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| Ahead of print publication |
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Clusters of malignant cysts in the gastric submucosal layer (with video)
Shan Li1, Minna Gao2, Li Tao3, Guomin Luo4, Qing Gao1, Kun Qian5, Liang Deng1
1 Department of Gastroenterology, The First Affiliated Hospital of Chongqing Medical University, Chongqing, China
2 Department of Pathology, Chongqing Medical University, Chongqing, China
3 Department of Radiology, The First Affiliated Hospital of Chongqing Medical University, Chongqing, China
4 Department of Oncology, Yongchuan Hospital, Chongqing Medical University, Chongqing, China
5 Department of Gastrointestinal Surgery, The First Affiliated Hospital of Chongqing Medical University, Chongqing, China
| Date of Submission | 22-May-2021 |
| Date of Acceptance | 30-Nov-2021 |
| Date of Web Publication | 02-May-2022 |
Correspondence Address:
Kun Qian,
Department of Gastrointestinal Surgery, The First Affiliated Hospital of Chongqing Medical University, Chongqing 400016
China
Liang Deng,
Department of Gastroenterology, The First Affiliated Hospital of Chongqing Medical University, Chongqing 400016
China
 Source of Support: None, Conflict of Interest: None DOI: 10.4103/EUS-D-21-00133
How to cite this URL:
Li S, Gao M, Tao L, Luo G, Gao Q, Qian K, Deng L. Clusters of malignant cysts in the gastric submucosal layer (with video). Endosc Ultrasound [Epub ahead of print] [cited 2022 Sep 24]. Available from: http://www.eusjournal.com/preprintarticle.asp?id=344245 |
Shan Li, Minna Gao, Li Tao
These authors contributed equally as co-first authors.
Kun Qian, Liang Deng
These authors contributed equally to the work as the corresponding authors.
A 68-year-old female was admitted for intolerable abdominal pain for 2 months, which can only be relieved by continuous analgesic treatment. Her medical history was unremarkable.
Laboratory examinations revealed a moderative elevation of serum CA 19-9 (122.8 U/mL, normal range: 0–27.0 U/mL). Standard endoscopy showed multiple bulges in the gastric body, but repeated mucosal biopsies solely suggested mild gastritis [Figure 1]. Numerous irregular cysts were seen throughout the gastric wall, forming a honeycomb look on computed tomography and EUS, while on EUS, the mucosal layer remained continuous [Figure 1], [Figure 2] and [Video 1 [Additional file 1]]. The entire gastric wall was involved, causing peristalsis loss, resulting in repeated vomiting and worsening abdominal pain, and following EUS-FNA indicated borderline cystadenocarcinoma with an elevation of CA 19-9 (499.7 U/mL) in the cystic fluid [Supplementary Table 1 [Additional file 2]]. Later, a palliative total gastrectomy was performed to release symptoms and verify the diagnosis. | Figure 1: The endoscopic view and EUS of the stomach. Standard gastroscopy showing multiple cyst-like bulging and compressible lesions with mild mucosal erosions (a). On the EUS image, numerous cysts were distributed through the submucosal layer to the serosa with irregular sizes (b). One large cyst was visualized on the EUS (c), and EUS-guided fine needle aspiration was performed to obtain cystic fluid (d)
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 | Figure 2: Metastatic mixed serous–mucinous cystadenocarcinoma originated from type II gastric ectopic pancreas in the stomach. (a) Enhanced abdominal computed tomography showing diffused and irregular cysts in the gastric body. (b) The resected stomach comprising a large cyst and several smaller ones in the gastric wall's deep layer. (c) Hematoxylin and eosin staining of the original excision specimen showing cystadenocarcinoma (shaped as cystic changes) extended from the muscularis mucosae to the deep serosa. The lining cells comprised both malignant mucous columnar epithelium (purple arrowhead) and malignant serous cuboidal epithelium (black arrowhead). Ducts with malignant change (one cancer cell with morphism and hyperchromatic nuclei, black arrow) and ducts with normal morphology (purple arrow) can be seen simultaneously
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In histological examinations, from muscularis mucosa to the serosa, many malignant cysts were seen, but the mucosal layer was normal, which was different from any reported gastric cancer [Supplementary Figure 1 [Additional file 3]]. Besides, cystic walls were comprised both mucinous columnar and serous cuboidal cells. Notably, near malignant cysts, some benign cystic structures with normal columnar epithelium can also be seen, shaped like pancreatic ducts [Figure 2] and [Supplementary Figure 2 [Additional file 4]],[Supplementary Figure 3 [Additional file 5]],[Supplementary Figure 4 [Additional file 6]],[Supplementary Figure 5 [Additional file 7]],[Supplementary Figure 6 [Additional file 8]]. As expected, the immunohistochemical test and whole-exome sequencing analysis confirmed their pancreatic origin [Supplementary Table 2 [Additional file 9]] and [Supplementary Figure 7 [Additional file 10]], as a mixed serous–mucinous cystadenocarcinoma originated from type II ectopic pancreas (consisting of ducts only) was diagnosed.[1] Postoperatively, abdominal pain was relieved, but she could not tolerate chemotherapy and refused further treatment. Unfortunately, 6 months later, this patient died of cancer relapse [Supplementary Figure 8 [Additional file 11]].
We reported the first case of gastric submucosal multiple cystic lesions originated from the ectopic pancreas, and it clearly showed the malignant transformation.[2] From this case, to investigate the nature of these submucosal cystic lesions, EUS and EUS-FNA played critical roles. In addition, the elevation of CA 19-9 in the serum and cystic fluid might be an important indicator for diagnosing such lesions.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient's guardians have given their consent for her images and other clinical information to be reported in the journal. The patient's guardians understand that her names and initials will not be published and due efforts will be made to conceal her identity, but anonymity cannot be guaranteed.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
Supplementary materials
Supplementary information is linked to the online version of the paper on the Endoscopic Ultrasound website.
| References | |  |
| 1. | Rezvani M, Menias C, Sandrasegaran K, et al. Heterotopic pancreas: Histopathologic features, imaging findings, and complications. Radiographics 2017;37:484-99.  |
| 2. | Cazacu IM, Luzuriaga Chavez AA, Nogueras Gonzalez GM, et al. Malignant transformation of ectopic pancreas. Dig Dis Sci 2019;64:655-68. |
[Figure 1], [Figure 2]
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