Os odontoideum with cervical mylopathy due to posterior subluxation of C1 presenting sleep apnea

doi:10.1007/s00776-011-0043-5

Journal of Orthopaedic Science

Volume 16, Issue 3, May 2011, Pages 329-333

https://doi.org/10.1007/s00776-011-0043-5 Get rights and content

Abstract

Sleep apnea is a breathing disorder that manifests as repetitive cessation of breathing during sleep. It has been reported that patients with upper cervical lesions caused by rheumatoid arthritis [1–5], Arnold-Chiari type 1 malformation [6] and anterior C1-2 osteochondroma [7] have sleep apnea. Os odontoideum is characterized by the radiolucent defect between the dens and the body of the axis. In some cases with os odontoideum, C1 is very unstable and causes compression of the spinal cord at the upper cervical level. We encountered a patient with os odontoideum presenting as sleep apnea. The patient had cervical myelopathy caused by posterior subluxation of C1. Spinal cord atrophy due to the compression was marked at the C1–2 level. To our knowledge, there are no reports of such a case. Here, we present the clinical findings of this patient, and the pathogenesis of sleep apnea is discussed.

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Cited by (5)

Treatment of os odontoideum in a patient with spastic quadriplegic cerebral palsy
2015, Journal of Clinical Neuroscience
Citation Excerpt :
There are other case reports of OO with respiratory compromise but without cerebral palsy. In a report on OO [22], instability caused symptoms of sleep apnea and myelopathy. An MRI demonstrated atrophy of the spinal cord and intramedullary high signal intensity at the level of C1.
Severe atlantoaxial instability due to os odontoideum in a patient with spastic cerebral palsy has not been well described. There is no consensus on treatment, particularly with regard to conservative or surgical options. Our patient was a 9-year-old girl with spastic cerebral palsy and unstable os odontoideum as an incidental finding. During the waiting period for elective surgical treatment, the patient developed respiratory compromise. Surgery was performed to reduce the subluxation and for C1–C2 arthrodesis and the girl regained baseline respiratory function. A CT scan was obtained 1 year after the initial surgery and revealed adequate maintenance of reduction and patency of the spinal canal. This patient highlights the fact that unstable os odontoideum can cause mortality due to respiratory distress in patients with spastic cerebral palsy. This is an important factor in deciding treatment options for cerebral palsy patients with low functional demand. We review the relevant literature.
The impact of spinal cord injury on breathing during sleep
2013, Respiratory Physiology and Neurobiology
Citation Excerpt :
We were able to identify 29 published studies examining the impact of SCI on breathing during sleep (see Table 1). In addition, we found 13 related clinical case reports (Biering-Sorensen et al., 1995; Danielpour et al., 2007; Goh and Li, 2004; Graham et al., 2004; Heike et al., 2007; Howard et al., 1998; Kam et al., 2009; Kawaguchi et al., 2011; Miyano et al., 2009; Nagaoka et al., 2006; Russian et al., 2011; Star and Osterman, 1988; Vella et al., 1984). Collectively, this literature demonstrates unequivocally that the incidence of SDB increases after SCI, with estimates ranging from 2 to 5 fold over the general population (Burns et al., 2000; Klefbeck et al., 1998b; Leduc et al., 2007; Stockhammer et al., 2002).
The prevalence of sleep disordered breathing (SDB) following spinal cord injury (SCI) is considerably greater than in the general population. While the literature on this topic is still relatively small, and in some cases contradictory, a few general conclusions can be drawn. First, while both central and obstructive sleep apnea (OSA) has been reported after SCI, OSA appears to be more common. Second, SDB after SCI likely reflects a complex interplay between multiple factors including body mass, lung volume, autonomic function, sleep position, and respiratory neuroplasticity. It is not yet possible to pinpoint a “primary factor” which will predispose an individual with SCI to SDB, and the underlying mechanisms may change during progression from acute to chronic injury. Given the prevalence and potential health implications of SDB in the SCI population, we suggest that additional studies aimed at defining the underlying mechanisms are warranted.
The Impact of Obstructive Sleep Apnea on Clinical, Perioperative, and Cost Outcomes in Patients Who Underwent Posterior Cervical Decompression and Fusion: A Single-Center Retrospective Analysis From 2008 to 2016
2022, International Journal of Spine Surgery
Unstable os odontoideum contributing to cervical myelopathy and obstructive sleep apnea
2021, Surgical Neurology International
Sleep apnea and cervical spine pathology
2014, European Spine Journal

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Case ReportOs odontoideum with cervical mylopathy due to posterior subluxation of C1 presenting sleep apnea

Abstract

J Clin Neurosci.

Sleep Med Rev.

Mayo Clin Proc.

Lancet.

Chest

Chest

Chest

Sleep apnea in rheumatoid arthritis patients with occipitocervical lesions: the prevalence and associated radiographic features

Eur Spine J.

Sleep apnoea caused by rheumatoid arthritis

Br J Rheumatol.

Sleep apnoea syndrome secondary to rheumatoid arthritis

Thorax.

Obstructive sleep apnea due to upper cervical lesions and temporomandibular joint destruction from rheumatoid arthritis: case report

Clin Rheumatol.

A case of Arnold-Chiari Type I malformation presenting with dysrhythmic breathing during sleep

Psychiatry Clin Neurosci.

Case Report
Os odontoideum with cervical mylopathy due to posterior subluxation of C1 presenting sleep apnea