Journal of Korean Society of Spine Surgery Paraplegia in an Ankylosing Spondylitis Patient with a Neglected Spine Fracture after Osteosynthesis for Fr

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Journal of Korean Society of Spine Surgery Paraplegia in an nkylosing Spondylitis Patient with a Neglected Spine Fracture after Osteosynthesis for Fracture of the Femur - Case Report - Jae Won You, M.D., Sin Wook Kang, M.D., Hong Moon Sohn, M.D. J Korean Soc Spine Surg 2017 Dec;24(4):246-251. Originally published online December 31, 2017; https://doi.org/10.4184/jkss.2017.24.4.246 Korean Society of Spine Surgery san Medical Center 88, Olympic-ro 43 Gil, Songpa-gu, Seoul, 05505, Korea Tel: +82-2-483-3413 Fax: +82-2-483-3414 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.2017.24.4.246 This is an Open ccess article distributed under the terms of the Creative Commons ttribution 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 Dec;24(4):246-251. https://doi.org/10.4184/jkss.2017.24.4.246 Paraplegia in an nkylosing Spondylitis Patient with a Neglected Spine Fracture after Osteosynthesis for Fracture of the Femur - Case Report - Jae Won You, M.D., Sin Wook Kang, M.D., Hong Moon Sohn, M.D. Department of Orthopaedic Surgery, School of Medicine, Chosun University, Gwangju, Korea Study Design: Case report. Objectives: To report a case of paraplegia in a patient with thoracic kyphosis after osteosynthesis for a fracture of the femur. Summary of the Literature Review: There are few reports about cases of paraplegia after low extremity fracture surgery in patients with thoracic kyphosis with ankylosing spondylitis. Materials and Methods: n 86-year-old female patient presented with right hip pain. She had undergone surgery for an intertrochanteric fracture of the femur in the supine position under general anesthesia. Immediately after surgery, she showed paraplegia. Postoperative thoracolumbar spine images revealed a fracture through the disc at T12 and L1. However, she did not complain of back pain or any neurologic deficits before surgery. Results: lthough the patient underwent emergent posterior decompression and fusion surgery, her neurologic compromise did not improve during 1 year of follow-up. Conclusions: It is necessary to check preoperative spine radiographs before surgery in elderly patients who have a kyphotic deformity and lower extremity fractures. Surgeons should consider changing the position of the patient and the type of anesthesia used during surgery when spine stability is in doubt. Key words: nkylosing spondylitis, Spine fracture, Lower extremity fracture, Paraplegia, Thoracic kyphosis 강직성척추염은전, 후종인대와황색인대및극간인대의골화와추간판의석회화로상하척추가융합되어탄력성, 운동성의상실과긴지렛대작용으로가장약한부위에응력이집중되고골다공증과동반되어사소한외상에도골절이잘발생한다. 강직성척추염은흉요추부의원형척추후만증 (round kyphosis) 를일으키고, 국소골절이있을경우예각상척추후만증 (acute angular kyphosis) 를일으키기도한다. 1) 후만변형이온상태에서골절이발생할경우주로추간판을통한골절부위전이전위가일어나신경학적증상이발생할수있고상하관절이모두유합되어있는상태라서골절부위가상대적으로매우불안정한상태라서가능한빨리고정수술을해야한다. 저자들이경험한증례는낙상에의해우측고관절전자간골절및추간판을통해제 12흉추- 제 1요추간골절및탈구가발생하였으나전혀신경학적인증상은없었다. 하지만전신마취및우측고관절골절수술에서앙와위자세를취하였고, 이로인한제 12흉추- 제 1요추간탈구악화로인한척수원추증후군이발생하여척추에대한응급수술시행하였던경우였다. 이 증례는고령의척추변형을동반한환자에서반드시척추방사 선사진을확인하고환자에따라수술자세의변화가필요함을 일깨워주는좋은경험이기에보고하는바이다. 본연구는본원기관생명윤리위원회의승인을얻어연구를 진행하였다 (IR No.2017-08-019). Received: ugust 28, 2017 Revised: September 5, 2017 ccepted: November 1, 2017 Published Online: December 31, 2017 Corresponding author: Hong Moon Sohn, M.D. ORCID ID: Jae Won You: https://orcid.org/0000-0002-7306-9813 Sin Wook Kang: https://orcid.org/0000-0003-0784-5076 Hong Moon Sohn: https://orcid.org/0000-0002-2121-9162 Department of Orthopaedic Surgery, Chosun University Hospital, 365 Pilmundae-ro, Dong-gu, Gwangju, 61453, Republic of Korea TEL: +82-62-220-3147, FX: +82-62-226-3379 E-mail: hmsohn@chosun.ac.kr * This study was supported by a research fund from Chosun University Hospital, 2013. 246 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 ccess article distributed under the terms of the Creative Commons ttribution 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 Paraplegia after Osteosynthesis for Fracture of the Femur 증례보고 86세여자환자로평소허리가좋지않아지팡이 1개이용하여보행하던자로계단에서넘어진이후발생한우측고관절통증이발생하여응급실로내원하였다. 기저질환으로고혈압외내과적질환은없었다. 환자는결핵감염의과거력없이점점진행하는척추후만변형으로내원 20여년전부터는직립보행이불가하였고, 수면시에도후만변형에의한상체를베개 3~4개정도로지지한다고하였다. 흉요추부의외상에대한병력청취결과본인은특별한외상병력이없다고하였다. 신체진찰에서우측대퇴골전자간골절로침상안정중이었고양측하지로신경학적이상소견은관찰되지않았다. 고관절의불안정골절로척추마취는포지션의제한으로인해시행하지못하여전신마취를시행하였다. 앙와위자세에서우측대퇴골전자간부의골 절 (oyd and griffin 분류 II, Evans unstable) (Fig. 1) 에대해근위대퇴골수정 (Proximal femur cephalomedullary nail) 을이용하여내고정시행하였다 (Fig. 1). 골절수술은골절수술대를이용하지않고일반적인수술테이블을이용하였으며골절의정복을위해심한견인력을가하지는않았다. 고관절수술직후병실에서시행한신체진찰에서양측서혜부에서부터하지로 Fig. 1. n intertrochanteric fracture of the right femur (oyd-griffin type II, Evans unstable) is observed on a preoperative pelvic radiograph (). Postoperative pelvic radiographs show that the fracture was fixed by a cephalomedullary nail (). Fig. 2. Lumbar spine radiography before hip surgery showed bamboo spine with extensive fusion across the entire spine (). On the lateral view, there was a gap between T12 and L1 (). Thoracolumbar lateral spine radiographs after hip surgery show a more displaced thoracolumbar fracture and deviated spine alignment (C). C www.krspine.org 247

Jae Won You et al Volume 24 Number 4 December 31 2017 완전한감각의소실및항문주위, 회음부, 족지와족장부등천골신경근분포영역의감각신경도완전히소실되어있었다. 양측고관절굴곡에서부터하지의주요근육의완전마비로미국척수손상학회 (merican Spinal Injury ssociation, SI) 의장애척도에따라모두 0등급에해당되어완전손상으로분류할수있었다. 환자의가족들도어머니의허리가곧게펴져완전한앙와위로누워있는자세를 20년만에보았다고하였다. 이에척추문제를의심하고수술전검사하였던척추방사선사진을다시확인하였다. 요추단순방사선상척추전체에걸쳐광범위한척추유합으로대나무척추 (bamboo spine) 소견을나타냈고 (Fig. 2), 측면사진에서제 12흉추와제 1요추사이간격이벌어져있었고, 제 2요추압박골절로제 1요추체가전방으로전위되어있어제 12흉추- 제 1요추사이의추간판을통한골절로제 1요추의전방전위가발생하였던것으로추정할수있었고, 흉요추부에원형척추후만증이관찰되었다 (Fig. 2). 고관절수술직후신경학적이상소견이인지된이후시행한흉-요추단순방사선사진상제 12흉추와제 1요추사이의디스크를통한골절및탈구가악화되었다 (Fig. 2C). 3차원컴퓨터단층촬영영상에서도제 1요추의전방굴곡으로인해척추관의연속성이소실되어있었고 (Fig. 3), 자기공명영상검사에서후방인대군의손상과함께척수및척수액의음영소실확인할수있 었다 (Fig. 4). 이를통해제 12흉추- 제 1요추의골절및탈구로발생한척추원추증후군으로진단할수있었다. 환자의마비를인지한이후약 8시간경과이후응급수술로후방접근법을통한골절의정복및후방기기고정술, 감압술, 골유합술을시행하였다. 수술소견에서제 1요추체가전하방으로전위되어있었고제 1요추의좌측척추경골절및우측횡돌기의골절그리고극돌기의골절소견관찰되었다. 제 11, 12흉추와제 1, 2, 3요추의양측척추경나사를삽입하고금속봉을연결후전하방으로전위된 L1에대해정복을위해신전시켜주었다. 이어 lamina 후방과횡돌기의골피질제거시행하여준이후동종골이식하여후방골유합술시행하여주었다 (Fig. 5). 수술이후의심되었던강직성척추염에대한평가위해진단기준을확인한결과양측천장관절면의경화및관절간격이좁아져있는 modified New York criteria grade III의대칭적인관절염으로관찰되었고, Fig. 4. Magnetic resonance imaging after hip surgery showed a discontinuity of the spinal canal due to fracture and dislocation of T12-L1 () and severe canal compromise (). Fig. 3. 3-dimensional reconstructed computed tomography scan after hip surgery revealed a wide range of upper and lower vertebral fusion and worsening of the thoracolumbar fracture in the anteroposterior view () and lateral view (). Fig. 5. Thoracolumbar spine radiographs after the emergent spine operation showed posterior instrumentation and posterolateral fusion of T11-L4 and restoration of the spine alignment in anteroposterior () and lateral views (). 248 www.krspine.org

Journal of Korean Society of Spine Surgery Paraplegia after Osteosynthesis for Fracture of the Femur Rheumatoid factor 는음성, HL 27는양성으로확인되어강직성척추염을진단할수있었다. 수술후신경학적회복은없었으며이후지속적으로침상안정상태로천추부의욕창이발생하여창상관리하고있는상태이다. 수술후 12개월경과한상태로수술전의지팡이이용한보행능력을회복하지못하고신경학적증상의호전은없다. 고찰 척추의후만변형은 Scheuermann 병, 선천성, 척수수막류등의질환과수술후후만변형, 외상성, 대사성, 골이형성및교원병, 종양, 그리고염증성변형등다양한원인에의한것으로알려져있다. 이중강직성척추염은흉요추부의원형척추후만증 (round kyphosis) 를일으키고, 국소골절이있을경우예각상척추후만증 (acute angular kyphosis) 를일으키기도한다. 1) 본증례는이중대표적인예로강직성척추염에의한척추골절및탈구가이미발생하였으나신경학적손상이전혀관찰되지않았던환자였고, 고관절골절수술을위해앙와위자세를하면서이전의척추골절및탈구가악화되어척수원추증후군이발생한예이다. 강직성척추염은전, 후종인대와황색인대및극간인대의골화와추간판의석회화로상하척추가융합되어탄력성, 운동성의상실과긴지렛대작용으로가장약한부위에응력이집중되어사소한외상에도골절이잘발생한다. 골절의호발부위는대부분경추하부이며흉추부및요추부의골절은경추부에비해빈도가낮으며각각 14%, 5% 로보고되고있다. 2) 경추부의전만및흉추부의후만변형이있어이미가동성이감소된척추의과다신전이수상의주된기전이다. Patel 등 3) 에의하면강직성척추염환자에서발생하는척추골절은대부분이응력이집중되는경추부의추간판을통한골절 (transdiscal fracture) 이며흉요추부의경우운동성이남아있는흉요추부이행부 (thoraco-lumbar junction) 에서잘발생하며추체를통한 (transvertebral) 골절은드물다고보고하였다. 강직성척추염의척추골절은매우불안정하며신경학적증상이경추골절의경우많게는 91% 에서동반되고, 흉-요추골절의경우빈도가경추골절에비하여드물지만 33% 의신경학적증상이동반된다고한다. 4) Good 5) 은전신마취하에고관절조작을한이후발생한요추골절및하반신마비가발생한강직성척추염환자를보고한바있다. Osgood 등 6) 은신경학적증상이있는경우에는감압술및내고정술을시행해야한다고주장하였다. 그리고치료시에강직성척추염에서발생한골절의치료는가벼운외상등에의한골절의발생으로과신전손상과같은손상기전에중점을두 어강직성척추염의병적특성인골다공증에대한고려가필요하다. 특히 Ghozlani 등 7) 은강직성척추염에서작은외상과함께골질의저하를동반한골다공증이강직성척추염에서척추골절이발생하는부수적인원인이아닌주원인이라고주장하였다. Olerud 등 8) 은골다공증이심한강직성척추염환자에서발생한제 12흉추의골절에대하여단순감압술및후방기기고정술후금속기기의이완을경험하였다고하며, 전후방고정술을동시에시행한경우기기의이완이없음을강조하며전후방고정술을시행하자고하였다. 저자들은후방접근법을통한골절의정복및후방기기고정술을시행하였고후궁판을충분히노출시켜망상골이최대한이식골과많이접촉할수있도록한후이식부위에충분한양의동종골이식을하는후외방골유합술을시행하였다. 저자들이경험한증례에서는수술부위가아닌척추손상을간과하여수술적치료이후하지마비가발생하였고이후이환기간의증가및재수술의위험도증가, 의료비증가라는손실이발생하였다. Danish 등 9) 은적절한처치를시행하여도신경학적회복이관찰되는예는드물어수술시행전일차적인예방을하는것이중요하다고하였다. 따라서강직성척추염이있는환자및심한후만변형이있는고령의환자에대해서는환자를이동시키거나, 자세변경, 전신마취하수술진행시반드시척추골절에대한위험성을인지하고대비하여야한다. REFERENCES 1. Labelle H, Roussouly P, erthonnaud É, et al. Spondylolisthesis, pelvic incidence, and spinopelvic balance: a correlation study. Spine (Phila Pa 1976). 2004 Sep;29(18):2049-54. DOI: 10.1097/01.brs.0000138279.53439.cc. 2. Murray GC, Persellin RH. Cervical fracture complicating ankylosing spondylitis: a report of eight cases and review of the literature. m J Med. 1981 May;70(5):1033-41. DOI: 10.1016/0002-9343(81)90860-3. 3. Patel SN, Turtz, Dixon, et al. Neurologically intact lumbar spine displaced fracture with ankylosing spondylitis. West J Emerg Med. 2011 Feb;12(1):142-3. 4. Chaudhary S, Hullinger H, Vives MJ. Management of acute spinal fractures in ankylosing spondylitis. ISRN Rheumatol. 2011 pr;2011(2011):1-9. DOI: 10.5402/2011/150484. 5. Good E. Nontraumatic fracture of the thoracic spine in ankylosing spondylitis. rthritis Rheum. 1967 Oct;10(5):467-9. DOI: 10.1002/art.1780100509. www.krspine.org 249

Jae Won You et al Volume 24 Number 4 December 31 2017 6. Osgood CP, bbasy M, Mathews T. Multiple spine fractures in ankylosing spondylitis. J Trauma 1975 Feb;15(2):163-6. DOI: 10.1097/00005373-197502000- 00011. 7. Ghozlani I, Ghazi M, Nouijai, et al. Prevalence and risk factors of osteoporosis and vertebral fractures in patients with ankylosing spondylitis. one. 2009 May;44(5):772-6. DOI: 10.1016/j.bone.2008.12.028. 8. Olerud C, Frost, ring J. Spinal fractures in patients with ankylosing spondylitis. Eur Spine J. 1996 Jan;5(1):51-5. DOI: 10.1007/F00307827. 9. Danish SF, Wilden J, Schuster J. Iatrogenic paraplegia in 2 morbidly obese patients with ankylosing spondylitis undergoing total hip arthroplasty. J Neurosurg Spine. 2008 Jan;8(1):80-3. DOI: 10.3171/SPI-08/01/080. 250 www.krspine.org

Case Report J Korean Soc Spine Surg. 2017 Dec;24(4):246-251. https://doi.org/10.4184/jkss.2017.24.4.251 강직성척추염이있는환자에서고관절골절수술이후발생한마비 - 증례보고 - 유재원 강신욱 손홍문조선대학교의과대학정형외과학교실연구계획 : 후향적연구목적 : 수술전흉추후만변형이있는환자에서대퇴골골절수술이후마비가발생한증례에대하여보고하고자한다. 선행연구문헌의요약 : 강직성척추염으로후만변형이있는환자에서하지골절수술후하지마비가발생한경우에대한보고는드물다. 대상및방법 : 86세여자환자가우측고관절통증으로내원하였다. 환자는전신마취하에앙와위로대퇴골전자간골절에대한수술을받았고수술직후양측하지마비가발생하였다. 수술후방사선촬영에서제 12흉추및 1요추추간판부위의골절이확인되었다. 수술전환자는요통이나하지신경학적증상을호소하지는않았었다. 결과 : 응급수술로후방감압술및유합술을받았으나 1년추시관찰후에도하지의마비는회복되지않았다. 결론 : 고령의척추변형을동반한환자에서하지골절이동반된경우반드시수술전에척추방사선사진을확인하고척추의안정성이의심될경우에는환자에따라마취및수술자세의변화에대한고려가필요합니다. 색인단어 : 강직성척추염, 척추골절, 하지골절, 하지마비, 흉추후만변형 약칭제목 : 대퇴골골절수술이후발생한마비 접수일 : 2017년 8월 28일수정일 : 2017년 9월 5일게재확정일 : 2017년 11월 1일교신저자 : 손홍문광주광역시동구필문대로 309 조선대학교의과대학정형외과학교실 TEL: 062-220-3147 FX: 062-226-3379 E-mail: hmsohn@chosun.ac.kr 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 ccess article distributed under the terms of the Creative Commons ttribution 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. 251