< DC1F5B7CA28B1E8C0B1BCF72DC0CCB9CCB6F D E696E6464>

Transcription

1 CASE REPORT Korean J Obstet Gynecol 2012;55(2): pissn eissn A CASE OF TREATMENT BY EMBOLIZATION OF UTERINE ARTERIAL PSEUDOANEURYSM FOLLOWING LAPAROSCOPIC SUPRACERVICAL HYSTERECTOMY Mi-Ra Lee, MD 1, Min-Jeong Kim, MD 1, Hye-Ji Jeon 1, MD, Sl-A Choi, MD 1, Yun-Sook Kim, MD 1, Dong-Han Bae, MD 1, Dong- Erk Goo, MD 2 Departments of 1 Obstetrics and Gynecology, 2 Interventional Radiology, Soonchunhyang University Chunan Hospital, University of Soonchunhyang College of Medicine, Chunan, Korea Laparoscopic supracervical hysterectomy is relatively new and minimally invasive surgery. The advantages of this operation are conserving the cervix. But it has complications like intraoperative bleeding, ureteral injury, bowel perforation and postoperative hematoma. Pseudoaneurysm can be acquired in association with trauma, previous surgery, trophoblastic disease. When a punctured artery does not seal completely, blood may escape and dissects the adjacent tissues, and collects in perivascular areas. If this maintains in communication with the parent vessel, a pseudoaneurysm could result. Typically the lesions are discovered because the patients have symptoms related to delayed rupture of the pseudoaneurysm, causing severe hemorrhage. Radiologic techniques have provided the opportunity to diagnose and treat pseudoaneurysm. We experienced a case of severe hemorrhage in the cervix four weeks following laparoscopic supracervical hysterectomy attributed to a pseudoaneurysm of the uterine artery and treated with arterial embolization. Keywords: Laparoscopic supracervical hysterectomy; Pseudoaneurysm; Arterial embolization 최근복강경수술이활발해지면서개복식전자궁절제술의빈도가줄고복강경자궁절제술의빈도가증가하고있으며, 마찬가지로개복식자궁상부절제술에비해복강경하자궁상부절제술의빈도또한증가하는추세이다 [1-3]. 복강경하자궁상부절제술은이론적으로개복수술에비해복부에남는흉터가적어미용적인면에서유리하고통증이적고재원기간단축및회복이빠르다는장점이있다. 자궁동정맥기형은자궁내에비정상적인혈관의증식및소통을보이는질환으로서현재까지국내에서여러증례보고가있었다 [4,5]. 증상은자궁출혈이대부분을차지하고이중약 30% 의환자는수혈을할정도의심각한출혈이다. 자궁출혈을유발하는다른질환과는다르게주로수술적외상후발생하며손상된혈관의치유실패때문에순차적으로섬유성피낭과손상된혈관조직이싸고있는연성조직으로혈액이유출됨으로써발생하는박동성혈종이다. 진단은초음파를이용한색도플러와전산화단층촬영이며, 혈관조영술이진단과치료에성공적이다 [6,7]. 저자들은복강경하자궁상부절제술시행 1개월후가성동맥류에의해발생한드문자궁경부내혈종에의한출혈을동맥색전술로진단및치료한매우드문예를경험하였기에보고하는바이다. 환자 : 손 O 숙 (41세) 산과력 : , 12년전과 10년전에 2회의질식분만을하였고, 당뇨나고혈압등은없었다. Received: Revised: Accepted: Corresponding author: Yun-Sook Kim, MD Department of Obstetrics and Gynecology, Soonchunhyang University Cheonan Hospital, University of Soonchunhyang College of Medicine, 31 Suncheonhyang 2-gil, Dongnam-gu, Cheonan , Korea Tel: Fax: drsook@schmc.ac.kr This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License ( by-nc/3.0/) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited. Copyright Korean Society of Obstetrics and Gynecology 137

2 KJOG Vol. 55, No. 2, 2012 월경력 : 14세에초경을시작하여 28일주기로규칙적이고기간은 7일이었으며, 양은중등도이었고월경통은없었다. 주소및현병력 : 1개월전개인병원에서자궁근종에의한생리통으로복강경하자궁상부절제술을시행했다. 해외여행중극심한질출혈로인한실신으로해외산부인과입원하여다섯팩의 red blood cell를수혈받았다. 이곳에서시행한질식초음파에서혈복강은없으나, 약 80 mm 크기의자궁경부입구쪽으로돌출된종괴소견을보여자궁육종과감별을위해조직검사가필요하다는이야기들었으나, 귀국하여하기로하고시행하지않았다. 귀국한이틀후다시성인기저귀 10장정도의출혈있어본원응급실통해입원하였다. 과거력 : 외상의기왕력, 약물복용력, 면역억제제복용력등의과거력은전혀없었다. 가족력과수술력 : 1개월전복강경하자궁상부절제술을시행받았다. 신체검사소견 : 입원당시신체검진에서혈압 80/50 mm Hg, 맥박 120회 / 분, 호흡수 20회 / 분, 체온 36.8 o C이었고의식은명료하였다. 흉부소견에서호흡음은깨끗하였고, 심박동은규칙적이었으며, 심잡음은들리지않았다. 결막은창백하였고, 복통은없었다. 질경을통해자궁경부를보니, 자궁경부의개구부가경부상부에생긴약 5 cm의혈종에의해아래쪽으로밀려있었고, 자궁경부상부는커져서마치자궁경부상부에종괴가생겨튀어나와있는것처럼보였다. 자궁경부개구부에서소량의질출혈은있었으나, 급성출혈소견은없었다. 질, 회음부, 서혜부에특이소견은없었다. 검사실검사소견 : 혈액검사에서혈색소는 6.0 g/dl ( 범위, g/dl) 로심한빈혈소견보였으며, 백혈구 12,030/mm 3 는정상, 혈소판은 130/mm 3 ( 범위, /mm 3 ) 으로정상소견보였고, 프로트롬빈시간 (prothrombin time) 12.8 sec ( 범위, sec), 부분트롬보플라스틴시간 (A activated partial thromboplastin time) 37.1 sec ( 범위, sec) 로약간연장되어있었다. 생화학검사에서 protein 5.2 g/dl ( 범위, g/dl), albumin 2.8 g/dl ( 범위, g/dl) 로경한감소소견을보였고, total bilirubin 0.7 mg/dl ( 범위, mg/dl) 이었고, apartate aminotransferase 38 IU/L ( 범위, 0-40 IU/ L), alanine aminotransferase 33 IU/L ( 범위, 0-40 IU/L) 로정상이었다. Alkaline phosphatase, urea nitrogen, creatinine 정상이었다. B형간염항원검사, 그리고요검사에서이상소견없었다. 흉부 X선촬영, 심전도에서이상소견없었다. 초음파소견 : 입원직후초음파에서복강내에는혈복강소견없었고, 자궁경관의앞입술에약 mm 크기의혈종이보였다. 컬러도플러에서혈액분포는증가되어있었으나, 가성동맥류인지여부는명확하 Fig. 2. The computed tomogram showed high attenuated sac like structure and extravasation from it after contrast infusion (arrow). Fig. 1. Transvaginal sonogram showed mm sized hematoma (arrow) in the anterior wall of the cervix, and no hemoperitoneum in the pelvic cavity. Fig. 3. The arterial angiography of the uterine artery showing pseudoaneurysm with a feeding vessel from the left uterine artery (arrow)

3 Mi-Ra Lee, et al. Embolization of uterine arterial pseudoaneurysm A B Fig. 4. The Diagram. (A) About 50 mm sized hematoma like mass was shown anterior lip of cervix, so the internal os of cervix (arrow) was displaced downward. We incised the mass (arrow head) and inserted Jones-Pitzerald catheter drain. (B) Five days after the operation, the internal os of cervix (arrow) was back again. 지않았다 (Fig. 1) 컴퓨터단층촬영소견 : 입원시응급실에서시행한컴퓨터단층촬영에서조영제를주사한후혈종내로조영증강을보이는주머니모양의구조와혈관밖으로의조영제유출이보여좌측자궁동맥의가성동맥류를강력히의심할수있었다 (Fig. 2). 치료및경과 : 당일중재시술방사선과에서우측대퇴동맥으로접근하여 5 French도관을이용하여혈관조영술을시행한결과좌측자궁동맥이영양혈관으로지지하는가성동맥류가관찰되었다 (Fig. 3). 좌측자궁동맥에색전용재료인 gelform, coil을주입하여색전술을시행하였다. 시술후시행한혈관조영사진에서혈관의완전한폐색을확인후카테터를제거하고천자부위를압박하여지혈하였다. 더이상의출혈은없어자궁경부부위에생긴혈종은메스로절개하여제거후 Jones- Pitzerald catheter 드레인을넣어 5일간거치하였다 (Fig. 4A). 5일후시행한초음파에서혈종의크기는 mm로감소하였고 (Fig. 4B), 추가수혈없이헤모글로빈수치유지되어드레인을제거후퇴원하였다. 퇴원 3주후외래에서시행한초음파에서더이상의혈종은보이지않았다. 부인과수술에서복강경을이용한미세침습수술법은작은수술절개, 통증감소, 상처의빠른회복, 일상생활로의빠른복귀등의장점으로인해널리이용되고있다. 1989년 Reich 등 [8] 에의해최초로복강경을이용한자궁절제술이시행된이래과거에복식으로시행되던많은자궁절제수술이복강경수술로대체되고있다 [9]. 복강경을이용한 자궁절제술에는완전자궁절제술인복강경하질식자궁절제술과복강경하완전자궁절제술등이있으며, 불완전자궁절제술에는복강경하자궁상부절제술이있다. 복강경하자궁상부절제술은비정상자궁출혈의치료를위해 1990년대에개발되었다. 수술기법의안정성, 유효성, 재현성이확인되었다 [10,11]. 수술중의합병증은출혈, 방광천공, 피하기종, 요관손상등이있을수있고, 수술후의합병증으로트로카삽입부위봉소염, 지속적인질출혈, 지연출혈, 지속적인질분비물이약 2 % 에서있을수있다고보고되고있으나, 아직까지가성동맥류에의한지연출혈에대한보고는없었다 [12]. 가성동맥류는치명적인출혈을일으킬수있는질환으로손상된혈관의치유실패때문에순차적으로섬유성피낭과손상된혈관조직을싸고있는연성조직으로혈액이유출되어발생하는박동성혈종이다. 발생률에대한보고는거의없으며, 원인은외상, 골반수술, 융모성질환, 종양, 감염또는 diethylstilbestrol 등에노출시생길수있다. 발생기전은확실하지않지만혈관손상에의한부분적외상에의한것으로생각되며동맥이천자되거나열상이일어났을때완전히지혈되지않은경우혈액이유출되면서주변조직을분리시키고혈관주변으로축적되며, 주변은혈전으로구성되어세층의동맥층으로둘러싸여있지않은점이진성동맥류와감별점이된다 [13]. 본증례의경우가성동맥류는복강경수술시좌측자궁동맥손상에의해생긴것으로생각된다. 증상은파열에의한대량출혈이대부분이며증상이없는경우발견이어렵고, 출혈양은다양하다. 진단방법은초음파, computed tomography, magnetic resonance imaging, 도플러초음파등이있고확진은동맥혈관조영술이다 [14]. 치료는급성출혈이있을때에는개복하여내장골동맥결찰술이나자궁동맥결찰술, 또는남아있는자궁경부를수술적으로제거하는방법등이있으나, 정확하게출혈부위를알 139

4 KJOG Vol. 55, No. 2, 2012 수없으나, 검사에서계속적으로출혈이의심되는경우조영술을통해출혈부위를쉽게찾을수있고, 진단과동시에동맥색전술을통해치료가가능한장점이있다. 선택적혈관조영술을통한동맥색전술은과거 1960년대이후부터복부와골반외상등의위장관질환, 악성신생물, 방사선으로인한출혈조절의목적으로광범위하게사용되어왔으나, 최근에는산부인과영역에서태반이상을동반한각종산과적출혈, 자궁수축부전, 자궁동정맥기형의치료, 가성동맥류, 자궁경부나제왕절개부위임신의보존적치료, 자궁근종의종괴축소목적이나진행성자궁경부암환자의심한출혈에대한지혈목적등산과및부인과영역의출혈에아주효과적인방법으로대두되고있다. Langer와 Cope 등은질식자궁절제술 14일후감염된골반내혈종으로의심하였던자궁동맥의가성동맥류를질식으로배액하여 (drain) 치료한예를보고하였고 [15], Zimon 등 [15] 은 31세여성에서분만후 8일동안하복부통증이있었던가성동맥류를혈관색전술로성공적으로치료한예를보고하였다. 색전물질로사용되는재료로는크게멸균흡수성 gelform 과비흡수성재료인 polyvinyl alcohol, 강철코일 (coil), isobutyl-2- cyanocrylate 및 N-butyl- cyanoacrylate 등으로나뉜다. Gelform은비교적저렴하고주입방법이용이하며항원성과자극성이비교적적다. 또한영구적이지않고시술후 21-23일정도면부분적으로재소통되고, 30-35일후에는완전히재소통되므로생식력의보존이가능하며생리재개가가능하면서장기적인독성이없다는장점과함께주로 1 mm 이하의직경을가진원위부동맥의색전에이용된다. Coil을포함한비흡수성재료등은미세혈관에정확한부위의장기적색전효과가뛰어나고 gelform으로조절이안되거나지속적이고영구적인폐색이필요한경우사용되며혈액응고장애가있는환자나외상후고혈류량을가진동정맥누공에사용가능하고주로큰분지를가진가성동맥류의근위부혈관이나큰동정맥누공의폐색을위해사용된다. 상기환자의경우 gelform으로색전을시도후출혈조절이용이하지않고, 지속적이고영구적인폐색이필요하여 coil을이용하여추가색전을시행하였다. 혈관색전술의합병증으로동맥파열, 천자부위에발생한혈종, 감염및농양, 조영제에따른과민반응, 신독성, 혈관폐색으로인한혈관말단부위허혈에의한통증, 부적절한혈관색전으로인한방광및소장괴사, 하지마비, 둔부근육괴사등이보고된바있다. 1997년 Vendantham 등은합병증발생률이약 6-7% 정도된다고보고하였는데, 가장흔한합병증은시술후발열로전형적으로 2-3일내에회복되고심한복통은시술후 24시간이내에주로나타나게되는데, 이러한통증은혈관폐색에의한허혈의결과로여겨지며대부분의환자에게 1주간의안정을권유한다. 상기환자의경우색전술후특별한합병증발생은없었다 [13]. 본환자의경우 1개월전복강경하자궁상부절제술을시행후생긴좌측자궁동맥가성동맥류의파열에의한대량의출혈로수혈을받았고, 현재까지복강경하자궁상부절제술후지연성대량출혈의합병증에대한보고가매우드물어자궁경부에다시발생한자궁경부근종으로생각하여진단이늦어졌으나, 컴퓨터단층촬영과초음파에서가성동맥류의심되는소견보여시행한혈관조영술로가성동맥류로진단이 되어색전술에의해치료한매우드문경우였다. 최근복강경하자궁상부절제술이증가하면서여러가지합병증발생의보고가늘고있어상기증례를간단한문헌고찰과함께보고하는바이다. References 1. Nam AN, Cho SH, Seo KS, Jeon YE, Kim HY, Choi YS. Laparoscopic total hysterectomy versus laparoscopic supracervical hysterectomy: the effect on female sexuality. J Womens Med 2008;1: Washington JL. Laparoscopic supracervical hysterectomy compared with abdominal, vaginal, and laparoscopic vaginal hysterectomy in a primary care hospital setting. JSLS 2005;9: Jin K. Comparative study of laparoscopic assisted vaginal hysterectomy and laparoscopic supracervical hysterectomy. Korean J Obstet Gynecol 2010;53: Lee JW, Kim HC, Chang SW, Kang SH, O YR, Seok HH, et al. Cases of transarterial embolization in arteriovenous malformation of uterus. Korean J Obstet Gynecol 2005;48: Kim MJ, Park CH, Kwen I, Lee HJ, Hur SY, Kim EJ, et al. An uncommon cause of postpartum hemorrhage after cesarean section treated with selective arterial embolization: pseudoaneurysm of the uterine pedicle. Korean J Obstet Gynecol 2004;47: Hoffman MK, Meilstrup JW, Shackelford DP, Kaminski PF. Arteriovenous malformations of the uterus: an uncommon cause of vaginal bleeding. Obstet Gynecol Surv 1997;52: Kwon JH, Kim GS. Obstetric iatrogenic arterial injuries of the uterus: diagnosis with US and treatment with transcatheter arterial embolization. Radiographics 2002;22: Reich H, DeCaprio J, McGlynn F. Laparoscopic hysterectomy. J Gynecol Surg 1989;5: Liu CY. Laparoscopic hysterectomy. A review of 72 cases. J Reprod Med 1992;37: Lyons T. Laparoscopic supracervical versus total hysterectomy. J Minim Invasive Gynecol 2007;14: Lyons TL. Laparoscopic supracervical hysterectomy. Obstet Gynecol Clin North Am 2000;27: van Evert JS, Smeenk JM, Dijkhuizen FP, de Kruif JH, Kluivers KB. Laparoscopic subtotal hysterectomy versus laparoscopic total hysterectomy: a decade of experience. Gynecol Surg 2010;7: Shin WK, Kim DJ, Kim HM, Park SH, Jun HA, Lee KY. A case of angiography and transarterial embolization in arteriovenous 140

5 Mi-Ra Lee, et al. Embolization of uterine arterial pseudoaneurysm malformation of uterus after a cesarean section episode. Korean J Obstet Gynecol 2007;50: Mitchell DG, Needleman L, Bezzi M, Goldberg BB, Kurtz AB, Pennell RG, et al. Femoral artery pseudoaneurysm: diagnosis with conventional duplex and color Doppler US. Radiology 1987;165: Zimon AE, Hwang JK, Principe DL, Bahado-Singh RO. Pseudoaneurysm of the uterine artery. Obstet Gynecol 1999;94: 복강경하자궁상부절제술후발생한자궁동맥가성동맥류의색전술에의한치료 1 예 순천향대학교의과대학 1 산부인과학교실, 2 중재시술방사선과학교실이미라 1, 김민정 1, 전혜지 1, 최슬아 1, 김윤숙 1, 배동한 1, 구동억 2 복강경하자궁상부절제술은비교적새롭고최소침습적인수술로자궁경부를보존하고부속기수술도동시에할수있는장점이있다. 그러나수술중출혈, 요관손상, 장천공, 그리고수술후혈종형성등의합병증이생길수있다. 가성동맥류는외상, 이전수술, 융모성질환, 종양또는 diethylstilbestrol에노출된경우발생한다. 동맥이천자되거나열상이일어났을때완전히지혈되지않은경우혈액이유출되면서주변조직을분리시키고혈관주변으로축적되어인접한혈관과교통되면서발생하게되며, 전형적으로출혈을일으키는가성동맥류의지연성파열에의해발견이되고, 방사선학적인기술로진단과동시에치료가된다. 저자들은최근복강경하자궁상부절제술시행 1 개월후가성동맥류파열에의해발생한매우드문자궁경부내지연성대량출혈을동맥색전술로진단, 치료한매우드문예를경험하였기에보고하는바이다. 중심단어 : 복강경하자궁상부절제술, 가성동맥류, 동맥색전술 141