Z Gastroenterol 2025; 63(02): 145-154
DOI: 10.1055/a-2442-9540
Kasuistik

Entirely Intramural Growth Pattern: A Rare Presentation of Esophageal Squamous Cell Carcinoma and Review of the Literature

Vollständig intramurales Wachstumsmuster: eine seltene Darstellung des Plattenepithelkarzinoms der Speiseröhre und Literaturrecherche
Jiayu Qiu
1   Department of Gastroenterology, Jiangxi Provincial Key Laboratory of Digestive Diseases, Jiangxi Clinical Research Center for Gastroenterology, Digestive Disease Hospital, The First Affiliated Hospital, Jiangxi Medical College, Nanchang University, Nanchang, Jiangxi, China (Ringgold ID: RIN117970)
,
Yi Tu
2   Department of Pathology, First Affiliated Hospital of Nanchang University, Nanchang, Jiangxi, China (Ringgold ID: RIN117970)
,
Chen Yu
3   Department of Radiology, First Affiliated Hospital of Nanchang University, Nanchang, Jiangxi, China (Ringgold ID: RIN117970)
,
Xu Shu
1   Department of Gastroenterology, Jiangxi Provincial Key Laboratory of Digestive Diseases, Jiangxi Clinical Research Center for Gastroenterology, Digestive Disease Hospital, The First Affiliated Hospital, Jiangxi Medical College, Nanchang University, Nanchang, Jiangxi, China (Ringgold ID: RIN117970)
,
Xiaolin Pan
1   Department of Gastroenterology, Jiangxi Provincial Key Laboratory of Digestive Diseases, Jiangxi Clinical Research Center for Gastroenterology, Digestive Disease Hospital, The First Affiliated Hospital, Jiangxi Medical College, Nanchang University, Nanchang, Jiangxi, China (Ringgold ID: RIN117970)
,
Yanxia Zhang
1   Department of Gastroenterology, Jiangxi Provincial Key Laboratory of Digestive Diseases, Jiangxi Clinical Research Center for Gastroenterology, Digestive Disease Hospital, The First Affiliated Hospital, Jiangxi Medical College, Nanchang University, Nanchang, Jiangxi, China (Ringgold ID: RIN117970)
› Institutsangaben
Gefördert durch: Science and Technology Department of Jiangxi Province 20192BAB215034
Gefördert durch: National Natural Science Foundation of China 81401983
Gefördert durch: Scientific Research of Health Commission of Jiangxi Province 202310393
Gefördert durch: Education Department of Jiangxi Province JJ210230

Abstract

Background

Esophageal squamous cell carcinoma (ESCC) is a malignant tumor originating from the squamous epithelium. In contrast, esophageal submucosal tumors are common benign lesions arising from mesenchymal tissues. To date, an entirely intramural growth of ESCC is very rare. This study described a case of an esophageal submucosal tumor resected by endoscopic submucosal dissection (ESD) that was finally diagnosed as ESCC.

Case report

A 51-year-old woman presented with progressive dysphagia and was provisionally diagnosed with esophageal leiomyoma by further diagnostic modalities. The patient did not have any obvious suspicious malignant features and underwent ESD. However, the histopathology of the resected specimen was reported as poorly differentiated infiltrating squamous cell carcinoma with normal overlying squamous epithelium. Consequently, the patient received additional chemoradiotherapy, and no recurrence was observed during the 2-year follow-up.

Literature review

A comprehensive literature search related to ESCC with entirely intramural growth was performed in PubMed and Embase from their inception up to November 2023, and 12 articles including 13 cases were finally included in the literature review. Subsequently, we extracted information about these cases.

Conclusion

It is concluded that ESCC may masquerade as a submucosal tumor with a complete submucosal growth pattern and is easily misdiagnosed because endoscopic biopsy and iodine staining are always negative. Therefore, if a patient with a submucosal tumor has dysphagia or weight loss in the short term, clinicians should be alert to the possibility of ESCC with a complete submucosal growth pattern. Endoscopic ultrasonography (EUS), chest computed tomography (CT), or positron emission tomography–computed tomography (PET-CT) may help assist in the diagnosis, and EUS-guided fine-needle aspiration (EUS-FNA) could be used to confirm the diagnosis.

Zusammenfassung

Hintergrund

Das Plattenepithelkarzinom der Speiseröhre (ESCC) ist ein bösartiger Tumor, der aus dem Plattenepithel stammt. Im Gegensatz dazu sind submuköse Tumoren der Speiseröhre häufig gutartige Läsionen, die aus mesenchymalen Geweben entstehen. Bis heute ist das vollständig intramurale Wachstum von ESCC sehr selten. In dieser Studie wurde ein Fall eines submukösen Tumors der Speiseröhre beschrieben, der durch endoskopische submuköse Dissektion (ESD) entfernt und schließlich als ESCC diagnostiziert wurde.

Fallbericht

Eine 51-jährige Frau stellte sich mit fortschreitender Dysphagie vor und wurde vorläufig mittels weiterer diagnostischer Verfahren mit einem Ösophagus-Leiomyom diagnostiziert. Die Patientin zeigte keine offensichtlichen verdächtigen malignen Merkmale und unterzog sich einer ESD. Allerdings wurde die Histopathologie des resezierten Präparats als schlecht differenziertes infiltrierendes Plattenepithelkarzinom mit normalem überliegendem Plattenepithel gemeldet. Folglich erhielt die Patientin zusätzliche Chemo- und Strahlentherapie, und während der 2-jährigen Nachbeobachtung wurde kein Rezidiv beobachtet.

Literaturübersicht

Eine umfassende Literaturrecherche zu ESCC mit vollständig intramuralem Wachstum wurde in PubMed und Embase von deren Anfängen bis November 2023 durchgeführt, und 12 Artikel mit 13 Fällen wurden schließlich in die Literaturübersicht aufgenommen. Anschließend extrahierten wir Informationen über diese Fälle.

Schlussfolgerung

Es wird geschlossen, dass ESCC als submuköser Tumor mit einem vollständigen submukösen Wachstumsmuster maskiert sein kann und leicht fehldiagnostiziert wird, da endoskopische Biopsien und Jodfärbungen immer negativ sind. Daher sollten Kliniker bei Patienten mit einem submukösen Tumor, die innerhalb kurzer Zeit Dysphagie oder Gewichtsverlust haben, auf die Möglichkeit eines ESCC mit vollständigem submukösem Wachstumsmuster achten. Die endoskopische Ultraschalluntersuchung (EUS), die Computertomografie des Brustkorbs (CT) oder die Positronen-Emissions-Tomografie-Computertomografie (PET-CT) können bei der Diagnose helfen, und die EUS-gesteuerte Feinnadelaspiration (EUS-FNA) könnte zur Bestätigung der Diagnose verwendet werden.



Publikationsverlauf

Eingereicht: 09. Juli 2024

Artikel online veröffentlicht:
25. November 2024

© 2024. Thieme. All rights reserved.

Georg Thieme Verlag KG
Oswald-Hesse-Straße 50, 70469 Stuttgart, Germany

 
  • References

  • 1 Bray F, Ferlay J, Soerjomataram I. et al. Global cancer statistics 2018: GLOBOCAN estimates of incidence and mortality worldwide for 36 cancers in 185 countries. CA: A Cancer Journal for Clinicians 2018; 68 (06) 394-424
  • 2 Abnet CC, Arnold M, Wei WQ. Epidemiology of Esophageal Squamous Cell Carcinoma. Gastroenterology 2018; 154 (02) 360-373
  • 3 Chen W, Zheng R, Baade PD. et al. Cancer statistics in China, 2015. CA: A Cancer Journal for Clinicians 2016; 66 (02) 115-132
  • 4 Puhr HC, Pablik E, Berghoff AS. et al. Viennese risk prediction score for Advanced Gastroesophageal carcinoma based on Alarm Symptoms (VAGAS score): characterisation of alarm symptoms in advanced gastro-oesophageal cancer and its correlation with outcome. ESMO Open 2020; 5 (02) e000623
  • 5 Sarbia M, Becker KF, Hofler H. Pathology of upper gastrointestinal malignancies. Semin Oncol 2004; 31 (04) 465-475
  • 6 Fockens P, Bartelsman J, Tytgat G. Benign and Malignant Esophageal Tumors Other than Squamous and Adenocarcinoma. Gastrointest Endosc Clin of N Am 1994; 4 (04) 791-801
  • 7 McGregor DH, Mills G, Boudet RA. Intramural squamous cell carcinoma of the esophagus. Cancer 1976; 37 (03) 1556-1561
  • 8 Kishino T, Yamaguchi Y, Yamagishi T. et al. Submucosal tumor (SMT)-like esophageal squamous cell carcinoma with gastric metastasis. Hepatogastroenterology 2000; 47 (36) 1581-1584
  • 9 von Rahden BH, Brucher BL, Sarbia M. Esophageal squamous cell carcinoma with entirely intramural growth pattern. Virchows Arch 2006; 448 (06) 862-866
  • 10 Schmitz KJ, Konig C, Riesener KP. Intramural carcinoma of the oesophagogastric junction. BMJ Case Rep 2012; 2012: bcr0320126080
  • 11 Sonthalia N, Jain SS, Surude RG. et al. Primary Esophageal Intramural Squamous Cell Carcinoma Masquerading as a Submucosal Tumor: A Rare Presentation of a Common Disease. Clin Med Insights Gastroenterol 2016; 9: 63-66
  • 12 Choudhary NS, Bansal RK, Puri R. et al. Esophageal squamous cell carcinoma presenting as submucosal lesion with repeatedly negative endoscopic biopsies. Journal of Digestive Endoscopy 2019; 7 (04) 153-154
  • 13 Shanmugam RM, Shanmugam C, Murugesan M. et al. Oesophageal carcinoma mimicking a submucosal lesion: A case report. World J Gastrointest Endosc 2019; 11 (11) 541-547
  • 14 Zhu H, Shi B, Li F. et al. Esophageal intramural squamous cell carcinomas presenting as subepithelial lesions removed by endoscopic submucosal dissection: Two case reports. Medicine (Baltimore) 2020; 99 (45) e21850
  • 15 Wang W, Li D, Zheng L. et al. Oesophageal squamous cell carcinoma mimicking submucosal tumour. BMC Gastroenterol 2021; 21 (01) 150
  • 16 Pan H, Zhou X, Zhao F. et al. The diagnosis of intramural esophageal squamous cell carcinoma without mucosal invasion using endoscopic ultrasound-guided fine needle aspiration biopsy. Medicine 2020; 99 (27) e21099
  • 17 Jia Y, Zhang Q, Li E. et al. Submucosal oesophageal squamous cell carcinoma with lymph node metastasis: a case report and literature review. BMC Gastroenterol 2022; 22 (01) 97
  • 18 Yang L, Zuo Z, Zhang H. et al. Esophageal squamous cell carcinoma presenting as a submucosal tumor. Asian J Surg 2023; 46 (08) 3075-3076
  • 19 Lee HL. Advances in the Management of Upper Gastrointestinal Subepithelial Tumor: Pathologic Diagnosis Using Endoscopy without Endoscopic Ultrasound-Guided Biopsy. Clin Endosc 2016; 49 (03) 216-219
  • 20 McCarty TR, Ryou M. Endoscopic diagnosis and management of gastric subepithelial lesions. Curr Opin Gastroenterol 2020; 36 (06) 530-537
  • 21 Arnold M, Soerjomataram I, Ferlay J. et al. Global incidence of oesophageal cancer by histological subtype in 2012. Gut 2015; 64 (03) 381-387
  • 22 Chheda NN. Upper Esophageal Dysphagia. Surgical Clinics of North America 2022; 102 (02) 199-207
  • 23 Iwaya T, Maesawa C, Uesugi N. et al. Coexistence of esophageal superficial carcinoma and multiple leiomyomas: A case report. World J Gastroenterol 2006; 12 (28) 4588-4592
  • 24 Mortensen MB, Pless T, Durup J. et al. Clinical impact of endoscopic ultrasound-guided fine needle aspiration biopsy in patients with upper gastrointestinal tract malignancies. A prospective study. Endoscopy 2001; 33 (06) 478-483
  • 25 Nishida T, Kawai N, Yamaguchi S. et al. Submucosal tumors: comprehensive guide for the diagnosis and therapy of gastrointestinal submucosal tumors. Dig Endosc 2013; 25 (05) 479-489
  • 26 Baysal B, Masri OA, Eloubeidi MA. et al. The role of EUS and EUS-guided FNA in the management of subepithelial lesions of the esophagus: A large, single-center experience. Endosc Ultrasound 2017; 6 (05) 308-316
  • 27 Aoki T, Nakamura T, Oshikiri T. et al. Strategy for esophageal non-epithelial tumors based on a retrospective analysis of a single facility. Esophagus 2018;
  • 28 Niimi K, Goto O, Kawakubo K. et al. Endoscopic ultrasound-guided fine-needle aspiration skill acquisition of gastrointestinal submucosal tumor by trainee endoscopists: A pilot study. Endosc Ultrasound 2016; 5 (03) 157-164
  • 29 Williams DB, Sahai AV, Aabakken L. et al. Endoscopic ultrasound guided fine needle aspiration biopsy: a large single centre experience. Gut 1999; 44 (05) 720-726
  • 30 Kanno A, Yasuda I, Irisawa A. et al. Adverse events of endoscopic ultrasound-guided fine-needle aspiration for histologic diagnosis in Japanese tertiary centers: Multicenter retrospective study. Dig Endosc 2021; 33 (07) 1146-1157
  • 31 Tae HJ, Lee HL, Lee KN. et al. Deep biopsy via endoscopic submucosal dissection in upper gastrointestinal subepithelial tumors: a prospective study. Endoscopy 2014; 46 (10) 845-850
  • 32 Jacobson BC, Bhatt A, Greer KB. et al. ACG Clinical Guideline: Diagnosis and Management of Gastrointestinal Subepithelial Lesions. Am J Gastroenterol 2023; 118 (01) 46-58