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CC BY-NC-ND 4.0 · Arquivos Brasileiros de Neurocirurgia: Brazilian Neurosurgery 2017; 36(02): 117-121
DOI: 10.1055/s-0037-1603554
Case Report | Relato de Caso
Thieme Revinter Publicações Ltda Rio de Janeiro, Brazil

Compressive Myelopathy Due to Ossification of the Ligamentum Flavum: Case Report and Review of the Literature

Mielopatia compressiva devido à ossificação do ligamento flavo: relato de caso e revisão da literatura
Alisson R. Teles
1   Department of Neurology and Neurosurgery, McGill Scoliosis & Spine Group, McGill University, Montreal, QC, Canada
,
Frederico A. Criscuoli de Farias
2   Universidade de Caxias do Sul, Caxias do Sul, RS, Brazil
,
Marcelo R. Roxo
3   Department of Neurosurgery, Universidade de Caxias do Sul, Caxias do Sul, RS, Brazil
,
Albert Vincent Berthier Brasil
4   Hospital São José - Santa Casa de Porto Alegre, Porto Alegre, RS, Brazil
› Author Affiliations
Further Information

Publication History

30 December 2016

16 March 2017

Publication Date:
24 May 2017 (online)

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Abstract

Ossification of the ligamentum flavum (OLF) is a rare condition in which the ligamentum flavum, due to mechanical, biological and genetic factors, becomes ossified. Due to its nature and anatomic location, OLF produces symptoms characteristic of spinal cord compression. The diagnostic confirmation is based primarily on imaging tests such as computed tomography (CT) and magnetic resonance imaging (MRI). Ossification of the ligamentum flavum most often affects Asian populations, rarely occurring in black people. The authors report a case of a 61-year-old black man with progressive paraparesis due to OLF, and review the literature regarding the pathology's prevalence, pathogenesis, clinical features, diagnosis, treatment and prognosis.

Resumo

A ossificação do ligamento flavo (OLF) é uma doença rara na qual o ligamento flavo se torna ossificado devido a fatores mecânicos, biológicos e genéticos. Devido à sua natureza e localização anatômica, a OLF produz sintomas característicos de compressão da medula espinhal. A confirmação diagnóstica baseia-se principalmente em exames de imagem, como tomografia computadorizada (TC) e ressonância magnética (RM). A OLF afeta mais comumente populações asiáticas, raramente sendo observada em pacientes negros. Os autores relatam o caso de um homem negro de 61 anos com paraparesia progressiva decorrente de OLF com uma revisão da literatura a respeito da prevalência, patogênese, aspectos clínicos, diagnóstico, tratamento e prognóstico desta patologia.

Keywords

ossification of the ligamentum flavum - myelopathy - paraparesis - ligamentum flavum - laminectomy - thoracic spine

Palavras-chave

ossificação do ligamento flavo - mielopatia - paraparesia - ligamento flavo - laminectomia - coluna torácica
 

  • References

  • 1 Christiano LD, Assina R, Goldstein IM. Ossification of the ligamentum flavum: a unique report of a Hispanic woman. Neurosurg Focus 2011; 30 (03) E15
    Google Scholar
  • 2 Ben Hamouda K, Jemel H, Haouet S, Khaldi M. Thoracic myelopathy caused by ossification of the ligamentum flavum: a report of 18 cases. J Neurosurg 2003; 99 (2, Suppl) 157-161
    PubMedGoogle Scholar
  • 3 Guo JJ, Luk KD, Karppinen J, Yang H, Cheung KM. Prevalence, distribution, and morphology of ossification of the ligamentum flavum: a population study of one thousand seven hundred thirty-six magnetic resonance imaging scans. Spine 2010; 35 (01) 51-56
    CrossrefPubMedGoogle Scholar
  • 4 Okada G, Hosoi S, Kato K. , et al. Case report 779. Carbonate apatite calcification of ligamentum flavum. Skeletal Radiol 1993; 22 (03) 211-213
    PubMedGoogle Scholar
  • 5 Okada K, Oka S, Tohge K, Ono K, Yonenobu K, Hosoya T. Thoracic myelopathy caused by ossification of the ligamentum flavum. Clinicopathologic study and surgical treatment. Spine 1991; 16 (03) 280-287
    CrossrefPubMedGoogle Scholar
  • 6 Pascal-Moussellard H, Cabre P, Smadja D, Catonné Y. Symptomatic ossification of the ligamentum flavum: a clinical series from the French Antilles. Spine 2005; 30 (14) E400-E405
    CrossrefPubMedGoogle Scholar
  • 7 Li Z, Ren D, Zhao Y. , et al. Clinical characteristics and surgical outcome of thoracic myelopathy caused by ossification of the ligamentum flavum: a retrospective analysis of 85 cases. Spinal Cord 2016; 54 (03) 188-196
    CrossrefPubMedGoogle Scholar
  • 8 Pascal-Mousselard H, Smadja D, Cabre P, Raynaud M, Catonne Y. Ossification of the ligamenta flava with severe myelopathy in a black patient. A case report. Spine 1998; 23 (14) 1607-1608
    CrossrefPubMedGoogle Scholar
  • 9 Mobbs RJ, Dvorak M. Ossification of the ligamentum flavum: diet and genetics. J Clin Neurosci 2007; 14 (07) 703-705
    CrossrefPubMedGoogle Scholar
  • 10 Sakou T, Taketomi E, Matsunaga S, Yamaguchi M, Sonoda S, Yashiki S. Genetic study of ossification of the posterior longitudinal ligament in the cervical spine with human leukocyte antigen haplotype. Spine 1991; 16 (11) 1249-1252
    CrossrefPubMedGoogle Scholar
  • 11 Koga H, Sakou T, Taketomi E. , et al. Genetic mapping of ossification of the posterior longitudinal ligament of the spine. Am J Hum Genet 1998; 62 (06) 1460-1467
    CrossrefPubMedGoogle Scholar
  • 12 Yayama T, Uchida K, Kobayashi S. , et al. Thoracic ossification of the human ligamentum flavum: histopathological and immunohistochemical findings around the ossified lesion. J Neurosurg Spine 2007; 7 (02) 184-193
    CrossrefPubMedGoogle Scholar
  • 13 Parekh HC, Gurusinghe NT, Perera SS, Prabhu SS. Ossification of the ligamentum flavum in a Caucasian: case report. Br J Neurosurg 1993; 7 (06) 687-690
    CrossrefPubMedGoogle Scholar
  • 14 Xu R, Sciubba DM, Gokaslan ZL, Bydon A. Ossification of the ligamentum flavum in a Caucasian man. J Neurosurg Spine 2008; 9 (05) 427-437
    CrossrefPubMedGoogle Scholar
  • 15 van Oostenbrugge RJ, Herpers MJ, de 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 1999; 24 (05) 486-488
    CrossrefPubMedGoogle Scholar
  • 16 Toledo JA, Isseldyk FV, Re M, Garrote M. Ossification of the ligamentum flavum as cause of thoracic cord compression: Case report of a Latin American man and review of the literature. Surg Neurol Int 2013; 4: 119
    CrossrefPubMedGoogle Scholar
  • 17 Cabrera-Aldana EE, De la Garza Ramos R, Zuluaga Gómez A, Rosales-Olivares LM, Reyes-Sánchez A. Multilevel thoracic ossification of the ligamentum flavum in a Hispanic woman with achondroplasia. Spine J 2016; 16 (11) e749-e750
    CrossrefPubMedGoogle Scholar
  • 18 Chana JS, Afshar F. Thoracic spondylosis presenting with spastic paraparesis. Postgrad Med J 1996; 72 (846) 243-244
    CrossrefPubMedGoogle Scholar
  • 19 al-Orainy IA, Kolawole T. Ossification of the ligament flavum. Eur J Radiol 1998; 29 (01) 76-82
    CrossrefPubMedGoogle Scholar
  • 20 Gupta RK, Agarwal P, Kumar S, Surana PK, Lal JH, Misra UK. Compressive myelopathy in fluorosis: MRI. Neuroradiology 1996; 38 (04) 338-342
    CrossrefPubMedGoogle Scholar
  • 21 Sushil P, Anant K. Ossified-calcified ligamentum flavum causing dorsal cord compression with computed tomography-magnetic resonance imaging features. Surg Neurol 1994; 41 (06) 441-442
    CrossrefPubMedGoogle Scholar
  • 22 Jaffan I, Abu-Serieh B, Duprez T, Cosnard G, Raftopoulos C. Unusual CT/MR features of putative ligamentum flavum ossification in a North African woman. Br J Radiol 2006; 79 (944) e67-e70
    CrossrefPubMedGoogle Scholar
  • 23 Kruse JJ, Awasthi D, Harris M, Waguespack A. Ossification of the ligamentum flavum as a cause of myelopathy in North America: report of three cases. J Spinal Disord 2000; 13 (01) 22-25
    CrossrefPubMedGoogle Scholar
  • 24 Gondim J, Ramos Júnior F. [Thoracic spinal cord compression at two levels due to ligamentum flavum calcification. Case report]. Arq Neuropsiquiatr 1998; 56 (02) 312-316
    CrossrefPubMedGoogle Scholar
  • 25 Uchida K, Yayama T, Cai HX. , et al. Ossification process involving the human thoracic ligamentum flavum: role of transcription factors. Arthritis Res Ther 2011; 13 (05) R144
    CrossrefPubMedGoogle Scholar
  • 26 Yin X, Chen Z, Guo Z, Liu X, Yu H. Tissue transglutaminase expression and activity in human ligamentum flavum cells derived from thoracic ossification of ligamentum flavum. Spine 2010; 35 (20) E1018-E1024
    CrossrefPubMedGoogle Scholar
  • 27 Moon SH, Park SR, Kim H. , et al. Biologic modification of ligamentum flavum cells by marker gene transfer and recombinant human bone morphogenetic protein-2. Spine 2004; 29 (09) 960-965
    CrossrefPubMedGoogle Scholar
  • 28 Hayashi K, Ishidou Y, Yonemori K. , et al. Expression and localization of bone morphogenetic proteins (BMPs) and BMP receptors in ossification of the ligamentum flavum. Bone 1997; 21 (01) 23-30
    CrossrefPubMedGoogle Scholar
  • 29 Kong Q, Ma X, Li F. , et al. COL6A1 polymorphisms associated with ossification of the ligamentum flavum and ossification of the posterior longitudinal ligament. Spine 2007; 32 (25) 2834-2838
    CrossrefPubMedGoogle Scholar
  • 30 Tanaka T, Ikari K, Furushima K. , et al. 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 2003; 73 (04) 812-822
    CrossrefPubMedGoogle Scholar
  • 31 Tsukamoto N, Maeda T, Miura H. , et al. 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 2006; 5 (03) 234-242
    CrossrefPubMedGoogle Scholar
  • 32 Li F, Chen Q, Xu K. Surgical treatment of 40 patients with thoracic ossification of the ligamentum flavum. J Neurosurg Spine 2006; 4 (03) 191-197
    CrossrefPubMedGoogle Scholar
  • 33 Yoshida M, Shima K, Taniguchi Y, Tamaki T, Tanaka T. Hypertrophied ligamentum flavum in lumbar spinal canal stenosis. Pathogenesis and morphologic and immunohistochemical observation. Spine 1992; 17 (11) 1353-1360
    CrossrefPubMedGoogle Scholar
  • 34 Ono K, Yonenobu K, Miyamoto S, Okada K. Pathology of ossification of the posterior longitudinal ligament and ligamentum flavum. Clin Orthop Relat Res 1999; (359) 18-26
    PubMedGoogle Scholar
  • 35 Tomita K, Kawahara N, Baba H, Kikuchi Y, Nishimura H. Circumspinal decompression for thoracic myelopathy due to combined ossification of the posterior longitudinal ligament and ligamentum flavum. Spine 1990; 15 (11) 1114-1120
    CrossrefPubMedGoogle Scholar
  • 36 Hukuda S, Mochizuki T, Ogata M, Shichikawa K. The pattern of spinal and extraspinal hyperostosis in patients with ossification of the posterior longitudinal ligament and the ligamentum flavum causing myelopathy. Skeletal Radiol 1983; 10 (02) 79-85
    CrossrefPubMedGoogle Scholar
  • 37 Ehara S, Shimamura T, Nakamura R, Yamazaki K. Paravertebral ligamentous ossification: DISH, OPLL and OLF. Eur J Radiol 1998; 27 (03) 196-205
    CrossrefPubMedGoogle Scholar
  • 38 Kojima T, Oonishi I, Kurokawa T. Ossification of the ligamentum flavum in the thoracolumbar spine of young adults report of two cases. Int Orthop 1992; 16 (01) 75-79
    PubMedGoogle Scholar
  • 39 Ishida Y, Kawai S. Effects of bone-seeking hormones on DNA synthesis, cyclic AMP level, and alkaline phosphatase activity in cultured cells from human posterior longitudinal ligament of the spine. J Bone Miner Res 1993; 8 (11) 1291-1300
    PubMedGoogle Scholar
  • 40 Terakado A, Tagawa M, Goto S, Yamazaki M, Moriya H, Fujimura S. Elevation of alkaline phosphatase activity induced by parathyroid hormone in osteoblast-like cells from the spinal hyperostotic mouse TWY (twy/twy). Calcif Tissue Int 1995; 56 (02) 135-139
    CrossrefPubMedGoogle Scholar
  • 41 Wang Z, Li XD, Li MQ, Wang QP. Changes in basic metabolic elements associated with the degeneration and ossification of ligamenta flava. J Spinal Cord Med 2008; 31 (03) 279-284
    CrossrefPubMedGoogle Scholar
  • 42 Li H, Jiang LS, Dai LY. Hormones and growth factors in the pathogenesis of spinal ligament ossification. Eur Spine J 2007; 16 (08) 1075-1084
    CrossrefPubMedGoogle Scholar
  • 43 Matsui H, Katoh Y, Tsuji H. Untreated hypophosphatemic vitamin D-resistant rickets with symptomatic ossification of the ligamentum flavum. J Spinal Disord 1991; 4 (01) 110-113
    PubMedGoogle Scholar
  • 44 Vera CL, Cure JK, Naso WB. , et al. Paraplegia due to ossification of ligamenta flava in X-linked hypophosphatemia. A case report. Spine 1997; 22 (06) 710-715
    CrossrefPubMedGoogle Scholar
  • 45 Epstein NE. Ossification of the yellow ligament and spondylosis and/or ossification of the posterior longitudinal ligament of the thoracic and lumbar spine. J Spinal Disord 1999; 12 (03) 250-256
    PubMedGoogle Scholar
  • 46 Kim TJ, Kim TH, Jun JB, Joo KB, Uhm WS. Prevalence of ossification of posterior longitudinal ligament in patients with ankylosing spondylitis. J Rheumatol 2007; 34 (12) 2460-2462
    PubMedGoogle Scholar
  • 47 Sato T, Kokubun S, Tanaka Y, Ishii Y. Thoracic myelopathy in the Japanese: epidemiological and clinical observations on the cases in Miyagi Prefecture. Tohoku J Exp Med 1998; 184 (01) 1-11
    CrossrefPubMedGoogle Scholar
  • 48 Aizawa T, Sato T, Sasaki H, Kusakabe T, Morozumi N, Kokubun S. Thoracic myelopathy caused by ossification of the ligamentum flavum: clinical features and surgical results in the Japanese population. J Neurosurg Spine 2006; 5 (06) 514-519
    CrossrefPubMedGoogle Scholar
  • 49 Aizawa T, Sato T, Sasaki H. , et al. Results of surgical treatment for thoracic myelopathy: minimum 2-year follow-up study in 132 patients. J Neurosurg Spine 2007; 7 (01) 13-20
    CrossrefPubMedGoogle Scholar
  • 50 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 1996; 9 (05) 446-447 , discussion 447–450
    PubMedGoogle Scholar
  • 51 Rivierez M, Vally P. [Ossification of ligamentum flavum unmasked by acute paraplegia]. Neurochirurgie 2001; 47 (06) 572-575
    PubMedGoogle Scholar
  • 52 Sagar H, Fernandez-Madrid F, Kupsky W. Rare cause of thoracic myelopathy: ossified ligamentum flavum. J Clin Rheumatol 2010; 16 (07) 326-329
    CrossrefPubMedGoogle Scholar
  • 53 Suojanen JN, Lipson SJ. Spinal cord compression secondary to ossified ligamentum flavum. J Spinal Disord 1989; 2 (04) 238-240
    PubMedGoogle Scholar
  • 54 Wiseman DB, Stokes JK, Toselli 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 2002; 15 (06) 542-545
    CrossrefPubMedGoogle Scholar
  • 55 Shiokawa K, Hanakita J, Suwa H, Saiki M, Oda M, Kajiwara M. Clinical analysis and prognostic study of ossified ligamentum flavum of the thoracic spine. J Neurosurg 2001; 94 (2, Suppl) 221-226
    PubMedGoogle Scholar
  • 56 Shiraishi T, Crock HV, Lewis P. Thoracic myelopathy due to isolated ossification of the ligamentum flavum. J Bone Joint Surg Br 1995; 77 (01) 131-133
    PubMedGoogle Scholar
  • 57 Omojola MF, Cardoso ER, Fox AJ, Drake CG, Durward QJ. Thoracic myelopathy secondary to ossified ligamentum flavum. J Neurosurg 1982; 56 (03) 448-450
    CrossrefPubMedGoogle Scholar
  • 58 Sugimura H, Kakitsubata Y, Suzuki Y. , et al. MRI of ossification of ligamentum flavum. J Comput Assist Tomogr 1992; 16 (01) 73-76
    CrossrefPubMedGoogle Scholar
  • 59 Muthukumar N. Dural ossification in ossification of the ligamentum flavum: a preliminary report. Spine 2009; 34 (24) 2654-2661
    CrossrefPubMedGoogle Scholar
  • 60 Miyasaka K, Kaneda K, Sato S. , et al. Myelopathy due to ossification or calcification of the ligamentum flavum: radiologic and histologic evaluations. AJNR Am J Neuroradiol 1983; 4 (03) 629-632
    PubMedGoogle Scholar
  • 61 Muthukumar N, Karuppaswamy U, Sankarasubbu B. Calcium pyrophosphate dihydrate deposition disease causing thoracic cord compression: case report. Neurosurgery 2000; 46 (01) 222-225
    CrossrefPubMedGoogle Scholar
  • 62 Sanghvi AV, Chhabra HS, Mascarenhas AA, Mittal VK, Sangondimath GM. Thoracic myelopathy due to ossification of ligamentum flavum: a retrospective analysis of predictors of surgical outcome and factors affecting preoperative neurological status. Eur Spine J 2011; 20 (02) 205-215
    CrossrefPubMedGoogle Scholar
  • 63 Kalevski SK, Peev NA, Haritonov DG. Incidental Dural Tears in lumbar decompressive surgery: Incidence, causes, treatment, results. Asian J Neurosurg 2010; 5 (01) 54-59
    PubMedGoogle Scholar
  • 64 Inamasu J, Guiot BH. A review of factors predictive of surgical outcome for ossification of the ligamentum flavum of the thoracic spine. J Neurosurg Spine 2006; 5 (02) 133-139
    CrossrefPubMedGoogle Scholar
  • 65 Kuh SU, Kim YS, Cho YE. , et al. Contributing factors affecting the prognosis surgical outcome for thoracic OLF. Eur Spine J 2006; 15 (04) 485-491
    CrossrefPubMedGoogle Scholar