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Grisel syndrome as a complication of Kawasaki disease: a case report and review of the literature

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Abstract

Grisel syndrome is a non-traumatic atlantoaxial subluxation and a rare complication of any inflammatory condition of the upper neck and otolaryngological procedures. Delayed diagnosis causes neurological impairment, ranging from radiculopathy to paralysis and death. Kawasaki disease is a very frequent and important acute febrile vasculitis of childhood that is seen worldwide, and upper neck involvement (cervical lymphadenopathy) is one of the common symptoms of Kawasaki disease. A case of Grisel syndrome that occurred as a complication of Kawasaki disease is reported. This is the first case report, in English, of Grisel syndrome as a complication of Kawasaki disease. Conclusion: Pediatricians should be aware of Grisel syndrome as a possible complication of Kawasaki disease.

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References

  1. Battiata AP, Pazos G (2004) Grisel’s syndrome: the two-hit hypothesis—a case report and literature review. Ear Nose Throat J 83:553–555

    PubMed  Google Scholar 

  2. Bell C (1830) The nervous system of the human body embracing papers delivered to the Royal Society on the subject of the nerves. Longman, Rees, and Orme, London, p 403

    Google Scholar 

  3. Cai Z, Zuo R, Liu Y (2011) Characteristics of Kawasaki disease in older children. Clin Pediatr 50:952–956

    Article  Google Scholar 

  4. Grisel P (1930) Enucleation de l’atlas et torticollis nasopharyngien. Presse Med 38:50–53

    Google Scholar 

  5. Harma A, Firat Y (2008) Grisel syndrome: nontraumatic atlantoaxial rotatory subluxation. J Craniofac Surg 19:1119–1121

    Article  PubMed  Google Scholar 

  6. Igarashi C, Shimono Y, Hoshii S, Abo W, Kadowaki J, Monji J, Fukuhara H, Tame Y (1989) A case of Kawasaki disease complicated with atlanto-axial rotator fixation. J Clin Pediatr Sapporo 37:93–97 [in Japanese]

    Google Scholar 

  7. Kaketa K, Miyawaki Y, Kitano K, Hirohasi K (2004) Atlanto-axial rotator fixation due to cervical lymphadenitis. Ann Saiseikai Nakatu Hosp Osaka 14:235–237 [in Japanese]

    Google Scholar 

  8. Karkos PD, Benton J, Leong SC, Mushi E, Sivaji N, Assimakopoulos DA (2007) Grisel’s syndrome in otolaryngology: a systematic review. Int J Pediatr Otorhinolaryngol 71:1823–1827

    Article  PubMed  CAS  Google Scholar 

  9. Kliegman RM, Stanton BMD, Geme JS, Schor MF, Behrman RE (2011) Nelson textbook of pediatrics, 19th edn. Saunders, Philadelphia

    Google Scholar 

  10. Konishi S, Watanabe M, Kaneda M, Takahashi N, Tonoki H, Koga Y (2007) A report of atlantoaxial rotatory fixation related with cervical lymphadenopathy in 3 pediatric cases. J Jpn Pediatr Soc 111:1191–1195 [in Japanese]

    Google Scholar 

  11. Nakamura Y, Yashiro M, Uehara R, Sadakane A, Tsuboi S, Aoyama Y, Kotani K, Tsogzolbaatar EO, Yanagawa H (2012) Epidemiologic features of Kawasaki disease in Japan: results of the 2009–2010 nationwide survey. J Epidemiol 22:216–221

    PubMed  Google Scholar 

  12. Oda Y, Nakayama I, Shiraishi H, Hirade T, Fukushima H (2009) Atlantoaxial rotator fixation caused by Kawasaki disease; a case report. Orthop Surg Traumatol 52:1563–1565 [in Japanese]

    Google Scholar 

  13. Oda Y, Nihei K, Uemura Y, Fujiwara J, Yamaguchi K, Musha Y, Kishida M (2009) Two cases of Kawasaki disease with atlanto-axial rotator fixation. Jpn J Pediatr 62:2095–2099 [in Japanese]

    Google Scholar 

  14. Ortega-Evangelio G, Alcon JJ, Alvarez-Pitti J, Sebastia V, Juncos M, Lurbe E (2011) Eponym: Grisel syndrome. Eur J Pediatr 170:965–968

    Article  PubMed  Google Scholar 

  15. Osiro S, Tiwari KJ, Matusz P, Gielecki J, Tubbs RS, Loukas M (2012) Grisel’s syndrome: a comprehensive review with focus on pathogenesis, natural history, and current treatment options. Childs Nerv Syst 28(6):821–825

    Article  PubMed  Google Scholar 

  16. Tashiro R, Oyama T, Mihara A, Kanai R, Yamaguchi S, Hiramatu Y, Matsuzaki M, Uchio Y (2011) 5 cases of atlanto-axial rotator fixation (Grisel syndrome) after infections. J Pediatr Pract 151:1171–1174 [in Japanese]

    Google Scholar 

  17. Uehara R, Belay ED (2012) Epidemiology of Kawasaki disease in Asia, Europe, and the United States. J Epidemiol 22:79–85

    Article  PubMed  Google Scholar 

  18. Wilson BC, Jarvis BL, Haydon RC 3rd (1987) Nontraumatic subluxation of the atlantoaxial joint: Grisel’s syndrome. Ann Otol Rhinol Laryngol 96:705–708

    PubMed  CAS  Google Scholar 

  19. Yanagawa H, Nakamura Y, Yashiro M, Uehara R, Oki I, Kayaba K (2006) Incidence of Kawasaki disease in Japan: the nationwide surveys of 1999–2002. Pediatr Int 48:356–361

    Article  PubMed  Google Scholar 

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All authors declare no conflict of interest.

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Correspondence to Fumihito Nozaki.

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Nozaki, F., Kusunoki, T., Tomoda, Y. et al. Grisel syndrome as a complication of Kawasaki disease: a case report and review of the literature. Eur J Pediatr 172, 119–121 (2013). https://doi.org/10.1007/s00431-012-1858-6

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  • DOI: https://doi.org/10.1007/s00431-012-1858-6

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