BACKGROUND: The ovary is conspicuous for the wide diversity of histolo
gic tumor types that it contains. Unusual or rare tumors may present d
iagnostic difficulty on cytologic examination. CASE: A 36-year-old wom
an presented with an 80-mm cystic lesion in the left ovary. Fine needl
e aspiration provided 30 mL of transparent, light yellow fluid. On cyt
ologic examination, numerous clusters and rosettes of benign cells, re
sembling choroid plexus cells, were seen. Surgical resection confirmed
the presence of an ependymal cyst adjacent to a mature cystic teratom
a (dermoid cyst).CONCLUSION: We report this unique case in order to al
ert cytologists unfamiliar with cerebrospinal fluid cytopathology to t
he rare entity of ovarian ependymal cysts.