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Purpose To report two unusual cases of lipid keratopathy in siblings with rheumatic fever and positive anti-phospholipid antibodies.Methods Clinical and histopathological examination along with complete systemic work-up was done after taking appropriate consent from the patients. A serum lipid profile showed elevated cholesterol and low-density lipoproteins. Systemic history revealed both the siblings were on treatment for rheumatic fever. A complete blood profile showed positive anti-phospholipid antibodies in one of them.Results Penetrating keratoplasty was performed in both cases one year apart. Histopathological examination showed lipid filled macrophages distributed throughout the corneal stroma. No episode of recurrence was noted in the follow-up period of three years.Conclusion These two cases highlight the importance of a thorough family history and a full systemic work-up in a patient with bilateral symmetrical lipid keratopathy. The co-existence of rheumatic fever and anti-phospholipid syndrome has been studied in literature but, their presence along with lipid keratopathy has not been previously described. It requires further research whether they have an association or it was just an incidental finding.