Evidence map›Paper›PMID 40418416›Full record

ReviewNeurological sciences : official journal of the Italian Neurological Society and of the Italian Society of Clinical Neurophysiology2025

Rare association of Tolosa-Hunt Syndrome with Sjogren's Syndrome: case report and review of immunological links across autoimmune disorders".

Hosna Elshony, Rakan Almuhanna, Rabia Muddassir

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Review in Neurological sciences : official journal of the Italian Neurological Society and of the Italian Society of Clinical Neurophysiology, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

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4 · The record

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5 · Who and what money

Authors and funding

3 authors.

Hosna ElshonyDepartment of Neuropsychiatry, Faculty of Medicine, Menoufiya University, Shebin elkom, Egypt. hosna.saad28@gmail.com.ORCID http://orcid.org/0000-0001-7078-4697
Rakan AlmuhannaDepartment of Internal Medicine, Neurology Unit, Security Forces Hospital Makkah, Makkah, Kingdom of Saudi Arabia.
Rabia MuddassirDepartment of Internal Medicine, Neurology Unit, Security Forces Hospital Makkah, Makkah, Kingdom of Saudi Arabia.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

backgroundTolosa-Hunt Syndrome (THS) is a rare inflammatory condition characterized by painful ophthalmoplegia, often associated with granulomatous inflammation in the cavernous sinus. While typically idiopathic, THS can sometimes be linked to underlying autoimmune diseases. This report presents the first documented case of THS as the initial manifestation of Sjögren's Syndrome (SS). CASE PRESENTATION: A 60-year-old woman presented with symptoms of left eye pain, diplopia, and ptosis, leading to a diagnosis of THS after extensive workup and exclusion of other potential causes. MRI findings indicated inflammatory changes in the cavernous sinus, and further autoimmune testing revealed positive ANA and anti-SSA (Ro), leading to a diagnosis of Sjogren's syndrome confirmed by lip biopsy. Following corticosteroid treatment, she experienced prompt symptom relief and was subsequently placed on immunosuppressive therapy for ongoing management. METHODS AND

resultsA literature review of 15 autoimmune-associated THS cases was conducted, revealing that 87% of patients presented with classic symptoms of periorbital pain, ptosis, and cranial nerve palsies, while 73% had cavernous sinus involvement on MRI. Most cases responded well to corticosteroids, with 60% requiring additional immunosuppressive treatment to manage ongoing autoimmune activity.

conclusionThis case highlights the importance of considering autoimmune etiologies in THS, especially in patients with limited response to initial therapies. Pathophysiological links may include immune dysregulation, granulomatous inflammation, and vascular compromise within the cavernous sinus. Early recognition and appropriate management are crucial for optimal outcomes. Further research into the immunological mechanisms linking THS and autoimmune diseases could improve diagnostic accuracy and therapeutic approaches.

Indexed as

Sjogren's SyndromeTolosa-Hunt SyndromeFemaleHumansMagnetic Resonance ImagingMiddle AgedAutoimmune diseaseGranulomatous inflammationNeuroinflammationSjogren’s SyndromeTolosa-Hunt Syndrome

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