Steroid-resistant Tolosa-Hunt syndrome with a de novo intracavernous aneurysm: A case report

Takasuna, Hiroshi; Sasaki, Rie; Shiraishi, Makoto; Doi, Masatomo; Wakui, Daisuke; Ito, Hidemichi; Oshio, Kotaro; Tanaka, Yuichiro

doi:10.4103/2152-7806.193925

Cited by 11 publications

(5 citation statements)

References 29 publications

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“…This case illustrates the difficulty in diagnosing a rare disease in a patient population which is rarely affected. Our patient's dramatic pain relief after steroid therapy was consistent with the diagnosis of THS [5]. In nearly half of patients however, long-term steroid treatment may be required to relieve recurrent symptoms.…”

Section: Discussionsupporting

confidence: 75%

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Tolosa-Hunt Syndrome, the Hunt for One in a Million

Rizvi¹,

Filatov²,

Swerdloff³

2020

Cureus

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Tolosa-Hunt Syndrome (THS) is a rare disorder characterized by severe unilateral periorbital headache, painful eye movements, and double vision. This syndrome affects males and females equally in their forties to fifties. THS is caused by granulomatous inflammation of the cavernous sinus, usually involving CN III, IV, and VI. We herein report a case of Tolosa-Hunt Syndrome in an 18-year-old female who was initially misdiagnosed as idiopathic intracranial hypertension. Initial treatment was ineffective and the delay may have allowed spread of the disease to involve CN V and VII. This case highlights the pitfalls which can lead to a delay in diagnosis and treatment.

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Section: Discussionsupporting

confidence: 75%

“…The differential diagnosis includes herpes zoster infection, bacterial sinusitis, or fungal infection [5]. Initially, our patient was diagnosed with idiopathic intracranial hypertension (IIH).…”

Section: Discussionmentioning

confidence: 99%

Tolosa-Hunt Syndrome, the Hunt for One in a Million

Rizvi¹,

Filatov²,

Swerdloff³

2020

Cureus

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“…The more ominous abnormalities included ICA aneurysm and dissection. 29 30 31 32 Out of 54 cases of benign THS [Supplementary Tables 2 and 3 ], four showed vascular anomalies, 33 34 35 36 two of which were late-onset dural arteriovenous (AV) fistulas.…”

Section: R Esultsmentioning

confidence: 99%

Tolosa–Hunt Syndrome

Dutta

Anand

2021

Journal of Current Ophthalmology

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Purpose: To review the diagnostic criteria for Tolosa–Hunt syndrome (THS) and utility of recent modifications. Methods: We searched PubMed for keywords Tolosa Hunt and magnetic resonance imaging. We compared the three editions of International Classification of Headache Disorders and isolated case reports and case series with the assessment of cavernous internal carotid artery (ICA) caliber to find the prevalence of vascular anomalies. We also evaluated cases of THS with the involvement of extracavernous structures and the possible role of idiopathic hypertrophic pachymeningitis (HP). Cases diagnosed falsely as THS were also reviewed for the presence of atypical features and relevance of criterion D. We assessed nonconforming cases (those with normal neuroimaging benign THS) and idiopathic inflammatory orbital pseudotumor (IIPO). Results: Vascular abnormalities were found in 36.36% of THS cases. Benign THS may also show changes in ICA caliber. Evidence suggestive of idiopathic HP could be found in 57% of cases with the involvement of extracavernous structures, such as facial nerve and pituitary gland. Both THS and IIPO are steroid-responsive pathologies with similar clinical and radiological features. False-positive diagnosis of THS results from early labeling, based solely on clinical features and symptom resolution after steroid therapy. Conclusions: Benign THS may be a result of limitation of resolution of available neuroimaging technique or early testing. Early and late vascular changes can be seen in both THS and its benign variant; some of them are not innocuous. THS may be considered a type of focal idiopathic HP. IIPO may represent an anterior variant of THS. In the absence of histopathological diagnosis, steroid-induced resolution of symptoms should be confirmed radiologically and followed-up.

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“…To date, only three reports have described the presence of an aneurysm in association with THS. [15][16][17] From a total of four reported aneurysms, a decrease in aneurysm size was observed in two following high-dose steroid administration. 15,16 In Kambe and colleagues' report of a patient with THS and a concurrent bilateral aneurysm, the aneurysm on one side decreased in size following treatment with steroids, while the contralateral side required endovascular intervention; Takasuna and colleagues furthermore reported an increase in aneurysm size following steroid administration 15,17 Though the mechanism underpinning aneurysm formation is unknown, we hypothesize that the perivascular inflammatory state led to the vessel wall integrity being compromised, perhaps owing to local matrix metalloproteinase activity.…”

Section: Discussionmentioning

confidence: 99%

Angiographic Findings in the Tolosa–Hunt Syndrome and Resolution after Corticosteroid Treatment

Ravindran

Schmalz

Torun

et al. 2017

Neuro-Ophthalmology

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The Tolosa-Hunt syndrome is a rare clinical condition characterized by painful opthalmoparesis associated with idiopathic granulomatous inflammation of the orbital apex and cavernous sinus. Historically, this condition was thought to result from arteritic changes in the internal carotid artery and cavernous sinus. Modern digital angiographic techniques were unavailable when THS was initially described, and few reports exist on its high-resolution angiographic findings. Painful ophthalmoparesis, especially of the oculomotor nerve, warrants vascular imaging because of the concern for an underlying aneurysm. Here, we describe angiographic findings of THS which may be useful for clinicians when encountering patients presenting with painful ophthalmoplegia.

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Steroid-resistant Tolosa-Hunt syndrome with a de novo intracavernous aneurysm: A case report

Cited by 11 publications

References 29 publications

Tolosa-Hunt Syndrome, the Hunt for One in a Million

Tolosa-Hunt Syndrome, the Hunt for One in a Million

Tolosa–Hunt Syndrome

Angiographic Findings in the Tolosa–Hunt Syndrome and Resolution after Corticosteroid Treatment

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