Journal of Korean Society of Spine Surgery Subacute Delayed Ascending Myelopathy after Spinal Cord Injury from Flexion-distraction Injury of Low Thora

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Journal of Korean Society of Spine Surgery Subacute Delayed Ascending Myelopathy after Spinal Cord Injury from Flexion-distraction Injury of Low Thoracic Spine - A Case Report - Sang Jae Park, M.D., Jae Hwan Cho, M.D., Sang Ik Shin, M.D., Bong-Soon Chang, M.D., Choon-Ki Lee, M.D. J Korean Soc Spine Surg 2013 Sep;20(3):123-128. Originally published online September 30, 2013; http://dx.doi.org/10.4184/jkss.2013.20.3.123 Korean Society of Spine Surgery Department of Orthopedic Surgery, Inha University School of Medicine #7-206, 3rd ST. Sinheung-Dong, Jung-Gu, Incheon, 400-711, Korea Tel: 82-32-890-3044 Fax: 82-32-890-3467 Copyright 2013 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:///doix.php?id=10.4184/jkss.2013.20.3.123 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.

Case Report pissn 2093-4378 eissn 2093-4386 J Korean Soc Spine Surg. 2013 Sep;20(3):123-128. http://dx.doi.org/10.4184/jkss.2013.20.3.123 Subacute Delayed Ascending Myelopathy after Spinal Cord Injury from Flexion-distraction Injury of Low Thoracic Spine - A Case Report - Sang Jae Park, M.D., Jae Hwan Cho, M.D., Sang Ik Shin, M.D., Bong-Soon Chang, M.D., Choon-Ki Lee, M.D. Department of Orthopedic Surgery, Seoul National University College of Medicine Study Design: A case report. Objectives: To report a rare case of subacute delayed ascending myelopathy. Summary of Literature Review: After low spinal cord injury, the cord injury may proceed to a proximal level and lead to subacute delayed ascending myelopathy. The patient suffered from orthostatic hypotension, weakness and sensory loss in the upper extremities and dyspnea. MRI showed more proximal progression of the spinal cord injury. There is no prevention or treatment for this condition. Materials and Methods: A 62-year-old man fell from heights and had 11th thoracic spine flexion-distraction injury. Upon arrival at the hospital, he was found to suffer from lower extremity weakness and sensory loss, but showed no neurologic symptom in his upper extremities. Two days later, we performed posterior instrumentation with fusion, and no postoperative neurologic symptom change was detected. One week after the fall, he suffered from dyspnea, upper extremity weakness and sensory loss. MRI was taken and we discovered that his spinal cord injury had proceeded to the 2nd cervical spine level. Results: Three months later, he showed little improvement in his upper extremity motor power, but not to the extent of the previous low spinal injury. Conclusion: Physicians should pay attention to the upper extremity and respiratory function of the patient with low spinal cord injury, because the level of spinal cord injury may proceed to a proximal level. Key Words: Subacute delayed ascending myelopathy, Low thoracic spine, Spinal cord injury 서론 흉추및요추부의외상에의한손상은척수손상을동반하는경우가흔히있다. 하부척수의손상이있은후수일에서수주후에손상부위보다근위부로척수손상이진행되는경우가드물게보고되고있으며이를아급성지연성상행성척수병증 (subacute delayed ascending myelopathy) 이라고한다. 기립성저혈압, 상지의근력및감각저하, 호흡곤란등의증상과동반하여자기공명영상에서수상당시의척수손상보다더근위부로손상이진행한양상을확인함으로써진단할수있으며심한경우경추부에까지이를수있어주의를요한다. 1-3) 예방법이나치료법은현재까지발표된바없으며한번진행한신경학적증상은원래수준까지호전되지는않는것으로알려져있다. 1,7) 저자들은드문질병인아급성지연성상행성척수병증을 1례경험하여이에대한경과를문헌고찰과함께보고하고자한다. Received: February 20, 2012 Revised: April 13, 2012 Accepted: October 18, 2012 Published Online: September 30, 2013 Corresponding author: Bong-Soon Chang, M.D. Department of Orthopedic Surgery, Seoul National University Hospital, Dae-hak-ro 101, Jongno-gu, Seoul, Korea TEL: 82-2-2072-3864, FAX: 82-2-764-2718 E-mail: bschang@snu.ac.kr 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. Copyright 2013 Korean Society of Spine Surgery 123

Sang Jae Park et al Volume 20 Number 3 September 2013 증례보고 Fig. 1. (A, B) Plain radiograph of D-L spine AP and Lateral. Showing T11 body and left pedicle, L2 body fracture 기저질환없이건강했던 62세남자환자가 3층높이에서낙상한후흉-요추부의통증및하지의마비증상이발생하여내원하였다. 환자는타병원을경유하여수상 30시간후본원으로전원되었다. 타병원에서자기공명영상을촬영한후척수손상을확인하여바로메틸프레드니솔론고용량치료 (30mg/kg 를 15분에걸쳐정맥주입한후 45분을쉰다음매시간당 5.4mg/kg 를 23시간에걸쳐정맥주입 ) 시행하였다. 본원내원당시신체검진상흉-요추이행부의심한통증을호소하였다. 감각은 T10 피부분절부위에서정상보다감소하기시작하였고 T12 피부분절부위이하로는전혀감각이없었다. 표재성복부근검사는정상이었으며대소변을보고자하는느낌이전혀없었으며구해면체반사도없었다. 하지의자발적운동은불가능하였다 (Grade 0). 하지의심부건반사는전혀나타나지않았으며바빈스키징후나 Fig. 2. Axial MRI of T10-11 spine, showing T11 left pedicle fracture Fig. 3. Sagittal T1 MRI of D-L spine. Showing T11, T12 and L2 body fracture 124

Journal of Korean Society of Spine Surgery Subacute Delayed Ascending Myelopathy after Low Spinal Cord Injury 족간대성경련도나타나지않았다. 하지의혈액순환에는문제가없었으며상지를포함한배꼽위쪽은감각이나운동기능의저하는전혀없었다. 혈액검사에서출혈경향은없었으며, 혈전이나색전증을의심할만한소견은없었다. 수상후 48시간이지나도대소변에대한느낌이돌아오지않았으며심부건반사도거의없었으며괄약근수축의징후및구해면체반사도없는상태가지속되었다. 본원내원당시촬영한단순방사선촬영및자기공명영상에서제11번흉추체및왼쪽후관절의골절, 제11번흉추의극돌기골절소견이보여굴곡-신연손상이있는것으로판단되었으며동반하여제 12흉추및제2번요추의압박골절소견이보였다. 또한시상면 T1 이미지에서제 5에서 12흉추체부위의광범위한범위에서척수의신호강도증가가관찰되어급성척수손상이동반된것으로보였다 (Fig. 1,2,3,4). 내원시촬영한경추부 CT에서는골절소견은보이지않았다 (Fig. 5). 수상 2일후제9-10-11-12 번흉추및제1번요추에대한후 방기기고정술및후방유합술을시행하면서제11번흉추의왼쪽후관절골절에대한후방감압술을시행하였다. 동반된제2 번요추에대해서는척추체성형술을동시에시행하였다 (Fig. 6). 수상 7일후 ( 술후 5일 ) 부터환자는양측상지의위약감및호 Fig. 5. Sagittal CT of C-spine. Showing no fracture Fig. 4. Sagittal T2 MRI of D-L spine. Showing increased signal intensity from T5 to T12. Fig. 6. (A, B) Plain radiograph of D-L spine AP and lateral. Showing Posterior instrumentation from T9 to L1, Kyphoplasty L2. 125

Sang Jae Park et al Volume 20 Number 3 September 2013 흡곤란을호소하였다. 신체검진에서근력을측정한결과삼각근 (C5) 은 5/5로유지되었으나상완이두근 (C5>C6) 은 3/3, 완관절신전근 (C6) 은 2/2, 상완삼두근 (C7) 은 2/2, 수지굴곡근 (C8) 및수지외전근 (T1) 은 1/1로감소되었다. 감각은양쪽젖꼭지부위 (T4) 에서전혀감각이없었으며약간위쪽 (T3) 에서감각이있었지만정상보다는감소되었다. 이에수상후 8일째자기공명영상을촬영하였고시상면 T2 영상에서고신호강도를보이는병변이제2번경추이하부위에서관찰되어아급성지연성상행성척수병증이발생된것으로판단되었다 (Fig 7, 8). 이에산소포화도및혈압을감시하면서산소투여및메틸프레드니솔론고용량치료 (30mg/kg 를 15분에걸쳐정맥주입한후 45분을쉰다음매시간당 5.4mg/kg 를 23시간에걸쳐정맥주입 ) 시행하였다. 이후환자의근력은수상후 11일 ( 술후 8일 ) 에상완이두근 (C5>C6) 은 4/4, 완관절신전근 (C6) 은 4/4, 상완삼두근 (C7) 은 3/3, 수지굴곡근 (C8) 은 2/2, 수지외전근 (T1) 은 1/1로호전되는 양상이었으며호흡곤란도호전되었다. 한편술후합병증으로장폐색이있어식이진행이어려웠으며재활훈련을위해기립경사테이블 (tilting table) 시행하였을때기립성저혈압으로약 30도정도기울였을때에도혈압이 60/40mmHg 까지떨어지는등어려움이있었다. 이에기립성저혈압치료위해미도드린 (midodrine) 처방하여사용하기시작하였다. 수상 1개월후에는상완이두근 (C5) 은 4/4, 완관절신전근 (C6) 은 4/4, 상완삼두근 (C7) 은 4/4, 수지굴곡근 (C8) 은 3/3, 수지외전근 (T1) 은 2/2 까지회복되었다. 감각은회복되지않아양쪽젖꼭지부위 (T4) 이하로전혀감각을느낄수없었다. 이때까지도장폐색및기립성저혈압이지속되어재활에어려움이많았다. 이후환자는타병원으로전원하여재활치료를지속하였고수상 6개월후평가했을때근력이나감각은술후 1달째와비교하여큰변화가없었으나기립성저혈압은미도드린 2.5mg 하루두번사용하면서기립경사테이블 60도까지혈압이떨어지지 Fig. 7. Sagittal T2 MRI of C-spine. Showing increased signal intensity of spinal cord from C2 to distal Fig. 8. Sagittal T2 MRI of T-spine. Showing increased signal intensity of spinal cord from fracture site. 126

Journal of Korean Society of Spine Surgery Subacute Delayed Ascending Myelopathy after Low Spinal Cord Injury 않고유지되었다. 침대형휠체어 (recline wheelchair) 로 50도정도상체를세워 30분가량거동가능하였다. 고찰 아급성지연성상행성척수병증은주로하부척수부위수상후수일에서수주후에수상부위보다근위부의감각저하를통해발견하게되며매우드문질환이다. 1-3) 1969 년 Frankel 7) 에의해서처음으로발표된후로증례보고가이어지고있다. 환자는주로기립성저혈압의증세를보이며병변의범위에따라흉부의감각저하, 상지의감각저하및근력의저하, 호흡곤란을호소할수있다. 자기공명영상 T2 강조영상에서수상부위보다근위부의척수에고신호강도를보이는병변을확인하여진단할수있다. 병변의범위는증례에따라다르게보고하고있는데, Schmidt 1) 는 35세여자환자에서제 11-12 흉추간의골절-탈구손상후수상 18일째에제5 경추부까지침범한증례를발표하였으며 Kumar 8) 등은 24세남자환자에서제 12흉추부의압박골절후수상 7일째에척수-연수연접부까지침범한증례를발표하였다. 아급성지연성상행성척수병증의원인으로는동맥을통한혈액공급에장애가생겨발생한다는가설과정맥을통한혈액의환류에장애가생겨발생한다는가설이있다. 2-6) 척수의혈류공급을담당하는가장큰동맥중의하나인 Adamkiewicz 동맥이하부흉추부에서기시하게되는데이부분에척추손상을받으면서혈전이생겨동맥을막아혈류의장애로인해발병한다는가설이있다. 3,5) 하지만 Belanger 2) 등은환자중한명에대해혈관조영술을해본결과 Adamkiewicz 동맥에혈전이없는경우에대해보고한바있다. 척수의정맥환류장애가아급성지연성상행성척수병증을일으킬수있다는가설도있다. 정상인에서는하대정맥의압력이척추측부정맥총의압력보다낮아척수를공급한혈액의정맥환류가잘일어나지만척수손상을입은환자의 68% 에서이압력이역전되어척수의혈액순환에장애가일어난다는보고가있었다. 6) 환자는수상후거동이불편하여침대에누워있게되며수술을받은후흉요천추보조기를착용하게되는데이로인한혈류장애를고려해볼수있겠으나이에대한연구는아직이루어지지않았다. 위에서보는바와같이척수를공급하는동맥과정맥의혈류의장애를그원인으로추측하고있으나아직명확하게밝혀진바는없다. 본증례는기존의증례들과같이기립성저혈압이있었으며 수상후일정한기간이지난후근위부로병변이진행하여증상이생겼다. 대부분의다른논문들의증례에서는비교적하부경추까지병변이진행되어호흡곤란에대한언급은없었고 Kumar 8) 등은척수-연수연접부까지병변이진행한증례를발표하였으나호흡곤란에대한언급은없었다. 본증례에서는병변의범위가제 2경추체부위까지포함되어호흡근력의저하로인한호흡곤란이있었다. 이에따라아급성지연성상행성척수병증이의심될때에는호흡부전에의한사망가능성이있으므로철저한혈압및산소포화도감시및산소투여, 필요에따라서는기관삽관에의한기도확보와중환자실치료등이필요하다. 아급성지연성상행성척추병증은보고된예가많지않은드문질환이지만간과할경우상지마비및호흡곤란으로사망에이르는경우도있어임상적으로중요하다. 환자가내원할당시에철저한신경학적검진을해야하며하부흉추손상이의심된다면시상면전장을포함하는자기공명영상을촬영하는것이필수적이다. 신경학적증상이근위부로진행하는경우에는추가적인자기공명영상촬영을통해척수손상의진행범위를파악하는것이필요하다. 이때척수손상의근위부침범범위에따라상지의근력저하, 호흡곤란, 기립성저혈압및장폐색등이발생할수있어이에대한보존적치료가필요하다. REFERENCES 1. Schmidt BJ. Subacute delayed ascending myelopathy after low spine injury: case report and evidence of a vascular mechanism. Spinal Cord. 2006;44:322-5. 2. Belanger E, Picard C, Lacerte D, Lavallee P, Levi AD. Subacute posttraumatic ascending myelopathy after spinal cord injury. Report of three cases. J Neurosurgery. 2000;93:294-9. 3. Aito S, El Masry WS, Gerner HJ, et al. Ascending myelopathy in the early stage of spinal cord injury. Spinal Cord. 1999;37:617-23. 4. Tator CH, Fehlings MG. Review of the secondary injury theory of acute spinal cord trauma with emphasis on vascular mechanisms. J Neurosurgery. 1991;75:15-26. 5. Sliwa JA, Maclean IC. Ischemic myelopathy: a review of spinal vasculature and related clinical syndromes. Arch Phys Med Rehabil. 1992;73:365-72. 6. Cassar-Pullicino VN, Colhoun E, McLelland M, Mc- Call IW, el Masry W. Hemodynamic alterations in the 127

Sang Jae Park et al Volume 20 Number 3 September 2013 paravertebral venous plexus after spinal injury. Radiology. 1995;197:659-63. 7. Frankel HL. Ascending cord lesions in the early stages following spinal injury. Paraplegia. 1969;7:111 8. 8. Kumar A, Kumar J, Garg M, Farooque K, Gamanagatti S, Sharma V. Posttraumatic subacute ascending myelopathy in a 24-year-old male patient. Emerg Radiol. 2010;17:249 52. 하부흉추부의굴곡 - 신연손상으로인한척수손상후에발생한아급성지연성상행성척수병증 - 증례보고 - 박상재 조재환 신상익 장봉순 이춘기서울대학교의과대학정형외과학교실 연구계획 : 증례보고 목적 : 드문척수손상중의하나인아급성지연성상행성척수병증환자를 1 례경험하였기에보고하고자한다. 선행문헌의요약 : 하부척수의손상후손상부위보다근위부로척수손상이진행되는경우가드물게보고되며이를아급성지연성상행성척수병증 (subacute delayed ascending myelopathy) 이라고한다. 기립성저혈압, 상지의근력및감각저하, 호흡곤란등의증상과동반하여자기공명영상에서 수상당시보다더근위부로척수손상이진행한양상을확인함으로써진단할수있다. 대상및방법 : 62 세남자환자가 3 층높이에서낙상하여제 11 번흉추부의굴곡 - 신연손상으로인한척수손상을받았다. 내원당시하지마비가있었으 나상지의신경학적이상은없었다. 수상 2 일후후방유합술및기기고정술시행하였으며수술전후로신경학적증상의변화는없었다. 수상 1 주후호 흡곤란, 상지의감각이상및위약감을호소하여자기공명영상을촬영하였고제 2 번경추이하부위의척수까지손상이진행된것이관찰되었다. 결과 : 수상후 3 개월까지경과관찰하여호흡곤란및상지의감각이상, 위약감이조금호전되었으나초기의하부흉추손상시의수준까지회복되지는 못하였다. 결론 : 하흉추부척수손상이있더라도상지의신경기능이나호흡기능평가에대해세심한주의를기울여야할것으로사료된다. 색인단어 : 아급성지연성상행성척수병증, 하부흉추, 척수손상 약칭제목 : 하부척수손상후에발생한아급성지연성상행성척수병증 128