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1 ORIGINAL ARTICLE pissn eissn J Korean Soc Surg Hand 2012;17(4): JOURNAL OF THE KOREAN SOCIETY FOR SURGERY OF THE HAND Recurrent Huge Benign Tumors in the Hands Min Wook Kim, So Min Hwang, Kwang Ryeol Lim, Yong Hui Jung, Jennifer K Song Hand and Microsurgery Center, Good Moonhwa Hospital, Busan, Korea Received: August 10, 2012 Revised: November 6, 2012 Accepted: November 19, 2012 Correspondence to: So Min Hwang Hand and Microsurgery Center, Good Moonhwa Hospital, 119 Beomil-ro, Dong-gu, Busan , Korea TEL: FAX: sominhwang@hanmail.net This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License ( licenses/bync/3.0/) which permits unrestricted noncommercial use, distribution, and reproduction in any medium, provided the original work is properly cited. Purpose: Huge benign tumors in the hands sometimes show aggressive nature clinically. We report the clinical features of patients with a large benign recurrent tumors in the hands. Methods: We retrospectively reviewed 139 benign tumors in hands excised by the authors between January, 2006 and March, There were 4 cases of huge benign tumors in hands that recurred after total excision. Results: The average initial tumor size was cm and the average recurrent tumor size was cm. The average period of recurrence from initial operation was 11.3 months. The pathologic findings involved one epidermal cyst, two fibromatosis, and one giant cell tumor of tendon sheath. Although radical removal of the tumors were successful, reoperation due to the tumor recurrence was required. Conclusion: Regardless of the tumor malignancy, a wide range of tumor resection and radiation therapy may be necessary in order to prevent the recurrence of tumors in the hand. Sufficient follow-up periods to determine recurrence were required. Keywords: Benign tumor, Hand, Recurrence 서론 일반적으로인체에서발생되는양성종양은악성종양에비해크기가자라는속도가느리고비침습적이며재발률이낮고생명에위협적이지않은것으로알려져있다. 그러나때때로임상에서조직학적으로양성종양에해당되지만, 종양의크기가크거나종양의특성상임상적으로는악성종양못지않게침습적이고공격적인성향을가지는양성종양을만나게되는경우가있다. 특히수부에발생하는종양은병리조직학적으로양성종양에해당하더라도크기가클경우주변건이나인대, 혈관, 신경등을침범하여수술적제거가쉽지않아임상적으로합병증을남기거나, 재발하는경우가발생하기도한다. 이 에저자들은드물지만수부에발생한 3 cm 이상의거대양성종양을제거한뒤다시재발한임상 4예를분석하여, 수부에발생한거대양성종양의치료후재발을막는데도움이되고자한다. 대상및방법 2006년 1월부터 2012년 3월까지기간중본원에서수부에발생한양성종양중단순종양절제술을시행받은환자들의의무기록을후향적으로조사하였다. 이들중종양의크기에관계없이수부동일부위에종양절제술을두번이상시행받은환자와두번이상의수술모두에서병리조직소견이동일 Copyright c The Korean Society for Surgery of the Hand 153
2 J Korean Soc Surg Hand Vol. 17, No. 4, December 2012 하였던환자를선별하였다. 모든환자는의무기록을통해성별, 나이, 처음수술뒤재발하기까지의기간, 처음수술당시종양의크기와재발후재수술시에종양의크기, 병리조직검사결과, 2차수술후치료가각각조사되었다. 결과 조사기간동안본원에서수부에발생한양성종양중단순종양절제술을시행받은환자는모두 139명이었다. 종양의종류는표피낭종 (epidermal cyst) 19예, 지방종 18예, 피부섬유종 13 예, 결절종 12예로가장많았고, 혈관종 (hemangioma) 9 예, 모기질세포종 (pilomatricoma) 6예, 섬유종증 (fibromatosis) 6예, 사마귀 (wart) 6예등으로다양하였다. 그밖에건막거대세포종 (giant cell tumor of tendon sheath), 신경섬유종, 사구체종양, 림프관종등이소수였다. 수부에서양성종양으로판별받은총 139명의환자중 4명이동일부위에서종양이재발하여다시종양절제술을시행받았다. 재수술받은 4명모두가병리조직검사에서도처음수술당시와동일한결과로확인되었다. 수부에서재발한양성종양환자 4명중남자가 3명, 여자가 1명이었고, 처음발생연령은 20세에서 55세까지로평균연령은 37.8세였다. 종양의병리조직학적진단은표피낭종1예, 섬유종증 2예, 경계성종양인건막거대세포종이 1예였다. 이들의처음수술뒤재발이의심되어다시내원하기까지의평균기간은 11.3개월 ( 범위 : 7-16개월 ) 이었다. 처음발견당시종양의평균크기는 cm ( 범위 : cm) 였으며, 재발이의심되어다시내원하였을당시종양의평균크기는 cm ( 범위 : cm) 였다 (Table 1). 재발이발견된 4예모두에서 2차수술이시행되었고, 2차수술후표피낭종을제외한 3예에서추가적인방사선치료가시행되었다. 환자들은평균 1일 Gy 씩주 5회의통상분할조사법으로총 Gy 방사선조사를받았다. 3예모두에서방사선치료이후최소 1년 6개월까지재발및수술부 위특별한이상소견은발견되지않았다. 1. 증례 1 20세남자환자로우연히발견된좌측손목부위의종물을주소로내원하였다 (Fig. 1). 이학적검사와함께, 종양에대한초음파검사를시행하였다 (Fig. 2). 검사결과, 요골동맥주변부에증식되어있는혈관음영이관찰되어혈관종의심하에단순종양경계부절제술 (marginal excision) 을시행하였다. Fig. 1. Patient 1, 3 2 cm sized tumor in wrist, Lt., palmar aspect. Fig. 2. Encircled radial artery in the tumor was identified sonographically. Table 1. Data of the patients Patient Sex Age (yr) Initial size (cm) Recurrent size (cm) Recurrent period (mo) Histological result 1 Male Giant cell tumor of tendon sheath 2 Male Epidermal cyst 3 Female Fibromatosis 4 Male 23 No data* Fibromatosis Average *The first surgery of the patient 4 was operated at other clinic
3 Min Wook Kim, et al. Recurrent Huge Benign Tumors in the Hands 좌측액와부위마취후에요골동맥과신경주위를둘러싸고있는형태의종양 (Fig. 3) 을미세현미경시야아래서절제한뒤, 3 2 cm 크기의종양덩이를병리조직학적검사한결과건막거대세포종으로확인되었다. 수술후13개월뒤이전수술한동일부위에 4 4 cm 크기의재발소견이관찰되어이전수술보다광범위하게재수술을시행받았다 (Fig. 4). 병리조직학적진단결과이전과같은건막거대세포종으로확인받은뒤, 환자는타병원으로의뢰되어방사선량 1일 2.0 Gy 씩주 5회의통상분할조사법으로총 30 Gy 방사선치료를받았다. 이후 1년 6개월까지환자는재발및치료와관련된후유증은발견되지않았다. 2 cm 크기의양성낭종성종물이손등쪽으로뻗어있는것으로의심되었다 (Fig. 6). 좌측액와부위마취후에요측수지동맥과신경을포함하는종양을미세현미경시야에서조심스럽게박리하여경계부절제술로떼어낸뒤 (Fig. 7), 7 3 cm 크기의종양을병리조직학적검사한결과표피낭종으로확인받았다. 수술후 7개월 2. 증례 2 55세남자환자로 4-5년전부터발견된좌측손바닥의종물이 1개월전부터는손등으로까지확대되는주소로내원하였다 (Fig. 5). 이학적검사와함께자기공명영상검사가시행되어졌다. 검사결과, 손바닥쪽에경계가잘지어진다엽성 5 Fig. 5. Patient 2, 7 3 cm sized tumor in the palmar aspect of the left hand. Fig. 3. The tumor encircled radial artery and nerve. The histological result was giant cell tumor of tendon sheath. Fig. 6. Coronal image high T1 weighted signals suggests a well defined tumor toward the dorsal aspect in the palm. Fig. 4. Patient 1, recurrent 4 4 cm sized tumor in the palmar aspect of the left wrist. Fig. 7. The tumor encircled radial digital artery and nerve. The histological result was epidermal cyst
4 J Korean Soc Surg Hand Vol. 17, No. 4, December 2012 뒤좌측손바닥에서다시종물이만져져 (Fig. 8) 영상학적검사를추천하였으나환자가거절하여바로종양제거술을다시시행하였다 cm 크기의종양을제거하여병리조직학적확인결과, 이전과같은표피낭종으로밝혀졌다. 이후 1년 8개월까지추적관찰결과환자는재발및수술부위특별한이상소견은보이지않았다. 고찰 인체에발생하는종양은병리조직학적특성상양성과악성으로나뉘어진다. 양성종양은대부분자라는속도가느리고, 비침습적이며, 경계도뚜렷하여수술적제거도쉬워재발도드문것으로알려져있다. 악성종양은대부분빠르게자라며, 주위조직에매우침습적이며, 진행한상태에서는수술적완전제거가어려울수있어그만큼재발률도높다. 그러나때때로수부에발생한양성종양은병리조직학적으로는양성에해당되지만, 임상적으로는크기가클수록주변조직에보다침습적이며, 수술적제거가쉽지않고, 수술적제거후에도재발이되는경우가있다. 본저자들의연구에서도전체수부양성종양 139예에서 4예가재발하여, 재발률 2.9% 를보였으며, 이들은종양의크기가최소 cm 이상의큰종양이었다. Larson 1 의연구에서양성종양재발의원인중하나가종양의수술적제거중파종 (seeding) 에의한것이가장크다고보고하였고, Lincoski 등 2 이보고한연구에따르면양성종양의재발은수술당시불완전한제거에의한것이라고보고하였다. 본저자들의증례들에서도수부에발생한양성종양의크기가모두 cm 이상으로크기가크면서건이나인대, 주위조직을둘러싸는형태라든지신경, 혈관등을포함하는형태라서제거를위해비교적어려운술기가요구되 Fig. 8. Patient 2, recurrent cm sized tumor in the palmar aspect of the left hand. 었다. 그로인해육안적으로는종양의완전제거가되었다고생각될지라도술기도중의파종이나, 눈으로확인되지못한종양의일부가남아있었을가능성도배제하기는힘들다. 양성종양의대부분은절제적생검술 (excisional biopsy) 로치료하게되는데, 이는일종의경계절제술이다. 양성공격성종양을완전히제거하기위해서, 그리고악성도가낮은악성종양을절제하기위해서는, 종양의경계부에서부터정상조직을붙여절제하는광범절제술 (wide excision) 이필요하다 3. 본증례들에있어서도처음수술은양성종양제거의일반적인원칙인경계부절제술로종양을제거하였지만종양이재발하였고, 이후두번째수술에서는정상조직의일부를포함하는광범위절제술을시행하였다. McCall 등 4 과 Ge 등 5 의연구에서는재발하는양성종양의악성화가능성을보고하였다. 종양의재발하기까지의기간이짧거나종양자체가가지고있는특성상악성화의경과를보일수가있고, 악성화가되고난다음의종양의치료와예후는악성화되기전보다어렵고좋지않을수밖에없다. 특히본연구의증례 1에서와같이악성화의가능성은낮지만재발률이높은것으로알려져있는건막거대세포종의경우많은연구에서수술적절제후방사선치료가재발률을낮추는것으로보고되고있다 6-9. 섬유종증의경우역시병리조직학적으로는양성종양임에도불구하고종양의특성상침습성이강하고, 국소재발률또한높아완전치료가어려운것으로알려져있다. Shin 등 10 과 Goy 등 11 은공격성섬유종증에서수술적제거후방사선치료가효과적으로국소재발률을낮출수있다고보고하였다. 본연구의섬유종증 2예에있어서도수술적제거를하였음에도불구하고재발이발견되었고, 재수술후에추가적인방사선치료가병행되어좋은결과를얻었다. 그러므로양성종양일지라도크기가큰경우의치료에있어서때로는과감한광범위절제라든지종양의특성에따라방사선치료와같은추가적인치료요법을고려하여야한다. 하지만, 방사선치료가모든양성종양에서재발률을낮출수있는것은아니다. 오히려방사선치료후양성종양의악성화나방사선유래원발성종양의발생이종종보고되기하여 Evans 등 12 은양성종양의방사선치료에있어서보다주의깊은접근을추천하기도하였다. Goo 등 13 은양성종양의성장속도가느린특성상재발이발견되는데시간이오래걸릴수있어종양이적절하게제거가되었다고판단될지라도추적관찰이꼭필요하고충분한추적관찰기간을가질것을주장하였다. 본저자들의연구에서도첫수술후재발이발견되기까지짧게는 7개월, 길게 156
5 Min Wook Kim, et al. Recurrent Huge Benign Tumors in the Hands 는 16 개월이지나서였다. 따라서양성종양의특성을고려하여보다긴추적관찰기간을가질필요가있다. 결론 재발이드문것으로알려진수부에발생한양성종양도크기가클경우, 수술후에도재발할가능성이있고, 임상적으로는침습적이며주위조직에공격적인성향을가질수가있다. 수부에발생한양성종양의재발을막고, 적절한치료를위해서는종양의병리조직학적인악성도와는관계없이, 양성종양일지라도수부에발생한경우, 그크기와침습성를고려하여경우에따라서는광범위절제술과방사선치료와같은적절한추가적치료가필요할수있다. 그리고치료후에도충분한추적관찰기간을통해재발유무를확인할필요가있다. 참고문헌 1. Larson DL. Management of the recurrent, benign tumor of the parotid gland. Plast Reconstr Surg. 2001; 108: Lincoski CJ, Bush DC, Millon SJ. Epidermoid cysts in the hand. J Hand Surg Eur Vol. 2009;34: Mankin HJ. Principles of diagnosis and management of tumors of the hand. Hand Clin. 1987;3: McCall T, Rao G, Jensen R. Development and rapid growth of a desmoid tumor in the surgical corridor after suboccipital craniotomy for recurrent low-grade astrocytoma. J Neurooncol. 2006;80: Ge P, Luo Y, Fu S, Ling F. Recurrent epidermoid cyst with malignant transformation into squamous cell carcinoma. Neurol Med Chir (Tokyo). 2009;49: Heyd R, Micke O, Berger B, Eich HT, Ackermann H, Seegenschmiedt MH, et al. Radiation therapy for treatment of pigmented villonodular synovitis: results of a national patterns of care study. Int J Radiat Oncol Biol Phys. 2010;78: Chin KR, Barr SJ, Winalski C, Zurakowski D, Brick GW. Treatment of advanced primary and recurrent diffuse pigmented villonodular synovitis of the knee. J Bone Joint Surg Am. 2002;84: Schnirring-Judge M, Lin B. Pigmented villonodular synovitis of the ankle-radiation therapy as a primary treatment to reduce recurrence: a case report with 8- year follow-up. J Foot Ankle Surg. 2011;50: Ravi V, Wang WL, Lewis VO. Treatment of tenosynovial giant cell tumor and pigmented villonodular synovitis. Curr Opin Oncol. 2011;23: Shin KH, Shin SJ, Lee DH, Kang ES, Suh CO. The role of radiotherapy in the treatment of aggressive fibromatosis. Yonsei Med J. 1999;40: Goy BW, Lee SP, Eilber F, Dorey F, Eckardt J, Fu YS et al. The role of adjuvant radiotherapy in the treatment of resectable desmoid tumors. Int J Radiat Oncol Biol Phys. 1997;39: Evans DG, Birch JM, Ramsden RT, Sharif S, Baser ME. Malignant transformation and new primary tumours after therapeutic radiation for benign disease: substantial risks in certain tumour prone syndromes. J Med Genet. 2006;43: Goo J, Jung YJ, Kim JH, Lee SY, Ahn SK. A case of recurrent superficial acral fibromyxoma. Ann Dermatol. 2010;22:
6 J Korean Soc Surg Hand Vol. 17, No. 4, December 2012 수부에서재발한거대양성종양 김민욱 황소민 임광열 정용휘 송제니퍼김좋은문화병원미용성형재건센터 목적 : 수부에발생한거대한양성종양은임상적으로공격적인임상소견을보일수있다. 수부에발생한재발성거대양성종양의임상양상에대하여보고하고자한다. 대상및방법 : 2006년 1월부터 2012년까지 3월까지수부에발생한양성종양을절제수술받은환자 139명에대해후향적조사를시행하였다. 그중 4명의수부에서재발한거대양성종양환자들을선별하였다. 각환자들의성별, 나이, 종양크기, 재발기간, 종양의병리조직학적결과에대한정보를분석하였다. 결과 : 처음발생종양의평균크기는 cm, 재발이발견될당시평균크기는 cm, 재발이발견되기까지걸린평균기간은 11.3개월이었다. 이들의병리조직학적결과는표피낭종 1예, 섬유종증 2예, 건막거대세포종이 1예였다. 4예모두에서종양이적절한수술적절제가이루어졌음에도불구하고재발하여다시수술적절제가필요하였다. 결론 : 종양의악성도와는관계없이수부에서종양의재발을막기위해서는광범위한종양절제술과방사선치료와같은추가적인치료가필요할수있다. 치료후에도충분한추적관찰기간을통해재발유무를확인해야한다. 색인단어 : 양성종양, 수부, 재발 접수일 2012 년 8 월 10 일수정일 2012 년 11 월 6 일게재확정일 2012 년 11 월 19 일교신저자황소민부산시동구범일로 119 좋은문화병원수부미세수술센터 TEL FAX sominhwang@hanmail.net 158
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