Amrita Journal of Medicine

: 2022  |  Volume : 18  |  Issue : 2  |  Page : 70--71

Prognostic factors for recurrence of Tolosa Hunt syndrome

Jamir Pitton Rissardo, Ana Letícia Fornari Caprara 
 Department of Medicine, Federal University of Santa Maria, Santa Maria, Brazil

Correspondence Address:
Jamir Pitton Rissardo
Av. Roraima, 1000 - Camobi, Santa Maria, RS 97105–900

How to cite this article:
Rissardo JP, Fornari Caprara AL. Prognostic factors for recurrence of Tolosa Hunt syndrome.Amrita J Med 2022;18:70-71

How to cite this URL:
Rissardo JP, Fornari Caprara AL. Prognostic factors for recurrence of Tolosa Hunt syndrome. Amrita J Med [serial online] 2022 [cited 2022 Sep 27 ];18:70-71
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Dear Editor,

We read the article titled ‘‘A Case of Idiopathic Hypertrophic Cranial Pachymeningitis Masquerading as Tolosa–Hunt Syndrome in an Elderly Female’’ in the esteemed ‘‘Amrita Journal of Medicine’’ with great interest. Parate et al. report a 68-year-old female presenting with left hemicranial headache and drooping of the left eyelid after cataract surgery. Brain MRI was suggestive of dural thickening with a pachymeningeal enhancement along the left frontoparietal and bilateral basitemporal regions. High-dose corticosteroids were started, with significant improvement in her retroorbital pain and ocular movements.[1]

Tolosa Hunt syndrome (THS), also known as painful ophthalmoplegia, recurrent ophthalmoplegia, or ophthalmoplegia syndrome, is characterized by unilateral periorbital headaches with painful and restricted eye movements. It is associated with nonspecific granulomatous inflammation of the cavernous sinus, superior orbital fissure, and/or the apex of the orbit. Glucocorticoids are the mainstay of management. However, there are no specific studies regarding recommendations about its dose or duration. Herein, we would like to discuss recent articles published about the prognostic factors for THS relapse [Figure 1].{Figure 1}

In a Korean study, 11 individuals with a diagnosis of THS fulfilling the International Classification of Headache Disorders 3 diagnostic criteria were assessed. One patient with optic neuritis failed to recover visual acuity, and another relapsed after five weeks of the total recovery. Kim and Oh suggested that initial IV methylprednisolone at 1 g/day may have better outcomes in optic neuritis. They explained that this presentation, although rare, has elevated inflammatory markers and can extend throughout the cavernous sinus and orbital apex.[2]

Ata et al. conducted a retrospective cohort study with 31 individuals with THS, of whom 71% were males. The majority of the individuals were Asian. A previous history of THS and female gender was associated with recurrence. Relapsing attacks were seen in 66.7% fully recovered and 33.3% partially recovered cases. It is worthy mentioning that the male-dominant population of Qatar may be due to more male expatriates (19 million males compared with 7 million females).[3]

THS affects mostly middle-aged adults. In this way, the occurrence of THS during childhood and adolescence is rare and poorly described. In a recent review, Gama and Silva-Néto showed that females are more commonly affected by THS in this group of individuals. Brain MRI showed inflammation in the cavernous sinus in all the patients. Also, 16% (2/12) of the subjects had a recurrence of the disease. This fact is interesting, because THS in childhood/adolescence appears to have a lower recurrence rate than when occurs in the adult population. A possible explanation for this finding is that the patients were not followed for enough time to develop another inflammatory painful ophthalmoplegic attack.[4]

Arthur et al. aim at studying the recurrence and role of steroid-sparing agents in THS. A total of 44 cases were investigated. The recurrence occurred in 48.6% of the individuals within a mean of 18 months. The authors did not identify any clinical or radiological predictors for relapse rate. Apparently, individuals who received steroid-sparing agents had a significantly lower recurrence. It is noteworthy that this study showed a high relapsing rate, which can be explained by the long-term follow-up of 120 months.[5]

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1Parate T, Tony KS, Chavan S, Khatri A A case of idiopathic hypertrophic cranial pachymeningitis masquerading as Tolosa–Hunt syndrome in an elderly female. Amrita J Med 2021;17:58-62.
2Kim H, Oh SY The clinical features and outcomes of Tolosa-Hunt syndrome. BMC Ophthalmol 2021;21:237.
3Ata F, Yousaf Z, Arachchige SNM, Rose S, Alshurafa A, Muthanna B, et al. The demographics of Tolosa-Hunt syndrome in Qatar. Eneurologicalsci 2021;24:100359.
4Gama BP, Silva-Néto RP Tolosa-hunt syndrome in childhood and adolescence: A literature review in the last 10 years. Neuropediatrics 2021;52:1-5.
5Arthur A, Sivadasan A, Mannam P, Prabakhar AT, Aaron S, Mathew V, et al. Tolosa-hunt syndrome: Long-term outcome and role of steroid-sparing agents. Ann Indian Acad Neurol 2020;23:201-5.