A 34-year-old man ingested more then 250 g of amodiaquine hydrochloride (for pain) during one year and was noted to have diffuse conjunctival, corneal, and skin changes and also abnormal results from retinal function tests. Ultrastructural examination of corneal and conjunctival biopsy specimens revealed intralysosomal membranous and amorphous inclusions in corneal epithelial cells, conjunctival epithelial cells, keratocytes, and vascular endothelial cells. Follow-up of more than two years and repeated conjunctival biopsy specimens revealed diminution in both the clinical and electron microscopic abnormalities, but persistence in abnormal results of retinal function tests. These changes are consistent with a drug-induced phospholipidosis.