279 pissn : , eissn : Case Report J Korean Orthop Assoc 2017; 52: 슬

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1 279 pissn : , eissn : Case Report J Korean Orthop ssoc 2017; 52: 슬관절통증을유발한사구체종양 박희곤 김성현 류지원 단국대학교의과대학정형외과학교실 Glomus Tumor Causing Knee Pain Hee-Gon Park, M.D., Ph.D., Sung-Hyun Kim, M.D., and Jee-Won Ryu, M.D. Department of Orthopaedic Surgery, Dankook University Medical College, Cheonan, Korea Glomus tumor is a kind of hemangioma that occurs at the glomerulus in the subcutaneous layer. It mainly occurs at the distal hand and subungual area, and rarely at the knee joint. Pain, tenderness, and cold intolerance are known symptoms; however, symptoms in practice are not so easily detectable, and the diagnosis can be delayed if it is presented at areas other than the hand. If the diagnosis is delayed, patients could suffer extreme pain. Therefore, early diagnosis and surgical treatment are important. Ultrasound and magnetic resonance imaging were used to diagnose glomus tumor in our cases, which were found in subcutaneous tissue and muscle fascia. We claim that, for patients with persistent pain, known symptoms extreme pain, cold intolerance, and tenderness should be examined carefully and rule out glomus tumor. We report 2 cases of glomus tumors around the knee joint, which is not a common location of occurrence. Key words: knee, glomus tumor, subcutaneous tissue, fascia, biopsy 사구란모세혈관의연결없이동정맥문합으로구성된말초기관 이며한층의내피층과이주위를둘러싸는평활근의특징을가 진사구세포들로구성되어있고신체중수부에가장많이분포 하고있는것으로알려져있다. 1) 사구종 (glomus tumor) 은피부와 피하인접부위에위치하는피부의온도를조절하는사구체 (glomus body) 에서발생하는혈관종 (vascular tumor) 의일종으로, 모 든연부조직종양의약 1.6% 의빈도를보이고수부종양의약 1% 를차지하는드문양성종양이다. 1,2) 단발형 (solitary type) 과다발 형 (multiple type) 으로분류되고단발형은대개수부에서발견되 며 25%-75% 에서조갑하에위치하고다발형은신체어느곳에서 나발견될수있다. 2) 40 대에서호발하며 20 세이하나 60 세이상에 서는드물게발생하고, 3) 통증, 압통, 추위민감성이삼주징으로알 려져있으나실제로증상이명확하지않은경우가많다. 2) 수지이 외의부위에서발생하는경우나심부조직에발생하는경우사구 Received September 5, 2016 Revised December 12, 2016 ccepted December 13, 2016 Correspondence to: Hee-Gon Park, M.D., Ph.D. Department of Orthopaedic Surgery, Dankook University Medical College, 119 Dandae-ro, Dongnam-gu, Cheonan 31116, Korea TEL: FX: lovebio1ogy@naver.com 종진단이늦어질수있으며슬관절주위에서는드물게발생하는것으로알려져있다. 4) 본교실에서는일반적으로호발하는부위가아닌슬관절주위에서발생한사구종 2예를경험하였기에이에대해보고하고자한다. 증례보고 1. 증례 1 51세여자환자가좌측슬개건전방의통증을동반한종괴를주소로내원하였다. 과거력상내원 5년전부터슬개건전방에종괴소견이있었고크기변화는없었다. 내원 6개월전부터압통이있었으며앉는자세에서통증이심해졌고추위에노출되었을때증상이심해지는소견을보여인근병원에서보존적치료를받았으나증상호전이없어본원외래로내원하였다. 이학적검사상좌측슬개건전방에위치하는약 cm 의청자색의색상변화를보이는종괴가관찰되었으며주위조직과의경계가비교적분명하였고좁은범위에서이동성을보였다 (Fig. 1). 압통은종양이있는정확한위치에서발생하였으며, 슬관절최대굴곡시통증심해졌고, ice test상에서냉온에민감한반 The Journal of the Korean Orthopaedic ssociation Volume 52 Number Copyright 2017 by The Korean Orthopaedic ssociation This is an Open ccess article distributed under the terms of the Creative Commons ttribution Non-Commercial License ( which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

the left knee (arrowhead). 응을보였다. 감각이상및운동이상등의다른증상은없었으며, 단순방사선검사및혈액검사소견상이상소견은없었다.

국소마취하에수술이시행되었고약 2 cm의절개를통해종양에접근하여주변조직과잘구분되는피막으로둘러싸인종양을확인하였다. 종양은피하지방층에위치하였고피막과함께적출하였다. 적출된종양은원형의약 0.4 0.3 0.

병리조직학적소견상정상혈관내피세포를갖는많은혈관구 (vascular channel) 가있었고그주위에운집하고있는둥근세포들이관찰되었으며면역조직화학염색에서평활근액틴 (smooth muscle actin, SM)

Ultrasound finding of the mass lesion. () Well-defined ovoid hypoechoic nodular lesion with a size of about 0.4 0.2 0.

2 280 Hee-Gon Park, et al. Figure 1. Preoperative clinical photo showing the elevated subcutaneous layer of patellar tendon due to the bluish tender lesion on the left knee (arrowhead). 응을보였다. 감각이상및운동이상등의다른증상은없었으며, 단순방사선검사및혈액검사소견상이상소견은없었다. 초음파검사상최대직경 5 mm 크기의저에코성병변이슬개건전방의피하층에서관찰되었으며도플러검사상고혈관성 (hypervascular) 병변이관찰되었다 (Fig. 2). 국소마취하에수술이시행되었고약 2 cm의절개를통해종양에접근하여주변조직과잘구분되는피막으로둘러싸인종양을확인하였다. 종양은피하지방층에위치하였고피막과함께적출하였다. 적출된종양은원형의약 cm 크기의경계가명확한섬유성피막 (fibrous capsule) 으로싸여있었다. 병리조직학적소견상정상혈관내피세포를갖는많은혈관구 (vascular channel) 가있었고그주위에운집하고있는둥근세포들이관찰되었으며면역조직화학염색에서평활근액틴 (smooth muscle actin, SM) 양성소견을보여사구종으로확진하였다 (Fig. 3). 수술후환자는좌측슬개건앞부분의통증및압통은없어졌으며수술후 1년추시상술전증상의재발은없었다. Figure 2. Ultrasound finding of the mass lesion. () Well-defined ovoid hypoechoic nodular lesion with a size of about cm in the subcutaneous fat layer of the prepatellar area on transverse sonograms (arrow). () Hypervascular mass lesion on longitudinal Doppler sonograms. Figure 3. Microscopic findings of the mass lesion. () It is composed of capillary sized vessels surrounded by solid proliferation of round-to-cuboidal epithelioid cells with round nuclei (H&E, 200). () Tumor cells are smooth muscle actin (SM) positive (SM, 200).

281 Glomus Tumor Causing Knee Pain Figure 4. Sagittal T2 () and axial T1 () magnetic resonance imaging findings.

femoral insertion site of medial head of gastrocnemius tendon. Separated from neurovascular bundle in popliteal fossa. 척추마취하에복와위로수술을시행하였고주변조직과잘구분되는피막으로둘러싸인종양을확인하였다. 종양은슬와동맥바로내측심부에위치하였고피막과함께조심스럽게적출하였다.

수술후좌측슬관절내부및슬와부분의통증및압통은없어졌으며수술후 6개월추시상증상의재발은없었고, 현재추시관찰중이다. 고찰 Figure 5. Gross photo of specimen shows a round-in-shape, wellencapsulated mass with a size of 1.3 1.5 1.5 cm. 2.

3 281 Glomus Tumor Causing Knee Pain Figure 4. Sagittal T2 () and axial T1 () magnetic resonance imaging findings. Well-defined 1-cm-sized T1 iso signal (black arrowhead), T2 heterogeneous high signal nodular lesion (white arrowhead) in just the medial and deep aspects of the popliteal artery at the level of femoral insertion site of medial head of gastrocnemius tendon. Separated from neurovascular bundle in popliteal fossa. 척추마취하에복와위로수술을시행하였고주변조직과잘구분되는피막으로둘러싸인종양을확인하였다. 종양은슬와동맥바로내측심부에위치하였고피막과함께조심스럽게적출하였다. 적출된종양은약 cm 크기의원형종괴였으며경계가명확한섬유성피막으로싸여있었고 (Fig. 5), 병리조직학적소견상사구종으로확진되었다 (Vimentin & SM: Positive, CE, EM, S-100, pan-ck [5,6,8,18], p53, Desmin, CD31, CD34: negative) (Fig. 6). 수술후좌측슬관절내부및슬와부분의통증및압통은없어졌으며수술후 6개월추시상증상의재발은없었고, 현재추시관찰중이다. 고찰 Figure 5. Gross photo of specimen shows a round-in-shape, wellencapsulated mass with a size of cm. 2. 증례 2 26 세여자환자가 8 년전부터간헐적으로좌측슬관절및슬와부 분의통증, 압통및저린감이있었고내원 1 년전부터증상이심 해져보존적치료를시행받았으나증상호전이없어본원에내 원하였다. 이학적검사상슬관절을신전시통증이발생하였고 슬와부분상방으로압통이있었으며촉지되는종괴는없었다. 감각이상및운동이상등의다른증상은없었고단순방사선촬 영및말초혈액검사소견상이상소견은없었다. 조영증강자기공명영상촬영에서 T1 강조영상에서동신호 강도 (iso-signal), T2 강조영상에서이성고신호강도 (heterogeneous high signal), 그리고약하게조영증강되는경계가명확한 결절성병변이관찰되었다. 이근막내종양은비복근내측두대 퇴기시부위의슬와동맥의내측에위치하였다 (Fig. 4). 정상사구체는모세혈관망없이직접동정맥문합으로이루어지는말초기관으로체온조절기능, 혈압조절기능, 세포간환경조절기능과연관이있다. 1,5) 사구종양의가장특징적인임상징후는동통으로부분적인압력이나온도변화, 특히저온노출에의해심해지는통증의양상을보이며, 심한경우환자가이환부위의절단을원할정도로동통을호소하는경우도있다. 3) 진단이지연되는경우가많고신경성호소 (neuropathic complaint), 관절염, 신경통 (neuralgia) 등으로잘못진단되기쉬우며, 교감신경절제술, 신경근절단술등잘못된치료를받게되는경우가흔히발생한다. 6) 이와유사하게동통성결절이있을때는섬유종, 지방종, 혈관평활근종, 결절종, 한선종, 신경섬유종, 평활근종, 육종, 혈전성정맥염등과감별해야하며, 가장많이오진되는병명은혈관종, 신경종, 신경섬유종이라고하였다. 4) 특히병리조직학적소견상사구종양은사구세포덩어리에의해둘러쌓인내피세포로싸여있는혈관강 (vascular spaces) 으로관찰되고혈관종은적혈구세포 (red blood cells) 또는삼출액 (transudate) 을포함하는혈관구조물

282 Hee-Gon Park, et al. Figure 6. Microscopic findings. () Regular round tumor cells forming solid sheets showed central nuclei, small nucleoli, and conspicuous cytoplasmic borders (H&E, 400).

하지만증상이경미하거나없는경우도있고호발부위인수부가아닌다른부위에발생하거나심부조직에위치한경우진단이늦어질수있다. 슬관절부위에발생한사구종의보고는발생부위의다양성을시사한다.

4 282 Hee-Gon Park, et al. Figure 6. Microscopic findings. () Regular round tumor cells forming solid sheets showed central nuclei, small nucleoli, and conspicuous cytoplasmic borders (H&E, 400). () Tumor cells are smooth muscle actin (SM) positive (SM, 200). (vascular structures) 의수적증가로나타나며단층내피세포로싸여있다. 7) 이러한사구종양은정확한병력청취와이학적검사만으로도진단이가능하며조직검사로서확진할수있다. 하지만증상이경미하거나없는경우도있고호발부위인수부가아닌다른부위에발생하거나심부조직에위치한경우진단이늦어질수있다. 슬관절부위에발생한사구종의보고는발생부위의다양성을시사한다. Lee 등 8,9) 은슬개건주위, 슬개골하지방괴등에서사구종이발생할수있으며이러한경우진단이늦어질수있고오진으로인한잘못된치료를할수있어정확한술전검사가필요하다고하였고, 슬관절통증의원인중의하나로사구종을감별해야한다고하였다. Shaikh 등 10) 은심부근막내에발생한 15예의사구종에대해보고하였다. 모두진피혹은피하조직의병변을동반한다발형이었으며, 단발형사구종은없었고심부의사구종진단을위해서는조기에자기공명영상검사가필요하다고하였다. 현재까지슬관절주위의심부근막내에발생한단발형사구종에대한보고는없으며증례 2와같이슬관절주위심부근막내에발생한단발형사구종의경우임상적의의가클것으로생각된다. 본교실에서경험한환자의경우는일반적호발부위가아닌슬관절부위에발생한사구종으로, 초음파검사 ( 증례 1) 와자기공명영상검사 ( 증례 2) 를통해사구종을진단하였다. 일반적인임상양상을보이지않아진단이지연된경우로, 슬관절의지속되는통증이있는경우철저한이학적검사와정확한술전검사를통해서사구종의가능성을항상고려해야할것으로생각된다. 슬관절주위의지속되는통증을호소하는환자에서사구종의삼주징인극심한통증, 추위민감성, 압통의증상유무를주의깊게관 찰하여사구종도감별해야함을위증례들을통해알수있었고, 진단이늦어질경우임상적으로환자에게고통을줄수있으므로빠른진단과수술적치료가중요할것으로생각된다. CONFLICTS OF INTEREST The authors have nothing to disclose. REFERENCES 1. Carroll RE, erman T. Glomus tumors of the hand: review of the literature and report on twenty-eight cases. J one Joint Surg m. 1972;54: Gombos Z, Zhang PJ. Glomus tumor. rch Pathol Lab Med. 2008;132: Takei TR, Nalebuff E. Extradigital glomus tumour. J Hand Surg r. 1995;20: Prabhakar S, Dhillon MS, Vasishtha RK, ali K. Glomus tumor of Hoffa's fat pad and its management by arthroscopic excision. Clin Orthop Surg. 2013;5: Kim SH, Suh HS, Choi JH, Sung KJ, Moon KC, Koh JK. Glomus tumor: a clinical and histopathologic analysis of 17 cases. nn Dermatol. 2000;12: Seo C, Oh DY, Park KS, et al. Glomus tumor in soleus muscle: a case report. J Korean Soc Plast Reconstr Surg. 2006;33: aek HJ, Lee SJ, Cho KH, et al. Subungual tumors: clinico-

5 283 Glomus Tumor Causing Knee Pain pathologic correlation with US and MR imaging findings. Radiographics. 2010;30: Lee MC, Seong SC, Moon YW, Park YK. Glomus tumor adjacent to patellar tendon, a case report. J Korean Knee Soc. 1996;8: Lee SH, Lee MC, Seong SC, Jeong GI. Glomus tumor in the infrapatellar fat pad, a case report. J Korean Knee Soc. 2001;13: Shaikh R, lomari I, Mulliken J, Fishman SJ, Kozakewich HP, Chaudry G. Subfascial involvement in glomuvenous malformation. Skeletal Radiol. 2014;43:895-7.

6 284 pissn : , eissn : Case Report J Korean Orthop ssoc 2017; 52: 슬관절통증을유발한사구체종양 박희곤 김성현 류지원 단국대학교의과대학정형외과학교실 사구종은피하지방층의사구체에서발생하는혈관종의일종으로수부의원위부, 조갑하에특징적으로호발하며슬관절주위에서는드물게발생하는것으로알려져있다. 통증, 압통, 추위민감성이삼주징으로알려져있으나실제로증상이명확하지않은경우가많고수지이외의부위에서발생하는경우에는사구종진단이늦어질수있다. 수년간지속적으로슬관절통증이있었던사구종증례 2 예를경험하였으며각각초음파와자기공명영상검사를통해진단하였다. 슬관절주위의지속되는통증을호소하는환자에서사구종의삼주징인극심한통증, 추위민감성, 압통의증상유무를주의깊게관찰하여사구종도감별해야함을이증례를통하여알수있었고빠른진단과수술적치료가중요할것으로생각되어이에대해보고하는바이다. 색인단어 : 슬관절, 사구종, 피하조직, 근막, 생검술 접수일 2016 년 9 월 5 일수정일 2016 년 12 월 12 일게재확정일 2016 년 12 월 13 일책임저자박희곤 31116, 천안시동남구단대로 119, 단국대학교의과대학정형외과학교실 TEL , FX , lovebio1ogy@naver.com 대한정형외과학회지 : 제 52 권제 3 호 2017 Copyright 2017 by The Korean Orthopaedic ssociation This is an Open ccess article distributed under the terms of the Creative Commons ttribution Non-Commercial License ( which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

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(49-54)Kjhps004.hwp Biliary Cystadenoma and Cystadenocarcinoma of the Liver Jae Hoon Lee, M.D., Dong Ho Choi, M.D. 1, Kyeong Geun Lee, M.D., Hwon Kyum Park M.D. and Kwang Soo Lee, M.D. Department of Surgery, College of Medicine,