DOI QR코드

DOI QR Code

Recurrent Shoulder Dislocation and Proximal Humerus Fracture in Neurofibromatosis Type I - A Case Report -

제 1형 신경 섬유종증 환자에서 발생한 재발성 견관절 탈구 및 근위 상완골 골절 - 1예 보고 -

  • Hwang, Tae Hyok (Department of Orthopaedic Surgery, Good Samsun Hospital) ;
  • Cho, Hyung Lae (Department of Orthopaedic Surgery, Good Samsun Hospital) ;
  • Wang, Tae Hyun (Department of Orthopaedic Surgery, Good Samsun Hospital) ;
  • Yang, Hui Sun (Department of Orthopaedic Surgery, Good Samsun Hospital)
  • 황태혁 (좋은삼선병원 정형외과) ;
  • 조형래 (좋은삼선병원 정형외과) ;
  • 왕태현 (좋은삼선병원 정형외과) ;
  • 양희순 (좋은삼선병원 정형외과)
  • Received : 2013.09.24
  • Accepted : 2013.12.30
  • Published : 2013.12.31

Abstract

We report a case of recurrent shoulder dislocation and combined proximal humerus fracture in neurofibromatosis type I. A twenty-year-old male patient with known neurofibromatosis type I presented with right shoulder dislocation and proximal humerus fracture following a minor trauma. His injured arm also had large plexiform neurofibromas on the elbow, and the dislocation of the shoulder joint was proven to be recurrent. Bony deformation of the humerus and scapula were revealed on X-ray and computed tomography, as well as multiple neurofibromatic changes of the deltoid and periscapular muscles were noted on magnetic resonance imaging. Open reduction and capsular reconstruction were performed and humeral fracture was stabilized with a reconstruction plate. Satisfactory union and functional result were achieved and the dislocation did not recur until the 7-month followup after the procedure. Recurrent shoulder dislocation and combined proximal humerus fracture in neurofibromatosis type I are rare complications. However, it should still be considered in the orthopaedic evaluation process of the diseases.

제 1형 신경 섬유종증 환자에서 발생한 재발성 견관절 탈구 및 동반된 근위 상완골 골절 1예를 보고하고자 한다. 제 1형 신경 섬유종증으로 진단된 20세 남자로 가벼운 외상 후 발생한 우측 견관절의 탈구와 근위 상완골 골절로 내원하였다. 수상측 주관절부에는 큰 망상형 신경 섬유종들이 있었으며 견관절 탈구는 재발성 병변이었다. 단순 방사선과 컴퓨터 단층 촬영 소견 상 상완골과 견갑골의 골성 변형을 보였고 자기 공명 영상 검사 상 삼각근과 회전근 개에 다발성 신경 섬유종성 변화가 관찰되었다. 관혈적 정복술 및 관절막 재건술을 시행하였으며 근위 상완골 골절은 재건 금속판을 이용하여 고정하였다. 골유합과 만족스런 기능 회복을 보였으며 수술 후 7개월 추시 상 견관절 탈구의 재발은 관찰되지 않았다. 제 1형 신경 섬유종증 환자에서 발생한 견관절 재발성 탈구와 근위 상완골 골절은 드문 합병증이나 질환의 정형외과적 접근 시 고려해야 할 것으로 사료된다.

Keywords

References

  1. Fienman NL, Yakovac WC. Neurofibromatosis in childhood. J Pediatr. 1970;76:339-46. https://doi.org/10.1016/S0022-3476(70)80472-3
  2. Crawford AH Jr, Bagamery N. Osseous manifestations of neurofibromatosis in childhood. J Pediatr Orthop. 1986;6:72-88. https://doi.org/10.1097/01241398-198601000-00015
  3. Vitale MG, Guha A, Skaggs DL. Orthopaedic manifestations of neurofibromatosis in children: an update. Clin Orthop Relat Res. 2002;Aug:107-18.
  4. Feldman DS, Jordan C, Fonesca L. Orthopaedic manifestations of neurofibromatosis type 1. J Am Acad Orthop Surg. 2010;18:346-57. https://doi.org/10.5435/00124635-201006000-00007
  5. Stevenson DA, Birch PH, Friedman JM, et al. Descriptive analysis of tibial pseudarthrosis in patients with neurofibromatosis 1. Am J Med Genet. 1999;84:413-9. https://doi.org/10.1002/(SICI)1096-8628(19990611)84:5<413::AID-AJMG5>3.0.CO;2-1
  6. Kolanczyk M, Kossler N, Kuhnisch J, et al. Multiple roles for neurofibromin in skeletal development and growth. Hum Mol Genet. 2007;16:874-86. https://doi.org/10.1093/hmg/ddm032
  7. Galbraith JG, Butler JS, Harty JA. Recurrent spontaneous hip dislocation in a patient with neurofibromatosis type 1: a case report. J Med Case Rep. 2011;5:106. https://doi.org/10.1186/1752-1947-5-106
  8. Haga N, Nakamura S, Taniguchi K, Iwaya T. Pathologic dislocation of the hip in von Recklinghausen’s disease: a report of two cases. J Pediatr Orthop. 1994;14:674-6. https://doi.org/10.1097/01241398-199409000-00023
  9. Phillips JE, McMaster MJ. Pathological dislocation of the hip in neurofibromatosis. J R Coll Surg Edinb. 1987;32:180-2.
  10. Tonsgard JH. Clinical manifestations and management of neurofibromatosis type 1. Semin Pediatr Neurol. 2006;13:2-7. https://doi.org/10.1016/j.spen.2006.01.005