Journal of Korean Society of Spine Surgery Acute Epidural Hematoma Following Cervical Spinal Fracture in a Patient with Ankylosing Spondylitis Sang Bu

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Journal of Korean Society of Spine Surgery Acute Epidural Hematoma Following Cervical Spinal Fracture in a Patient with Ankylosing Spondylitis Sang Bum Kim, M.D., Youn Moo Heo, M.D., Byung Hak Oh, M.D., Tae Gyun Kim, M.D., You Sun Jung, M.D. J Korean Soc Spine Surg 2017 Mar;24(1):44-48. Originally published online March 31, 2017; https://doi.org/10.4184/jkss.2017.24.1.44 Korean Society of Spine Surgery Department of Orthopedic Surgery, Gangnam Severance Spine Hospital, Yonsei University College of Medicine, 211 Eunju-ro, Gangnam-gu, Seoul, 06273, Korea Tel: 82-2-2019-5410 Fax: 82-2-573-5393 Copyright 2017 Korean Society of Spine Surgery pissn 2093-4378 eissn 2093-4386 The online version of this article, along with updated information and services, is located on the World Wide Web at: http://www.krspine.org/doix.php?id=10.4184/jkss.2017.24.1.44 This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http:// creativecommons.org/licenses/by-nc/4.0) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited. www.krspine.org

Case Report J Korean Soc Spine Surg. 2017 Mar;24(1):44-48. https://doi.org/10.4184/jkss.2017.24.1.44 Acute Epidural Hematoma Following Cervical Spinal Fracture in a Patient with Ankylosing Spondylitis Sang Bum Kim, M.D., Youn Moo Heo, M.D., Byung Hak Oh, M.D., Tae Gyun Kim, M.D., You Sun Jung, M.D. Department of Orthopedic Surgery, Konyang University College of Medicine, Daejeon, Korea Study Design: Case report. Objectives: To report a case of epidural hematoma following cervical spinal fracture in a patient with ankylosing spondylitis. Summary of Literature Review: An early surgical intervention for acute epidural hematoma following cervical spinal fracture led to improvements in the patient s neurological deficits. Materials and Methods: A 76-year-old male with ankylosing spondylitis presented with neck pain and motor weakness of both upper and lower extremities after falling. He sustained fractures of the C7 body and the spinous processes of C5 and C6. Magnetic resonance imaging showed an extensive epidural hematoma from C7 to T5. The authors performed decompression from C6 to T2, and posterior instrumentation and fusion from C4 to T3. Results: An urgent surgical intervention was performed, and a good result was obtained. Conclusions: The authors describe an early surgical intervention in a case of acute epidural hematoma following cervical spinal fracture in a patient with ankylosing spondylitis. Key words: Ankylosing spondylitis, Epidural hematoma, Cervical spinal fracture 서론 척추외상에동반된경막외혈종의경우자발성경막외혈종보다는빈도가적어약 1.7% 정도로보고되고있으나강직성척추염이나류마티스관절염과같은척추질환이있을경우발생빈도가더높은것으로알려져있다. 경추골절에동반하여신경학적증상이있을경우경막외혈종뿐만아니라디스크관련질환및종양, 농양등의병변과의감별및신속한진단을위해자기공명영상검사를조기에시행하는것이도움이된다. 1) 다른척추의질환들과마찬가지로외상에의한경막외혈종에의하여진행성신경학적증상을보이는경우수술을통한빠른감압술이치료원칙이며, 신경학적증상이경미하거나환자의상태가수술을고려하기힘든상황일경우에는보존적치료를고려해볼수있다. 2) 저자들은강직성척추염환자에서경추골절과동반되어발생한경막외혈종에대해빠른수술적치료를통해우수한결과를얻었기에이를문헌고찰과함께보고하고자한다. 증례보고 평소독립보행이가능하였던 76 세강직성척추염남자환자 가침상에서낙상하며발생한경부통증및사지의근력과감각 저하로내원하였다. 고혈압및당뇨과거력이있으며아스피린 을복용중이었던환자로, 내원시신체검진상에서양측주관절 굴곡근 (C5), 수근신전근 (C6) 및주관절신전근 (C7) 에서근력 4 단계, 양측수지굴곡근 (C8) 및수지외전근 (T1) 이근력 2-3 단계, 양측고관절굴곡근 (L2), 슬관절신전근 (L3), 족배굴곡근 (L4), 족무지신전근 (L5), 및족저굴곡근 (S1) 에는근력 1 단계 측정되었다. 수의적항문조임은측정되지않으나, 구해면체 반사는양성소견을보였다. 감각은전신의가벼운촉각은정상 이나침통각은제 2 흉추이하로측정되지않았다. 수상후촬영한단순방사선검사상척추전반적으로죽상척 주죽상형척추 (bamboo spine) 및석회화, 제 2-5 경추에이르 Received: August 12, 2016 Revised: October 27, 2016 Accepted: February 2, 2017 Published Online: March 31, 2017 Corresponding author: Byung-Hak Oh, M.D. Department of Orthopedic Surgery, Konyang University Hospital, 158 Gwanjeodong-ro, Seo-gu, Daejeon 35365, KoreaDaejeon, Korea TEL: +82-42-600-9863, FAX: +82-42-600-9090 E-mail: sebslab@hanmail.net 44 Copyright 2017 Korean Society of Spine Surgery Journal of Korean Society of Spine Surgery. www.krspine.org. pissn 2093-4378 eissn 2093-4386 This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/4.0/) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

Journal of Korean Society of Spine Surgery Traumatic Epidural Hematoma with Ankylosing Spondylitis Patient A B A Fig. 2. Postoperative radiologic findings. (A) A plain radiograph shows in situ posterior fixation from C4 to T3. (B) Cervical spine computed tomography shows adequate posterior decompression from C6 to T2. B C Fig. 1. Initial radiologic findings taken 2 hours after the trauma. (A) A lateral X-ray shows the ankylosed cervical spine. (B) Computed tomography shows fracture of the C7 body. (C, D) On magnetic resonance imaging, a T2-weighted sagittal view and a Dixon view delineate a posterior epidural lesion with an iso to high signal from C7 to T5, corresponding to acute hematoma. 는후종인대골화증소견을보였으나골절의소견은없었다. CT 및자기공명영상에서제 7 경추체및제 5, 6 경추극돌기골절소 견관찰되었으며 T1 에서중등도의신호및 T2 에서고신호강도 를보이는병변이제 7 경추에서제 5 흉추에걸쳐관찰되어, 저자 들은이를급성경막외혈종에합당한소견으로판단하였다 (Fig. 1). 진행하는신경학적증상의악화에근거하여내원후약 2 시간 경과후응급수술을시행하였으며제 6 경추에서제 2 흉추에걸 친후궁절제및혈종제거를시행하였다. 제 7 경추체골절에대 해서는제 4 경추에서제 3 흉추까지후방기기고정술및유합술 D 을시행하였다. 과신전으로인한신경손상의방지를위하여손상전상태를감안한원위치 (in situ) 로고정술을시행하였으며, 술후시행한전산화단층촬영상에서감압및견고한고정을확인할수있었다 (Fig. 2). 과거력상의아스피린복용은예방적목적인것으로확인되어수술후중단하였다. 내원시 1단계였던하지근력은술후 3주째 3단계로호전되었다. 환자는술후 2개월째보행기를이용한보행까지가능한상태로퇴원하였으며, 지속적재활및운동치료를시행하였다. 술후 1년외래추시상특이소견없는상태로지팡이보행이가능한상태까지기능회복하였다. 양하지근력은 4단계로호전되었으며지팡이보행이가능한상태로회복되었다. 고찰 강직성척추염은류마티스질환중하나로유병률은 0.1~0.5% 로드문질환이나, 이중 6% 에서일생중척추의골절이발생하며이는일반인에비해약 4배정도높은빈도로알려져있다. 1,3,4) 이는강직성척추염의초기에도발생할수있는골다공증및죽상형척추죽상형척추 (bamboo spine) 가원인으로, 골밀도감소에의해약해진척추의생체역학의변화로인하여매우미세한손상이나약한강도의외상에의해서도골절이생겨치명적합병증이발생할수있다. 특히유병기간이길고척추의유합이있는환자에서치사율과사망률이높은데, 이경우신경손상가능성이 29~91%, 사망률도 35% 로높은확률을보이는것으로알려져있으며, 5) 이역시약해진골조직내신경조직이정상골조직내에있는신경조직의경우보다정상보다낮은 www.krspine.org 45

Sang-Bum Kim et al Volume 24 Number 1 March 2017 골밀도로인해골절가능성이높아지고따라서신경조직의손상가능성도높아지기때문이다. 6) 특히경추부에발생한갑작스런동통과진행하는신경학적증상은경추부의골절및이로인한경추부경막외혈종을의심할수있는지표가되며, 완전마비로급격히진행할수있어주의를요한다. 즉, 완전마비로진행하는증상은빠르게커지는혈종에의해척수가압박되어나타나는증상임을시사할수있으므로초기에적절한검사와치료가필수적이다. 7,8) 그러므로조기에자기공명영상검사를통하여경막외혈종과경막하또는지주막하출혈, 추간판탈출증과척수부종등을감별해야할뿐만아니라, 주위연부조직손상이나척수의상태를진단하여야한다. 8) 또한환자의과거력혹은내과적질환파악을통하여항응고제등을복용한경력이확인된다면더욱더출혈의가능성을염두에두어야할것으로생각한다. 본증례의경우아스피린을복용하던환자였기에출혈경향을감안하여신속한정밀검사를시행하였다. 항응고제의중단여부는동반질환의중증도를고려하여결정하여야하나이는추가적인연구가필요할것으로생각한다. 경추부경막외혈종의수술적치료는일반적으로빨리시행할수록신경학적회복의정도가우수한것으로알려져있다. 술후신경학적인회복의정도에관여하는여러요인들중에증상이발생한시점과관련하여수술시기가가장중요하며, 수상후 12 시간이내에감압술을시행하는것이가장좋은예후를보인다고한다. 9) 골절의치료에대해서는여러의견이많은데, 강직성척추염에서골절이발생하는경우삼주 (three column) 모두를침범하는불안정골절인경우가흔하며이로인한전위가동반된경우에는견고한내고정및유합술이보편적으로추천된다. 8) 하지만신경학적결손이없거나악화되지않고임상증상이호전되는양상을보이는안정성골절및경막외혈종의경우에는보존적치료도고려될수있다. Jeon 등 10) 은강직성척추염환자에서전위없는경추골절과동반되어발생한경막외혈종을 Halo vest 보조기등을통한보존적치료사례를보고한바있다. 저자들의증례에서는환자가수상후내원하였을때단순방사선상특이소견이관찰되지않았으나신경학적결손이발생한상태로내원하였기에신속한 CT 및 MRI 검사를통하여급성경막외혈종을정확히진단할수있었다. 또한환자의항응고제복용력및내원후에도급격히악화되는신경학적증상으로미루어볼때보존적치료로증상의호전을기대하기어려울것으로판단하여신속한수술적치료를결정하였고, 이에따라양호한결과를얻을수있었던것으로생각한다. 결론 광범위한척추의강직과유합이발생한강직성척추염환자의경우, 약한강도의외상으로도척추골절이발생할수있으며이로인한합병증발생및사망률이높은것으로알려져있다. 외상후경추부통증과함께신경학적증상을보이는강직성척추염환자에서는신속히영상평가를시행하여경추부골절및경막외혈종등의여부를감별하는것이필요하다. 저자들은강직성척추염환자에서경추부골절과동반하여진행하는신경학적증상을보이는경막외혈종을자기공명영상을통하여빠르게진단하고즉각적인수술적치료를시행하여좋은결과를얻었기에이를문헌고찰과함께보고하는바이다. REFERENCES 1. Qunfeng G, Yidong C, Liang W, et al. Single anterior approach for cervical spine fractures at C5-T1complicating ankylosing spondylitis. Clinical neurology and Neurosurgery. 2016;147:1-5. 2. Gourav G, Rambir S, Kishan R. Anticoagulant induced spontaneous spinal epidural hematoma, conservative management or surgical intervention A dilemma. J. Acute medicine. 2016;6:38-42. 3. Mundwiler ML, Siddique K, Dym JM, et al. Complications of the spine in ankylosing spondylitis with a focus on deformity correction. Neurosurgical Focus. 2008;24:E6. 4. Westerveld LA, Verlaan JJ, Oner FC. Spinal fractures in patients with ankylosing spinal disorders: a systematic review of the literature on treatment, neurological status and Complications. Eur Spine J. 2009;18:145 56. 5. Vosse D, Feldtkeller E, Erlendsson J, et al. Clinical vertebral fractures in patients with ankylosing spondylitis. J Rheumatology. 2004;31:1981-5. 6. Gartman JJ Jr, Bullitt E, Baker ML. Axis fracture in ankylosing spondylitis: case report. Neurosurgery. 1991;29:590-4. 7. Duffill J, Sparrow OC, Millar J, et al. Can spontaneous spinal epidural hematoma be managed safely without operation? A report of four cases. J Neurol Neurosurg Psychiatry. 2000;69:816-9. 8. Chaudhary SB, Hullinger H, Michael JV. Management of Acute Spinal Fractures in Ankylosing Spondylitis. ISRN Rheumatol. 2011 Jun 30. 46 www.krspine.org

Journal of Korean Society of Spine Surgery Traumatic Epidural Hematoma with Ankylosing Spondylitis Patient 9. Chong KH, Hsu HT, Tu CW, et al. Spontaneous spinal epidural hematoma: Early surgical intervention provides ideal neurological outcome. Formosan J of Surgery. 2014;47:156-9. 10. Jeon TS, Chung WY, Lee WS, et al. Spinal Epidural Hematoma Associated with Cervical Fracture in Ankylosing Spondylitis. J Korean spine surg. 2003;10:191-5. www.krspine.org 47

Case Report J Korean Soc Spine Surg. 2017 Mar;24(1):44-48. https://doi.org/10.4184/jkss.2017.24.1.48 강직성척추염환자의경추골절에서발생한급성경막외혈종 김상범 허윤무 오병학 김태균 정유선건양대학교의과대학정형외과학교실연구계획 : 증례보고목적 : 강직성척추염환자의경추골절에서발생한급성경막외혈종의증례보고선행문헌의요약 : 강직성척추염환자에발생한경추골절로생긴경막외혈종에서조기의수술적치료로좋은결과를얻었다. 대상및방법 : 강직성척추염환자인 76세남자로낙상후발생후경부통증및사지의근력저하를보였다. 단순방사선및 CT 검사상제 7경추추체와제 5, 6경추극돌기골절이확인되었으며, 자기공명영상상제7경추에서제 5흉추에걸쳐경막외혈종소견확인되었다. 이에저자들은제 6경추에서제 2흉추까지감압술및제 4흉추에서제 3흉추까지후방기기고정술및유합술을시행하였다. 결과 : 조기수술로서좋은결과를얻을수있었다. 결론 : 저자들은강직성척추염환자에서경추골절과동반되어발생한경막외혈종의치험사례에대하여보고하고자한다. 색인단어 : 강직성척추염, 경막외혈종, 경추골절 약칭제목 : 강직성척추염환자에발생한외상성경막외혈종 접수일 : 2016년 8월 12일 수정일 : 2016년 10월 27일 게재확정일 : 2017년 2월 2일 교신저자 : 오병학 대전광역시서구관저동로 158 건양대학교의과대학정형외과학교실 TEL: 042-600-9863 FAX: 042-600-9090 E-mail: sebslab@hanmail.net 48 Copyright 2017 Korean Society of Spine Surgery Journal of Korean Society of Spine Surgery. www.krspine.org. pissn 2093-4378 eissn 2093-4386 This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/4.0/) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.