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Metastatic disease is the most frequent intraocular malignancy in adults. The most common sites of origin are breast and lung. The vast majority of metastatic intraocular tumours are carcinomas, and rarely neuroendocrine tumours and sarcomas. Gastrointestinal stromal tumours (GIST) constitute a distinct group of gastrointestinal tumours arising from the interstitial cells of Cajal, regulators of peristalsis. The development of imatinib mesylate, a receptor tyrosine kinase inhibitor, has made a major impact on the management of advanced GISTs. We report the case of a patient with choroidal and systemic disease from a GIST, who underwent therapy with imatinib. An accurate diagnosis is required since therapy may differ from the most common metastatic carcinomas.