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1 대한골관절종양학회지 J. of Korean Bone & Joint Tumor Soc. Volume 12, Number 2, December, 2006 비골에발생한원발성골종양 연세대학교의과대학정형외과학교실 박장원 이승환 신규호 목적 : 본연구에서는골종양의발생부위중비교적드문부위인비골에서원발하는골종양의종류와그치료후경과에대하여연구하고자하였다. 대상및방법 : 1993년 1월 1일부터 2005년 12 월 31 일까지하지의골종양을진단받고수술적치료를받은환자중비골의원발성골종양환자에대하여조사하여그임상적결과를조사하였다. 결과 : 상기기간에비골의원발성골종양을진단받고수술적치료를받은환자는총 17 명 ( 남자 10 명, 여자 7명 ) 이었으며평균연령은 22 세, 평균추시는 18 개월이었다. 병리조직검사결과골연골종이 8예로가장흔한진단이었으며그외에골육종, 섬유성골이형성증이 2예씩진단되었다. 악성골종양의빈도는 10.5% 였다. 그부위별로는근위부, 간부, 원위부가각각 12 예, 4예, 1예였다. 최종추시시의임상적인만족도는우수혹은양호의결과가 14 명으로나타났다. 결론 : 비골은골이식시에가장흔한공여부중의하나로병변이경골까지침범되거나원위부에위치하지지않는이상절제후에재건술이특별히필요하지않기때문에적극적인수술적인치료가가능하며특히간부의경우에는충분한절제경계를확보하기용이하다. 그러나전체적으로그빈도가낮은편이며증상이명확하지않고병변이진행될경우에절단술이필요할수있어진단에주의를요한다. 색인단어 : 원발성골종양, 비골 서론원발성골종양의호발부위는그조직학적진단에따라서차이가나지만하지에서는슬관절, 골에대해서는대퇴골에서가장많이발생하는것으로알려 져있다. 전이성골종양을제외하고가장발생빈도가높은악성골종양인골육종과양성골종양중거대세포종에서모두대퇴골원위부와경골근위부가가장빈도가높은호발부위이다. 비골은전체골종양중에서원발성골종양이발생하는빈도가낮은 통신저자 : 신규호서울특별시서대문구신촌동 134 연세대학교의과대학정형외과학교실 Tel: 02) , Fax: 02) , qshin@yumc.yonsei.ac.kr * 본논문의요지는 2006 년도대한정형외과학회추계학술대회에서발표되었음. 112

2 박장원외 : 비골에발생한원발성골종양 골에속하며비골에서발생한골종양에대해서는대개골육종, 거대세포종, 유잉씨육종이흔한것으로알려져있으나 2,8,9,10,12) 우리나라에서는그종류나분포가잘알려져있지않다. 비골내의위치에있어서는근위부에서가장호발하며그다음간부, 원위부순인것으로알려져있다. 또한가장흔한편인근위부에서는비골자체의크기나기능적인중요성에비해서비골에인접한총비골신경이나전경골동맥같은구조가있기때문에완전한절제가쉽지않은경우가빈번하다 7-10). 본연구에서는비골에발생한원발성골종양의종류와그분포, 치료후의경과에대한분석을시행하고자하였다. 연구대상및방법 1. 연구대상 평균연령은 22 세, 평균추시는 18 개월이었다. 2. 연구방법각각의환자에대해서방사선사진으로병변을확인한후에는 MRI를시행하였으며 MRI 소견에따라서전신골주사검사, 흉부방사선및 CT를추가로시행하였다. 비골종양의수술적절제방법에대해서는 Malawer에의한절제방법 8) 으로조사하였다. 악성종양의경우필요에따라서술전및술후항암화학요법을추가하였다. 조직학적분류를조사하고, 비골에서의위치별분포는 3등분하여근위부, 간부및원위부로나누어조사하였다. 환자의진단당시의주요호소증상에대해서조사하였으며수술후의임상적결과는 Enneking score system 4) 을통하여조사하였다. 1993년 1월 1일부터 2005년 12 월 31 일까지세브란스병원에서하지의원발성골종양으로수술적인치료를받고병리조직학적으로확진이이루어진환자를대상으로후향적인연구를시행하였다. 상기기간중에하지의원발성골종양으로수술적치료를받은환자는총 409 명이었으며이중비골의원발성골종양으로수술적인치료를받은환자는 17 명이었다. 전체환자중남녀성비는 10:7 이었으며 결과수술후에조직학적으로확진된진단은골연골종이 8예로가장많았고골육종이 2예, 섬유성이형성증이 2예에서나타났다 (Table 1). 9명의환자가통증을주소로내원하였고 7명은촉지되는종괴를주소로내원하였다 (Table 2). 비골의부위별로분류하였을때에는근위부에서 Table 1. Pathologic diagnosis Pathologic diagnosis number of cases percentage (%) Osteochondroma 8 46 Osteosarcoma 2 12 Fibrous dysplasia 2 12 Giant cell tumor 1 16 Enchondroma 1 16 Hemangioma 1 16 Intraosseous ganglion 1 16 Ossifying fibroma 1 16 Table 2. Chief complains Chief complains number of cases percentage (%) Pain 9 53 Palpable mass 7 41 No subjective symptoms

3 대한골관절종양학회지 : 제 12 권제 2 호 2006 년 12 예로가장많이발생하였고간부 4예, 원위부 1예로나타났다. 수술적치료의방법을조사하여표시하였는데 marginal resection이 7예로가장많았다. En bloc resection의 type 은 K. Erler 등 5) 에의해제시된 modified Malawer s resection을기준으로분류하였다 (Table 3). 2예의골육종환자중 1예에서는술전과술후항암화학요법을각각 3회씩실시하였고수술은비골신경을보존하면서 modified type II Malawer s resection을시행하였으며진단후 5년 2개월째까지생존상태 (Fig. 1) 이며 1예에서는진단후에술전항암화학요법을시행후 intercalary resection을시행하였으나수술후원격전이가있어진단 15 개월째사망하였다 (Table 4). 수술후의임상적결과는 Enneking의기능평가항목에의하여운동, 통증, 안정성, 변형, 힘, 기능적활동, 환자의만족도를측정하였고 17 예중 9예에서우수, 5예에서양호, 1예에서보통, 2예에서불량으로나타났다. 고찰비골은원발성골종양이나전이성골종양모두에서드물게발생하는해부학적위치다. 비골내에서는근위부가가장흔하게종양이발생하는것으로 알려져있으며그다음으로간부, 원위부순이다. 비골내에서는골육종, 유잉씨육종, 거대세포종이가장흔한것으로알려져있다 12). 비골근위부의악성육종의절제를위해서는비골의근위부와근위경비관절, 총비골신경, 그리고근육군을포함해서절제를하여야하며이것을 type II resection으로부른다. 이것은총비골신경을희생해야하기때문에족하수가발생하게된다. Type I resection의경우에는비골근위부를절제하면서근육층과비골신경을남겨두게된다. Erler K 등이제시한 modified type II resection 경우에서는 type II resection을시행하면서비골신경을보존하게되는데본연구에서는 modified type II 술식으로시행하였고술후에족하수발생없이우수한임상결과를얻을수있었다 3-6). 이것은주로는 benign-aggressive, lowgrade malignant tumor, metastatic lesion에주로적합하다 7,8). 원위비골의원발성육종의경우에주변연부조직이거의없고주위의근육이나인대를잘침범하는속성때문에절단술이필요한경우가많다. 비골의근위부골종양에서 type II resection을시행할경우외측측부인대의부착부가같이절제되기때문에슬관절에내반의불안정성이발생하게된다. Bickels J 등 1) 은외측측부인대를근위경골의골간단에 stapling을통해고정하여우수한결과를보고한바있다. 본연구에서는외측 Table 3. Operative methods Operative methods number of cases percentage (%) Curettage 4 24 Curettage & cementation 3 18 Marginal excision 7 40 Intercalary resection 2 12 Type II resection* 1 16 * modified type II Malawer s resection described by Erler et al. Table 4. Malignant fibular tumor patients Age Gender Diagnosis Site Stage margins follow functional oncological up month outcome outcome 15 F osteosarcoma proximal IIB wide 62 Excellent CDF 29 M osteosarcoma middle III wide 15 Poor DOD CDF continuously disease free. DOD died of disease. 114

4 박장원외 : 비골에발생한원발성골종양 측부인대의원위이동하여근위경골의피질골에봉합하여양호한결과를얻을수있었다. 비골의간부의절제술후에는하지의역학적안정성에별다른영향을미치지않기때문에별다른재건술은필요하지않다. 원위부의골종양의경우에는족근관절의외과가족근관절복합체의일부이기때문에재건술이필요하다 11). 비골은그자체의해부학적특성으로도피질골이얇고다양한근육이연결되어있기때문에다른부 위에비해서 extra-compartmental로더일찍진행을하게된다. 따라서적절한진단에이은수술적인치료가필요하며수술시에 intra-compartmental인경우에는사지구제의가능성이높다. 적절한시기에비골절제술이행해질경우에는사지구제가비교적용이하며수술후재건술이필요없는장점이있다 2,6,8). 본연구에서비골의골종양은전체하지골종양중 4.2% 로조사되었는데이것은 Unni 12) 에의해 A Fig year old female with osteosarcoma of proximal fibula. (A) pre-operative x ray (B) pre-operative MRI (C) photograph of surgical specimen, peroneal nerve was preserved. (D) final x ray at post op 62 months, the patient shows no evidence of recurrence and has excellent functional outcome. B C D 115

5 대한골관절종양학회지 : 제 12 권제 2 호 2006 년 보고된 2.5% 보다높은것이지만비골의골종양자체가드문것이고전체환자수에있어서본연구에서의전체환자군이작았기때문에정확한비교는어렵다고하겠다. 본연구에서는비골의원발성골종양을전체적으로조사하였는데추후에는양성골종양과악성골종양을구분하고더많은증례에대한조사가필요하다고하겠다. 결 비골은골이식시에가장흔한공여부중의하나로병변이경골까지침범되거나원위부에위치하지지않는이상절제후에재건술이특별히필요하지않기때문에적극적인수술적인치료가가능하며특히간부의경우에는충분한절제경계를확보하기용이하다. 그러나전체적으로그빈도가낮은편이며증상이명확하지않고병변이진행될경우에절단술이필요할수있어진단에주의를요한다. 론 REFERENCES 01) Bickels J, Kollender Y, Pritsch T, Meller I, Malawer MM.: Knee stability after resection of the proximal fibula. Clin Orthop Relat Res, 24, Aug, ) Capanna R, van Horn JR, Biagini R, Ruggieri R, Betelli G, and Campanacci M: Reconstruction after resection of the distal fibulae for bone tumors. Acta Orthop Scand, 57(4): , ) Draganich LF, Nicholas RW, Shuster JK, Sathy MR, Chang AF, and Simon MA: The effects of resection of the proximal part of the fibula on stability of the knee and on gait. J Bone Joint Surg Am, 73: , ) Enneking WF, Dunham W, Gebherdt MC, Malawer N, and Pritchard DJ: A system for the functional evaluation of reconstructive procedures after surgical treatment of tumours of musculoskeletal system. Clin Orthop, 286: , ) Erler K, Demiralp BM, Ozdemir T, and Basbozkurt M: Treatment of proximal fibula tumors with en bloc resection. The Knee, 11: , ) Farooque M, Biyani A, Adhikari A: Giant cell tumors of the proximal fibula. J Bone Joint Surg, ;72-B(4): , ) Lushiku HB, Gebhart M: Osteosarcoma of the proximal fibula:report of 3 cases. Acta Chir Belg;97(5): ) Malawer MM: Surgical management of aggressive and malignant tumors of the proximal fibula. Clin Orthop;186: , ) Marcove RC, Jensen MJ: Radical resection for osteogenic sarcoma of fibula with preservation of the limb. Clin Orthop, 125: , ) Natarajan M, Paraskumar M, Rajkumar G, Sivaseelam A, and Natarajan S: Limb salvage in aggressive and malignant tumours of the fibula. International Orthopaedics, 28: , ) Norman-Taylor FH, Sweetnam DI, Fixsen JA: Distal fibulectomy for Ewing s sarcoma. J Bone Joint Surg, 76: , ) Unni KK.: Dahlin s bone tumors: general aspects and data on cases, 19. Illinois: Lippincott- Raven; p. 9-10, 172,264,

6 박장원외 : 비골에발생한원발성골종양 Abstract Primary bone tumors of fibula Jang-won Park, M.D., Seung-Hwan Lee M.D., Kyoo-Ho Shin, M.D. Department of Orthopedic Surgery, Yonsei University, College of Medicine, Seoul, Korea Purpose: The purpose of this study was to investigate on primary bone tumors of fibula. Materials and Methods: Patients who received operative treatment from January 1993 to December 2005 for primary bone tumors of fibula were analyzed for clinical outcomes. Results: There were 17 patients(10 males, 7 females) who were diagnosed for primary bone tumors of fibula. Osteochondroma was the most common diagnosis and others were osteosarcoma, fibrous dysplasia. The malignant bone tumors were 10.5% of all fibula tumors. There were 12 cases on proximal, 4 cases on mid shaft, and 1 cases on distal part of fibula. At the final follow up, 14 patients reported patient satisfaction of either excellent or good. Conclusion: Bone tumor of fibula doesn t need reconstruction after the resection unless it invades tibia or its site distal, which enables easy surgical resection. However, generally it is rare and accompany with vague symptoms, so careful approach to diagnosis is necessary because late diagnosis can mean the needs for amputation. Key Words: Fibula, Primary bone tumor Address reprint requests to Kyoo-Ho Shin, M.D. Department of Orthopedic Surgery, Yonsei University, College of Medicine, 134 Shinchon-dong, Seodaemun-gu, Seoul, , Korea TEL: , Fax: , qshin@yumc.yonsei.ac.kr 117

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