Analysis of Kawasaki disease complicated with atlantoaxial rotatory subluxation / 中华实用儿科临床杂志

Objective:To investigate the clinical characteristics of children with Kawasaki disease (KD) complicated with atlantoaxial rotatory subluxation (AARS).Methods:Clinical characteristics of 60 AARS patients complicated with KD (the atlantoaxial rotatory subluxation group) and 60 patients with KD only diagnosed (the control group)in the Children′s Hospital of Chongqing Medical University from December 2010 to December 2019 were retrospectively analyzed.Differences between groups were compared by the Chi- square test and the t test. Results:A total of 8 365 KD patients were diagnosed during the study period, involving 60 cases (0.72%) complicated with AARS.which usually occurred in the acute phase with the onset ages of 3 to 6 years ( P<0.001). Initial clinical symptoms of KD complicated with AARS included fever with restricted neck movement (100.00%), neck mass (66.67%), torticollis (21.67%) and neck pain (11.67%). CT or X-ray exa-mination of the neck indicated AARS, with thickening and swelling of the cervical soft tissues in some cases.Compared with those of control group, red, dry, cracked lips ( P=0.01) and cervical lymph node swollen ( P<0.001) were significantly pronounced in KD patients complicated with AARS.The absolute and relative count of neutrophils were significantly higher in KD patients complicated with AARS (all P< 0.05). Cervical soft tissue swelling and thickening in B-ultrasound were more obvious than those in the control group( P<0.05). However, there were no significant differences in coronary artery lesions and the response to intravenous immunoglobulin (IVIG) combined with Aspirin between groups ( P>0.05). Head traction could relieve neck symptoms to a certain extent, but there was no significant difference between groups ( P>0.05). Conclusions:Cervical lymphadenopathy, red, dry, cracked lips, increase of absolute and relative count of neutrophils, and swelling and thicke-ning of cervical soft tissues were the high-risk factors of KD complicated with AARS.The complication of AARS in KD patients did not increase the risk of coronary artery injury and IVIG resistance.IVIG combined with aspirin achieved a good prognosis in the majority of KD patients complicated with AARS.

Traumatic Atlantoaxial Rotatory Subluxation in Adult: Case Report

Traumatic atlantoaxial rotatory subluxation (AARS) is generally found in pediatric patients, rarely found in adults, being a life-threatening condition especially when early diagnosis is not possible, which can lead to severe late neurological deficits.We describe a 38-year-old patient, victim of physical aggression caused by strangulation attempt who developed AARS, an uncommontraumatic cause. During the hospital care, the early diagnosis allowed us to institute a conservative treatment, which made the case uncommon, since most of the time surgical treatment is imperative. With the patient awake and under analgesia, a closed reduction was performed that promoted immediate pain relief, followed by a prescription of wearing a Philadelphia-type collar for 8 weeks. During the follow-up, cervical spine radiographies demonstrated no subluxation after removing the cervical collar. The patient was asymptomatic after 6months of treatment. This case supports the importance of nonoperative management of AARS in selected cases.

Grisel's Syndrome Induced by Mycobacterium tuberculosis

Grisel's syndrome is a non-traumatic subluxation of the atlantoaxial joints, which is caused by an inflammatory process in the upper neck. It is rare to find literary reports of Grisel's syndrome with an evident pathogen in a lesion. For the first time in Korea, we report a 36-year-old female with Grisel's syndrome having an atlantoaxial subluxation, which was caused by a retropharyngeal abscess secondary to pulmonary Mycobacterium tuberculosis. The patient was treated with an anti-tuberculosis regimen and was prescribed a Philadelphia collar for the control of torticollis. The result of magnetic resonance imaging (MRI) showed an improved atlantoaxial alignment, after drug treatment and immobilization. This patient was neurologically intact and free from symptomatic complaints at follow-up visit. Dynamic cervical radiograph confirmed that the atlantoaxial joints had been stable. The pathophysiology of Grisel's syndrome, along with anatomical attributes, was explained on the basis of the patient's clinical course.

Clinical analysis of acute limited neck motion in 420 children / 中华实用儿科临床杂志

Objective To study the clinical features of acute limited neck motion in children and its differential diagnosis, and to improve doctors' awareness and the early diagnosis of the disease.Methods A total of 420 patients with acute limited neck motion as the chief complaint and atlantoaxial rotary subluxation as the first diagnosis between January 2005 and January 2014 in Nanjing Children's Hospital Affiliated to Nanjing Medical University were enrolled.The clinical manifestations, imaging and consultation data of the 420 patients were retrospectively analyzed.Results There were totally 248 male patients and 172 female patients.The ages ranged from 2 to 14 years old(the mean age was 7.2 years old) ,the course ranged from 2 hours to 2 days.Among 420 patients,353 patients were diagnosed as atlantoaxial rotatory subluxation.Forty-two patients were diagnosed as cervical intervertebral disc calcification.Seven patients were diagnosed as Tourette syndrome.Four patients were diagnosed as intracranial tumors.Three patients were diagnosed as cervical eosinophilic granuloma.Two patients were diagnosed as cervical spinal tuberculosis.Two patients were diagnosed as viral encephalitis.Two patients were diagnosed as benign paroxysmal torticollis.Two patients were diagnosed as Kawasaki disease.One patient was diagnosed as neuroblastoma.One patient was diagnosed as hemophilia A.One patient was diagnosed as drug adverse reaction of metoclopramide.According to Fielding clinical classification,204 patients were type Ⅰ, 122 patients were type Ⅱ,21 patients were type Ⅲ, and 6 patients were type Ⅳ.Conclusions Based on disease distribution of acute limited neck motion in children, atlantoaxial rotatory subluxation seems to be the predominant while non-atlantoaxial rotatory subluxation is not uncommon.Differential diagnoses should be considered to avoid misdiagnosis.For atlantoaxial rotatory subluxation, conservative treatment including continuous mandible occipital belt traction and brace has been proved to be very effective methods.

A Morphologically Atypical Case of Atlantoaxial Rotatory Subluxation

A rare case of atlantoaxial rotatory subluxation occurred after pediatric cervical spine surgery performed to remove a dumbbell-shaped meningioma at the level of the C1/C2 vertebrae. This case is classified as a post-surgical atlantoaxial rotatory subluxation, but has a very rare morphology that has not previously been reported. Although there are several reports about post-surgical atlantoaxial rotatory subluxation, an important point of this case is that it might be directly related to the spinal cord surgery in C1/C2 level. On day 6 after surgery, the patient presented with the Cock Robin position, and a computed tomography scan revealed a normal type of atlantoaxial rotatory subluxation. Manual reduction was performed followed by external fixation with a neck collar. About 7 months after the first surgery, the subluxation became severe, irreducible, and assumed an atypical form where the anterior tubercle of C1 migrated to a cranial position, and the posterior tubercle of C1 and the occipital bone leaned in a caudal direction. The pathogenic process suggested deformity of the occipital condyle and bilateral C2 superior facets with atlantooccipital subluxation. A second operation for reduction and fixation was performed, and the subluxation was stabilized by posterior fixation. We encountered an unusual case of a refractory subluxation that was associated with an atypical deformity of the upper spine. The case was successfully managed by posterior fixation.