- Pars Advanced and Minimally Invasive Medical Manners Research Center, Pars Hospital, Iran University of Medical Sciences, Tehran, Iran.
DOI:10.25259/SNI_663_2020Copyright: © 2020 Surgical Neurology International 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: Abolfazl Rahimizadeh, Walter Williamson, Shaghayegh Rahimizadeh, Mahan Amirzadeh. Painful torticollis due to tubercular atlantoaxial rotatory fixation: A case report. 16-Dec-2020;11:440
How to cite this URL: Abolfazl Rahimizadeh, Walter Williamson, Shaghayegh Rahimizadeh, Mahan Amirzadeh. Painful torticollis due to tubercular atlantoaxial rotatory fixation: A case report. 16-Dec-2020;11:440. Available from: https://surgicalneurologyint.com/surgicalint-articles/10461/
Background: Tubercular atlantoaxial, rotary dislocation warranting fixation (AARF) is an extremely rare event.
Case Description: AARF was suspected in a 23-year-old female with painful torticollis. When diagnostic studies documented unilateral destruction of the left lateral mass of the atlas, she underwent removal of the lateral mass, reduction of the deformity, and C1-C2 fusion/reconstruction utilizing an iliac bone graft. Laboratory tests and the pathologic surveys were all consistent with the diagnosis of underlying tuberculosis.
Conclusion: We present a case of tubercular atlantoaxial, rotary dislocation (AARF) in a patient who warranted C1-C2 decompression, reduction, and fusion.
Keywords: Atlantoaxial dislocation, Atlantoaxial rotatory fixation, Atlas, Craniovertebral junction, Tuberculosis, Upper cervical
Tuberculosis (TB) involving the atlantoaxial complex is rare, accounting for approximately 0.1% of all spinal tubercular infections.[
A 23-year-old female presented with a 4-week history of severe neck pain (VAS:10) and torticollis with a classic “Cock Robin” deformity. Laboratory studies demonstrated an increased erythrocyte sedimentation rate (ESR) of 42. The lateral cervical plain radiographs and computed tomography (CT) axial, 2D, and 3D reconstructed images showed C1–C2 AARF with destruction of the left lateral mass of the atlas [
3D computed tomography scan of the atlantoaxial complex (a) frontal view showing destruction of the left lateral mass and a part of anterior ring of atlas, note forward displacement of the right C1 lateral mass. (b) Occipital view demonstrates rotation of the posterior ring of atlas to the right.
As cervical traction failed to reduce the deformity, surgical intervention was warranted. Surgery required; the initial insertion of bilateral C2 pedicle screws, isolation of the V3 segment of the vertebral artery, removal of the destroyed left C1 lateral mass in a piecemeal fashion, and fusion (e.g., utilizing a tricortical iliac bone graft secured with left C1 laminar hook-C2 pedicle screw, and an additional right C1 lateral mass-C2 pedicle screw rod construct) [
Bacteriology and pathology
The operative specimens demonstrated: a positive polymerase chain reaction (PCR) for TB, and the pathology was compatible with a granulomatous infection.
Four-drug therapy was warranted for TB; isoniazid (5 mg/kg), rifampicin (10 mg/kg), ethambutol (15 mg/kg), and pyrazinamide (25 mg/kg). They were administered as a first-line of treatment for 4 months. This was followed by an additional 12 months of rifampicin and isoniazid.
Spinal TB, presenting as AARF with painful torticollis, is extremely rare.
Severe painful torticollis may be the only clinical indication that AARP is present. Patients may exhibit unilateral destruction of the lateral mass of the atlas with/ without infiltration/disruption of the alar ligaments. The ESRs are typically increased, and the Mantoux test is typically positive.[
AARF is the best documented utilizing axial reconstructed 2D and 3D CT images and CTA.[
The following findings are classical for TB; an osteolytic, fragmented lesion involving the C1 lateral mass, deviation of the odontoid to the affected lateral mass side, and forward displacement of the contralateral lateral mass of atlas.[
Magnetic resonance imaging also may help to establish the diagnosis of tuberculous involvement of the C1 lateral mass, by demonstrating heterogeneous intensity on the T1, and hyperintensity on the T2-weighted and fat-suppressed images.
The differential diagnoses for painful torticollis with unilateral involvement of the C1 lateral mass include TB tumors, rheumatoid arthritis, and other types of pyogenic spondylitis.[
In classic tubercular atlantoaxial dislocation, management strategies range from purely conservative treatment to radical operations.[
Here, we presented an extremely rare cause of painful torticollis due to tubercular AARF involving a unilateral C1 lateral mass requiring decompression, reduction, and fixation.
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