We describe the first two cases of diffuse unilateral subacute neuroretinitis (DUSN) in the UK successfully treated with albendazole. Two patients are described who presented with unilateral reduction in vision, with clusters of yellow-white retinal lesions fading and reappearing in new locations of the fundus over a period of weeks. Based on the clinical picture, the diagnosis of DUSN was made, even though no subretinal worm could be found. Both patients were treated with albendazole 400 mg daily for 30 days. After 4 weeks the vision in the affected eye improved to 6/9 in each patient and the retinal lesions resolved completely. Over a follow-up period of 28 months and 13 months respectively, there was no recurrence of any lesion and the vision was maintained. DUSN is a sight-threatening disease and the preservation of vision relies on early diagnosis and treatment. Laser photocoagulation of the worm is the treatment of choice, but this was not possible in either case, as no worm could be found; however, oral albendazole proved to be a safe and effective alternative treatment.