DOI QR코드

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Cervical Spondylotic Myelopathy due to the Ochronotic Arthropathy of the Cervical Spine

  • Li, Nan (Department of Spine Surgery, Beijing Jishuitan Hospital) ;
  • Tian, Wei (Department of Spine Surgery, Beijing Jishuitan Hospital) ;
  • Yuan, Qiang (Department of Spine Surgery, Beijing Jishuitan Hospital) ;
  • He, Da (Department of Spine Surgery, Beijing Jishuitan Hospital)
  • 투고 : 2014.08.27
  • 심사 : 2015.01.05
  • 발행 : 2016.01.01

초록

Ochronosis is a musculoskeletal manifestation of alkaptonuria, a rare hereditary metabolic disorder occurs due to the absence of homogentisic acid oxidase and leading to various systemic abnormalities related to deposition of homogentisic acid pigmentation (ochronotic pigmentation). The present case reports the clinical features, radiographic findings, treatments and results of a cervical spondylotic myelopathy woman patient due to the ochronotic arthropathy of the cervical spine. The patient aged 62 years was presented with gait disturbance and hand clumsiness. Physical examination, X-rays, computed tomography and lab results of the urine sample confirmed the presence of ochronosis with the involvement of the cervical spine. The patient underwent a modified cervical laminoplasty due to multi-segment spinal cord compression. The postoperative follow-up showed a good functional outcome with patient satisfaction. The present study concludes the conditions and important diagnostic and surgical aspects of a patient. It is necessary to identify the condition clinically and if cord compression is observed, appropriate surgical interventions needs to be instituted.

키워드

참고문헌

  1. Alrehaily A, Pope JE : Alkaptonuria with atypical joint involvement. J Rheumatol 29 : 198-199, 2002
  2. Aynaci O, Onder C, Turhan AU : Bilateral hip arthroplasty for ochronotic arthropathy. Clin Rheumatol 19 : 150-152, 2000 https://doi.org/10.1007/s100670050035
  3. Chua SY, Chang HC : Bilateral spontaneous rupture of the quadriceps tendon as an initial presentation of alkaptonuria--a case report. Knee 13 : 408-410, 2006 https://doi.org/10.1016/j.knee.2006.06.008
  4. Corra T, Zaccala M, Galante M : Ochronotic arthropathy : rapid destructive hip osteoarthritis associated with metabolic disease. Clin Rheumatol 14 : 474-477, 1995 https://doi.org/10.1007/BF02207686
  5. Dom K, Pittevils T : Ochronotic arthropathy : the black hip. Case report and review of the literature. Acta Orthop Belg 63 : 122-125, 1997
  6. Emel E, Karagoz F, Aydín IH, Hacisalihoglu S, Seyithanoglu MH : Alkaptonuria with lumbar disc herniation : a report of two cases. Spine (Phila Pa 1976) 25 : 2141-2144, 2000 https://doi.org/10.1097/00007632-200008150-00021
  7. Fernandez-Canon JM, Granadino B, Beltran-Valero de Bernabe D, Renedo M, Fernandez-Ruiz E, Penalva MA, et al. : The molecular basis of alkaptonuria. Nat Genet 14 : 19-24, 1996 https://doi.org/10.1038/ng0996-19
  8. Gaines JJ Jr : The pathology of alkaptonuric ochronosis. Hum Pathol 20 : 40-46, 1989 https://doi.org/10.1016/0046-8177(89)90200-1
  9. Justesen P, Anderson PE Jr : Radiologic manifestations in alcaptonuria. Skeletal Radiol 11 : 204-208, 1984 https://doi.org/10.1007/BF00349495
  10. Kusakabe N, Tsuzuki N, Sonada M : Compression of the cervical cord due to alcaptonuric arthropathy of the atlanto-axial joint. A case report. J Bone Joint Surg Am 77 : 274-277, 1995 https://doi.org/10.2106/00004623-199502000-00016
  11. Van Offel JF, De Clerck LS, Francx LM, De Jonge MJ, Stevens WJ : A patient with ochronotic arthropathy and spondylopathy : a difficult differential diagnosis with spondylitis ankylosans. Clin Exp Rheumatol 13 : 259-261, 1995
  12. Van Offel JF, De Clerck LS, Francx LM, Stevens WJ : The clinical manifestations of ochronosis : a review. Acta Clin Belg 50 : 358-362, 1995 https://doi.org/10.1080/17843286.1995.11718475
  13. Wolff JA, Barshop B, Nyhan WL, Leslie J, Seegmiller JE, Gruber H, et al. : Effects of ascorbic acid in alkaptonuria : alterations in benzoquinone acetic acid and an ontogenic effect in infancy. Pediatr Res 26 : 140-144, 1989 https://doi.org/10.1203/00006450-198908000-00015
  14. Zhao BH, Chen BC, Shao de C, Zhang Q : Osteoarthritis? Ochronotic arthritis! A case study and review of the literature. Knee Surg Sports Traumatol Arthrosc 17 : 778-781, 2009 https://doi.org/10.1007/s00167-009-0778-0

피인용 문헌

  1. Ochronotic arthropathy—a rare clinical case vol.2018, pp.9, 2018, https://doi.org/10.1093/omcr/omy069
  2. “As Black as Ink”: A Case of Alkaptonuria-Associated Myelopathy and a Review of the Literature vol.44, pp.1, 2019, https://doi.org/10.1097/brs.0000000000002755
  3. Cervical Myelopathy Due to Ochronosis: An Intraoperative Suspicion vol.21, pp.None, 2016, https://doi.org/10.12659/ajcr.924575