Tolosa-Hunt syndrome and comorbidity of obsessive compulsive disorder and aortic aneurysm.

To cite: Ceylan ME, Evrensel A, Noyan CO, et al. BMJ Case Rep Published online: [please include Day Month Year] doi:10.1136/ bcr-2016-216727 DESCRIPTION A 59-year-old man with obsessive compulsive disorder (OCD) was on antidepressant treatment for years. Six months ago, he got a severe headache on the left side of his head, reported pain behind his left eye, diplopia and ptosis displayed on his left eyelid. The patient had mild migraine attacks occasionally. However, his previous headache was very intense when compared with earlier ones. By cranial MRI, a lesion (22×7 mm horizontally, 13×8 mm vertically) adjacent to carotid segment 4 (C4) of the left internal carotid artery was found. The lesion showed a hyperintense signal in contrast-enhanced T2-weighted images, which was the result of enhanced abnormal soft tissue extending through the orbital fissure and into the orbital apex. This did not cause compression on the optic nerve and there was slight bulging into the left cavernous sinus (figure 1). The patient’s blood and cerebrospinal fluid biochemical parameters were within normal limits. An aneurysm was found in the ascending aorta with a width of 4.5 cm (normal width=2.5 cm) and followed-up for further risk of dissection. The patient was diagnosed with Tolosa-Hunt syndrome (THS), and he was started on prednisolone 64 mg/day. Periorbital pain, diplopia and left eyelid ptosis disappeared after 2 months of highdose cortisone treatment. THS is an episodic, painful ophthalmoplegia of unknown aetiology, involving ipsilateral paralysis of one or more of the third, fourth and sixth cranial nerves and the ophthalmic and/or maxillary branches of trigeminal nerve. There is inflammation in the cavernous sinus or the superior orbital fissure. Severe orbital and periorbital pain usually starts prior to paralysis of cranial nerves. Symptoms improve rapidly with corticosteroid treatment. THS is diagnosed according to the criteria defined by International Headache Society Classification, 2004. MRI findings are important for clarifying the diagnosis. The incidence of THS is estimated to be 1–2 cases per million people. Vascular malformations and deformities involving intracranial arteries have been reported in some cases of THS. Zhou et al reported a case of dissection aneurysm of the internal carotid artery; the suggested cause was inflammation related to THS. In another case, a mycotic abdominal aortic aneurysm was found to be associated with THS. None of the reported cases had comorbid psychopathology. In our patient, the aneurysm in the ascending aorta may have been caused by inflammation. Proinflammatory markers have been reported to be elevated in children and adolescents with OCD. For example, in patients with paediatric autoimmune neuropsychiatric disorders associated