Grisel's syndrome in otolaryngology: A systematic review

Objective At the conclusion of the report, the readers should be able to recognize the rare complication of adenotonsillectomy of Grisel syndrome, discuss its pathogenesis, and provide early, effective treatment



Study Design Analysis of the clinical presentation, plain radiographs, computed tomography scans, and magnetic resonance imaging scans of a child with Grisel syndrome after adenotonsillectomy



Methods Retrospective study of the case record and a review of the literature regarding the pathogenesis, treatment modalities, outcome, and prognosis after early and delayed treatment of Grisel syndrome



Results Pathophysiology of atlantoaxial subluxation revolves around the periodontoid vascular plexus that drains the posterosuperior pharyngeal region Infectious and inflammatory emboli cause synovial engorgement, weakening paraspinal ligaments Radiological studies play an important role in diagnosis Treatment consists of cervical immobilization, muscle relaxants, analgesics, and antibiotics Full resolution is expected with early conservative management Failure to recognize the syndrome can lead to catastrophic results



Conclusions Recognition of Grisel syndrome in addition to other rare complications of adenotonsillectomy requires a high index of suspicion Early intervention is the critical factor for a positive outcome However, delayed diagnosis is common and can result in catastrophic consequences, including neurological deficits, cosmetic deformity and, in rare instances, paralysis and death in the short term

Nontraumatic Atlantoaxial Subluxation (Grisel Syndrome): A Rare Complication of Otolaryngological Procedures

Three extraordinary complications of adenotonsillectomy.

Background and Objective. Grisel's syndrome is a rare syndrome characterized by nontraumatic rotatory subluxation of the atlantoaxial joint. It usually affects children and typically presents with torticollis after ear, nose, and throat (ENT) surgery or head and neck infections. In the pediatric literature, there is only a small amount of available data; moreover, no systematic review has been previously done with focus on the pediatric population. We report our experience of two cases, and we provide a systematic review on Grisel's syndrome in children in order to offer a deeper insight about its clinical presentation, its current diagnosis, and principles of treatment. Case Reports and Review. We describe two boys of 9 and 8 years old, who developed atlantoaxial subluxation after adenoidectomy. Considering the early diagnosis, a conservative treatment was chosen, with no recurrence and no sequelae at follow-up. We identified 114 case reports, of which 90 describe children, for a total of 171 pediatric patients. Of the 154 cases in which cause was reported, 59.7% presented a head and neck infection and 35.7% had previous head and neck surgery. There is no sex prevalence (49.7% males versus 50.2% females). Mean delay in diagnosis is 33 days. Eight % of the patients had neurological impairment of the 165 cases which mentioned treatment, 96% underwent a conservative treatment, of whom the 8.8% recurred with the need of surgery. As a whole, 12% underwent surgery as a first- or second-line treatment. 3 6% of the patients whose follow-up was reported developed a sequela, minor limitation of neck movement being the most frequent. Conclusion. Grisel's syndrome should be suspected in children with painful unresponsive torticollis following ENT procedures or head and neck inflammation. CT scan with 3D reconstruction is the gold standard for diagnosis, allowing the identification of the subluxation and the classification according to the Fielding-Hawkins grading system. Surgical treatment is indicated in case of high-grade instability or failure of conservative treatment. Review of the literature shows how early diagnosis based on clinical and radiological evaluation is crucial in order to avoid surgical treatment and neurologic sequelae.

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Grisel's Syndrome in Children: Two Case Reports and Systematic Review of the Literature.

Grisel Syndrome in Pediatric Age: A Single-Center Italian Experience and Review of the Literature

Acute torticollis due to non-traumatic atlanto-axial subluxation (AAS) is often seen in children presenting with inflammatory conditions of the upper respiratory tract and the neck. Grisel's syndrome is the eponym given to this condition. These patients may present earlier in the disease process without evident subluxation. Thus, early recognition of the condition with prompt commencement of appropriate conservative treatment could halt the progression into Grisel's syndrome. The purpose of this study is to address the importance of early recognition of inflammatory torticollis that can be treated successfully by conservative methods. A retrospective review was made of the case files and radiological investigations of 13 children with fever and torticollis who were treated in the neurosurgery unit of Hamad General Hospital in Qatar, over a two-year period from July 1996 to July 1998. The children were aged between three and 12 years with a male to female ratio of 3:1. All patients arrived at the hospital within 48 hours of onset of torticollis and almost all had manifestations of upper respiratory tract or head and neck infections. Radiological examination by cervical spine X-rays, computerized tomography (CT) or magnetic resonance images (MRI) revealed that only three cases out of 13 had AAS. All patients underwent conservative treatment that included rest, neck collar, simple analgesics and antibiotics, where appropriate. A muscle relaxant was used in nine cases and Halter traction was applied to the three with AAS. All patients responded well to treatment and none required surgical intervention for AAS. We conclude that the majority of children presenting acutely with inflammatory torticollis have rotational deformity only without AAS. Progression to the latter, i.e. Grisel's syndrome, may be aborted should the diagnosis be made early and conservative treatment initiated in time. On the other hand, delay in diagnosis would deprive these children an opportunity of receiving effective conservative treatment.

Fever and acquired torticollis in hospitalized children.

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Fever and acquired torticollis in hospitalized children.