Grisel’s Syndrome: Non-traumatic Atlantoaxial Rotatory Subluxation—Report of Five Cases and Review of the Literature

Iaccarino, Corrado; Francesca, Ormitti; Piero, Spennato; Rubini, Monica; Rapanà, Armando; Bonis, Pasquale De; Aliberti, Ferdinando; Trapella, Giorgio; Mongardi, Lorenzo; Cavallo, Michele Alessandro; Cinalli, Giuseppe; Servadei, Franco

doi:10.1007/978-3-319-62515-7_40

Cited by 20 publications

(20 citation statements)

References 41 publications

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“…However, no female dominance has been observed [34]. In accordance with the prior findings by Fath et al, we found that GS was more likely to involve females, but that female dominance was not found in patients with GS resulting from other inflammatory conditions of the upper neck, namely tonsillitis and pharyngotonsillitis [28,35]. Regrettably, the cause of female dominance in patients with KD complicated by GS remains unknown.…”

Section: Discussionsupporting

confidence: 88%

Grisel’s syndrome in Kawasaki disease

Liu

Zhou

Hua

et al. 2020

Orphanet J Rare Dis

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Background Approximately 50–70% of patients with Kawasaki disease (KD) could present with cervical lymphadenopathy associated with deep neck inflammation, which may result in Grisel’s syndrome (GS). Given the possibility of neurological impairment owing to GS, it is important to understand the disease profile in KD. Therefore, we carried out this study to investigate this possible complication of KD, with the aim of improving pediatricians’ recognition and awareness. Methods Patients with KD complicated by GS in our hospital were retrospectively recruited for our study. The profiles of patients with GS (n = 10) were compared to those patients without GS (n = 1254). All the available literature describing these complications of KD was reviewed. Results The incidence of GS in KD was 0.6% in our population. Compared to patients without GS, KD patients with GS were older, presented with a significantly lower male:female ratio, and a higher incidence of cervical lymphadenopathy, a higher level of neutrophil count, and erythrocyte sedimentation rate. Ten articles reporting 14 KD patients with GS were reviewed. Of the total 24 patients, GS affected 7 males and 17 females, aged from 3.5 to 9 years old. Encouragingly, no delayed diagnosis and treatment of KD was found, and all patients received conservative therapy for GS, without intravenous immunoglobulin resistance, coronary artery lesions, and neurological impairment. Conclusions GS is a rare complication of KD with an incidence of 0.6%, predominantly affecting older, female children. The overall outcome of this disorder in KD was satisfactory with conservative therapy. Pediatricians, especially pediatric surgeons, should recognize and be aware of this possible complication of KD to avoid misdiagnosis and overtreatment.

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Section: Discussionsupporting

confidence: 88%

Grisel’s syndrome in Kawasaki disease

Liu

Zhou

Hua

et al. 2020

Orphanet J Rare Dis

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“…There are 4 types of atlantoaxial subluxation according to Fielding classification [11] . The first type is the rotation of atlas above axis without presence of anterior displacement.…”

Section: Discussionmentioning

confidence: 99%

Conservative approach for treatment of Grisel's syndrome after resection of lympahadenitis tuberculosis of the neck: A rare case report

Tobing

Hendriarto

Wikanjaya

2021

International Journal of Surgery Case Reports

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Introduction Grisel's syndrome is a rare condition characterized by nontraumatic rotatory subluxation of the atlantoaxial joint, which was caused by previous inflammation around the head and neck. It is usually seen in children and signed as torticollis. There was no consensus for management, yet early diagnosis and treatment is paramount. Presentation of case A 5-year-old girl came to outpatient clinic complaining of wry neck 1 day after surgical excision of her TB lymphadenitis and got worsen by time. There was no history of trauma around the neck. Examination under general anesthesia and CT scan revealed acquired severe torticollis consistent with Grisel's Syndrome Fielding type 3 accompanied by TB lymphadenitis, and TB myositis of the neck. Manipulation under general anesthesia and immobilization using Minerva cast followed by Lerman Cervicothoracal Osthosis was conducted. Discussion This patient was diagnosed with Grisel's syndrome and underwent conservative treatment consisting of reduction under general anesthesia and immobilization using Minerva cast for 6 weeks. The patient was then applied Lherman Cervical Thoracic Orthosis (CTO) halo brace for another 3 months. Anti-tuberculous drug was given to control tuberculous infection. Eight months follow-up showed neither residual deformity, neck pain, nor movement limitation of the neck. Conclusion Grisel's syndrome has excellent result that is treated with conservative treatment using reduction under general anesthesia and Minerva cast.

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“…Grisel's syndrome presents as sudden onset painful torticollis, neck rigidity, and decreased neck mobility of neck due to uncertain etiopathology. [1] The clinical complaints usually follow an infective or inflammatory disorders of the head and neck or nasopharyngeal region. [2] It is also reported after surgeries of the ear, nose, and throat region.…”

Section: Pearls and Discussionmentioning

confidence: 99%