C1‑C2 subluxation in enthesitis‑related arthritis: two case reports and literature review of ten cases

Pediatric Rheumatology, 2023 · DOI: https://doi.org/10.1186/s12969-023-00862-3 · Published: July 24, 2023

Rheumatology Pediatrics Spinal Disorders

Simple Explanation

C1-C2 subluxation is a rare complication of enthesitis-related arthritis (ERA). If left untreated, it may lead to functional impairment or cervical spinal cord compression. We present two cases of C1-C2 subluxation: an 8-year-old boy with ERA and 16-year-old boy with ERA with bilateral sacroiliitis. Cervical spine protection and ruling out spinal cord compression should be prioritized, in addition to controlling the underlying inflammation in ERA.

Study Duration

Not specified

Participants

Two case reports of ERA patients with C1-C2 subluxation (8-year-old boy and 16-year-old boy) and a literature review of ten cases

Evidence Level

Case reports and literature review

Key Findings

1
The diagnosis of C1-C2 subluxation is mostly based on radiographs and cervical spine computed tomography.
2
All patients were treated with non-steroidal anti-inflammatory drugs. Six ERA patients were treated surgically for cervical fusion.
3
Most ERA patients with sacroiliitis had cervical collar protection. Neurologic abnormalities after treatment were not reported.

Research Summary

C1-C2 subluxation refers to the partial displacement of C1 and C2 that limits the movement or rotation of the neck. Initial management of C1-C2 subluxation in ERA should be focused on protecting the cervical spine and ruling out spinal cord compression. Early recognition and treatment of C1-C2 subluxation in ERA are crucial for minimizing the risk of complications.

Practical Implications

Early Recognition and Treatment

Early diagnosis and appropriate treatment of C1-C2 subluxation in ERA are essential to reduce the risk of complications like permanent fusion and spinal cord compression.

Cervical Spine Protection

Prioritize cervical spine protection and rule out spinal cord compression in ERA patients presenting with neck pain or torticollis.

Comprehensive Evaluation

Consider minor trauma, previous upper respiratory infection, and post-operative retropharyngeal inflammation as inciting events in ERA patients with suspected C1-C2 subluxation.

Study Limitations

1
Small sample size (two case reports and a literature review of ten cases).
2
Retrospective nature of the literature review.
3
Unclear whether C1-C2 subluxation found in children with rheumatic disorders is likely to be triggered by minor trauma.

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