1. Background and Importance
Tolosa Hunt syndrome (THS) is a rare syndrome characterized by eye pain. It occurs in one to two people per million people [
1]. Diagnostic criteria include ophthalmodynia, paresis of the cranial nerves for days to weeks, and spontaneous improvement. Ophthalmoplegic attacks can recur for months or years [
1,
2]. Cavernous sinus involvement can occur as granuloma [
3].
THS responds well to steroids, but there is no consensus on dosage and duration. A patient’s cranial nerve defect can remain permanent or recover for a long time [
2].
On magnetic resonance imaging (MRI), isointense lesions in the cavernous sinus are seen on T1-weighted images and isointense to hypointense lesions on T2-weighted images, which show increased enhancement after injection. To diagnose, MRI findings and patient history and eye pain that responds to steroids will be helpful [
1,
4].
2. Case Presentation
A 59-year-old man of Hispanic race with a history of diabetes was hospitalized for cervical laminectomy. Two weeks after surgery, surgical site infection (SSI) occurred, and the patient was treated with clindamycin 600 milligrams (mg) every 6 hours. Five days after hospitalization, he experienced pain in the right eye and the right side of the head without nausea and vomiting. Forty-eight hours after the pain started, the patient had ptosis of the right eye. The other cranial nerves were normal. Also, the pupils in both eyes responded to light. Forty-eight hours after ptosis, he developed right eye movement paralysis (frozen eyes with cranial nerve III, IV, and VI involvement). No problems were observed in biochemical, thyroid, and liver functional tests. After brain MRI, FLAIR hyperintensity in the right cavernous sinus was observed (
Figure 1).
Suspecting THS, the patient was treated with low-dose dexamethasone (4 mg every 8 hours) for 5 days. The patient’s eye pain partially improved 24 hours after receiving the first dose of dexamethasone. Two weeks after stopping dexamethasone, the patient’s eye pain recurred. Therefore, high-dose dexamethasone was started (8 mg every 6 hours) and continued for 10 days. At follow-up, the patient’s symptoms improved, and no new episodes occurred after one year. Only paresis of the right third cranial nerve remained.
3. Discussion
THS is a rare painful ophthalmological syndrome involving cranial nerves III, IV, VI, and V1 [
1,
5]. Hunt described six patients with recurrent unilateral painful ophthalmoplegia and suggested that non-specific cavernous sinus inflammation can cause THS [
6].
Because our patient was hospitalized for SSI, the syndrome was diagnosed earlier. Also, because THS is an inflammatory condition and our patient was admitted with SSI, it suggests that THS can be triggered by SSI inflammation. On MRI, FLAIR hyperintensity of the right cavernous sinus was observed. Other studies reported that the soft tissue of the supraorbital fissure area was markedly enlarged [
1,
5].
In the first treatment course with low-dose dexamethasone, the patient responded well to treatment, but his symptoms relapsed after two weeks. In the second course of treatment with high-dose dexamethasone, no recurrence of the syndrome occurred during the one-year follow-up. Herrera et al. treated a 17-year-old woman with one gram per day of methylprednisolone for 3 days, followed by 40 mg of prednisolone for 6 weeks [
5].
Recurrent THS over a short period may be due to the low dose of dexamethasone in the first course of treatment. In other studies, short-term recurrence has not been observed.
4. Conclusion
Comparing the findings of our study with those of other research studies revealed that, a higher corticosteroid dose and a longer treatment period can prevent short-term recurrence of the syndrome. In addition, THS can be triggered by various types of infections (SSI after surgery).
Ethical Considerations
Compliance with ethical guidelines
Written consent was obtained from all patients for participation.
Funding
This research did not receive any grant from funding agencies in the public, commercial, or non-profit sectors.
Authors' contributions
Conception and design: Abdolhadi Daneshi, Omid Masoudi, and Seyed Mohammad Reza Mohajeri; Data collection: Omid Masoudi, Seyed Mohammad Reza Mohajeri, and Hossein Ghazvini; Data analysis and interpretation: Omid Masoudi, and Seyed Mohammad Reza Mohajeri; Drafting the article: Omid Masoudi, Saina Darvishnia, and Seyed Mohammad Reza Mohajeri; Critically revising the article: Abdolhadi Daneshi; Reviewing submitted version of manuscript, and approving the final version of the manuscript: All authors.
Conflict of interest
The authors declared no conflict of interest.
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