PURPOSE: To describe the clinical and multimodal imaging characteristics of an atypical clinical and imaging presentation of ocular toxoplasmosis. METHODS: serology, and exclusion of alternative infectious etiologies. Intraocular PCR and Goldmann-Witmer coefficient analysis were not available. Clinical presentation, multimodal imaging findings, treatments and outcomes were reviewed. RESULTS: Six eyes of five patients were included. All eyes demonstrated a consistent inner foveal SD-OCT imaging pattern characterized by focal inner retinal cavitation, ellipsoid zone disruption, and outer nuclear layer involvement, with variable but generally limited intraocular inflammation. Two eyes (Case 1 OD and Case 3) presented with predominantly macular involvement without adjacent active retinochoroiditis. Following treatment with trimethoprim-sulfamethoxazole and systemic corticosteroids, all eyes showed anatomical improvement, although residual inner retinal thinning persisted in several eyes. CONCLUSIONS: This case series described an atypical clinical and imaging presentation of ocular toxoplasmosis characterized by a distinctive inner foveal SD-OCT imaging pattern (acute inner foveal toxoplasmic maculopathy). Although the absence of intraocular microbiological confirmation limits diagnostic certainty, recognition of this presentation may facilitate earlier diagnosis and appropriate treatment in selected patients. The term "acute" refers to the active ocular presentation and does not imply recently acquired systemic infection. Further studies are warranted to validate these findings and clarify their underlying pathophysiology.
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