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DOI: 10.4103/ajns.AJNS_35_19
Dystopic Os odontoideum causing cervical myelopathy: A rare case report and review of literature

Os odontoideum (OO) was first described by Giacomini in 1886 as separation of the odontoid process from the body of the axis. Instability can consequently occurs at this level due to the failure of the transverse atlantal ligament (TAL) and this atlantoaxial instability can be a cause of progressive neurological deficits. It is considered a rare anomaly of the odontoid process. It is a disease with controversial etiology, debatable incidence, and only a partly known natural history owing to the paucity of the literature on this topic. There are insufficient demographic data about the occurrence of the disease, and most of the management is dictated by the isolated case reports and few studies which have been carried out at handful of institutes. OO is classified into two types by Fielding et al. based on the anatomic location: orthotopic and dystopic. Orthotopic OO consists of an ossicle that moves with the anterior arch of the atlas, whereas the dystopic type presents as an ossicle near the basion or one that is fused with the clivus. In one magnetic resonance imaging (MRI) study of odontoid morphology, a 0.7% (1 case of 133 patients) incidence was reported. The spectrum of the clinical presentation varies from completely asymptomatic individuals to patients presenting with features of cervical myelopathy. Here, we present a case of 35-year-old-male with dystopic OO who presented to us with features of gradually progressing cervical myelopathy without any obvious history of neck trauma. On investigations, he was found to have atlantoaxial instability with wide atlanto-dens interval. He was treated with the posterior C1-C2 stabilization and reduction of atlantoaxial instability.
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Publication History
Received: 07 February 2019
Accepted: 10 January 2020
Article published online:
16 August 2022
© 2020. Asian Congress of Neurological Surgeons. This is an open access article published by Thieme under the terms of the Creative Commons Attribution-NonDerivative-NonCommercial License, permitting copying and reproduction so long as the original work is given appropriate credit. Contents may not be used for commercial purposes, or adapted, remixed, transformed or built upon. (https://creativecommons.org/licenses/by-nc-nd/4.0/)
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References
- 1 Giacomini C. Sull' esistenza dell' “os odontoideum” nell' uomo. Gior Accad Med Torino 1886;49:24-8.
- 2 Fielding JW, Hensinger RN, Hawkins RJ. Os Odontoideum. J Bone Joint Surg Am 1980;62:376-83.
- 3 Perdikakis E, Skoulikaris N. The odontoid process: Various configuration types in MR examinations. Eur Spine J 2014;23:1077-83.
- 4 Tejada Meza H, Modrego Pardo P, Gazulla Abio J. Cervical myelopathy as the initial manifestation of os odontoideum. Neurologia 2016;31:278-9.
- 5 Zhang Z, Wang H, Liu C. Acute traumatic cervical cord injury in pediatric patients with os odontoideum: A series of 6 patients. World Neurosurg 2015;83:1180.e1-6.
- 6 Sankar WN, Wills BP, Dormans JP, Drummond DS. Os odontoideum revisited: The case for a multifactorial etiology. Spine (Phila Pa 1976) 2006;31:979-84.
- 7 White IK, Mansfield KJ, Fulkerson DH. Sequential imaging demonstrating os odontoideum formation after a fracture through the apical odontoid epiphysis: Case report and review of the literature. Childs Nerv Syst 2013;29:2111-5.
- 8 Stevens JM, Chong WK, Barber C, Kendall BE, Crockard HA. A new appraisal of abnormalities of the odontoid process associated with atlanto-axial subluxation and neurological disability. Brain 1994;117 (Pt 1):133-48.
- 9 Morgan MK, Onofrio BM, Bender CE. Familial os odontoideum. Case report. J Neurosurg 1989;70:636-9.
- 10 Yukata K, Katoh S, Sairyo K, Matsui Y, Hamada Y, Yasui N. Os odontoideum in achondroplasia: A case report. J Pediatr Orthop B 2008;17:103-5.
- 11 Kornreich L, Horev G, Schwarz M, Karmazyn B, Laron Z. Laron syndrome abnormalities: Spinal stenosis, os odontoideum, degenerative changes of the atlanto-odontoid joint, and small oropharynx. AJNR Am J Neuroradiol 2002;23:625-31.
- 12 Dias RP, Buchanan CR, Thomas N, Lim S, Solanki G, Connor SE, et al. Os odontoideum in Wolcott-Rallison syndrome: A case series of 4 patients. Orphanet J Rare Dis 2016;11:14.
- 13 Ricciardi JE, Kaufer H, Louis DS. Acquired os odontoideum following acute ligament injury. Report of a case. J Bone Joint Surg Am 1976;58:410-2.
- 14 Visocchi M, Di Rocco C. Os odontoideum syndrome: Pathogenesis, clinical patterns and indication for surgical strategies in childhood. Adv Tech Stand Neurosurg 2014;40:273-93.
- 15 Sasaki H, Itoh T, Takei H, Hayashi M. Os odontoideum with cerebellar infarction: A case report. Spine (Phila Pa 1976) 2000;25:1178-81.
- 16 Arvin B, Fournier-Gosselin MP, Fehlings MG. Os odontoideum: Etiology and surgical management. Neurosurgery 2010;66:22-31.
- 17 Hughes TB Jr., Richman JD, Rothfus WE. Diagnosis of os odontoideum using kinematic magnetic resonance imaging. A case report. Spine (Phila Pa 1976) 1999;24:715-8.
- 18 Dai L, Yuan W, Ni B, Jia L. Os odontoideum: Etiology, diagnosis, and management. Surg Neurol 2000;53:106-8.
- 19 Michaels L, Prevost MJ, Crang DF. Pathological changes in a case of os odontoideum (separate odontoid process). J Bone Joint Surg Am 1969;51:965-72.