Case Report J Korean Orthop Assoc 2011; 46: 507-511 http://dx.doi.org/10.4055/jkoa.2011.46.6.507 www.jkoa.org Simple Subperiosteal Hematoma with a Periosteal Reaction Mimicking a Malignancy 차수민 신현대 김경천 황정모 김보건 * 충남대학교의학전문대학원정형외과학교실, * 동아대학교의과대학정형외과학교실 외상또는스트레스등에의해서도골막반응이나타날수있으며이는주로양성의소견을보이는균질하면서도연속적인양상이다. 원위요골의성장판손상병력이있는 11세남아의단순방사선사진에서원위요골의골간단에미만성의골용해성소견을보이며, 외측피질골의음영이감소되었다. 코드만삼각양상의반응이원위요골의외측에, 수개의침골양상의골막반응이내측에관찰되었다. MRI 소견상관상면의 T2 강조영상에서골막을박리하는골성기원의종괴가관찰되었으며축성사진상비균질의고신호강도의종괴와내부의액체- 액체경계 (fluid-fluid level) 가관찰되었으며종괴의경계는불분명하고조영증강되는양상이었다. 일차성악성병변을의심하여생검을시행하였으나단순혈종의육안적소견및병리적결과를얻은드문증례를경험하였기에문헌고찰과함께보고하는바이다. 색인단어 : 골막, 요골, 혈종 골막주위반응은골막의일련의염증또는자극으로인한신생골형성에기인한방사선학적소견이다. 이러한골막의신생골형성을유발할수있는원인은다양하며반응의양상을양성 (benign) 과악성또는공격적 (aggressive) 반응으로나누기도한다. 1,2) 특히, 코드만삼각반응, 침골양상 (spicule), 햇살모양 (sunburst appearance) 은공격적반응으로분류하며이는종종원발성악성종양과연관된다. 한편, 잦은외상또는스트레스등에의해서도골막반응이나타날수있으며이는주로양성소견을보이며균질하면서도연속적인 (solid uninterrupted) 양상이다. 2) 현재까지저자들의문헌고찰결과, 운동선수에서만성적스트레스 (stress) 로야기된 " 악성종양에서보이는골막반응 " 이 2예, 3,4) 치골부위의외상으로역시 " 악성종양에서의골막반응 " 이보인 1예, 5) 결핵으로인한 " 악성종양의골막반응 " 이보인 1예 6) 정도의보고가있었으나소아에서성장판손상으로인한혈종때문에전형적악성종양으로오인될만한골막반응을보고한사례는아직없었다. 저자들은 11세소아에서단순방사선사진상악성의골막반응을보여추가로시행한 MRI에서도악성을배제하기어려운예에서조직검사를시행하였고, 단순혈종의병리학적결과를얻은 1예 접수일 2011 년 5 월 30 일게재확정일 2011 년 8 월 22 일교신저자신현대대전시중구대사동 640, 충남대학교의학전문대학원정형외과학교실 TEL 042-280-7349, FAX 042-252-7098 E-mail hyunsd@cnu.ac.kr 를보고하고자한다. 증례보고 11세된남아로 3주전부터악화된좌측수근관절의수장측에압통을동반한종창 (swelling) 을주소로내원하였다. 약 2 4 cm 넓이의종괴가만져졌으며경계는명확하지않았고다소딱딱한양상이었다. 약간가동성 (mobility) 은존재하였으며, 피부색깔등의변화는없었다. 4주전수상병력이있었으며, 환아스스로수상 3 일후장상지반부목을풀었다고하였다. 측면단순방사선소견상원위요골수장측에 Salter-Harris 분류 I의성장판손상이보였고수상당시통증이 2-3일지속후없어졌다가 3주전부터서서히통증과종창이증가하였다. 2주전의상기도감염증상은 4일만에완치되었으며내원당시열 (fever) 등의증상은없었다. 수근관절의관절가동범위는정상이었으며수지의굴곡시통증이다소악화되는양상이었다. 가족력상특이한사항은없었다. 수근관절방사선사진에서종괴의윤곽으로보이는방사선비투과성 (radiopaque) 음영이관찰되었으며, 원위요골의골간단에미만성의골용해성소견 (osteolytic) 을보이며, 골간단의외측에피질골음영이감소되었다. 코드만삼각양상의반응이원위요골의외측에, 수개의침골 (spiculated) 골막반응이내측피질에서관찰되었다. 측면사진상선형의골막신생골의음영이관찰되었다 (Fig. 대한정형외과학회지 : 제 46권제 6호 2011 Copyright 2011 by The Korean Orthopaedic Association 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.
508 차수민 신현대 김경천외 2 인 1A, 1B). 혈액학적검사상혈색소치나백혈구수치및 AST, ALT, LDH, ESR, CRP, ALP (alkaline phospatase), bleeding time, PT, PTT는모두정상이었다. 시행한 MRI 소견상 T2 강조, 관상면의영상에서골막을박리 (stripping) 하는골성기원의종괴 (intraosseus lesion) 가관찰되었으며골간단과성장판의부위에종괴의원위경계 (margin) 가관찰되었다 (Fig. 1C, 1D). 시상면및축성사진상비균질의고신호강도 (inhomogeneous high signal intensity) 의종괴와내부의액체-액체경계 (fluid-fluid level) 가관찰되었으며종괴의경계는불분명하고조영증강되는양상이었다. 전반적으로팽창하는양상의 (bulging out) 종괴로골수 (bone marrow) 는정상적신호강도였다. 전형적인원발성악성종양또는염증반응을의심하는소견으로저자들은동맥류성골낭종을동반한골막성골육종가능성, 골수염또는유잉육종 (Ewing's sarcoma) 등을의심하였다. 방사선판독의는모세혈관확장성골육종 (telangiectatic osteosarcoma), 골막의동맥류성낭종 (periosteal aneurismal cyst) 등의가능성을제시하였다. 저자들은악성종양의가능성을배제할수없어종양절제의원칙을따라절개생검 (incisional biopsy) 을하였고병리적결과는 " 섬유소성퇴행 (fibrinoid degeneration) 을동반한미만성출혈과염증반응 " 으로나와재차시행한동일절개상의절제생검 (excisional biopsy) 및주변의동결건조조직검사를시행하였다. 수술시시행한임시동결조직검사에서는악성세포가관찰되지않았으며종괴는골막하에서육안적으로저명하고경계도명확하였으며회내방형근 (pronator quadratus) 을압박하였다 (Fig. 2A). 최종조직검사의결과역시단순혈종으로나타났다 (Fig. 2B). 골막박리후얇은혈종의피막을관찰하였고소파상혈종성분만이확인되었다. 혈종으로인한원위요골피질의자극소견은육안적으로심하지않았으며골괴사역시관찰할수없었다. 10주후시행한추시방사선사진상침골양상의골막반응과코드만삼각양상의반응은저명하지않았고이중피질의음영은 Figure 1. (A, B) A radiopaque shadow of the mass border was present on anteroposterior and lateral simple wrist radiographs, and diffuse osteolysis was seen at the distal radial metaphysis. The cortical shadow decreased on the lateral side of the metaphysis. A Codman s triangle-like lesion was seen on the lateral side of the distal radius, and a few spicules were observed on the medial cortex. A Salter-Harris classification I growth plate injury was found on the palmar side of the distal radius on a lateral radiograph, and a linear shadow of periosteal new bone formation was visible. (C, D) Coronal images showed an inhomogeneous high signal-intensity mass. The mass margins were ambiguous and enhanced. The mass was bulging, but the bone marrow showed normal signal intensity. Figure 2. (A) The mass was located on the periosteum. The margin was clear, and the pronator quadratus was compressed. (B) Diffuse hemorrhage with fibrinoid degeneration and acute inflammation was shown at permanent biopsy (H&E stain, 200).
509 Figure 3. (A, B) The spiculated pattern of the periosteal reaction and Codman s triangle-like lesion were not significant on follow-up radiographs taken 10 weeks later, but a double cortical shadow was present. The patient did not complain of any symptoms. 존재하고있었고환아는증상이없는상태였다 (Fig. 3). 고찰 골막반응은어떠한이유에서든골막하공간의석회화또는골화가방사선사진에서관찰되는것이며크게생리적골막반응과병적골막반응으로나뉜다. 생리적반응은 6개월미만의영아에서성장급진기 (growth spurt) 기에장관골 (long tubular bone) 주변에대칭적으로생기는것으로임상적의미는없으며병적골막하반응을배제하는것이중요하다. 7) 대개골막반응은병적반응을의미하는것이며다시국소적, 미만성으로구분하며국소적인반응 (focal periosteal reaction) 은대개외상, 감염, 종양 ( 유골골종, 골육종, 유잉육종등 ), 연부조직종괴등에서관찰할수있다. 미만성반응은혈우병, 백혈병등의혈액학적이상, 구루병과같은대사및영양적이상, 영아피질골과골증 (infantile cortical hyperostosis), 연소기형류마토이드관절염등의염증성질환등에서관찰된다. 2,7) 한편, 골막반응의양상에따라단일층 (unilaminar), 다층 (multilaminar or onion skin), 치밀 (compact), 침골 (spiculated) 또는햇살모양 (sunburst), 코드만삼각 (Codman triangle), 배엽성껍데기 (lobulated shell) 등이있다. 골절이동반되지않은외상의경우단층 (unilaminar) 의골막반응이대개 7-10일후에관찰되며육안적골절이동반되는경우는 2-3주경과후혈종이성숙되면서기질화되어치밀 (compact) 양상의골막반응이골절부위와근처에서관찰된다. 8,9) 본증례의 첫외상시점은내원 4주전이었고당시보존적치료로수일만에증상이호전된후환아스스로가장상지반석고부목을제거하였다. 그상태에서수일을지낸후, 내원 3주전에통증과종창이서서히발생한것으로보아골막하혈종은수상받은성장판에서급성이아닌만성적, 지속적으로발생한것이거나통증과종창이서서히증가하기시작하는시점에서골절편의미세전위또는손상받은성장판의재손상등으로지연성으로보는것이타당할것이다. 한편으로는, 반석고부목제거 4일만에통증이재발되었고이는외상에대한보호를하지않아서발생한통증일가능성이높은만큼, 보호되지않아서거대가골형성이이루어질수있었다는개연성또한충분히있는것이사실이다. 다만외상으로인한혈종형성시침골또는햇살모양의골막반응은거의나타나지않는다고알려져있으며, 3,9) 문헌고찰상이번증례와같은성장판손상으로인한경우는없었고, 치골부위의외상후악성골종양으로오인된국내의 1예보고가있었다. 만성적자극으로유발된경우가 Koskinen 등 3) 과 Ward 등 4) 에의해보고되었으며, 이들은만성부하 (stress) 로유발된침골또는햇살모양의골막반응의 1예를각각보고하였고두예모두골프선수및풋볼선수에서나타난스포츠손상이었으며요척골간부에서골간막으로작용하는인장력 (pulling force) 으로초래되었다고가정하였다. 침골양상및햇살모양의반응이골간막의방향과일치하게나타났으며수개월간의보존적치료로임상증상및방사학적소견의호전을보고하였다. 한편혈우병에서도골막하혈종만으로도골막반응의양상은매우공격적일수있으나이경우는대개매우어린나이에진단된환아들이며, 전신에다발성으로골막하혈종이관찰되며침골및햇살모양의골막반응이악성종양에서관찰되는소견보다매우저명하고골수침범및주변의연부조직종괴를만드는점, 관절내에서관절파괴의형태로나타난다는점에서 10) 저자들의증례와는쉽게구분된다. 저자들의증례는성장판손상으로혈종이만성적, 지속적으로서서히생겼을가능성또는아직치유가안된성장판에대한추가적손상등의가능성을원인으로고려할수있다. MRI 역시악성을의심하는소견들이었고전이성악성병변의경우골수강내의부종이동반되어피질골, 골막, 골수에서전형적고신호강도가관찰되나본증례에서는이러한소견이없었으며어린연령과도잘맞지않아그가능성은떨어진다고판단하였다. 다만절제생검및소파술시행후아쉬웠던점은 1차절개생검후단순혈종의병리결과를얻은후핵의학검사를시행하지않았던것이며핵의학검사를시행하였다면저자들이가정하는원위요골의성장판손상을확인할수있었을것이며다소가능성은떨어지지만다른부위의조영증강을확인하여전이성병변등을확실히배제할수있었을것이다.
510 차수민 신현대 김경천외 2 인 결론 저자들은 11세소아에서원위요골골간단의미만성골용해성병변과침골양상, 코드만삼각양상등의골막양상반응이관찰되어 MRI 촬영및절제생검을시행하였고그결과단순혈종으로진단된 1예의보고를통해외상 ( 성장판손상 ) 과연관된혈종에서도악성종양을의심할수있는방사선소견이매우드물게관찰됨을문헌고찰과함께보고하는바이다. 참고문헌 1. Miller TT. Bone tumors and tumorlike conditions: analysis with conventional radiography. Radiology. 2008;246:662-74. 2. Ragsdale BD, Madewell JE, Sweet DE. Radiologic and pathologic analysis of solitary bone lesions. Part II: periosteal reactions. Radiol Clin North Am. 1981;19:749-83. 3. Koskinen SK, Mattila KT, Alanen AM, Aro HT. Stress fracture of the ulnar diaphysis in a recreational golfer. Clin J Sport Med. 1997;7:63-5. 4. Ward WG Sr, Sekiya JK, Pope TL Jr. Traumatic ossifying periostitis of the ulna masquerading as a malignancy in a football player. A case report and literature review. Am J Sports Med. 1996;24:852-6. 5. Song KS, Kim HG, Park BM, Kim JM, Jung SH, Yang BS. Post traumatic osteolysis of the pubic bone simulating malignancy or osteomyelitis - a case report -. J Korean Bone Joint Tumor Soc. 2007;13:180-4. 6. Jahng J, Kim YH, Lee KS. Tuberculosis of the lower lumbar spine with an atypical radiological presentation - a case mimicking a malignancy -. Asian Spine J. 2007;1:102-5. 7. Shopfner CE. Periosteal bone growth in normal infants. A preliminary report. Am J Roentgenol Radium Ther Nucl Med. 1966;97:154-63. 8. Wenaden AE, Szyszko TA, Saifuddin A. Imaging of periosteal reactions associated with focal lesions of bone. Clin Radiol. 2005;60:439-56. 9. Ishibashi Y, Okamura Y, Otsuka H, Nishizawa K, Sasaki T, Toh S. Comparison of scintigraphy and magnetic resonance imaging for stress injuries of bone. Clin J Sport Med. 2002;12:79-84. 10. Resnick D. Diagnosis of bone and joint disorders. 4th ed. Philadelphia: WB Saunders; 2002. 2346-73.
511 Simple Subperiosteal Hematoma with a Periosteal Reaction Mimicking a Malignancy Soo-Min Cha, M.D., Hyun-Dae Shin, M.D., Ph.D., Kyung-Cheon Kim, M.D., Ph.D., Jung-Mo Hwang, M.D., and Bo-Kun Kim, M.D.* Department of Orthopedic Surgery, Chungnam National University School of Medicine, Daejeon, *Dong-A University School of Medicine, Busan, Korea A benign periosteal reaction, which can occur after trauma or stress, has a solid and uninterrupted appearance on radiography. In contrast, an aggressive periosteal reaction, which may indicate a malignancy, appears as a Codman's triangle or with a spiculated and sunburst pattern. In the present case, an 11-year-old boy with a previous injury to the distal radial growth plate presented with diffuse osteolysis on the distal radial metaphysis and decreased opacity of the lateral side cortex on plain radiograph. A Codman s triangle-like lesion was seen on the lateral side of the distal radius, and a few spicules were observed on the medial side of the distal radius. A T2-weighted coronal magnetic resonance image revealed a mass that had stripped the periosteum; the mass had heterogeneous signal intensity and a fluid-fluid level on axial views. The margins of the mass were unclear, but enhanced. Suspecting a primary malignancy, we performed a biopsy. The pathology revealed that the mass was a simple hematoma. Key words: periosteum, radius, hematoma Received May 30, 2011 Accepted August 22, 2011 Correspondence to: Hyun-Dae Shin, M.D., Ph.D. Department of Orthopaedic Surgery, Chungnam National University School of Medicine, 640, Daesa-dong, Jung-gu, Daejeon 301-721, Korea TEL: +82-42-280-7349 FAX: +82-42-252-7098 E-mail: hyunsd@cnu.ac.kr