Progress in the diagnosis and treatment of Grisel’s syndrome after adenotonsillectomy_West China Medical Journal

Authors：

 XUE Di ¹ , LIANG Peng ^1,2

1. Department of Anesthestology, West China Hospital, Sichuan University, Chendu, Sichuan 610041, P. R. China;
2. Day Surgery Center, General Practice Medical Center, West China Hospital, Sichuan University, Chendu, Sichuan 610041, P. R. China;

Corresponding author：

XUE Di, Email: 528031012@qq.com

Keywords：

Adenotonsillectomy; Grisel’s syndrome; torticollis

DOI：

10.7507/1002-0179.202412226

Video：

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Grisel’s syndrome is a rare cervical spine disorder characterized by non-traumatic rotary subluxation of the atlantoaxial joint. This article provides a systematic review to collect evidence on its pathogenesis, clinical manifestations, diagnosis, treatment, complications, and risk management, in order to guide clinical diagnosis and treatment. The syndrome is often associated with adenotonsillectomy. Patients typically present with neck stiffness, pain, and a “cock robin posture” (chin tucked in and head tilted forward). Diagnosis relies on MRI and CT scans. After timely diagnosis, most patients can control the condition through conservative treatment. However, those with ineffective conservative treatment or severe subluxation may require surgical intervention. Therefore, early diagnosis and treatment are crucial. This article focuses on the progress in the diagnosis and treatment of Grisel’s syndrome after adenotonsillectomy, which will provide new insights into the diagnosis and treatment of this rare disease.

Citation： XUE Di, LIANG Peng. Progress in the diagnosis and treatment of Grisel’s syndrome after adenotonsillectomy. West China Medical Journal, 2025, 40(2): 295-299. doi: 10.7507/1002-0179.202412226 Copy

1.	Bell C. The nervous system of the human body: embracing the papers delivered to the Royal Society on the subject of the nerves. London: Longman, Rees, Orme, Brown, and Green, 1830.
2.	Grisel P. Enucleation de l’atlas et torticollis nasopharyngien. Presse Médicale, 1930, 38: 50-53.
3.	Al-Driweesh T, Altheyab F, Alenezi M, et al. Grisel’s syndrome post otolaryngology procedures: a systematic review. Int J Pediatr Otorhinolaryngol, 2020, 137: 110225.
4.	孙宝春, 王丰, 杨淑芝, 等. 低温等离子辅助下儿童腺样体、扁桃体手术并发症分析. 临床耳鼻咽喉头颈外科杂志, 2017, 31(22): 1720-1723.
5.	Spinnato P, Zarantonello P, Guerri S, et al. Atlantoaxial rotatory subluxation/fixation and Grisel’s syndrome in children: clinical and radiological prognostic factors. Eur J Pediatr, 2021, 180(2): 441-447.
6.	Parke WW, Rothman RH, Brown MD. The pharyngovertebral veins: an anatomical rationale for Grisel’s syndrome. J Bone Joint Surg Am, 1984, 66(4): 568-574.
7.	Yu KK, White DR, Weissler MC, et al. Nontraumatic atlantoaxial subluxation (Grisel syndrome): a rare complication of otolaryngological procedures. Laryngoscope, 2003, 113(6): 1047-1049.
8.	赵喜红, 王运升. 扁桃体切除术后并发寰枢关节脱位 2 例. 临床耳鼻咽喉头颈外科杂志, 2019, 33(11): 1099-1101.
9.	Hong WJ, Lee JK, Hong JH, et al. Iatrogenic atlantoaxial rotatory subluxation after thyroidectomy in a pediatric patient: a case report. World J Clin Cases, 2023, 11(14): 3351-3355.
10.	Hashimoto K, Nishimura S, Shinyashiki Y, et al. Grisel’s syndrome after COVID-19 in a pediatric patient: a case report. Cureus, 2024, 16(6): e62028.
11.	Oshita Y, Takata K, Someya R, et al. Retrospective analysis of atlantoaxial rotatory fixation describing age distribution and gender ratio in children and adolescents: a preliminary report. J Orthop Sci, 2024, 29(2): 486-488.
12.	Al-Hussain OH, Al-Hussain G. Diagnostic approach and treatment options for pediatric cases of Grisel’s syndrome post otolaryngology procedure: a systematic review. Cureus, 2024, 16(1): e51739.
13.	Aldamouni MB, Albitar MH, Alhosainy ZH, et al. Challenges in Grisel’s syndrome management in a two-month-old infant. Cureus, 2023, 15(2): e35128.
14.	K P D, Cherian A. Pearls & Oy-sters: Grisel syndrome presenting as pseudodystonia: a twist in the neck. Neurology, 2022, 98(11): 462-465.
15.	Raucci U, Roversi M, Ferretti A, et al. Pediatric torticollis: clinical report and predictors of urgency of 1409 cases. Ital J Pediatr, 2024, 50(1): 86.
16.	Barcelos AC, Patriota GC, Netto AU. Nontraumatic atlantoaxial rotatory subluxation: grisel syndrome. Case report and literature review. Global Spine J, 2014, 4(3): 179-186.
17.	Ortiz GL, Pratts I, Ramos E. Grisel’s syndrome: an unusual cause of torticollis. J Pediatr Rehabil Med, 2013, 6(3): 175-180.
18.	Iaccarino C, Francesca O, Piero S, et al. Grisel’s syndrome: non-traumatic atlantoaxial rotatory subluxation-report of five cases and review of the literature. Acta Neurochir Suppl, 2019, 125: 279-288.
19.	Wenger KJ, Hattingen E, Porto L. Magnetic resonance imaging as the primary imaging modality in children presenting with inflammatory nontraumatic atlantoaxial rotatory subluxation. Children (Basel), 2021, 8(5): 329.
20.	Fielding JW, Hawkins RJ. Atlanto-axial rotatory fixation. (Fixed rotatory subluxation of the atlanto-axial joint). J Bone Joint Surg Am, 1977, 59(1): 37-44.
21.	Viscone A, Brembilla C, Gotti G. The importance and effectiveness of conservative treatment in Grisel’s syndrome. J Pediatr Neurosci, 2014, 9(2): 200-201.
22.	Ghodke A, Gadkari A, Trivedi R, et al. Torticollis in an 8-year-old child due to Grisel’s syndrome: a case report. Surg Neurol Int, 2022, 13: 502.
23.	Yeung CY, Feng CK. Halter traction for the treatment of atlantoaxial rotatory fixation. J Bone Joint Surg Am, 2022, 104(3): 229-238.
24.	Deichmueller CM, Welkoborsky HJ. Grisel’s syndrome: a rare complication following “small” operations and infections in the ENT region. Eur Arch Otorhinolaryngol, 2010, 267(9): 1467-1473.
25.	Ferdinandov D, Yankov D, Bussarsky A. Grisel’s syndrome in adults: a case report. Surg Neurol Int, 2021, 12: 15.
26.	Miller BJ, Lakhani R, Rashid A, et al. Painful torticollis following adenotonsillectomy: a cardinal sign of atlantoaxial subluxation. BMJ Case Rep, 2018, 2018: bcr2017223567.
27.	Salpietro V, Polizzi A, Granata F, et al. Upper respiratory tract infection and torticollis in children: differential diagnosis of Grisel’s syndrome. Clin Neuroradiol, 2012, 22(4): 351-353.
28.	高志伟, 王俊. 扁桃体术后继发性多次出血 1 例. 临床耳鼻咽喉头颈外科杂志, 2014, 28(19): 1530-1531.

1. Bell C. The nervous system of the human body: embracing the papers delivered to the Royal Society on the subject of the nerves. London: Longman, Rees, Orme, Brown, and Green, 1830.
2. Grisel P. Enucleation de l’atlas et torticollis nasopharyngien. Presse Médicale, 1930, 38: 50-53.
3. Al-Driweesh T, Altheyab F, Alenezi M, et al. Grisel’s syndrome post otolaryngology procedures: a systematic review. Int J Pediatr Otorhinolaryngol, 2020, 137: 110225.
4. 孙宝春, 王丰, 杨淑芝, 等. 低温等离子辅助下儿童腺样体、扁桃体手术并发症分析. 临床耳鼻咽喉头颈外科杂志, 2017, 31(22): 1720-1723.
5. Spinnato P, Zarantonello P, Guerri S, et al. Atlantoaxial rotatory subluxation/fixation and Grisel’s syndrome in children: clinical and radiological prognostic factors. Eur J Pediatr, 2021, 180(2): 441-447.
6. Parke WW, Rothman RH, Brown MD. The pharyngovertebral veins: an anatomical rationale for Grisel’s syndrome. J Bone Joint Surg Am, 1984, 66(4): 568-574.
7. Yu KK, White DR, Weissler MC, et al. Nontraumatic atlantoaxial subluxation (Grisel syndrome): a rare complication of otolaryngological procedures. Laryngoscope, 2003, 113(6): 1047-1049.
8. 赵喜红, 王运升. 扁桃体切除术后并发寰枢关节脱位 2 例. 临床耳鼻咽喉头颈外科杂志, 2019, 33(11): 1099-1101.
9. Hong WJ, Lee JK, Hong JH, et al. Iatrogenic atlantoaxial rotatory subluxation after thyroidectomy in a pediatric patient: a case report. World J Clin Cases, 2023, 11(14): 3351-3355.
10. Hashimoto K, Nishimura S, Shinyashiki Y, et al. Grisel’s syndrome after COVID-19 in a pediatric patient: a case report. Cureus, 2024, 16(6): e62028.
11. Oshita Y, Takata K, Someya R, et al. Retrospective analysis of atlantoaxial rotatory fixation describing age distribution and gender ratio in children and adolescents: a preliminary report. J Orthop Sci, 2024, 29(2): 486-488.
12. Al-Hussain OH, Al-Hussain G. Diagnostic approach and treatment options for pediatric cases of Grisel’s syndrome post otolaryngology procedure: a systematic review. Cureus, 2024, 16(1): e51739.
13. Aldamouni MB, Albitar MH, Alhosainy ZH, et al. Challenges in Grisel’s syndrome management in a two-month-old infant. Cureus, 2023, 15(2): e35128.
14. K P D, Cherian A. Pearls & Oy-sters: Grisel syndrome presenting as pseudodystonia: a twist in the neck. Neurology, 2022, 98(11): 462-465.
15. Raucci U, Roversi M, Ferretti A, et al. Pediatric torticollis: clinical report and predictors of urgency of 1409 cases. Ital J Pediatr, 2024, 50(1): 86.
16. Barcelos AC, Patriota GC, Netto AU. Nontraumatic atlantoaxial rotatory subluxation: grisel syndrome. Case report and literature review. Global Spine J, 2014, 4(3): 179-186.
17. Ortiz GL, Pratts I, Ramos E. Grisel’s syndrome: an unusual cause of torticollis. J Pediatr Rehabil Med, 2013, 6(3): 175-180.
18. Iaccarino C, Francesca O, Piero S, et al. Grisel’s syndrome: non-traumatic atlantoaxial rotatory subluxation-report of five cases and review of the literature. Acta Neurochir Suppl, 2019, 125: 279-288.
19. Wenger KJ, Hattingen E, Porto L. Magnetic resonance imaging as the primary imaging modality in children presenting with inflammatory nontraumatic atlantoaxial rotatory subluxation. Children (Basel), 2021, 8(5): 329.
20. Fielding JW, Hawkins RJ. Atlanto-axial rotatory fixation. (Fixed rotatory subluxation of the atlanto-axial joint). J Bone Joint Surg Am, 1977, 59(1): 37-44.
21. Viscone A, Brembilla C, Gotti G. The importance and effectiveness of conservative treatment in Grisel’s syndrome. J Pediatr Neurosci, 2014, 9(2): 200-201.
22. Ghodke A, Gadkari A, Trivedi R, et al. Torticollis in an 8-year-old child due to Grisel’s syndrome: a case report. Surg Neurol Int, 2022, 13: 502.
23. Yeung CY, Feng CK. Halter traction for the treatment of atlantoaxial rotatory fixation. J Bone Joint Surg Am, 2022, 104(3): 229-238.
24. Deichmueller CM, Welkoborsky HJ. Grisel’s syndrome: a rare complication following “small” operations and infections in the ENT region. Eur Arch Otorhinolaryngol, 2010, 267(9): 1467-1473.
25. Ferdinandov D, Yankov D, Bussarsky A. Grisel’s syndrome in adults: a case report. Surg Neurol Int, 2021, 12: 15.
26. Miller BJ, Lakhani R, Rashid A, et al. Painful torticollis following adenotonsillectomy: a cardinal sign of atlantoaxial subluxation. BMJ Case Rep, 2018, 2018: bcr2017223567.
27. Salpietro V, Polizzi A, Granata F, et al. Upper respiratory tract infection and torticollis in children: differential diagnosis of Grisel’s syndrome. Clin Neuroradiol, 2012, 22(4): 351-353.
28. 高志伟, 王俊. 扁桃体术后继发性多次出血 1 例. 临床耳鼻咽喉头颈外科杂志, 2014, 28(19): 1530-1531.

West China Medical Journal

Progress in the diagnosis and treatment of Grisel’s syndrome after adenotonsillectomy

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