Background: Ligneous change of the genital tract is a very rare condition and may result in infertility.
Case: A 27-year-old woman presented with primary infertility and ligneous changes of the genital tract. Cervical and endometrial samples revealed subepithelial deposition of dense, amorphous, eosinophilic material associated with intensive inflammatory cell infiltration. Plasminogen functional activity was 16%.
Conclusion: The clinician must be aware of the systemic nature of this disorder and the multisystem complications.