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LETTER TO EDITOR
Year : 2012  |  Volume : 60  |  Issue : 3  |  Page : 339-341

Spontaneous arthrodesis of atlanto-axial complex in a case of rheumatoid arthritis


Department of Neurosurgery, Nizam's Institute of Medical Sciences, Punjagutta, Hyderabad, Andhra Pradesh, India

Date of Submission05-Feb-2012
Date of Decision20-Feb-2012
Date of Acceptance11-Mar-2012
Date of Web Publication14-Jul-2012

Correspondence Address:
M Vijayasaradhi
Department of Neurosurgery, Nizam's Institute of Medical Sciences, Punjagutta, Hyderabad, Andhra Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0028-3886.98533

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How to cite this article:
Vijayasaradhi M, Kumar M R. Spontaneous arthrodesis of atlanto-axial complex in a case of rheumatoid arthritis. Neurol India 2012;60:339-41

How to cite this URL:
Vijayasaradhi M, Kumar M R. Spontaneous arthrodesis of atlanto-axial complex in a case of rheumatoid arthritis. Neurol India [serial online] 2012 [cited 2021 Sep 17];60:339-41. Available from: https://www.neurologyindia.com/text.asp?2012/60/3/339/98533


Sir,

Atlanto-axial dislocation (AAD) and superior migration of odontoid (cranial settling) and sub-axial subluxation in rheumatoid arthritis (RA) can present with cervical myelopathy. [1] The underlying pathology includes destruction of synovial joints, ligamentous laxity, and pannus formation. [2],[3] Usual treatment of these patients includes fusion of atlanto-axial joint with implants, aiming for a goal of achieving bony fusion of atlanto-axial complex. [4] We present a patient of RA with spontaneous arthrodesis of C1-C2 (atlanto-axial) complex with extensive pan-ligamental and soft tissue calcification around the C1-C2 complex with anterior migration of  Atlas More Details causing high cervical compressive myelopathy.

A 60-year-old female, a known case of RA for the last 10 years, presented with neck pain since 2 years, weakness in the both hands, and paresthesiae in both upper and lower limbs since 2 years. On neurological examination, power in all the four limbs was normal except for mild bilateral hand grip weakness. Deep tendon reflexes were exaggerated and bilateral plantar was extensor. Erythrocyte sedimentation rate was 25 in the first hour and rheumatoid factor was positive. Dynamic X-ray of craniovertebral (CV) junction showed evidence of extensive calcification at C1-C2 complex with atlanto-axial subluxation [Figure 1]. Computed tomography (CT) of CV junction showed extensive pan-ligamental and soft tissue calcification at C1-C2 complex with resultant fusion of atlanto-axial bones with anterior migration of atlas causing spinal canal narrowing at CV junction [Figure 2]. Magnetic resonance imaging (MRI) of CV junction showed cervico-medullary compression by posterior arch of C1 [Figure 3] along with hypointense lesion around C1-C2 complex in T1-weighted and T2-weighted sequences, suggestive of calcification. X-ray of both hands with wrist joints showed evidence of periarticular osteopenia, narrowing carpal and metacarpophalangeal joints, and ulnar deviation of metacarpophalangeal joints, which are the features of chronic RA [Figure 4]. Patient was planned for surgical decompression. As there was spontaneous fusion of atlanto-axial complex, there was no necessity for additional fusion. Hence, only surgical decompression in the form of posterior C1 arch excision was performed. The patient showed remarkable improvement in her symptoms. She was asymptomatic at the end of 1 year and postoperative X-rays at the end of 1 year did not show any instability.
Figure 1: X-ray CV junction lateral view with extension and flexion showing fused atlantoaxial complex anteriorly arrow) with no movement at C1C2 joint

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Figure 2: CT CV junction sagittal reconstruction showing evidence of pan ligamental and soft tissue calcification ( thick arrow) with anterior migration of atlas (thin arrow)

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Figure 3: MRI CV junction n sagittal section showing calcification at C1C2 joint anteriorly (arrow) and compression of cervicomedullay junction by posterior arch of C1

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Figure 4: X-ray both hands demonstrating periarticular osteopenia (arrow)

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Spontaneous arthrodesis of atlanto-axial joint with pan-ligamental and soft tissue calcification causing high cervical compressive myelopathy has not been reported in patients with RA till date. Patients with RA present with various types of anatomical CV junction pathologies, [2],[3] the most common being AAD due to the formation of pannus at predental space. Pannus is a chronic inflammatory granulation tissue composed of mesenchymal and bone narrow derived cells, and it stimulates interleukin-1, platelet-derived growth factor, prostaglandins, and substance P, which ultimately cause cartilage, bone, and ligament destruction. [2],[3] In RA, AAD usually occurs as a consequence of destruction of synovial joint, ligaments, and bone. The major ligaments that maintain the integrity of AA joint are the transverse ligament, alar ligament, apical ligament, tectorial membrane, and other ligaments, which in RA are destroyed by the pannus formation. In our patient, there was spontaneous fusion of C1 and C2 with anterior subluxation of atlas due to pan-ligamental and soft tissue calcification resulting in cervico-medullary compression by posterior arch of C1. Patients with AAD or superior migration of odontoid are managed with cervical immobilization followed by various surgical procedures of fusion depending on the instability of the joint and compression, viz. occipito-cervical fusion, transoral odontoidectomy, transarticular srew fixation, and C1-C2 lateral mass fixation. [4] The pannus resolves with immobilization of CV junction with surgery. But in our patient, there was already spontaneous arthrodesis of C1-C2 complex, as a result of which natural means of immobilization has already been achieved. Thus, only posterior decompression in the form of posterior C1 arch excision was performed. In the extensive literature review, there were only few reported cases of punctate (non-homogeneous) calcifications involving retro-odontoid mass or along the transverse ligament of atlas causing cervico-medullary compression in patients with calcium pyrophosphate dehydrate (pseudo-gout). [5]

We could find only one case regarding chronic AAD with bony bar between C1 anterior arch and body of C2 with os odontoideum following minor trauma [6] There are few other cases showing retro-odontoid mass without calcification causing cervico-medullary compression in patients with Forestier disease. [7] In calcium phyrophosphate dehydrate (CPPD) deposition disease (pseudo-gout) and Forestier disease, the type of calcification was punctate unlike in our patient where extensive calcification led to spontaneous fusion. [8],[9] There are few cases of focal or nodular calcification of alar ligament reported where calcification was punctuate, contrary to our case. [10] Similar punctate calcifications of transverse and alar ligaments were also reported. [11],[12],[13] Presentation similar to meningoencephalitic syndrome was observed in pseudo-gout where there was punctate calcification of transverse ligament, described as crown dense syndrome. [13] Ovoid calcification of the right alar ligament mimicking fracture of the craniovertebral junction can an incidental finding following trauma. [14] Thus, the spontaneous isolated fusion of C1-C2 complex adds up to the vast literature of rheumatoid AAD. The probable role of osteogenic inflammatory markers in the pathogenesis of rheumatoid pannus needs to be confirmed in future research.

 
  References Top

1.Kobayashi Y, Mochida J, Saito I, Matui S. Eiren Toh: Calcification of alar ligament of the cervical spine: Imaging findings and clinical course. Skeletal Radiol 2001;3:295-7.  Back to cited text no. 1
    
2.Patel NP, Wright NM, Choi WW, Mcbridy DQ, Johnson P. Forestier disease associated with a retroodontoid mass causing cervicomedulary compression. J Neurosurg spine 2002;96:190-6.  Back to cited text no. 2
    
3.Zunkeler B, Schlper R, Meneze AH. Periodontoid calcium pyrophosphate dehydrate deposition disease: "Pseudogout" mass lesions of craniocervical junction. J Neurosurg 1996;85:803-9.  Back to cited text no. 3
    
4.Doita M, Shimomura T, Maeno K, Nishida K, Fujioka H, Kurosaka M. Calcium pyrophosphate deposition in the transverse ligament of atlas: An unusual cause of cervical myelopathy. Skeletal Radiol 2007;36:699-702.  Back to cited text no. 4
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5.Robinson HS, Vancouver BC. Rhematoid arthritis- Atlanto-axial subluxation and it's clinical presentation. Can Med Assoc J 1996;94:470-7.  Back to cited text no. 5
    
6.Heselson NG, Marus G. Chronic atlanto axial dislocation with spontaneous bony fusion. Clin Radiol 1998;39:555-7.  Back to cited text no. 6
    
7.Fenoy AF, Menezes AH, Donovan KA, Kralik SF. Calcium pyrophosphate dihydratedeposition in the craniovertebral junction. J Neurosurg Spine 2008;8:22-9.  Back to cited text no. 7
    
8.Narvaez JA, Narvaez J, Serrallonga M, De Lama E, de Albert M, Mast R, et al. Cervical spine involvement in rheumatoid arthritis: Correlation between neurological manifestations and magnetic resonance imaging findings. Rheumatology 2008;47:1814-9.  Back to cited text no. 8
    
9.Conaty JP, Monagan ES. Cervical fusion in rheumatoid arthritis. J Bone Joint Surg Am 1981;63:1218-27.  Back to cited text no. 9
    
10.Zapletel J, Hekster RE, Straver JS, Wilmink JT, Hermans J. Association of transeverse transeverse ligament calcification with anterior atlanto-odontoid osteoarthritis: CT findings. Neuroradiology 1995;37:667-9.  Back to cited text no. 10
    
11.Pellicci PM, Ranawat CS, Tsairis P, Bryan WJ. A prospective study of progression of rheumatoid arthritis of the cervical spine. J Bone Joint Surg Am 1981;63:342-50.  Back to cited text no. 11
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12.Constantin A, Marin F, Bone E, Fedel M, Lagarrigue B, Bouteiller G. Calcification of transeverse ligament of the atlas in chondrocalcinosis: Computed tomography study. Ann Rheum Dis 1996;55:137-9.  Back to cited text no. 12
    
13.Sato Y, Yasuda T, Konno SS, Kuwayama A, Komatasu K. Pseudogout showing meningoencephalitic symptoms: Crowned Dens Syndrome. Intern Med 2004;43:865-8.  Back to cited text no. 13
    
14.Che Mohamed SK, Abd Aziz A. Calcification of Alar ligament mimics fracture of the Craniovertebral junction (CVJ): An Incidental finding from computerized tomography of cervical spine following trauma. Malays J Med Sci 2009;16:69-72.  Back to cited text no. 14
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