Midline congenital abnormalities of the skin may be related to subjacent visceral developmental defects. We report on three unrelated children presenting a small midline pit on the bridge of the nose from which a small tuft of hair was emerging. This presentation is the hallmark of a nasal dermoid sinus cyst. In one of the patients the sinus tract was connected to the anterior cerebral fossa and required neurosurgical management. Although midline cutaneous abnormalities may look trivial, dermatologists should keep a high index of suspicion for possible association with more severe underlying defect. The diagnosis of nasal dermoid sinus cyst requires appropriate imaging studies to determine the extension of the sinus tract thus allowing proper management.
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