Tolosa-Hunt Syndrome in Childhood and Adolescence: A Literature Review in the Last 10 Years
Objectives The aim of the study is to review the articles published in the last 10 years on Tolosa-Hunt syndrome (THS) in childhood and adolescence to learn about its clinical and epidemiological characteristics and its therapeutic management.
Methods Based on the literature search in the major medical databases and using the descriptors “Tolosa-Hunt syndrome and children” and “Tolosa-Hunt syndrome and adolescent” we included articles published between 2009 and 2019. We found 51 articles, but only 11 met the inclusion criteria.
Results There were eight women and four men with THS, whose average age was 12.0 ± 4.3 years, ranging from 4 to 17 years. Magnetic resonance imaging showed inflammation in the cavernous sinus (100%), orbit (25%), and superior orbital fissure (8.3%), unilateral in all patients. The internal carotid artery (58.3%), third (66.7%), sixth (33.3%), and fourth (25%) cranial nerves were injured. Headache was located in the orbital region, ipsilaterally to nerve involvement and had mild to severe intensity. Corticosteroid was the treatment used in 91.7% of the patients. They were followed for an average of 13.4 ± 12.4 months. Only two patients had recurrence of the disease.
Conclusion THS is infrequent in childhood and adolescence and knowledge of its clinical and epidemiological characteristics and its therapeutic management is necessary for pediatricians.
Received: 30 March 2020
Accepted: 20 May 2020
06 September 2020 (online)
© 2020. Thieme. All rights reserved.
Georg Thieme Verlag KG
Rüdigerstraße 14, 70469 Stuttgart, Germany
- 1 Hung CH, Chang KH, Wu YM. et al. A comparison of benign and inflammatory manifestations of Tolosa-Hunt syndrome. Cephalalgia 2013; 33 (10) 842-852
- 2 Mullen E, Green M, Hersh E, Iloreta AM, Bederson J, Shrivastava R. Tolosa-Hunt syndrome: appraising the ICHD-3 beta diagnostic criteria. Cephalalgia 2018; 38 (10) 1696-1700
- 3 Bhatkar S, Goyal MK, Takkar A. et al. Cavernous sinus syndrome: a prospective study of 73 cases at a tertiary care centre in Northern India. Clin Neurol Neurosurg 2017; 155: 63-69
- 4 Serralta SMG, Torrecillas ND, Soler RL, Ibáñez SL, Gómez CJ. Tolosa-Hunt syndrome [in Spanish]. Rev Clin Esp 2013; 213 (02) e11-12
- 5 Zanus C, Furlan C, Costa P, Cosentini D, Carrozzi M. The Tolosa-Hunt syndrome in children: a case report. Cephalalgia 2009; 29 (11) 1232-1237
- 6 Pienczk-Reclawowicz K, Pilarska E, Lemka M, Konieczna S. Paediatric Tolosa-Hunt syndrome: the need for treatment guidelines and renewed criteria. Dev Med Child Neurol 2010; 52 (09) 873-874
- 7 Benzohra DE, Damry N, Delpierre I, Huybrechts S, Monier A, Christophe C. Tolosa-Hunt syndrome in children. JBR-BTR 2011; 94 (05) 290-292
- 8 Cerisola A, González G, Scavone C. Tolosa-Hunt syndrome preceded by facial palsy in a child. Pediatr Neurol 2011; 44 (01) 61-64
- 9 Cornish KS, Cornish DS, Vella M. An unusual case of pediatric painless ophthalmoplegia. Semin Ophthalmol 2011; 26 (02) 47-49
- 10 Kang CH, Huang YC, Lui CC, Chang YC, Hung PL. Unusual clinical course in pediatric Tolosa-Hunt syndrome. Pediatr Neurol 2011; 44 (06) 471-474
- 11 Slattery E, White AJ, Gauthier M, Linscott L, Hirose K. Tolosa-Hunt syndrome masquerading as Gradenigo syndrome in a teenager. Int J Pediatr Otorhinolaryngol 2013; 77 (07) 1219-1221
- 12 Maji B, Das D. Tolosa-Hunt syndrome. Sri Lanka J Child Heal 2014; 43 (04) 244-245
- 13 Pérez CA, Evangelista M. Evaluation and management of Tolosa-Hunt Syndrome in children: a clinical update. Pediatr Neurol 2016; 62: 18-26
- 14 Rodriguez-Homs LG, Goerlitz-Jessen M, Das SUA. 17-year-old girl with unilateral headache and double vision. J Investig Med High Impact Case Rep 2019; 7: 2324709619838309
- 15 Tsirigotaki M, Ntoulios G, Lioumpas M, Voutoufianakis S, Vorgia P. Tolosa-Hunt Syndrome: clinical manifestations in children. Pediatr Neurol 2019; 99: 60-63
- 16 La Mantia L, Curone M, Rapoport AM, Bussone G. International Headache Society. Tolosa-Hunt syndrome: critical literature review based on IHS 2004 criteria. Cephalalgia 2006; 26 (07) 772-781
- 17 Headache Classification Subcommittee of the International Headache Society. The International Classification of Headache Disorders. 3rd ed. Cephalalgia. 2018; 38 (01) 1-211
- 18 Anagnostou E, Kouzi I, Kararizou E. Painful ophthalmoplegia: the role of imaging and steroid response in the acute and subacute setting. J Neurol Sci 2013; 331 (1-2): 145-149
- 19 Tolosa E. Periarteritic lesions of the carotid siphon with the clinical features of a carotid infraclinoidal aneurysm. J Neurol Neurosurg Psychiatry 1954; 17 (04) 300-302
- 20 Hunt WE, Meagher JN, Lefever HE, Zeman W. Painful opthalmoplegia. Its relation to indolent inflammation of the carvernous sinus. Neurology 1961; 11: 56-62
- 21 Colnaghi S, Versino M, Marchioni E. et al. ICHD-II diagnostic criteria for Tolosa-Hunt syndrome in idiopathic inflammatory syndromes of the orbit and/or the cavernous sinus. Cephalalgia 2008; 28 (06) 577-584
- 22 Yeung MC, Kwong KL, Wong YC, Wong SN. Paediatric Tolosa-Hunt syndrome. J Paediatr Child Health 2004; 40 (07) 410-413
- 23 Zurawski J, Akhondi H. Tolosa-Hunt syndrome—a rare cause of headache and ophthalmoplegia. Lancet 2013; 382 (9895): 912
- 24 Iaconetta G, Stella L, Esposito M, Cappabianca P. Tolosa-Hunt syndrome extending in the cerebello-pontine angle. Cephalalgia 2005; 25 (09) 746-750
- 25 Monzillo PH, Saab VM, Protti GG, Costa AR, Sanvito WL. Tolosa-Hunt syndrome: analysis of six cases. Arq Neuropsiquiatr 2005; 63 (3A): 648-651