Abstract

Background:Ossification of the ligamentum flavum (OLF) occurs mostly in adult males, typically in the thoracolumbar spine where it may contribute to neurological deficits. Here we reviewed 68 cases of cervical OLF resulting in progressive quadriparesis.

Methods:The literature on cervical OLF was reviewed between 1962 and 2018 along with the case of an 81-year-old male with progressive quadriparesis attributed to cervical OLF.

Results:Most patients with cervical OLF are Asian, with Caucasians constituting the second most frequently impacted population.

Conclusions:Cervical OLF is typically reported in the Asian, followed by the Caucasian population, and is most often found in the thoracolumbar spine. Here we presented an 81-year-old male with cervical OLF contributing to quadriparesis.

Keywords: Cervical myelopathy, cervical spine, ossification of ligamentum flavum, ossification of posterior longitudinal ligament, ossification of yellow ligament, review article

INTRODUCTION

Ossification of the ligamentum flavum (OLF) typically occurs in adults involving the thoracolumbar spine. It is less frequently encountered in the cervical region (<1%).[ 27 44 ] In 1962, Koizumi described cervical OLF at autopsy in a 55-year-old male who had developed progressive quadriparesis.[ 20 ] Since then only 68 more cases of cervical OLF have been published, typically involving the East Asian populations presenting varying degrees of myelopathy.[ 1 4 6 7 9 14 20 21 27 37 40 44 48 ] Here, an 81-year-old Caucasian male with unilateral large nodular OLF at the C4-C5 level and a smaller lesion at C5-C6 presented with quadriparesis adequately treated with laminectomy and OLF resection.

CASE REPORT

An 81-year-old bed-ridden male presented a spastic quadriparesis. The T1-weighted sagittal magnetic resonance image (MRI) showed a large heterogeneous mass resulting in dorsolateral cord compression at C4-C5, and a smaller lesion at the C5-C6 levels, consistent with OLF [ Figure 1 ]. On the T2-weighted MRI, the mass was isointense with a hypointense peripheral rim [ Figure 1 ]. The CT scan confirmed ossification of both OYL lesions [ Figure 2 ].

Figure 1

Cervical MRI: (a) T1-weighted sagittal image showing a heterointense epidural mass at the posterior aspect of the spinal cord at the C4-C5 level, a smaller one is visualized below this level. (b) T2-weighted sagittal image demonstrating a hypointense mass with an isointensity at the center of the mass. (c) It also reveals a significant reduction in the cervical canal diameter

Figure 2

Cervical computed tomography (CT) scan. (a) Sagittal reconstructed CT showing a calcified mass between the spinal processes of C4 and C5 with canal compromise present at the site of the ligamentum flavum. (b) Axial CT demonstrated an oval-shaped calcified mass

The patient underwent a C4-C6 laminectomy for resection of large dorsolateral OLF masses (C45, C56) [ Figure 3 ]. Neurolysis and durolysis was accomplished without a cerebrospinal fluid fistula. Postoperatively, the patient improved. The histological examination confirmed OLF [ Figure 4 ]. The MRI taken 2 weeks later confirmed adequate canal decompression [ Figure 5 ]. Three months later, the patient was able to ambulate with a walker.

Figure 3

The surgical specimen shows the calcified mass that is almost removed en-block

Figure 4

Histological examination of the surgical specimen (a) and (b) shows areas of endochondral ossification or new bone formation. At the edge, elastic bundles compatible with ligaments are noted

Figure 5

Post-op sagittal cervical MRI, (a) and (b) laminectomy and decompression of the cord is shown both in T1- and T2-weighted images

DISCUSSION

About 80% of the ligamentum flavum is composed of elastic fibers, and 20% with collagen.[ 1 14 27 35 36 44 48 ] Ossification of the ligamentum flavum (OLF) involves heterotopic ossification of this ligament.

Incidence

There are 69 previously reported cases of cervical OLF Table 1 ,[ 1 4 6 7 14 27 37 44 48 ] which most typically occur in the East Asian population, followed by Caucasians [ Table 2 ]. Patients with cervical OLF are between 27 and 84 years of age (average 62), and are mostly males [Tables 3 and 4 ].

Table 1

Review of all patients with cervical OLF

Table 2

The ethnicity of the patients with cervical OLF

Table 3

The gender of the patients with cervical OLF

Table 4

(a) The range of the age and the mean of the patients with cervical OLF

Clinical picture

Patients with cervical OLF often present late in the clinical course with cord compression/spondylosis resulting in chronic myeloradiculopathy and an evolving quadriparesis.[ 1 14 27 44 ] Cervical OLF and ossification of the posterior longitudinal ligament (OPLL) rarely appear together (e.g. 9/55 cases reported).

Imaging

Lateral cervical plain radiographs may demonstrate OLF located between the bases of two spinal processes.[ 23 ] However, the location and severity of OLF are better demonstrated on MRI and CT studies. On T2-weighted sagittal MRI, OLF may be isointense, hypointense, or both,[ 27 35 36 44 ] and there may be an accompanying intramedullary hyperintense signal [ Table 5 ]. On CT scans examination, OLF lesions are usually seen as ossified masses extending from the facet joint to the base of the spinal processes, either unilaterally or bilaterally.

Table 5

The frequency of OLF in different cervical levels

Treatment

A decompressive laminectomy with excision of OLF may be warranted in symptomatic patients. The OLF dissection from the dura should be accomplished under the operating microscope to lyse adhesions. Li et al. described decompressive en-block laminectomy for removal of OLF.[ 24 ] Epstein, in 1999, proposed posterior stabilization for multilevel cervical OLF following extensive laminectomy.[ 9 ] Dural tears should largely be avoided routinely using an operating microscope. If they occur, closure with 7-0 Gortex sutures and microdural stapes is warranted.

Outcome

Typically following a cervical laminectomy with resection of OLF, patients should significantly recover from their preoperative myeloradicular syndrome.

Summary

Cervical OLF rarely causes cervical myeloradiculopathy. Following both MR and CT studies to adequately document the location/extent of disease, laminectomy alone often suffices to decompress the cord.

Declaration of patient consent

Written informed consent was obtained from the patient for publication and corresponding images.

Contribution

The steps of this article from design to writing were made by Abolfazl Rahimizadeh, Naser Asgari, Housain Soufiani, and Shaghayegh Rahimizadeh, retrospectively.

Ethical approval

Approved.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

References

1. Chachan S, Kasat NS, Keng PTL. Cervical myelopathy secondary to combined ossification of ligamentum flavum and posterior longitudinal ligament—A case report. Int J Spine Surg. 2018. 12: 121-5

2. Chachan S, Kasat NS, Thng PK. Cervical myelopathy secondary to combined ossification of ligamentum flavum and posterior longitudinal ligament. J Spinal Surg. 2016. 3: 157-9

3. Chen P-Y, Lin C-Y, Tzaan W-C, Chen H-C. Brown-Sequard syndrome caused by ossification of the ligamentum flavum. J Clin Neurosci. 2007. 14: 887-90

4. Chitoku S, Nishiura I, Fukuda M, Amano S. Ossification of the yellow ligament combined with ossification of the posterior longitudinal ligament at the cervicothoracic junction. World Neurosurg. 2017. 103: 952.e19-952.e22

5. Chou Y-C, Lee C-C, Yen P-S, Lin J-F, Su C-F, Lin S-Z. Cough induced by ossification of the ligamentum flavum in the high cervical spine: Case report. J Neurosurg Spine. 2004. 100: 364-6

6. Christiano LD, Assina R, Goldstein IM. Ossification of the ligamentum flavum: A unique report of a Hispanic woman. Neurosurg Focus. 2011. 30: E15-

7. Dewachter L, Govaere F, Crevits I, De Man R. Images in clinical radiology. JBR–BTR. 2011. 94: 219-

8. Doi S, Kageyama Y, Nagahusa T, Okada M. Ossification of the ligamentum flavum of the cervical spine. Case report. Seikei Geka. 2000. 51: 1659-62

9. Epstein NE. Laminectomy with posterior wiring and fusion for cervical ossification of the posterior longitudinal ligament, spondylosis, ossification of the yellow ligament, stenosis, and instability: A study of 5 patients. J Spinal Disord. 1999. 12: 461-6

10. Fotakopoulos G, Alexiou G, Mihos E, V oulgaris S. Ossification of the ligamentum flavum in cervical and thoracic spine. Report of three cases. Acta Neurol Belg. 2010. 110 N2: 186-9

11. Fujiwara M, Bitoh S, Hasegawa H, Nakata M, Hata H. A case of nodular calcification of the ligamentum flavum with ossification of the posterior longitudinal ligament in the cervical spine. No Shinkei Geka. 1982. 10: 769-74

12. Fukao S, Hanakita J, Kitahama Y, Minami M, Tamiya A. Ossification of the ligamentum flavum of the upper cervical spine a case report. Spinal Surg. 2006. 20: 49-54

13. Hoshida T. Three cases of ossified and calcified nodules in the cervical ligamentum flavum. Osaka Keisatsu Byoin Igaku Zasshi. 1989. 13: 71-9

14. Inoue H, Seichi A, Kimura A, Endo T, Hoshino Y. Multiple-level ossification of the ligamentum flavum in the cervical spine combined with calcification of the cervical ligamentum flavum and posterior atlanto-axial membrane. Eur Spine J. 2013. 22: 416-20

15. Kamakura K, Furukawa T, Takasu T, Mannen T, Toyokura Y. Cervical myelopathy due to ossification of ligamenta flava: Report of a case (author's transl). Rinsho Shinkeigaku. 1978. 18: 96-102

16. Kaneda K. Study of an operative case with a myelopathy due to the ossification of the thoracic spinal canal. Rinsho Seikeigeka. 1977. 12: 387-94

17. Kim K, Isu T, Nomura R, Kobayashi S, Teramoto A. Cervical ligamentum flavum ossification. Neurol Med Chir (Tokyo). 2008. 48: 183-7

18. Kirita Y, Miyazaki K, Lin D, Nosaka K, Shima M, Yamamura H. Surgical treatment for the ossification of the ligamentum flavum. Chubu Nippon Seikeigeka Saigaigeka Gakkai Zasshi. 1973. 16: 814-7

19. Kobayashi S, Okada K, Onoda K, Horikoshi S. Ossification of the cervical ligamentum flavum. Surg Neurol. 1991. 35: 234-8

20. Koizumi M. Three cases of spinal cord paralysis proved by ligamenta flava ossification. Rinsho-Geka (Clin Surg). 1962. 17: 1181-8

21. Kotani Y, Takahata M, Abumi K, Ito M, Sudo H, Minami A. Cervical myelopathy resulting from combined ossification of the ligamentum flavum and posterior longitudinal ligament: Report of two cases and literature review. Spine J. 2013. 13: e1-6

22. Kruse JJ, Awasthi D, Harris M, Waguespack A. Case report. Ossification of the ligamentum flavum as a cause of myelopathy in North America: Report of three cases. Clin Spine Surg. 2000. 13: 22-5

23. Kubota M, Baba I, Sumida T. Myelopathy due to ossification of the ligamentum flavum of the cervical spine a report of two cases. Spine. 1981. 6: 553-9

24. Li K-K, Chung O-M, Chang Y-P, So Y-C. Myelopathy caused by ossification of ligamentum flavum. Spine. 2002. 27: E308-12

25. Mak K-h, Mak K-l, Gwi-Mak E. Ossification of the ligamentum flavum in the cervicothoracic junction: Case report on ossification found on both sides of the lamina. Spine. 2002. 27: E11-4

26. Minami Y, Maeyama I, Kawakami T, Sakinaga Y. A case of cervical myelopathy due to ossification of the ligamentum flavum. Orthop Traumatol. 1985. 34: 335-7

27. Miyazawa N, Akiyama I, Di Lorenzo N, Mouchaty H. Ossification of the ligamentum flavum of the cervical spine/comment. J Neurosurg Sci. 2007. 51: 139-44

28. Mizuno J, Nakagawa H. Unilateral ossification of the ligamentum flavum in the cervical spine with atypical radiological appearance. J Clin Neurosci. 2002. p. 9462-4

29. Mohindra S, Gupta R, Chhabra R, Gupta SK. Compressive myelopathy due to ossified yellow ligament among South Asians: Analysis of surgical outcome. Acta Neurochir (Wien). 2011. 153: 581-7

30. Nadkarni TD, Menon RK, Desai KI, Goel A. Ossified ligamentum flavum of the atlantoaxial region. J Clin Neurosci. 2005. 12: 486-9

31. Nagashima C, Takahama M, Nakayama Y, Asano T. Giant occipital hemangioendothelioma with thrombocytopenia, anemia, and hypofibrinogenemia: Case report. J Neurosurg. 1975. 43: 74-9

32. Nishiura I. Cervical radiculopathy due to ossification of yellow ligament: A case report. Sekitsui Sekizui J. 1992. 5: 383-5

33. Ohnishi Y-I, Iwatsuki K, Yoshimura K, Ishihara M, Yoshimine T. Incarcerated herniation of the cervical spinal cord after laminectomy for an ossification of the yellow ligament. Eur Spine J. 2010. 19: 140-3

34. Ota S. A case of cervical myelopathy due to ossification of the ligamentum-flavum. Chubu Nippon Seikeigeka Saigaigeka Gakkai Zasshi. 1982. 25: 966-8

35. Rahimizadeh A. Isolated proximal thoracic ossified ligamentum flavum causing paraparesis in a caucasian: A case report and review of literature. World Spinal Column J. 2012. 3: 38-43

36. Rahimizadeh A, Soufiani H, Amirzadeh M, Rahimizadeh S. Ossification of the ligamentum flavum of the lumbar spine in caucasians: Case series. J Spine Neurosurg. 2017. 6: 5-

37. Sampanis N, Gavriilaki E, Paschou E, Kalaitzoglou A, Vasileiou S. Ossification of the cervical ligamentum flavum and osseous brown tumor: Late manifestations of primary hyperparathyroidism misdiagnosed in a case of parathyroid carcinoma. Clin Cases Miner Bone Metab. 2016. 13: 54-6

38. Sato K, Hayashi M, Kubota T, Kawano H, Handa Y, Kabuto M. Symptomatic calcification and ossification of the cervical ligamentum flavum: Clinical, radiological and pathological features. Br J Neurosur. 1989. 3: 597-602

39. Seichi A, Katoh H, Tanaka H, Tuzuki N. Radiculomyelopathy due to ossification of the ligamentum flavum of the cervical spine; a case report. Seikeigeka. 1988. 39: 929-32

40. Shepard NA, Shenoy K, Cho W, Sharan AD. Extensive ossification of the ligamentum flavum treated with triple stage decompression: A case report. Spine J. 2015. 15: e9-14

41. Shimada Y. Ossification of the ligamentum flavum of the cervical spine. Case report. Seikeigeka To Saigaigeka. 1990. 33: 399-403

42. Singhal U, Jain M, Jaiswal AK, Behari S. Unilateral ossified ligamentum flavum in the high cervical spine causing myelopathy. Indian J Orthop. 2009. 43: 305-8

43. Song JY, Park JH, Roh SW. Ossified ligamentum flavum causing cervical myelopathy. Korean J Spine. 2012. 9: 24-7

44. Sonntag VK. Ossification of the ligamentum flavum (OLF): An increasing cause of cervical myelopathy. World Neurosurg. 2011. 3: 445-6

45. Sugimura H, Kakitsubata Y, Suzuki Y, Kakitsubata S, Tamura S, Uwada O. MRI of ossification of ligamentum flavum. J Comput Assist Tomogr. 1992. 16: 73-6

46. Takayama S, Kuribayashi K, Miyamoto Y, Nakasu Y, Handa J. Ossification and calcification of the cervical ligamentum flavum--case reports. No to shinkei. 1993. 45: 859-63

47. Tanaka H, Tsuzuki N, Seichi A, Hotta Y, Iizuka T. Anatomical study of ossification and calcification of the yellow ligament of spine with reference to the distribution of the yellow ligament. Rinsho Seikei Geka. 1988. 23: 411-7

48. Yang J, Ni B, Xie N, Guo Q, Wang L. Surgical treatments of myelopathy caused by cervical ligamentum flavum ossification. World Neurosurg. 2011. 75: 546-50

49. Yang JC, Lin CP, Chan JY, Liu YK, Huang JS. Surgical treatment of multilevel cervical radiculomyelopathy caused by the concomitant ossification of the ligamentum flavum and the posterior longitudinal ligament. Surg Pract. 2005. 9: 111-4