A Case Report of Ichthyosis Uteri


 Background: Ichthyosis uteri is a rare condition in which the entire surface of the endometrium is replaced by squamous epithelium. The condition often is considered as benign, despite anaplastic and dysplastic changes have been reported. Case presentation: A 36-year-old female present with extensive squamous metaplasia and dispersed normal endometrium. The case displayed with a benign ichthyosis uteri showing heavy keratinization without dysplasia. The cervix showed no dysplastic changes without HPV infection. Conclusion: We conclude that a benign ichthyosis uteri was not associated with HPV infection.


Background
Ichthyosis uteri is an exceedingly rare condition in which the entire surface of the endometrium is replaced by strati ed squamous epithelium. It was initially reported in 1885 by Zeller. Originally described as an endometrial response to iatrogenically-introduced caustic substances such as formalin or iodine, similar changes have since been described in association with a variety of in ammatory conditions of the endometrium.
Progesterone is a steroids released by ovary. As a sex hormone, it is applied to prevent miscarriage, emergency contraception et al in clinical practice. We describe herein an undescribed example of an ichthyosis uteri following after oral administration of progesterone.

Case Presentation
A 36-year-old female presented with complaints of massive vaginal bleeding for ten days. Her past medical history was insigni cant with no history of tuberculosis, in ammatory conditions of the uterus or iatrogenically-introduced substances in uterus.
Ultrasound abdomen (Fig. 1) showed endometrial thickness of 1.2 cm. Hysteroscopy examination showed uterus were signi cantly enlarged like a barrel, with endometrial cavity full lled with friable, greywhite mass. Cervical biopsy displayed strati ed squamous epithelium without dysplasia and HPV infection. Endometrial curetting was grey-white and measured 3.5x3x1cm in size. Curettages were totally sampled and revealed strati ed squamous epithelium showing heavy keratinization and no dysplastic changes ( Fig. 2A, 2B). Extensive squamous metaplasia of endometrium and dispersed normal endometrium were seen ( Fig. 2C) according to full sampling of endometrium and careful observation.
She was without any complications follow-up 9 months after her curettage.

Discussion & Conclusion
Zeller coined "ichthyosis uteri" in 1885 to describe extensive squamous metaplasia of the surface endometrium following iatrogenically introduced caustic substances such as formalin or iodine(1). The condition is exceedingly rare with few cases reported in the literature. Malignancies have rarely been associated with ichthyosis uteri. The case in the literature that most closely resembles our case was reported by Kucukali in 1996(2). The case described in that report underwent a benign papilloma with a background of severe ichthyosis uteri. Microscopic evaluation showed simulating koilocytosis in some papilloma cells without sign of dysplasia or malignancy. Human papillomavirus was not detected immunohistochemically.
Although ichthyosis is considered as a benign condition, anaplastic and dysplastic changes have been reported (3)(4)(5). These cases were associated with dysplasia or carcinoma of cervix. After discussion for direct extension from cervical for ndings in endometrium. They conclude that squamous cell carcinoma of endometrium was either developed directly from(6) or in association with pre-existing ichthyosis uteri, and ichthyosis may associate with human papillomavirus(3).
A case of endometrial adenocarcinoma covered almost by a plaque-like, keratinizing mature squamous epithelium was described by Bewtra(7). Therefore, ichthyosis uteri could be diagnosed after extensive observation to exclude endometrial adenocarcinoma.
In summary, a case of a benign ichthyosis uteri without HPV infection is described. The possibility of squamous cell carcinoma should be considered after extensive examination (such as CEA in blood), especially when plaques of squamous epithelium with dysplastic changes are identi ed in an endometrial biopsy or curettage.  Figure 1 Ultrasound abdomen showed endometrial thickness of 1.2 cm.

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