Ossification of the ligamentum flavum in a Caucasian man

Case report

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Abnormal ossification of spinal ligaments is a well-known cause of myelopathy in East Asian populations, with ossification of the ligamentum flavum (OLF) and the posterior longitudinal ligament being the most prevalent. In Caucasian populations, OLF is rare, and there has been only 1 documented case of the disease affecting more than 5 spinal levels. In this report, the authors describe the clinical presentation, imaging characteristics, and management of the second published case of a Caucasian man with OLF affecting almost the entire thoracic spine. The literature is then reviewed with regard to OLF epidemiology, pathogenesis, presentation, and treatment.

Abbreviations used in this paper:CLF = calcification of the ligamentum flavum; OLF = ossification of ligamentum flavum; OPLL = ossification of the posterior longitudinal ligament.
Article Information

Contributor Notes

Address correspondence to: Daniel M. Sciubba, M.D., 600 North Wolfe Street, Meyer 8-161, Baltimore, Maryland 21287. email: dsciubb1@jhmi.edu.

© AANS, except where prohibited by US copyright law.

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References
  • 1

    Adams JEDavies M: Intra-spinal new bone formation and spinal cord compression in familial hypophosphataemic vitamin D resistant osteomalacia. Q J Med 61:111711291986

    • Search Google Scholar
    • Export Citation
  • 2

    Aizawa TSato TSasaki HKusakabe TMorozumi NKokubun S: Thoracic myelopathy caused by ossification of the ligamentum flavum: clinical features and surgical results in the Japanese population. J Neurosurg Spine 5:5145192006

    • Search Google Scholar
    • Export Citation
  • 3

    Aizawa TSato TSasaki HMatsumoto FMorozumi NKusakabe T: Results of surgical treatment for thoracic myelopathy: minimum 2-year follow-up study in 132 patients. J Neurosurg Spine 7:13202007

    • Search Google Scholar
    • Export Citation
  • 4

    Akhaddar AMansouri AZrara IGazzaz MMaftah MMostarchid B: Thoracic spinal cord compression by ligamentum flavum ossifications. Joint Bone Spine 69:3193232002

    • Search Google Scholar
    • Export Citation
  • 5

    Arafat QWJackowski AChavda SVWest RJ: Case report: ossification of the thoracic ligamenta flava in a Caucasian: a rare cause of myelopathy. Br J Radiol 66:119311961993

    • Search Google Scholar
    • Export Citation
  • 6

    Ben Hamouda KJemel HHaouet SKhaldi M: Thoracic myelopathy caused by ossification of the ligamentum flavum: a report of 18 cases. J Neurosurg 99:1571612003

    • Search Google Scholar
    • Export Citation
  • 7

    Celli PCaroli ETrillo GFerrante L: Ossification of the ligamentum flavum in a Caucasian. Case report. J Neurosurg Sci 46:96992002

    • Search Google Scholar
    • Export Citation
  • 8

    Couto ARBrown MA: Genetic factors in the pathogenesis of CPPD crystal deposition disease. Curr Rheumatol Rep 9:2312362007

  • 9

    Debiais FBataille BDebiais PAzais IBontoux DAl-calay M: Femoral neuropathy secondary to ossification of the ligamentum flavum. J Rheumatol 27:131313142000

    • Search Google Scholar
    • Export Citation
  • 10

    Epstein N: Thoracic ossification of the posterior longitudinal ligament, ossification of the yellow ligament from T9–T12 with superimposed acute T10/T11 disc herniation: controversies in surgical management. J Spinal Disord 9:446 4501996

    • Search Google Scholar
    • Export Citation
  • 11

    Epstein NE: The frequency and etiology of intraoperative dural tears in 110 predominantly geriatric patients undergoing multilevel laminectomy with noninstrumented fusions. J Spinal Disord Tech 20:3803862007

    • Search Google Scholar
    • Export Citation
  • 12

    Farooq MTChao TBennett M: Paraplegia following posttraumatic thoracic spinal stenosis: a case report. Arch Phys Med Rehabil 77:84851996

    • Search Google Scholar
    • Export Citation
  • 13

    Folinais DLassale BBenacerraf RDeburge A: [Dorsal radiculo- myelopathy due to ossification of the yellow ligaments. Apropos of a case.]. Rev Rhum Mal Osteoartic 55:5955991988. (Fr)

    • Search Google Scholar
    • Export Citation
  • 14

    Fong SYWong HK: Thoracic myelopathy secondary to ligamentum flavum ossification. Ann Acad Med Singapore 33:3403462004

  • 15

    Gil-Salú JLLópez-Escobar MGómez-Cárdenas ERodríguez- Peña FRepeto-López JALópez-López J: [Calcification of the thoracic yellow ligament as a cause of myelopathy.]. Rev Neurol 34:7507532002. (Span)

    • Search Google Scholar
    • Export Citation
  • 16

    Gondim JRamos F Junior: [Thoracic spinal cord compression at two levels due to ligamentum flavum calcification. Case report.]. Arq Neuropsiquiatr 56:3123161998. (Port)

    • Search Google Scholar
    • Export Citation
  • 17

    Goodman JMKuzma BB: Ossification of the ligamentum flavum with myelopathy. Surg Neurol 46:3963971996

  • 18

    Hirabayashi HEbara STakahashi JNarasaki KTakahara KMurakami G: Surgery for thoracic myelopathy caused by ossification of the ligamentum flavum. Surg Neurol 69:1141162008

    • Search Google Scholar
    • Export Citation
  • 19

    Ilkko EPyhtinen JSajanti J: Thoracic spinal canal stenosis: a case report. Eur J Radiol 18:1261281994

  • 20

    Kaneyama SDoita MNishida KShimomura TMaeno KTamura Y: Thoracic myelopathy due to ossification of the yellow ligament in young baseball pitchers. J Spinal Disord Tech 21:68712008

    • Search Google Scholar
    • Export Citation
  • 21

    Kawano NMatsuno TMiyazawa SIida HYada KKobayashi N: Calcium pyrophosphate dihydrate crystal deposition disease in the cervical ligamentum flavum. J Neurosurg 68:6136201988

    • Search Google Scholar
    • Export Citation
  • 22

    Kobayashi SOkada KOnoda KHorikoshi S: Ossification of the cervical ligamentum flavum. Surg Neurol 35:2342381991

  • 23

    Kong QMa XLi FGuo ZQi QLi W: COL6A1 polymorphisms associated with ossification of the ligamentum flavum and ossification of the posterior longitudinal ligament. Spine 32:283428382007

    • Search Google Scholar
    • Export Citation
  • 24

    Kosaka TImakiire AMizuno FYamamoto K: Activation of nuclear factor kappaB at the onset of ossification of the spinal ligaments. J Orthop Sci 5:5725782000

    • Search Google Scholar
    • Export Citation
  • 25

    Kruse JJAwasthi DHarris MWaguespack A: Ossification of the ligamentum flavum as a cause of myelopathy in North America: report of three cases. J Spinal Disord 13:22252000

    • Search Google Scholar
    • Export Citation
  • 26

    Kuh SUKim YSCho YEJin BHKim KSYoon YS: Contributing factors affecting the prognosis surgical outcome for thoracic OLF. Eur Spine J 15:4854912006

    • Search Google Scholar
    • Export Citation
  • 27

    Lee KSShim JJDoh JWYoon SMBae HGYun IG: Transient paraparesis after laminectomy in a patient with multi-level ossification of the spinal ligament. J Korean Med Sci 19:6246262004

    • Search Google Scholar
    • Export Citation
  • 28

    Li FChen QXu K: Surgical treatment of 40 patients with thoracic ossification of the ligamentum flavum. J Neurosurg Spine 4:1911972006

    • Search Google Scholar
    • Export Citation
  • 29

    Li KKChung OMChang YPSo YC: Myelopathy caused by ossification of ligamentum flavum. Spine 27:E308E3122002

  • 30

    Maiuri FIaconetta GGambardella A: Ossification of the yellow ligament causing thoracic cord compression. Arch Orthop Trauma Surg 120:3463482000

    • Search Google Scholar
    • Export Citation
  • 31

    Matsui HKatoh YTsuji H: Untreated hypophosphatemic vitamin D-resistant rickets with symptomatic ossification of the ligamentum flavum. J Spinal Disord 4:1101131991

    • Search Google Scholar
    • Export Citation
  • 32

    Miyakoshi NShimada YSuzuki THongo MKasukawa YOkada K: Factors related to long-term outcome after decompressive surgery for ossification of the ligamentum flavum of the thoracic spine. J Neurosurg 99:2512562003

    • Search Google Scholar
    • Export Citation
  • 33

    Miyasaka KKaneda KSato SIwasaki YAbe STakei H: Myelopathy due to ossification or calcification of the ligamentum flavum: radiologic and histologic evaluations. AJNR Am J Neuroradiol 4:6296321983

    • Search Google Scholar
    • Export Citation
  • 34

    Miyazawa NAkiyama I: Ossification of the ligamentum flavum of the cervical spine. J Neurosurg Sci 51:1391442007

  • 35

    Mobbs RJDvorak M: Ossification of the ligamentum flavum: diet and genetics. J Clin Neurosci 14:7037052007

  • 36

    Mohindra SChhabra RMukherjee KKGupta SKVaiphei KKhosla VK: Spinal compression due to ossified yellow ligament: a short series of 5 patients and literature review. Surg Neurol 65:3773842006

    • Search Google Scholar
    • Export Citation
  • 37

    Muthukumar N: Ossification of the ligamentum flavum as a result of fluorosis causing myelopathy: report of two cases. Neurosurgery 56:E6222005

    • Search Google Scholar
    • Export Citation
  • 38

    Nakamura IIkegawa SOkawa AOkuda SKoshizuka YKawaguchi H: Association of the human NPPS gene with ossification of the posterior longitudinal ligament of the spine (OPLL). Hum Genet 104:4924971999

    • Search Google Scholar
    • Export Citation
  • 39

    Okawa ANakamura IGoto SMoriya HNakamura YIkegawa S: Mutation in Npps in a mouse model of ossification of the posterior longitudinal ligament of the spine. Nat Genet 19:2712731998

    • Search Google Scholar
    • Export Citation
  • 40

    Omojola MFCardoso ERFox AJDrake CGDurward QJ: Thoracic myelopathy secondary to ossified ligamentum flavum. J Neurosurg 56:4484501982

    • Search Google Scholar
    • Export Citation
  • 41

    Pantazis GTsitsopoulos PBibis AMihas CChatzistamou IKouzelis C: Symptomatic ossification of the ligamentum flavum at the lumbar spine: a retrospective study. Spine 33:306 3112008

    • Search Google Scholar
    • Export Citation
  • 42

    Parekh HCGurusinghe NTPerera SSPrabhu SS: Ossification of the ligamentum flavum in a Caucasian: case report. Br J Neurosurg 7:6876901993

    • Search Google Scholar
    • Export Citation
  • 43

    Pascal-Mousselard HSmadja DCabre PRaynaud MCatonne Y: Ossification of the ligamenta flava with severe myelopathy in a black patient. A case report. Spine 23:160716081998

    • Search Google Scholar
    • Export Citation
  • 44

    Pascal-Moussellard HCabre PSmadja DCatonne Y: Symptomatic ossification of the ligamentum flavum: a clinical series from the French Antilles. Spine 30:E400E4052005

    • Search Google Scholar
    • Export Citation
  • 45

    Pascal-Moussellard HCabre PSmadja DKaidomar SCatonne Y: Myelopathy due to calcification of the cervical ligamenta flava: a report of two cases in West Indian patients. Eur Spine J 8:2382401999

    • Search Google Scholar
    • Export Citation
  • 46

    Payer MBruder EFischer JABenini A: Thoracic myelopathy due to enlarged ossified yellow ligaments. Case report and review of the literature. J Neurosurg 92:1 Suppl1051082000

    • Search Google Scholar
    • Export Citation
  • 47

    Polgar F: Uber interarkuelle wirbel verkalkung. Fortschr Geb Rontgenstrahlen Nuklearmed Erganzungsbd 40:2922981929

  • 48

    Renier JCGuy GSeret PFrancois H: [Dorsal spinal cord disease caused by ossification of the ligamentum flavum and D10–D11 posterior arthrosis.]. Rev Rhum Mal Osteoartic 55:6656671988. (Fr)

    • Search Google Scholar
    • Export Citation
  • 49

    Rivierez MVally P: [Ossification of ligamentum flavum unmasked by acute paraplegia.]. Neurochirurgie 47:5725752001. (Fr)

  • 50

    Sakou TTaketomi EMatsunaga SYamaguchi MSonoda SYashiki S: Genetic study of ossification of the posterior longitudinal ligament in the cervical spine with human leukocyte antigen haplotype. Spine 16:124912521991

    • Search Google Scholar
    • Export Citation
  • 51

    Sato TKokubun STanaka YIshii Y: Thoracic myelopathy in the Japanese: epidemiological and clinical observations on the cases in Miyagi Prefecture. Tohoku J Exp Med 184:1111998

    • Search Google Scholar
    • Export Citation
  • 52

    Sharma RRMahapatra APawar SJSousa JLad SDAthale SD: Spinal cord and cauda equina compression in 'DISH'. Neurol India 49:1481522001

    • Search Google Scholar
    • Export Citation
  • 53

    Sharma RRRamakantan RBhama BAMathad NV: Late post-traumatic spinal stenotic progressive myelo-radiculopathy. J Postgrad Med 36:33371990

    • Search Google Scholar
    • Export Citation
  • 54

    Shimamura TKato SToba TYamazaki KEhara S: Sagittal splitting laminoplasty for spinal canal enlargement for ossification of the spinal ligaments (OPLL and OLF). Semin Musculoskelet Radiol 5:2032062001

    • Search Google Scholar
    • Export Citation
  • 55

    Shiraishi TCrock HVLewis P: Thoracic myelopathy due to isolated ossification of the ligamentum flavum. J Bone Joint Surg Br 77:1311331995

    • Search Google Scholar
    • Export Citation
  • 56

    Singh PKar AMGarg RK: Calcification of the thoracic ligamentum flavum: a rare cause of myelopathy. J Assoc Physicians India 49:8438442001

    • Search Google Scholar
    • Export Citation
  • 57

    Sugimura HKakitsubata YSuzuki YKakitsubata STamura SUwada O: MRI of ossification of ligamentum flavum. J Comput Assist Tomogr 16:73761992

    • Search Google Scholar
    • Export Citation
  • 58

    Suojanen JNLipson SJ: Spinal cord compression secondary to ossified ligamentum flavum. J Spinal Disord 2:2382401989

  • 59

    Tanaka TIkari KFurushima KOkada ATanaka HFurukawa K: Genomewide linkage and linkage disequilibrium analyses identify COL6A1, on chromosome 21, as the locus for ossification of the posterior longitudinal ligament of the spine. Am J Hum Genet 73:8128222003

    • Search Google Scholar
    • Export Citation
  • 60

    Tokala DPLam KSPrince HG: Ossification of the proximal thoracic ligamenta flava causing acute myelopathy in a Caucasian: case report and literature review. Spinal Cord 45:3103132007

    • Search Google Scholar
    • Export Citation
  • 61

    Trivedi PBehari SPaul LBanerji DJain VKChhabra DK: Thoracic myelopathy secondary to ossified ligamentum flavum. Acta Neurochir (Wien) 143:7757822001

    • Search Google Scholar
    • Export Citation
  • 62

    Tsukamoto NMaeda TMiura HJingushi SHosokawa AHarimaya K: Repetitive tensile stress to rat caudal vertebrae inducing cartilage formation in the spinal ligaments: a possible role of mechanical stress in the development of ossification of the spinal ligaments. J Neurosurg Spine 5:234 2422006

    • Search Google Scholar
    • Export Citation
  • 63

    Ugarriza LFCabezudo JMPorras LFRodriguez-Sanchez JA: Cord compression secondary to cervical disc herniation associated with calcification of the ligamentum flavum: case report. Neurosurgery 48:6736762001

    • Search Google Scholar
    • Export Citation
  • 64

    van Oostenbrugge RJHerpers MJde Kruijk JR: Spinal cord compression caused by unusual location and extension of ossified ligamenta flava in a Caucasian male. A case report and literature review. Spine 24:4864881999

    • Search Google Scholar
    • Export Citation
  • 65

    Vera CLCure JKNaso WBGelven PLWorsham FRoof BF: Paraplegia due to ossification of ligamenta flava in X-linked hypophosphatemia. A case report. Spine 22:710 7151997

    • Search Google Scholar
    • Export Citation
  • 66

    Wang KChen X: Thoracic cord compression caused by contiguous multilevel ossification of ligamentum flavum in Chinese patients. Chin J Traumatol 10:2132172007

    • Search Google Scholar
    • Export Citation
  • 67

    Wiseman DBStokes JKToselli RM: Paraparesis in a black man brought on by ossification of the ligamentum flavum: case report and review of the literature. J Spinal Disord Tech 15:5425452002

    • Search Google Scholar
    • Export Citation
  • 68

    Xiong LZeng QYJinkins JR: CT and MRI characteristics of ossification of the ligamenta flava in the thoracic spine. Eur Radiol 11:179818022001

    • Search Google Scholar
    • Export Citation
  • 69

    Yayama TUchida KKobayashi SKokubo YSato RNakajima H: Thoracic ossification of the human ligamentum flavum: histopathological and immunohistochemical findings around the ossified lesion. J Neurosurg Spine 7:1841932007

    • Search Google Scholar
    • Export Citation
  • 70

    Yonenobu KEbara SFujiwara KYamashita KOno KYamamoto T: Thoracic myelopathy secondary to ossification of the spinal ligament. J Neurosurg 66:5115181987

    • Search Google Scholar
    • Export Citation
  • 71

    Zhang YBrown MAPeach CRussell GWordsworth BP: Investigation of the role of ENPP1 and TNAP genes in chondrocalcinosis. Rheumatology (Oxford) 46:5865892007

    • Search Google Scholar
    • Export Citation
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