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1 증례접수번호 :09-029(2 차 -0710) 을지대학교의과대학을지대학병원신경과, 정형외과 a 윤동주이현정최영철윤수진오건세이수주김하용 a Piriformis Syndrome Following Transarterial Embolization of Bilateral Uterine Arteriovenous Malformation Dong Joo Yun, MD, Hyun Jeong Lee, MD, Young Chul Choi, MD, Soo Jin Yoon, MD, Gun-Sei Oh, MD, Soo Joo Lee, MD, Hayong Kim, MD a Departments of Neurology and Orthopaedic Surgery a, Eulji University Hospital, Eulji University School of Medicine, Daejeon, Korea Piriformis syndrome is a rare entrapment neuropathy in which the sciatic nerve is compromised by the piriformis muscle or other local structures. We report a case of sciatic and gluteal nerve lesions with infarction of the piriformis muscle following internal iliac artery embolization of the bilateral uterine vascular malformation. The surgical intervention revealed anatomical variation of the right sciatic nerve. To our knowledge, this is the first case of piriformis syndrome following endovascular treatment of uterine vascular malformation in Korea. J Korean Neurol Assoc 30(1):37-41, 2012 Key Words: Piriformis syndrome, Uterine arteriovenous malformation 궁둥구멍근육증후군 (piriformis syndrome) 은궁둥구멍근육과좌골신경의해부학적이상때문에생기는좌골신경통을말한다. 골반부나둔부에외상이나궁둥구멍근육의비정상적인비후, 하지길이의차이나궁둥구멍근육근염과같은병변에의해좌골신경이압박을받는경우에둔근부위의동통과하지의방사통을호소하게된다. 1 자궁동정맥기형 (uterine arteriovenous malformation) 은자궁내에비정상적인혈관의증식과교통이생기는매우드문질환으로과거에는대부분전자궁적출술을하였으나최근에는동맥경유색전술 (transarterial embolization) 을이용한보존치료를한다. 2 저자들은자궁동정맥기형에서동맥경유색전술후우측하지 Received April 28, 2011 Revised May 31, 2011 Accepted May 31, 2011 *Dong Joo Yun, MD Department of Neurology, Eulji University Hospital, 1306 Dunsan-dong, Seo-gu, Daejeon , Korea Tel: Fax: djy@eulji.ac.kr 통증과위약감이발생한환자가전기생리검사에서좌골신경, 상둔신경 (superior gluteal nerve), 하둔신경 (inferior gluteal nerve) 병변과골반자기공명영상에서궁둥구멍근육의허혈성근괴사를보여궁둥구멍근육증후군으로판단한환자를경험하였기에보고하는바이다. 증례 32세여자환자가 5년전과다자궁출혈때문에시행한복부대동맥과자궁혈관조영술에서양측자궁동맥에서혈류공급을받는동정맥기형이있었다. 각각의자궁동맥을선택하여미세도관을동정맥기형핵 (nidus) 근처까지접근시킨뒤겔거품 (gelfoam) 과폴리비닐알코올입자 (polyvinyl alcohol particles) 를혼합하여양측동맥색전술을하였다. 색전술후에영양혈관 (feeding vessels) 은모두차단되었고양측자궁동맥에서공급받는혈류는현저히감소되었으나약간의혈류공급이남아있는것이확인되었다. 환자는특별한문제없이퇴원하였다. 이후반복적인자궁출혈로 2차례의색전술을추가로시행하였고다 J Korean Neurol Assoc Volume 30 No. 1,

2 윤동주이현정최영철윤수진오건세이수주김하용 른신경학적증상은없었다. 내원 10일전시행한추적골반혈관조영술에서양측자궁동맥과내장골동맥 (internal iliac artery) 에서기시하는새로운측부순환이형성된것을확인할수있었고각각의자궁동맥과내장골동맥을선택하여리피오돌 (lipiodol) 과아교물질 (glue material) 을 1대 1로혼합하여양측동맥색전술을하였다. 그결과더이상의동정맥기형은없었다. 환자는시술이후우측다리전체에저린증상을호소하였고 5일전부터는우측발바닥과종아리에전기오듯이찌릿찌릿하고화끈거리는통증이발생하였고발목의근위약도나타났다. 과거력과가족력에이상은없었다. 다리펴들기검사 (straight-leg raising test) 는 30 로제한되었고궁둥구멍근육징후가나타났다. Freiberg 씨징후와 Pace 씨징후는없었다. 의식은명료하였으며뇌신경검사는정상이었다. 근력검사에서 MRC (Medical Research Council) 등급으로우측엉덩이의외전 (abduction), 내전 (adduction), 굴곡 (flexion), 신전 (extension) 은정상이었다. 우측무릎신전은정상이었고무릎굴곡은 4등급이었다. 우측발등굽힘 (dorsiflexion), 발바닥쪽굽힘 (plantar flexion), 발목의내번 (ankle inversion) 과외번 (eversion) 은모두 4등급이었다. 감각기능검사에서이질통증 (allodynia) 과통각과민 (hyperalgesia) 이우측무릎이하에서나타났고특히발바닥에서심하였다. 심부건반사는우측발목반사가소실되었으나병적반사는없었다. 감각신경전도검사에서양측천비골신경과비복신경의신경전도속도와진폭은정상범위였지만우측진폭이좌측보다감소되었다. 복재신경 (saphenous nerve) 은양측차이가없었다. 운동신경전도검사에서우측비골신경의진폭이감소되었고, 양측경골신경과좌측비골신경은정상이었다. 대퇴신경전도검사는 Stohr 방법에의하여시행하였고말단잠복기와진폭은정상범위였다. 우측비골신경과경골신경의 F- 파잠복기가증가되었고, H- 반사도우측에서잠복기가증가되어있었다. 침근전도검사에서우측앞경골근, 내측장딴지근, 긴비골근, 뒤경골근, 대퇴두갈래근, 중간둔부근, 큰둔부근에서삽입전위가증가되었고안정시비정상적인자발전위가있었으며근수축시에간섭양상이감소되었다. 우측가쪽넓은근, 큰내향근, 요천추주위 Table. Nerve conduction study and electromyography of the lower extremities A) Nerve conduction study Motor nerve Terminal latency (ms) Amplitude (mv) Conduction F wave latency (ms) distal/proximal velocity (m/s) Right peroneal / Left peroneal / Right tibial / Left tibial / Right femoral Left femoral H reflex (ms) 31.5 (Right)/24.8 (Left) Sensory nerve Latency (ms) Amplitude (μv) Conduction velocity (m/s) Right superficial peroneal Left superficial peroneal Right sural Left sural Right saphenous Left saphenous B) Electromyography Muscle (Right side) Insertional activity Spontaneous activity Volitional activity (fibrillation/psw) (duration/amplitude) Recruitment Tibialis anterior Increased None/1+ Normal/Increased Reduced Gastrocnemius medial head Increased 2+/2+ Normal/Normal Normal Peroneus longus Increased 3+/3+ Normal/Normal Reduced Tibialis posterior Increased 1+/2+ Normal/Increased Reduced Biceps femoris long head Increased 2+/2+ Normal/Normal Reduced Gluteus medius Increased 1+/2+ Increased/Normal Normal Gluteus maximus Increased 1+/2+ Normal/Normal Normal Other muscles a Normal Normal Normal Normal PSW; positive sharp wave. a right vastus lateralis, adductor magnus, lumbosacral paraspinal muscles. 38 대한신경과학회지제 30 권제 1 호, 2012

A C B Figure 1. Pelvis MRI of the patient.

increase in signal intensity on T2-weighted image (arrow).

right iliac and superior gluteal vessels (arrow) anterior to the right priformis muscle on

(C) Gadolinium-enhanced T1-weighted coronal image demonstrates contrast enhancement of

nerve enhancement (arrow). A B Figure 2. Photography of the operating field.

3 A C B Figure 1. Pelvis MRI of the patient. (A) MR scan demonstrates diffuse thickening of the right piriformis muscle and slight increase in signal intensity on T2-weighted image (arrow). (B) The lesion shows peripheral enhancement with central unenhanced area with dilatation of right iliac and superior gluteal vessels (arrow) anterior to the right priformis muscle on gadolinium-enhanced T1-weighted axial image. (C) Gadolinium-enhanced T1-weighted coronal image demonstrates contrast enhancement of surrounding soft tissue in the right sciatic nerve without remarkable thickening or abnormal nerve enhancement (arrow). A B Figure 2. Photography of the operating field. (A) The common peroneal division of sciatic nerve passes superior to the piriformis muscle. (B) The pale and fibrotic gluteus minimus muscle (long white arrow) compresses proximal portion of the sciatic nerve. J Korean Neurol Assoc Volume 30 No. 1,

4 윤동주이현정최영철윤수진오건세이수주김하용 근에서는정상이었다 (Table). 전기생리학적검사에서상둔근신경, 하둔근신경, 좌골신경에서이상이있었고동맥경유색전술과의연관성을의심하여시행한골반자기공명영상에서우측궁둥구멍근육이비후되고 T1 강조영상에서불균질한조영증강을보여허혈성근괴사가의심되었고좌골신경주행주위를따라연부조직의조영증강이있었다 (Fig. 1). 약물요법, 요추부신경근차단술같은보존치료를시행하였으나호전되지않았다. 궁둥구멍근육과근막의염증, 부종때문에좌골신경이눌려서좌골신경통이발생하였을것으로판단하고정확한진단과치료를위하여수술을하기로했다. 수술시야에서좌골신경이정상주행과는다르게궁둥구멍근육위로주행하였으며심한섬유성변화에의해창백해진작은둔부근에의해좌골신경이압박되었다. 작은둔부근을절제하여좌골신경에가해지는압력을감압하였다 (Fig. 2). 조직검사에서절제한작은둔부근에섬유화를확인하였다. 수술 3개월후에환자의하지방사통은상당히호전되었다. 다시시행한신경전도검사에서우측비복신경진폭이 8.3 μv에서 17.3 μv로호전되었고우측비골신경의진폭도 1.2 mv에서 4.6 mv 로정상소견을보였다. 고찰 궁둥구멍근육증후군이란궁둥구멍근육의근긴장이나비후로인해둔근신경이나좌골신경이압박되어이들이분포하는둔부와하지에통증, 저림, 당김, 이상감각이나타나는질환으로서다음의네가지임상양상이특징이다. 1) 둔부통증, 2) 대좌골절흔부분의압통, 3) 앉을때악화되는통증, 4) 궁둥구멍근육의긴장이증가되는수기에의한통증증가이다. 3 대표적인검사법으로고관절을신전시킨상태에서내회전시통증이유발되는 Freiberg 씨징후와고관절을굴곡한상태에서외전하려고하는것을방해할때근력약화와동통이나타나는 Pace 씨징후가있으며, 이외에환자를앙와위로눕히면이환된부위가외회전되어비대칭적인모습을보이는궁둥구멍근육징후가있다. 3 그외에음부신경, 하둔부신경, 상둔부신경이침범되어성교불쾌증, 고관절외전근의약화가동반될수있다. 4 궁둥구멍근육증후군의원인으로는궁둥구멍근육의기형이나둔부외상에의해궁둥구멍근육에염증이생겨좌골신경을압박하거나궁둥구멍근육의유발점증후군, 좌골신경의해부학적변이를생각할수있다. 1 일반적으로좌골신경은분지되지않은상태로궁둥구멍근육의밑을통과하며 10-21% 에서해부학적변이가나타난다 % 정도는비골신경부와경골신경부로분지되어비골신경 부만궁둥구멍근육을관통하고, 2-3% 는분지되어궁둥구멍근육의위, 아래로지나가고, 약 0.5% 정도는궁둥구멍근육위로주행하고, 1% 정도는분지되지않은상태로근육을관통한다. 5 본환자의경우에서도비골분지가궁둥구멍근육위로주행하는변형을보였다. 좌골신경의해부학적변이만으로도원인이될수있지만해부학적변이와함께궁둥구멍근육과둔부근육의비후나주변조직의섬유화나유착이동반된경우도원인이될수있다. Hare 등은내장골동맥색전술후에하지위약과통증이발생한환자 3명을보고하였고, 색전에의한좌골신경과대퇴신경에혈액을공급하는혈관의폐색을기전으로제시하였다. 6 본환자는양측자궁동정맥기형을치료하기위하여총 4차례에걸쳐자궁동맥과내장골동맥색전술을받은후에우측궁둥구멍근육의허헐성근괴사와우측상둔근신경, 하둔근신경, 좌골신경의허혈성손상이발생하였다. 이는궁둥구멍근육의허혈성근괴사와염증이좌골신경과주위신경을압박하여허혈성손상을초래하였을것으로판단하였다. 수술시야에서확인된우측좌골신경의해부학적변이가이러한손상과함께증상을유발하였을것이다. 하지만내장골동맥색전술당시에좌골신경, 상둔근신경, 하둔근신경을담당하는동맥이색전에의해동시에폐색되어신경의허혈성손상이발생하였을가능성도배제할수없다. 자궁동정맥기형은 1926 년 Dubreuil 등이보고한뒤지금까지전세계적으로 100 여개미만으로보고된드문질환이다. 7 치료는주로자궁적출술을시행하였으나최근에는임신을원하는여성에게동맥경유색전술을시행하며, 치료성적도비교적좋은것으로알려져있다. 8 합병증으로는외장골동맥파열, 방사선조영제사용같은시술과직접연관된합병증, 서혜부혈종, 골반내감염, 허혈성합병증, 골반통증이있다. 8 동정맥기형에서동맥경유색전술후에신경손상이발생한경우는극히드물게보고되었다. 9,10 Ozguclu 등은우측내장골동맥에서기시하는동정맥기형에서동맥색전술후발생한좌골신경손상을보고하였으나, 10 본증례와같이좌골신경의해부학적변이가동반된상태에서동맥경유색전술후에발생한궁둥구멍근육과주변근육의허혈성손상과이로인한신경압박은아직까지없다. 본증례는자궁동정맥기형에서동맥경유색전술후에우측하지에심한통증과위약감이발생하였으며이학검사와신경계진찰에서좌골신경손상이의심되었다. 신경전도와근전도검사에서상둔근신경, 하둔근신경, 좌골신경에이상이확인되었고골반자기공명영상에서궁둥구멍근육의허헐성근괴사와비후, 좌골신경주행부위의염증이있었다. 수술시야에서좌골신경의비골분지가궁둥구멍근육위로주행하는해부학적변이가 40 대한신경과학회지제 30 권제 1 호, 2012

5 보였고심한섬유화가진행된작은둔부근에의해좌골신경이압박되어있었다. 작은둔부근을절제하여좌골신경에가해지는압박을감압시켜줌으로써증상이호전되었다. 본증례와같이동맥경유색전술후좌골신경손상증상이나타나는환자에서궁둥구멍근육증후군이의심된다면자기공명영상을시행하여비후된궁둥구멍근육을확인하는것이진단에도움이될수있으며확실한진단과치료를위하여수술도고려해야할것이다. REFERENCES 1. Synek VM. The pyriformis syndrome: review and case presentation. Clin Exp Neurol 1987;23: Hoffman MK, Meilstrup JW, Shackelford DP, Kaminski PF. Arteriovenous malformations of the uterus: an uncommon cause of vaginal bleeding. Obstet Gynecol Surv 1997;52: Hopayian K, Song F, Riera R, Sambandan S. The clinical features of the piriformis syndrome: a systematic review. Eur Spine J 2010;19: Ahn SH, Moon JH, Kim YC. Clinical studies on piriformis syndrome. J Korean Acad Rehab Med 1993;17: Hallin RP. Sciatic pain and the piriformis muscle. Postgrad Med 1983;74: Hare WS, Holland CJ. Paresis following internal iliac artery embolization. Radiology 1983;146: Timmerman D, Van den Bosch T, Peeraer K, Debrouwere E, Van Schoubroeck D, Stockx L, et al. Vascular malformations in the uterus: ultrasonographic diagnosis and conservative management. Eur J Obstet Gynecol Reprod Biol 2000;92: Kwon JH, Kim GS. Obstetric iatrogenic arterial injuries of the uterus: diagnosis with US and treatment with transcatheter arterial embolization. Radiographics 2002;22: Ney JP, Shih W, Landau ME. Sciatic neuropathy following endovascular treatment of a limb vascular malformation. J Brachial Plex Peripher Nerve Inj 2006;1: Ozguclu E, Kilic E. Sciatic nerve paralysis following arteriovenous malformation embolization. Am J Phys Med Rehabil 2009;88:82. J Korean Neurol Assoc Volume 30 No. 1,

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Microsoft PowerPoint - 김호성 Myofascial Manual Therapy of L/E - Principle & Treatment - Myofascial Manual Therapy of L/E Principles of Treatment 2012 KATA Kim, Ho-Sung (Ph D, PT Kim Ho Sung (Ph D, PT Myofascial Manual Therapy of L/E