Clin J Gastroenterol (2009) 2:80–84 DOI 10.1007/s12328-008-0042-z
CASE REPORT
Bronchogenic cyst of the stomach involved with gastric adenocarcinoma Hiroaki Shibahara Æ Toshiyuki Arai Æ Shunpei Yokoi Æ Seijun Hayakawa
Received: 8 February 2008 / Accepted: 2 October 2008 / Published online: 2 December 2008 Ó Springer 2008
Abstract Bronchogenic cyst, a congenital anomaly mostly found in the mediastinum, rarely arises in the stomach. A 43-year-old man had epigastric pain and was diagnosed as having gastric adenocarcinoma. Abdominal ultrasonography showed hepatic cyst, and computed tomography and magnetic resonance imaging revealed a cystic lesion near the stomach. At surgery, the cystic lesion was found to be located at the lesser curvature of the stomach where the cancer invasion was seen. Total gastrectomy with combined resection of the cystic lesion was performed. Pathologically, the cyst wall was lined by pseudostratified ciliated columnar epithelium, subepithelial mixed seromucinous glands and smooth muscle bundles. The pathological diagnosis was bronchogenic cyst of the stomach involved with gastric adenocarcinoma. Based on a similar association between gastric diffuse submucosal cysts and gastric cancer in the previous reports, it is possible that chronic inflammation from bronchogenic cysts to the gastric mucosa may cause adenocarcinoma in the stomach. At surgery, complete combined resection without rupture of the bronchogenic cyst involved with the gastric adenocarcinoma is needed for treatment of gastric cancer to prevent dissemination of cancer cells considering when cancer cells have invaded beyond the pseudostratified
H. Shibahara T. Arai S. Yokoi Department of Surgery, Anjo Kosei Hospital, 28 Higashi-Hirokute, Anjo-cho, Anjo 446-8602, Japan H. Shibahara (&) Department of Surgery, Fukuroi Municipal Hospital, 2515-1 Kunou, Fukuroi 437-0061, Japan e-mail: [email protected] S. Hayakawa Department of Pathology, Anjo Kosei Hospital, Anjo, Japan
123
ciliated columnar epithelium and within the bronchogenic cyst. Keywords Bronchogenic cyst Stomach Adenocarcinoma
Introduction Bronchogenic cysts are foregut-derived developmental anomalies most commonly encountered in the mediastinum and rarely in the abdomen or retroperitoneum [1]. Bronchogenic cysts of the stomach are uncommon [2–12], and no case of bronchogenic cyst of the stomach with cancer invasion has been reported. We herein report the first case of bronchogenic cyst of the stomach involved with gastric adenocarcinoma.
Case report A 43-year-old man had epigastric pain, and gastrointestinal endoscopy in a nearby hospital revealed irregular mucosa at the cardia of the stomach. He was referred to our hospital for further examination. Abdominal ultrasonography showed hepatic cyst including internal low echoic pattern with extrahepatic growth (Fig. 1). Upper gastrointestinal series showed an irregular ulcerative lesion with compression by a cyst looking like a submucosal tumor at the lesser curvature of the cardia (Fig. 2). Endoscopy of the stomach showed an irregular mucosal lesion at the lesser curvature of the cardia (Fig. 3). Biopsy specimens of the lesion revealed group V adenocarcinoma. Computed tomography (Fig. 4) and magnetic resonance imaging (Fig. 5) revealed a cystic lesion near the stomach. Our
Clin J Gastroenterol (2009) 2:80–84
Fig. 1 Abdominal ultrasonography shows hepatic cyst (arrowhead) including internal low echoic pattern with extrahepatic growth
81
preoperative diagnoses for these lesions were gastric cancer and hepatic cyst. At surgery, the cystic lesion was found to be located at the lesser curvature of the stomach and to be invaded by the cancer. The cystic lesion had no communication with liver parenchyma. Combined resection of the cystic lesion without rupture and total gastrectomy with D2 lymph node dissection was performed. Grossly, the cystic lesion of the resected specimen was a soft mass measuring 9 9 4 cm. The gastric cancer showed a type-2 lesion compressed by the cystic lesion macroscopically (Fig. 6a). The cut surface after fixation by formalin revealed that the cystic lesion was unilocular and filled with green-colored gelatinous content, and that the gastric cancer had invaded the cystic lesion (Fig. 6b). Pathologically, the cystic lesion was located at the opposite side of the gastric wall from the adenocarcinoma (Fig. 7a). The cyst wall was lined by pseudostratified ciliated columnar epithelium (Fig. 7b) and smooth muscle bundles, which did not resemble the muscularis propria of the stomach. Mixed seromucinous glands (Fig. 7c) were identified in the subepithelial layer of the cyst. No hyaline cartilage was identified. The gastric cancer was moderately differentiated adenocarcinoma infiltrated to the subserosa with involvement in one right paracardial lymph node, which showed tubular pattern without cystic formation, and both lymphatic and vascular invasion was present. The stage was II (pT2 pN1 sH0 sP0 sM0) according to the Japanese classification of gastric carcinoma [13]. The adenocarcinoma cells invaded the cystic lesion, but not the pseudostratified ciliated columnar epithelium (Fig. 7d). The pathological diagnosis was bronchogenic cyst of the stomach invaded by the adjacent gastric adenocarcinoma. The patient was discharged uneventfully.
Discussion
Fig. 2 Upper gastrointestinal series shows irregular ulcerative lesion (arrowhead) with compression by cyst (arrows) appearing like submucosal tumor at the lesser curvature of cardia
Bronchogenic cysts are congenital anomalies arising from the ventral foregut during the 3rd to 7th week of development. Most commonly, bronchogenic cysts migrate caudally with the esophagus and are eventually found in the posterior mediastinum near the carina attached to the tracheobronchial tree or the esophagus [5]. In the present case, the bronchogenic cyst was located at the lesser curvature of the cardia. The proximity of the cyst to the esophagus indicates that the bronchogenic cyst might migrate caudally with the growing esophagus to the stomach in the development. Subdiaphragmatic bronchogenic cysts are rare, and Liang et al. [1] reported 38 cases in the English literature. They reported that the most common organ involved was the diaphragm, followed by the stomach. In our review of the literature, a search was performed using PubMed
123
82
Fig. 3 Endoscopy of stomach shows an irregular mucosal lesion at the lesser curvature of the cardia
Clin J Gastroenterol (2009) 2:80–84
Fig. 6 The gastric cancer shows a type-2 lesion (black arrowheads) compressed by the cystic lesion (white arrows) macroscopically on the fresh specimen (a). The cystic lesion (white line) is unilocular and filled with green-colored gelatinous content on the cut surface after fixation by formalin. Gastric cancer appearing as a white solid mass (arrowheads) is located at the opposite side from the cystic lesion (b)
Fig. 4 Computed tomography shows a cystic lesion (arrow) located between the liver and the stomach (arrowhead)
Fig. 5 Magnetic resonance imaging shows a cystic lesion (arrow) located between the liver and the stomach (arrowhead). L liver
123
Fig. 7 Microscopically, the cystic lesion and gastric cancer are adjacent (a). The cyst wall is lined by pseudostratified ciliated columnar epithelium (b) along the internal surface of the cystic lesion with smooth muscle bundles, and subepithelial mixed seromucinous glands (c) are identified. The pseudostratified ciliated columnar epithelium of the cystic lesion is not involved by adenocarcinoma cells (d). Pseud pseudostratified ciliated columnar epithelium, W wall of the cystic lesion, Ad adenocarcinoma of gastric cancer, Sq squamous epithelium of the esophagus
Clin J Gastroenterol (2009) 2:80–84
83
Table 1 Bronchogenic cyst of the stomach Case no.
References
Age/gender
Symptom
Location
Preoperative diagnosis
Operation
1
Keohane et al. [2]
64/Female
Nausea, vomiting and epigastric discomfort
–/Post
Multiloculated cysts
Resection
Braffman et al. [3]
64/Female
Nausea and vomiting
U/Post
Multiloculated mass
Resection
2
Matsubayashi et al. [4]
62/Male
–
–/Post
Lymphangioma/benign neurogenic tumor
Resection
3
Hedayati et al. [5]
59/Female
–
–/Post
Adrenal incidentaloma
Resection
4
Melo et al. [6]
39/Female
Rib pain
U/–
GIST
Laparoscopic resection
5
Rubio et al. [7, 8]
26/Male
Periodic epigastric pain
U/–
–
–
6
Song et al. [9]
62/Female
–
U/Less
Benign stromal tumor
Resection
7
Lee et al. [10]
38/Female
–
U/–
GIST, developmental or complicated cyst
Endoscopic mucosal resection
8
Wakabayashi et al. [11]
37/Male
Upper abdominal dull pain and dysphagia
U/Less
Duplication cyst
Laparoscopic resection
9
Sato et al. [12]
60/Female
–
U/Less
Cystic neoplasm
–
10
Shibahara et al. (present case)
43/Male
Epigastric pain
U/Less
Hepatic cyst
Resection
U upper portion of the stomach, post posterior wall of the stomach, less lesser curvature of the stomach, GIST gastrointestinal stromal tumor
(http://www.ncbi.nlm.nih.gov/sites/entrez) from 1950 to August 2008 with the following keywords: bronchogenic cyst and stomach. This search yielded 24 citations. Of these, 16 were in the English literature. Of the 16 citations, 11 were identified to be articles describing bronchogenic cyst of the stomach [2–12]. Four citations were redundant reports of two cases [2, 3, 7, 8]. There were nine cases of bronchogenic cyst of the stomach in the English literature, and they are summarized in Table 1, including the present case. Several cases of malignancy in bronchogenic cysts were reported [14–17]. However, there has been no report describing the relation of bronchogenic cyst and gastric cancer. An association between gastric diffuse submucosal cysts and gastric cancer has been reported [18–23]. Iwanaga et al. [19] examined 12 cases of the stomach with submucosal cysts, carcinoma and atypical hyperplasia, and their results showed that erosion or regenerative epithelia were evident on the gastric mucosa of all 12 cases. There was erosion at the superficial mucosa immediately above the heterotopic glands in many cases. They described that gastritis may give rise to heterotopic glands, and the development of heterotopic cysts in the submucosa may make the surface mucosa prone to erosion; repeated erosion and regeneration may cause carcinoma or atypical hyperplasia. Pillay et al. [20] reported a case of diffuse cystic glandular malformation and adenocarcinoma of the stomach, and in their case variable degrees of acute and chronic inflammatory changes in the mucosa and in the submucosa around the glands were seen. Martel et al. [21] reported a case of heterotopic submucosal gastric cysts
with adenocarcinoma. In their case, numerous large cysts were evident in the submucosa beneath the adenocarcinoma, and the invasive neoplasm was focally evident adjacent to the cysts; gastric mucosa manifested extensive and diffuse chronic inflammation with lymphocytes and focal intestinalization. No malignant epithelium was seen within the bronchogenic cyst; however, adenocarcinoma from the gastric mucosa at the opposite side of the stomach wall had invaded to the bronchogenic cyst in the present case. Based on the interpretation of diffuse submucosal cysts and cancer of the stomach described previously, it is possible that chronic inflammation from the bronchogenic cyst to the gastric mucosa may cause adenocarcinoma in the stomach. Bronchogenic cyst, for which the imaging modality does not indicate malignancy and is without symptoms, does not need resection clinically. The present case showed bronchogenic cyst involved with gastric cancer and needing to be resected. There was no report about the surgical procedure for cases with bronchogenic cyst involved with gastric cancer. The authors recommend complete combined resection of the bronchogenic cyst located near gastric cancer with gastrectomy. Furthermore, a surgical procedure without rupture of the cyst is needed to prevent dissemination of gastric cancer cells considering when cancer cells have invaded beyond the pseudostratified ciliated columnar epithelium and within the bronchogenic cyst. In conclusion, we present the first reported case of bronchogenic cyst of the stomach involved with gastric adenocarcinoma. Complete combined resection without
123
84
rupture of the bronchogenic cyst involved with gastric adenocarcinoma is needed for treatment of gastric cancer.
References 1. Liang MK, Yee HT, Song JW, Marks JL. Subdiaphragmatic bronchogenic cysts: a comprehensive review of the literature. Am Surg. 2005;71:1034–41. 2. Keohane ME, Schwartz I, Freed J, Dische R. Subdiaphragmatic bronchogenic cyst with communication to the stomach: a case report. Hum Pathol. 1988;19:868–71. 3. Braffman B, Keller R, Gendal ES, Finkel SI. Subdiaphragmatic bronchogenic cyst with gastric communication. Gastrointest Radiol. 1988;13:309–11. 4. Matsubayashi J, Ishida T, Ozawa T, Aoki T, Koyanagi Y, Mukai K. Subphrenic bronchopulmonary foregut malformation with pulmonary-sequestration-like features. Pathol Int. 2003;53:313–6. 5. Hedayati N, Cai DX, McHenry CR. Subdiaphragmatic bronchogenic cyst masquerading as an ‘‘adrenal incidentaloma’’. J Gastrointest Surg. 2003;7:802–4. 6. Melo N, Pitman MB, Rattner DW. Bronchogenic cyst of the gastric fundus presenting as a gastrointestinal stromal tumor. J Laparoendosc Adv Surg Tech A. 2005;15:163–5. 7. Rubio CA, Orrego A, Willen R. Bronchogenic gastric cyst. A case report. In Vivo. 2005;19:383–5. 8. Rubio CA, Orrego A, Wille´n R. Congenital bronchogenic cyst in the gastric mucosa. J Clin Pathol. 2005;58:335. 9. Song SY, Noh JH, Lee SJ, Son HJ. Bronchogenic cyst of the stomach masquerading as benign stromal tumor. Pathol Int. 2005;55:87–91. 10. Lee SH, Park DH, Park JH, Kim HS, Park SH, Kim SJ, et al. Endoscopic mucosal resection of a gastric bronchogenic cyst that was mimicking a solid tumor. Endoscopy. 2006;38:E12–3. 11. Wakabayashi H, Okano K, Yamamoto N, Suzuki Y, Inoue H, Kadota K, et al. Laparoscopically resected foregut duplication cyst (bronchogenic) of the stomach. Dig Dis Sci. 2007;52:1767–70.
123
Clin J Gastroenterol (2009) 2:80–84 12. Sato M, Irisawa A, Bhutani MS, Schnadig V, Takagi T, Shibukawa G, et al. Gastric bronchogenic cyst diagnosed by endosonographically guided fine needle aspiration biopsy. J Clin Ultrasound. 2008;36:237–9. 13. Japanese Gastric Cancer Association. Japanese classification of gastric carcinoma. 13th ed. Tokyo: Kanehara and Co Ltd.; 1999. 14. Krous HF, Sexauer CL. Embryonal rhabdomyosarcoma arising within a congenital bronchogenic cyst in a child. J Pediatr Surg. 1981;16:506–8. 15. Murphy JJ, Blair GK, Fraser GC, Ashmore PG, LeBlanc JG, Sett SS, et al. Rhabdomyosarcoma arising within congenital pulmonary cysts: report of three cases. J Pediatr Surg. 1992; 27:1364–7. 16. Sullivan SM, Okada S, Kudo M, Ebihara Y. A retroperitoneal bronchogenic cyst with malignant change. Pathol Int. 1999;49: 338–41. 17. Endo C, Imai T, Nakagawa H, Ebina A, Kaimori M. Bronchioloalveolar carcinoma arising in a bronchogenic cyst. Ann Thorac Surg. 2000;69:933–5. 18. Ignatius JA, Armstrong CD, Eversole SL. Case reports. Multiple diffuse cystic disease of the stomach in association with carcinoma. Gastroenterology. 1970;59:610–4. 19. Iwanaga T, Koyama H, Takahashi Y, Taniguchi H, Wada A. Diffuse submucosal cysts and carcinoma of the stomach. Cancer. 1975;36:606–14. 20. Pillay I, Petrelli M. Diffuse cystic glandular malformation of the stomach associated with adenocarcinoma: case report and review of the literature. Cancer. 1976;38:915–20. 21. Martel W, Oberman HA. Heterotopic submucosal gastric cysts: report of two cases, one in association with carcinoma. Gastrointest Radiol. 1978;3:391–5. 22. Nakamura T, Kobayashi S, Nakamura S. Diffuse submucosal cysts of the stomach with multifocal gastric carcinoma: diagnostic role of endoscopic ultrasonography. Gastrointest Endosc. 1993;39:87–9. 23. Inaba T, Mizuno M, Kawai K, Okada H, Tsuji T. Diffuse submucosal cysts of the stomach: report of two cases in association with development of multiple gastric cancers during endoscopic follow up. J Gastroenterol Hepatol. 1999;14:1161–5.
CASE REPORT
Bronchogenic cyst of the stomach involved with gastric adenocarcinoma Hiroaki Shibahara Æ Toshiyuki Arai Æ Shunpei Yokoi Æ Seijun Hayakawa
Received: 8 February 2008 / Accepted: 2 October 2008 / Published online: 2 December 2008 Ó Springer 2008
Abstract Bronchogenic cyst, a congenital anomaly mostly found in the mediastinum, rarely arises in the stomach. A 43-year-old man had epigastric pain and was diagnosed as having gastric adenocarcinoma. Abdominal ultrasonography showed hepatic cyst, and computed tomography and magnetic resonance imaging revealed a cystic lesion near the stomach. At surgery, the cystic lesion was found to be located at the lesser curvature of the stomach where the cancer invasion was seen. Total gastrectomy with combined resection of the cystic lesion was performed. Pathologically, the cyst wall was lined by pseudostratified ciliated columnar epithelium, subepithelial mixed seromucinous glands and smooth muscle bundles. The pathological diagnosis was bronchogenic cyst of the stomach involved with gastric adenocarcinoma. Based on a similar association between gastric diffuse submucosal cysts and gastric cancer in the previous reports, it is possible that chronic inflammation from bronchogenic cysts to the gastric mucosa may cause adenocarcinoma in the stomach. At surgery, complete combined resection without rupture of the bronchogenic cyst involved with the gastric adenocarcinoma is needed for treatment of gastric cancer to prevent dissemination of cancer cells considering when cancer cells have invaded beyond the pseudostratified
H. Shibahara T. Arai S. Yokoi Department of Surgery, Anjo Kosei Hospital, 28 Higashi-Hirokute, Anjo-cho, Anjo 446-8602, Japan H. Shibahara (&) Department of Surgery, Fukuroi Municipal Hospital, 2515-1 Kunou, Fukuroi 437-0061, Japan e-mail: [email protected] S. Hayakawa Department of Pathology, Anjo Kosei Hospital, Anjo, Japan
123
ciliated columnar epithelium and within the bronchogenic cyst. Keywords Bronchogenic cyst Stomach Adenocarcinoma
Introduction Bronchogenic cysts are foregut-derived developmental anomalies most commonly encountered in the mediastinum and rarely in the abdomen or retroperitoneum [1]. Bronchogenic cysts of the stomach are uncommon [2–12], and no case of bronchogenic cyst of the stomach with cancer invasion has been reported. We herein report the first case of bronchogenic cyst of the stomach involved with gastric adenocarcinoma.
Case report A 43-year-old man had epigastric pain, and gastrointestinal endoscopy in a nearby hospital revealed irregular mucosa at the cardia of the stomach. He was referred to our hospital for further examination. Abdominal ultrasonography showed hepatic cyst including internal low echoic pattern with extrahepatic growth (Fig. 1). Upper gastrointestinal series showed an irregular ulcerative lesion with compression by a cyst looking like a submucosal tumor at the lesser curvature of the cardia (Fig. 2). Endoscopy of the stomach showed an irregular mucosal lesion at the lesser curvature of the cardia (Fig. 3). Biopsy specimens of the lesion revealed group V adenocarcinoma. Computed tomography (Fig. 4) and magnetic resonance imaging (Fig. 5) revealed a cystic lesion near the stomach. Our
Clin J Gastroenterol (2009) 2:80–84
Fig. 1 Abdominal ultrasonography shows hepatic cyst (arrowhead) including internal low echoic pattern with extrahepatic growth
81
preoperative diagnoses for these lesions were gastric cancer and hepatic cyst. At surgery, the cystic lesion was found to be located at the lesser curvature of the stomach and to be invaded by the cancer. The cystic lesion had no communication with liver parenchyma. Combined resection of the cystic lesion without rupture and total gastrectomy with D2 lymph node dissection was performed. Grossly, the cystic lesion of the resected specimen was a soft mass measuring 9 9 4 cm. The gastric cancer showed a type-2 lesion compressed by the cystic lesion macroscopically (Fig. 6a). The cut surface after fixation by formalin revealed that the cystic lesion was unilocular and filled with green-colored gelatinous content, and that the gastric cancer had invaded the cystic lesion (Fig. 6b). Pathologically, the cystic lesion was located at the opposite side of the gastric wall from the adenocarcinoma (Fig. 7a). The cyst wall was lined by pseudostratified ciliated columnar epithelium (Fig. 7b) and smooth muscle bundles, which did not resemble the muscularis propria of the stomach. Mixed seromucinous glands (Fig. 7c) were identified in the subepithelial layer of the cyst. No hyaline cartilage was identified. The gastric cancer was moderately differentiated adenocarcinoma infiltrated to the subserosa with involvement in one right paracardial lymph node, which showed tubular pattern without cystic formation, and both lymphatic and vascular invasion was present. The stage was II (pT2 pN1 sH0 sP0 sM0) according to the Japanese classification of gastric carcinoma [13]. The adenocarcinoma cells invaded the cystic lesion, but not the pseudostratified ciliated columnar epithelium (Fig. 7d). The pathological diagnosis was bronchogenic cyst of the stomach invaded by the adjacent gastric adenocarcinoma. The patient was discharged uneventfully.
Discussion
Fig. 2 Upper gastrointestinal series shows irregular ulcerative lesion (arrowhead) with compression by cyst (arrows) appearing like submucosal tumor at the lesser curvature of cardia
Bronchogenic cysts are congenital anomalies arising from the ventral foregut during the 3rd to 7th week of development. Most commonly, bronchogenic cysts migrate caudally with the esophagus and are eventually found in the posterior mediastinum near the carina attached to the tracheobronchial tree or the esophagus [5]. In the present case, the bronchogenic cyst was located at the lesser curvature of the cardia. The proximity of the cyst to the esophagus indicates that the bronchogenic cyst might migrate caudally with the growing esophagus to the stomach in the development. Subdiaphragmatic bronchogenic cysts are rare, and Liang et al. [1] reported 38 cases in the English literature. They reported that the most common organ involved was the diaphragm, followed by the stomach. In our review of the literature, a search was performed using PubMed
123
82
Fig. 3 Endoscopy of stomach shows an irregular mucosal lesion at the lesser curvature of the cardia
Clin J Gastroenterol (2009) 2:80–84
Fig. 6 The gastric cancer shows a type-2 lesion (black arrowheads) compressed by the cystic lesion (white arrows) macroscopically on the fresh specimen (a). The cystic lesion (white line) is unilocular and filled with green-colored gelatinous content on the cut surface after fixation by formalin. Gastric cancer appearing as a white solid mass (arrowheads) is located at the opposite side from the cystic lesion (b)
Fig. 4 Computed tomography shows a cystic lesion (arrow) located between the liver and the stomach (arrowhead)
Fig. 5 Magnetic resonance imaging shows a cystic lesion (arrow) located between the liver and the stomach (arrowhead). L liver
123
Fig. 7 Microscopically, the cystic lesion and gastric cancer are adjacent (a). The cyst wall is lined by pseudostratified ciliated columnar epithelium (b) along the internal surface of the cystic lesion with smooth muscle bundles, and subepithelial mixed seromucinous glands (c) are identified. The pseudostratified ciliated columnar epithelium of the cystic lesion is not involved by adenocarcinoma cells (d). Pseud pseudostratified ciliated columnar epithelium, W wall of the cystic lesion, Ad adenocarcinoma of gastric cancer, Sq squamous epithelium of the esophagus
Clin J Gastroenterol (2009) 2:80–84
83
Table 1 Bronchogenic cyst of the stomach Case no.
References
Age/gender
Symptom
Location
Preoperative diagnosis
Operation
1
Keohane et al. [2]
64/Female
Nausea, vomiting and epigastric discomfort
–/Post
Multiloculated cysts
Resection
Braffman et al. [3]
64/Female
Nausea and vomiting
U/Post
Multiloculated mass
Resection
2
Matsubayashi et al. [4]
62/Male
–
–/Post
Lymphangioma/benign neurogenic tumor
Resection
3
Hedayati et al. [5]
59/Female
–
–/Post
Adrenal incidentaloma
Resection
4
Melo et al. [6]
39/Female
Rib pain
U/–
GIST
Laparoscopic resection
5
Rubio et al. [7, 8]
26/Male
Periodic epigastric pain
U/–
–
–
6
Song et al. [9]
62/Female
–
U/Less
Benign stromal tumor
Resection
7
Lee et al. [10]
38/Female
–
U/–
GIST, developmental or complicated cyst
Endoscopic mucosal resection
8
Wakabayashi et al. [11]
37/Male
Upper abdominal dull pain and dysphagia
U/Less
Duplication cyst
Laparoscopic resection
9
Sato et al. [12]
60/Female
–
U/Less
Cystic neoplasm
–
10
Shibahara et al. (present case)
43/Male
Epigastric pain
U/Less
Hepatic cyst
Resection
U upper portion of the stomach, post posterior wall of the stomach, less lesser curvature of the stomach, GIST gastrointestinal stromal tumor
(http://www.ncbi.nlm.nih.gov/sites/entrez) from 1950 to August 2008 with the following keywords: bronchogenic cyst and stomach. This search yielded 24 citations. Of these, 16 were in the English literature. Of the 16 citations, 11 were identified to be articles describing bronchogenic cyst of the stomach [2–12]. Four citations were redundant reports of two cases [2, 3, 7, 8]. There were nine cases of bronchogenic cyst of the stomach in the English literature, and they are summarized in Table 1, including the present case. Several cases of malignancy in bronchogenic cysts were reported [14–17]. However, there has been no report describing the relation of bronchogenic cyst and gastric cancer. An association between gastric diffuse submucosal cysts and gastric cancer has been reported [18–23]. Iwanaga et al. [19] examined 12 cases of the stomach with submucosal cysts, carcinoma and atypical hyperplasia, and their results showed that erosion or regenerative epithelia were evident on the gastric mucosa of all 12 cases. There was erosion at the superficial mucosa immediately above the heterotopic glands in many cases. They described that gastritis may give rise to heterotopic glands, and the development of heterotopic cysts in the submucosa may make the surface mucosa prone to erosion; repeated erosion and regeneration may cause carcinoma or atypical hyperplasia. Pillay et al. [20] reported a case of diffuse cystic glandular malformation and adenocarcinoma of the stomach, and in their case variable degrees of acute and chronic inflammatory changes in the mucosa and in the submucosa around the glands were seen. Martel et al. [21] reported a case of heterotopic submucosal gastric cysts
with adenocarcinoma. In their case, numerous large cysts were evident in the submucosa beneath the adenocarcinoma, and the invasive neoplasm was focally evident adjacent to the cysts; gastric mucosa manifested extensive and diffuse chronic inflammation with lymphocytes and focal intestinalization. No malignant epithelium was seen within the bronchogenic cyst; however, adenocarcinoma from the gastric mucosa at the opposite side of the stomach wall had invaded to the bronchogenic cyst in the present case. Based on the interpretation of diffuse submucosal cysts and cancer of the stomach described previously, it is possible that chronic inflammation from the bronchogenic cyst to the gastric mucosa may cause adenocarcinoma in the stomach. Bronchogenic cyst, for which the imaging modality does not indicate malignancy and is without symptoms, does not need resection clinically. The present case showed bronchogenic cyst involved with gastric cancer and needing to be resected. There was no report about the surgical procedure for cases with bronchogenic cyst involved with gastric cancer. The authors recommend complete combined resection of the bronchogenic cyst located near gastric cancer with gastrectomy. Furthermore, a surgical procedure without rupture of the cyst is needed to prevent dissemination of gastric cancer cells considering when cancer cells have invaded beyond the pseudostratified ciliated columnar epithelium and within the bronchogenic cyst. In conclusion, we present the first reported case of bronchogenic cyst of the stomach involved with gastric adenocarcinoma. Complete combined resection without
123
84
rupture of the bronchogenic cyst involved with gastric adenocarcinoma is needed for treatment of gastric cancer.
References 1. Liang MK, Yee HT, Song JW, Marks JL. Subdiaphragmatic bronchogenic cysts: a comprehensive review of the literature. Am Surg. 2005;71:1034–41. 2. Keohane ME, Schwartz I, Freed J, Dische R. Subdiaphragmatic bronchogenic cyst with communication to the stomach: a case report. Hum Pathol. 1988;19:868–71. 3. Braffman B, Keller R, Gendal ES, Finkel SI. Subdiaphragmatic bronchogenic cyst with gastric communication. Gastrointest Radiol. 1988;13:309–11. 4. Matsubayashi J, Ishida T, Ozawa T, Aoki T, Koyanagi Y, Mukai K. Subphrenic bronchopulmonary foregut malformation with pulmonary-sequestration-like features. Pathol Int. 2003;53:313–6. 5. Hedayati N, Cai DX, McHenry CR. Subdiaphragmatic bronchogenic cyst masquerading as an ‘‘adrenal incidentaloma’’. J Gastrointest Surg. 2003;7:802–4. 6. Melo N, Pitman MB, Rattner DW. Bronchogenic cyst of the gastric fundus presenting as a gastrointestinal stromal tumor. J Laparoendosc Adv Surg Tech A. 2005;15:163–5. 7. Rubio CA, Orrego A, Willen R. Bronchogenic gastric cyst. A case report. In Vivo. 2005;19:383–5. 8. Rubio CA, Orrego A, Wille´n R. Congenital bronchogenic cyst in the gastric mucosa. J Clin Pathol. 2005;58:335. 9. Song SY, Noh JH, Lee SJ, Son HJ. Bronchogenic cyst of the stomach masquerading as benign stromal tumor. Pathol Int. 2005;55:87–91. 10. Lee SH, Park DH, Park JH, Kim HS, Park SH, Kim SJ, et al. Endoscopic mucosal resection of a gastric bronchogenic cyst that was mimicking a solid tumor. Endoscopy. 2006;38:E12–3. 11. Wakabayashi H, Okano K, Yamamoto N, Suzuki Y, Inoue H, Kadota K, et al. Laparoscopically resected foregut duplication cyst (bronchogenic) of the stomach. Dig Dis Sci. 2007;52:1767–70.
123
Clin J Gastroenterol (2009) 2:80–84 12. Sato M, Irisawa A, Bhutani MS, Schnadig V, Takagi T, Shibukawa G, et al. Gastric bronchogenic cyst diagnosed by endosonographically guided fine needle aspiration biopsy. J Clin Ultrasound. 2008;36:237–9. 13. Japanese Gastric Cancer Association. Japanese classification of gastric carcinoma. 13th ed. Tokyo: Kanehara and Co Ltd.; 1999. 14. Krous HF, Sexauer CL. Embryonal rhabdomyosarcoma arising within a congenital bronchogenic cyst in a child. J Pediatr Surg. 1981;16:506–8. 15. Murphy JJ, Blair GK, Fraser GC, Ashmore PG, LeBlanc JG, Sett SS, et al. Rhabdomyosarcoma arising within congenital pulmonary cysts: report of three cases. J Pediatr Surg. 1992; 27:1364–7. 16. Sullivan SM, Okada S, Kudo M, Ebihara Y. A retroperitoneal bronchogenic cyst with malignant change. Pathol Int. 1999;49: 338–41. 17. Endo C, Imai T, Nakagawa H, Ebina A, Kaimori M. Bronchioloalveolar carcinoma arising in a bronchogenic cyst. Ann Thorac Surg. 2000;69:933–5. 18. Ignatius JA, Armstrong CD, Eversole SL. Case reports. Multiple diffuse cystic disease of the stomach in association with carcinoma. Gastroenterology. 1970;59:610–4. 19. Iwanaga T, Koyama H, Takahashi Y, Taniguchi H, Wada A. Diffuse submucosal cysts and carcinoma of the stomach. Cancer. 1975;36:606–14. 20. Pillay I, Petrelli M. Diffuse cystic glandular malformation of the stomach associated with adenocarcinoma: case report and review of the literature. Cancer. 1976;38:915–20. 21. Martel W, Oberman HA. Heterotopic submucosal gastric cysts: report of two cases, one in association with carcinoma. Gastrointest Radiol. 1978;3:391–5. 22. Nakamura T, Kobayashi S, Nakamura S. Diffuse submucosal cysts of the stomach with multifocal gastric carcinoma: diagnostic role of endoscopic ultrasonography. Gastrointest Endosc. 1993;39:87–9. 23. Inaba T, Mizuno M, Kawai K, Okada H, Tsuji T. Diffuse submucosal cysts of the stomach: report of two cases in association with development of multiple gastric cancers during endoscopic follow up. J Gastroenterol Hepatol. 1999;14:1161–5.
