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Summary: Wound infection due to a preauricular sinus after craniotomy has not been previously reported. A 71-year-old woman visited our institute with subarachnoid hemorrhage. The aneurysm was surgically clipped with external decompression. Sixteen days after surgery, a focal erythema and discharging were observed at the inferior end of the skin incision. Careful inspection revealed a small pit there, which was diagnosed as a preauricular sinus. After the infection subsided, the sinus was completely excised. Neurosurgeons must be aware of this rare condition and must opt for complete excision to prevent infectious sequelae even if the preauricular sinus is asymptomatic.