Journal of Korean Society of Spine Surgery Sequestrated Intradural Disc Herniation Around Couns Medullaris - A Case Report - Jaewon Lee, M.D., Wan-Sik

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Journal of Korean Society of Spine Surgery Sequestrated Intradural Disc Herniation Around Couns Medullaris - A Case Report - Jaewon Lee, M.D., Wan-Sik Seo, M.D., Ye-Soo Park, M.D., Young-Ha Oh, M.D. J Korean Soc Spine Surg 2014 Sep;21(3):134-138.

Sinheung-Dong, Jung-Gu, Incheon, 400-711, Korea Tel: 82-32-890-3044 Fax: 82-32-890-3467 Copyright 2014 Korean Society of Spine Surgery pissn 2093-4378 eissn 2093-4386 The online version of this

1 Journal of Korean Society of Spine Surgery Sequestrated Intradural Disc Herniation Around Couns Medullaris - A Case Report - Jaewon Lee, M.D., Wan-Sik Seo, M.D., Ye-Soo Park, M.D., Young-Ha Oh, M.D. J Korean Soc Spine Surg 2014 Sep;21(3): Originally published online September 30, 2014; Korean Society of Spine Surgery Department of Orthopedic Surgery, Inha University School of Medicine #7-206, 3rd ST. Sinheung-Dong, Jung-Gu, Incheon, , Korea Tel: Fax: Copyright 2014 Korean Society of Spine Surgery pissn eissn The online version of this article, along with updated information and services, is located on the World Wide Web at: This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License ( 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.

2 Case Report pissn eissn J Korean Soc Spine Surg Sep;21(3): Sequestrated Intradural Disc Herniation Around Couns Medullaris - A Case Report - Jaewon Lee, M.D., Wan-Sik Seo, M.D., Ye-Soo Park, M.D., Young-Ha Oh, M.D.* Department of Orthopaedic Surgery, Department of Pathology, Guri Hospital, Hanyang University College of Medicine* Study Design: A case report. Objectives: To report a rare case of intradural disc herniation (IDH) around conus medullaris. Summary of Literature Review: IDH is rare with an incidence of less than 1% of all lumbar disc herniations. It is important to differentiate IDH from other condition with accurate diagnosis and subsequent surgical treatment. IDH has a higher risk of neurologic deficit, like conus medullaris syndrome and cauda equina syndrome. Materials and Methods: A 62 year-old male was affected by lumbar back pain radiating to the anterolateral aspect of the right thigh for 5 days. MRI showed a mass that existed on the anterior portion of the conus medullaris. We performed partial laminectomy at the L1- L2level. The mass located anteriorly in the intradural space was eliminated after durotomy by a posterior approach. Results: We confirmed the IDH for histopathology. Conclusions: IDH usually needs accurate differential diagnosis. Preoperative MRI scans are necessary to differentiate IDH from other intradural lesions. The confirmative diagnosis can be done only in the operative field. Key Words: Intradural disc herniation, Conus medullaris 경막내추간판탈출은추간판의수핵이경막내공간으로침범되어있는것으로정의하며발생빈도는추간판탈출증에서 0.27% 만을차지한다. 1) 이중요추부가 92% 로경막내추간판탈출의대부분을차지하며흉추부, 경추부에서의발생은매우드물다. 1) 요추부에서의발생빈도를자세히살펴보면제 4-5요추간, 제 3-4요추간, 제 5요추- 제 1천추간추간판탈출증의순으로높게나타나며제 2-3요추간및제 1-2요추간에는아주드물게발생하는것으로되어있다. 2,3) 이에본저자들은척수원추주변에서발생한경막내추간판탈출증을보고하고자한다. 증례보고 62세남자가내원 5일전등산중넘어지며발생한요통, 우측하지의방사통을주소로외래에내원하였다. 내원전에요통이나하지방사통등을경험하여병원치료를받은과거력은없었으며고혈압외에기저질환은없었다. 내원시환자는요추부의통증과우측대퇴부전외측으로저린감각을호소하였다. 이학적검사상상위요추부의경한압통이있었으며하지직거상검사 (Straight Leg Raise Test) 상우측하지 40도거상시양성소견보였고양측하지근력은정상소견을보였으며항문주위감각 (perianal sense) 과구해면체반사 (bulvocavernous reflex) 는정 상이었다. 단순방사선사진에서제 1-2 요추추간판높이의감 소소견이있었으며 (Fig. 1) 자기공명영상검사에서경막 (dura mater) 을경계로내측또는외측에존재하는지구분이불분명한 타원형의종물을확인할수있었다 (Fig. 2). 척수원추부위에발 생한종물로신경학적결손의진행가능성및감별진단을위해 수술적치료를시행하였다. 전신마취하에후방접근법으로제 Received: January 8, 2014 Revised: March 13, 2014 Accepted: June 12, 2014 Published Online: September 30, 2014 Corresponding author: Ye-Soo Park, M.D. Department of Orthopaedic Surgery, Guri Hospital, Hanyang University College of Medicine, 153, Gyungchun-ro(St), Guri city, Gyunggi-do, , Korea TEL: , FAX: hyparkys@hanyang.ac.kr This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License ( 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. 134 Copyright 2014 Korean Society of Spine Surgery

T1- and T2-weighted images in non-contrast sagittal and axial MRI (T1WI) (A, B). Gadoliniumenhanced T1-weighted MRI shows ring enhancement of the mass(c).

3 Journal of Korean Society of Spine Surgery Intradural Disc Herniation - A Case Report- Fig. 1. Preoperative AP (A) and lateral (B) x-rays. Intervertebral disc space of L1-2 is narrower than other intervertebral disc spaces. Fig. 2. T1- and T2-weighted images in non-contrast sagittal and axial MRI (T1WI) (A, B). Gadoliniumenhanced T1-weighted MRI shows ring enhancement of the mass(c). 1-2요추간까지도달한후추궁판 (lamina) 과황색인대 (ligament flavum) 를제거하고경막을노출시켰다. 미세현미경으로경막절개술 (Durotomy) 을시행하였고계란형종물을제거하였다 (6 x 10 x 16 mm, 0.35g) (Fig. 3). 병리조직생검으로추간판조직을확인하였다 (Fig. 4). 고찰 경막내추간판탈출증은평균 세에발병하며, 대부분요추에서발생하는데, 2/3에서마미증후군등의신경학적손상을동반하며드물기는하지만경추에서발생시심각한신경학적 135

Jaewon Lee et al Volume 21 Number 3 September 2014 Fig. 3. Intra-operative clinical photo shows intradural egg-shaped mass after dural incision(a, B). Fig. 4.

2,4) 병태생리학적으로추간판의섬유륜 (annulus fibrosus), 후방종인대 (posterior longitudinal ligament), 그리고경막이모두천공되어야발생하며이는선천적으로척수관이좁아서척추관탈출증이발생시쉽게척수주위조직을압박하게되어천공되거나해부학적으로추간판의섬유륜과후방종인대, 경막의전방부가함께유착되어있을경우발생한다고알려져있다.

4 Jaewon Lee et al Volume 21 Number 3 September 2014 Fig. 3. Intra-operative clinical photo shows intradural egg-shaped mass after dural incision(a, B). Fig. 4. H-E stained histologic findings show degenerative disc. (A) original magnification x 40, (B) original magnification x 200. 손상을가져올수있다. 2,4) 병태생리학적으로추간판의섬유륜 (annulus fibrosus), 후방종인대 (posterior longitudinal ligament), 그리고경막이모두천공되어야발생하며이는선천적으로척수관이좁아서척추관탈출증이발생시쉽게척수주위조직을압박하게되어천공되거나해부학적으로추간판의섬유륜과후방종인대, 경막의전방부가함께유착되어있을경우발생한다고알려져있다. 5,6) 원인으로써선천적또는후천적으로경막조직이얇아지거나수술, 외상, 감염, 노화등으로섬유륜과후방종인대, 경막전방부의유착을들수있으나아직까지원인은뚜렷이밝혀지지않았다. 경막내추간판탈출증의진단은가돌리늄 (gadolinium) 조영증강자기공명영상검사가유용하다. 경막내추간판탈출증은 T1, T2 강조영상에서모두추간판과동일한저신호강도 (low intensity signal) 를보이는경우가많으며시상면상에서후방종 인대의연속성이끊어져있을수있다. 또한후방섬유륜을통과하여탈출된추간판조직과추간판사이에줄기 (stalk) 가관찰될수있다. 7) Hidalgo-Ovejero 등 8) 은척수관내에서가스 (gas) 를확인할시에경막내추간판탈출증이동반되었을것이라고하였으며, 이가스는척추관의외상또는수술로기인할수있지만대부분탈출된추간판조직이경막을뚫고이동시같이이동하여존재하는경우라고하였다. Hida 등 9) 은 T2 강조축상영상의추간판공간레벨에서새부리모양의종물과함께가돌리늄조영시주위가반지 (ring) 형태로조영증강될때진단에도움이된다고하였다. 하지만탈출된추간판주위의조영증강은장시간이지난후주위에생긴육아조직으로인한것으로급성기에는육아조직이발달되지않아조영증강되지않을수있다. 본증례에서는 T1, T2 강조영상에서모두주위추간판과동일한신호강도로보이나후방종인대의연속성소실이나추간 136

5 Journal of Korean Society of Spine Surgery Intradural Disc Herniation - A Case Report- 판과연결되어있는줄기소견은보이지않았다. 다른특이점으로환자의증상이 5일전넘어지며갑자기발생하였다고진술하였음을고려할때급성기소견이보여야하지만가돌리늄조영 T1 강조영상에서반지형태의조영증강소견이뚜렷하게관찰되었다. 본증례는경막내추간판탈출증이거의발생하지않는척수원추주변에서발생하였고자기공명영상검사상경막내추간판탈출증을의미하는소견이뚜렷하지않았기에본증례에서는척수종양과감염성질환을반드시감별하여야했다. 경막내추간판탈출증과척수종양, 감염성질환의감별은각각의질환에따른치료방법과예후가달라지기때문에매우중요하다. 임상적으로척수종양은단순한배부통, 방사통, 서서히진행되는신경학적이상증상등비특이적으로나타나는경우가많으며감염성질환은균주에따라증상의발현속도가다르며척수종양과같이비특이적으로나타나는경우가많다. 감별진단시증상의급성발현과함께발열이동반되는경우에는급성감염성질환가능성이높으며이때적혈구침강속도 (erythrocyte Sedimentation Rate: ESR) 가일반적으로높고백혈구수치의상승은비특이적으로나타난다. 하지만아급성또는만성의경과를보이는경우, 대표적으로결핵균 (mycobacterium tuberculosis) 은증상이비특이적이다. 감염성질환일경우에는주위다른조직으로의침범이동반되어있는경우가많으며경막외공간 (epidural space) 에국한되어농양 (abscess) 형성시에경막내추간판탈출증과달리광범위한분절을침범하는경우가좀더많다. 10) 무엇보다도감별진단시가장중요한것은임상증상뿐만아니라환자의나이와성별, 동반된질환, 영상검사소견, 병변의병리조직소견등을종합적으로분석하여판단하여야한다는점이다. 본증례에서와같이경막내추간판탈출증이척수원추주변에발생하는경우신경학적결손을동반하여발생할가능성이높기때문에수술전후정확하고세심한이학적검사가필요하다. 수술전진단이어렵고, 척수종양및감염성질환과감별이필요하며수술중시행한병리조직을통해정확한진단이가능하다. 또한수술중병태생리학적특징으로인해주변구조물들과유착되어병변의절제시세심한주의를기울여야한다. 결론 대해수술적치료를통한임상증상의호전을가져온드문증례에대하여문헌고찰과함께보고한다. REFERENCES 1. Epstein NE, Syrquin MS, Epstein JA, Decker RE. Intradural disc herniations in the cervical, thoracic, and lumbar spine: report of three cases and review of the literature. J Spinal Disord. 1990;3: Kataoka O, Nishibayashi Y, Sho T. Intradural lumbar disc herniation. Report of three cases with a review of the literature. Spine (Phila Pa 1976). 1989;14: Connolly PJ, Rosenbaum AE, Sacks T, Kopacz KJ. Incomplete intradural lumbar disk herniation. Orthopedics. 1997;20: Eisenberg RA, Bremer AM, Northup HM. Intradural herniated cervical disk: a case report and review of the literature. AJNR Am J Neuroradiol. 1986;7: Blikra G. Intradural herniated lumbar disc. J Neurosurg. 1969;31: Yildizhan A, Pasaoglu A, Okten T, Ekinci N, Aycan K, Aral O. Intradural disc herniations pathogenesis, clinical picture, diagnosis and treatment. Acta Neurochir (Wien). 1991;110: Liu CC, Huang CT, Lin CM, Liu KN. Intradural disc herniation at L5 level mimicking an intradural spinal tumor. Eur Spine J. 2011;20 Suppl 2:S Hidalgo-Ovejero AM, Garcia-Mata S, Gozzi-Vallejo S, Izco-Cabezon T, Martinez-Morentin J, Martinez-Grande M. Intradural disc herniation and epidural gas: something more than a casual association? Spine (Phila Pa 1976). 2004;29:E Hida K, Iwasaki Y, Abe H, Shimazaki M, Matsuzaki T. Magnetic resonance imaging of intradural lumbar disc herniation. J Clin Neurosci. 1999;6: Tali ET, Gultekin S. Spinal infections. Eur Radiol. 2005;15: 제 1-2 요추간신경증상을동반하는경막내추간판탈출증에 137

6 Jaewon Lee et al Volume 21 Number 3 September 2014 척수원추주변에발생한경막내추간판탈출증 - 증례보고 - 이재원 서완식 박예수 오영하 * 한양대학교의과대학구리병원정형외과학교실, 병리과학교실 * 연구계획 : 증례보고목적 : 척수원추주변에발생한경막내추간판탈출증에대하여보고한다. 선행문헌의요약 : 경막내추간판탈출증은매우드문질환으로전체추간판탈출증중 1% 미만으로보고되고있다. 다른경막내종물과감별진단이필요하며본증례에서와같이척수원추주변에발생하는경우는매우드물고, 또한병변의위치로인해척수원추증후군 (conus medullaris syndrome) 이나마미증후군 (cauda equina syndrome) 등의신경학적손상을초래할수있어정확한진단과수술적제거가필요하다. 대상및방법 : 62세남자가내원 5일전부터시작된요추부의통증과우측대퇴부전외측으로저린감각을호소하였다. 자기공명영상검사에서척수원추부위에발생한종물을발견하였다. 후방접근법으로제1-2 요추간추궁판을일부제거하고경막에절개를하여경막내공간 (intradural space) 으로돌출되어있는종물을제거하였다. 결과 : 종물은병리조직생검을통해퇴행성추간판으로확진하였다. 결론 : 경막내요추부추간판탈출증은정확한감별진단을요한다. 수술전자기공명영상검사는다른경막내병변과감별하기위하여반드시필요하며확진은수술중가능하다. 색인단어 : 경막내추간판탈출증, 척수원추 약칭제목 : 경막내추간판탈출증 138

Lumbar spine

Lumbar spine CT 32 111 DOI : 10.3831/KPI.2010.13.2.111 Lumbar Spine CT 32 Received : 10. 05. 23 Revised : 10. 06. 04 Accepted : 10. 06. 11 Key Words: Disc herniation, CT scan, Clinical analysis The Clinical