There is a wide spectrum of cilial anomalies; however, ectopic cilia are the rarest of the cilial anomalies. We report, to the best of our knowledge, the third worldwide case of this eyelid anomaly in the inferior palpebral conjunctiva and the first with a long-term follow-up of 18 months without recurrence following excision. The 41-year-old Saudi male presented with posterior ectopic cilia in the palpebral conjunctiva of the left lower eyelid. This rare anomaly was not associated with other findings. Eighteen months after complete excision of the cilia (along with the adjacent conjunctiva and tarsal plate), there was no recurrence of the ectopic cilia. This case highlights the importance of considering ectopic cilia when patients present with focal punctate keratopathy and the importance of excision of the cilia along with the adjacent conjunctiva and tarsal plate to prevent recurrence.

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