Tolosa-Hunt syndrome relapse during pregnancy following chronic intake of combined oral contraceptives

Abstract

Tolosa-Hunt syndrome (THS) is a rare syndrome of painful ophthalmoplegia secondary to an idiopathic granulomatous inflammation affecting the cavernous sinus, superior orbital fissure or orbit. Pregnancy and pregnancy-related hormones have been identified as potential triggers. A 39-year-old gravida-2 para-1 woman with prior chronic intake of combined oral contraceptives (COC) suffered two episodes of painful ophthalmoplegia—the first event with spontaneous remission and the relapse occurring during pregnancy and with complete resolution following steroid treatment. MRI revealed a postinflammatory mass at the junction of the left orbital apex and anterior cavernous sinus, supporting the diagnosis of THS. To our knowledge, this is the first report of a THS relapse occurring during pregnancy following a chronic history of COC intake. This case adds to the growing evidence supporting the relationship between immune and hormonal factors that may be present during pregnancy and the disease pathogenesis of THS.

Frozen overnight: Acute orbital apex syndrome caused by aspergillosis

“…In fact, THS, a rare syndrome of painful ophthalmoplegia secondary to an idiopathic granulomatous inflammation affecting the cavernous sinus, requests steroid treatment and should thus be differentiated by an infectious process involving the orbital apex. 2 In fact, THS is not a diagnosis, but a cluster of symptoms, which may result from a number of pathological conditions. 3 In the Authors’ opinion, THS should be a diagnosis of exclusion, since systemic steroid given without appropriate evidence can even be harmful in case of infectious etiology.…”

Complete ophthalmoplegia diagnosed as Tolosa-Hunt syndrome on interval MRI

“…The causative inflammation is often idiopathic but can be traumatic, neoplastic or aneurysmal in aetiology. Though Tolosa-Hunt syndrome is inflammatory and has been reported in conjunction with SLE, other autoimmune diseases, trauma and pregnancy, it is not associated with systemic inflammation 7…”