“…Besides that, a small amount of GS patients develop for no reason. Infection mainly involves the upper respiratory tract or head and neck regions, such as pharyngitis, otitis, mastoiditis, lymphadenitis and retropharyngeal abscess ( 6 ), tonsillitis, sinusitis, and submandibular abscess ( 16 ). Some rare infections include bronchitis and gastroenteritis ( 7 ), acute rheumatic fever ( 1 ), Kawasaki disease ( 7 ), cervical tuberculosis ( 8 ), infectious mononucleosis ( 9 ), and mycoplasma pneumoniae infection ( 10 ).…”
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.
“…Besides that, a small amount of GS patients develop for no reason. Infection mainly involves the upper respiratory tract or head and neck regions, such as pharyngitis, otitis, mastoiditis, lymphadenitis and retropharyngeal abscess ( 6 ), tonsillitis, sinusitis, and submandibular abscess ( 16 ). Some rare infections include bronchitis and gastroenteritis ( 7 ), acute rheumatic fever ( 1 ), Kawasaki disease ( 7 ), cervical tuberculosis ( 8 ), infectious mononucleosis ( 9 ), and mycoplasma pneumoniae infection ( 10 ).…”
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.
Inflammatory nontraumatic atlantoaxial rotatory subluxation (AAS) in children is an often-missed diagnosis, especially in the early stages of disease. Abscess formation and spinal cord compression are serious risks that call for immediate surgical attention. Neither radiographs nor non-enhanced computed tomography (CT) images sufficiently indicate inflammatory processes. Magnetic resonance imaging (MRI) allows a thorough evaluation of paraspinal soft tissues, joints, and ligaments. In addition, it can show evidence of vertebral distraction and spinal cord compression. After conducting a scoping review of the literature, along with scientific and practical considerations, we outlined a standardized pediatric MRI protocol for suspected inflammatory nontraumatic AAS. We recommend contrast-enhanced MRI as the primary diagnostic imaging modality in children with signs of torticollis in combination with nasopharyngeal inflammatory or ear nose and throat (ENT) surgical history.
“…The Fielding and Hawkins system was used to classify the displacement of the atlas with respect to the odontoid in patients with GS [ 24 ]. The therapeutic strategy for GS is based on the Fielding-Hawkins classification, including non-steroidal anti-inflammatory drugs, physiotherapy, analgesics and surgery [ 25 ].…”
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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