13-09 박재형

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1 /jkhs Case Report Lipoma rborescens in the Hip Joint - Case Report - Jai-Hyung Park, MD, Hyung-Soo Kim, MD, Soo-Tae Chung, MD, Jeong-Hyun Yoo, MD, Joo-Hak Kim, MD, Seung-Do Cha, MD, Jin-Ha Jung, MD Department of Orthopaedic Surgery, Myongji Hospital, College of Medicine, Kwandong University, Goyang, Korea Lipoma arborescens is a very rare disease in which adipocytes form a synovial villous lipoma. In addition, it has the potential for dysplastic proliferation. Hypertrophic synovial lipoma is stimulated by movement of joints, which results in joint pain, effusion, limitation of joint movement and swelling. definite cause has not yet been proven, but trauma, rheumatic arthritis, inflammation and degenerative change are believed to be possible causes. There have been reports of lipoma arborescens in several joints, including the knee joint and elbow joint. However, it is very rare to find this disease in hip joints. In fact, only one such case has been reported in Korea. We report here on a rare case of lipoma arborescens in the hip joint along with a review of the relevant literature. Key Words: Lipoma arborescens, Lipoma 서 론 증례보고 수지상지방종 (Lipoma arborescens) 은활막의지방세포가융모형태로이상증식하는양성병변으로활액막을비후시키고이로인하여관절의종창과통증을유발하는질환으로매우드문관절내질환이다. 발생원인으로외상, 감염, 퇴행성변화, 종양, 결핵등이알려져있으나뚜렷한원인은확실하지않다. 대부분이슬관절에서발견되고있으나완관절, 견관절, 족관절등에도발생이보고되고있다 1). 이중고관절에서의수지상지방종은매우드물게보고되고있으며저자들은고관절전구획에발생된수지상지방종을치료하였기에문헌고찰과함께보고하고자한다. Submitted: September 19, st revision: November 15, nd revision: November 29, 2010 Final acceptance: December 3, 2010 ddress reprint request to Jai-Hyung Park, MD Department of Orthopedic Surgery, Myongji Hospital, College of Medicine, Kwandong University, Hwajung-dong, Deokyanggu, Goyang , Korea TEL: FX: wonnypia@hanmail.net Copyright c 2011 by Korea Hip Society 34 세남자환자로내원 10 년전에등산한이후부터시작된우측고관절의동통이활동량이많을경우일년에두세차례증상의호전과악화가반복되어개인병원에서대증적치료를받았었지만증상지속되어내원하였다. 과거력과가족력상특이소견은없으며이학적검사상우측고관절주위의종창은관찰되지않았으나보행시중등도의통증을호소하였고휴식시에통증은경미하였다. 우측고관절의운동범위는굴곡과신전을제외하고는전반적으로감소되어으며특히내회전 10, 외전 30 로건측에비해크게감소된것이관찰되었고내회전시에중등도의통증을호소하였으며 Patrick 검사도양성이었다. 고관절의단순방사선소견에서는우측대퇴골두의상외측과하내측부의연부조직의음영및골의미란을관찰할수있었다 (Fig. 1). 자기공명영상촬영상우측고관절의전반적인지방성신호와동일하거나약간감소를보이는종괴와국소적으로골수의부종, 대퇴골두와비구의부종및낭종이관찰되었고 Tc-99 MDP 골주사검사상에서는우측고관절의흡수가증가된소견이관찰되었다 (Fig. 2, 3). 전신마취하에앙와위자세에서근위대퇴부의전상장골극에서슬개골의외측을향하여 13 cm 의피부절개후에봉공근과대퇴근막장근사이를절개한후대퇴직근을외측으로당겨고관절을노출시키고관절낭을절개하였다. 83

Fig. 3. Images demonstrated uneven mild increased uptake diffuse increased uptake in right hip joint.

4). 병리조직소견상다양한크기의융모상지방성종괴가관찰되었으며각각의지방성종괴는활액성세포들에의해둘러싸인성숙한지방세포로구성되어있었다 (Fig. 5).

수술후 1 개월추시에서내회전및외전등관절운동범위가호전되었으며 6, 12 개월추시에서는관절운동제한은 Fig. 1. (, ) nteroposterior & axial Hip radiograph shows soft tissue density and bony erosion of the right femoral head.

2 Fig. 3. Images demonstrated uneven mild increased uptake diffuse increased uptake in right hip joint. 고관절내에대퇴골두와경부를둘러싸고있는여러개의황색의난원성종물이관찰되었고이는활액막과연결되어있었으며적황색의종물과혼돈되어있는양상이었다. 종괴는골조직으로의침범은관찰되지않았고검체는지방성종괴가융모상으로비후된양상이었다 (Fig. 4). 병리조직소견상다양한크기의융모상지방성종괴가관찰되었으며각각의지방성종괴는활액성세포들에의해둘러싸인성숙한지방세포로구성되어있었다 (Fig. 5). 수술후 5 일째퇴원하였고대퇴직근을부분절제하고다시건봉합을시행했기때문에수술일주일후부터조심스럽게하지직거상운동및부분체중부하를시작하였다. 수술후 1 개월추시에서내회전및외전등관절운동범위가호전되었으며 6, 12 개월추시에서는관절운동제한은 Fig. 1. (, ) nteroposterior & axial Hip radiograph shows soft tissue density and bony erosion of the right femoral head. Fig. 2. (, ) Coronal & xial T1-weighted MR image shows focal marrow edema and proliferative fatty mass of intermediate signal density in distended hip joint space. 84

Jai-Hyung Park et al.: Lipoma rborescens in the Hip Joint - Case Report - 없었고합병증이나재발소견은발견되지않았다. 고 수지상지방종 (Lipoma arborescens) 은관절강내에서성숙한지방세포로인하여활액막지방조직이융모성으로과도하게증가및변형되어생기는매우희귀한질환이다 2).

Microscopic examination of the tumor reveals villous masses composed of mature adipocytes and lined by synovial cells (x40, inlet x200). 찰 이동반되는것이보고되고있다 1,3).

3 Jai-Hyung Park et al.: Lipoma rborescens in the Hip Joint - Case Report - 없었고합병증이나재발소견은발견되지않았다. 고 수지상지방종 (Lipoma arborescens) 은관절강내에서성숙한지방세포로인하여활액막지방조직이융모성으로과도하게증가및변형되어생기는매우희귀한질환이다 2). 대부분의수지상지방종환자에서다른근골격계질환은발견되지않으며슬관절의상낭이호발부위이지만이외에도견관절, 완관절, 주관절에서도보고되었다. 정확한병태생리및원인은밝혀지지않았으나관절염, 반복적인관절외상, 당뇨, 결핵등과관련가능성이알려져있으며슬관절의수지상지방종은 20% 에서베이커씨낭종 Fig. 5. Microscopic examination of the tumor reveals villous masses composed of mature adipocytes and lined by synovial cells (x40, inlet x200). 찰 이동반되는것이보고되고있다 1,3). 호발연령은 40 대에서 60 대이며외측성으로발생하고남자에서더호발하는것으로알려져있으나양측성및극소수에서다발성의발생도보고되고있다 4,5). 임상증상으로이환관절의운동범위감소, 종창, 동통, 삼출액의증가등이있으며이는관절내종양의기계적인효과에의해생긴것으로생각된다. 관절내와건초의지방종은두종류의변형을보이는데하나는관절내와건초에활액막의변화없이고형의단일종물로생기는활액막성지방종과다른하나는활액막성융모가비후되고변형되어생기는수지상지방종이있다. 단일종괴로발생하는활액막성지방종은수부와완관절에주로생기며족부와족관절에는잘발생하지않는다 4). 관절내의수지상지방종은대부분슬관절의상낭에영향을미치며이외에주관절, 견관절, 족관절등에발생한다 1). 후자인수지성지방종은지방세포의비후로인한지방조직의비후외에도염증세포가활액막에다수분포되어있다 4). 이로인하여이차적인재형성과정에의해서퇴행성관절염과만성류마티스성관절염, 외상에의한손상의악화등이일어날수있다 4). 단순방사선검사상에서일정공간의관절내종괴효과에의해관절간격이감소를보일수있고오래지속되면골경화및골극등의관절의퇴행성변화를보일수있다 4). 종괴에크기에따라서는연부조직음영을확인할수도있다. 컴퓨터단층촬영에의해서직접적인융모모양의결절을확인할수도있지만활액막성연골종증등의감별진단에도움이될수있으며수술시에수지상지방종의절제범위를정하는데도움이될수있다. 초음파검사상으로도관절강내에다수의고음영융모돌기를확인할수있으며관절을수기로조작함으로써융모성돌기가흔들리는것을확인할수있다 6). 자기공명영상은이질환진단에필수적인데자기공명영상상에서는 T1 WI, T2 WI 에서모두지방의신호강도와비슷한 Fig. 4. () Intraoperative photograph shows pale, proliferating villi arising from synovial surface. () postoperative photograph showing the villous architecture of the synovium. 85

4 강도와엽상의모양을보이고지방억제 T1 WI 에서피하지방과같이신호가감소된다 7). 또한관절내의삼출액을확인할수있어진단에중요하다. 슬관절의수지상지방종에서는관절경도이용되고있는데관절경상특징적인다수의엽상모양의황색융모성돌기가관찰되며그주변으로활액막의비후등이관찰되어진단과동시에치료가가능하다. 병리조직소견은성숙된지방세포가융모의기질를이루고있으며이를활액막세포가둘러싸고있고융모의활액막은림프구에의한침윤이관찰된다 4). 감별진단으로색소융모결절성활액막염, 활액막성지방종, 활액막성연골종증, 류마토이드관절염, 활액막성혈관종, 아밀로이드성관절병증등이있으며전산화단층촬영, 초음파검사, 자기공명영상검사등으로감별이어렵지는않다 8). 자기공명영상상류마토이드관절염은수지상지방종과다르게 T1 WI 에서는중등도, T2 WI 에서는저신호를보이며색소융모결절성활액막염은헤모시데린의축적에의해서 T1 WI, T2 WI 에서저신호를보이기때문에수지상지방종과감별이가능하다 7). 활액막성연골종증과혈관종은 T1 WI 와 T2 WI 둘다중등도에서저신호를보이므로감별이가능하다 7). 수지상지방종은다발성일경우재발한예가있으나관절내의활액막조직과지방종괴의확실한절제술만으로대부분재발없이치료가가능하다. 슬관절에서는관절경적치료가이용되고있으며재발없이치료결과가좋다고보고되고있다 9). 고관절에서도관절경을이용하여절제술을시행하기도한다. 이는주변근육과조직에큰손상없이절제가능하므로개방적절제술보다회복및재활이빠를것으로생각되며앞으로고관절에서의수지상지방종에서관절경절제술이치료에있어우선선택되어져야할것으로생각된다. 다만본례와같이고관절전구획에걸쳐형성된수지상지방종은관절경으로종괴의확실한절제연을갖게절제하는것이쉽지않으므로고관절전구획에걸친종괴및활액막의확실한절제를위해서는개방적절제술이필요할것으로생각된다. 본례에서고관절의운동범위감소및운동시통증은수지상지방종이고관절의전구획에걸쳐기계적인효과에의한관절낭의자극되었을것으로생각된다. 수년간의간헐적인통증은관절과사용에의한활액막염이동반되었던것으로생각되며수술시에서도수지상지방종주위의중등도의활액막염소견이관찰되었었다. 진단에있어 Tc-99 MDP 골주사검사에서우측고관절부위의흡수증가가있었지만화농성관절염, 무혈성괴사증등과감별이 필요하므로단독검사로는크게유용성이없을것으로생각되며오히려초음파검사가수지상지방종의고관절내위치와범위를파악하는데도움이될것이다. 대부분의수지상지방종은종창의증상을동반한다고하지만이는피부와인접한슬관절, 주관절등과같은경우에해당할것으로생각되며고관절과같이신체깊이있는관절에서는신체검진상종창의증상이쉽게보이지않으므로신체검진시주위를필요할것으로생각된다. 결 고관절전구획에발생한수지상지방종 1 예에대해자기공명검사로진단하여개방적절제술로치료하였으며 1 년간의추시에서재발및합병증의발생이없었다. 앞으로고관절에서의이질환은종괴의크기가크지않고제한된범위에있다면관절경적절제술이종괴의제거와술후관절기능의회복에도도움이될것으로생각된다. 저자들은고관절전구획에걸쳐발생한수지상지방종 1 예를경험하고치료하였기에문헌고찰과함께보고하는바이다. 론 REFERENCES 01. ryan RS, Dahlin DC, Sullivan CR. Lipoma of tendon sheath. J one Joint Surg m. 1956;38-: Edamitsu S, Mizuta H, Kubota K, Matsukawa, Takagi K. Lipoma arborescens with hemarthrosis of the knee. case report. cta Orthop Scand. 1993;64: rmstrong SJ, Watt I. Lipoma arborescens of the knee. r J Radiol. 1989;62: Hallel T, Lew S, ansal M. Villous lipomatous proliferation of the synovial menbrane (lipoma arborescens). J one Joint Surg m. 1988;70: Sheldon PJ, Forrester DM, Learch TJ. Imaging of intraarticular masses. Radiographics. 2005;25: Learch TJ, raaton M. Lipoma arborescens: highresolusion ultrasonographic findings. J Ultrasound Med. 2000;19: Kloen P, Keel S, Chandler HP, Geiger RH, Zarins, Rosenberg E. Lipoma arborescens of the knee. J one Joint Surg r. 1998;80: Martinez D, Millner P, Coral, Newman RJ, Hardy GJ, utt WP. Case report 745: Synovial lipoma arborescens. Skeletal Radiol. 1992;21: Sola J, Wright RW. rthroscopic treatment for lipoma arborescens of the knee: a case report. J one Joint Surg m. 1998;80:

5 Jai-Hyung Park et al.: Lipoma rborescens in the Hip Joint - Case Report - 국문초록 고관절의수지상지방종 - 1 예보고 - 박재형 김형수 정수태 유정현 김주학 차승도 정진하 관동대학교의과대학명지병원정형외과학교실 수지상지방종 (Lipoma arborescens) 은관절낭에서지방세포가활액막의융모상지방종을이루며이형증식하는질환으로매우희귀한질환이다. 비후된활액막성지방종이관절의움직임에따라자극되어관절의동통, 삼출, 관절운동의제한및종창등의증상을일으키며발생원인으로는외상, 류마티스관절염, 염증, 퇴행성변화등이알려져있으나명확한원인은아직판명되지않았다. 대부분의수지상지방종은슬관절및주관절등여러관절에서의발생이보고되고있으나고관절에서의수지상지방종은드물어국내에서는 1예정도만보고되고있다. 저자들은고관절에발생한수지상지방종환자 1예를치료하였기에문헌고찰과함께보고한다. 색인단어 : 수지상지방종, 고관절 87

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( ) Jkra076.hwp Vol. 11, No. 4, December, 2004 Objective: Ultrasonography (USG) of joints has a unique position for the diagnosis of joint diseases. Bone surface, cartilage, periarticular soft tissue and their pathologic