Combined Carcinoid Carcinoma and Squamous Cell Carcinoma of the Endometrium: A Case Report and Survey of Related Literature

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Abstract

Introduction

Histologically, endometrial neuroendocrine carcinoma is an extremely rare pathological type of endometrial cancer. In addition, this type is characterized by high invasiveness and poor clinical outcome, which was classified into carcinoid (low grade), and small-cell and large-cell neuroendocrine carcinoma (high grade). Globally, reports on endometrial carcinoid carcinoma are limited. Clinically, it is also rare to see primary squamous cell carcinoma of endometrium.

Case Presentation

Here, we report an interesting case of mixed carcinoma of endometrium with both carcinoid and squamous cell carcinoma, which presented with persistent vaginal bleeding and hyponatremia.

Conclusion

Careful pathologic review is necessary to diagnose this rare disease. More studies in the future are warranted to demonstrate the primary surgical treatments and the efficacy of adjuvant therapy of this disease.

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Most cited references 12

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Pathology of Neuroendocrine Tumours of the Female Genital Tract

Brooke E Howitt, Paul Kelly, W. McCluggage (2017)

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Neuroendocrine Carcinoma of the Endometrium

Cady E Pocrnich, Preetha Ramalingam, Elizabeth D Euscher … (2016)

Neuroendocrine carcinoma (NECa) of the endometrium is an uncommon tumor. In this study, we present the clinicopathologic features of 25 such cases. The patients ranged in age from 37 to 87 years (median, 57 y) and most commonly presented with vaginal bleeding. The tumors were either pure NECa (10) or mixed with other histotypes (15), most commonly endometrioid carcinoma. The NECas were large cell type (15), small cell type (4), or a mixture of both (6). NECa was underrecognized in 89% of referral/consultation cases. All tumors were positive for ≥1 neuroendocrine marker (chromogranin, synaptophysin, CD56). Additional immunohistochemical (IHC) studies were obtained in 18 cases, with positive results as follows: keratin cocktail (17), diffuse p16 (6), PAX-8 (6), CD117 (6), and TTF-1 (1). Mismatch-repair protein expression by IHC was abnormal in 8 of 18 cases (6 MLH1/PMS2 loss; 1 MSH2/MSH6 loss; 1 MSH6 loss). According to FIGO staging, cases were distributed as follows: I (6), II (2), III (10), and IV (7). All patients underwent surgical treatment, and 20 patients received adjuvant therapy. Twelve patients died of disease (mean survival 12.3 mo). Eleven patients were alive 5 to 134 months after diagnosis, including 7 who achieved a 5-year survival (3 stage I; 4 stage III). In summary, most of our endometrial NECas were large cell type, mixed with other histotypes, and underrecognized. These tumors tend to be PAX-8 negative and may be associated with microsatellite instability. The recognition of NECa may have an impact on the treatment of the patients affected by this disease. Although NECa usually has an aggressive behavior, 28% of our patients survived at least 5 years.

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Clinicopathologic features, treatment, prognosis and prognostic factors of neuroendocrine carcinoma of the endometrium: a retrospective analysis of 42 cases from the Kansai Clinical Oncology Group/Intergroup study in Japan

Harunobu Matsumoto, Mototsugu Shimokawa, Kaei Nasu … (2019)

Objective We conducted a retrospective, multi-institutional, collaborative study to accumulate cases of neuroendocrine carcinoma of the endometrium, to clarify its clinicopathologic features, treatment, prognosis and prognostic factors to collate findings to establish future individualized treatment regimens. To our knowledge, this is the largest case study and the first study to statistically analyze the prognosis of this disease. Methods At medical institutions participating in the Kansai Clinical Oncology Group/Intergroup, cases diagnosed at a central pathologic review as neuroendocrine carcinoma of the endometrium between 1995 and 2014 were enrolled. We retrospectively analyzed the clinicopathologic features, treatment, prognosis and prognostic factors of this disease. Results A total of 65 cases were registered from 18 medical institutions in Japan. Of these, 42 (64.6%) cases were diagnosed as neuroendocrine carcinoma of the endometrium based on the central pathological review and thus included in the study. Advanced International Federation of Gynecology and Obstetrics stages (stage III and IV) and pure type small cell neuroendocrine carcinoma cases had a significantly worse prognosis. Upon multivariate analysis, only histologic subtypes and surgery were significant prognostic factors. Pure type cases had a significantly worse prognosis compared to mixed type cases and complete surgery cases had a significantly better prognosis compared to cases with no or incomplete surgery. Conclusion Our findings suggest that complete surgery improves the prognosis of neuroendocrine carcinoma of the endometrium. Even among cases with advanced disease stages, if complete surgery is expected to be achieved, clinicians should consider curative surgery to improve the prognosis of neuroendocrine carcinoma of the endometrium.

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Author and article information

Journal

Journal ID (nlm-ta): Case Rep Oncol

Journal ID (iso-abbrev): Case Rep Oncol

Journal ID (hwp): CRO

Journal ID (publisher-id): CRO

Title: Case Reports in Oncology

Publisher: S. Karger AG (Basel, Switzerland )

ISSN (Electronic): 1662-6575

Publication date (Electronic): 15 November 2023

Publication date Collection: Jan-Dec 2023

Publication date PMC-release: 15 November 2023

Volume: 16

Issue: 1

Pages: 1402-1408

Affiliations

[a ]Department of Obstetrics and Gynecology, Beijing Chao-Yang Hospital, Capital Medical University, Beijing, China

[b ]Department of Pathology, Beijing Chao-Yang Hospital, Capital Medical University, Beijing, China

[c ]Department of Radiology, Beijing Chao-Yang Hospital, Capital Medical University, Beijing, China

Author notes

Correspondence to: CuiQin Sang, sangcuiqin2020@ 123456126.com

Article

Publisher ID: 534622

DOI: 10.1159/000534622

PMC ID: 10651180

SO-VID: ebef0e1e-a875-44e2-8795-280895283e71

License:

This article is licensed under the Creative Commons Attribution-NonCommercial 4.0 International License (CC BY-NC) ( http://www.karger.com/Services/OpenAccessLicense). Usage and distribution for commercial purposes requires written permission.

History

Date received : 6 September 2023

Date accepted : 9 October 2023

Date: 2023

Page count

Figures: 2, References: 12, Pages: 7

Funding

No funding was received.

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Combined Carcinoid Carcinoma and Squamous Cell Carcinoma of the Endometrium: A Case Report and Survey of Related Literature

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Abstract

Introduction

Case Presentation

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Karger: Oncology

Most cited references 12

Pathology of Neuroendocrine Tumours of the Female Genital Tract

Neuroendocrine Carcinoma of the Endometrium

Clinicopathologic features, treatment, prognosis and prognostic factors of neuroendocrine carcinoma of the endometrium: a retrospective analysis of 42 cases from the Kansai Clinical Oncology Group/Intergroup study in Japan

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