A large retro-odontoid cystic mass caused by transverse ligament degeneration with atlantoaxial subluxation leading to granuloma formation and chronic recurrent microbleeding case report.

BACKGROUND CONTEXT

Noninfectious nontumorous retro-odontoid masses are rare, and masses have not been reported to extensively compress the spinal cord. We encountered a case of a large retro-odontoid lesion that extensively compressed the spinal cord.

CASE REPORT

A 76-year-old-man reported experiencing a sudden onset of neck pain, hand and foot paresthesia, dysarthria, and dysphagia. When symptoms had not eased by 10 days of treatment with external stabilization and bed rest, he was referred to our hospital. Dynamic radiographs of the cervical spine showed that the atlantodental interval widened from 2 mm on extension to 7 mm on flexion. Computed tomography did not reveal abnormality of the odontoid process or the presence of a high-density area that could suggest calcification in or near the cystic mass. Fluid-attenuated inversion recovery axial magnetic resonance image showed a mass that was 3.0-cm wide, 2.7-cm high, and 2.5-cm thick that severely compressed the lower brain stem. T2-weighted magnetic resonance imaging showed that the mass contained a solid part posterior to the C2 dense area, extending rostrally, compatible with the presence of degenerated and hypertrophic ligaments. We performed surgical decompression of the lesion combined with atlantoaxial fixation. The partly cystic mass, which was located extradurally, had xanthochromic content, indicating microbleeding. Dysarthria and dysphagia immediately disappeared, and neurologic symptoms disappeared by 1 month. At 1-year follow-up, the patient remained symptom free, and computed tomography scans did not show recurrence of the mass. The pathologic diagnosis of degenerative ligament tissue with chronic recurrent microbleeding and associated granulation was made.

DISCUSSION

A possible explanation why the cyst grew to an exceptionally large size is that the transverse ligament of axis became degenerated and hypertrophic because of chronic mechanical stress by atlantoaxial subluxation. Then, a part of the ligament developed reactive granulation with small vessel formation. Finally, rupture of these small vessels caused repeated episodes of microbleeding, resulting in formation of a large cyst. The observation of degenerative ligament tissue, granulation formation, and microbleeding differentiated it from a synovial cyst or a ganglion cyst. The presence of hemosiderin deposits suggested chronic recurrent microbleeding. Taking all our findings together, we believe that our case of retro-odontoid cystic mass is different from the others that have been reported. Atlantoaxial instability may cause a large mass, such as we described here, so that careful observation is important.