Early diagnosis of Grisel’s syndrome in children with favorable outcome

Özalp, Hakan; Hamzaoğlu, Vural; Avcı, Emel; Karataş, Derya; İsmi, Onur; Talaş, Derya Ümit; Bağdatoğlu, Celal; Dağtekin, Ahmet

doi:10.1007/s00381-018-3996-2

Cited by 9 publications

(11 citation statements)

References 16 publications

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“…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: 64%

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: 64%

Grisel’s syndrome in Kawasaki disease

Liu

Zhou

Hua

et al. 2020

Orphanet J Rare Dis

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“…GS also presents after maxillofacial or otorhinolaryngology procedures, including tonsillectomy and adenoidectomy [2]. For the patient presented in this report, though the tonsillectomy was documented as having no postoperative complications, the onset of neck pain, as well as the finding of enlarged lymph nodes, around the same time as the tonsillectomy suggest recurrence or exacerbation of existing torticollis secondary to GS [6].…”

Section: Etiologymentioning

confidence: 73%

Recurrent Torticollis and Cervical Subluxation in a Pediatric Patient

Iqbal¹,

Mazzola²

2020

SCR

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Background: Atlantoaxial subluxation (AAS), also referred to as C1-C2 subluxation, is a misalignment of the first two vertebrae of the cervical spine. AAS typically presents with a head tilt (torticollis) with limited range of motion (ROM). Torticollis is quite common in infants, but in older children, torticollis may be an indication of AAS. Method: In this retrospective case study, the clinical history of a female pediatric patient diagnosed with atlantoaxial subluxation presenting with recurrent torticollis is reviewed. Result: The patient was initially diagnosed with torticollis during infancy; torticollis partially resolved. However, after an ear infection, the child again developed torticollis secondary to Grisel’s Syndrome. Despite undergoing physical therapy treatments, torticollis persisted. The patient was referred to neurosurgery at age nine. AAS was diagnosed after a three-dimensional (3D) computed tomography (CT) scan. The child was subsequently placed in halo-traction to reduce the C1-C2 subluxation. Once the alignment was acceptable, the child was placed in a halo vest. However, even after several months of noninvasive cervical spine immobilization with a halo vest and hard cervical collar, the head tilt and cervical subluxation recurred due to bone remodeling. Conclusion: The diagnosis of AAS requires both a comprehensive physical examination and imaging following presentation of torticollis. Understanding the etiology of the torticollis early on is critical in preventing the occurrence of AAS after treatment.

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“…Nontraumatic atlantoaxial rotatory subluxation is a rare condition, and it should be included in the differential diagnosis of a child with painful torticollis after infection of the upper respiratory tract, postadenotonsillectomy, and other surgical procedures, such as pharyngoplasty and ear operation. [ 6 ] Torticollis with the head in the oblique position associated with muscular stiffness and reduced range of movements are the typical clinical findings. Usually, this syndrome affects children under the age of 12 years, without predisposition for gender.…”

Section: Discussionmentioning

confidence: 99%

Rotational dislocation C1–C2 after otoplasty under local anesthesia

Matos

Pinheiro

Costa

et al. 2020

J Craniovert Jun Spine

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Non-traumatic rotational atlantoaxial subluxation (NTARS) is rare and mostly reported after infection of the upper respiratory tract and named Grisel's syndrome. NTARS has also been reported after head-and-neck surgery, but it is extremely rare after otoplasty. A case of NTARS after bilateral otoplasty is reported under local anesthesia, a 15-year-old female being presented with painful torticollis. The diagnosis of atlantoaxial rotatory subluxation was performed using radiographs and computed tomography 2 weeks after the surgery. Closed reduction was performed by traction of the head and transoral direct pressure over an anterior dislocated C1 mass. The reposition of the joint was achieved, but it was very unstable, and it was not possible to keep the reduction. Open posterior reduction and posterior C1–C2 arthrodesis were performed followed by the use of a soft collar during 3 months.

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Early diagnosis of Grisel’s syndrome in children with favorable outcome

Cited by 9 publications

References 16 publications

Grisel’s syndrome in Kawasaki disease

Grisel’s syndrome in Kawasaki disease

Recurrent Torticollis and Cervical Subluxation in a Pediatric Patient

Rotational dislocation C1–C2 after otoplasty under local anesthesia

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