Acta Medica International

CASE REPORT
Year
: 2015  |  Volume : 2  |  Issue : 1  |  Page : 172--173

Occipito-thoracic fusion in incidental dystropic os odontoideum in a patient suspected with osteogenesis imperfecta: A case report


P Reyes Rechilda Rhea1, P Tangente Ronald2, A Soriano James3, G Lagapa Euvin Paul4,  
1 Neurological Surgery Resident Physician, Southern Integrated Neurological Surgery Training Program, Davao City, Philippines
2 Spine Surgery, Department of Orthopedics, Davao Doctors Hospital, Southern Integrated Neurological Surgery Training Program, Davao City, Philippines
3 Program Director, Department of Neurosurgery, Southern Integrated Neurological Surgery Training Program, Davao City, Philippines
4 Training Officer, Department of Neurosurgery, Southern Integrated Neurological Surgery Training Program, Davao City, Philippines

Correspondence Address:
P Reyes Rechilda Rhea
Neurological Surgery Resident Physician, Southern Philippines Integrated Neurological Surgery Training Program, Davao City
Philippines

Abstract

Introduction: Os odontoideum is a rare lesion of the axis. Described as an ossicle, consisting of smooth and separate caudal portions of the odontoid process wherein its etiology has remained controversial. Os odontoideum complicating a possible osteogenesis imperfecta has not been reported before in the literature. Methods: We report the case of a 14-yr male patient presented with progressive weakness of both lower and upper extremities after a head trauma 10 months prior to admission, which presented as transient quadreparesis. Results: Magnetic resonance imaging of cervical spine showing cervicomedullary junction compression. Patient underwent surgical intervention and 3 months post operation, patient was reported to have steady gait and muscle grading of 5/5 on all extremities. Conclusion: Surgical fixation and fusion in patients with instability may prevent catastrophic neurologic insult after minor trauma in the future.



How to cite this article:
Rhea P R, Ronald P T, James A S, Paul G L. Occipito-thoracic fusion in incidental dystropic os odontoideum in a patient suspected with osteogenesis imperfecta: A case report.Acta Med Int 2015;2:172-173


How to cite this URL:
Rhea P R, Ronald P T, James A S, Paul G L. Occipito-thoracic fusion in incidental dystropic os odontoideum in a patient suspected with osteogenesis imperfecta: A case report. Acta Med Int [serial online] 2015 [cited 2021 Jan 20 ];2:172-173
Available from: https://www.actamedicainternational.com/text.asp?2015/2/1/172/209442


Full Text



 Introduction



Os odontoideum is a rare lesion of the axis described in 1886 by Giacomini as an ossicle, consisting of smooth and separate caudal portions of the odontoid process wherein its etiology has remained controversial.[1],[2] To the best of our knowledge, Os odontoideum complicating a possible osteogenesis imperfecta[3],[4],[5] has not been reported before in the literature.

 Case Report



We report a case of a 14-year-old male presented with progressive weakness of both lower and upper extremities. He reported an unsteady gait requiring assistance associated with lack of fine motor coordination in his hands. He had a trauma of his head and neck 10 months prior to admission resulting to a transient quadriparesis. Physical examination revealed limited cervical spine flexion and extension. On neurological examination, his gait was wide-based, unsteady and spastic. Both lower and upper extremities had a muscle weakness grade 3 of 5. Sensory deficits are present in both upper and lower extremities. He had a symmetrical hyperreflexia, as well as positive Hoffman sign, Babinski sign and clonus bilaterally. Radiographs of the cervical spine showed atlantoaxial instability from a dystrophic Os odontoideum with associated cervicomedullary compression [Figure 1].{Figure 1}

CT scan studies demonstrated a displaced ossicle located near the basion. The bodies of C3 to T1 are deformed with areas of nonfusion [Figure 2].{Figure 2}

The patient underwent suboccipital craniectomy, C1 laminectomy, occipito-thoracic fusion using rods and screws [Figure 3] and [Figure 4]. At the time of the latest follow-up at 3 months, his unsteady gait had fully recovered and the muscle grade has improved to 5 of 5.{Figure 3}{Figure 4}

 Discussion



Os odontoideum is rare and an important cervical spine abnormality that can be associated with significant spinal cord injury and should be considered when a young patient has symptoms consistent with upper cervical spine compression.[6],[7] Surgical fixation and fusion in patients with instability may prevent catastrophic neurologic insult after minor trauma in the future.

References

1Wollin DG: The osodontoideum. Separate odontoid process. J Bone Joint Surg Am 45:1459–1471, 1963.
2Giacomini C: Sull' esistenza dell' “osodontoideum” nell' vomo. Gior Accad Med Torino 49:24–28, 1886.
3Gordon CM, Bachrach LK, Carpenter TO, Crabtree N, El-Hajj Fuleihan G, Kutilek S, et al. Dual energy X-ray absorptiometry interpretation and reporting in children and adolescents: the 2007 ISCD Pediatric Official Positions. J Clin Densitom. Jan-Mar 2008;11(1):43–58. [Medline].
4Rauch F, Plotkin H, DiMeglio L, Engelbert RH, Henderson RC, Munns C, et al. Fracture prediction and the definition of osteoporosis in children and adolescents: the ISCD 2007 Pediatric Official Positions. J Clin Densitom. Jan-Mar 2008;11(1):22–8. [Medline].
5Baim S, Leonard MB, Bianchi ML, Hans DB, Kalkwarf HJ, Langman CB, et al. Official Positions of the International Society for Clinical Densitometry and executive summary of the 2007 ISCD Pediatric Position Development Conference. J Clin Densitom. Jan-Mar 2008;11(1):6–21. [Medline].
6Clements WD, Mezue W, Mathew B. Osodontoideum: congenital or acquired? That's not the question. Injury. 1995;26(9):640–642.
7Fielding JW, Hensinger RN, Hawkins RJ. Osodontoideum. J Bone Joint Surg Am. 1980;62(3):376–383.