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400 pissn : 1226-2102, eissn : 2005-8918 Case Report J Korean Orthop Assoc 2014; 49: 400-404 http://dx.doi.org/10.4055/jkoa.2014.49.5.400 www.jkoa.org 강민수 문규필 김경택 박원로 박원석 이경신 * 동의의료원정형외과, * 병리과 An Extradural En-Plaque Meningioma Involving Thoracic Spine Min Soo Kang, M.D., Kyu Pill Moon, M.D., Kyung Taek Kim, M.D., Won Ro Park, M.D., Won Seok Park, M.D., and Kyoung Shin Lee, M.D.* Departments of Orthopaedic Surgery and *Pathology, Dong-Eui Medical Center, Busan, Korea Reports of extradural spinal meningioma are rare, and differentiation from a metastatic lesion is important. We treated a case of thoracic extradural meningioma with surgical excision and obtained a favorable outcome without recurrence during one-year follow-up. Thus, we report on a case with review of the literature. Key words: meningioma, extradural 수막종은중추신경계종양의하나로, 대부분천막상 (supratentorial) 병변이나척추부위발생도많은편이다. 척추수막종은지주 막융모에있는수막세포형 (meningothelial) 세포에서기원하기 때문에대부분이경막내병변이다. 그러나일부는경막외에서 발생되며, 경막외수막종은위치, 수술중종양의전반적소견, 급 격한증세의진행및방사선학적골구조변화때문에경막외척 추전이성병변과의감별이중요하다. 1) 척추경막외수막종은보 고된바가적고, 국내에서보고된예는없다. 저자들은흉추에발 생한경막외수막종 1 예를수술적제거후 1 년간추시관찰하여 양호한결과를경험하였기에이를문헌고찰과함께보고하고자 한다. 증례보고 45 세여자환자로내원전 2 개월부터시작된양하지무력감, 이상 보행, 양하지감각저하및이상감각을주소로내원하였다. 내과 적질환이나외상의기왕력은없었다. 내원당시이학적검사상 Received April 25, 2014 Revised July 3, 2014 Accepted July 11, 2014 Correspondence to: Won Ro Park, M.D. Department of Orthopaedic Surgery, Dong-Eui Medical Center, 62 Yangjeong-ro, Busanjin-gu, Busan 614-710, Korea TEL: +82-51-850-8937 FAX: +82-51-850-8943 E-mail: wonro@hanmail.net 유두와배꼽사이이하로감각저하및이상감각소견을보였으며, 고관절이하로양하지근력이 4/5 정도로감소되어있었다. 심부건반사는양측으로항진되어있었고, 양측족간대성경련양성소견을보였다. 단순방사선사진에서는경미한측만변화가관찰되었고, 뚜렷한골파괴소견등은보이지않았다. 자기공명영상소견에서는 T1 및 T2 강조영상에서척수와비슷한신호강도를보이는비교적경계가명확한약 33 mm의경막외종양이흉추 6, 7번에걸쳐서우측흉추 6번신경근을포함하여척수를후방에서압박하고있는소견이관찰되었으며, 우측흉추 6번신경공의확장소견이관찰되었다. 이종양은가돌리니움 (gadolinium) 조영증강영상에서는경막과명확한경계의균질의조영증강소견을보였다 (Fig. 1). 경막외종양으로는거대세포종, 혈관종, 동맥류성골낭종등척추의전방또는후방의골을침범하는종양이대부분이나, 본증례에서는골파괴소견이보이지않아진단에서배제시켰고, 판형의경막외종양으로는전이성병변과임파종의감별이필요했다. 술전검사에서혈액검사, 혈액도말검사및단순흉부방사선사진에서이상소견은없었으며, 종양표지자검사는정상이었다. 흉부및복부전산화단층촬영이전이성병변과임파종의감별에도움이되나환자의병력상내원전체중감소나사지및추체의동통등특이소견이없고, 여러혈액검사에서정상소견이보여시행하지않았으며, 전이성병변이나임파종은수막종에비해골파괴소견이많아가능성은적으 The Journal of the Korean Orthopaedic Association Volume 49 Number 5 2014 Copyright 2014 by The Korean Orthopaedic Association This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/3.0/) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

401 Figure 1. (A) On T1-weighted images, a mass separated from dura mater that compressed the spinal cord at the 6th and 7th thoracic vertebral level was seen on a sagittal image and a widened right neural foramen with compression of the 6th thoracic nerve root by the mass was seen on an axial image. (B) On T2-weighted images, the mass showed relatively homogeneous similar signal intensity with the spinal cord. (C) On gadoliniumenhanced images, the mass was enhanced homogeneously and distinguished from dura mater. Figure 2. (A) The mass was seen on posterior dura mater intraoperatively. (B) The mass was completely excised. 나완전히배제하지는못하고수술을시행하였다. 수술적치료는 흉추 6, 7 번척추궁전절제술을시행하고, 우측내측관절돌기일 부를제거하여신경근을충분히관찰할수있도록하였다. 중심 부종양은경막과유착소견이거의없어하나의바늘구멍손상 외에박리가비교적잘이루어졌으며바늘구멍손상부위로경막 내종양소견은없었다 (Fig. 2). 우측신경근부위는신경근출구 를철저히확인해가면서잔여조직없이종양을제거하려하였 다. 종양주변의잔여지방조직을제거하였으며, 술중획득한자 가골은이식하지않았다. 고정은흉추 5, 6, 7 번에척추경나사삽 입을하였다 (Fig. 3). Figure 3. Postoperative plain radiograph showed posterior fixation. 해부병리소견상수막세포형및혈관종형 (angiomatous) 이혼재되어있는세계보건기구 (World Health Organization, WHO) 1단계의수막종으로진단되었고, 유두체 (psammoma body) 소견은많지않았으며, 악성소견인유사분열은보이지않았으나주변지방조직침범은관찰되었다 (Fig. 4). 술후 6주에양하지근력은거의정상소견이었으며, 경미한이상감각소견만잔존하였다. 술후 1년추시에서신경적기능은완전회복하였고, 운동시흉추부에경미한불편감및동통을호소하였다. 1년추시방사선소견에서경도의전만및측만변화를보였으나, 이는술후 3개월보다진행하지는않았으며, 추시자기공명영상에서국소재발의소견은관찰되지않았다 (Fig. 5).

402 강민수 문규필 김경택외 3 인 Figure 4. (A) The meningotheliomatous tumor cells were arranged in nests reminiscent of paraganglioma with hyalinized stromal vasculature. The tumor cells invaded into adjacent fat tissue (H&E, 40). (B) Nests of meningotheliomatous tumor cells with several psammoma bodies and proliperated hyalinized vasculatures are noted. Mitosis is not seen (H&E, 100). 고찰 경막의외부에는거미막세포가없으므로실질상경막외수막 종이라는종양은존재할수없다. 일부저자들은시신경의막과 연결이없는안구내수막종처럼거미막조직의섬들이경막밖 의공간으로이동하였을것이라상정하였고, 다른저자들은코나 피부에서발생하는두개외수막종처럼경막외공간에비정상 적인거미막조직덩어리가있었을것이라상정하였다. 2,3) Haft 와 Shenkin 4) 은주된거미막층에서뒤늦게분리된거미막융모세포 의경막침습가능성을제시했다. Pecker 등 5) 은신경근근처의경막이상대적으로얇고배아지주 막과융모의표재층퇴화잔존물을포함하고있어, 이것이경막 외수막종이신경근근처에서발생하는것과관련이있다고하였 다. 일부연구자들은경막외주변지방조직으로의침범소견을 기술하였다. 6) 저자들의경우에도종양이흉추 6 번우측신경근을 포함하고있었으며, 현미경적소견에서주변지방조직으로의침 범이관찰되었다. 저자들은수술시내측관절돌기일부를제거하였으나흉추는 경추나요추에비해늑골의지지에의한안정성이있고종양발 생부위가흉곽의중심부로상대적으로안정적이라판단하였다. 술중획득한자가골이나동종골을이식하지않고척추경나사만 삽입하였으나추시에서경도의전만및측만변화가있었다. 충 분히지방조직을제거한후자가골을이식하거나동종골이식을 시행했다면변화의정도를더감소시킬수도있었을것으로생각 된다. 병리학적으로수막종은경막부착부와잘분리되는경향이있 으며, 경막표면을따라얇게퍼져 en plaque 형태로자란다. 현미 Figure 5. (A) On T2-weighted image, there was no local recurrence at one-year follow-up. (B) On a plain radiograph at one-year follow-up, mild kyphosis was seen. 경적소견으로는 WHO 1 단계는공격적인성장이나재발의위 험이적어서좋은예후를보이고, 수막세포형과섬유모세포형 (fibroblastic) 및두가지형이혼재되어있는이행형 (transitional) 이 가장흔하다. 2 단계는비정형 (atypical) 수막종으로재발의빈도가 높고보다공격적인성장을보이며, 술후방사선치료가필요할 수있다. 3 단계는악성수막종으로, 수막세포에서기원하는조직 학적소견이있으나고도의육종모양을지닌매우공격적인종 양이다. 7) Mariniello 등 8) 은경추부를침범한 WHO 1 단계의판형 경막외수막종을수술적으로제거하였으나광범위한경막으로 의침투소견이있어서제거하지못하고술후 50 Gy 의방사선치 료를시행하여임상적으로호전된 1 예를보고하였다. 저자들의 경우수막세포형에혈관이많이증식한혈관종형이혼재되어있 는 1 단계종양이며, 수술소견에서경막과잘분리되고침투소견 이뚜렷하지않아술후방사선치료는시행하지않았다. Barbanera 등 9) 은경추 5 번과흉추 1 번사이에발생한경막내, 외병변이혼재되어있는양성수막종제거 2 년후악성으로의재 발소견을보고한바있으나대부분의경막외수막종은양성으로 보고되고있으며 Solero 등 10) 은 9 명의경막외수막종수술환자 4-17 년간의추시에서 1 명의재발만보고하였다. 경막내병변이혼재되어있지않은순수한경막외수막종은 매우드물며전이성병변과의감별또한중요하다. 저자들의경 우경막외수막종제거후 1 년추시에서국소재발소견이없는 양호한결과를치험하였고대부분의수막종은양성이며재발의 빈도도적으나지속적추시관찰은필요할것으로생각된다. REFERENCES 1. Milz H, Hamer J. Extradural spinal meningiomas. Report of

403 two cases. Neurochirurgia (Stuttg). 1983;26:126-9. 2. Fortuna A, Gambacorta D, Occhipinti EM. Spinal extradural meningiomas. Neurochirurgia (Stuttg). 1969;12:166-80. 3. Sato N, Sze G. Extradural spinal meningioma: MRI. Neuroradiology. 1997;39:450-2. 4. Haft H, Shenkin HA. Spinal epidural meningioma: case report. J Neurosurg. 1963;20:801-4. 5. Pecker MJ, Javalet A, Simon J, Loussouarn Y. Benign epidural tumors of the spinal cord. Neurochirurgie. 1967;13:647-60. 6. Stern J, Whelan MA, Correll JW. Spinal extradural meningiomas. Surg Neurol. 1980;14:155-9. 7. Kumar V, Abbas AK, Fausto N, Aster JC. Robbins & Cotran pathologic basis of disease. 8th ed. Philadelphia: Saunders; 2010. 1338-9. 8. Mariniello G, Briganti F, De Caro ML, Maiuri F. Cervical extradural "en-plaque" meningioma. J Neurol Surg A Cent Eur Neurosurg. 2012;73:330-3. 9. Barbanera A, Nina P, Serchi E, Ascanio F. Aggressive recurrence of intra-extradural cervico-thoracic meningothelial meningioma. Acta Neurochir (Wien). 2007;149:83-6. 10. Solero CL, Fornari M, Giombini S, et al. Spinal meningiomas: review of 174 operated cases. Neurosurgery. 1989;25:153-60.

404 pissn : 1226-2102, eissn : 2005-8918 Case Report J Korean Orthop Assoc 2014; 49: 400-404 http://dx.doi.org/10.4055/jkoa.2014.49.5.400 www.jkoa.org 강민수 문규필 김경택외 3 인 강민수 문규필 김경택 박원로 박원석 이경신 * 동의의료원정형외과, * 병리과 척수경막외수막종은보고된바가적고, 전이성병변과의감별도중요하다. 저자들은흉추부에발생한경막외수막종 1 예를수술적 제거후, 1 년간추시관찰하여재발없이양호한결과를경험하였기에, 이를문헌고찰과함께보고하고자한다. 색인단어 : 수막종, 경막외 접수일 2014 년 4 월 25 일수정일 2014 년 7 월 3 일게재확정일 2014 년 7 월 11 일책임저자박원로부산시부산진구양정로 62, 동의의료원정형외과 TEL 051-850-8937, FAX 051-850-8943, E-mail wonro@hanmail.net 대한정형외과학회지 : 제 49 권제 5 호 2014 Copyright 2014 by The Korean Orthopaedic Association This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/3.0/) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.