Bilateral C1 laminar hooks combined with C2 pedicle screw fixation in the treatment of atlantoaxial subluxation after Grisel syndrome

Abstract

Background context: Many etiologies can lead to atlantoaxial subluxaion. In Grisel syndrome (GS), this subluxation occurs spontaneously after inflammatory processes of the head and neck. Diagnosis is typically based on clinical history and a strong suspicion of this syndrome. Nonsurgical treatment most often resolves the symptoms; however, in some cases surgical treatment is necessary to repair the subluxation. Various surgical techniques and instrumentation systems have been used to treat atlantoaxial subluxation, although there is no consensus regarding the best treatment method for the pediatric population.

Purpose: To describe a case of atlantoaxial subluxation in a child with GS treated surgically with an alternative construct.

Study design/setting: This is a case report and literature review.

Materials and methods: Our case study involves a 5-year-old girl with a 6-month history of unresolved Fielding type II atlantoaxial subluxation caused by GS. Despite conservative treatment, the patient's symptoms continued to progress. After two failed closed reduction attempts, open reduction and C1-C2 fusion were performed with atlas laminar hook and axis pedicle polyaxial screws. A literature review of the surgical treatment of GS was also performed.

Results: After surgery, the patient exhibited full clinical and functional recovery with complete resolution of symptoms. At the 36-month follow-up examination, there was continual evidence of satisfactory reduction and fusion. No complications were observed. Upon completion of the literature review, eight GS cases were found to have been treated surgically with the minimum patient age being 9 years.

Conclusions: Conservative management of GS is the most common and effective treatment; however, a few surgical cases have been reported in the literature with good results. Satisfactory clinical results and fusion at 36 months post surgery were seen in a pediatric patient with atlantoaxial subluxation and instability using atlas laminar hook and axis pedicle polyaxial screws.

Keywords: Atlantoaxial subluxation; Atlas laminar hook; Axis pedicle polyaxial screws; C1–C2 arthrodesis; Goel-Harms arthrodesis; Grisel syndrome.