Ichthyosis uteri associated with squamous cell carcinoma of the endometrium - a case report

Acta Chir Belg. 2023 Dec;123(6):679-681. doi: 10.1080/00015458.2022.2101332. Epub 2022 Jul 20.

Abstract

Background: Ichthyosis uteri is an extremely rare condition in which the entire or extensive parts of endometrial lining are replaced by stratified squamous epithelium. Malignant potential of this entity is unclear and its association with dysplastic changes and primary squamous cell carcinoma of the endometrium and endometrial adenocarcinoma has been reported. However, lack of data makes difficult to interpret the significance of neoplasms arising from this condition.

Patients and methods: We report a case of ichthyosis uteri associated with squamous cell carcinoma of the endometrium in a 62-year-old female who presented with postmenopausal bleeding and thin endometrium on ultrasound.

Results: Endometrial curettage was performed and revealed high grade squamous intraepithelial lesion. The patient underwent total laparoscopic hysterectomy with bilateral salpingo-oophorectomy and bilateral pelvic lymph node dissection. Microscopic examination of sections revealed squamous cell cancer along with extensive replacement of the endometrial lining by stratified squamous epithelium, consistent with ichthyosis uteri.

Conclusion: If ichthyosis uteri is suspected we recommend hysterectomy in order to rule out possibility of coexisting carcinoma. Also, thin endometrium in women with postmenopausal bleeding does not reliably exclude endometrial cancer.

Keywords: Ichthyosis uteri; case report; hysterectomy; malignant potential; postmenopausal bleeding.

Publication types

  • Case Reports

MeSH terms

  • Carcinoma, Squamous Cell* / diagnosis
  • Carcinoma, Squamous Cell* / surgery
  • Endometrial Neoplasms* / complications
  • Endometrial Neoplasms* / diagnosis
  • Endometrial Neoplasms* / surgery
  • Endometrium / pathology
  • Endometrium / surgery
  • Female
  • Humans
  • Ichthyosis* / complications
  • Ichthyosis* / diagnosis
  • Ichthyosis* / pathology
  • Middle Aged
  • Uterus / pathology