Superficial Endometrial Spread of Squamous Cell Carcinoma of the Cervix (Cake Icing): Two Case Reports and Review of the Literature

Abstract

Background: Although squamous cell carcinoma of the cervix is the most common gynecologic malignancy in India, superficial spreading carcinoma of the endometrium is rare. The uterine corpus can be involved by direct extension from cervical growth, but superficial spread without invasion of the underlying myometrium is uncommon. Case: Two cases of squamous cell carcinoma of the cervix with superficial extension to the endometrium are reported. Clinically, both cases presented with pyometra. The tumor had spread superficially into the entire endometrial cavity up to the fundus, totally replacing the columnar epithelium. Results: Both patients were diagnosed as having stage 1b cervical squamous cell carcinoma and were treated with hysterectomy and bilateral salpingo-oophrectomy. One patient was lost to follow-up and the other has remained disease free for 48 months. Conclusions: As squamous cell carcinoma of the cervix with superficial extension to the endometrium is a rare phenomenon, with fewer than 40 cases reported in literature so far, this article has been written to emphasize the need for keeping this possibility in mind in cases of carcinoma of the cervix. (J GYNCOL SURG 28:375)

Introduction

Squamous cell carcinoma of the cervix is the most common gynecologic malignancy in India. Recognized patterns of local tumor spread include direct invasion of the parametrium and endometrial cavity; however, superficial spread without invasion of the underlying myometrium is uncommon.¹ Two interesting cases of squamous cell carcinoma of the cervix, in which there was replacement of the entire endometrial surface by an intraepithelial squamous cell carcinoma, are reported, along with a review of literature. This is a very rare phenomenon, with fewer than 40 cases reported so far in the literature.

Cases

Case 1

A 65-year-old gravida 5, para 4, abortus 1, 10 years postmenopausal woman was investigated for an abnormal Papanicolaou smear. Clinically, there was a mild cervical stenosis with pyometra. There was no contributing medical history. A punch biopsy confirmed the diagnosis of an infiltrating squamous cell carcinoma of the cervix, nonkeratinizing, large cell type. Wertheim's hysterectomy with bilateral salpingo-oophrectomy was performed. Grossly, the uterus and cervix measured 8×5×4 cm. The cervix showed an irregular gray-white growth measuring 3.5×3×2 cm. The endometrial cavity was dilated and the endometrial surface had a gray-white corrugated appearance (Fig. 1). Microscopically, the cervical lesion consisted of squamous cell carcinoma moderately differentiated (Fig. 2) and extending up and over the endometrial surface of the lower uterine segment as carcinoma in situ, without involvement of the underlying myometrium (Fig. 3). The tumor extended to the right parametrium, but the left parametrium, vaginal cuff, both fallopian tubes, and the ovaries were unremarkable. Eleven lymph nodes were isolated, and all showed reactive hyperplasia. Systemic examination, including abdominal ultrasonography and computed tomographic (CT) chest and bone scan, was negative for evidence of distant metastasis. The patient was diagnosed as having stage 1b cervical squamous cell carcinoma.

FIG. 1.

Gross photograph revealing dilated endometrial cavity with corrugated appearance and cervix replaced by gray-white growth.

FIG. 2.

Photomicrograph from the cervical growth revealing invasive moderately differentiated squamous cell carcinoma (hematoxylin and eosin ×100).

FIG. 3.

Photomicrograph revealing carcinoma of the cervix extending over the endometrial surface as carcinoma in situ, without involvement of the underlying myometrium (hematoxylin and eosin ×40).

The immediate postoperative course was uneventful, and the patient was put on chemotherapy, but was lost to follow up after one cycle.

Case 2

A 60-year-old gravida 2, para 2, 5 years postmenopausal woman presented with a 4 month history of vaginal bleeding and discharge. On pelvic examination, the uterus was enlarged and there was a firm mass occupying the cervix. Cervical biopsy revealed invasive squamous cell carcinoma, large cell nonkeratinizing type. A radical vaginal hysterectomy with bilateral salpingo-oophrectomy, in addition to pelvic and para-aortic lymph node dissection, was performed. Grossly, the cervix revealed a gray-white growth measuring 2.5×2×1 cm. A cut section of growth was gray-white, with areas of necrosis and hemorrhage. The endometrial cavity was roughened with gray-white patches. Microscopically, the cervical lesion consisted of squamous cell carcinoma, large cell nonkeratinizing type, extending up and over the endometrial surface. There was no evidence of remaining normal endometrial gland and stroma. The entire endometrial surface was replaced with squamous cell carcinoma in situ, with few small foci of micro- invasion, associated with extensive inflammatory cell infiltrate. Right and left parametrium, vaginal cuff, both the fallopian tubes and ovaries, and all the pelvic and para-aortic lymph nodes isolated were negative for metastasis and showed reactive hyperplasia. Systemic examination, including abdominal ultrasonography and CT chest and bone scan, was negative for evidence of distant metastasis.

The patient was diagnosed as having stage 1b cervical squamous cell carcinoma. She has been on regular follow-up since, and clinical examination is being performed at regular intervals. The patient is free of disease, both locally and distantly, 48 months after the presentation of primary symptoms.

Discussion

Primary squamous cell carcinoma of the endometrium is very rare.² The presence of squamous epithelium in the endometrium, variously termed as “ichthyosis uteri,” “leukoplakia epidermalization,” “psoriatic uteri,” “epidermoid heteroplasia,” “cholesteometra,” and “indirect regenerative squamous metaplasia,” has been described under a variety of conditions, and is benign in the majority of cases. Primary squamous cell carcinoma may arise through the process of squamous metaplasia, as proposed by Baggish and Woodruff.³

To be accepted as primary carcinoma of the endometrium, the tumor must satisfy the criteria established by Fluhmann and modified by Kay.⁴ These criteria are:

1. No co-existent endometrial adenocarcinoma

2. No demonstrable connection between the endometrial tumor and the stratified squamous epithelium of the cervix

3. No primary cervical carcinoma

Pyometra, nonspecific and specific endometritis, tuberculosis, syphilis, vitamin A deficiency, irradiation, foreign bodies including intrauterine devices (IUDs), chemical agents, and exogenous and endogenous estrogens have been mentioned as associated with, or as precursors of, this type of lesion.⁴

More commonly, squamous cell carcinoma of endometrium can occur as direct extension from carcinoma of the cervix. The common pattern of uterine corpus involvement by cervical cancer is through deep myometrial penetration or via lymphatic dissemination. The superficial spread of in situ or invasive squamous cell carcinoma of the cervix over the contagious endometrial surface may occur in rare instances. The intrauterine spread of cervical carcinoma in the endometrium may be evident on gross inspection as whitish patches, a condition called “cake icing” or “Zukerguss” carcinoma, in which superficial squamous tumor sweeps over or replaces the entire endometrium.¹ Such involvement was seen in both of the cases described here. Such a lesion may involve the entire endometrial cavity and may extend into the tubal mucosa, fimbria, and ovary.

Cervical stenosis and subsequent pyometra may promote surface propagation of cervical cancer. An extensive survey of the literature revealed a report of 38 cases of cervical carcinoma with endometrial surface involvement. Analysis of these cases presented by various authors showed 11 cases of carcinoma in situ,^1,5–7 2 cases of micro-invasive carcinoma,^5,8 and 25 cases of invasive cervical carcinoma^9,5,10–14 (Table 1). In 8 cases, the fallopian tube was also involved in direct continuity with cervical and endometrial lesions.^5,10 In 4 cases, bilateral ovaries were also involved.^1,7,11,14 One case in addition showed extensive superficial spread to almost the entire genital tract, with associated endometrial stromal sarcoma.¹¹ The case discussed here showed endometrium lined by malignant squamous epithelium with foci of micro-invasion into surrounding stroma. In all cases, inflammatory changes were present and pyometra was reported in many cases. In both of the cases discussed here, the patient presented in the sixth decade, more than 10 years above the expected age for cervical cancer. The usual complaints mentioned in the literature are vaginal bleeding, abdominal pain, and abdominal lump, which were seen in the present cases.

Table 1.

A Summary of Reported Cases of Superficial Spread to the Endometrium in Association with Squamous Cell Lesions of the Cervix

No.	Authors	Year	Mean age (years)	Total cases	In situ	Micro-invasive	Invasive carcinoma	Associated other involvement
1	Schmitt and Schafer⁹	1977	-	4	-	-	4	-
2	Kanbour and Stock⁵	1978	60	25	8	1	16	Fallopian tube
3	Gupta et al.⁶	1979	-	1	1	-	-	-
4	Sandhyamani¹⁰	1983	-	1	-	-	1	Fallopian tube and vagina
5	Motayama and Watanabe¹¹	1988	-	1	-	-	1	Genital tract and endometrial stromal sarcoma
6	Pins et al.⁷	1997	55	1	1	-	-	Fallopian tube and ovary
7	Tan et al.⁸	2004	70	1	-	1	-	-
8	Fadare¹²	2006	38	1	-	-	1	-
9	Agashe et al.¹	2006	55	1	1	-	-	Fallopian tube and ovary
10	Adler et al.¹³	2007		1			1	-
11	Gungor et al.¹⁴	2011	53	1	-	-	1	Fallopian tube and ovary
12	Present study	2011	62	2	-	-	2	-

On radiologic investigations, magnetic resonance imaging (MRI) may suggest layers of early enhancing tumor lining the endometrial cavity.

Although the data are limited, the survival in treated cases of squamous cell carcinoma of the cervix with contiguous spread of the tumor into the endometrium does not appear to be altered and, therefore, does not change clinical staging.⁵

Conclusion

To conclude, squamous cell carcinoma of the cervix with superficial extension to the endometrium is a rare phenomenon, with <40 cases reported in the literature so far, which has prompted this report, whose purpose was to emphasize the need for keeping this possibility in mind in cases of carcinoma of the cervix.

Footnotes

Acknowledgments

Dr. Nisha Marwah thank all the co-authors and the technicians for making significant contributions to this study.

Disclosure Statement

No competing financial conflicts exist.

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