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Mixed high-grade serous and large cell neuroendocrine carcinoma arising from rectal endometriosis 11 years after hysterectomy

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Abstract

The malignant gastrointestinal endometriosis transformation is represented by endometriosis-associated intestinal tumors. Endometrioid adenocarcinoma and clear cell adenocarcinoma are most common among the endometrial cancers of all organs. Only four cases of mixed serous carcinoma and large cell neuroendocrine carcinoma have been reported, and all these cases originated from the uterus. A 59-year-old woman with a month’s history of bloody stools was admitted. She was stable until the hematochezia occurred but is 11 years post-hysterectomy. A circumferential type-3 advanced upper rectum tumor was seen on colonoscopy. Adenocarcinoma was revealed from the forceps biopsies of the type-3 tumor component. Computed tomography showed narrowed lumen with a thickened rectum wall, a continuing mass, and a component on the anorectal side. Swollen lymph nodes were observed around the rectum, but no distant metastatic lymph nodes or organs were found. To treat the lesion, rectal surgical resection with D3 lymph node dissection was performed. Histological examination revealed combined high-grade serous and large cell neuroendocrine carcinomas. Tumor was contiguous to the endometrium in the sub-serosa. Endometriosis was determined to be the origin of both carcinomas. Therefore, endometriosis-associated intestinal tumors should be included in the differential diagnosis when rectal tumors with cystic structures are found post-hysterectomy.

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References

  1. Yantiss RK, Clement PB, Young RH. Endometriosis of the intestinal tract: a study of 44 cases of a disease that may cause diverse challenges in clinical and pathologic evaluation. Am J Surg Pathol. 2001;25:445–54.

    Article  CAS  PubMed  Google Scholar 

  2. Macafee CH, Greer HL. Intestinal endometriosis. A report of 29 cases and a survey of the literature. J Obstet Gynaecol Br Emp. 1960;67:539–55.

    Article  CAS  PubMed  Google Scholar 

  3. Orbuch IK, Reich H, Orbuch M, et al. Laparoscopic treatment of recurrent small bowel obstruction secondary to ileal endometriosis. J Minim Invasive Gynecol. 2007;14:113–5.

    Article  PubMed  Google Scholar 

  4. Paksoy M, Karabiçak I, Ayan F, et al. Intestinal obstruction due to rectal endometriosis. Mt Sinai J Med. 2005;72:405–8.

    PubMed  Google Scholar 

  5. Heaps JM, Nieberg RK, Berek JS. Malignant neoplasms arising in endometriosis. Obstet Gynecol. 1990;75:1023–8.

    CAS  PubMed  Google Scholar 

  6. Mulvany NJ, Allen DG. Combined large cell neuroendocrine and endometrioid carcinoma of the endometrium. Int J Gynecol Pathol. 2008;27:49–57.

    Article  PubMed  Google Scholar 

  7. Albores-Saavedra J, Martinez-Benitez B, Luevano E. Small cell carcinomas and large cell neuroendocrine carcinomas of the endometrium and cervix: polypoid tumors and those arising in polyps may have a favorable prognosis. Int J Gynecol Pathol. 2008;27:333–9.

    Article  PubMed  Google Scholar 

  8. Posligua L, Malpica A, Liu J, et al. Combined large cell neuroendocrine carcinoma and papillary serous carcinoma of the endometrium with pagetoid spread. Arch Pathol Lab Med. 2008;132:1821–4.

    Article  PubMed  Google Scholar 

  9. Terada T. Large cell neuroendocrine carcinoma with sarcomatous changes of the endometrium: a case report with immunohistochemical studies and molecular genetic study of KIT and PDGFRA. Pathol Res Pract. 2010;206:420–5.

    Article  PubMed  Google Scholar 

  10. Deodhar KK, Kerkar RA, Suryawanshi P, et al. Large cell neuroendocrine carcinoma of the endometrium: an extremely uncommon diagnosis, but worth the efforts. J Cancer Res Ther. 2011;7:211–3.

    Article  PubMed  Google Scholar 

  11. Shahabi S, Pellicciotta I, Hou J, et al. Clinical utility of chromogranin A and octreotide in large cell neuro endocrine carcinoma of the uterine corpus. Rare Tumors. 2011;3: e41.

    Article  PubMed  PubMed Central  Google Scholar 

  12. Makihara N, Maeda T, Nishimura M, et al. Large cell neuroendocrine carcinoma originating from the uterine endometrium: a report on magnetic resonance features of 2 cases with very rare and aggressive tumor. Rare Tumors. 2012;4: e37.

    Article  PubMed  PubMed Central  Google Scholar 

  13. Rivera G, Niu S, Chen H, et al. Collision tumor of endometrial large cell neuroendocrine carcinoma and low-grade endometrial stromal sarcoma: a case report and review of the literature. Int J Surg Pathol. 2020;28:569–73.

    Article  PubMed  Google Scholar 

  14. Nguyen ML, Han L, Minors AM, et al. Rare large cell neuroendocrine tumor of the endometrium: a case report and review of the literature. Int J Surg Case Rep. 2013;4:651–5.

    Article  PubMed  PubMed Central  Google Scholar 

  15. Erhan Y, Dikmen Y, Yucebilgin MS, et al. Large cell neuroendocrine carcinoma of the uterine corpus metastatic to brain and lung: case report and review of the literature. Eur J Gynaecol Oncol. 2004;25:109–12.

    CAS  PubMed  Google Scholar 

  16. Pocrnich CE, Ramalingam P, Euscher ED, et al. Neuroendocrine carcinoma of the endometrium: a clinicopathologic study of 25 cases. Am J Surg Pathol. 2016;40:577–86.

    Article  PubMed  PubMed Central  Google Scholar 

  17. Tu YA, Chen YL, Lin MC, et al. Large cell neuroendocrine carcinoma of the endometrium: a case report and literature review. Taiwan J Obstet Gynecol. 2018;57:144–9.

    Article  PubMed  Google Scholar 

  18. Jenny C, Kimball K, Kilgore L, et al. Large cell neuroendocrine carcinoma of the endometrium: a report and review of the literature. Gynecol Oncol Rep. 2019;28:96–100.

    Article  PubMed  PubMed Central  Google Scholar 

  19. Hu R, Jiang J, Song G, et al. Mixed large and small cell neuroendocrine carcinoma of the endometrium with serous carcinoma: a case report and literature review. Medicine. 2019;98: e16433.

    Article  PubMed  PubMed Central  Google Scholar 

  20. Hardy LE, Chaudry Z, Wan K, et al. Primary mixed large cell neuroendocrine and high grade serous carcinoma of the endometrium. BMJ Case Rep. 2020;13: e234977.

    Article  PubMed  PubMed Central  Google Scholar 

  21. Slavin RE, Krum R, Van Dinh T. Endometriosis-associated intestinal tumors: a clinical and pathological study of 6 cases with a review of the literature. Hum Pathol. 2000;31:456–63.

    Article  CAS  PubMed  Google Scholar 

  22. Howitt BE, Kelly P, McCluggage WG. Pathology of neuroendocrine tumours of the female genital tract. Curr Oncol Rep. 2017. https://doi.org/10.1007/s11912-017-0617-2.

    Article  PubMed  Google Scholar 

  23. Sampson JA. Endometrial carcinoma of the ovary, arising in endometrial tissue in that organ. Arch Surg. 1925;10:1–72.

    Article  Google Scholar 

  24. Ariura M, Kasajima R, Miyagi Y, et al. Combined large cell neuroendocrine carcinoma and endometrioid carcinoma of the endometrium: a shared gene mutation signature between the two histological components. Int Cancer Conf J. 2017;6:11–5.

    Article  PubMed  Google Scholar 

  25. Abu MA, Sinha P, Totoe L, et al. Endometrial cancer thirteen years after total abdominal hysterectomy and bilateral salpingo-oophorectomy and hormone replacement therapy: a case report. Eur J Gynaecol Oncol. 1997;18:482–3.

    CAS  PubMed  Google Scholar 

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Acknowledgements

We would like to express our gratitude to Dr. Yuko Omori of the Tohoku University Department of Investigative Pathology for her pathological evaluation, Dr. Kazunori Otsuka of the Miyagi Cancer Center Department of Medical Oncology for chemotherapy, and all medical staff for their assistance at the Soma General Hospital.

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Authors and Affiliations

  1. Department of Gastroenterology, Soma General Hospital, Fukushima, Japan

    Takumi Yanagita, Yuichi Waragai, Hiroshi Shimizu, Yuta Takahashi, Naoto Abe, Choichiro Hashimoto & Hiromi Kumakawa

  2. Department of Gastroenterology, Fukushima Medical University School of Medicine, Fukushima, Japan

    Takumi Yanagita, Yuichi Waragai, Hiroshi Shimizu, Naoto Abe, Choichiro Hashimoto & Hiromasa Ohira

  3. Department of Endoscopy, Fukushima Medical University Hospital, 1 Hikarigaoka, Fukushima, 960-1295, Japan

    Takuto Hikichi, Yuichi Waragai & Masao Kobayakawa

Authors
  1. Takumi Yanagita
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  2. Takuto Hikichi
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  3. Yuichi Waragai
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  4. Hiroshi Shimizu
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  5. Yuta Takahashi
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  8. Hiromi Kumakawa
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  9. Masao Kobayakawa
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  10. Hiromasa Ohira
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Contributions

Conceptualization, TY and TH. Methodology, TY, TH, and YW. Formal Analysis, TY Investigation, TY, YW, HS, YT, NA, CH, and HK. Resources, TY, TH, and YW Writing–Original Draft Preparation, TY and TH Writing–Review and Editing, YW and MK. Supervision, HO. All authors have read and approved the final manuscript.

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Correspondence to Takuto Hikichi.

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Takumi Yanagita, Takuto Hikichi, Yuichi Waragai, Hiroshi Shimizu, Yuta Takahashi, Naoto Abe, Choichiro Hashimoto, Hiromi Kumakawa, Masao Kobayakawa, Hiromasa Ohira declare that they have no conflict of interest.

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All procedures followed have been performed in accordance with the ethical standards laid down in the 1964 Declaration of Helsinki and its later amendments.

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Yanagita, T., Hikichi, T., Waragai, Y. et al. Mixed high-grade serous and large cell neuroendocrine carcinoma arising from rectal endometriosis 11 years after hysterectomy. Clin J Gastroenterol 16, 366–371 (2023). https://doi.org/10.1007/s12328-023-01769-y

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  • DOI: https://doi.org/10.1007/s12328-023-01769-y

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