입술을단계적으로안전하고효과적으로시행한증례를보고하였으며국내에서는 Lee 등 [10] 이스텐트이식편의근위부안착지점확보를위하여 1단계로대동맥에서양쪽온목동맥으로의우회로술을시행하고 2단계로왼쪽온목동맥에서왼쪽빗장밑동맥으로의우회로술을, 3단계로오른쪽온목동맥에서이상성오른쪽빗장밑동맥으로의우회로술을시행한후흉부대동맥혈관내스텐트이식편삽입술을시행하여좋은결과를얻어보고한증례가있다. 이증례의경우는여러단계의우회로술을통하여양쪽빗장밑동맥으로의안정적인혈류를확보할수있다는장점이있을수있으나여러차례의수술로인한부작용증가우려와대동맥에서양쪽온목동맥으로의우회로술의경우심폐체외순환기의보조와저체온순환정지법을사용하는개흉술이필요하여시술및수술에따른위험도가증가할수있는단점이있을수있다. 그에반하여본증례는왼쪽온목동맥에서왼쪽빗장밑동맥으로의우회로술의한차례시행만으로흉부대동맥혈관내스텐트이식편삽입술시행후에양쪽빗장밑동맥이막히더라도왼쪽뿐만아니라오른쪽혈류까지안정적으로확보할수있었기때문에의미가있었다. 본증례는 Kommerell s diverticulum에의한대동맥류는아니었으

6 - The Korean Journal of Medicine: Vol. 87, No. 2, 나이상성오른쪽빗장밑동맥의위치이상으로인하여실제로는이식편근위부안착지점분류상 Zone 2에해당함에도불구하고양쪽빗장밑동맥의혈류장애가예상되는환자였다. 하지만양쪽우회로술을모두실시할경우환자가목을움직일때불편함을야기할뿐만아니라추가수술에따른부작용의가능성이있었다. 또한시술전자기공명혈관조영술에서왼쪽이우세 (dominant) 한척추동맥임을확인하였기때문에시술후오른쪽팔의통증이나어지럼등의심한허혈증상이있을때추가로오른쪽우회로술을시행하기로하고왼쪽온목동맥에서왼쪽빗장밑동맥으로의우회로술만을시행한후흉부대동맥혈관내스텐트이식편삽입술을시행하였다. 이후이상성오른쪽빗장밑동맥의위치이상으로인하여양쪽빗장밑동맥의기시부가모두스텐트이식편에의하여덥히게되어만약빗장밑동맥의기시부를막지않으면우회로를통한혈류가대동맥류안으로다시들어오는 Type II endoleak 가능성이있어추가로양쪽빗장밑동맥모두코일색전술을실시하였다. 왼쪽빗장밑동맥의폐쇄에대하여는아직여러가지이론들이있으나일반적으로왼쪽빗장밑동맥에서분지하는척추동맥이뇌후부순환을담당하기때문에척추기저부전 (vertebrobasilar insufficiency) 이나빗장밑동맥도류증후군 (subclavian steal syndrome) 등의발생가능성이높아진다고알려져있다. 특히왼쪽척추동맥이크고우세한경우에는신경학적인합병증이증가하는것으로알려져있어이러한부작용이예상되는경우에는왼쪽빗장밑동맥재개통술 (revascularization) 을고려하여야한다고알려져있다. 본증례에서는시술후시행한대동맥조영술에서왼쪽빗장밑동맥은왼쪽온목동맥에서의우회로를통하여혈류가원활하였고오른쪽빗장밑동맥은우회로술을시행하지않았음에도오른쪽온목동맥에서윌리스서클 (circle of willis) 을통하여오른쪽척추동맥으로우회하는혈류를통하여혈류를공급받을수있었다. 빗장밑동맥을보존하여야하는문제에대하여서는아직이론이많은실정으로추후많은자료들의검토와연구가이루어져야할것으로생각되며본증례에서와같이이상성오른쪽빗장밑동맥으로인하여시술후양쪽빗장밑동맥의혈류장애가예상되는경우에왼쪽온목동맥- 왼쪽빗장밑동맥우회로술만을시행한이후에흉부대동맥혈관내스텐트이식편삽입술을시행하였을때도양쪽팔의혈류가모두 원활한결과를얻을수있었다. 요 흉부대동맥류는크기가클수록파열로인한사망의가능성이높아지는위험한질환이다. 이러한경우수술적치료를통한대동맥류의제거가필요하나위험인자가많은고위험군환자에있어서는수술에따른부작용이매우높은편이다. 따라서최근에이러한환자에서는흉부대동맥혈관내스텐트이식편삽입술이수술을대체하여널리시행되고있다. 이에저자들은고령및다발성골수종으로수술고위험군인대동맥궁대동맥류환자에서왼쪽온목동맥에서왼쪽빗장밑동맥우회로술과흉부대동맥혈관내스텐트이식편삽입술을함께시행하였으며이상성오른쪽빗장밑동맥이있음에도불구하고특별한합병증없이양쪽팔의혈류가원활한좋은결과를경험하였기에문헌고찰과함께 1예를보고하는바이다. 약 중심단어 : 대동맥 ; 대동맥류 ; 스텐트 ; 빗장밑동맥 REFERENCES 1. Bickerstaff LK, Pairolero PC, Hollier LH, et al. Thoracic aortic aneurysms: a population-based study. Surgery 1982; 92: Troxler M, Mavor AI, Homer-Vanniasinkam S. Penetrating atherosclerotic ulcers of the aorta. Br J Surg 2001;88: Crawford ES, Svensson LG, Coselli JS, Safi HJ, Hess KR. Surgical treatment of aneurysm and/or dissection of the ascending aorta, transverse aortic arch, and ascending aorta and transverse aortic arch: factors influencing survival in 717 patients. J Thorac Cardiovasc Surg 1989;98(5 Pt 1): Dake MD, Miller DC, Semba CP, Mitchell RS, Walker PJ, Liddell RP. Transluminal placement of endovascular stentgrafts for the treatment of descending thoracic aortic aneurysms. N Engl J Med 1994;331: Jonker FH, Trimarchi S, Verhagen HJ, Moll FL, Sumpio BE, Muhs BE. Meta-analysis of open versus endovascular repair for ruptured descending thoracic aortic aneurysm. J Vasc Surg 2010;51: Makaroun MS, Dillavou ED, Wheatley GH, Cambria RP; Gore TAG Investigators. Five-year results of endovascular

7 - Jong Hyun Choi, et al. Aortic aneurysm with aberrant right subclavian artery - treatment with the Gore TAG device compared with open repair of thoracic aortic aneurysms. J Vasc Surg 2008;47: Mitchell RS, Ishimaru S, Ehrlich MP, et al. First International Summit on Thoracic Aortic Endografting: roundtable on thoracic aortic dissection as an indication for endografting. J Endovasc Ther 2002;9(Suppl 2):II Stone WM, Ricotta JJ 2nd, Fowl RJ, Garg N, Bower TC, Money SR. Contemporary management of aberrant right subclavian arteries. Ann Vasc Surg 2011;25: Lacroix V, Astarci P, Philippe D, et al. Endovascular treatment of an aneurysmal aberrant right subclavian artery. J Endovasc Ther 2003;10: Lee JH, Ko YG, Yoon YN, Choi D, Lee DY. Hybrid treatment for thoracic aortic aneurysm combined with aberrant right subclavian artery. Korean Circ J 2013;43:

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Jksvs019(8-15).hwp Grade I Grade II Grade III 12 대한혈관외과학회지 : 제 20 권 제 1 호 2004 Control Group A Group B Fig. 4. Microscopic findings of vein wall in control, group A and group B on the day of 7 after venous occlusion. The