Rare Case of Squamous Cell Cancer – Mixed Neuroendocrine Neoplasm in the tubular Esophagus (MiNEN) discovered by Endoscopic Submucosal Dissection of the entire Tumor

Abstract

Background

Mixed neuroendocrine-non-neuroendocrine neoplasms (MiNENs) are rare in the gastrointestinal tract.

Case report

We report the case of a patient in whom complete endoscopic resection of a tumorous lesion in the tubular esophagus, initially diagnosed as squamous cell carcinoma (SCC) led to the diagnosis of SCC-MiNEN, which changed the patient´s treatment course.

Conclusion

With improvements in endoscopic techniques, we advocate resection of small malignant lesions in the gastrointestinal tract for both curative and diagnostic reasons.

Zusammenfassung

Hintergrund

Gemischte neuroendokrine/nicht-neuroendokrine Neoplasmen (MiNENs) sind im Gastrointestinaltrakt selten.

Fallbericht

Wir berichten über den Fall eines Patienten, bei dem die vollständige endoskopische Resektion einer tumorösen Läsion im tubulären Ösophagus, die ursprünglich als Plattenepithelkarzinom (PLECA) diagnostiziert wurde, zur Diagnose eines PLECA-MiNEN führte, was den Therapieverlauf des Patienten änderte.

Schlussfolgerung

Angesichts der Verbesserungen bei den endoskopischen Techniken befürworten wir die Resektion kleiner maligner Läsionen im Gastrointestinaltrakt sowohl aus kurativen als auch aus diagnostischen Gründen.

Keywords

MiNEN - mixed neuroendocrine-non-neuroendocrine neoplasm - squamous cell carcinoma (SCC) - neuroendocrine carcinoma (NEC) - endoscopic submucosal dissection (ESD) - Esophagus - neuroendocrine tumor (NET)

Schlüsselwörter

MiNEN - gemischtes neuroendokrin-nichtneuroendokrines Karzinom - neuroendokrines Karzinom (NEC) - neuroendokriner Tumor (NET) - Plattenepithelkarzinom (PLECA) - Ösophagus - Endoskopische Submukosadissektion (ESD)

Publication History

Received: 10 April 2025

Accepted after revision: 28 May 2025

Article published online:
16 July 2025

• References

• 1 Mastracci L, Rindi G, Grillo F. et al. Neuroendocrine neoplasms of the esophagus and stomach. Pathologica 2021; 113: 5-11
• 2 Giannetta E, Guarnotta V, Rota F. et al. A rare rarity: Neuroendocrine tumor of the esophagus. Crit Rev Oncol Hematol 2019; 137: 92-107
• 3 Huang Q, Wu H, Nie L. et al. Primary high-grade neuroendocrine carcinoma of the esophagus: a clinicopathologic and immunohistochemical study of 42 resection cases. Am J Surg Pathol 2013; 37: 467-483
• 4 Fukui H, Dohi O, Miyazaki H. et al. A case of endoscopic submucosal dissection for neuroendocrine carcinoma of the esophagus with invasion to the muscularis mucosae. Clin J Gastroenterol 2022; 15: 339-344
• 5 Tanaka Y, Hirata D, Fujii S. et al. Early-stage Neuroendocrine Carcinoma of the Esophagus Observed with Annual Endoscopy for Three Years. Intern Med 2019; 58: 1727-1731
• 6 Watanabe K, Hikichi T, Sato M. et al. A case of endocrine cell carcinoma combined with squamous cell carcinoma of the esophagus resected by endoscopic submucosal dissection. Fukushima J Med Sci 2014; 60: 187-191
• 7 Kobayashi K, Aoki T, Nishioka K. et al. A Case of Esophageal Endocrine Cell Carcinoma that recurred in the Lymph Nodes around the thoracic Aorta after ESD. Gastroenterological Endoscopy 2011; 53: 28-34
• 8 Fujihara S, Kobayashi M, Nishi M. et al. Composite neuroendocrine carcinoma and squamous cell carcinoma with regional lymph node metastasis: a case report. J Med Case Rep 2018; 12: 227
• 9 Dasari CS, Ozlem U, Kohli DR. Composite Neuroendocrine and Squamous Cell Tumor of the Esophagus. ACG Case Rep J 2019; 6: e00248
• 10 Yang L, Sun X, Zou Y. et al. Small cell type neuroendocrine carcinoma colliding with squamous cell carcinoma at esophagus. Int J Clin Exp Pathol 2014; 7: 1792-1795 https://www.ncbi.nlm.nih.gov/pubmed/24817981
• 11 Sulaiman M, Agarwal S, Mandal A. et al. High-grade mixed neuroendocrine non-neuroendocrine neoplasm of the gastroesophageal junction: A rare case report and review of literature. Indian J Pathol Microbiol 2022; 65: 918-920
• 12 Adachi K, Tateno Y, Okada H. An esophageal collision tumor. Clin Gastroenterol Hepatol 2014; 12: e17-18
• 13 Zhang L, Yu B, Liu Z. et al. Analysis of the Clinicopathological Characteristics, Prognosis, and Lymphocyte Infiltration of Esophageal Neuroendocrine Neoplasms: A Surgery-Based Cohort and Propensity-Score Matching Study. Cancers (Basel) 2023; 15
• 14 Wu IC, Chu YY, Wang YK. et al. Clinicopathological features and outcome of esophageal neuroendocrine tumor: A retrospective multicenter survey by the digestive endoscopy society of Taiwan. J Formos Med Assoc 2021; 120: 508-514
• 15 Takubo K, Nakamura K, Sawabe M. et al. Primary undifferentiated small cell carcinoma of the esophagus. Hum Pathol 1999; 30: 216-221
• 16 Kuwano H, Yokobori T, Ide M. et al. Coexistence of superficial carcinogenesis of resident epithelium besides neuroendocrine neoplasm of the digestive tract. Cancer Med 2022; 11: 983-992
• 17 Maru DM, Khurana H, Rashid A. et al. Retrospective study of clinicopathologic features and prognosis of high-grade neuroendocrine carcinoma of the esophagus. Am J Surg Pathol 2008; 32: 1404-1411
• 18 Frizziero M, Wang X, Chakrabarty B. et al. Retrospective study on mixed neuroendocrine non-neuroendocrine neoplasms from five European centres. World J Gastroenterol 2019; 25: 5991-6005
• 19 Ebert MP, Fischbach W, Hollerbach S. et al. S3-Leitlinie Diagnostik und Therapie der Plattenepithelkarzinome und Adenokarzinome des Ösophagus [German Guidelines for Squamous Cell Carcinoma and Adenocarcinoma of the esophagus]. Z Gastroenterol 2024; 62: 535-642
• 20 Kitagawa Y, Ishihara R, Ishikawa H. et al. Esophageal cancer practice guidelines 2022 edited by the Japan esophageal society: part 1. Esophagus 2023; 20: 343-372
• 21 Beaufort IN, Frederiks CN, Overwater A. et al. Endoscopic submucosal dissection for early esophageal squamous cell carcinoma: long-term results from a Western cohort. Endoscopy 2024; 56: 325-333
• 22 La Rosa S, Sessa F, Uccella S. Mixed Neuroendocrine-Nonneuroendocrine Neoplasms (MiNENs): Unifying the Concept of a Heterogeneous Group of Neoplasms. Endocr Pathol 2016; 27: 284-311
• 23 Morita M, Saeki H, Nakaji YU. et al. Conversion to Neuroendocrine Carcinoma from Squamous Cell Carcinoma of the Esophagus After Definitive Chemoradiotherapy. Anticancer Res 2016; 36: 4045-4049 https://www.ncbi.nlm.nih.gov/pubmed/27466511
• 24 Borghesani M, Reni A, Lauricella E. et al. Efficacy and Toxicity Analysis of mFOLFIRINOX in High-Grade Gastroenteropancreatic Neuroendocrine Neoplasms. J Natl Compr Canc Netw 2024; 22
• 25 Welin S, Sorbye H, Sebjornsen S. et al. Clinical effect of temozolomide-based chemotherapy in poorly differentiated endocrine carcinoma after progression on first-line chemotherapy. Cancer 2011; 117: 4617-4622
• 26 Zhang P, Li Z, Li J. et al. Clinicopathological features and lymph node and distant metastasis patterns in patients with gastroenteropancreatic mixed neuroendocrine-non-neuroendocrine neoplasm. Cancer Med 2021; 10: 4855-4863