Journal of Korean Society of Spine Surgery Kyphotic Deformity after Spinal Fusion in a Patient with Diffuse Idiopathic Skeletal Hyperostosis - A Case

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Journal of Korean Society of Spine Surgery Kyphotic Deformity after Spinal Fusion in a Patient with Diffuse Idiopathic Skeletal Hyperostosis - A Case Report - Jaedong Kim, M.D., Jaewon Lee, M.D., Ye-Soo Park, M.

1 Journal of Korean Society of Spine Surgery Kyphotic Deformity after Spinal Fusion in a Patient with Diffuse Idiopathic Skeletal Hyperostosis - A Case Report - Jaedong Kim, M.D., Jaewon Lee, M.D., Ye-Soo Park, M.D. J Korean Soc Spine Surg 2017 Jun;24(2): Originally published online June 30, 2017; Korean Society of Spine Surgery Asan Medical Center, 88 Olympic-ro 43 Gil, Songpa-gu, Seoul, 05505, Korea Tel: Fax: Copyright 2017 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/4.0) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

2 Case Report J Korean Soc Spine Surg Jun;24(2): Kyphotic Deformity after Spinal Fusion in a Patient with Diffuse Idiopathic Skeletal Hyperostosis - A Case Report - Jaedong Kim, M.D., Jaewon Lee, M.D., Ye-Soo Park, M.D. Department of Orthopedic Surgery, Guri Hospital, Hanyang University College of Medicine, Guri, Korea Study Design: Case report. Objectives: To report a case of progressive kyphotic deformity after spinal fusion in a patient with diffuse idiopathic skeletal hyperostosis (DISH). Summary of Literature Review: DISH is characterized by spinal and peripheral enthesopathy, and is a completely different disease from ankylosing spondylitis (AS). Though DISH can be associated with thoracic kyphosis, no reports have described a progressive thoracolumbar kyphotic deformity after spinal fusion surgery in a DISH patient. Materials and Methods: A 47-year-old male presented with pain in the thoracolumbar region. After excluding the possibility of AS and confirming the diagnosis of DISH, we performed spinal fusion for the treatment of a T11-T12 flexion-distraction injury. The kyphotic deformity was found to be aggravated after the first operation, and we then performed corrective osteotomy and additional spinal fusion. Results: The kyphotic deformity of the patient was corrected after the second operation. Conclusions: In DISH patients in whom AS must be excluded in the differential diagnosis, a kyphotic deformity can become aggravated despite spinal fusion surgery, so regular and continuous follow-up is required. Key words: Diffuse idiopathic skeletal hyperostosis (DISH), Kyphotic deformity 미만성특발성골격과골증 (Diffuse Idiopathic Skeletal Hyperostosis, DISH) 은비염증성척추관절증으로척추의관절강직증과골부착부병증을보인다. 1) 미만성특발성골격과골증은강직성척추염 (Ankylosing spondylitis, AS) 과근본적으로다른질환으로임상적, 방사선학적특징을통해감별할수있다. 2) 미만성특발성골격과골증과강직성척추염을감별하는데도움이되는소견으로는미만성특발성골격과골증은강직성척추염에비하여상대적으로발병나이가늦고임상적으로추체부의통증과추체관절운동범위의제한을환자가호소하나일반적으로강직성척추염에비해서는그정도가경미하다. 2) 강직성척추염은천장골관절의하부를침범하는반면, 미만성특발성골격과골증은상부의전인대를비대칭적으로침범하고강직성척추염에서는특징적으로척추체의사각화 (squaring) 나골단성관절의강직이있는반면, 미만성특발성골격과골증에서는해당소견이없다. 2) HLA B-27 검사결과에있어서도강직성척추염에서는대부분양성소견을보이나미만성특발성골격과골증에서는매우드물다. 2) 후종인대의골화는강직성척추염보다는미만성특발성골격과골증에서더 흔한증상이다. 2) 척추후만변형은나이, 성별, 외상력등다양한원인에의해 발생할수있고미만성특발성골격과골증과척추후만변형의 관련성은기존의보고에서찾을수있으나 3,4) 미만성특발성골 격과골증으로진단된환자에서척추유합술을시행후진행하 는척추후만변형을보고한사례는없었다. 이에저자들은강 직성척추염과감별을요하는미만성특발성골격과골증환자 에서골절로인한수술적치료후진행하는후만변형 1 예를경 험하고치료하였기에문헌고찰과함께보고하고자한다. Received: November 5, 2016 Revised: December 30, 2016 Accepted: April 25, 2017 Published Online: June 30, 2017 Corresponding author: Ye-Soo Park, M.D. ORCID ID: Department of Orthopedic Surgery, Hanyang University Guri Hospital, 153 Gyeongchun-ro, Guri , Korea TEL: , FAX: hyparkys@hanyang.ac.kr Copyright 2017 Korean Society of Spine Surgery Journal of Korean Society of Spine Surgery. pissn eissn This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License ( which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited. 103

Jaedong kim et al Volume 24 Number 2 June 2017 A B C Fig. 1. Preoperative radiographs. (A) Anteroposterior view of a thoracolumbar spine radiograph shows osteophytes connecting the thoracic vertebrae.

3 Jaedong kim et al Volume 24 Number 2 June 2017 A B C Fig. 1. Preoperative radiographs. (A) Anteroposterior view of a thoracolumbar spine radiograph shows osteophytes connecting the thoracic vertebrae. (B) Lateral view of a thoracolumbar spine radiograph shows a melted candle wax appearance. (C) An axial computed tomography image of the pelvis shows no evidence of fusion, erosion, or sclerosis of the sacroiliac joint. 증례보고 47세남성이흉요추부동통을주소로내원하였다. 환자는평소특별한허리통증없이지내던자로내원전계단에서넘어진후흉-요추이행부의심한통증이시작되었다고하였다. 내원당시생체징후는안정된상태로이학적소견상흉-요추이행부에동통및운동제한이있었으나신경학적검사상상하지의감각및운동마비는없었다. 내원후시행한흉요추부단순방사선검사상전후면영상에서인접한척추의외측변연을이어주는골화소견이관찰되며 Bamboo Spine 소견과유사하게관찰되었고측면영상에서 melted candle wax 소견이관찰되었으며흉추부추간판높이는비교적보전되어있었다 (Fig. 1A, B). 천추부전산화단층촬영소견상천장관절의미란, 경화, 융합소견은관찰되지않았다 (Fig. 1C). 흉요추부전산화단층촬영및자기공명영상소견상제 11, 12흉추체의골절과극돌기골절소견이보여 (Fig. 2) 제 흉추간의굴곡-신연손상을진단하였다. 미만성특발성골격과골증와강직성척추염을감별하기위하여 HLA-B27 를추가로시행하였고검사결과음성소견을보였다. 증상의발현시점이비교적늦고영상소견상흉추부위에인접한 4개이상의척추전인대의골화가뚜렷하며추간판높이가비교적보전되어있고천장관절의미란, 경화, 융합등의소견은보이지않았다는점과 HLA-B27 검사상음성소견을종합하여강직성척추염의가능성을배제하고, 미만성특발성골격과골증을진단하였다. 제 흉추의굴곡-신연손상에대하여제 11흉추관혈적정복과제 10흉추- 제 1요추의분절간척추경고정술및척추유합술을시행하였다. 수술중이나수술후에별다른합병증은없었고수술직후시행한단순방사선사진에서흉추후만각은 49도, 요추전만각 -35도, 시상수직축은 8.6 mm로확인되었고골반입사각은 65.2도, 골반경사 22.6도, 골반기울기 42.6도로확인되었다 (Fig. 3A). 이후환자는본원외래에내원하지않다가수술후 2년이지나서흉요추부의통증및진행하는척추후만변형으로인한일상생활의제한을주소로재내원하였다. 당시시행한단순방사선사진에서흉추후만각은 52도, 요추전만각 -41도, 시상수직축은 69.3 mm로확인되었고골반입사각은 65.2도, 골반경사 21.0도, 골반기울기 64.2도로확인되었다 (Fig. 3B). 환자가호소하는보행및전방주시장애를개선하고자교정수술을결정하였고교정각도는수술전 Surgimap spine software(nemaris, Inc., New York, NY, USA) 을이용한컴퓨터시뮬레이션을통해결정하였다. 5) 시뮬레이션결과교정술기는다분절 Smith-Peterson 절골술을시행하기보다는한분절 Smith-Peterson 절골술과척추경후방절골술을동시에시행하는것이보다좋은교정각을보여기존삽입물을제거하고제 11-제 12흉추의 Smith-Peterson 절골술과제 2요추의척추경후방절골술 (Pedicle subtraction osteotomy) 을시행하였고제 10흉추-제 4요추까지척추유합술을시행하였다. 수술후단순방사선검사에서흉추후만각은 42도, 요추전만각 -50도, 시상수직축은 13.1 mm로측정되었고골반입사각은 65.2도, 골반경사 22.7도, 골반기울기 42.5도로확인되었으며 (Fig. 3C) 104

$(A) Sagittal CT shows a fracture of the vertebral body of T11 and T12. (B) Sagittal CT (arrowheads) shows a fracture of the spinous process of T11.$ $(C) Three-dimensional CT shows the fracture of the vertebral body of T11 and T12.$

4 Journal of Korean Society of Spine Surgery Kyphotic Deformity After Spinal Fusion in DISH A B C D Fig. 2. Preoperative CT and MRI. (A) Sagittal CT shows a fracture of the vertebral body of T11 and T12. (B) Sagittal CT (arrowheads) shows a fracture of the spinous process of T11. (C) Three-dimensional CT shows the fracture of the vertebral body of T11 and T12. (D) Sagittal MRI shows the fracture of the vertebral body and spinous process of T11 and the fracture of the vertebral body of T12. All the images indicate a T11-T12 flexion-distraction injury. CT, computed tomography; MRI, magnetic resonance imaging. 현재외래추시중이다. 고찰 A B C Fig. 3. (A) A radiograph taken 2 months after the first operation, showing a sagittal vertical axis (dashed line) of 8.6 mm and a thoracic kyphosis angle (solid line) of 49. (B) A radiograph taken 2 years after the first operation, showing a sagittal vertical axis (dashed line) of 69.3 mm and a thoracic kyphosis angle (solid line) of 52 (C). A radiograph taken 2 months after the second operation, showing a sagittal vertical axis (dashed line) of 13.1 mm and a thoracic kyphosis angle (solid line) of 42. 미만성특발성골격과골증은척추와척추외관절의골화와인대의석회화를특징으로하는질환으로국내유병률은진단기준에따라서 2.9% 에서 4.1% 에이른다. 6) 골화는경추부, 흉추부, 요추부한부위에국한될수도있고동시에여러부위에나타날수도있는데주로흉추부에발병한다. 2) 일반적으로미만성특발성골격과골증의진단은흉추부의침범을기본전제로하고강직성척추염과의감별을위해 Resnick 의기준을따른다. 즉, 척추의추체-추간판인접부위에병소의국소적돌출유무에관계없이적어도 4개의연속된척추에서전외측을따라석회화및골화형성을보이고침범된부위에추간판높이가비교적보존되어있으며, 진공현상, 척추외연경화를포함한퇴행성추간판질병의방사선학적변화가없고골단성관절의골강직과천장골관절의미란, 경화, 융합이없는경우로미만성특발성골격과골증을진단한다. 1) 본증례에서는증상및방사선소견, HLA B-27 검사결과를토대로강직성척추염을배제하고미만성특발성골격과골증 105

5 Jaedong kim et al Volume 24 Number 2 June 2017 을진단하였다. 이러한미만성특발성골격과골증과강직성척추염의감별이최근더욱중요해진이유는강직성척추염의치료에있어서항 TNF-alpha 치료 (Anti-TNF-a therapy) 가중요하기때문이다. 2) 역으로, 미만성특발성골격과골증의경우항 TNF-alpha 치료의효과를기대할수없으므로본증례에서환자의수술전두질환의감별은수술후치료의측면에있어서매우중요했다. 미만성특발성골격과골증환자의경우척추골절의위험도가상대적으로증가한다. 7) Diederichs 등 8) 은 65세이상 342명을대상으로한연구에서미만성특발성골격과골증환자군의경우대조군에비해척추골절의빈도가흔하다고보고하였다. Westerveld 등 9) 은문헌고찰 (systemic review) 을통해미만성특발성골격과골증환자의척추손상중 69.1% 는저에너지충격에의해발생하였고고에너지충격에의한척추손상은 23.6% 에불과하였다고보고하였다. 일반적으로척추의경우외부의충격이가해지면유동성이있는여러추체를따라서에너지가분산되지만척추의유동성이상대적으로사라진미만성특발성골격과골증의경우외부충격의분산이이루어지지않으므로 7.8.9) 이번증례에서보는바와같이약한충격에도쉽게골절이일어날것으로추측할수있다. 본증례에서환자는척추골절로인해척추유합술을받고 2 년후척추후만변형이진행되어내원하였다. 미만성특발성골격과골증과척추후만변형의관련성에대해서는기존의보고에서살펴볼수있다. Nardo 등 3) 은 1,172명의환자를대상으로미만성특발성골격과골증과척추후만변형의상관관계를 Cobb 각도를측정하여분석하였다. 이중흉추부에국한된 DISH 환자는 101명으로대조군에비하여 Cobb 각도가통계학적으로유의하게증가하였고이는인종에따라서도유의한차이를보였다. Yamada 등 4) 은 40세이상척추협착증에대한치료가필요한환자 132명을대상으로한후향적연구에서 39.4% 에해당하는 52명의환자가미만성특발성골격과골증으로진단되었고나이가들수록발병률은점차적으로증가한다고보고하였다. 미만성특발성골격과골증환자의경우대조군에비하여흉추부의척추후만변형이증가하고요추부의전만각은통계적으로유의하게감소하였으며특히 DISH 환자중추체골화수가증가할수록통계적으로유의하게흉추후만각은증가하고요추전만각은감소하였다. 흉추부는해부학적으로척추후만이있는부위인데미만성특발성골격과골증이라는질환의특성상전종인대와주변의결체조직등의석회화및골화양상, 척추체전면의새로운골형성, 추체전면부의퇴행성변화등으로인해본증례에서처럼척추후만변형이진행될것으로추측해볼수있다. 3,4) 미만성특발성골격과골증환자에서발생한척추골절에 대한치료는보조기착용또는침상안정과같은보존적치료를고려할수있으나일반적으로수술적치료가더선호된다. Westerveld 등 9) 은문헌고찰을통해척추손상을받은미만성특발성골격과골증환자중 54.5% 가수술적치료를받은반면, 45.5% 가보존적치료를받았으며보존적치료를받은주요이유는단지수술위험도가높거나환자가수술적치료를거부했기때문이라고보고하였다. 또한수술적치료를받은미만성특발성골격과골증환자군의경우보존적치료를받은환자에비해신경학적이상증상이더호전됐다고보고하였다. 단, 미만성특발성골격과골증환자의경우흉요추이행부의유연성이감소하므로척추에가해지는피로가요추부에집중될수있다. Otsuki 등 10) 은요추부척추협착증을진단받고단분절요추부척추체간유합술을시행받은환자 208명을대상으로한연구에서미만성특발성골과골증환자의경우수술후가관절증 (pseudoarthrosis) 또는인접분절질환 (ASD) 으로인한재수술비율이일반환자군에비해높다고보고하였다. 따라서본증례의환자의경우에도지속적인추적관찰이필요할것으로판단된다. REFERENCES 1. Resnick D, Niwayama G. Radiographic and pathologic features of spinal involvement in diffuse idiopathic skeletal hyperostosis. Radiology. 1976;119: Olivieri I, D Angelo S, Palazzi C, et al. Diffuse idiopathic skeletal hyperostosis: differentiation from ankylosing spondylitis. Curr Rheumatol Rep. 2009;5: Nardo L, Lane NE, Parimi N, et al. Diffuse idiopathic skeletal hyperostosis association with thoracic spine kyphosis: a cross-sectional study for the Health Aging and Body Composition Study. Spine (Phila Pa 1976). 2014;24: Yamada K, Toyoda H, Terai H, et al. Spinopelvic alignment of diffuse idiopathic skeletal hyperostosis in lumbar spinal stenosis. Eur Spine J. 2014;6: Park YS, Kim HS, Baek SW, et al. Preoperative computerbased simulations for the correction of kyphotic deformities in ankylosing spondylitis patients. Spine J. 2014;14: Kim SK, Choi BR, Kim CG, et al. The prevalence of diffuse idiopathic skeletal hyperostosis in Korea. J Rheumatol. 2004;10: Mader R, Verlaan JJ, Buskila D. Diffuse idiopathic skeletal hyperostosis: clinical features and pathogenic mechanisms. Nat Rev Rheumatol. 2013;12:

6 Journal of Korean Society of Spine Surgery Kyphotic Deformity After Spinal Fusion in DISH 8. Diederichs G, Engelken F, Marshall LM, et al. Osteoporotic fractures in men research group. Diffuse idiopathic skeletal hyperostosis (DISH): relation to vertebral fractures and bone density. Osteoporos Int. 2011;6: 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;2: Otsuki B, Fujibayashi S, Takemoto M, et al. Diffuse idiopathic skeletal hyperostosis(dish) is a risk factor for surgery in short segment lumbar interbody fusion. Eur Spine J. 2015;11:

7 Case Report J Korean Soc Spine Surg Jun;24(2): 미만성특발성골격과골증환자에서척추유합술후발생한후만변형 - 증례보고 - 김재동 이재원 박예수한양대학교의과대학구리병원정형외과학교실연구계획 : 증례보고목적 : 강직성척추염과감별을요하는미만성특발성골격과골증환자에서골절로인한척추유합술후진행하는척추후만변형 1예를경험하였기에문헌고찰과함께보고하고자한다. 선행문헌의요약 : 미만성특발성골격과골증은주로인대및골부착부의석회화및골화를특징으로하는질병으로강직성척추염과유사하나두질환은근본적으로다르다. 미만성특발성골격과골증은척추후만증과관련되어있으나골절로인해시행한척추유합술후흉추부의진행하는척추후만변형을보고한사례는없다. 대상및방법 : 47세남성이흉요추부동통으로내원하였다. 강직성척추염의가능성을배제하고미만성특발성골격과골증을진단하였으며제 11-12흉추간의굴곡-신연손상을진단한후척추유합술을시행하였다. 이후척추후만변형이진행되어기존삽입물을제거한뒤절골술및척추유합술을시행하였다. 결과 : 수술후후만척추변형은교정되었고외래추시중이다. 결론 : 미만성특발성골격과골증은강직성척추염과의감별을요하고골절에취약하며척추유합술후에도척추후만변형이진행될수있으므로지속적인추적관찰이필요하다. 색인단어 : 미만성특발성골격과골증, 후만변형 약칭제목 : 미만성특발성골격과골증에서척추유합술후발생한후만변형 접수일 : 2016년 11월 5일 수정일 : 2016년 12월 30일 게재확정일 : 2017년 4월 25일 교신저자 : 박예수 경기도구리시경춘로 153 한양대학교구리병원정형외과학교실 TEL: FAX: hyparkys@hanyang.ac.kr 108 Copyright 2017 Korean Society of Spine Surgery Journal of Korean Society of Spine Surgery. pissn eissn This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License ( 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 Acute Epidural Hematoma Following Cervical Spinal Fracture in a Patient with Ankylosing Spondylitis Sang Bu

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.,