Clin J Gastroenterol (2012) 5:82–87 DOI 10.1007/s12328-011-0275-0
CASE REPORT
A case of foregut gastric duplication cyst with pseudostratified columnar ciliated epithelium Naohiro Hosomura • Hiroshi Kono • Hiromichi Kawaida • Hidetake Amemiya Jun Itakura • Hideki Fujii
•
Received: 27 July 2011 / Accepted: 30 October 2011 / Published online: 22 December 2011 Ó Springer 2011
Abstract Gastrointestinal duplication is a congenital rare disease. Duplication cyst of the stomach with pseudostratified columnar ciliated epithelium is extremely rare. A 44-year-old Japanese woman visited University of Yamanashi Hospital for evaluation of an abnormal tumor detected by abdominal ultrasonography at an annual general health examination. Abdominal computed tomography indicated a subserosal cystic lesion 6 cm in diameter on the posterior wall of the stomach. The cystic lesion was resected through partial resection of the stomach. Histopathology showed that the cyst did not communicate with the gastric lumen, was covered with gastric epithelium and pseudostratified columnar ciliated epithelium with circular muscle layers, and did not contain cartilaginous tissue. Consequently, the patient was diagnosed as having foregut duplication cyst of the stomach. Gastrointestinal duplication can occur in any region of the gastrointestinal tract, but foregut duplication cyst of the stomach is rare. The present case was a subserosal cyst on the greater curvature that did not communicate with the gastric lumen and was covered with gastric epithelium and pseudostratified columnar ciliated epithelium, suggesting a foregut cyst caused by an aberrant respiratory organ. Keywords Gastrointestinal duplication cyst Columnar ciliated epithelium Stomach
N. Hosomura H. Kono (&) H. Kawaida H. Amemiya J. Itakura H. Fujii First Department of Surgery, Faculty of Medicine, University of Yamanashi, 1110 Shimokato, Chuo, Yamanashi 409-3898, Japan e-mail: [email protected]
123
Introduction Gastrointestinal duplications are rare and even more exceptional are those occurring in the stomach. As a general definition, a gastrointestinal duplication is a spherical hollow structure with a smooth muscle coat, lined by a mucous membrane and attached to any part of the gastrointestinal tract from the base of the tongue to the anus [1]. These malformations are believed to be congenital and are formed before differentiation of the lining epithelium and, therefore, are named after the organs with which they are associated [2]. Foregut duplications may or may not communicate with the gastrointestinal tract and are usually diagnosed at a younger age; in adults non-specific symptoms delay diagnosis, which is established during surgical exploration [3]. Here we report a case of gastric duplication cyst with respiratory epithelium.
Clinical summary A 44-year-old Japanese woman visited University of Yamanashi Hospital for evaluation of an abnormal tumor detected by abdominal ultrasonography at an annual general health examination. Past medical history, present status and physical examination were non-disease-specific. Furthermore, laboratory studies were within the normal range, and carcinoma embryonic antigen or carbohydrate antigen 19-9 was unremarkable. Abdominal computed tomography (CT) demonstrated a well-circumscribed homogenous lowdensity mass measuring 6 9 7 cm between the posterior wall of the upper third of the stomach and the tail of the pancreas (Fig. 1). Furthermore, the pancreas was strongly pressed by this cystic tumor. Magnetic resonance imaging (MRI) demonstrated a homogenous low-intensity mass on
Clin J Gastroenterol (2012) 5:82–87
83
Fig. 1 Preoperative CT showing a well-circumscribed subserosal cystic mass on the greater curvature of the stomach. a Plain CT, and b enhanced CT
Fig. 2 Preoperative MRI showing a well-circumscribed subserosal cystic mass on the greater curvature of the stomach. a T1-weighted image, and b T2-weighted image
T1-weighted imaging and a homogenous high-intensity mass on T2-weighted imaging (Fig. 2). Although upper gastrointestinal endoscopy showed a bulging deformation in the posterior wall of the upper third of the stomach (Fig. 3), it was difficult to identify the origin of this cystic tumor. From these findings, the tumor was suspected to be a benign cyst of the pancreas or a gastrointestinal stromal tumor. A laprotomy was performed and complete excision of the mass lesion with stomach wall was performed. The postoperative course was uneventful. Macroscopically, the cystic lesion was intimately associated with the subserosal posterior wall of the upper body of the stomach and measured 7.5 9 6.5 9 6.5 cm in size (Fig. 4a, b). Furthermore, the cyst did not communicate with the gastric lumen. The lining epithelium consisted of
the gastric foveolar epithelium with pyloric gland. The wall of the cyst consisted of smooth muscle and the fibrotic tissue (Fig. 4c). Cartilaginous tissue was not identified; however, gastric epithelium was identified (Fig. 4d). Furthermore, at high magnification the lining was seen to be composed of cuboidal to pseudostratified columnar (respiratory type) epithelium (Fig. 4e).
Discussion Alimentary tract duplications occur in any region from mouth to anus, and those in the terminal ileum are most common. Gastric duplications account for between 3 and 20% of gastrointestinal duplications [3] and occur twice as
123
84
often in females than in males. They are usually solitary, less than 12 cm in diameter and located along the greater curvature. Due to their position and mass effect, gastric
Fig. 3 Upper gastrointestinal endoscopy showing a bulging deformation in the posterior wall of the upper third of the stomach
Fig. 4 Subserosal cystic lesion. a Gross appearance of the resected specimen; b gross appearance of the resected specimen after fixation; c subserosal cystic wall and circular muscle layer (H&E); d gastric
123
Clin J Gastroenterol (2012) 5:82–87
duplication cysts are usually diagnosed at a younger age; however, in adults diagnosis may be difficult. A wide range of symptoms and signs have been reported and vary from asymptomatic to non-disease-specific presentations, e.g., vague abdominal complaints, nausea, vomiting, epigastric fullness, weight loss, anemia, dysphagia, dyspepsia, etc. In general, the structure is defined as tubular when the lumen is contiguous and as cystic when it is completely separate. Approximately 80% of gastric duplication cysts do not communicate with the gastric lumen, consistent with the present case [4]. Histologically, they have a gastrointestinal mucosal membrane (usually gastric) with the submucosal smooth muscle layer. Respiratory mucosa is usually found in an esophageal duplication cyst; however, it is extremely rare in the gastric duplication cyst [13]. Previously, there were twenty reported cases of adult duplication cyst of the stomach with respiratory lining epithelium (Table 1) [2, 5– 13, 19–27]. Eight cases including the present patient were shown to have both gastric epithelium and respiratory mucosa [5–8, 19–21]. The other twelve cases were shown to have pseudostratified columnar ciliated epithelium
epithelium (H&E); and e fine cilia on pseudostratified columnar ciliated epithelium (H&E)
Age
Size (cm)
F
8.
35
44
39
29
62
35
34
25
5.5 9 2.5 9 2
7.5 9 6.5 9 6.5
695
8.5 9 5.5 9 4.8
10 9 3 9 3
79695
Unknown
6.5 9 5 9 5
F
M
14.
15.
M
M
F
18.
19.
20.
M
F
13.
M
F
12.
16.
F
11.
17.
F F
9. 10.
40
30
42
76
37
48
72
46
63
39
46 61
39392
795
4.5 9 5.2
494
494
2.5 9 2 9 2
2 9 1.5
1, 8 9 5.5; 2, 3
10 9 7.6
4 9 2.5 9 1
698 2 9 1.5
Absence of gastric mucosa
M
M
6.
M
5.
7.
M
M
3.
4.
F
M
1.
2.
Presence of gastric mucosa
Sex
Dysphagia
Epigastralgia
Left lumbago
None
Abdominal pain
Abdominal pain
No complaints
Loss of consciousness
Abdominal pain
No complaints
No complaints Heart failure
No complaints
None
Abdominal pain
None
Epigastric discomfort
No complaints
No complaints
Epigastric pain, nausea
Complaints
Fundus
Cardia
Lesser curvature
Lesser curvature
Lesser curvature
Lesser curvature
Lesser curvature
1, Fundus; and 2, Gastro-splenic ligament
Posterior wall
Posterior fundus
Greater curvature Cardia, intramural
Posterior wall
Greater curvature
Fundus
Posterior wall
Lesser curvature
Greater curvature
Greater curvature
Posterior wall
Locations
Table 1 Adult gastric duplication cyst with pseudostratified columnar ciliated epithelium
N/A
Bronchogenic cyst
N/A
N/A
N/A
N/A
N/A
N/A
N/A
Squamous metaplasia
Focal squamous metaplasia Ulcer (?) cartilage
Normal
Fundic gland
Antrum type
Fundic gland, mature lung tissue
Ulcer (?)
CA19-9 staining-positive cell
Focal squamous metaplasia
N/A
Pathological findings
Itoh et al. [27]
Kizu et al. [26]
Mardi et al. [25]
Jiang et al. [24]
Wakabayashi et al. [23]
Wang et al. [22]
Murakami et al. [13]
Theodosopoulos et al. [12]
Cunningham et al. [2]
Melo et al. [11]
Gensler et al. [9] Shireman [10]
Present case
Ohta et al. [21]
Khoury and Rivera [20]
Matsubayashi et al. [19]
Kim et al. [7]
Ikehata and Sakuma [8]
Takahara et al. [6]
Laraja et al. [5]
References
Clin J Gastroenterol (2012) 5:82–87 85
123
86
without gastric epithelium [2, 9–13, 22–27]. There was only one case of subserosal cyst [6]. The site of the cyst in the other cases was continuous with the stomach wall or surrounded by smooth muscle that is continuous with the muscle of the stomach. It was difficult to explain the occurrence mechanism of a gastric duplication cyst with columnar ciliated epithelium in the stomach. The origin of most duplication cysts is still unclear. There are no established mechanisms which provide definitive explanations for all types of duplication. Gensler et al. [9] reported that the histogenesis of a duplication cyst with columnar ciliated epithelium in the stomach is derived from the anomalous laryngotracheal outgrowth, which remains attached to the portion of the primitive foregut embryo logically destined to become the stomach. The presence of respiratory epithelium in the cyst indicates that the undifferentiated epithelium of the foregut may undergo transition to differentiated, specialized epithelium during the embryonic period [7]. Duplication cysts with ciliated columnar epithelium are classified into three groups—cysts containing cartilage or seromucous respiratory glands are designated ‘bronchogenic cysts’, cysts containing two well-developed layers of smooth muscle without cartilage are designated ‘esophageal cysts’, and cysts with none of these distinguishing features are ‘foregut cysts’ [14]. Furthermore, Cunningham et al. [2] suggested that ‘gastric duplication’ implies the presence of gastric epithelium, and that when pseudostratified columnar ciliated epithelium is present in the cyst, it should be called ‘foregut duplication cyst’ of the stomach. The present case involved neither cartilaginous tissue nor the seromucous gland, nor two layers of smooth muscle. Therefore, the present case is the fifth reported case of a foregut gastric duplication cyst with pseudostratified columnar ciliated epithelium. The patient had no symptoms, and the cystic lesion was found on preoperative CT. Most patients with the gastric duplication cyst are asymptomatic and have no specific clinical sign. CT or ultrasonography indicates a cystic lesion incidentally, as in the present case [13]. Some cases include perforation, bleeding, fistula formation and obstruction [13]. Furthermore, gastrointestinal bleeding often occurs in the cystic type; this may be due to regeneration of the mucosa refashioned by intracystic pressure or chemical erosion in the cyst [15]. MRI is also useful to diagnose cysts. Furthermore, endoscopic ultrasound (EUS) is used for characterizing intramural duplication cyst and demonstrating the relationship between the cyst and the gastric wall or adjacent organ [6]. As for therapy, surgical excision is considered to be the best treatment, since some clinical case reports indicate that adenocarcinoma is arising in duplication of the stomach [16, 17]. Complete resection of the cyst is the ideal technique achieved with both open and laparoscopic surgery [11]. For a cyst communicating
123
Clin J Gastroenterol (2012) 5:82–87
with the gastric lumen, partial gastrectomy may be required. On the other hand, it was reported that surgery is not necessary if respiratory-type epithelium cells are diagnosed on EUS-guided fine-needle aspiration biopsy in gastric duplication cyst [18]. Indeed, there has been no malignancy in the previous 11 gastric duplication cysts with pseudostratified columnar ciliated epithelium. In conclusion, foregut duplication cysts of the stomach are rare entities diagnosed incidentally and usually intraoperatively in adults and should be treated surgically by complete resection. Conflict of interest of interest.
The authors declare that they have no conflict
References 1. Glaser C, Kuzinkovas V, Maurer C, Glattli A, Mouton WG, Baer HU. A large duplication cyst of the stomach in an adult presenting as pancreatic pseudocyst. Dig Surg. 1998;15:703–6. 2. Cunningham SC, Hansel DE, Fishman EK, Cameron JL. Foregut duplication cyst of the stomach. J Gastrointest Surg. 2006;10: 620–1. 3. Perek A, Perek S, Kapan M, Goksoy E. Gastric duplication cyst. Dig Surg. 2000;17:634–6. 4. Spivak H, Pascal RR, Wood WC, Hunter JG. Enteric duplication presenting as cystic tumors of the pancreas. Surgery. 1997;121: 597–600. 5. Laraja RD, Rothenberg RE, Chapman J, Imran UH, Sabatini MT. Foregut duplication cyst: a report of a case. Am Surg. 1995;61: 840–1. 6. Takahara T, Torigoe T, Haga H, Yoshida H, Takeshima S, Sano S, et al. Gastric duplication cyst: evaluation by endoscopic ultrasonography and magnetic resonance imaging. J Gastroenterol. 1996;31:420–4. 7. Kim DH, Kim JS, Nam ES, Shin HS. Foregut duplication cyst of the stomach. Pathol Int. 2000;50:142–5. 8. Ikehata A, Sakuma T. Gastric duplication cyst with markedly elevated concentration of carbohydrate antigen 19-9. Am J Gastroenterol. 2000;95:842–3. 9. Gensler S, Seidenberg B, Rifkin H, Rubinstein BM. Ciliated lined intramural cyst of the stomach: case report and suggested embryogenesis. Ann Surg. 1966;163:954–6. 10. Shireman PK. Intramural cyst of the stomach. Hum Pathol. 1987;18:857–8. 11. 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. 12. Theodosopoulos T, Marinis A, Karapanos K, Vassilikostas G, Dafnios N, Samanides L, et al. Foregut duplication cysts of the stomach with respiratory epithelium. World J Gastroenterol. 2007;13:1279–81. 13. Murakami S, Isozaki H, Shou T, Sakai K, Toyota H. Foregut duplication cyst of the stomach with pseudostratified columnar ciliated epithelium. Pathol Int. 2008;58:187–90. 14. Harvell JD, Macho JR, Klein HZ. Isolated intra-abdominal esophageal cyst. Case report and review of the literature. Am J Surg Pathol. 1996;20:476–9. 15. Gupta S, Sleeman D, Alsumait B, Abrams L. Duplication cyst of the antrum: a case report. Can J Surg. 1998;41:248–50. 16. Coit DG, Mies C. Adenocarcinoma arising within a gastric duplication cyst. J Surg Oncol. 1992;50:274–7.
Clin J Gastroenterol (2012) 5:82–87 17. Kuraoka K, Nakayama H, Kagawa T, Ichikawa T, Yasui W. Adenocarcinoma arising from a gastric duplication cyst with invasion to the stomach: a case report with literature review. J Clin Pathol. 2004;57:428–31. 18. Ponder TB, Collins BT. Fine needle aspiration biopsy of gastric duplication cysts with endoscopic ultrasound guidance. Acta Cytol. 2003;47:571–4. 19. 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. 20. Khoury T, Rivera L. Foregut duplication cysts: a report of two cases with emphasis on embryogenesis. World J Gastroenterol. 2011;17:130–4. 21. Ohta M, Konno H, Baba M, Tanaka T, Nakamura T, Nishino N, et al. A case of gastric duplication. Gastroenterol Endosc. 1999;4: 1101–6. 22. Wang B, Hunter WJ, Bin-Sagheer S, Bewtra C. Rare potential pitfall in endoscopic ultrasound-guided fine needle aspiration biopsy in gastric duplication cyst: a case report. Acta Cytol. 2009;53:219–22.
87 23. 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. 24. Jiang W, Zhang B, Fu YB, Wang JW, Gao SL, Zhang SZ, et al. Gastric duplication cyst lined by pseudostratified columnar ciliated epithelium: a case report and literature review. J Zhejiang Univ Sci B. 2011;12:28–31. 25. Mardi K, Kaushal V, Gupta S. Foregut duplication cysts of stomach masquerading as leiomyoma. Indian J Pathol Microbiol. 2010;53:160–1. 26. Kizu T, Nishida T, Tsutsui S, Yakushijin T, Egawa S, Miyazaki M, et al. Bronchogenic cyst in the abdomen—a case report. Nihon Shokakibyo Gakkai Zasshi. 2011;108:769–77. 27. Itoh K, Sudo H, Tokita H, Mukaide M, Katayanagi S, Takagi Y, et al. A case of the cyst with underdeveloped cilia on the inner side of the cystic wall directly beneath the esophagogastric junction. Jpn J Gastroenterol Surg. 2004;37:1390–4.
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CASE REPORT
A case of foregut gastric duplication cyst with pseudostratified columnar ciliated epithelium Naohiro Hosomura • Hiroshi Kono • Hiromichi Kawaida • Hidetake Amemiya Jun Itakura • Hideki Fujii
•
Received: 27 July 2011 / Accepted: 30 October 2011 / Published online: 22 December 2011 Ó Springer 2011
Abstract Gastrointestinal duplication is a congenital rare disease. Duplication cyst of the stomach with pseudostratified columnar ciliated epithelium is extremely rare. A 44-year-old Japanese woman visited University of Yamanashi Hospital for evaluation of an abnormal tumor detected by abdominal ultrasonography at an annual general health examination. Abdominal computed tomography indicated a subserosal cystic lesion 6 cm in diameter on the posterior wall of the stomach. The cystic lesion was resected through partial resection of the stomach. Histopathology showed that the cyst did not communicate with the gastric lumen, was covered with gastric epithelium and pseudostratified columnar ciliated epithelium with circular muscle layers, and did not contain cartilaginous tissue. Consequently, the patient was diagnosed as having foregut duplication cyst of the stomach. Gastrointestinal duplication can occur in any region of the gastrointestinal tract, but foregut duplication cyst of the stomach is rare. The present case was a subserosal cyst on the greater curvature that did not communicate with the gastric lumen and was covered with gastric epithelium and pseudostratified columnar ciliated epithelium, suggesting a foregut cyst caused by an aberrant respiratory organ. Keywords Gastrointestinal duplication cyst Columnar ciliated epithelium Stomach
N. Hosomura H. Kono (&) H. Kawaida H. Amemiya J. Itakura H. Fujii First Department of Surgery, Faculty of Medicine, University of Yamanashi, 1110 Shimokato, Chuo, Yamanashi 409-3898, Japan e-mail: [email protected]
123
Introduction Gastrointestinal duplications are rare and even more exceptional are those occurring in the stomach. As a general definition, a gastrointestinal duplication is a spherical hollow structure with a smooth muscle coat, lined by a mucous membrane and attached to any part of the gastrointestinal tract from the base of the tongue to the anus [1]. These malformations are believed to be congenital and are formed before differentiation of the lining epithelium and, therefore, are named after the organs with which they are associated [2]. Foregut duplications may or may not communicate with the gastrointestinal tract and are usually diagnosed at a younger age; in adults non-specific symptoms delay diagnosis, which is established during surgical exploration [3]. Here we report a case of gastric duplication cyst with respiratory epithelium.
Clinical summary A 44-year-old Japanese woman visited University of Yamanashi Hospital for evaluation of an abnormal tumor detected by abdominal ultrasonography at an annual general health examination. Past medical history, present status and physical examination were non-disease-specific. Furthermore, laboratory studies were within the normal range, and carcinoma embryonic antigen or carbohydrate antigen 19-9 was unremarkable. Abdominal computed tomography (CT) demonstrated a well-circumscribed homogenous lowdensity mass measuring 6 9 7 cm between the posterior wall of the upper third of the stomach and the tail of the pancreas (Fig. 1). Furthermore, the pancreas was strongly pressed by this cystic tumor. Magnetic resonance imaging (MRI) demonstrated a homogenous low-intensity mass on
Clin J Gastroenterol (2012) 5:82–87
83
Fig. 1 Preoperative CT showing a well-circumscribed subserosal cystic mass on the greater curvature of the stomach. a Plain CT, and b enhanced CT
Fig. 2 Preoperative MRI showing a well-circumscribed subserosal cystic mass on the greater curvature of the stomach. a T1-weighted image, and b T2-weighted image
T1-weighted imaging and a homogenous high-intensity mass on T2-weighted imaging (Fig. 2). Although upper gastrointestinal endoscopy showed a bulging deformation in the posterior wall of the upper third of the stomach (Fig. 3), it was difficult to identify the origin of this cystic tumor. From these findings, the tumor was suspected to be a benign cyst of the pancreas or a gastrointestinal stromal tumor. A laprotomy was performed and complete excision of the mass lesion with stomach wall was performed. The postoperative course was uneventful. Macroscopically, the cystic lesion was intimately associated with the subserosal posterior wall of the upper body of the stomach and measured 7.5 9 6.5 9 6.5 cm in size (Fig. 4a, b). Furthermore, the cyst did not communicate with the gastric lumen. The lining epithelium consisted of
the gastric foveolar epithelium with pyloric gland. The wall of the cyst consisted of smooth muscle and the fibrotic tissue (Fig. 4c). Cartilaginous tissue was not identified; however, gastric epithelium was identified (Fig. 4d). Furthermore, at high magnification the lining was seen to be composed of cuboidal to pseudostratified columnar (respiratory type) epithelium (Fig. 4e).
Discussion Alimentary tract duplications occur in any region from mouth to anus, and those in the terminal ileum are most common. Gastric duplications account for between 3 and 20% of gastrointestinal duplications [3] and occur twice as
123
84
often in females than in males. They are usually solitary, less than 12 cm in diameter and located along the greater curvature. Due to their position and mass effect, gastric
Fig. 3 Upper gastrointestinal endoscopy showing a bulging deformation in the posterior wall of the upper third of the stomach
Fig. 4 Subserosal cystic lesion. a Gross appearance of the resected specimen; b gross appearance of the resected specimen after fixation; c subserosal cystic wall and circular muscle layer (H&E); d gastric
123
Clin J Gastroenterol (2012) 5:82–87
duplication cysts are usually diagnosed at a younger age; however, in adults diagnosis may be difficult. A wide range of symptoms and signs have been reported and vary from asymptomatic to non-disease-specific presentations, e.g., vague abdominal complaints, nausea, vomiting, epigastric fullness, weight loss, anemia, dysphagia, dyspepsia, etc. In general, the structure is defined as tubular when the lumen is contiguous and as cystic when it is completely separate. Approximately 80% of gastric duplication cysts do not communicate with the gastric lumen, consistent with the present case [4]. Histologically, they have a gastrointestinal mucosal membrane (usually gastric) with the submucosal smooth muscle layer. Respiratory mucosa is usually found in an esophageal duplication cyst; however, it is extremely rare in the gastric duplication cyst [13]. Previously, there were twenty reported cases of adult duplication cyst of the stomach with respiratory lining epithelium (Table 1) [2, 5– 13, 19–27]. Eight cases including the present patient were shown to have both gastric epithelium and respiratory mucosa [5–8, 19–21]. The other twelve cases were shown to have pseudostratified columnar ciliated epithelium
epithelium (H&E); and e fine cilia on pseudostratified columnar ciliated epithelium (H&E)
Age
Size (cm)
F
8.
35
44
39
29
62
35
34
25
5.5 9 2.5 9 2
7.5 9 6.5 9 6.5
695
8.5 9 5.5 9 4.8
10 9 3 9 3
79695
Unknown
6.5 9 5 9 5
F
M
14.
15.
M
M
F
18.
19.
20.
M
F
13.
M
F
12.
16.
F
11.
17.
F F
9. 10.
40
30
42
76
37
48
72
46
63
39
46 61
39392
795
4.5 9 5.2
494
494
2.5 9 2 9 2
2 9 1.5
1, 8 9 5.5; 2, 3
10 9 7.6
4 9 2.5 9 1
698 2 9 1.5
Absence of gastric mucosa
M
M
6.
M
5.
7.
M
M
3.
4.
F
M
1.
2.
Presence of gastric mucosa
Sex
Dysphagia
Epigastralgia
Left lumbago
None
Abdominal pain
Abdominal pain
No complaints
Loss of consciousness
Abdominal pain
No complaints
No complaints Heart failure
No complaints
None
Abdominal pain
None
Epigastric discomfort
No complaints
No complaints
Epigastric pain, nausea
Complaints
Fundus
Cardia
Lesser curvature
Lesser curvature
Lesser curvature
Lesser curvature
Lesser curvature
1, Fundus; and 2, Gastro-splenic ligament
Posterior wall
Posterior fundus
Greater curvature Cardia, intramural
Posterior wall
Greater curvature
Fundus
Posterior wall
Lesser curvature
Greater curvature
Greater curvature
Posterior wall
Locations
Table 1 Adult gastric duplication cyst with pseudostratified columnar ciliated epithelium
N/A
Bronchogenic cyst
N/A
N/A
N/A
N/A
N/A
N/A
N/A
Squamous metaplasia
Focal squamous metaplasia Ulcer (?) cartilage
Normal
Fundic gland
Antrum type
Fundic gland, mature lung tissue
Ulcer (?)
CA19-9 staining-positive cell
Focal squamous metaplasia
N/A
Pathological findings
Itoh et al. [27]
Kizu et al. [26]
Mardi et al. [25]
Jiang et al. [24]
Wakabayashi et al. [23]
Wang et al. [22]
Murakami et al. [13]
Theodosopoulos et al. [12]
Cunningham et al. [2]
Melo et al. [11]
Gensler et al. [9] Shireman [10]
Present case
Ohta et al. [21]
Khoury and Rivera [20]
Matsubayashi et al. [19]
Kim et al. [7]
Ikehata and Sakuma [8]
Takahara et al. [6]
Laraja et al. [5]
References
Clin J Gastroenterol (2012) 5:82–87 85
123
86
without gastric epithelium [2, 9–13, 22–27]. There was only one case of subserosal cyst [6]. The site of the cyst in the other cases was continuous with the stomach wall or surrounded by smooth muscle that is continuous with the muscle of the stomach. It was difficult to explain the occurrence mechanism of a gastric duplication cyst with columnar ciliated epithelium in the stomach. The origin of most duplication cysts is still unclear. There are no established mechanisms which provide definitive explanations for all types of duplication. Gensler et al. [9] reported that the histogenesis of a duplication cyst with columnar ciliated epithelium in the stomach is derived from the anomalous laryngotracheal outgrowth, which remains attached to the portion of the primitive foregut embryo logically destined to become the stomach. The presence of respiratory epithelium in the cyst indicates that the undifferentiated epithelium of the foregut may undergo transition to differentiated, specialized epithelium during the embryonic period [7]. Duplication cysts with ciliated columnar epithelium are classified into three groups—cysts containing cartilage or seromucous respiratory glands are designated ‘bronchogenic cysts’, cysts containing two well-developed layers of smooth muscle without cartilage are designated ‘esophageal cysts’, and cysts with none of these distinguishing features are ‘foregut cysts’ [14]. Furthermore, Cunningham et al. [2] suggested that ‘gastric duplication’ implies the presence of gastric epithelium, and that when pseudostratified columnar ciliated epithelium is present in the cyst, it should be called ‘foregut duplication cyst’ of the stomach. The present case involved neither cartilaginous tissue nor the seromucous gland, nor two layers of smooth muscle. Therefore, the present case is the fifth reported case of a foregut gastric duplication cyst with pseudostratified columnar ciliated epithelium. The patient had no symptoms, and the cystic lesion was found on preoperative CT. Most patients with the gastric duplication cyst are asymptomatic and have no specific clinical sign. CT or ultrasonography indicates a cystic lesion incidentally, as in the present case [13]. Some cases include perforation, bleeding, fistula formation and obstruction [13]. Furthermore, gastrointestinal bleeding often occurs in the cystic type; this may be due to regeneration of the mucosa refashioned by intracystic pressure or chemical erosion in the cyst [15]. MRI is also useful to diagnose cysts. Furthermore, endoscopic ultrasound (EUS) is used for characterizing intramural duplication cyst and demonstrating the relationship between the cyst and the gastric wall or adjacent organ [6]. As for therapy, surgical excision is considered to be the best treatment, since some clinical case reports indicate that adenocarcinoma is arising in duplication of the stomach [16, 17]. Complete resection of the cyst is the ideal technique achieved with both open and laparoscopic surgery [11]. For a cyst communicating
123
Clin J Gastroenterol (2012) 5:82–87
with the gastric lumen, partial gastrectomy may be required. On the other hand, it was reported that surgery is not necessary if respiratory-type epithelium cells are diagnosed on EUS-guided fine-needle aspiration biopsy in gastric duplication cyst [18]. Indeed, there has been no malignancy in the previous 11 gastric duplication cysts with pseudostratified columnar ciliated epithelium. In conclusion, foregut duplication cysts of the stomach are rare entities diagnosed incidentally and usually intraoperatively in adults and should be treated surgically by complete resection. Conflict of interest of interest.
The authors declare that they have no conflict
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