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 Table of Contents  
CASE REPORT
Year : 2021  |  Volume : 8  |  Issue : 2  |  Page : 187-190

Ichthyosis uteri with carcinoma cervix: A case report


1 Department of Pathology and Lab Medicine, All India Institute of Medical Sciences (AIIMS), Bhopal, Madhya Pradesh, India
2 Department of Pathology, MGM Medical College and Hospital, Kamothe, Navi Mumbai 410209, Maharashtra, India

Date of Submission06-Apr-2021
Date of Acceptance06-Apr-2021
Date of Web Publication02-Jun-2021

Correspondence Address:
Dr. Abeer M Ilyas
Department of Pathology and Lab Medicine, All India Institute of Medical Sciences (AIIMS), Bhopal, Madhya Pradesh.
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/mgmj.mgmj_80_20

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  Abstract 

Ichthyosis uteri is a rare condition in which the entire surface of the endometrium undergoes metaplasia and is replaced by stratified squamous epithelium. This change is the endometrial response to iatrogenically introduced caustic substances and a variety of inflammatory conditions of the endometrium. We describe herein microinvasive squamous cell carcinoma of the uterine cervix with extensive ichthyosis uteri-like changes in the adjacent endometrium. The possible origin of these complex findings and the neoplastic potential of ichthyosis uteri is also discussed.

Keywords: Carcinoma cervix, ichthyosis uteri, pyometra, squamous metaplasia of the endometrium


How to cite this article:
Ilyas AM, Maheswari U. Ichthyosis uteri with carcinoma cervix: A case report. MGM J Med Sci 2021;8:187-90

How to cite this URL:
Ilyas AM, Maheswari U. Ichthyosis uteri with carcinoma cervix: A case report. MGM J Med Sci [serial online] 2021 [cited 2021 Oct 21];8:187-90. Available from: http://www.mgmjms.com/text.asp?2021/8/2/187/317453




  Introduction Top


Ichthyosis uteri, a rare condition, is used when the entire surface of the endometrium is replaced by stratified squamous epithelium. Although the condition is often benign, anaplastic and dysplastic changes have been reported.[1] The term “ichthyosis uteri” was initially coined in 1885 by Zeller to refer to the extensive squamous metaplasia of the entire surface of the endometrium following iatrogenically introduced caustic substances such as formalin or iodine.[2] Etiological factors associated with the conditions are tuberculous endometritis, puerperal endometritis, endometrial polyps, hyperplasia, and pyometra secondary to cervical stenosis or malignancies.[2]


  Case history Top


A 55-year-old multiparous, lean, postmenopausal woman presented at a tertiary care hospital, Mumbai, India with foul-smelling vaginal discharge for the past few months. She was a multipara with G2P2A0. She had been postmenopausal for 5 years. On abdomen examination, the abdomen was soft and non-tender. As per vaginal examination, the cervix appeared healthy. On ultrasonography, uterus was enlarged measuring 8 × 4 cm, and there was a tiny cyst in the right ovary, suggestive of a postmenopausal simple ovarian cyst. A Papanicolaou smear (Pap smear) of the cervix was suggestive of severe dysplasia. A laparotomy and a total abdominal hysterectomy with right salpingo-oophorectomy were performed. The procedure was carried out with ease, and there were no postoperative complications.

On gross examination, on cut section 50 mL of pus was present and showed widened endometrial cavity and the uterus was enlarged. The inner surface of the cavity had a smooth appearance. The cut section showed widened endometrial cavity with the thin cervix. [Figure 1]A and B, along with attached unilateral adnexa with ovary, shows the presence of a simple cyst measuring 1 × 0.6 × 0.3 cm.
Figure 1: A. Specimen of the uterus along with the cervix with unilateral adnexa (arrow). B. Cut section of the uterus with smooth endometrial (thick arrow) and cervical surface (thin arrow)

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Microscopy: Sections from ectocervix show malignant epithelial cells arranged in nests, sheets, as well a few singly scattered invading into the underlying stroma. Individual cells are round to oval to polygonal, showing increased nuclear-cytoplasmic ratios, a moderate degree of nuclear pleomorphism having a vesicular nucleus with prominent nucleoli and scanty cytoplasm. Atypical mitotic figures are also seen (WHO; early invasive squamous cell carcinoma). Serial sections from the endometrium to the fundus show the presence of dysplastic squamous cells covering the entire surface of the endometrium. A focus of malignant squamous cells is seen extending up to 0.2 cm into the myometrium [Figure 2]A and B. Sections from the ovary showed a simple cyst. Additionally, HPV testing was carried out at an outsourced laboratory using Diagen DNA hybrid capture methodology “Hybrid Capture 2 High-Risk HPV DNA Test.” The endometrium and cervix were found positive for high-risk HPV.
Figure 2: A. (H&E stain 40×) Extensive replacement of the entire endometrial lining by sheets of atypical squamoid cells in nests (arrow). B. (H&E stain 20×) High-grade squamous intraepithelial lesion cells extending over the entire surface of the endometrium up to 0.2 cm into the myometrium

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  Discussion Top


The term “ichthyosis uteri” and the phenomenon it describes have been acknowledged and have been used now and again in the literature.[2],[3],[4],[5] Here, we report a case of ichthyosis uteri-like changes of the entire innermost layer of the uterus, which is the endometrium; moreover, it shows dysplasia. In addition to the endometrium finding, there is the presence of cervical squamous cell carcinoma with microinvasion. Our findings were similar to the findings by Patton and Squires.[6] They mention in their case report that the patient underwent a hysterectomy for severe pyometra; microscopic evaluation showed extensive dysplasia of the cervix (with a focal area of microinvasion) and ichthyosis uteri of the endometrium. Moreover, the metaplastic endometrium showed some degree of “cellular anaplasia,” which the authors interpreted as a direct extension from the cervix.[6]

This complex conclusion can be explained in two scenarios: the first being that squamous cell carcinoma developed in a background of ichthyosis uteri and the second being extension of the cervical carcinoma into the endometrium.

In our case, the patient is postmenopausal with no evident mass over the cervix, and the endometrial lesion shows extension into the myometrium. HPV testing turned positive for the high-risk group for both the endometrium and the cervix. These findings give us a new insight, perhaps dysplasia developed on the background of ichthyosis uteri with superimposed HPV infection. Goodrich et al.[7] have suggested in their study primary squamous cell carcinoma of the endometrium that HPV may play a role in its pathogenesis.

Development of invasive squamous cell carcinoma at the base of ichthyosis uteri[8] reported its association with ichthyosis uteri.[9] Also, a case of endometrial adenocarcinoma is covered almost entirely by a plaque-like, keratinizing mature squamous epithelium.[10] Clement and Young[11] report that the finding of plaque-like squamous epithelium lesion is rarely seen in endometrioid adenocarcinoma of the endometrium.

In a population-based study from Norway, the prevalence of primary squamous cell carcinoma is 0.1%.[12] For a tumor to be accepted as a primary endometrial carcinoma, it should fulfill the criteria established by Fluhmann et al. and modified by Kay et al. that there must be no coexisting endometrial adenocarcinoma; there must be no connection between endometrial tumor and squamous epithelium of the cervix; there must be absences of primary squamous carcinoma of the cervix; and if the cervix shows an in situ carcinoma, there must be no connection between this and independent endometrial neoplasm.[13] In our case, the cervix shows the presence of squamous cell carcinoma (microinvasion), and HPV for high-risk groups was detected in both the cervix and the endometrium. Hence, a more plausible explanation is that there is an extension of cervical cancer into the endometrium. Direct extension hypothesis coined by the investigators reported a case of the cervical intraepithelial lesion with contiguous spread to the endometrium, fallopian tubes, and ovaries, and tumors from all sites showed the presence of HPV-16.[14] On literature search, many authors thought that ichthyosis uteri had negligible malignant potential.[15],[16]


  Conclusion Top


Summing up, the discovery of squamous epithelium in endometrial curettage or biopsy specimen slide should be judicially examined to rule out the possibility of squamous cell carcinoma.

A proper history of etiological factors such as chronic trauma, repair, irritation, inflammation, foreign material, and estrogenic effects should be considered on findings of squamous plaques in a curettage or biopsy of the endometrium.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
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. 1
    
2.
Marcus SL Adenoacanthoma of the endometrium. A report of 24 cases and a review of squamous metaplasia. Am J Obstet Gynecol 1961;81:259-67.  Back to cited text no. 2
    
3.
Bewtra C, Xie QM, Hunter WJ, Jurgensen W Ichthyosis uteri: A case report and review of the literature. Arch Pathol Lab Med 2005;129:e124-5.  Back to cited text no. 3
    
4.
Kücükali T, Ertoy D, Ayhan A Ichthyosis uteri associated with a uterine squamous papilloma. Eur J Gynaecol Oncol 1996;17:37-41.  Back to cited text no. 4
    
5.
Zschoch H, Ertel G [Ichthyosis uteri–squamous cell epithelial metaplasia of the endometrium]. Zentralbl Allg Pathol 1987;133:355-60.  Back to cited text no. 5
    
6.
Patton WT, Squires GV Icthyosis uteri. A case report. Am J Obstet Gynecol 1962;84:858-60.  Back to cited text no. 6
    
7.
Goodrich S, Kebria-Moslemi M, Broshears J, Sutton GP, Rose P Primary squamous cell carcinoma of the endometrium: Two cases and a review of the literature. Diagn Cytopathol 2013;41:817.  Back to cited text no. 7
    
8.
Gargade CB, Deshpande AH Ichthyosis uteri with dysplasia — A case report. Indian J Pathol Microbiol 2020;63:637-9.  Back to cited text no. 8
    
9.
Anderson MC, Robboy SJ, Russell P, Morse A Endometritis, metaplasia, polyps and miscellaneous changes. In: Robboy, SJ, Anderson, MC, Russell, P, editors. Pathology of the Female Reproductive Tract. London: Churchill-Livingstone; 2002. p. 303.  Back to cited text no. 9
    
10.
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:ED24-5.  Back to cited text no. 10
    
11.
Clement PB, Young RH Endometrioid carcinoma of the uterine corpus: A review of its pathology with emphasis on recent advances and problematic aspects. Adv Anat Pathol 2002;9:145-84.  Back to cited text no. 11
    
12.
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. 12
    
13.
Kay S Squamous-cell carcinoma of the endometrium. Am J Clin Pathol 1974;61:264-9.  Back to cited text no. 13
    
14.
Pins MR, Young RH, Crum CP, Leach IH, Scully RE Cervical squamous cell carcinoma in situ with intraepithelial extension to the upper genital tract and invasion of tubes and ovaries: Report of a case with human papilloma virus analysis. Int J Gynecol Pathol 1997;16:272-8.  Back to cited text no. 14
    
15.
Zaino RJ Metaplasia. In: Silverberg, SG, editor. Interpretation of Endometrial Biopsies and Curettings. Philadelphia: Lippincott-Raven; 1996. p. 241-61.  Back to cited text no. 15
    
16.
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. 16
    


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