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대한정형외과학회지 : 제 42 권제 4 호 2007 J Korean Orthop Assoc 2007; 42: 426-432 장골의양성골종양절제술후골결손에대한 Ilizarov 외고정기를이용한치료 정성택ㆍ조성범ㆍ이진호 전남대학교의과대학정형외과학교실 Treatment of Bone Loss using Ilizarov Fixation after Resection of Benign Tumor in the Long Bone Sung Taek Jung, M.D., Seong Beom Cho, M.D., and Jin Ho Lee, M.D. Department of Orthopaedic Surgery, Chonnam National University Hospital, Gwangju, Korea Purpose: This study analyzed the results of treatment of bone loss using Ilizarov fixation after resection of benign tumor in the long bone radiologically and clinically. Materials and Methods: Initial diagnoses were 5 osteofibrous dysplasia, 1 aneurysmal bone cyst and 1 giant cell tumor. Three indices were used to evaluate the results; percentage transport, healing index and percentage increase. The function of the affected limb was assessed according to Ennecking rating. And we assessed the radiological and functional results according to A.S.A.M.I.'s classification. Results: Percentage transport averaged 75% (range: 29-144), healing index averaged 64.5 day/cm (range: 34.8-108.6) and the percentage increase averaged 13% (range: 11-20). In Enneking rating, there were 6 cases that were classified as being better than good. In A.S.A.M.I. s classification, there were 6 good cases, and 1 fair case each in the bone result and functional result, respectively. Conclusion: Ilizarov technique is a reconstruction method using living bone. It is safe and effective for the treatment of bone loss after the resection of a benign tumor in the long bone. Key Words: Benign bone tumor, Bone defect, Ilizarov 서론경골, 대퇴골등의장골 (long bone) 에발생하는골섬유성이형성증, 동맥류성골낭종, 거대세포종등은양성의성격을갖는골종양이다. 더욱이골섬유성이형성증은경과관찰만으로도더이상의진행없이자연치유를보이는경우도있어보존적인치료가원칙이며수술이필요하더라도가능하면골격성장이멈추어질환의활성이감소하는 15세이후까지수술을연기하는것이바람직하다. 그러나상기양성골종양은재발이많으며과도한골파괴나골취약성을가져와병적골절의위험성을갖는다 16). 이처럼병변이광범위하여골이취약해진경우, 급격한진행을보이는경우, 병적골절이반복되거나심한 만곡이나가관절증을형성한경우등에서는수술적치료가불가피하다. 이런경우그치료방법으로는국소소파술부터골병변부위전범위골절제술 (en bloc resection) 까지다양하며일반적인치료로는주로소파술 (curettage) 이이용되어왔다. 하지만이는재발및장골의각변형등의많은문제점을보이고있어병변부위의광범위골절제술이필요한경우가많으며이후발생한골결손에대해골이식술및내고정술, 전기자극, 생화학물질이식술, 외고정기를이용한치료방법등이이용되고있다 14,16). 이에본연구에서는양성골종양치료후발생한골결손의해결방법으로아직많은보고가없는피질절골술후신연골생성술로써 Ilizarov 외고정기 통신저자 : 조성범광주시동구학 1 동 8 번지전남대학교의과대학정형외과학교실 TEL: 062-227-1640 ㆍ FAX: 062-225-7794 E-mail: madkid@dreamwiz.com Address reprint requests to Seong Beom Cho, M.D. Department of Orthopaedic Surgery, Chonnam National University Hospital, 8, Hak 1-dong, Dong-gu, Gwangju 501-757, Korea Tel: +82.62-227-1640, Fax: +82.62-225-7794 E-mail: madkid@dreamwiz.com 426

장골의양성골종양절제술후골결손에대한 Ilizarov 외고정기를이용한치료 427 를이용한치료의임상적, 방사선적결과및여러가지합병증등을알아보고자한다. 대상및방법본원에서 1997년 10월부터 2005년 5월까지장골에발생한양성종양에대해광범위골병변절제술후 Ilizarov 외고정기를이용한신연골생성술로치료받고술후최소 1년이상추시가가능했던 7명의환자를대상으로하였다. 총 7명중남자 6명, 여자 1명이었으며수술당시의평균연령은 13.9 (4.8-36.8) 세였다. 원인질환으로는골섬유성이형성증이총 5예로가장많았으며동맥류성골낭종이 1예, 거대세포종이 1예였다. 본연구에서는 Ilizarov 기구를이용하여치료한장골의골결손치료결과의평가를위해방사선적으로골편이동비율 (percentage transport: 골결손크기 / 이동골편크기 100) 과치유지수 (healing index: 기구장치부터제거까지의개월수 / 연장된길이 cm) 그리고술전길이에대한연장율 (percentage increase: 신연된골길이 / 술전원골길이 ) 를분석하였으며 10,17) 임상적으로 Enneking에따른환측의기능적면을평가하였다 5). 또한 A.S.A.M.I. group 의분류에따라골적결과와기능적결과를평가하였다 11-13). 골적결과는골유합, 감염여부, 변형, 하지부동유무등을기준하여우수, 양호, 보통, 불량으로구분하여평가하였다. 우수는골유합을얻고감염소실, 6도이내의변형, 2.5 cm 이하의하지부동이있을때로하였고양호는골유합이되었고나머지기준에서두개를만족할때이며, 보통은골유합을얻고나머지기준중한개를만족할때, 마지막으로골유합을얻지못한경우를불량으로분류하였다. 기능적결과는다섯가지를기준으로하였는데첫째동통의유무, 둘째관절구축유무, 셋째연부조직위축유무, 넷째보행시파행유무, 다섯째일상생활수행능력정도를평가하였다. 기능적결과상우수는환자가어려움없이일상생활을수행할수있고파행, 관절구축, 연부조직위축그리고동통이없을때로하였다. 양호는일상생활을어려움없이수행할수있으나다른기준중한가지나혹은두가지가있을때로하였고, 보통은일상생활에어려움은없으나나머지기준중세가지혹은네가지전부있거나또는절단을하였을경우이며, 불량은어떤경우라도일상생활에심각한장애가있는경우로하였다 5,13). 종양제거술후발생한골결손의길이는평균 43.9 (28-74) mm 였다. 수술방법으로는수술전환자의예상골결손부위에남아있는근위부및원위부각각두부위에서고정될수있도록미리적절한크기의 Ilizarov 기구를조립하였으며, 장골의정렬상태를방사선투시하여확인후상하관절에평행하고근위부와원위부에평행하게 K강선으로고정후강선긴장기구 (dynamometric wire tensioner) 를이용하여 110-130 kg의긴장을가하여 Ilizarov 링을단단히고정하였다. 수술후평균 8.6 (6-11) 일의절골치유기간의휴지기를갖고하루에 1 mm 또는 0.5 mm 를 4회에나누어신장시켰다. 피질절골술후근위부로이동시킨경우 1예, 원위부로이동시킨경우가 6예였다. 골편신연중에도가능하면환자에게관절운동과체중부하운동을시켰다. 주기적으로방사선촬영을하면서골형성정도에따라신장속도를조절하였고 Ilizarov 외고정기를평균 243 (123-568) 일유지하였으며계획된길이의골연장을얻은후에는목발보행과더불어적극적인체중부하보행을더욱권장하여신연간격 (distraction gap) 의고정화 (consolidation), 피질골화 (corticalization) 와골수강의재형성이어느정도진행되면 Ilizarov 기구를제거하였고, 필요할경우피질골화가완전히보일때까지석고붕대를고정하거나보조기를착용하여골절이발생하지않도록하였다. 결과골연장의목표길이는환자의나이, 골연령, 환측및건측골의길이등을고려하여결정하였으며최소 3.0 cm에서최대 7.2 cm로평균 4.4 cm을연장하였다 (Fig. 1-7). 골결손의골재건방법은신연신생골형성분류 (classification of reconstruction by distraction osteogenesis) 에따라골이동 (bone transport) 에의한골간골형성 (diaphyseal reconstruction) 5예, 골단축신연 (shortening distraction) 에의한골간단골형성 (metaphyseal reconstruction) 2예로분류되었다 5,7,17). 평균골유합기간은 8.0 (4.1-18.9) 개월이었다. 치유지수 (Healing index) 는평균 64.5 (34.8-108.6) 일 /cm였고골편이동비율은평균 75 (29-144)% 였다. 또한술전길이에대한연장율은평균 13 (11-20)% 로술전연장목표에도달하였다 10). 또한환측의기능적평가인 En-

428 정성택ㆍ조성범ㆍ이진호 Fig. 1. (A) Radiographs of a 12-year-old male patient show an osteolytic lesion of left tibial shaft. (B) Coronal and sagittal images of computed tomograph show the same lesions. (C) We performed resection of the lesion and applied Ilizarov fixator. Pathologic diagnosis was osteofibrous dysplasia. (D) Radiographs of at 5 months postoperative show nonunion of docking site. (E) We performed a fibular strut bone graft at docking site. (F) Radiographs from the last follow-up show a healed bone lesion. Fig. 2. (A) Radiographs of a 14-year-old male patient show an osteolytic lesion of the shaft of the right tibia. (B) We resected the lesion and inserted calcium sulfate into the bone defect and applied Ilizarov fixator. We simutaneously performed corticotomy at the proximal metaphysis. Radiographs at 1 month postoperative showed that the proximal fragment was transported distally. (C) Radiographs from the last follow-up show healed bone lesion. Fig. 3. (A) Radiographs of a 15-year-old female patient show a pathologic fracture of the right distal femur. (B) We performed resecting of the lesion and applied Ilizarov fixator. (C) Radiographs at 2 months postoperative show lengthening of the corticotomy site. (D) Radiographs from the last follow-up show a healed bone lesion.

장골의양성골종양절제술후골결손에대한 Ilizarov 외고정기를이용한치료 429 Fig. 4. (A) The image shows the pathologic fracture due to fibrous dysplasia of the diaphysis. (B) We resected the lesion and applied Ilizarov fixator. (C) We performed gradual lengthening at the proximal metaphysis. (D) Radiographs at the last follow-up show a healed bone lesion. Fig. 5. (A) Radiographs of a 4-year-old male patient show an osteolytic lesion of the right tibial diaphysis. (B) We performed resecting of the lesion and applied Ilizarov fixator. (C) Radiographs at 12 months postoperative show lengthening of the corticotomy site. (D) Radiographs at the last follow-up show a healed bone lesion. Fig. 6. (A) Radiographs of a 36-year-old male patient show an osteolytic lesion of the left dital femur. (B) We resected the lesion and applied Ilizarov fixator. (C) Radiographs at 9 months postoperative show lengthening of the corticotomy site. (D) Radiographs at 2 years postoperative show a healed bone lesion. neking 에따른분석상탁월 1예, 우수 5예, 양호 1예를보였다 5) (Table 1). A.S.A.M.I. 의분류상골적결과는우수 6예, 양호 1예였고보통및불량은없었으며기능적결과또한우수 6예, 양호 1예로보통이나불량은없었다 (Table 1). 전예에서장골계측촬영을시행하였으며 2 cm 이상의하지부동을보이는경우는한예에서도발생 하지않았다. 총 7예중 2예에서접촉부위 (docking site) 의불유합이발생하여비골이식과함께 4일간의매일 1 mm씩압박단축후신연하여골유합및원하는길이의골신연을가졌다 (Fig. 1). 또한 1예에서 K강선삽입부위에서염증소견을보였으나일주일간의경구항생제투여후염증소견은소실되었다. 술후골절, 종양의재발,

430 정성택ㆍ조성범ㆍ이진호 Fig. 7. (A) Radiographs of a 10-year-old male patient show an osteolytic lesion of the shaft of the right tibia. (B) We resected the lesion and inserted calcium sulfate and a fibular strut bone graft at the bone defect. (C) The image shows union processing at the graft site. (D) Radiographs at the last follow-up show a healed bone lesion. Table 1. Summary of Treated Patients No.* Age Sex Diagnosis Site A.S.A.M.I. Defect Enneking's classification Method size (cm) rating Bone Function 1 10.6 M OFD Proximal tibia 3.10 Diaphyseal Fair Good Good 2 14.1 M OFD Tibial shaft 7.40 Diaphyseal Good Good Fair 3 12.2 M OFD Proximal tibia 4.90 Diaphyseal Good Fair Good 4 15.8 F ABC Distal femur 4.50 Diaphyseal Excellent Good Good 5 4.8 M OFD Tibial shaft 2.80 Metaphyseal Good Good Good 6 7.2 M OFD Tibial shaft 3.80 Diaphyseal Good Good Good 7 36.8 M GCT Distal femur 4.23 Metaphyseal Good Good Good *No., Number; M, Male; F, Female; OFD, Osteofibrous dysplasia; ABC, Aneurysmal bone tumor; GCT, Giant cell tumor. 족근관절의첨족, 재형성골의조기경화 (consolidation) 등의다른합병증은보이지않았다. 고찰 1905년 Codivilla 가대퇴골절골술후종골견인방법을제시함으로써수술적방법에의한하지연장술이시작된이래 Putti, Abbott, Anderson 등이여러방법을고안하였으나, 대부분기술상의어려움과많은합병증등으로좋은성과를얻지못하였다 1,2,4). 1960년대새로운방법들이나타나게되었는데, Vassershtein 은신연간격을동종의피질골이식을통하여채워지게하였고 1971년 Wagner 는견고한골신연장치를이용하여골연장을얻은후신연간격에장골이식을하고내고정하는방법을고안하여좋은결과를보고하였다 18,19). Ilizarov 는신연골생성술 (distraction osteogenesis) 이라는개념을도입하였는데, 이는장골방향으로지속적인골형성이일어나도록유도하는방법으로그는독특한형태의 골외고정장치를고안하여하지뿐만아니라상지연장에도이용하였고그밖에복잡한변형의고정, 골절의치료에까지이용하는등정형외과영역에서치료가용이하지않았던여러질환의치료에좋은결과를보고하였다 3,6-8,14). 하지만근골격계종양의치료에있어서의그이용은아직많은보고가없는실정이다 9,16,17). 양성골종양에대한일반적인치료로지금까지는소파술을시행하고있으며이는수술후국소적재발, 골용해에의한골절위험성, 각변형등의문제점을야기할수있다 9,15-17). Ilizarov 기구를이용한신연신생골형성방법은오랜기간외고정기를유지해야하고이로인해핀삽입부의감염, K-강선의파괴, 환자의심적부담감등의문제점을가지고있으나 15,16) 자가생골을이용한신연신생골형성술을이용함으로써궁극적으로안정성및물리적안정성을제공하며골대체물이나골이식술으로인한합병증을줄일수있음을알수있었다 9). 또한골신연부분의

장골의양성골종양절제술후골결손에대한 Ilizarov 외고정기를이용한치료 431 말초신경, 혈관, 근육피부등의신연도점진적으로얻을수있어이들의문제점도해소가능하였다 14). Tsuchiya 등의보고에의하면 15예 (10예의골육종및 5예의거대세포종 ) 의골종양환자에서신연신생골형성법을이용한결과장기생존율및합병증에관해서만족할만한결과를얻었다 17). 그들은또한 5예의거대세포종환자에서 Ilizarov 방법을이용하여평균 5.7 cm의결손부위에대해 233일의외고정을통하여평균 40.5일 /cm의치유지수를보였고합병증으로는 1예의접촉부위의골절, 족근관절의첨족, 재형성된골의경화, 비골두의아탈구의합병증을보였다. 본연구에서도이들과비슷한좋은결과를얻을수있었다. 이처럼 Ilizarov 외고정기를이용한골신연신생골형성술은양성골병변의치료에효과적으로사용해볼수있는한방법이다. 골섬유성이형성증, 동맥류성골낭종, 거대세포종과같은양성종양성골병변이있는경우간단한소파술이나제거술로는재발, 각변형등의단점이있으나본방법은건강한피부및조직의결손없이치료가가능하며효과적인골치유가기대되기때문이다. 또한골신연신생골형성술은광범위제거술후에발생한과도한골결손을채울수있으며변형의교정또한가능한방법이었다 9). 결론장골의양성골종양제거술후골결손에대한 Ilizarov 외고정기를이용한골신연신생골형성술은자가생골을이용한재건이라는장점을가지며안전하고효과적인방법으로양성골병변의치료에효과적으로사용해볼수있는한방법임을알수있었다. 참고문헌 1. Abbott LC: The operative lengthening of the tibia and fibula. J Bone Joint Surg, 9: 128, 1927. 2. Anderson WV: Leg-lengthening, in proceedings of the British Orthopedic Association. J Bone Joint Surg Br, 34: 150, 1952. 3. Cierny G 3rd, Zorn KE: Segmental tibial defects. Comparing conventional and Ilizarov methodologies. Clin Orthop Relat Res, 301: 118-123, 1994. 4. Codivillar A: On the means of lengthening, in the lower limbs, the muscles and tissues which are shortened through deformity. 1904. Clin Orthop Relat Res, 301: 4-9, 1994. 5. Enneking WF: A system for functional evaluation of the surgical management of musculoskeletal tumors. In: Ennecking WF. ed. Limb salvage in musculoskeletal oncology. New York: Churchill Livingstone, 5-16, 1987. 6. Hahn SB, Park EH, Park HW, Kim HW, Kim BH: Comparison of treating tibial nonunion with bone and soft tissue defect: Ilizarov only versus free flap and Ilizarov. J Korean Orthop Assoc, 37: 754-758, 2002. 7. Ilizarov GA: The tension-stress effect on the genesis and growth of tissue: Part II. The influence of the rate and frequency of distraction. Clin Orthop Relat Res, 239: 263-285, 1989. 8. Ilizarov GA: Clinical application of tension-stress effect for limb lengthening. Clin Orthop Relat Res, 250: 8-26, 1990. 9. Karita M, Tsuchiya H, Sakurakichi K, Tomita K: Osteofibrous dysplasia treated with distraction osteogenesis: a report of two cases. J Orthop Sci, 9: 516-520, 2004. 10. Lee DY, Choi IH, Chung CY, Lee KH, Kim HS: Our experience on leg lengthening by the Ilizarov technique. -A preliminary report on the first seventeen patients-. J Korean Orthop Assoc, 25: 1611-1623, 1990. 11. Paley D, Catagni MA, Argnani F, Villa A, Benedetti GB, Cattaneo R: Ilizarov treatment of tibial nonunions with bone loss. Clin Orthop Relat Res, 241: 146-165, 1989. 12. Paley D, Fleming B, Pope M, Kristansen T: A comparative study of fracture gap motion and shear in external fixation. Presented at the Conference on Recent Advances in External Fixation. Riva Del Garda, 28-30, 1986. 13. Song HR, Cho SH, Koo KH, et al: Treatment of tibial bone defect by internal bone transport using Ilizarov method. J Korean Orthop Assoc, 31: 1071-1079, 1996. 14. Subasi M, Kapukaya A: Distraction osteogenesis for treatment of bone loss in the lower extremity. J Orthop Sci, 8: 882-884, 2003. 15. Tsuchiya H, Tomita K: Distraction osteogenesis for treatment of bone loss in the lower extremity. J Orthop Sci, 8: 116-124, 2003. 16. Tsuchiya H, Tomita K, Shinokawa Y, Minematsu K, Katsuo S, Taki J: The Ilizarov method in the management

432 정성택ㆍ조성범ㆍ이진호 of giant-cell tumours of the proximal tibia. J Bone Joint Surg Br, 78: 264-269, 1996. 17. Tsuchiya H, Tomita K, Minematsu K, Mori Y, Asada N, Kitano S: Limb salvage using distraction osteogensis. A classification of the technique. J Bone Joint Surg Br, 79: 403-411, 1997. 18. Vassershteĭn IS: Distraction-compression method of elongation of the lower extremity with use of bone tubular homotransplant. Orthop Travmatol Protez, 29: 44-48, 1968. 19. Wagner H: Operative lengthening of the femur. Clin Orthop Relat Res, 136: 125-142, 1978. = 국문초록 = 목적 : 양성골종양에대해절제술후발생한골결손의치료방법으로 Ilizarov 외고정기를이용한치료의결과에대해방사선및임상적으로분석해보고자한다. 대상및방법 : 원인질환으로는골섬유성이형성증이 5 예로가장많았으며동맥류성골낭종이 1 예, 거대세포종이 1 예였다. 방사선적으로골편이동비율과치유지수및술전길이에대한연장율을분석하였으며임상적으로 Enneking 에따른환측의기능적면을평가하였다. 또한 A.S.A.M.I. group 의분류에따라골적결과와기능적결과를평가하였다. 결과 : 골편이동비율은평균 75 (29-144)% 였으며치유지수는평균 64.5 (34.8-108.6) 일 /cm 였으며술전길이에대한연장율은평균 13 (11-20)% 였다. 환측의기능적평가인 Enneking 에따른분석상 6 예에서우수이상의결과를보였다. A.S.A.M.I. 분류상골적결과및기능적결과는각각우수 6 예, 양호 1 예였다. 결론 : 장골의양성골종양제거술후골결손에대한 Ilizarov 를이용한골신연신생골형성술은자가생골을이용한재건이라는장점을가지며안전하고효과적인방법으로양성골병변의치료에효과적으로사용해볼수있는한방법임을알수있었다. 색인단어 : 양성골종양, 골결손, Ilizarov