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370 Minho Han. Subclavian Steal Syndrome Treated by Stent Insertion CASE REPORT Korean J Clin Lab Sci. 2018;50(3):370-374 https://doi.org/10.15324/kjcls.2018.50.3.370 pissn 1738-3544 eissn 2288-1662 Duplex Sonography in Subclavian Steal Syndrome Treated by Stent Insertion Minho Han Department of Neurology, Yonsei University Severance Hospital, Seoul, Korea 이중초음파검사상빗장밑혈류빼앗김증후군이관찰된환자에서의스텐트삽입후추적 한민호 연세의료원세브란스병원신경과 Subclavian steal syndrome is a type of obstructive artery disease caused by severe stenosis and occlusion of a left proximal subclavian artery or brachiocephalic trunk. The resulting vertebrobasilar insufficiency symptoms are due to retrograde blood flow from the contralateral vertebral and basilar arteries into the low-pressure ipsilateral upper extremity vessels. For that reason, patients usually experience dizziness or arm ischemic symptoms. Neurointervention is an effective treatment for ischemic stroke, including the subclavian artery stenosis. This paper reports a patient with subclavian steal syndrome who had a vertebral artery Doppler waveform change in duplex sonography and a 12 mmhg difference in interarm systolic blood pressure. A stenotic lesion of the subclavian artery was treated effectively by inserting a precise stent. The follow up examination showed that the vertebral artery Doppler waveform change disappeared and recovered and that the 12 mmhg difference in interarm systolic blood pressure decreased to 5 mmhg after treatment. Key words: Duplex sonography, Interarm systolic blood pressure difference, Subclavian steal syndrome Corresponding author: Minho Han Department of Neurology, Yonsei University Severance Hospital, 50-1 Yonsei-ro, Seodaemun-gu, 03722 Seoul, Korea Tel: 82-2-2228-5283 Fax: 82-2-2228-5283 E-mail: umsthol18@gmail.com This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/4.0) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited. Copyright 2018 The Korean Society for Clinical Laboratory Science. All rights reserved. Received: July 2, 2018 Revised 1 st : July 11, 2018 Revised 2 nd : July 11, 2018 Revised 3 rd : July 11, 2018 Revised 4 th : July 11, 2018 Accepted: July 13, 2018 서론빗장밑혈류빼앗김증후군 (subclavian steal syndrome) 은오른쪽팔머리동맥 (brachiocephalic trunk) 혹은왼쪽근위부빗장밑동맥 (subclavian artery) 에심한협착이나폐색이있을때반대쪽척추동맥 (vertebral artery) 에서질환쪽척추동맥으로역행성혈류 (retrograde flow) 가형성되는혈류장애 ( 빗장밑혈류빼앗김현상, subclavian steal phenomenon) 로인해척추- 뇌바닥혈류부전 (vertebro-basilar insufficiency) 증상및질환 쪽팔에허혈성변화가나타나는질환을말한다. 그러나역행성척추동맥의혈류가보인다고해서모든환자가그러한증상을보이는것은아니다 [1-3]. 빗장밑동맥협착을포함한허혈성뇌졸중환자에서신경중재술 (neurointervention) 중하나인혈관성형술및스텐트설치술 (carotid angioplasty and stenting) 이일반적으로치료에이용되고있으며, 신경중재술로인한혈류의회복및방향교정은환자의증상과예후를좋게한다고알려져있다 [4]. 저자는허혈성뇌졸중환자중이중초음파검사 (duplex

Korean J Clin Lab Sci. Vol. 50, No. 3, September 2018 371 sonography) 에서빗장밑혈류빼앗김증후군에서관찰되는특이적인혈류변화를발견하였고, 스텐트 (stent) 를삽입한후 6개월뒤추적검사에서특이적인혈류변화가사라지고, 스텐트삽입이성공적으로장치된것을재확인하였기에질환의소개와함께진단및치료후추적검사소견을보고하고자한다. 증례 71세남자가기상후어지러움과두통을주된호소로신경과에내원하여실시한자기공명영상 (magnetic resonance imaging, MRI) 에서깊은백색질에오래된허혈변화와양쪽목동맥과오른쪽근위부뒤대뇌동맥에협착을보여이후외래에서추적하던중 3개월뒤오후에자고일어나서오른팔에힘이빠지는증상이발생하여정확한진단과적절한치료를위해입원하였다. 환자는폐쇄성심장동맥질환, 바닥핵경색을진단받았으며고혈압이있어항고혈압제를복용중이고, 하루에 1갑씩 50년간흡연하였으나입원 1년전부터는담배를피우지않고있었다. Figure 1. The retrograde blood is found in a left vertebral artery with the carotid duplex. 입원당시위팔-발목맥파전파속도 (brachial-ankle pulse wave velocity, bapwv) 와발목-위팔동맥압지수 (anklebrachial index, ABI) 를동시에측정하는검사 (VP-1000, Colin Co. Ltd, Komaki, Japan) 에서맥박수와체질량지수는정상범위였고, 위팔혈압은오른쪽이 131/63 mmhg, 왼쪽이 119/70 mmhg로측정되었으며, bapwv는연령에비해높아동맥경화 (arteriosclerosis) 가진행되고있음을알수있었고, ABI는오른쪽다리가 1.08, 왼쪽다리가 1.15로정상범위에포함되어말초동맥질환 (peripheral arterial disease) 은관찰되지않았다. bapwv & ABI는전체적인혈관의동맥경화도 (arterial stiffness) 와말초동맥질환을진단하는선별검사이다. 약 10분정도가소요되며, 심전도및심음도기록과함께양쪽팔과다리에서혈압을측정할수있다. 경두개도플러초음파검사 (transcranial Doppler, TC8080, Viasys healthcare, Pennsylvania, USA) 에서는특이적인혈류속도의상승이나역방향성의혈류흐름은관찰되지않았다. 이중초음파검사 (IU22, Philips, Washington, USA) 에서는양쪽목동맥에죽상동맥경화증 (atherosclerosis) 이관찰되었으며, 왼쪽속목동맥 (internal carotid artery) 에서 Area법으로협착정도를측정했을때, 약 80% 의혈관이좁아져있는것이발견되었다. 특히왼쪽척추동맥은 B-mode상에서죽상동맥경화증을관찰할수없었으나 Doppler mode로혈류를관찰하였을때수축기혈류는바닥선 (baseline) 아래로역전되고, 이완기혈류는정방향을유지하는특이적인혈류변화 ( 전후유동파형, to-and-fro waveform) 를발견하였으며 (Figure 1), 반응성충혈검사 (reactive hyperemia test) 에서는역행성혈류의흐름이더욱심해지는것을관찰할수있었다 (Figure 2). 반응성충혈검사란빗장밑혈류빼앗김증후군진단의정확성을높이는매우유용한검사이다. 외부에서다양한방법으로압력을주어특정혈관의혈류를차단하고, 일정시간이지난후차단한혈류를개통시켜해당혈관에일시적으로과잉혈류공급을유도하는것 Figure 2. A fully retrograde flow pattern of left vertebral artery by a reactive hyperemia test.

372 Minho Han. Subclavian Steal Syndrome Treated by Stent Insertion 이다. 빗장밑혈류빼앗김증후군이있을경우, 충혈검사직후척추동맥에서더욱심각한혈류의역행이관찰되고, 심지어파형이모두뒤집어지기도한다. 다음과같은순서로진행되었다 [1]. 1) 병변부위의위팔에혈압계밴드를감고, 수축기혈압보다약 20 mmhg 더높은압력으로혈류를차단시켰다. 2) 약 3분이지난시점에서급히공기를배출시켜혈류를개통함으로써혈류파형에서역행하는혈류양상이일시적으로더욱두드러지는지를관찰하였다. 머리와목부위의자기공명혈관촬영 (magnetic resonance angiography) 과디지털감산혈관조영술 (digital subtraction angiography) 에서는왼쪽속목동맥의협착과더불어왼쪽근위부빗장밑동맥에궤양성죽상판 (ulcerative atheromatous plaque) 을동반한약 80% 의협착이관찰되었다 (Figure 3). 협착된부위를치료하기위해오른쪽넙다리동맥 (femoral artery) 을천자한후유도철심을왼쪽빗장밑동맥까지삽입하여풍선카테터로혈관성형술을시행한후직경 10 mm, 길이 40 mm의스텐트 (precise stent) 를삽입하였다. 스텐트삽입후오른쪽척추동맥으로부터의역행성혈류소견은관찰되지않았으며, 왼쪽빗장밑동맥에서시작하여같은쪽척추동맥으로흐르는혈류가원활해짐을확인할수있었다 (Figure 4). 시술후 6개월이지나본검사실에방문한환자는경두개도플러초음파에서는별다른변화가없었으나, bapwv & ABI에서오른쪽위팔혈압이 142/85 mmhg, 왼쪽위팔혈압이 137/83 mmhg로이전의기록은양팔의수축기혈압의차이가 12 mmhg였다면, 추적검사에서는혈압의차이가 5 mmhg로감소하였다 (Figure 5). 그외의 bapwv 와 ABI 는큰변화가없었다. 이중초음파검사에서빗장밑동맥에설치되어있는스텐트는확인할수없지만, 정방향혈류 (anterograde flow) 를관찰함으 Figure 3. Digital subtraction angiography showing a 80% stenosis in the proximal left subclavian artery. Figure 4. The subclavian arterial blood flows smoothly to the ipsilateral upper extremity vessels and the proximal vertebral artery. Figure 5. The interarm systolic blood pressure difference decreased after treatment.

Korean J Clin Lab Sci. Vol. 50, No. 3, September 2018 373 Figure 6. The vertebral artery Doppler waveform change disappeared and recovered after treatment. 로써, 빗장밑혈류빼앗김현상은사라지고간접적이지만스텐트가잘장치되고있음을추측할수있었다 (Figure 6). 고찰 빗장밑혈류빼앗김현상은주로척추동맥시작마디이전의왼쪽근위부빗장밑동맥에심한협착이나폐색이있을때질환쪽빗장밑동맥혈류의압력이낮아져서반대쪽척추동맥이나뇌바닥동맥의혈류가질환쪽척추동맥을통해같은쪽빗장밑동맥으로역류하는것을말한다. 이러한현상에의해척추-뇌바닥혈류부전에의한어지럼증이나팔의허혈증상이동반되는것을빗장밑혈류빼앗김증후군이라고한다 [1-3]. 척추-뇌바닥혈류부전은근위부빗장밑동맥의협착에의해주로발생하며, 질환이있는팔에허혈성증상이나타나는것은원위부빗장밑동맥의협착이원인이되는경우가많다 [4]. 그러나척추동맥역류가관찰되어도이러한증상의원인으로는다양한원인이있을수있기때문에신중히판단해야하며, 온목동맥 (common carotid artery) 이나반대쪽척추동맥을통한곁순환 (collateral circulation) 이발달하여혈류공급이보충될수있기때문에모든환자가증상을호소하는것은아니다 [3]. 건강한성인에서양쪽위팔혈압의차이는 5 mmhg 이내로거의비슷한소견을보이지만, 빗장밑혈류빼앗김증후군의원인이되는빗장밑동맥또는팔머리동맥의협착이있을경우양쪽위팔혈압은 10 mmhg 이상으로큰혈압차를보인다 [5]. 이와더불어, 이중초음파검사는척추동맥에서역행성혈류빼앗김현상이관찰되었을때진단적특이도가높은필수검사로서, 비침습적이며환자침상에서빠르게시행될수있고, 검사자가직접특정척추동맥을발견한후혈류역학적정보를실시간으로측정할수있기때문에경두개도플러초음파검사에비해빗장밑혈류 빼앗김증후군을발견하는데민간도가높은장점을가지고있다 [1]. 본증례에서도경두개도플러초음파검사는본질적으로혈관의위치및구조를가시화하지못하기때문에검사자는왼쪽척추동맥의역행성혈류를관찰하지못하고주변에위치한다른동맥의혈류를측정함으로써혈류빼앗김현상을발견하지못하였다. 척추동맥에역류가관찰되는빗장밑혈류빼앗김증후군환자는수술과시술에의한혈류방향의교정이증상개선에도움이되는것으로알려져있다 [6]. 혈류방향의교정을위한치료방법은수술적치료와신경중재술이있으며, 수술적치료에는겨드랑동맥간우회술, 목동맥- 빗장밑동맥우회술, 가슴절개를통한동맥내막절제술등이있다 [5]. 이중동맥내막절제술은수술고위험군에서주수술기 (perioperative state) 의위험성을더욱증가시키고, 심각한심장동맥질환이있는경우에는목동맥내막절제술에의한유병률및사망률이높게나타난다. 또한재협착이발생할경우에흉터조직으로인해재수술이어려우며, 일차수술에비해합병증도높다 [7]. 우회술은개존율이 5년을두고보았을때스텐트삽입술보다우수하다는보고가있지만뇌혈관질환, 허혈성심장질환이동반된환자의경우에는수술적치료가어렵다 [4]. 최근에는이러한단점의효과적인대안으로신경중재술중하나인스텐트 (stent) 를이용한혈관성형술이많이이용되고있다 [7, 8]. 혈관성형술및스텐트설치술은수술적치료가어려운경우에최적의치료로추천되고있으며혈관조영술을하는동시에스텐트삽입을시행하여입원기간을줄일수있을뿐만아니라간단한국소마취로이루어지며, 시술직후의유병률이수술에비해낮고높은치료성공률을보이고있다 [2, 5]. 하지만심장동맥의경우에스텐트시술후재협착발생률이 20 50% 인것으로보고되어 [9], 시술후에도비교적간단하고환자에게심적부담을줄일수있는비침습적인검사를이용하여정기적으로감시할필요가있다. 따라서적절한치료후이중초음파검사를통해빗장밑혈류빼앗김현상이사라졌는지, 심혈관및말초혈관질환의지표로알려져있는 10 mmhg 이상의양팔혈압차가 5 mmhg 이내로작아졌는지확인하는것은환자의예후를예측할수있는좋은정보가될수있다 [5, 10]. 본검사실에방문한 71세, 남자환자는양팔수축기혈압이 12 mmhg로유의한차이를보였으며, 이중초음파검사에서는빗장밑혈류빼앗김증후군에서볼수있는특이적인혈류변화가관찰되어, 정밀진단후에스텐트를삽입하였다. 2010년국내에서보고된빗장밑혈류빼앗김증후군에대한증례는특이적인혈류양상을제시하였지만, 본증례보고와다르게치료후빗장밑

374 Minho Han. Subclavian Steal Syndrome Treated by Stent Insertion 혈류빼앗김현상이사라졌는지에대한추적검사소견은제시하지못하였다 [11]. 저자는 6개월후추적검사를시행하였고혈압의차이가 5 mmhg로감소되었음을확인하였으며특이적인혈류변화가사라져스텐트삽입이성공적으로장치됨을재확인하였다. 따라서이중초음파검사는빗장밑혈류빼앗김증후군의진단및치료후추적검사에서혈류역학적정보를파악하여환자의예후를예측하는데유용하게이용될수있다. 요약빗장밑혈류빼앗김증후군은왼쪽근위부빗장밑동맥이나오른쪽팔머리동맥에심한협착혹은폐색으로인해반대쪽척추동맥으로부터관류압이감소된병변쪽빗장밑동맥으로혈류가역으로유입되는현상이나타나게되고, 그결과로척추-뇌바닥혈류부전이나허혈성말초동맥질환증상이동반되는경우를말한다. 빗장밑동맥을포함한뇌혈관협착증에서신경중재술을이용한혈류의회복및방향교정은환자의증상과예후를좋게한다고알려져있다. 저자는오른팔에비해왼팔혈압이 12 mmhg 낮은환자에서이중초음파검사를시행하였고, 왼쪽척추동맥에서빗장밑혈류빼앗김증후군의특징적인혈류파형이관찰되어혈관성형술및스텐트설치술을통해성공적으로치료된증례를보고하고자한다. 6개월후추적검사에서왼쪽척추동맥의혈류파형은정상으로회복되었고, 양팔의혈압차는 5 mmhg로감소하였다. Acknowledgements: None Conflict of interest: None REFERENCES 1. Han M, Jin BH, Nam HS. The role of duplex sonography in right subclavian double steal syndrome. Korean J Clin Lab Sci. 2017; 49:316-321. 2. Clark CE, Campbell JL, Evans PH, Millward A. Prevalence and clinical implications of the interarm blood pressure difference:a systematic review. J Hum Hypertens. 2006;20:923-931. 3. Jung MS, Hong SI, Kim HS, Lee JH, Kim HJ, Ryu BY, et al. Subclavian steal syndrome. J Korean Surg Soc. 2009;76:203-206. 4. Park HK. Recent issues about neurointervention. J Neurocrit Care. 2011;4:42-49. 5. Kim BJ, Cho YP, Sohn HY, Lee EJ, Park SM, Kang DH, et al. Subclavian steal syndrome treated by axilloaxillary bypass surgery:the role of duplex sonography. J Korean Neurol Assoc. 2009;27:260-263. 6. Kim WB, Oh MK, Lee SH, Roh J, Kim HG, Kim JY, et al. A case of successful correction of subclavian steal syndrome by percutaneous transluminal angioplasty with stenting who found incidentally significant interarm blood pressure difference. Korean J Fam Med. 2009;30:979-984. 7. Lee SH, Kim DI, Kim JW, Cha KS, Lee SY, Kim SG, et al. Angiographic outcomes and predictors of recurrent restenosis after cutting balloon angioplasty for the treatment of in-stent restenosis. Korean Circulation J. 2003;33:196-204. 8. Alhaidar M, Algaeed M, Amdur R, Algahtani R, Majidi S, Sigounas D, et al. Early outcomes after carotid endarterectomy and carotid artery stenting for carotid stenosis in the ACS-NSQIP Database. J Vasc Interv Neurol. 2018;10:52-56. 9.Pollard H, Rigby S, Moritz G, Lau C. Subclavian Steal Syndrome:A review. Australas Chiropr Osteopathy. 1998;7:20-28. 10. Shim WH. Extracranial carotid stenting. Korean Circulation J. 1997;27:906-908. 11. Jang HC, Kim DH, Lee SS, Kim SH. Two cases of subclavian steal detected by duplex ultrasonography. Journal of Neurosonology. 2010;2:31-33.