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CASE REPORT  
Year : 2020  |  Volume : 63  |  Issue : 4  |  Page : 637-639
Ichthyosis uteri with dysplasia - A case report


Department of Pathology, Andaman and Nicobar Islands Institute of Medical Sciences, Port Blair, India

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Date of Submission19-Mar-2019
Date of Decision22-Oct-2019
Date of Acceptance04-Nov-2019
Date of Web Publication28-Oct-2020
 

   Abstract 


Ichthyosis uterus is an uncommon condition in which the entire surface of the endometrium is replaced by stratified squamous epithelium. This condition most commonly develops secondary to longstanding cervical obstruction or chronic inflammation. It is considered a benign lesion, but its association with endometrial malignancy has been reported in the literature. We are reporting a case of ichthyosis uteri with dysplasia associated with cervical intraepithelial neoplasm (CIN III). The case is reported for its rarity and to aware about neoplastic potential of ichthyosis uteri.

Keywords: Carcinoma cervix, CIN, dysplasia, endometrial carcinoma, Ichthyosis uteri, squamous metaplasia

How to cite this article:
Gargade CB, Deshpande AH. Ichthyosis uteri with dysplasia - A case report. Indian J Pathol Microbiol 2020;63:637-9

How to cite this URL:
Gargade CB, Deshpande AH. Ichthyosis uteri with dysplasia - A case report. Indian J Pathol Microbiol [serial online] 2020 [cited 2020 Nov 25];63:637-9. Available from: https://www.ijpmonline.org/text.asp?2020/63/4/637/299301





   Introduction Top


Uterine ichthyosis is an exceedingly rare condition in which the entire surface of the endometrium is replaced by stratified squamous epithelium. Originally described as an endometrial response to iatrogenically introduced caustic substances, similar changes have since been described in association with a variety of inflammatory conditions of the endometrium.[1] Ichthyosis uteri associated with dysplasia, in situ carcinoma, and frank squamous cell carcinoma of endometrium has been reported in the literature. We describe a case of extensive ichthyosis uteri with dysplasia along with high-grade intraepithelial neoplasia of cervix. The importance of thorough sampling of the uterus and cervix cannot be undermined so that dysplasia, in situ carcinoma, or invasive malignancy cannot be missed.


   Case History Top


A 67-year-old postmenopausal female presented with a history of per vaginal bleeding since 6 to 8 months. She had recurrent complaints of per vaginal foul-smelling discharge since last 3 years and had undergone drainage of pyometra twice for this. There was no history of tuberculosis or iatrogenically introduced substance in the uterus. She had no other significant clinical complaints.

Examination revealed a hypertrophied cervix flushed with the vagina and an enlarged uterus. Bilateral adnexa could not be palpated. CT scan showed bulky uterus with distended fluid-filled endometrial cavity, hypertrophic cervix, and unremarkable adnexa. A diagnosis of pyometra was made. Endometrial cavity was drained and an endometrial curettage along with cervical biopsy was done. Endometrial curettage showed only necrotic material. Cervical biopsy showed cervical intraepithelial neoplasia (CIN III). The patient underwent total abdominal hysterectomy.

The specimen showed an enlarged uterus measuring 10 × 4.5 × 3 cm. The endometrial cavity was dilated with shaggy necrotic material filling the endometrial cavity [Figure 1]a. Cervix and bilateral adnexa were unremarkable grossly.
Figure 1: (a) Gross specimen of the uterus showing dilated irregular endometrial cavity. (b) Squamous metaplasia of endometrium (H and E 40X). (c) Metaplastic endometrium showing dysplasia (H and E 100X). (d) Cervix showing endocervical glands and CIN III (H and E 40X). Inset: CIN III at higher magnification (H and E 400X)

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Sections from endometrium showed replacement of the entire endometrium by stratified squamous epithelium, inflammatory infiltrate, and few residual atrophic endometrial glands [Figure 1]b. Squamous epithelial lining showed moderate dysplasia [Figure 1]c. Cervix showed cervical intraepithelial neoplasia-CIN III [Figure 1]d. Fallopian tubes and ovaries were unremarkable. A diagnosis of ichthyosis of uteri with moderate dysplasia and cervical intraepithelial neoplasia–CIN III was made.


   Discussion Top


Endometrial metaplasia is divided into squamous, mucinous, ciliated (tubal), hobnail, clear cell, and eosinophilic—squamous metaplasia being the commonest. Squamous metaplasia of endometrium is mostly manifested by morules or nodules of benign non-keratinizing squamous cells associated with benign or malignant endometrial glands. It has been described with low-grade endometrial adenocarcinoma and with various benign conditions, including endometrial hyperplasia, chronic endometritis, and endometrial polyps. Extensive plaque-like keratinizing squamous change is distinctly uncommon and is known as ichthyosis uteri. Abnormal uterine bleeding and other endometrial disorders were often treated by introduction of caustic substances like formalin or iodine into the uterine cavity in the past. Extensive squamous metaplasia would often occur as a result of this treatment.

The presence of squamous epithelium in the endometrium is variously termed as ichthyosis uteri, leukoplakia, epidermalization psoriasis uteri, epidermoid heteroplasia, cholesterometra, acanthosis, and indirect regenerative squamous metaplasia.[2] Chronic endometritis, tuberculous endometritis, pyometra, estrogen deficiency, senile involution, irradiation, chronic irritants such as intrauterine devices are considered causes of squamous metaplasia of endometrium. Ichthyosis has sometimes been reported as an incidental finding.[3] The endometrial glands may be normal or atrophic or may show squamous metaplasia similar to that seen in surface endometrium. Although ichthyosis is a benign condition, varying degrees of dysplasia arising within the squamous lining of the endometrium,[4],[5] as well as frank squamous cell carcinoma[6] and adenocarcinoma, have been described in ichthyosis.[7]

The cervical lining may also show CIN.[1],[4] This may range from CIN I to microinvasive carcinoma.[1],[4],[5] Squamous cell carcinoma arising from the cervix and extending upward into the endometrium may be difficult to distinguish from squamous cell carcinoma arising in ichthyosis uteri. A continuum of changes in the endometrium from metaplasia to dysplasia, carcinoma in situ, and microinvasive and invasive carcinoma may be seen, which make the diagnosis easier since a carcinoma extending from the cervix will not show these changes.[8] Ichthyosis of uteri with in situ and invasive SCC of endometrium may not show cervical dysplasia.[2],[9]

Ichthyosis uteri may not be associated with endometrial and cervical dysplasia.[3],[10]


   Conclusion Top


The number of cases of ichthyosis uteri associated with either uterine or cervix malignancies is comparatively more than just squamous metaplasia of endometrium seen in ichthyosis uteri. Hence, extensive sampling of both endometrium and cervix should be done to look for evidence of malignancy in a case of ichthyosis of uteri.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to b'e reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Patton WT, Squires GV. Ichthyosis uteri: A case report. Am J Obstet Gynaecol 1962;84:858-60.  Back to cited text no. 1
    
2.
Pailoor K, Pai MR, Gatty RC, Fernandes H, Jayaprakash CS, Marla NJ. A rare case of primary insitu squamous cell carcinoma of the endometrium with extensive icthyosis uteri. Online J Health Allied Sci. 2014;13:10.  Back to cited text no. 2
    
3.
Wahal SP, Mardi K. Ichthyosis uteri: An incidental finding with review of literature. Muller J Med Sci Res 2014;5:89-91.  Back to cited text no. 3
  [Full text]  
4.
Malla KV, Shrestha B. Cervical intraepitheial neoplasm with ichthyosis uteri- A case report. J Pathol Nep 2018;8:1320-2.  Back to cited text no. 4
    
5.
Thumma RR, Krishna R, Pidakala P. Dysplastic ichthyosis uteri of endometrium associated with squamous cell carcinoma insitu of cervix- does it have a malignant potential. IOSR-JDMS 2014;13:64-7.  Back to cited text no. 5
    
6.
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 icthyosis uteri. Diagn Pathol 2008;3:4.  Back to cited text no. 6
    
7.
Bhardwaj N, Diwaker P, Gogoi P, Wadhwa N, Mishra K. Ichthyosis uteri associated with endometrial adenocarcinoma: A case report. J Clin Diagn Res 2017;11:24-5.  Back to cited text no. 7
    
8.
Kennedy AS, DeMars LR, Flannagan LM, Varia MA. Primary squamous cell carcinoma of the endometrium: A first report of adjuvant chemoradiation. Gynecol Oncol 1995;59:117-23.  Back to cited text no. 8
    
9.
Amita K, Padmini J. Squamous cell carcinoma of the endometrium with extensive icthyosis uteri. Online J Health Allied Scs 2010;9:25-7.  Back to cited text no. 9
    
10.
Nisal MK, Raje G, Sule M, Tyler X, Crocker S. A case of ichthyosis uteri. J Obstet Gynaecol 2011;31:94-5.  Back to cited text no. 10
    

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Correspondence Address:
Archana H Deshpande
Department of Pathology, Andaman and Nicobar Islands Institute of Medical Sciences, Port Blair . 744 101
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/IJPM.IJPM_223_19

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    Abstract
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