21-이승준

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1 대한견 주관절학회지제 10 권제 1 호 J. of Korean Shoulder and Elbow Society Volume 10, Number 1, June, 2007 견관절색소융모결절성활막염의관절경적치료 - 증례보고 - 건국대학교의과대학정형외과학교실 성균관대학교의과대학삼성서울병원정형외과학교실 이승준 유재철 * 임경섭 Arthroscopic Treatment of Pigmented Villonodular Synovitis of the Shoulder - A Case Report - Seoung-Joon Lee, M.D., Jae Chul Yoo, M.D. *, Kyung-Sub Lim, M.D. Department of Orthopedic Surgery, Konkuk University Hospital, Seoul, Korea Department of Orthopedic Surgery, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul, Korea Pigmented villonodular synovitis (PVNS) is a benign proliferative lesion, involving synovial tissue in joints, tendon sheaths, and bursae. Pigmented villonodular synovitis is a rare and usually monoarticular condition and primarily affects the knee joint and hand. Polyarticular PVNS appears in less than 1% of all case and its occurrence in the shoulder is rare (<2%). We present a 64-year-old male who had pigmented villonodular synovitis of both shoulder joints, which was treated by arthroscopic total synovectomy. Key Words: Pigmented villonodular synovitis, Shoulder, Arthroscopy 서론색소융모결절성활막염 (PVNS, Pigmented Villonodular Synovitis) 은관절이나건초, 점액낭을침범하는양성증식성활막의병변으로, 1941년 Jaffe 등에의해처음보고되었다. 이는슬관절및수부에주로발생하며, 견관절에서발생하는경우는현재까지세계적으로 28 예만보고 될정도로드물다. 1% 미만의환자에서동시에두관절이상을침범하는것으로보고되고있으며, 실제로양측견관절을동시에침범하는경우는현재까지그보고를찾아보기가힘들다. 색소융모결절성활막염의치료는종괴절제및활막전절제술이표준치료로알려져있으나견관절의경우대해서는적절한수술적치료의방법및치료의결과에대한정보가적기때문에아직까지 통신저자 : 유재철 * 서울특별시강남구일원동 50번지 성균관대학교의과대학삼성서울병원정형외과학교실 Tel: 02) , Fax: 02) , coolshoulder@hotmail.com 140

이승준 : 견관절색소융모결절성활막염의관절경적치료 - 증례보고 - 논란이많은상태이다. 저자들은양측견관절에동시에발생한색소융모결절성활막염으로내원한 64 세환자에대해서양측견관절관절경적활액막전절제술을시행하여, 이에대해수술전소견과수술소견및임상경과에대해문헌고찰과함께보고하고자한다. 증례보고 64 세남자환자로 4개월전부터발생한양측견관절의통증및종창을주소로내원하였다.

자기공명영상 (MRI) 검사상양측견관절에전반적인활액막증식소견및관절내삼출액의증가, 관절내비특이적인결절성종괴소견이관찰되었다. 또한회전근개의파열과 이와관련한관절내병변의견봉하공간및특징적으로삼각근으로의파급 (invasion) 소견이관찰되었다 (Fig. 3). 이런소견을바탕으로감염 (low grade infection) 또는종양등을의심할수있었다.

2 이승준 : 견관절색소융모결절성활막염의관절경적치료 - 증례보고 - 논란이많은상태이다. 저자들은양측견관절에동시에발생한색소융모결절성활막염으로내원한 64 세환자에대해서양측견관절관절경적활액막전절제술을시행하여, 이에대해수술전소견과수술소견및임상경과에대해문헌고찰과함께보고하고자한다. 증례보고 64 세남자환자로 4개월전부터발생한양측견관절의통증및종창을주소로내원하였다. 내원시시행한신체검진상양측견관절의종창및삼각근의경한위축소견관찰되었으며 (Fig. 1), 양측견관절의거상및외전, 회전운동이모두감소되어있었다. 특히좌측견관절의경우는종괴와비슷하게견관절전방부의돌출이있었다. 단순방사선검사상양측상완골두대결절에경화소견및견갑상완관절의퇴행성변화관찰되었으며, 좌측상완골두외측으로 2 2 cm 크기의골낭종소견이관찰되었다 (Fig. 2). 자기공명영상 (MRI) 검사상양측견관절에전반적인활액막증식소견및관절내삼출액의증가, 관절내비특이적인결절성종괴소견이관찰되었다. 또한회전근개의파열과 이와관련한관절내병변의견봉하공간및특징적으로삼각근으로의파급 (invasion) 소견이관찰되었다 (Fig. 3). 이런소견을바탕으로감염 (low grade infection) 또는종양등을의심할수있었다. 이에진단및치료적목적으로관절경적검사를시행하였다. 관절경적검사상양측관절와-상완골관절내관절낭전반에걸친특징적인황갈색색소침착을보이는활액막증식소견이관찰되어활액막증식부분에대해집게겸자를이용하여조직 Fig. 1. Left shoulder area show huge swelling and bulging of the skin due to bursitis and PVNS. Fig. 2. Preoperative radiograph of both shoulder shows degenerative periosteal sclerosis combined with humeral head proximal migration and cystic lucency of left humeral head. 141

대한견 주관절학회지제 10 권제 1 호 A B Fig. 3. (A) Preoperative axial MRI of both shoulders show diffuse synovial proliferation with deltoid muscle infiltration and pigmented soft tissue mass (arrow).

3 대한견 주관절학회지제 10 권제 1 호 A B Fig. 3. (A) Preoperative axial MRI of both shoulders show diffuse synovial proliferation with deltoid muscle infiltration and pigmented soft tissue mass (arrow). (B) Preoperative coronal MRI of both shoulders show bilateral massive supraspinatus tendon tear and cystic mass on left humeral head. 검사후관절경적인활액막전절제술을시행하였다 (Fig. 4A-B, 5A-B). 또만성적인염증으로인해발생하였을것으로사료되는관절내유착이전후방관절낭부분에관찰되어이에대해유착해리술시행하였다. 관절경적견봉하공간관찰시양측견관절의회전근개광범위전층파열이있었으며 (Fig. 4C, 5C), 또한병변으로파급되었을것이라고사료되는점액낭의증식및황갈색색소침착의소견이관찰되었다. 관절경적변연절제시행후병변관찰하였을때병변은양측견관절모두삼각근의근막을뚫고근층까지파급되어있었다 (Fig. 5D). 좌측견관절의회전근개파열에대해서는추가적인봉합이가능할수있는것처럼보였지만수술당시감염을완전히배제하지못해비흡수성봉합사를이용하여부분봉합술만시행하고, 고정나사 (anchor) 를이용한봉합은시행하지않았다 (Fig. 4D). 우측에대해서는남아있는극상건및극하건이거의없어봉합을시행하지못하였다. 또단순방사선검사및자기공명영상에서관찰되었던좌측상완골두골낭종에대해서도관절경적인골낭종제거술시행하였다. 수술후환자는좌측견관절에대해서만 3주간외전보조기착용을하였으며, 우측견관절에대해서는술후 2일째부터수동적인견관절운동을시작하였다. 환자는술후 3주째퇴원하였으며, 퇴원시통증은많이감소하였으나관절운동범위와근력은크게호전되지않았다. 고찰 색소융모결절성활액막염은병인이밝혀지지않은증식성질환으로관절활액낭이나건막활액 142

(D) Arthroscopic photograph showing deltoid muscle infiltration at the subdeltoid bursa area. D A B C Fig. 4.

4 이승준 : 견관절색소융모결절성활막염의관절경적치료 - 증례보고 - A B C Fig. 5. (A, B) Right shoulder showing somewhat similar figures with left. (C) Arthroscopic photograph showing massive rotator cuff tear on the right shoulder. (D) Arthroscopic photograph showing deltoid muscle infiltration at the subdeltoid bursa area. D A B C Fig. 4. (A) Arthroscopic photograph shows proliferation of yellow-brown villous synovial lesion on left shoulder. (B) So we performed arthroscopic biopsy using pituitary forcep. (C) Arthroscopic pictures show large size rotator cuff tear on the left shoulder. (D) Arthroscopic view of the subacromial space after partial rotator cuff side to side repair. D 143

5 대한견 주관절학회지제 10 권제 1 호 막에결절성구조를형성하며, 결절의내부에는혈철소 (hemosiderin), 콜레스테롤과다핵성거대세포가침범하여노란색및황갈색을띄면서융모의증식을가져오는질환이다 10). 연간 100만명당 1.8 명의발병률을보이며, 이중 80% 는슬관절에발생한다고보고되고있으며 5), 견관절의발생은현재까지 28 예에서만보고될정도로매우드문것으로알려져있다 2,9). 국소형과미만형으로분류되며국소형은활액막의일부에결절이나종물의형태로나타나며주변의활액막은대체로정상소견을보이는반면에, 미만형은관절내활액막전체를침범한다. 견관절에서는미만형이흔하며, 미만형의경우회전근개의파열이동반된경우가많다고보고되고있다. 이런회전근개파열의원인으로종괴의국소침범보다는충돌증후군에의해이차적으로발생한것이라는주장이부각되고있다 8). 대부분의견관절색소융모결절성활액막염은관절내병변으로국한된경우가많고, 경우에따라점액낭과같은관절외구조물로파급되는것으로알려져있다 6). 하지만본증례의경우에는관절내병변과견봉하공간및삼각근부분까지파급되어있는관절외병변이동반되어있었다. 또한본증례처럼양측견관절동시침범에대해서는현재까지보고된바가없다. 흔한임상증상은비특이적이나침범된관절의종창, 통증및종괴의촉지이며관절천자시특징적인혈액섞인관절액을볼수있다. 단순방사선검사상압력에의한골미란이나연골하낭종, 연부조직종괴등을관찰할수있으며, 진행된병기에서는이차성골관절염소견이나이환된골의다낭성변화등을관찰할수있다 3,4). 자기공명영상 (MRI) 은비교적특이도가높은검사로진단적가치가높다. 이는연부조직종괴의양상을잘보여주며, 활액막의증식소견을관찰할수있다. 또 T1 강조영상및 T2 강조영상모두에서저신호강도를보이는특징적인혈철소의침착을관찰할수있다 3). 색소융모결절성활액막염의치료는국소형의경우종괴절제및국소활액막절제술이권장되며, 미만형의경우활액막의전절제술이표준치료로알려져있다. Beguin 등 1) 은슬관절의색소 융모결절성활액막염에서관절경적인활액막절제및종괴절제술로좋은결과를얻었다고보고하였으며, 이들은재율에있어서도관혈적인관절절제술과비교하여차이가없다고보고한바있다. 견관절색소융모결절성활액막염의치료에대해기존의문헌을고찰해볼때, 대다수의저자들은개방적활액막절제술및종괴절제술에의해좋은결과를얻었다고보고하였으며, Mahieu 등이 2례의견관절색소융모결절성활액막염환자에대해서관절경적인활액막절제술을통해좋은결과를얻었다는보고이외에는관절경적치료에대한보고를찾아보기힘들다 2,7,9). 이는대부분의환자들이진단의어려움으로인해진행된병기에진단되어골파괴및광범위한관절손상에대한치료가부가적으로필요하여개방적술식의선택이불가피하였고, 또한관절경술식및장비의미발달등의기술적인문제로관절경적인접근이어려웠던것으로판단된다. 최근, 관절경적인술식은관절내상태의정확한탐색이가능하고개방적술식으로접근이힘든관절낭의후하방부분도관찰및관절경적인활액막절제가가능하다는장점이있다. 견관절의색소융모결절성활막염은매우드물기때문에진단및치료에대한정보가적어논란이많은상태이다. 현재까지개방적술식에의한종괴절제및활막절제술이표준치료로알려져있으나본증례의경우처럼골침범이심하지않아관절치환이필요하지않는경우, 다른질환과감별을요하는경우에는관절경적인접법도견관절색소융모결절성활막염의좋은치료방법으로사료된다. 본증례에서회전근개의많은침범으로관절기능이저하된것은추후추가적인치료의대상이되는지는추시관찰이필요할것으로사료된다. REFERENCES 01) Beguin J, Locker B, Vielpeau C, Souquieres G: Pigmented villonodular synovitis of the knee: results from 13 cases. Arthroscopy, 5: 62-64, ) Chiffolot X, Ehlinger M, Bonnomet F, Kempf JF: Arthroscopic resection of pigmented villon- 144

6 이승준 : 견관절색소융모결절성활막염의관절경적치료 - 증례보고 - odular synovitis pseudotumor of the shoulder: a case report with three year follow-up. Rev Chir Orthop Reparatrice Appar Mot, 91: , ) Cotten A, Flipo RM, Mestdahg H, Chastanet P: Diffuse pigmented villonodular synovitis of the shoulder. Skeletal Radiol, 24: , ) Dorwart RH, Genant HK, Johnston WH, Morris JM: Pigmented villonodular synovitis of the shoulder: radiologic-pathologic assessment. AJR Am J Roentgenol, 143: , ) Johansson JE, Ajjoub S, Coughlin LP, Wener JA, Cruess RL: Pigmented villonodular synovitis of joints. Clin Orthop Relat Res, , ) Konrath GA, Nahigian K, Kolowich P: Pigmented villonodular synovitis of the subacromial bursa. J Shoulder Elbow Surg, 6: , ) Mahieu X, Chaouat G, Blin JL, Frank A, Hardy P: Arthroscopic treatment of pigmented villonodular synovitis of the shoulder. Arthroscopy, 17: 81-87, ) Mulier T, Victor J, Van Den Bergh J, Fabry G: Diffuse pigmented villonodular synovitis of the shoulder. A case report & review of literature. Acta Orthop Belg, 58: 93-96, ) Muller LP, Bitzer M, Degreif J, Rommens PM: Pigmented villonodular synovitis of the shoulder: review and case report. Knee Surg Sports Traumatol Arthrosc, 7: , ) Myers BW, Masi AT: Pigmented villonodular synovitis and tenosynovitis: a clinical epidemiologic study of 166 cases and literature review. Medicine (Baltimore), 59: , 초록 색소융모결절성활막염은양성증식성활막의병변으로관절이나건초, 점액낭등을침범한다. 이는주로한관절에발생하며, 다관절침범은 1% 미만에서보고되고있다. 또슬관절및수부에주로발생하며, 견관절에서발생하는경우는드물다. 색소융모결절성활막염의치료에대해서는아직까지논란이많은상태이나일반적으로활액막전절제술이표준치료로알려져있다. 저자들은양측견관절에동시에발생한색소융모결절성활막염으로내원한환자에대해서관절경적활액막절제술을시행하여수술소견과임상경과에대해문헌고찰과함께보고하고자한다. 색인단어 : 색소융모결절성활막염, 견관절, 관절경 145

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untitled Case Report J Korean Knee Soc, Vol. 22, No. 3, September 2010 A Case of Isolated, Localized Form, Pigmented Villonodular Synovitis of the Proximal Tibiofibular Joint Soo-Yong Kang, M.D., Han-Jun Lee, M.D.,