Purpose To describe the clinical and epidemiologic characteristics of sporotrichosis in ocular adnexa and give an insight into factors associated with this condition. Design Retrospective case series and literature review. Methods We retrospectively reviewed all cases of sporotrichosis in ocular adnexa between 2004 and 2014 in the Santa Teresa Clinic of Abancay, Peru and reviewed all case reports of sporotrichosis in ocular adnexa in the literature. Results We reviewed records of 21 patients with sporotrichosis in ocular adnexa; 12 (57.1%) of them were male and their median age surrounded 9 years. In our series, 19 patients had lesions in the eyelids and 2 in the eyebrows. The lymphocutaneous form occurred in 62% of them. Ten patients (47.6%) were cured with potassium iodide. Among 65 patients with sporotrichosis in ocular adnexa (our 21 patients and 44 from the literature), the average age was 9 years, and 78% were ≤15 years of age; 54% were male. The lesions were more frequent on the eyelids (n = 53 [82%]), followed by the lacrimal gland (n = 5), conjunctiva (n = 4), and eyebrows (n = 3). The lymphocutaneous clinical form (54%) was the most frequent. Fifty patients were cured: 31 of them with potassium iodide, 16 with itraconazole, and 3 with a combination including potassium iodide, itraconazole, and fluconazole. Twenty-nine patients (44.6%) resided in a hyperendemic region, and 5 patients reported contact with cats that had sporotrichosis. Conclusions Sporotrichosis in ocular adnexa is associated with children ≤15 years of age, and in 82% of these cases the disease is limited to the eyelids.