PMC6186371_01
Angiography brain CT obtained to investigate the sudden right hemiparesis of the patient, showing a well-corticated bone fragment located superiorly of odontoid process in coronal reconstruction (a, white arrow) and in axial plane (c, white arrow) in bone window. This fragment seems an os odontoideum, with a pseudoarthrosis of a fracture of the dens as main differential diagnosis. In sagittal plane with bone window, it is associated with an atlantoaxial subluxation at this level (anterior arch of C1 above C2 body), with a narrowing of the space available for spinal cord between the previous 2006 CT exam (e, white line measuring 1.5 cm) and recent 2014 CT exam (f, white line measuring 1.4 cm) in bone window. There is no enlargement of the space between anterior surface of os odontoideum and the anterior arch of C1 on sagittal plane (b, black arrow). We can already see a pseudomass hyperattenuated but without enhancement in the late phase, posteriorly of os odontoideum in the cervical spinal canal in axial plane and soft tissue window (d, white arrowheads), compatible with a retroodontoid pseudotumor in ROP.
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fig1
The exam revealed a smooth and well-corticated bone ossicle measuring 14 mm and located superiorly to the odontoid process corresponding to an os odontoideum (Figures 1(a)-1(c)). The ossicle was associated with an atlantoaxial subluxation and with the posterior wall of 14 mm on spinal canal (Figures 1(e) and 1(f), white lines) that has increased since a previous CT 8 years ago. The late enhanced phase showed an intracanal hyperattenuated but no enhancing pseudomass situated just posterior to the ossicle (Figure 1(d)).
Complementary cervical enhanced MRI with Gadolinium administration, acquired to investigate the cervical spinal cord compression, showing the ROP as low signal intensity on both T1w and T2w images in sagittal planes surrounding the body of C2 and this ROP (a,b,d, white arrows). It appears without enhancement on T1w fat sat images after gadolinium administration in sagittal (c, white arrow) and axial (g, white arrows) planes. We can see some enhancement on axial T1w fat sat after contrast administration around interfacetar articulations of C1-C2 (h, white arrows), probably from degenerative origin. A sub centimetric geode in the basis of C2 shows low signal on T1w image in sagittal plane (b, black arrow) with an enhancement after contrast administration in axial plane (c, black arrow). The T2w image in sagittal plane reveals an area of high intensity in the intramedullary regions of C1 and C2 (d, white circle), due to compression by the ROP, but without diastasis between anterior arch of C1 and the dens (e, white arrow).
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A complementary cervical enhanced MRI with administration of Gadolinium confirmed a well-corticated ossicle and demonstrated a tissular retroodontoid process (Figures 2(a) and 2(b)). T2-weighted images and no enhancement (Figure 2(g)), compatible with a ROP. The main diagnosis was a noninflammatory ROP developed on atlantoaxial instability, secondary to an os odontoideum. The main differential diagnosis was pseudoarthrosis of an old fracture of the dens of axis. There was no enlargement of space between this os odontoideum and the anterior arch of C1 (Figure 2(e)). T1-weighted images and enhancement after contrast administration, was seen in the posterior dens basis of C2 (Figures 2(b) and 2(c)). The pseudotumor indenting into cervicomedullary cord and resulting in cord compression is shown (Figure 2(a)), with cervical myelopathy seen in high signal on T2-weighted images in sagittal plane (Figure 2(d)).
Unenhanced CT after posterior C1-C2 fixation. We see in coronal plane with bone window the transarticular C1-C2 screws (a, black arrows) and on volume rendering in coronal and sagittal reconstructions (b,c, the screws are highlighting). Spinal canal decompression by laminectomy is also made, seen in axial plane with bone window (d, white arrowheads) showing the absence of posterior arch of C1 (in comparison to image c of Figure 1). The intracanal ROP is not removed but regressed in size compared to preoperative CT.
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Given the severity of the radiological findings and the clinical impact due to spinal cord compression and the life-threatening risk, patient was treated with cervical posterior screw fixation and a decompressive laminectomy at C1-C2 level (Figures 3(a)-3(d)).
Comparison of the size of ROP on preoperative CT and postoperative CT with soft tissue window and in axial plane. We see the regression of size of the ROP between preoperative CT (a) with thickness of 16.6 mm under the os odontoideum and the postoperative CT just after surgical management (b) with thickness of 14.6 mm, at 4 months with thickness of 13 mm and at 6 months with thickness of 11.2 mm after surgery.
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The intracanal ROP is not removed but regressed in size in each postoperative CT compared to preoperative CT (Figures 4(a)-4(d)).
Postoperative X-rays of the cervical spine are also acquired for the follow-up. We see the posterior C1-C2 fixation with transarticular screws (a,b, black arrows). These X-rays show better degenerative changes of the cervical spine; disco-uncarthrosis (a, white arrowheads), interarticular posterior staggered arthrosis (a, b, black arrowheads), and anterior marginal osteophytosis (b, black-framed white arrowheads).
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In the postoperative follow-up, entire cervical spine showed marked degenerative changes, best viewed on postoperative cervical spine X-rays (Figures 5(a) and 5(b)) as disco-uncarthrosis (white arrowheads), interarticular posterior arthrosis (black arrowheads), and anterior marginal osteophytosis (black-framed white arrowheads).