545395
research-article2014
IJSXXX10.1177/1066896914545395International Journal of Surgical PathologyJung et al
Case Report
Gastric Inverted Hyperplasic Polyp Composed Only of Pyloric Glands: A Rare Case Report and Review of the Literature
International Journal of Surgical Pathology 2015, Vol. 23(4) 313–316 © The Author(s) 2014 Reprints and permissions: sagepub.com/journalsPermissions.nav DOI: 10.1177/1066896914545395 ijs.sagepub.com
Minsun Jung, MD1, Kyueng-Whan Min, MD2, and Young-Joon Ryu, MD, PhD1
Abstract Inverted hyperplastic polyp (IHP) in stomach is a rare benign gastric polypoid lesion, characterized by downward growth of hyperplastic mucosal glands into the submucosal layer. In most previous reported cases, gastric IHP showed mixtures of fundic-type gland, pyloric-type gland, and foveolar-type epithelium. Also, a case of IHP composed of only one type of gland is extremely rare. This report describes a case of a 70-year-old man with gastric IHP, composed only of pylorictype gland. It was removed completely by endoscopic submucosal dissection, and patient showed no recurrence over 2 years after treatment. Keywords stomach, hyperplasia, polyps, pylorus, humans
Introduction Gastric inverted hyperplastic polyp (IHP) is a rare gastric polyp, characterized by the downward growth of hyperplastic mucosal components into the submucosal layer. It has also been called as hamartomatous polyp1 and solitary polypoidhamartoma.2 To our knowledge, there are only 12 reports in the English literature,1-12 and only 1 report of gastric IHP composed of pure pyloric-type glands.4 Gastric IHP is usually treated by endoscopic submucosal dissection. It has clinical importance, because it can be misdiagnosed as submucosal tumor or malignant neoplasm, and adenocarcinoma can likely form in benign gastric IHP.8,9,11,12 Therefore, it is difficult and challenging for preoperative diagnosis. In this report, we describe endoscopic, gross, histological findings, and immunohistochemical expression of a rare gastric IHP, only composed of pyloric glands.
Case Report A 70-year-old man with long-standing epigastric discomfort underwent gastric endoscopy. He was otherwise healthy and had no notable medical records of any previous malignant disease. The endoscopic examination revealed a lesion in the mid-body posterior wall side of the stomach. The lesion exhibited a centrally depressed and peripherally elevated appearance. It also exhibited mild
erosion and hemorrhaging on the surface area (Figure 1A). Suspecting malignancy, endoscopic submucosal dissection (ESD) was performed. Gross examination revealed a lesion measuring 1.6 × 1.5 cm in width and 0.4 cm in depth (Figure 1B). On microscopic examination, the lesion showed well-circumscribed inverted growth pattern into the submucosal layer (Figure 2A). It only consisted of homogenous pyloric-type glands without any other glandular epithelium. The lesion showed closely packed type glands with a monolayer of cuboidal to low columnar epithelial cells showing a bland-looking round nuclei and pale to eosinophilic ground glass cytoplasm (Figure 2B). No architectural or cytological atypia was found. Immunohistochemical staining for MUC-5AC and MUC-6 were performed to define gastric gland mucosal cells.13 All composing glands were evenly positive to MUC-5AC and MUC-6 (Figure 2C and D). On staining for Ki-67 and p53, no glandular cells expressed immunopositivity. The patient was discharged after ESD and exhibited no recurrence during 2 years. 1
Kangwon National University, Chuncheon, Korea KangBuk Samsung Hospital, Seoul, Korea
2
Corresponding Author: Young-Joon Ryu, Department of Pathology, College of Medicine, Kangwon National University, Hyo-Ja-3-Dong, Chuncheon, Korea. Email: [email protected]
314
International Journal of Surgical Pathology 23(4)
Figure 1. Endoscopic and gross features of gastric inverted hyperplastic polyp.
(A) Centrally depressed with erosion and peripherally elevated appearance. (B) Postendoscopic submucosal dissection status. The lesion measures 1.6 × 1.5 × 0.4 cm.
Figure 2. Histopathologic findings of gastric inverted hyperplastic polyp.
(A) Low-power view (H&E stain, 12.5×). The lesion shows inverted growth pattern with surface erosion. (B) High-power view. Pyloric-type glands are lined by closely packed tubular cells (H&E stain, 400×). (C) Immunohistochemical stain to MUC-5A (200×) and (D) MUC-6 (100×).
315
Jung et al Table 1. Clinical and Histological Characteristic of Previously Reported Gastric Inverted Hyperplastic Polyp in the English Literature. Histologic Findings Reported Year and Reference
Age (Years)/ Gender
Location
Size (cm)
Foveolar-Type
1982 19821 19832
85/female 51/female 47/female
Antrum Antrum Fundus
5 × 11 4×6 0.9 × 1.3
+ + +
+
+
19873 19934
66/female 79/male
Fundus Body
1.5 × 1.5 2.5 × 1.5
+ +
+ −
+ +
19985 20026 20026 20026 20026 20047 20078 20079 200810 201211 201312
41/female 69/male 58/male 34/female 81/male 43/female 54/male 59/male 37/male 40/female 77/male
Fundus Body Cardia Body Fundus Body Antrum Body Fundus Body Body
2.3 × 1.8 × 0.9 1 × 0.9 2.6 × 2.2 3×3 0.5 × 0.5 2.8 × 2.8 4.5 × 3.5 ND 2.5 × 2.5 3.5 × 3.2 × 1.8 4.5 × 3 × 0.5
+ + + + + + + + + + ND
− − − − − ND
+ + + − + +
1
Fundic-Type
Pyloric-Type
ND ND
ND + − − −
+ + − ND
Associated Findings Intestinal metaplasia Intestinal metaplasia, AC Parietal-like cell ECL cell, AC, GCP ECL cell ECL cell ECL cell, AC, GCP AC AC Parietal-like cell AC, SC AC, GCP
Abbreviations: AC, adenocarcinoma; ECL cell, enterochromaffin-like cell; GCP, gastritis cysticaprofunda; GIST, gastrointestinal stromal tumor; SC, signet ring cell carcinoma, ND; no description.
Discussion Gastric polyps can be classified according to morphology as “protruding type” or “inverted type.” Most gastric polyps are of the protruding type, and the inverted type is a rare and is characterized by marked submucosal glandular proliferation. So far, 13 articles of benign gastric IHP have been reported (Table 1). In most reported IHP cases, it shows a mixture of foveolar-type epithelium and various gastric glands, including pyloric-type or fundic-type glands. In some cases, smooth muscle bundles, intestinal metaplasia, and enterochromaffin-like (ECL) cells were noted.6 The mean age was 59.5 years (range = 34-85 years), with significance between genders (males– females = 1.25:1). Eight polyps were located in the fundus of the stomach, 5 polyps in the body and 3 polyps in the antrum; and only 1 polyp was located in the cardia. The size of gastric IHPs varied between 0.5 and 11 cm. Glandular components of gastric IHPs were composed of fundic-type gland in 3 cases, pyloric-type gland in 12 cases, and foveolar-type epithelium in all cases. Gastric adenocarcinoma was reported in 7 cases in 4 articles, and of those, signet ring cell carcinoma was found simultaneously in 1 case (Table 1). Most cases of gastric IHP is often asymptomatic and found incidentally. However, it may occasionally manifest as an intestinal obstruction or as an anemia secondary to chronic bleeding.7 It is reported to be related with
dysplasia and even adenocarcinoma. Therefore, all IHPs should be completely removed and examined thoroughly. Due to its noninvasive and economic benefits, ESD is recommended in treating gastric IHP.12 Although the pathogenesis of gastric IHP is unclear, recent studies have suggested that an inflammation and subsequent healing may displace the mucosal component into the submucosal layer.8,11 In the present case, the gastric IHP consisted of pure pyloric-type glands without foveolar-type epithelium or any other glandular epithelium. Although foveolar-type epithelium is one of the major components of gastric IHP, and is contained in all gastric IHPs, its relationship to pathogenesis is still unclear. In summary, we have described a case of gastric IHP present in a 70-year-old male patient. The polyp consisted only of pyloric-type glands without foveolar-type epithelium. This is the second report of gastric IHP comprised only of pyloric-type glands. Declaration of Conflicting Interests The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.
Funding The author(s) disclosed receipt of the following financial support for the research, authorship, and/or publication of this article:
316 This report was supported by 2013 Research Grant of Kangwon National University. The control tissue for this study was provided by the Kangwon National University Hospital Biobank, a member of the National Biobank of Korea, which is supported by the Ministry of Health, Welfare and Family Affairs.
References 1. Katz LB, Tenembaum MM, Kreel I: Gastric hamartomatous polyps in the absence of familial polyposis: report of two cases. Mt Sinai J Med 49: 426-429, 1982. 2. Hanada M, Takami M, Hirata K, Kishi T, Nakajima T: Hyperplastic fundic gland polyp of the stomach. Acta Pathol Jpn 33: 1269-1277, 1983. 3. Carfagna G, Pilato FP, Bordi C, Barsotti P, Riva C: Solitary polypoid hamartoma of the oxyntic mucosa of the stomach. Path Res Pract 182: 326-330, 1987. 4. Kamata Y, Kuruotaki H, Onodera T, Nishida N: An unusual heterotopia of pyloric glands of the stomach with inverted downgrowth. Acta Pathol Jpn 43: 192-197, 1993. 5. Itoh K, Tsuchigame T, Matsukawa T, Takahashi M, Honma K, Ishimaru Y: Unusual gastric polyp showing submucosal proliferation of glands: Case report and literature review. J Gastroenterol 33: 720 -723, 1998. 6. Yamashita M, Hirokawa M, Nakasono M, Kiyoku H, Sano N, Fujii M, Koyama T, Yoshida S, Sano T: Gastric inverted hyperplastic polyp. Report of four cases and relation to gastritis cystic profunda. APMIS 110: 717 -723, 2002. 7. Aoki M, Yoshida M, Saikawa Y, Otani Y, Kubota T, Kumai K, Wakabayashi G, Omori T, Mukai M, Kitajima M:
International Journal of Surgical Pathology 23(4) Diagnosis and treatment of a gastric hamartomatous inverted polyp: report of a case. Surg Today 34: 532 -536, 2004. 8. Kono T, Imai Y, Ichihara T, Miyagawa K, Kanemitsu K, Ajiki T, Kawasaki K, Kamigaki T, Ikuta H, Ohbayashi C, Yokozaki H, Fujimori T, Kuroda Y: Adenocarcinoma arising in gastric inverted hyperplastic polyp: a case report and review of the literature. Pathol Res Pract 203: 53-56, 2007. 9. Ono S, Kamoshida T, Hiroshima Y, Okawara A, Matsuo T, Kakinoki N, Ishikawa A, Kishimoto Y, Hirai S, Oka Y, Shimokama T: A case of early gastric cancer accompanied by a hamartomatous inverted polyp and successfully managed with endoscopic submucosal dissection. Endoscopy 39: E202, 2007. 10. Odashima M, Otaka M, Nanjo H, Jin M, Horikawa Y, Matsuhashi T, Ohba R, Koizumi S, Kinoshita N, Takahashi T, Shima H, Watanabe S: Hamartomatous inverted polyp successfully treated by endoscopic submucosal dissection. Intern Med 47: 259-262, 2008. 11. Kim HS, Hwang EJ, Jang JY, Lee JH, Kim YW: Multifocal adenocarcinomas arising within a gastric inverted hyperplastic polyp. Korean J Pathol 46: 387-391, 2012. 12. Lee SJ, Park JK, Seo HI, Han KH, Kim YD, Jeong WJ, Cheon GJ, Eom DW: A case of gastric inverted hyperplastic polyp found with gastritis cystica profunda and early gastric cancer. Clin Endosc 46: 568-571, 2013. 13. Ho SB, Takamura K, Anway R, Shekels LL, Toribara NW, Ota H: The adherent gastric mucous layer is composed of alternating layers of MUC5AC and MUC6 mucin proteins. Dig Dis Sci 49: 1598-1606, 2004.
Copyright of International Journal of Surgical Pathology is the property of Sage Publications Inc. and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use.
research-article2014
IJSXXX10.1177/1066896914545395International Journal of Surgical PathologyJung et al
Case Report
Gastric Inverted Hyperplasic Polyp Composed Only of Pyloric Glands: A Rare Case Report and Review of the Literature
International Journal of Surgical Pathology 2015, Vol. 23(4) 313–316 © The Author(s) 2014 Reprints and permissions: sagepub.com/journalsPermissions.nav DOI: 10.1177/1066896914545395 ijs.sagepub.com
Minsun Jung, MD1, Kyueng-Whan Min, MD2, and Young-Joon Ryu, MD, PhD1
Abstract Inverted hyperplastic polyp (IHP) in stomach is a rare benign gastric polypoid lesion, characterized by downward growth of hyperplastic mucosal glands into the submucosal layer. In most previous reported cases, gastric IHP showed mixtures of fundic-type gland, pyloric-type gland, and foveolar-type epithelium. Also, a case of IHP composed of only one type of gland is extremely rare. This report describes a case of a 70-year-old man with gastric IHP, composed only of pylorictype gland. It was removed completely by endoscopic submucosal dissection, and patient showed no recurrence over 2 years after treatment. Keywords stomach, hyperplasia, polyps, pylorus, humans
Introduction Gastric inverted hyperplastic polyp (IHP) is a rare gastric polyp, characterized by the downward growth of hyperplastic mucosal components into the submucosal layer. It has also been called as hamartomatous polyp1 and solitary polypoidhamartoma.2 To our knowledge, there are only 12 reports in the English literature,1-12 and only 1 report of gastric IHP composed of pure pyloric-type glands.4 Gastric IHP is usually treated by endoscopic submucosal dissection. It has clinical importance, because it can be misdiagnosed as submucosal tumor or malignant neoplasm, and adenocarcinoma can likely form in benign gastric IHP.8,9,11,12 Therefore, it is difficult and challenging for preoperative diagnosis. In this report, we describe endoscopic, gross, histological findings, and immunohistochemical expression of a rare gastric IHP, only composed of pyloric glands.
Case Report A 70-year-old man with long-standing epigastric discomfort underwent gastric endoscopy. He was otherwise healthy and had no notable medical records of any previous malignant disease. The endoscopic examination revealed a lesion in the mid-body posterior wall side of the stomach. The lesion exhibited a centrally depressed and peripherally elevated appearance. It also exhibited mild
erosion and hemorrhaging on the surface area (Figure 1A). Suspecting malignancy, endoscopic submucosal dissection (ESD) was performed. Gross examination revealed a lesion measuring 1.6 × 1.5 cm in width and 0.4 cm in depth (Figure 1B). On microscopic examination, the lesion showed well-circumscribed inverted growth pattern into the submucosal layer (Figure 2A). It only consisted of homogenous pyloric-type glands without any other glandular epithelium. The lesion showed closely packed type glands with a monolayer of cuboidal to low columnar epithelial cells showing a bland-looking round nuclei and pale to eosinophilic ground glass cytoplasm (Figure 2B). No architectural or cytological atypia was found. Immunohistochemical staining for MUC-5AC and MUC-6 were performed to define gastric gland mucosal cells.13 All composing glands were evenly positive to MUC-5AC and MUC-6 (Figure 2C and D). On staining for Ki-67 and p53, no glandular cells expressed immunopositivity. The patient was discharged after ESD and exhibited no recurrence during 2 years. 1
Kangwon National University, Chuncheon, Korea KangBuk Samsung Hospital, Seoul, Korea
2
Corresponding Author: Young-Joon Ryu, Department of Pathology, College of Medicine, Kangwon National University, Hyo-Ja-3-Dong, Chuncheon, Korea. Email: [email protected]
314
International Journal of Surgical Pathology 23(4)
Figure 1. Endoscopic and gross features of gastric inverted hyperplastic polyp.
(A) Centrally depressed with erosion and peripherally elevated appearance. (B) Postendoscopic submucosal dissection status. The lesion measures 1.6 × 1.5 × 0.4 cm.
Figure 2. Histopathologic findings of gastric inverted hyperplastic polyp.
(A) Low-power view (H&E stain, 12.5×). The lesion shows inverted growth pattern with surface erosion. (B) High-power view. Pyloric-type glands are lined by closely packed tubular cells (H&E stain, 400×). (C) Immunohistochemical stain to MUC-5A (200×) and (D) MUC-6 (100×).
315
Jung et al Table 1. Clinical and Histological Characteristic of Previously Reported Gastric Inverted Hyperplastic Polyp in the English Literature. Histologic Findings Reported Year and Reference
Age (Years)/ Gender
Location
Size (cm)
Foveolar-Type
1982 19821 19832
85/female 51/female 47/female
Antrum Antrum Fundus
5 × 11 4×6 0.9 × 1.3
+ + +
+
+
19873 19934
66/female 79/male
Fundus Body
1.5 × 1.5 2.5 × 1.5
+ +
+ −
+ +
19985 20026 20026 20026 20026 20047 20078 20079 200810 201211 201312
41/female 69/male 58/male 34/female 81/male 43/female 54/male 59/male 37/male 40/female 77/male
Fundus Body Cardia Body Fundus Body Antrum Body Fundus Body Body
2.3 × 1.8 × 0.9 1 × 0.9 2.6 × 2.2 3×3 0.5 × 0.5 2.8 × 2.8 4.5 × 3.5 ND 2.5 × 2.5 3.5 × 3.2 × 1.8 4.5 × 3 × 0.5
+ + + + + + + + + + ND
− − − − − ND
+ + + − + +
1
Fundic-Type
Pyloric-Type
ND ND
ND + − − −
+ + − ND
Associated Findings Intestinal metaplasia Intestinal metaplasia, AC Parietal-like cell ECL cell, AC, GCP ECL cell ECL cell ECL cell, AC, GCP AC AC Parietal-like cell AC, SC AC, GCP
Abbreviations: AC, adenocarcinoma; ECL cell, enterochromaffin-like cell; GCP, gastritis cysticaprofunda; GIST, gastrointestinal stromal tumor; SC, signet ring cell carcinoma, ND; no description.
Discussion Gastric polyps can be classified according to morphology as “protruding type” or “inverted type.” Most gastric polyps are of the protruding type, and the inverted type is a rare and is characterized by marked submucosal glandular proliferation. So far, 13 articles of benign gastric IHP have been reported (Table 1). In most reported IHP cases, it shows a mixture of foveolar-type epithelium and various gastric glands, including pyloric-type or fundic-type glands. In some cases, smooth muscle bundles, intestinal metaplasia, and enterochromaffin-like (ECL) cells were noted.6 The mean age was 59.5 years (range = 34-85 years), with significance between genders (males– females = 1.25:1). Eight polyps were located in the fundus of the stomach, 5 polyps in the body and 3 polyps in the antrum; and only 1 polyp was located in the cardia. The size of gastric IHPs varied between 0.5 and 11 cm. Glandular components of gastric IHPs were composed of fundic-type gland in 3 cases, pyloric-type gland in 12 cases, and foveolar-type epithelium in all cases. Gastric adenocarcinoma was reported in 7 cases in 4 articles, and of those, signet ring cell carcinoma was found simultaneously in 1 case (Table 1). Most cases of gastric IHP is often asymptomatic and found incidentally. However, it may occasionally manifest as an intestinal obstruction or as an anemia secondary to chronic bleeding.7 It is reported to be related with
dysplasia and even adenocarcinoma. Therefore, all IHPs should be completely removed and examined thoroughly. Due to its noninvasive and economic benefits, ESD is recommended in treating gastric IHP.12 Although the pathogenesis of gastric IHP is unclear, recent studies have suggested that an inflammation and subsequent healing may displace the mucosal component into the submucosal layer.8,11 In the present case, the gastric IHP consisted of pure pyloric-type glands without foveolar-type epithelium or any other glandular epithelium. Although foveolar-type epithelium is one of the major components of gastric IHP, and is contained in all gastric IHPs, its relationship to pathogenesis is still unclear. In summary, we have described a case of gastric IHP present in a 70-year-old male patient. The polyp consisted only of pyloric-type glands without foveolar-type epithelium. This is the second report of gastric IHP comprised only of pyloric-type glands. Declaration of Conflicting Interests The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.
Funding The author(s) disclosed receipt of the following financial support for the research, authorship, and/or publication of this article:
316 This report was supported by 2013 Research Grant of Kangwon National University. The control tissue for this study was provided by the Kangwon National University Hospital Biobank, a member of the National Biobank of Korea, which is supported by the Ministry of Health, Welfare and Family Affairs.
References 1. Katz LB, Tenembaum MM, Kreel I: Gastric hamartomatous polyps in the absence of familial polyposis: report of two cases. Mt Sinai J Med 49: 426-429, 1982. 2. Hanada M, Takami M, Hirata K, Kishi T, Nakajima T: Hyperplastic fundic gland polyp of the stomach. Acta Pathol Jpn 33: 1269-1277, 1983. 3. Carfagna G, Pilato FP, Bordi C, Barsotti P, Riva C: Solitary polypoid hamartoma of the oxyntic mucosa of the stomach. Path Res Pract 182: 326-330, 1987. 4. Kamata Y, Kuruotaki H, Onodera T, Nishida N: An unusual heterotopia of pyloric glands of the stomach with inverted downgrowth. Acta Pathol Jpn 43: 192-197, 1993. 5. Itoh K, Tsuchigame T, Matsukawa T, Takahashi M, Honma K, Ishimaru Y: Unusual gastric polyp showing submucosal proliferation of glands: Case report and literature review. J Gastroenterol 33: 720 -723, 1998. 6. Yamashita M, Hirokawa M, Nakasono M, Kiyoku H, Sano N, Fujii M, Koyama T, Yoshida S, Sano T: Gastric inverted hyperplastic polyp. Report of four cases and relation to gastritis cystic profunda. APMIS 110: 717 -723, 2002. 7. Aoki M, Yoshida M, Saikawa Y, Otani Y, Kubota T, Kumai K, Wakabayashi G, Omori T, Mukai M, Kitajima M:
International Journal of Surgical Pathology 23(4) Diagnosis and treatment of a gastric hamartomatous inverted polyp: report of a case. Surg Today 34: 532 -536, 2004. 8. Kono T, Imai Y, Ichihara T, Miyagawa K, Kanemitsu K, Ajiki T, Kawasaki K, Kamigaki T, Ikuta H, Ohbayashi C, Yokozaki H, Fujimori T, Kuroda Y: Adenocarcinoma arising in gastric inverted hyperplastic polyp: a case report and review of the literature. Pathol Res Pract 203: 53-56, 2007. 9. Ono S, Kamoshida T, Hiroshima Y, Okawara A, Matsuo T, Kakinoki N, Ishikawa A, Kishimoto Y, Hirai S, Oka Y, Shimokama T: A case of early gastric cancer accompanied by a hamartomatous inverted polyp and successfully managed with endoscopic submucosal dissection. Endoscopy 39: E202, 2007. 10. Odashima M, Otaka M, Nanjo H, Jin M, Horikawa Y, Matsuhashi T, Ohba R, Koizumi S, Kinoshita N, Takahashi T, Shima H, Watanabe S: Hamartomatous inverted polyp successfully treated by endoscopic submucosal dissection. Intern Med 47: 259-262, 2008. 11. Kim HS, Hwang EJ, Jang JY, Lee JH, Kim YW: Multifocal adenocarcinomas arising within a gastric inverted hyperplastic polyp. Korean J Pathol 46: 387-391, 2012. 12. Lee SJ, Park JK, Seo HI, Han KH, Kim YD, Jeong WJ, Cheon GJ, Eom DW: A case of gastric inverted hyperplastic polyp found with gastritis cystica profunda and early gastric cancer. Clin Endosc 46: 568-571, 2013. 13. Ho SB, Takamura K, Anway R, Shekels LL, Toribara NW, Ota H: The adherent gastric mucous layer is composed of alternating layers of MUC5AC and MUC6 mucin proteins. Dig Dis Sci 49: 1598-1606, 2004.
Copyright of International Journal of Surgical Pathology is the property of Sage Publications Inc. and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use.
