View/Download PDF
CROSSMARK LOGO Buy Reprints
PDF

Translate this page into:
Notice: Please configure GTranslate from WP-Admin -> Settings -> GTranslate to see it in action.

Case Report
2021
:12;
403
doi:
10.25259/SNI_588_2021

Periodontoid pseudotumoral lesions

Department of Surgery, Santa Casa de Sao Paulo School of Medical Sciences, São Paulo, Brazil.
Corresponding author: Guilherme Brasileiro de Aguiar, Department of Surgery, Santa Casa de Sao Paulo School of Medical Sciences, São Paulo, Brazil. guilhermebraguiar@yahoo.com.br
Licence

This is an open-access article distributed under the terms of the Creative Commons Attribution-Non Commercial-Share Alike 4.0 License, which allows others to remix, tweak, and build upon the work non-commercially, as long as the author is credited and the new creations are licensed under the identical terms.

How to cite this article: de Souza Batista AV, de Aguiar GB, Bennett P, Veiga JC. Periodontoid pseudotumoral lesions. Surg Neurol Int 2021;12:403.

Abstract

Background:

Periodontoid pseudotumoral lesions (PPL) are an uncommon cause of cervical pain and myelopathy. In addition, they may be associated with atlantoaxial instability (AAI).

Case Description:

Two patients over 60 years of age presented with neck pain alone. Their MR scans showed expansive lesions involving the odontoid process. One patient with AAI required an occipitocervical arthrodesis, while the other patient without instability was managed with an external orthosis (Philadelphia collar). Both of them experienced full resolution of pain and remained neurologically intact an average 36 months later (range 24–48).

Conclusion:

Here, we discussed the clinical, MR, and non-surgical (without AAI) versus surgical management (with AAI) for different types of PPL.

Keywords

Atlanto-axial joint
Magnetic resonance imaging
Odontoid process
Skull base
Spinal cord diseases

INTRODUCTION

In this study, we present the clinical, MR appearance, non-surgical, and surgical management for two cases of periodontoid pseudotumoral lesions (PPL), and selectively reviewed the appropriate literature.

CASE DESCRIPTION

Case 1: Non-surgical management of PPL

A 72-year-old male presented with neck pain alone, no neurological deficit, and a cervical MR that showed an expansive lesion posterior to the odontoid process [Figure 1]. As dynamic radiographs did not demonstrate atlantoaxial instability (AAI), conservative treatment (i.e. Philadelphia-type cervical collar) was utilized. Four years later, the patient’s neck pain had improved, and he remained neurologically intact [Table 1].

Figure 1:: Sagittal (a) and axial (b) sections of magnetic resonance images of the craniocervical junction, presenting an expansive lesion in the posterior aspect of the odontoid process, with. Low signal at both T1 (a) and T2 (b). There is no evidence of spinal cord compression.
Table 1:: Summarization of the presented cases.

Case 2: Surgical management of PPL

A 61-year-old female developed the rapid onset cervical pain exacerbated with cervical rotation but no neurological deficit.

Here, the cervical MRI revealed an intense inflammatory reaction involving the atlantoaxial joint (i.e. hyperintense T2 signal in anomalous tissue around the axis dens, with widening of the atlantodental interval) [Figure 2]. Further, the C-reactive protein and erythrocyte sedimentation rates were increased. A diagnosis of AA joint synovitis led to the administration of 60 mg of prednisone daily for 3 weeks. When subsequent dynamic cervical X-rays showed AAI, she underwent an occipitocervical arthrodesis with biopsy of the odontoid process (which later showed crystal deposition). Within a month, her cervical symptoms improved, and at 2 postoperative years, she had neither pain or any neurological deficits [Table 1].

Figure 2:: Sagittal sections from preoperative magnetic resonance imaging (a-c) indicating tissue formations that suggest an expansive process at the atlantodental joint, with low signal at T1 (a), intense enhancement after contrast injection (b) and heterogeneous signal at T2 (c). In (d), it is presented an axial section of a computed tomography image of the cervical spine obtained at the immediate postoperative period of occipitocervical arthrodesis; the arrow points to the calcification halo around the axon odontoid process. In (e) and (f), it is presented the final result of the occipitocervical arthrodesis.

DISCUSSION

Retro-odontoid pseudotumor (ROP)

ROP tumor is most often attributed to rheumatoid arthritis,[11] cervical spondyloarthropathy,[5] primary amyloidosis, and hemodialysis.[2] Tanaka et al.[13] classified these lesions into three types, those with: (1) atlantoaxial subluxation, (2) subaxial spondylosis, or (3) secondary to herniation of an intervertebral disc.

The first patient we presented had a Tanaka’s type 2 ROP lesion without AAI.

Crowned dens syndrome (CDS)

In CDS, calcium pyrophosphate crystal deposition leads to expansion of the soft tissues and local inflammatory reaction (i.e. similar to pseudogout). CT is the most useful method for diagnosis.[4,10] Clinical treatment, which includes nonsteroidal anti-inflammatories (in some cases, corticosteroids with or without colchicine),[10] is usually sufficient and is associated with a good prognosis.[12]

MR imaging of PPL and documentation of three types of lesions

Yonezawa et al. classified retro-odontoid lesions in three types based on MR signals: type I (inflammatory, typical of pannus) – hyposignal in T1 and hypersignal in T2; type II (fibrous) - hyposignal in T1 and T2; and type III (mixed) – heterogeneous signal in T2.[14]

Non-surgical and surgical management of PPL

The ideal approach to cases of PPL may be non-surgical or surgical [Table 2].

Table 2:: Review of PPL casuistic in the literature.

Since those without instability do not typically warrant fusions,[3] some authors advocate simple decompression, but others indicate fixation if subaxial spondylosis is present.[13] In this sense, Kobayashi et al found no statistical differences between the fusion and non-fusion groups regarding neurological outcome, although lesion regression was significantly greater in those undergoing fixation.[7]

For those with AAI, fusions are usually warranted. Atlantoaxial and occipitocervical fixations are associated with good rates of recovery and reduction or disappearance of PPL.[1,9]

Here, case 1 of ROP was successfully managed with cervical collar immobilization for an average of 18 months.[6,8]

CONCLUSION

The best treatment for PPL requires MR evaluation and X-ray assessment of AAI; the latter may warrant fusion.

Declaration of patient consent

Patient’s consent not required as patients identity is not disclosed or compromised.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

REFERENCES

  1. , , , , , . Disappearance of degenerative, non-inflammatory, retro-odontoid pseudotumor following posterior C1-C2 fixation: Case series and review of the literature. Eur Spine J. 2013;22:S879-88.
    [Google Scholar]
  2. , , , . Retro-odontoid pseudotumor: A poorly recognized alteration of the craniocervical junction. Rev Assoc Med Bras. 2020;66:507-11.
    [Google Scholar]
  3. , , , , , , . Radiographic analysis of the cervical spine in patients with retro-odontoid pseudotumors. Spine (Phila Pa 1976). 2009;34:E110-4.
    [Google Scholar]
  4. , , , . Crowned dens syndrome. J Bone Joint Surg Am. 2007;89:2732-6.
    [Google Scholar]
  5. , , . Retro-odontoid pseudotumor in diffuse idiopathic skeletal hyperostosis. Spine (Phila Pa 1976). 2002;27:E266-70.
    [Google Scholar]
  6. , , . Regression of degenerative retro-odontoid pseudotumor treated in a collar. Can J Neurol Sci. 2018;45:599-600.
    [Google Scholar]
  7. , , , , . Postoperative regression of retro-odontoid pseudotumors treated with and without fusion. Eur Spine J. 2018;27:3105-12.
    [Google Scholar]
  8. , , . Regression of retro-odontoid pseudotumor using external orthosis without atlantoaxial fusion: A case report. JBJS Case Connect. 2019;9:e0329.
    [Google Scholar]
  9. , , , , , , . Postoperative regression of retro-odontoid pseudotumor after atlantoaxial posterior fixation: 11 years of experience in patients with atlantoaxial instability. Spine (Phila Pa 1976). 2017;42:1763-71.
    [Google Scholar]
  10. , . Retro-odontoid pseudotumor without radiologic atlantoaxial instability: A systematic review. World Neurosurg. 2019;121:100-10.
    [Google Scholar]
  11. , , , , , , . The effects of clinical factors and retro-odontoid soft tissue thickness on atlantoaxial instability in patients with rheumatoid arthritis. World Neurosurg. 2017;103:364-70.
    [Google Scholar]
  12. , , , , , . Crowned dens syndrome: Reports of six cases and review of the literature. Acute Med Surg. 2016;3:155-8.
    [Google Scholar]
  13. , , , , , . Retro-odontoid pseudotumor without atlantoaxial subluxation. J Clin Neurosci. 2010;17:649-52.
    [Google Scholar]
  14. , , , , , , . Retrodental mass in rheumatoid arthritis. J Spinal Disord Tech. 2013;26:E65-9.
    [Google Scholar]
Show Sections