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Purpose: To report on a case of diffuse unilateral subacute neuroretinitis (DUSN) that was successfully treated with a short course of oral albendazole. Observations: A 51-year-old male presented with severe visual loss secondary to DUSN associated with a positive Toxocara serology. Because the parasite could not be detected on fundoscopy, first-line treatment with photocoagulation could not be administered. Treatment with a 6-day course of oral albendazole resulted in the resolution of DUSN as well as the restoration of visual acuity. Conclusion and importance: Although DUSN is characterized by the presence of a parasitic organism in the retina, there are cases in which the parasite is not visible. Albendazole has been used to treat such cases, but a standard treatment regimen has not been determined yet. Our case suggests that the resolution of DUSN can be achieved with short-term albendazole therapy.

Purpose: Diffuse unilateral subacute neuroretinitis (DUSN) is often a challenging diagnosis to make. We present a DUSN case with its multimodal imaging to aid in the diagnosis, emphasizing the observations on optical coherence tomography angiography (OCTA). Observations: The evolution of a DUSN case is presented. Fundus photography and OCTA aided in the identification of the nematode. Conclusions and importance: DUSN is a difficult diagnosis to establish. We report the first case to our knowledge in which OCTA aided in the diagnosis of DUSN.

Diffuse unilateral subacute neuroretinitis is an ocular infectious disease caused by several distinct nematodes. Definite identification of the involved nematodes is rarely achieved. We report on the molecular-based genetic identification of an Ancylostoma ceylanicum hookworm implicated in a case of diffuse unilateral subacute neuroretinitis in a child.