453 pissn : 1226-2102, eissn : 2005-8918 Case Report J Korean Orthop Assoc 2018; 53: 453-457 https://doi.org/10.4055/jkoa.2018.53.5.453 www.jkoa.org 소아에서발생한진행성 Kienbock 병 김동희 어일주 정민 성균관대학교의과대학삼성창원병원정형외과학교실 Advanced Pediatric Kienbock s Disease Dong-Hee Kim, M.D., Yil-Ju Eho, M.D., and Min Jeong, M.D. Department of Orthopedic Surgery, Samsung Changwon Hospital, Sungkyunkwan University School of Medicine, Changwon, Korea Kienbock s disease is very rare in pediatrics. The treatment of Kienbock s disease is controversial and depends on an understanding of the natural history of the disease. The methods proposed vary from conservative treatment to surgical treatment such as radial shortening or scaphotrapezoidal joint fixation, but there are few therapeutic guidelines for paediatric Kienbock s disease. We encountered a 14-year-old male with Lichtman stage IIIB disease and treated him with conservative management. We report this rare case of advanced paediatric Kienbock s disease with good clinical results at the 2-year follow-up along with a review of the relevant literature. Key words: lunate, osteonecrosis, Kienbock s disease, adolescent Kienbock 병은월상골에무혈성괴사를보이는병으로병리및 자연경과, 치료에있어서아직도명확히정립되어있지는않지만 급, 만성의외상및반복적육체적활동으로인하여월상골을지 지하는인대에열상이발생하고이것이혈류공급을차단하여골 괴사가점차적으로진행하는것으로알려져있다. 이병은전형 적으로 20 대에서 40 대남성에분포해있으며청소년기에는드물 다. 1,2) Kienbock 병의치료는이견이분분하여보존적치료부터요 골단축술, 삼주상유합술등수술적치료까지다양하지만소아 에서명확한치료가이드라인은제시되어있지는않다. 일반적으로성인 Kienbock 병에서보존적치료를하는동안진 행성단계로가는것과달리소아는성인보다는골격적으로미성 숙하며높은재생잠재력을가지고있어보다양호한결과를보 이고있다. 3) 이러한차이로인하여일부문헌에서는 15 세보다어 린 Kienbock 병에서적어도 6 주동안은석고고정술을이용한비 수술적치료를받기를추천하고있다. 4) Irisarri 등 5) 에따르면영아 Received March 12, 2017 Revised July 2, 2017 Accepted November 21, 2017 Correspondence to: Dong-Hee Kim, M.D. Department of Orthopedic Surgery, Samsung Changwon Hospital, 158 Paryong-ro, Masanhoewon-gu, Changwon 51353, Korea TEL: +82-55-233-5201 FAX: +82-55-233-8357 E-mail: dhkim1149@gmail.com ORCID: https://orcid.org/0000-0003-4358-9356 또는초기청소년 Kienbock 병은비수술적치료에양호한결과를보였으나 15세이상의 Kienbock 병에서는불량한결과를보여예후인자에나이가중요한것으로보고하였다. 소아에서는발생빈도가낮고대부분증례보고이기때문에정립된치료방법이없으나대부분보존적치료가우선고려되며저자들은 14세의 Kienbock 병을가진환아에서 3개월간석고고정술을이용한비수술적치료에좋은결과를보인 1예를경험하였다. 국외에서는 15세이하의진행성 Kienbock 병에서 15주간의석고고정을통하여성인과다르게보존적치료가성공적이었다는보고도있으나, 2,6) 국내에서는보고된적이없어문헌고찰과함께보고하고자하는바이다. 증례보고 14세남자환자가 7개월전배구시합중낙상으로인한좌측손목의과굴곡손상후발생한좌측손목의통증으로내원하였다. 체조등과같이반복적으로손목에하중이가해지는운동에참여한적은없었고배구시합중낙상외에손상의경험은없었다. 이학적검사상발적이나열감등감염을의심할만한소견은보이지않았고좌측수배측손목은중앙부위의압통을호소하였으며완관절능동적운동범위는건측과비교하여신전 50도, 굴곡 50 The Journal of the Korean Orthopaedic Association Volume 53 Number 5 2018 Copyright 2018 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/4.0/) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.
454 Dong-Hee Kim, et al. 도로감소되어있었다. 초기방사선검사상에서월상골의경화와함몰및측면방사선검사에서월상골의붕괴소견을보였다. 척골변위는중성을보였으며수근골높이비율도 0.5로정상범위였다 (Fig. 1). 이후불유합또는무혈성괴사와의감별을위해시행한컴퓨터단층촬영에서월상골의분절화소견을보였고자기공명영상검사에서는월상골근위부로하여 T1 강조영상에서는저신호강도, T2 강조영상에서고신호강도로보여 Lichtman IIIB의 Kienbock 병으로진단하였다 (Fig. 2). 본증례의경우외상이전에는환부의통증은없었으며이전방사선혹은다른영상의학적검사가없었기때문에과거무증상의 Kienbock 병의발현유무는정확히알기어려우나적극적인운동활동이나반복적인외상력이없는환아에서발현빈도는낮을것으로생각된다. 환자의경우초기청소년 Kienbock 병으로연령을고려하여우선적으로석고고정을이용한보존적치료를결정하였으며 4주후시행한추시방사선검사상월상골의근부위붕괴가국소적으로발생하였다. 방사선사진상병변이일부진행되었으나임상적으로통증이완화되는소견을보여보존적석고고정치료를지속하였다. 고정 8주후에는더이상의방사선적변화는관찰되지않았으며 12주후에는월상골붕괴소견이관찰되지않아석고고정해제후적극적재활치료를시작하였다 (Fig. 3). 이후 2년간추시관찰에서환자는완관절의동통은없어졌으며 Mayo wrist score 80점, Disabilities of the Arm, Shoulder and Hand score 26.7점으로임상적으로양호한결과를보였다. 또한능동적관절운동범위는신전 60도, 굴곡 60도로회복되었다 (Fig. 4). 단순방사선검사에서월상골의음영이함몰은있으나정상화된것으로보였고, 컴퓨터단층촬영에서도월상골이유합된소견이보이며자기 공명영상검사에서신호강도회복을관찰할수있었다 (Fig. 5). 고찰 저자들은 14세남자환자가배구시합중낙상으로인하여발생한좌측손목의통증으로내원한병기 IIIB의진행성 Kienbock 병을가진환자에서 3개월간석고고정술을이용한보존적치료로성공적인치료결과를경험하였다. Kienbock 병을가진환자의 1% 정도가 15세이하이며증상은제한적이고비진행성이며종종정상적인경과를밟는다. 7) Irisarri 등 5) 은소아에서의 Kienbock 병을영아월상골연화증 (infantile lunatomalacia) 과청소년월상골연화증 (juvenile lunatomalacia) 으로구분지었다. 영아월상골연화증은작은크기와자연적경과로인한심한변형을보여성인에서와같은단계적분류를적용할수가없었고청소년기월상골연화증은 13세부터골성숙이이루어지기까지의기간에발생하는경우로통증이심한편이다. 5) Herzberg 등 7) 에서 14세여자환자의 Kienbock 병에대해보존적으로치료하였고약 12개월간의추시관찰결과좋은결과를보였다. 그들은 15세이하의 Kienbock 병을가진환자에서는초기치료로적어도 6개월간의비수술적치료를받아야하며조영증강자기공명영상이초기진단및예후평가를위해꼭시행되어야한다고주장하였다. 7) Kim 등 4) 은 9-15세까지 Lichtman 제2, 3단계의 Kienbock 병을앓는 7명의아이에서 6주간의비수술적치료를시행하였다. 12세보다어린 3명의환아는월상골의회복및혈관재형성을보였으나 12세보다많은 4명의환아들은보존적치료에실패하였다. A B Figure 1. A 14-year-old male with wrist pain due to stage IIIA Kienbock s disease. Standard anteroposterior and lateral radiographs at presentation show that the lunate has sclerotic changes with a collapse of height. A neutral ulnar variance can be seen. Figure 2. (A) Sagittal computed tomography image shows bony fragmentation of the lunate bone. (B) Coronal T1-weighted magnetic resonance imaging (MRI) shows diffuse hypointensity of the lunate bone. Coronal T2-weighted fat-suppressed MRI shows diffuse hyperintensity and collapse of the lunate bone.
455 Advanced Pediatric Kienbock s Disease Figure 3. Anteroposterior and lateral radiographs at 12 weeks after casting. The carpal height ratio did not change, and the collapse of the lunate did not progress. Figure 4. Twenty-four months after the cast was removed, the patient reported a resolution of their wrist pain and showed a preservation of some wrist motions. A B C Figure 5. Radiograph (A), magnetic resonance imaging (B) and computed tomography (C) at the last follow-up evaluation showed no recurrence of Kienbock s disease. 보존적치료혹은수술적치료모두월상골의감압및부하를감소시켜재혈관화를촉진하는것이며, 3) 성인에서는수근골배열의변화를초래한진행성 Kienbock 병의치료방법으로수근골간유합술이흔히시행된다. 하지만소아에서는제한된증례보고로인한명확한치료가이드라인이제시되어있지않으며골격적으로미성숙하며높은재생잠재력으로인한비진행성및정상적인경과를밟기때문에일부문헌에서는 15 세이하의 Kienbock 병에서초기치료로석고고정술을이용한비수술적치료를권고하고있다. 4,7) Ferlic 등 1) 은석고고정술은 15주에서 6개월까지요구된다고보고하였으며장기간의석고고정술은관절강직등의 합병증이발생할수있다고하였다. 보존적치료에실패하였을경우수술적치료를시행할수있으며일시적삼주상고정술이나요골단축술등이사용될수있다. 8,9) 요골단축술은음성척골변위를가진 Kienbock 병에서월상골을감압하는데유용하나골격적으로미성숙한소아의경우수술후요골과성장으로인한음성척골변위의위험성때문에주의가필요하다. 10) 소아 Kienbock 병은소수로만보고되었으며, 그치료법으로는보존적치료부터수술적치료까지다양하여치료적가이드라인은거의없는실정이다. 저자들이경험한 14세남자환자의청소년기 Kienbock 병은국내에서는처음으로보고되는증례로초기
456 Dong-Hee Kim, et al. 외상에의한불유합과의감별진단을위해컴퓨터단층촬영과자기공명영상검사가도움이되었다. 방사선적으로임상적병기는진행된 III기였지만환자의나이를고려하였을때보존적치료를일차적으로시행하였고이후약 3개월간의석고고정술및재활치료를이용한비수술적치료에만족할만한결과를보여이를문헌고찰과함께보고하는바이다. CONFLICTS OF INTEREST The authors have nothing to disclose. REFERENCES 1. Ferlic RJ, Lee DH, Lopez-Ben RR. Pediatric Kienböck's disease: case report and review of the literature. Clin Orthop Relat Res. 2003;408:237-44. 2. Cvitanich M, Solomons M. Juvenile lunatomalacia is this Kienböck's disease? J Hand Surg Br. 2004;29:288-92. 3. Kazuki K, Uemura T, Okada M, Egi T. Time course of magnetic resonance images in an adolescent patient with Kienböck's disease treated by temporary scaphotrapezoidal joint fixation: a case report. J Hand Surg Am. 2006;31:63-7. 4. Kim TY, Culp RW, Osterman AL, Bednar JM. Kienbock s disease in children. Jefferson Orthopedic J. 1997;25:53-7. 5. Irisarri C, Kalb K, Ribak S. Infantile and juvenile lunatomalacia. J Hand Surg Eur Vol. 2010;35:544-8. 6. Greene WB. Kienböck disease in a child who has cerebral palsy. A case report. J Bone Joint Surg Am. 1996;78:1568-73. 7. Herzberg G, Mercier S, Charbonnier JP, Got P. Kienböck's disease in a 14-year-old gymnast: a case report. J Hand Surg Am. 2006;31:264-8. 8. Iwasaki N, Minami A, Ishikawa J, Kato H, Minami M. Radial osteotomies for teenage patients with Kienböck disease. Clin Orthop Relat Res. 2005;439:116-22. 9. Yajima H, Ono H, Tamai S. Temporary internal fixation of the scaphotrapezio-trapezoidal joint for the treatment of Kienböck's disease: a preliminary study. J Hand Surg Am. 1998;23:402-10. 10. Herdem M, Ozkan C, Bayram H. Overgrowth after radial shortening for Kienböck's disease in a teenager: case report. J Hand Surg Am. 2006;31:1322-5.
457 pissn : 1226-2102, eissn : 2005-8918 Case Report J Korean Orthop Assoc 2018; 53: 453-457 https://doi.org/10.4055/jkoa.2018.53.5.453 www.jkoa.org 소아에서발생한진행성 Kienbock 병 김동희 어일주 정민 성균관대학교의과대학삼성창원병원정형외과학교실 소아에서의 Kienbock 병은아주드물게발생한다. Kienbock 병의치료는이견이분분하며질병의자연경과에대한이해도에따라매우다르며보존적치료부터요골단축술, 삼주상유합술등수술적치료까지다양하지만소아에서명확한치료가이드라인은제시되어있지않다. 저자들은병기 IIIB의진행성 Kienbock 병으로진단된 14세소아에서석고고정을이용한보존적치료로성공적인치료결과를경험하여문헌고찰과함께보고하고자한다. 색인단어 : 월상골, 무혈성괴사, Kienbock 병, 소아 접수일 2017 년 3 월 12 일수정일 2017 년 7 월 2 일게재확정일 2017 년 11 월 21 일책임저자김동희 51353, 창원시마산회원구팔용로 158, 삼성창원병원정형외과 TEL 055-233-5201, FAX 055-233-8357, E-mail dhkim1149@gmail.com ORCID https://orcid.org/0000-0003-4358-9356 대한정형외과학회지 : 제 53 권제 5 호 2018 Copyright 2018 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/4.0/) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.