Ichthyosis Uteri with High Grade Dysplasia and Microinvasion in a Case of Cervical Intraepithelial Neoplasia: A Rare Case Report with Review of Literature

Focal squamous metaplasia of endometrial gland is frequently associated with several inflammatory and neoplastic conditions. However, near complete replacement of endometrial lining with squamous cells (Ichthyosis uteri) is extremely rare. Dysplastic changes and invasive malignancy is rarely reported in Ichthyosis uteri.

A 69 year-old lady with multiple co-morbidities presented with postmenopausal bleeding and foul-smelling vaginal discharge. Clinical and imaging evaluation showed pyometra and cervical stenosis. Hysteroscopic biopsy showed cervical intraepithelial neoplasm with full-thickness dysplasia (CIN3) with focal suspicious invasion. Extrafascial hysterectomy and microscopic evaluation showed CIN3 of cervix and Ichthyosis uteri involving the entire endometrium with high grade dysplasia and focal invasion. Association with HPV infection was established by p16 immunohistochemistry.

Ichthyosis uteri is an extremely rare condition leading to metaplastic transformation of entire endometrium to squamous mucosa. Association with CIN3 is rarely reported. HPV infection in the metaplastic squamous lining can lead to malignant transformation. Knowledge about this distinct lesion and a close clinical and pathological association is important to prevent misdiagnosis.