| Summary: | Background: In this case, we reported the pseudobulbar affect (PBA) in a patient with Susac’s syndrome—a rare condition that was caused by a rare syndrome. Previous case reports of Susac syndrome described psychiatric symptoms such as emotional disturbances or personality changes. Only a few case reports have reported psychiatric disorders in patients with Susac’s syndrome. There were no reported cases of Susac syndrome with PBA as an initial presentation. Case presentation: Our patient was 56 years old and presented with involuntary crying, left-sided headache, left-sided hearing loss, and tinnitus. Brain MRI showed numerous areas of restricted diffusion and enhancement involving the corpus callosum, bilateral hemispheres, and brainstem. Ophthalmological evaluation showed bilateral branch retinal artery occlusion. She was diagnosed with Susac’s syndrome and PBA. She was treated with cyclophosphamide and dextromethorphan hydrobromide/quinidine sulfate with excellent recovery. This is a 2-year clinical course. Discussion and conclusions: Recognition of the clinical presentation of Susac’s syndrome and PBA with early diagnosis and treatment are the keys to preventing further disability and impact on patients and their families. © 2022, The Author(s).
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