277 pissn : , eissn : Case Report J Korean Orthop Assoc 2021; 56: 인

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277 pissn : 1226-2102, eissn : 2005-8918 Case Report J Korean Orthop ssoc 2021; 56: 277-281 https://doi.org/10.4055/jkoa.2021.56.3.277 www.jkoa.org 인공슬관절전치환술후발생한 May Thurner 증후군 심창헌 박진우 왕립 동아대학교의과대학정형외과학교실 May Thurner Syndrome after Total Knee rthroplasty Chang Heon Shim, M.D., Jin Woo Park, M.D., and Lih Wang, M.D., Ph.D. Department of Orthopaedic Surgery, Dona- University College of Medicine, usan, Korea Iliac vein compression syndrome, which results in thrombosis of the left iliac veins, was first described by May and Thurner in 1957. May Thurner syndrome should be considered when deep vein thrombosis-like symptoms appear, especially in the left lower extremities without an invasive procedure. The authors encountered an interesting case of a middle-aged female patient, who presented with sudden pain, swelling and skin color changes to the left lower extremity after right total knee arthroplasty and was diagnosed May Thurner syndrome by computed tomography venography. This case is of clinical significance in that the early diagnosis of May Thurner syndrome in the left lower extremity was made, which might have been overlooked after right total knee arthroplasty. This case is reported with a review of the literature review. Key words: knee, arthroplasty, vonous thrombosis, May-Thurner syndrome 아시아인에서인공슬관절전치환술후심부정맥혈전증 (deep vein thrombosis, DVT) 의발생빈도는 12.3% 에서 71.0% 로다 양하게보고되고있다. 1) 비록발생률에있어서저자마다차이가 있지만일반적으로혈전은수술후동측하지에서주로발생하고 반대측하지에서발생하는경우는흔치않다. 2) 그러나인공슬관 절전치환술후동통, 부종및피부색변화를동반하는 DVT 유사 증상이반대측하지에서갑자기발생한경우 May Thurner 증 후군 (May Thurner syndrome) 의가능성을항상염두에두어야 한다. 장골정맥눌림증후군 (iliocaval compression syndrome, ICS) 으로알려져있는 May Thurner 증후군은 1957 년 May 와 Thurner 에의해기술되었다. 3) 그들은 430 예의사체에대해부 검을시행한연구에서 22% 의좌측총장골정맥 (left common Received pril 29, 2020 Revised June 24, 2020 ccepted June 26, 2020 Correspondence to: Lih Wang, M.D., Ph.D. Department of Orthopaedic Surgery, Dong- University Hospital, 26 Daesingongwonro, Seo-gu, usan 49201, Korea TEL: +82-51-240-2593 FX: +82-51-254-6757 E-mail: libi33@dau.ac.kr ORCID: https://orcid.org/0000-0001-9360-8637 *This study was supported by research funds from Dong- University. iliac vein) 이우측총장골동맥 (right common iliac artery) 에의한압박을관찰하였다. 만성압박으로인해정맥돌기 (venous spur) 의형성을초래하며좁아진혈관내강은혈류역학적이상이생겨혈전증발생에영향을준다. 3) May Thurner 증후군은좌측장골정맥의급성또는만성혈전증을유발하는질환으로좌측하지에서침습적인처치없이 DVT의증상이나타날때 May Thurner 증후군을의심할수있다. 저자들이아는바로본증례는우측인공슬관절전치환술후좌측하지에서발생한 May Thurner 증후군의첫번째증례이며술후 2주경과한상태에서갑작스러운좌측하지의통증, 부종및피부색변화를보이는중년여성환자의흥미로운사례를문헌고찰과함께보고하고자한다. 증례보고 만성신장질환 (chronic kidney disease, 2 3단계 ; glomerular filtration rate, 47.1 65.6 ml/min/1.73 m 2 ) 외에특이과거병력이없는 71세여자환자로양쪽퇴행성슬관절염진단하에대증치료에도불구하고증상이지속되어단계적인공슬관절전 The Journal of the Korean Orthopaedic ssociation Volume 56 Number 3 2021 Copyright 2021 by The Korean Orthopaedic ssociation This is an Open ccess article distributed under the terms of the Creative Commons ttribution 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.

278 Chang Heon Shim, et al. 치환술을시행하기로하였으며, 증상이심한우측슬관절에대해우선수술을시행하기로계획하였다. 우측인공슬관절전치환술 (Fig. 1) 후, 환자는혈전예방목적으로 bemiparin sodium 3500 IU을술후 6시간후피하주사하고이후 24시간마다같은용량으로술후 5일까지피하주사를시행하였다. 양측하지에모두간헐적공기압박기구 (intermittent pneumatic compression) 를술후 7일까지사용하였고수술후 3일째부터점진적보행을시작하였다. 술후 1주일이경과한상태에서우측종아리의부종및통증을호소하여도플러초음파 (Doppler sonography) 를시행하였고검사결과에서 DVT는관찰되지않았다 (Fig. 2). 이후환자는수술부위통증및부종감소등양호한결과를보였고보행에서도만족할만한결과를보여우측인공슬관절전치환술시행 2주후에 2차수술인좌측인공슬관절전치환술을계획하였다. 그러나술후 14일째, 좌측인공슬관절전치환술시행하루전, 환자는갑작스러운좌측하지부종및피부색변화를호소하였다 (Fig. 3). 증상은갑자기발생하였고이후급격히악화되어통증또한동반되었다. 좌측하지의증상외에흉부압박감, 호흡곤란등의증상은없었다. 증상발생시점의활력징후는혈압 130/75 mmhg, 맥박 76회 / 분, 온도 36.3 C, 호흡수분당 18 회, 실내공기에서산소포화도 99% 로정상범위를보였다. 그러나시행한혈액검사결과상 D-dimer 33.25 μg/ml로높게측정되었고, 양측하지모두정상적인맥박이만져졌으나좌측서혜 부에서족부말단까지전반적인부종및피부색변화가관찰되었다. 인공슬관절전치환술후동측이아닌반대측하지에서발생한전반적인급성부종및피부색변화라는점에주목하여원위보다는근위심부정맥혈전증을강력하게의심할수있었다. 이에선행적으로저분자헤파린 (low-molecular-weight heparin) 치료를시작하였고복부에서양측하지까지의컴퓨터단층촬영정맥조영술 (computed tomography venography) 을시행하여혈전증의진단및범위를파악하였다. 결과적으로좌측총장골정맥에서대퇴정맥까지혈전이확인되어 May Thurner 증후군을진단하였다 (Fig. 4). 진단후예정이었던좌측인공슬관절전치환술은취소하였고혈관외과협진하에경증의혈전성 May Thurner 증후군환자에해당하여항응고제치료를이용한보존적치료를우선시행하였으며 enoxaparin sodium 60 mg을퇴원까지총 8일간 1일 2회피하주사를시행하였다. 점자증상호전을보였고술후 3주째에환자는증상이회복되어퇴원하였다. 퇴원시 Cnoxane을중단하고경구항응고제인 edoxaban 60 mg을처방하였으며 1일 1회복용하도록교육하였다. 2주후본원정형외과및혈관외과외래를방문하였고 May Thurner 증후군증상은회복되어추가적치료없이 6개월간의항응고제치료만을시행하였다. 술후 2개월외래방문때에는아무런증상도호소하지않았으며술후 6개월재방문했을때에도증상재발은없었다. 당시시행한 D-dimer는 0.73 μg/ml로감소하였고추시 Figure 1. Simple plain radiographs show osteoarthritis of both knee joints at the anteroposterior (P) view (), and the right total knee arthroplasty at the P and lateral views (). Figure 2. Doppler sonography was performed to identify thrombosis in the veins. Thrombus was not present in the superficial femoral vein (SFV) () and popliteal vein (POV) (). The veins were well pressed when compressed using a probe.

279 May-Thurner Syndrome after TK 도플러초음파검사에서양하지모두혈전은관찰되지않았다. 고찰 May Thurner 증후군의정확한유병률은알려져있지않지만 May Thurner 증후군의유병률은사체연구로부검을통해약 22% 로보고되었다. 3) 유증상 DVT 에있어서 May Thurner 증 Figure 3. Clinical photograph shows that swelling and pigmentation from inguinal area to the toes of the contralateral lower extremity were observed abruptly after right total knee arthroplasty two weeks after surgery. 후군은전체하지 DVT의 2% 3% 만해당하기때문에대부분의 May Thurner 증후군은무증상인경우가많다. 4) 위험요인에는여성, 비만, 그리고 20 40세연령대가포함된다. 병태생리학적으로우측총장골동맥의만성적이고, 반복적인박동이좌측총장골정맥을손상시키고, 엘라스틴 (elastin) 및콜라겐 (collagen) 의침착을유발하여정맥돌기가형성된다. 결과적으로국소혈관내막증식, 정맥혈전증및정맥순환장애가순차적으로발생한다. 5) May Thurner 증후군환자는일반적으로만성편측성좌측하지팽창및통증을호소하고때때로피부색의변화, 만성통증, 재발성피부궤양또는정맥류가동반되기도하며치명적인사례로장골정맥파열또는폐색전증 (pulmonary thromboembolism) 이보고되기도하였다. 6) May Thurner 증후군의진단에있어서컴퓨터단층촬영정맥조영술, 자기공명정맥조영술 (magnetic resonance venography), 정맥내초음파 (intravenous ultrasound) 또는고식적정맥조영술은 May Thurner 증후군을탐지하는유용한진단도구가될수있다. 본증례에서는 May Thurner 증후군의진단을위해 D-dimer 검사와컴퓨터단층촬영정맥조영술을시행하여혈전의양과위치를파악하였고이를통해치료방법을결정하였다. May Thurner 증후군의치료일반적으로 DVT 증상이있을때시행하게된다. 치료방법은혈전의정도및범위에따라다를수있고, 주된치료법은약물기계적혈전용해술 (pharmacomechanical thrombolysis) 이며항응고제의사용이우선적으로시행하게된다. 최근에는중증 May Thurner 증후군에대해항응고제치료와함께스텐트또는풍선정맥성형술 (balloon venoplasty) 과같은덜침습적인중재시술이각광받고있고응 C Figure 4. Computed tomography venography axial views show the clot (arrows), which has an enlarged diameter of vessels and intraluminal filling defect into the left iliac vein at the pelvis inlet level (), and into the femoral vein at the pelvic outlet level (). Dilatation of the vein and the formation of intravenous thrombi (arrow) were observed in the transitional region of the left iliac vein to the femoral vein in the coronal view (C). LEI, the left external iliac artery; LII, the left internal iliac artery; LCIV, the left common iliac vein; LEIV, the left external iliac vein; LF, the left femoral artery; LFV, the left femoral vein.

280 Chang Heon Shim, et al. 고성향이높은고위험환자에게는하대정맥필터 (inferior vena cava filter) 치료또한권장되고있으며 7) 광범위한혈전이있을경우에는혈전제거술 (thrombectomy) 이필요하다. 본증례를보면국소적혈전의양이많지않았고증상또한심한경우가아니라혈관외과협진후우선항응고제단독요법인보존적치료를결정하였다. 이후증상호전여부에따라중재적시술을고려하였으나점진적인호전을보였고 1주째증상이거의소실되어경구항응고제로변경하여퇴원하였다. 본증례인경우전형적인 May Thurner 증후군으로좌측하지의경미한증상이나타났음에도불구하고처음에는 DVT와유사한증상이수술을시행한우측하지가아닌좌측하지에서발생했기때문에혼란스러웠다. May Thurner 증후군을야기할수있는선행요인으로혈전성향증 (thrombophilia), 대퇴정맥의몸통정맥기형 (truncal venous malformation) 또는후복막혈종 (retroperitoneal hematoma) 등이보고되고있으나 8,9) 본증례의경우혈전증을야기할만한과거력또는가족력은없었다. 본증례는우측인공슬관절전치환술시행이후반대쪽인좌측하지에서 DVT가발생한 May Thurner 증후군을보고한첫번째증례이다. 좌측총장골정맥압박및대퇴정맥까지의혈전형성의정확한원인은알수없으나저자는장골혈관이취약한해부학적구조에의해자발적으로압축될수있으며인공슬관절전치환술이무증상 May Thurner 증후군에있어서 DVT 발생또는악화의유발요인이될수있을것으로생각된다. ergen 등 10) 의연구에따르면잠재적좌측총장골정맥의압박은인공고관절전치환술후정맥혈전색전증 (venous thromboembolism) 의발생확률을유의하게증가시켰으나인공슬관절전치환술에서는유의한증가를보이지않았다. 이러한결과는인공고관절전치환술이인공슬관절전치환술에비해술후골반부동 (pelvic immobility) 의정도가높기때문에정맥혈전색전증의발생률이높을것으로추정되나향후지속적인원인분석및발생기전에관한추가연구가필요할것으로생각된다. 환자가수술이력에관계없이좌측하지에서편측 DVT 증상을나타낼때감별진단에있어서 May Thurner 증후군을고려하는것은매우중요하다. 임상의가 May Thurner 증후군을즉시진단하지못하면이환율과사망률이현저히높아질수있기때문이다. 이러한합병증을예방하기위해서는인공슬관절전치환술시행후반대쪽, 특히좌측하지에도주의를기울여야한다. 이러한점에서본증례는임상의가드물게마주하게될좌측하지에서의편측 DVT 발생시에대한조기진단및적절한치료에있어서충분한임상적가치가있다고생각된다. CONFLICTS OF INTEREST The authors have nothing to disclose. ORCID Chang Heon Shim, https://orcid.org/0000-0001-7746-5269 Jin Woo Park, https://orcid.org/0000-0001-6506-3027 Lih Wang, https://orcid.org/0000-0001-9360-8637 REFERENCES 1. Kim YH, Yoo JH, Kim JS. Factors leading to decreased rates of deep vein thrombosis and pulmonary embolism after total knee arthroplasty. J rthroplasty. 2007;22:974-80. 2. Davidson HC, Mazzu D, Gage F, Jeffrey R. Screening for deep venous thrombosis in asymptomatic postoperative orthopedic patients using color Doppler sonography: analysis of prevalence and risk factors. JR m J Roentgenol. 1996;166:659-62. 3. May R, Thurner J. The cause of the predominantly sinistral occurrence of thrombosis of the pelvic veins. ngiology. 1957;8:419-27. 4. Kibbe MR, Ujiki M, Goodwin L, Eskandari M, Yao J, Matsumura J. Iliac vein compression in an asymptomatic patient population. J Vasc Surg. 2004;39:937-43. 5. O'Sullivan GJ, Semba CP, ittner C, et al. Endovascular management of iliac vein compression (May-Thurner) syndrome. J Vasc Interv Radiol. 2000;11:823-36. 6. Oğuzkurt L, Ozkan U, Tercan F, Koç Z. Ultrasonographic diagnosis of iliac vein compression (May-Thurner) syndrome. Diagn Interv Radiol. 2007;13:152-5. 7. aron HC, Shams J, Wayne M. Iliac vein compression syndrome: a new method of treatment. m Surg. 2000;66:653-5. 8. Dheer S, Joseph E, Drooz. Retroperitoneal hematoma caused by a ruptured pelvic varix in a patient with iliac vein compression syndrome. J Vasc Interv Radiol. 2003;14:387-90. 9. Uhl JF, Gillot C, Chahim M. natomical variations of the femoral vein. J Vasc Surg. 2010;52:714-9. 10. ergen M, Wall KC, Kim CY, Garrigues GE. Occult left common iliac vein compression increases postoperative venous thromboembolism risk following total hip arthroplasty. J rthroplasty. 2019;34:375-8.

281 pissn : 1226-2102, eissn : 2005-8918 Case Report J Korean Orthop ssoc 2021; 56: 277-281 https://doi.org/10.4055/jkoa.2021.56.3.277 www.jkoa.org 인공슬관절전치환술후발생한 May Thurner 증후군 심창헌 박진우 왕립 동아대학교의과대학정형외과학교실 장골정맥눌림증후군으로알려져있는 May Thurner 증후군은 May와 Thurner에의해 1957년처음으로기술되었다. 좌측하지에서침습적인처치없이심부정맥혈전증의증상이나타날때 May Thurner 증후군을의심할수있다. 저자들은우측인공슬관절전치환술후좌측하지의급작스러운통증, 부종및피부색변화를보여컴퓨터단층촬영정맥조영술시행후 May Thurner 증후군을진단받은중년여성환자의흥미로운증례를경험하였다. 본증례를통해서우측인공슬관절전치환술후간과할수있는좌측하지 May Thurner 증후군을조기에진단하여합병증을예방하는데임상적의의가있다고본다. 이에문헌고찰과함께증례를보고하고자한다. 색인단어 : 슬관절, 관절성형술, 정맥혈전증, May-Thurner 증후군 접수일 2020 년 4 월 29 일수정일 2020 년 6 월 24 일게재확정일 2020 년 6 월 26 일책임저자왕립 49201, 부산시서구대신공원로 26, 동아대학교병원정형외과 TEL 051-240-2593, FX 051-254-6757, E-mail libi33@dau.ac.kr, ORCID https://orcid.org/0000-0001-9360-8637 * 본연구는동아대학교연구과제지원으로이루어졌음. 대한정형외과학회지 : 제 56 권제 3 호 2021 Copyright 2021 by The Korean Orthopaedic ssociation This is an Open ccess article distributed under the terms of the Creative Commons ttribution 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.