Proboscis lateralis is a very rare congenital craniofacial malformation characterized by a finger‐like tubular appendage arising usually from the medial canthal region. It is mostly unilateral and associated with other craniofacial malformations. Occasionally, proboscis lateralis is seen with holoprosencephaly. A rare case of bilateral proboscis lateralis which was diagnosed prenatally by ultrasound and magnetic resonance imaging has been presented. In this case of bilateral proboscis lateralis, both lesions arose from a very lateral location and were associated with various central nervous system anomalies other than holoprosencephaly.
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