To report clinical features and optical coherence tomographic findings of presumed atypical ocular tuberculosis associated with tuberculosis lymphadenitis and encephalomeningitis.

A 28-year-old female with lymphadenitis in the axillary area presented with a fever and headache of a one week duration. CSF study and MRI findings implied tuberculosis encephalomeningitis, and presumed tuberculosis uveitis manifested with visual disturbance after five days. Ocular symptoms were aggravated and showed anterior iridocyclitis, vitritis, macular edema, and multifocal retinitis with miliary granuloma that was distinct from choroiditis or typical tuberculosis granuloma. After the patient received anti-tuberculosis medication and systemic corticosteroids, significant improvements in visual acuity, ocular findings and OCT results were observed.

Ocular tuberculosis can present with various clinical findings, and caution should be taken so as not to misdiagnose based on these characteristics. In the present case, anti-tuberculosis medication and systemic steroids resulted in the resolution of inflammation. In such cases, monitoring the posterior pole lesion via OCT may be helpful in determining improvement.