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Aim: Polythelia is a rare condition. It ıs known to be associated with urogenital anomalies. Also, there are other types of rare anomalies like hand or mandible anomalies related with this anomaly. However, our case presents the first case of polythelia with ophthalmic abnormalities.
Presentation of Case: A 22-year-old male with congenital bilateral nodular inframammarian macules with aesthetic complaints and high-grade myopia. Lesions were excised and concluded as polythelia. Ultrasonic examinations revealed no structural abnormalities in urogenital system.
Discussion: Polythelia is a rare condition with several accompanying abnormalities. Mostly, it is associated with urogenital anomalies. Our case showed no signs of urogenital deformities. Only deformity he had was high-grade myopia which began at infancy.
Conclusion: Our report is the only article that reports the possible association of polythelia with ophthalmic deformities.