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

Anania, Pasquale; Pavone, Piero; Pacetti, Mattia; Truffelli, Monica; Pavanello, Marco; Ravegnani, Marcello; Consales, Alessandro; Cama, Armando; Piatelli, Gianluca

doi:10.1016/j.wneu.2019.02.035

Cited by 15 publications

(11 citation statements)

References 55 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%

“…The mean size of the cervical lymphadenopathy was 2.9 cm, which was greater than in the general KD population (1.5 cm) [27]. In accordance with patients with upper respiratory tract infection (URTI) regarded as a common cause of GS [28,29], we found this disorder in KD prone to involve older children (median age: 5.9 years) than in the general KD population. This is associated with a high prevalence of cervical lymphadenopathy in older children with KD [30], which is partly in line with the prior findings by Nozaki [9].…”

Section: Discussionsupporting

confidence: 65%

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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%

Section: Discussionsupporting

confidence: 65%

Grisel’s syndrome in Kawasaki disease

Liu

Zhou

Hua

et al. 2020

Orphanet J Rare Dis

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“…5 However; the gold standard diagnostic tool of atlantoaxial rotary subluxation is a CT scan with three-dimensional reconstruction which evaluates the atlanto-dens interval, supporting classification according to the Fielding-Hawkins grading system (Table 1). 8,9 It should be suspected in patients presenting with fever, painful torticollis, restricted range of motion in the absence of trauma, but with a history of upper airway or head and neck infections, or otolaryngology surgery. 10,11 There is no gold standard management for this condition, even though various recommendations have been proposed.…”

Section: Discussionmentioning

confidence: 99%

Hyaluronic acid augmentation pharyngoplasty complicated by retropharyngeal abscess and grisel syndrome: Case report and literature review

Alkhaldi

Alwadi

Alenezi

et al. 2022

Clinical Case Reports

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Augmentation pharyngoplasty, in which tissue filler or grafts are used to augment the posterior nasopharynx, is an accepted option to treat velopharyngeal insufficiency. It is generally well tolerated and safe with limited side effects. In this study, we describe a case of a retropharyngeal abscess and Grisel syndrome following hyaluronic acid augmentation pharyngoplasty. Grisel syndrome is a serious condition that requires early diagnosis and prompt intervention to prevent further complications.

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“…It is worth mentioning that so far there is no gold standard for the management of atlantoaxial joint rotation and subluxation, although it is agreed that external immobilization alone or followed by surgical fixation is the most used therapy. Anania et al ( 15 ) developed a protocol for the management of AARS according to the major evidence in the literature. They recommended different treatment options in accordance with the duration of GS and Fielding–Hawkins classification, which includes drugs, Philadelphia collar, Halo jacket fixation, and surgical procedures.…”

Section: Discussionmentioning

confidence: 99%

Grisel's syndrome associated with mumps: A case report

et al. 2022

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Grisel's syndrome (GS) is defined as atlantoaxial rotatory subluxation/fixation not associated with trauma or bone disease, usually following head and neck infection/inflammation or ear, nose, and throat (ENT) surgery. Many conditions could lead to Grisel's syndrome, of which mumps is rarely to be seen. This report discusses a case of GS in children with Type I atlantoaxial joint subluxation and previously diagnosed mumps. A 6-year-old boy who had cervical pain and torticollis for 2 weeks was admitted to our hospital. There was no trauma and he had not had ENT surgery but was diagnosed with mumps 2 weeks previously due to swelling of the left cheek and cervical lymph node. Physical examination and computed tomography confirmed a diagnosis of Grisel's syndrome with an ADI (atlanto-dens interval) of 1.6 mm. The patient then received occipito-mandibular traction for 6 days and recovered. No recurrence was observed at 1 year follow-up. Physicians should raise awareness of this rare complication of mumps to avoid life-threatening neurological impairments owing to Grisel's syndrome.

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

Cited by 15 publications

References 55 publications

Grisel’s syndrome in Kawasaki disease

Grisel’s syndrome in Kawasaki disease

Hyaluronic acid augmentation pharyngoplasty complicated by retropharyngeal abscess and grisel syndrome: Case report and literature review

Grisel's syndrome associated with mumps: A case report

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