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LETTER TO EDITOR
Year : 2014  |  Volume : 5  |  Issue : 1  |  Page : 89-91

Ichthyosis uteri: An incidental finding with review of literature


Department of Pathology, Indira Gandhi Medical College, Shimla, Himachal Pradesh, India

Date of Web Publication15-Mar-2014

Correspondence Address:
Shailja Puri Wahal
Department of Pathology, Indira Gandhi Medical College, Shimla - 171 001, Himachal Pradesh
India
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/0975-9727.128968

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How to cite this article:
Wahal SP, Mardi K. Ichthyosis uteri: An incidental finding with review of literature. Muller J Med Sci Res 2014;5:89-91

How to cite this URL:
Wahal SP, Mardi K. Ichthyosis uteri: An incidental finding with review of literature. Muller J Med Sci Res [serial online] 2014 [cited 2019 Oct 16];5:89-91. Available from: http://www.mjmsr.net/text.asp?2014/5/1/89/128968

Dear Editor,

Ichthyosis uteri is an uncommon condition in which the entire surface of the endometrium is replaced by stratified squamous epithelium. [1] It is considered a benign lesion, but its association with malignancy has been reported in the literature. We report an incidental finding of squamous metaplasia of the endometrium in a uterus removed for prolapse.

A 48-year-old female P 5 + 0 presented with chief complaints of something coming out of the introitus for 1 year. On examination, there was a third degree descent. A hysterectomy with bilateral salpingo-oophorectomy was performed and sent for histopathological examination. Grossly, a hysterectomy specimen measuring 7.5 cm × 6 cm × 3 cm was received. Endometrium measured 1 mm and myometrium measured 2 cm. The cervical lips were everted, hypertrophied and keratinized. Bilateral tubes and ovaries were within normal limits. Microscopic examination of the endometrium showed the endometrial lining replaced by stratified squamous epithelium [Figure 1]. The sub-epithelial tissue showed atrophic endometrial glands and the normal endometrial stroma. The stratified squamous epithelial lining was benign and showed no features of dysplasia. The cervix was lined by keratinized hyperplastic stratified squamous epithelium with flattened rete ridges and chronic inflammatory cell infiltrate in the stroma. The  Fallopian tube More Detailss and ovaries showed normal histology. A report of endometrium-ichthyosis uteri was signed out.
Figure 1: Endometrium lined by stratified squamous epithelium, sub-epithelial tissue shows atrophic endometrial gland (H and E, ×40)

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The term ichthyosis uteri was first coined by Zeller in 1885 to describe the extensive keratinization of the endometrium seen following intrauterine application of caustic substances, like hot formalin. [2] Similar changes have been seen in association with tuberculous endometritis, puerperal endometritis, endometrial polyps, hyperplasias and pyometra secondary to cervical stenosis or malignancies. [3] A commoner finding is the presence of non-keratinizing squamous cells occurring either diffusely (adenoacanthosis) or in the form of berry-like aggregates (morules). [4] Most are seen in premenopausal women, in those receiving exogenous hormones, or in association with polycystic ovarian disease. [5] This change is distinguished from well-differentiated endometrial adenocarcinoma with squamous metaplasia (adenoacanthoma) because of the benign appearance of the glandular elements. [6] Ichthyosis uteri associated with dysplasia, in situ carcinoma and frank squamous cell carcinoma has been reported by Murheka et al. [7] in 2008, Bagga et al. [8] in 2008 and Takeuchi et al. [9] in 2012. Heckeroth and Ziegler [10] in their study have reported development of invasive cell carcinoma at the base of ichthyosis uteri. The carcinoma developed from discrete squamous metaplasia of the endometrial body in the presence of slight endometritis over a period of 3 years. In a population based study from Norway, the prevalence of primary squamous cell carcinoma is 0.1%. [11] To be accepted as a primary carcinoma of the endometrium, the tumor must satisfy the criteria established by Fluhmann and modified by Kay: [12] there must be no coexisting endometrial adenocarcinoma; there must be no connection between endometrial tumor and squamous epithelium of cervix; there must be no primary squamous carcinoma of the cervix; and if cervix shows an in situ carcinoma, there must be no connection between this and independent endometrial neoplasm.

The etiology of endometrial keratinization is not well understood. Chronic trauma, repair, irritation, inflammation, foreign material and estrogenic effects have all been implicated. No such predisposing factors could be found in our case. If widespread squamous epithelium covering the entire endometrial surface is detected in the curettage or biopsy an advanced examination should be done because of the possibility of the underlying malignancy association.

 
  References Top

1.Robert JK. Endometrial carcinoma. Blaustein's Pathology of Female Genital Tract. 5 th ed. New York: Springer; 2002. p. 542-3.  Back to cited text no. 1
    
2.Patton WT, Squires GV. Icthyosis uteri. A case report. Am J Obstet Gynecol 1962;84:858-60.  Back to cited text no. 2
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3.Hendrickson MR, Kempson RL. Surgical pathology for the uterine corpus. Major Problems in Pathology. Philadelphia, Pa: WB Saunders; 1980. p. 170-1.  Back to cited text no. 3
    
4.Dutra FR. Intraglandular morules of the endometrium. Am J Clin Pathol 1959;31:60-5.  Back to cited text no. 4
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5.Miranda MC, Mazur MT. Endometrial squamous metaplasia. An unusual response to progestin therapy of hyperplasia. Arch Pathol Lab Med 1995;119:458-60.  Back to cited text no. 5
    
6.Hendrickson MR, Kempson RL. Surgical pathology of the uterine corpus. In: Bennington JL, editor. Major Problems in Pathology. Vol. 12. Philadelphia: WB Saunders; 1980.  Back to cited text no. 6
    
7.Murhekar K, Majhi U, Sridevi V, Rajkumar T. Does "ichthyosis uteri" have malignant potential? A case report of squamous cell carcinoma of endometrium associated with extensive ichthyosis uteri. Diagn Pathol 2008;3:4.  Back to cited text no. 7
    
8.Bagga PK, Jaswal TS, Datta U, Mahajan NC. Primary endometrial squamous cell carcinoma with extensive squamous metaplasia and dysplasia. Indian J Pathol Microbiol 2008;51:267-8.  Back to cited text no. 8
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9.Takeuchi K, Tsujino T, Yabuta M, Kitazawa S. A case of primary squamous cell carcinoma of the endometrium associated with extensive "ichthyosis uteri". Eur J Gynaecol Oncol 2012;33:552-4.  Back to cited text no. 9
    
10.Heckeroth V, Ziegler HK. Ichthyosis uteri. Case report on a rare clinical entity. Geburtshilfe Frauenheilkd 1986;46:248-9.  Back to cited text no. 10
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11.Abeler VM, Kjørstad KE, Berle E. Carcinoma of the endometrium in Norway: A histopathological and prognostic survey of a total population. Int J Gynecol Cancer 1992;2:9-22.  Back to cited text no. 11
    
12.Kay S. Squamous-cell carcinoma of the endometrium. Am J Clin Pathol 1974;61:264-9.  Back to cited text no. 12
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