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Simultaneous ossification of the posterior longitudinal ligament and ossification of the ligamentum flavum causing upper thoracic myelopathy in DISH: case report and literature review

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Abstract

A rare case of a 44-year-old Chinese male with diffuse idiopathic skeletal hyperostosis (DISH) and simultaneous ossification of the posterior longitudinal ligament (OPLL) and ossification of the ligamentum flavum (OLF) at T1–2 causing thoracic myelopathy is reported herein. Posterior decompression without extirpating the OPLL was performed at T1–2. Postoperatively, symptoms were greatly improved, with remaining hyperreflexia and Grade 4/5 muscle strength in the lower extremities. The Japanese Orthopedic Association score improved from 5 preoperatively to 9 at final follow-up. The presence of a cyst due to leakage of cerebrospinal fluid was confirmed by MRI at day 27, but it resolved after conservative management. The clinical manifestation of DISH, the relationship among DISH, OPLL, and OLF, and management of thoracic myelopathy due to OPLL and OLF were reviewed.

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References

  1. Matge G (2005) Surgical management of cervical radiculopathy in Forestier’s disease. Case report and review. Neurochirurgie 51:15–18

    Article  PubMed  CAS  Google Scholar 

  2. Ozkalkanli MY, Katircioglu K, Ozkalkanli DT, Savaci S (2006) Airway management of a patient with Forestier’s disease. J Anesth 20:304–306. doi:10.1007/s00540-006-0418-5

    Article  PubMed  Google Scholar 

  3. McCafferty RR, Harrison MJ, Tamas LB, Larkins MV (1995) Ossification of the anterior longitudinal ligament and Forestier’s disease: an analysis of seven cases. J Neurosurg 83:13–17. doi:10.3171/jns.1995.83.1.0013

    Article  PubMed  CAS  Google Scholar 

  4. Pappone N, Di Girolamo C, Del Puente A, Scarpa R, Oriente P (1996) Diffuse idiopathic skeletal hyperostosis (DISH): a retrospective analysis. Clin Rheumatol 15:121–124

    Article  PubMed  CAS  Google Scholar 

  5. Chacko AG, Daniel RT (2007) Multilevel cervical oblique corpectomy in the treatment of ossified posterior longitudinal ligament in the presence of ossified anterior longitudinal ligament. Spine (Phila Pa 1976) 32:E575–E580

    Article  Google Scholar 

  6. Jun BY, Yoon KJ, Crockard A (2002) Retro-odontoid pseudotumor in diffuse idiopathic skeletal hyperostosis. Spine (Phila Pa 1976) 27:E266–E270

    Article  Google Scholar 

  7. Storch MJ, Hubbe U, Glocker FX (2008) Cervical myelopathy caused by soft-tissue mass in diffuse idiopathic skeletal hyperostosis. Eur Spine J 17(Suppl 2):S243–S247. doi:10.1007/s00586-007-0508-6

    Article  PubMed  Google Scholar 

  8. Patel NP, Wright NM, Choi WW, McBride DQ, Johnson JP (2002) Forestier disease associated with a retroodontoid mass causing cervicomedullary compression. J Neurosurg 96:190–196

    PubMed  Google Scholar 

  9. Goto S, Tanno T, Moriya H (1995) Cervical myelopathy caused by pseudoarthrosis between the atlas and axis associated with diffuse idiopathic skeletal hyperostosis. Spine (Phila Pa 1976) 20:2572–2575

    Article  CAS  Google Scholar 

  10. Pascal-Moussellard H, Drossard G, Cursolles JC, Catonne Y, Smadja D (2006) Myelopathy by lesions of the craniocervical junction in a patient with forestier disease. Spine (Phila Pa 1976) 31:E557–E560

    Article  Google Scholar 

  11. Griffiths ID, Fitzjohn TP (1987) Cervical myelopathy, ossification of the posterior longitudinal ligament, and diffuse idiopathic skeletal hyperostosis: problems in investigation. Ann Rheum Dis 46:166–168

    Article  PubMed  CAS  Google Scholar 

  12. Pouchot J, Watts CS, Esdaile JM, Hill RO (1987) Sudden quadriplegia complicating ossification of the posterior longitudinal ligament and diffuse idiopathic skeletal hyperostosis. Arthritis Rheum 30:1069–1072

    Article  PubMed  CAS  Google Scholar 

  13. Chiba H, Annen S, Shimada T, Imura S (1992) Atlantoaxial subluxation complicated by diffuse idiopathic skeletal hyperostosis. A case report. Spine (Phila Pa 1976) 17:1414–1417

    Article  CAS  Google Scholar 

  14. Kawabori M, Hida K, Akino M, Yano S, Saito H, Iwasaki Y (2009) Cervical myelopathy by C1 posterior tubercle impingement in a patient with DISH. Spine (Phila Pa 1976) 34:E709–E711

    Article  Google Scholar 

  15. Epstein NE (2000) Simultaneous cervical diffuse idiopathic skeletal hyperostosis and ossification of the posterior longitudinal ligament resulting in dysphagia or myelopathy in two geriatric North Americans. Surg Neurol 53:427–431 (discussion 431)

    Article  PubMed  CAS  Google Scholar 

  16. Razmi R, Khong KS (2001) Cervical cord injury in an elderly man with a fused spine—a case report. Singap Med J 42:477–481

    CAS  Google Scholar 

  17. Alenghat JP, Hallett M, Kido DK (1982) Spinal cord compression in diffuse idiopathic skeletal hyperostosis. Radiology 142:119–120

    PubMed  CAS  Google Scholar 

  18. Reisner A, Stiles RG, Tindall SC (1990) Diffuse idiopathic skeletal hyperostosis causing acute thoracic myelopathy: a case report and discussion. Neurosurgery 26:507–511

    Article  PubMed  CAS  Google Scholar 

  19. Wilson FM, Jaspan T (1990) Thoracic spinal cord compression caused by diffuse idiopathic skeletal hyperostosis (DISH). Clin Radiol 42:133–135

    Article  PubMed  CAS  Google Scholar 

  20. Chi D, Miyamoto K, Hosoe H, Kawai G, Ohnishi K, Suzuki N, Sumi H, Shimizu K (2008) Symptomatic lumbar mobile segment with spinal canal stenosis in a fused spine associated with diffused idiopathic skeletal hyperostosis. Spine J 8:1019–1023

    Article  PubMed  Google Scholar 

  21. Utsinger PD (1985) Diffuse idiopathic skeletal hyperostosis. Clin Rheum Dis 11:325–351

    PubMed  CAS  Google Scholar 

  22. Crockard HA, Sett P, Geddes JF, Stevens JM, Kendall BE, Pringle JA (1991) Damaged ligaments at the craniocervical junction presenting as an extradural tumour: a differential diagnosis in the elderly. J Neurol Neurosurg Psychiatry 54:817–821

    Article  PubMed  CAS  Google Scholar 

  23. Ono M, Russell WJ, Kudo S, Kuroiwa Y, Takamori M, Motomura S, Murakami J (1982) Ossification of the thoracic posterior longitudinal ligament in a fixed population. Radiological and neurological manifestations. Radiology 143:469–474

    PubMed  CAS  Google Scholar 

  24. Ehara S, Shimamura T, Nakamura R, Yamazaki K (1998) Paravertebral ligamentous ossification: DISH, OPLL and OLF. Eur J Radiol 27:196–205

    Article  PubMed  CAS  Google Scholar 

  25. Resnick D, Guerra J Jr, Robinson CA, Vint VC (1978) Association of diffuse idiopathic skeletal hyperostosis (DISH) and calcification and ossification of the posterior longitudinal ligament. AJR Am J Roentgenol 131:1049–1053

    PubMed  CAS  Google Scholar 

  26. Hukuda S, Mochizuki T, Ogata M, Shichikawa K (1983) The pattern of spinal and extraspinal hyperostosis in patients with ossification of the posterior longitudinal ligament and the ligamentum flavum causing myelopathy. Skeletal Radiol 10:79–85

    Article  PubMed  CAS  Google Scholar 

  27. Wiberg C, Klatt AR, Wagener R, Paulsson M, Bateman JF, Heinegard D, Morgelin M (2003) Complexes of matrilin-1 and biglycan or decorin connect collagen VI microfibrils to both collagen II and aggrecan. J Biol Chem 278:37698–37704

    Article  PubMed  CAS  Google Scholar 

  28. Tsukahara S, Miyazawa N, Akagawa H, Forejtova S, Pavelka K, Tanaka T, Toh S, Tajima A, Akiyama I, Inoue I (2005) COL6A1, the candidate gene for ossification of the posterior longitudinal ligament, is associated with diffuse idiopathic skeletal hyperostosis in Japanese. Spine (Phila Pa 1976) 30:2321–2324

    Article  Google Scholar 

  29. Havelka S, Vesela M, Pavelkova A, Ruzickova S, Koga H, Maeda S, Inoue I, Halman L (2001) Are DISH and OPLL genetically related? Ann Rheum Dis 60:902–903

    PubMed  CAS  Google Scholar 

  30. Yamazaki M, Mochizuki M, Ikeda Y, Sodeyama T, Okawa A, Koda M, Moriya H (2006) Clinical results of surgery for thoracic myelopathy caused by ossification of the posterior longitudinal ligament: operative indication of posterior decompression with instrumented fusion. Spine (Phila Pa 1976) 31:1452–1460

    Article  Google Scholar 

  31. Hioki A, Miyamoto K, Hosoe H, Shimizu K (2008) Two-staged decompression for thoracic paraparesis due to the combined ossification of the posterior longitudinal ligament and the ligamentum flavum: a case report. Arch Orthop Trauma Surg 128:175–177. doi:10.1007/s00402-007-0336-5

    Article  PubMed  Google Scholar 

  32. Tomita K, Kawahara N, Baba H, Kikuchi Y, Nishimura H (1990) Circumspinal decompression for thoracic myelopathy due to combined ossification of the posterior longitudinal ligament and ligamentum flavum. Spine (Phila Pa 1976) 15:1114–1120

    Article  CAS  Google Scholar 

  33. Hanai K, Ogikubo O, Miyashita T (2002) Anterior decompression for myelopathy resulting from thoracic ossification of the posterior longitudinal ligament. Spine (Phila Pa 1976) 27:1070–1076

    Article  Google Scholar 

  34. Fujimura Y, Nishi Y, Nakamura M, Toyama Y, Suzuki N (1997) Long-term follow-up study of anterior decompression and fusion for thoracic myelopathy resulting from ossification of the posterior longitudinal ligament. Spine (Phila Pa 1976) 22:305–311

    Article  CAS  Google Scholar 

  35. Kurosa Y, Yamaura I, Nakai O, Shinomiya K (1996) Selecting a surgical method for thoracic myelopathy caused by ossification of the posterior longitudinal ligament. Spine (Phila Pa 1976) 21:1458–1466

    Article  CAS  Google Scholar 

  36. Takahata M, Ito M, Abumi K, Kotani Y, Sudo H, Minami A (2008) Clinical results and complications of circumferential spinal cord decompression through a single posterior approach for thoracic myelopathy caused by ossification of posterior longitudinal ligament. Spine (Phila Pa 1976) 33:1199–1208

    Article  Google Scholar 

  37. Matsumoto M, Chiba K, Toyama Y, Takeshita K, Seichi A, Nakamura K, Arimizu J, Fujibayashi S, Hirabayashi S, Hirano T, Iwasaki M, Kaneoka K, Kawaguchi Y, Ijiri K, Maeda T, Matsuyama Y, Mikami Y, Murakami H, Nagashima H, Nagata K, Nakahara S, Nohara Y, Oka S, Sakamoto K, Saruhashi Y, Sasao Y, Shimizu K, Taguchi T, Takahashi M, Tanaka Y, Tani T, Tokuhashi Y, Uchida K, Yamamoto K, Yamazaki M, Yokoyama T, Yoshida M, Nishiwaki Y (2008) Surgical results and related factors for ossification of posterior longitudinal ligament of the thoracic spine: a multi-institutional retrospective study. Spine (Phila Pa 1976) 33:1034–1041

    Article  Google Scholar 

  38. Shen FH, Samartzis D (2007) Successful nonoperative treatment of a three-column thoracic fracture in a patient with ankylosing spondylitis: existence and clinical significance of the fourth column of the spine. Spine (Phila Pa 1976) 32:E423–E427

    Article  Google Scholar 

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Authors and Affiliations

  1. Department of Orthopedics, Changzheng Hospital, The Second Military Medical University, 415 Fengyang Road, Huangpu District, Shanghai, 200003, People’s Republic of China

    Qunfeng Guo, Bin Ni, Jun Yang, Zhuangchen Zhu & Jian Yang

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  1. Qunfeng Guo
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  2. Bin Ni
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  3. Jun Yang
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  4. Zhuangchen Zhu
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Correspondence to Bin Ni.

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Guo, Q., Ni, B., Yang, J. et al. Simultaneous ossification of the posterior longitudinal ligament and ossification of the ligamentum flavum causing upper thoracic myelopathy in DISH: case report and literature review. Eur Spine J 20 (Suppl 2), 195–201 (2011). https://doi.org/10.1007/s00586-010-1538-z

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