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준현 해
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1 심부정맥혈전증으로오인된급속파괴형고관절증 1 예 증례보고 연세대학교의과대학재활의학교실및근육병재활연구소 홍지성ㆍ정철오ㆍ정강재ㆍ김동현ㆍ최정화ㆍ박윤길 A Case of Rapid Destructive Coxarthrosis Misdiagnosed as Deep Vein Thrombosis A Case Report Ji Seong Hong, M.D., Chul Oh Jung, M.D., Kang Jae Jung, M.D., Dong Hyun Kim, M.D., Jung Hwa Choi, M.D. and Yoon-Ghil Park, M.D. Department of Rehabilitation Medicine and Rehabilitation Institute of Muscular Disease, Yonsei University College of Medicine, Seoul, Korea Rapidly destructive coxarthrosis of hip joint (RDA) is a rare condition characterized by rapid joint degeneration and destruction similar to findings of infection, osteonecrosis, or rhuematoid arthritis but without a definitive diagnosis. We report a case of RDA mis-diagnosed as deep vein thrombosis (DVT) in a 69-year-old male patient. He complained of left hip joint pain which became progressively worse. Moreover, within 2 months after onset of pain, joint space narrowing and destruction of femur head with swelling was rapidly progressed. Initially, He was diagnosed clinically as DVT related avascular necrosis (AVN). However, there was no evidence of DVT on computerized tomoraphy. He was treated by total hip replacement surgery. According to the literature and experience from this case, total hip arthroplasty at early stage of disease could be utilized as a treatment of choice due to the severity of the symptoms and the natural history. (Clinical Pain 2011;10: ) Key Words: Rapidly destructive coxarthrosis, Deep vein thrombosis 서 론 은 1970 년 Postel 과 Kerboull 등 1) 에의해 1970 년에처음소개되었으며, 주로 60 대이상의노년기여성에서편측성으로발생하고, 임상적으로는약 1 년이내의고관절부의통증과방사선학적으로급속하게진행되는대퇴골두의파괴를특징으로하는매우드문질환이다. 2) 본질환은결정유발성관절병 (crystal induced arthropathy), 약물유발성관절병증 (Drug induced arthropathy), 무혈성골괴사 (Avascular osteonecrosis), 연골하부전골절, 패혈성관절염 (Septic arthritis), 신경병증 (Neuropathy), 류마티스관절 (Rheumatic arthritis) 등과관련되어발생할수있으나현재까지정확한원인은알려지지않은상태이다. 3-6) 본질환은다른관절염이나이형성증, 감염후유증등의보존적치료를하거나경과관찰하다수술적치료를고려하는유사한증상을보이는질환과달리인공고관절치환술을시행하는것이반드시그리고조기에필요하며, 수술이후보행이가능해지며통증이감소되는등의수술적치료의예후도좋으므로, 치료를위하여조기에정확한감별진단이필요하다. 3,7) 국내에서는대퇴골두무혈성골괴사가급속파괴형고관절증의원인중하나일수있다는보고가있었으며, 8) Yun 등 9) 은류마티스고관절염과매우유사한검사소견을보였던급속파괴형고관절증 3 예에대한증례를보고하였고 Min 등 10) 은고령의여자환자에게연골하부전골절과동반된금속파괴형고관절증에관한증례를보고하였으며마찬가지로 2008 년 Watarai 등 11), 2005 년 Niimi 등 12) 도고령의노인에게서단기간만에연골하부전골절이동반되는급속파괴형고관절증을보고하였다. 최근본저자들은좌측고관절통증발생 2 개월만에좌측하지의부종과통증을주소로입원하여방사선학적검사상급속한대퇴골두파괴소견을보였던급속파괴형고관절증의 1 예를경험하였기에이의임상증상, 진단및치료과정을보고하는바이다. 급속파괴형고관절증 (Rapid Destructive Coxarthrosis) 증 례 접수일 : 2011 년 7 월 30 일, 게재승인일 : 2011 년 11 월 9 일책임저자 : 박윤길, 서울시강남구도곡동 , 강남세브란스병원재활의학과 Tel: , Fax: drtlc@yuhs.ac 69 세남자환자로내원 2 개월전부터발생한좌측고관절부위의동통으로진통소염제등의약물치료를시행하였으나증상의호전이없었고, 내원 2 주전부터악화된좌측하지전체의부종과통증그리고보행불능을주소로외래 126

2 홍지성외 5 인 : 심부정맥혈전증으로오인된급속파괴형고관절증 1 예 Table 1. Results of Blood Test at Admission Hemoglobin (g/dl) WBC (/mm 3 ) PLT (/mm 3 ) Glucose (mg/dl) BUN (mg/dl) Cr (mg/dl) Total protein (g/dl) Albumin (g/dl) T. bilirubin (mg/dl) AST (OT) (IU/L) ALT (PT) (IU/L) Na (mmol/l) K (mmol/l) Ca (mmol/l) Phosphate (mg/dl) Total cholesterol (mg/dl) ESR (mm/hr) Antistreptolysin O (IU/ml) Rheumatoid Factor (IU/ml) CRP (mg/l) D-dimer (ng/ml) Results , , References ,000 10, , , 방문하여내원당일시행한단순방사선검사에서좌측고관절의파괴형변화및전위소견을보여입원하였다. 과거력상고혈압으로외부병원에서약물조절중인것이외특이사항없었고가족력상에도특이사항관찰되지않았다. 입원당일환자는만성병색을보였으며혈압은 114/64 mmhg, 맥박수 77 회 / 분, 호흡수 20 회 / 분, 체온 36.2 o C 이었다. 두경부, 흉부, 그리고복부진찰소견상특이소견없었으며좌측하지의부종과고관절부터발목까지전체적인동통및부종이관찰되었다. 부종은양측하지에서우측과좌측둘레차이가 36.5:46 cm ( 무릎관절의슬개골상극 10 cm 상방 ), 30:34 cm ( 무릎관절의슬개골하극 10 cm 하방 ) 로좌측하지부종소견을보였다. 또한좌측고관절은통증이심하여능동적및수동적운동범위를측정할수없었으며통증으로인해보행이불가능하였다. 입원당일말초혈액검사상혈색소 11.3 g/dl, 백혈구 8,940/mm 3, 혈소판 304,000/mm 3, 일반화학검사상공복시혈당 104 mg/dl, 혈중요소질소 4.4 mg/dl, 크레아티닌 0.8 mg/dl, 총단백질 5.5 g/dl ( 알부민 3.4 g/dl), 총빌리루빈 0.7 mg/dl, 알라닌아미노기전이효소 / 아스파르트산염아미노전이효소 (ALT/AST) 41/32 IU/L, 알칼리성인산분해효소 135 IU/L, 나트륨 122 meq/l, 칼륨 4.0 meq/l, 칼슘 8.0 mg/dl, 인 3.6 mg/dl, 총콜레스테롤 151 mg/dl, 항트립신 O 28.0 IU/ml, 류마토이드인자 6.3 IU/ml 이었으며소변검사는정상범주이었다 (Table 1). 적혈구 침강속도는 11 mm/hr, C 반응단백질은 87.2 mg/l, D- dimer 6.5 mcg/ml 으로알라닌아미노기전이효소, 알칼리성인산분해효소및 C 반응단백질, D-dimer 수치가기준치보다증가소견을보였다. 입원당일고관절단순방사선사진에서좌측대퇴골두및경부에비정상적인골파괴성변화가관찰된반면우측대퇴골두는정상이었다 (Fig. 1). 좌측하지부종이있어좌측심부정맥혈전증을의심하여내원 2 일째시행한컴퓨터단층촬영소견에는심부정맥혈전증을의심할만한소견은보이지않았으며좌측대퇴골두가용해되어있었고고관절주위에삼출물이동반되어있었다 (Fig. 2). 또한근육의비정형성골화성근염이동반된급속파괴형고관절증이의심되었다. 더불어시행한전신골스캔검사 (whole body bone scan) 상에서도좌측고관절부위로대퇴골을비롯한주위근육및연부조직에 uptake 가증가된소견을보였다 (Fig. 2). 내원 4 일째, 하지의부종및동통이지속되었고자기공명영상검사상좌측고관절삼출물을동반한대퇴골부의골용해및주위활막염이관찰되었고, 이로인해해당부위주위의근육들의변형이발생하여급속파괴형고관절증으로진단되었다 (Fig. 2). 이후환자는입원 1 주째좌측인공고관절치환술을시행받았으며이후수술후 2 일째 2 차례, 이후에도 3 차례이상의재발성고관절탈구가발생하였고이후수술후약 2 개월째근위부인공관절주위골절로내고정술시행하였고이후대퇴골중간부위, 인공관절주위골절등의 3 차례골절로내고정술시행후퇴원하였다. 고 본증례는특발성좌측고관절동통발생이후부종을동반한급속하게진행된동통및관절가동범위및보행장애로내원하여초기진단시심부정맥혈전증이의심되었으나방사선검사상급속파괴형고관절증으로진단된사례이다. 급속파괴형고관절증에대해서는 1970 년 Postel 과 Kerboull 이한쪽고관절의심한통증을호소하며여성에게빈번하게발생하고고관절의파괴속도가 6 12 개월내에급속하게일어나는질환에대해서급속파괴형고관절증으로명명하였다. 1) 이의원인에대해서는분명하게알려져있지않으며, 대부분특발성으로발생하는것으로알려져있지만여러저자들에의해다양한연관이론들이제시되고있다. Rosenberg 등 2) 은고관절괴사가편측성으로나타나며 60 세이상의중년여성에게서흔히발생하고편측성으로급속히진행하는보행장애및고관절동통등의특징적인임상증상을동반하는퇴행성관절염의아형으로규정하였고그외에도 serum type I collagen C-telopeptide 또는 urinary type II collagen helical (Helix-II) 의증가를동반하는 찰 대한임상통증학회지 127

Fig. 1. X-ray of the hip joint. Anterior (A) and axial (B) views of abnormal destructive bone change (arrow) at left femoral head.

(D) Anterior image of dislocation (arrow) of left femur.

13-15) 이러한급속파괴형고관절증의기전으로는 matrix metalloproteinases (MMPs), interleukin-1 (IL-1), prostaglandins (PGs) 와다른 cytokine 등에의해관절파괴가유발되고,

급속파괴형고관절증의경우대퇴골연골하골절과골괴사및다른질환들을감별하는데방사선검사중자기공명영상검사가유용하다고알려져있으며대퇴골골두와비구에서 T1 강조영상에서골수의부종소견과함께낭포성상의연골하결손등의소견이보이는것으로알려져있다.

3 Fig. 1. X-ray of the hip joint. Anterior (A) and axial (B) views of abnormal destructive bone change (arrow) at left femoral head. Upward displacement of remained left proximal femur. (C) Anterior image of postoperative status (arrow) of left femur. (D) Anterior image of dislocation (arrow) of left femur. 류마티스관절염, 관절내 hydroxyapatite 나 pyrophosphate crystal 의침착이나 indomethacin 등의진통소염제사용, 쿠싱증후군와연관된골다공증, 과체중등이관련요소로제기되었다 ) 이러한급속파괴형고관절증의기전으로는 matrix metalloproteinases (MMPs), interleukin-1 (IL-1), prostaglandins (PGs) 와다른 cytokine 등에의해관절파괴가유발되고, 그밖에대퇴골두의연골하부전골절같은기계적요인에의해서도관절파괴에영향을주는것으로알려져있다. 급속파괴형고관절증의경우대퇴골연골하골절과골괴사및다른질환들을감별하는데방사선검사중자기공명영상검사가유용하다고알려져있으며대퇴골골두와비구에서 T1 강조영상에서골수의부종소견과함께낭포성상의연골하결손등의소견이보이는것으로알려져있다. 이와더불어조직학적검사상다핵거핵세포및육아종성병변등으로다른질환과감별할수있는점으로알려져있다. 본증례에서는병력상국소및전신발열이나발적양상이없었고고관절부위에상처나수술반흔등에서비롯되는세균성고관절염을배제할수있었으며, 혈액 학적검사를통해다른류마티스관절염등과관련된요소를배제할수있었다. 또한고관절동통외신경학적증상이없어신경병성고관절증과감별이가능했다. 하지만 60 세이상의남성, 또한극심한통증을감소시키기위한비스테로이드성항염증제재등의진통소염제를사용하였던병력등이고관절골절및급속파괴형고관절증의진행을가속시키는악화요인으로생각할수있었다. 심부정맥혈전증은임상소견으로해당부위의온기및압통, 호프만징후등을보이고검사실소견상 D-dimer 증가와방사선학검사상정맥 scanning 과도플러초음파혈류검사에서분명한하지혈류장애가보이는것이특징이다. 16,17) 류마티스관절염은급속한골파괴소견과혈액학적이상및다른관절의침범등의소견으로감별할수있다. 18) 세균성관절염은임상소견으로발열과열감등이존재하고, 혈액검사상염증수치의증가를보이며, 방사선학검사상경계가불분명한골파괴와이차적인골경화를수반한다. 따라서혈액검사와방사선검사소견에서이들질환의가능성을배제할수있었다. 또한단측성으로침범한관절 128

홍지성외 5 인 : 심부정맥혈전증으로오인된급속파괴형고관절증 1 예 Fig. 2.

externalrotators, adductor muscles (B). Computerized tomography showed resorption (arrow) of left femoral head with hip joint effusion (C).

이외에도류마티스관절염을앓고있던환자가급속파괴형고관절증을진단받으면서동측의요근확장과장요근의윤활낭염이발생하는등의보고가있었고, 19) 2005년에 Yamakawa 등 20) 이급속파괴형고관절증을진단받은환자에게서조직병리학적인검사를시행하였을때고관절내로과혈관성및파골세포의이상증식이보여급속파괴형고관절증의골용해와파괴에대한논의가있었다.

21) 본증례에서는좌측고관절동통발생이후진통소염제등으로치료하였으나증상의호전없이증상발현 2개월만에보행불능을동반한동통및관절가동범위악화와단순방사선사진과컴퓨터단층촬영, 자기공명영상검사상좌측대퇴골두의급속한골파괴로급속파괴형고관절증으로진단받고고관절치환술을시행하였다.

4 홍지성외 5 인 : 심부정맥혈전증으로오인된급속파괴형고관절증 1 예 Fig. 2. MRI study showed abnormal signal change (arrow) at left femoral head with joint effusion (A), synovitis including extensive signal alteration (arrow) of hip abductors, externalrotators, adductor muscles (B). Computerized tomography showed resorption (arrow) of left femoral head with hip joint effusion (C). Increased uptake (arrow) on left femur in whole body bone scan (D). 을비롯하여자기공명영상에서대퇴골두전체를침범하는괴사및대퇴골의부종, 비구주위의골수부종, 관절내삼출액등은전형적인대퇴골두무혈성괴사와는구별될수있는소견으로유의하여야할소견이라고할수있다. 이외에도류마티스관절염을앓고있던환자가급속파괴형고관절증을진단받으면서동측의요근확장과장요근의윤활낭염이발생하는등의보고가있었고, 19) 2005년에 Yamakawa 등 20) 이급속파괴형고관절증을진단받은환자에게서조직병리학적인검사를시행하였을때고관절내로과혈관성및파골세포의이상증식이보여급속파괴형고관절증의골용해와파괴에대한논의가있었다. 또한이질환을진단받고나서수술적치료를하기전스테로이드주사를고관절주위에맞았을때수술적치료에어떤예후를보이는가에대해서도보고가있었으나당시특별한예후에변화는없었다. 21) 본증례에서는좌측고관절동통발생이후진통소염제등으로치료하였으나증상의호전없이증상발현 2개월만에보행불능을동반한동통및관절가동범위악화와단순방사선사진과컴퓨터단층촬영, 자기공명영상검사상좌측대퇴골두의급속한골파괴로급속파괴형고관절증으로진단받고고관절치환술을시행하였다. 특히이전관련논문에서보고되지않았던좌측하지의심한부종과관절운동장애로인한보행장애는임상적경과상심부정맥 혈전증을시사하는소견으로생각될수있었으나이에신속한심부정맥혈전증컴퓨터촬영및혈액검사로이를배제할수있었고해당관절부위의적절한수술적치료를시행할수있었다. 본증례에서는자기공명영상검사후급속한수술적치료가필요한상태로확진을위한조직병리학적검사는시행하지않았다. 또한본증례에있어수술적치료후 2 차례의고관절탈구및 3 차례의골절이발생하였는데다른질환과급속파괴형고관절증에있어수술후고관절탈구및골절에대해서는 2009 년 Kuo 등 22) 이급속파괴형고관절증을진단받고인공고관절전치환술을받은 8 명의환자에게서 6 년뒤추적관찰하였을때 1 명에서수술부위의인공고관절해리가발견되었던보고외에는알려진바가없어본환자의경우탈구가발생한원인에대해서는유추하기어려우나상기질환에대해고관절탈구를염두에두고보조기착용및관절보호등으로예방할수있도록해야할것으로생각된다. 결 고령의환자가고관절의통증과하지전체의부종을주소로내원했을때단순히고관절무혈성괴사, 심부정맥혈전증뿐아니라연골하부전골절의가능성을염두에두고적극적인검사와지속적인관찰을통해급속파괴형고관 론 대한임상통증학회지 129

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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