Sporadic diffuse gastric polyposis: Report of a case

Surg Today (2011) 41:1428–1431 DOI 10.1007/s00595-010-4440-8

Case Report Sporadic Diffuse Gastric Polyposis: Report of a Case ERASMO SPAZIANI1, MARCELLO PICCHIO2, ANNALISA DI FILIPPO1, PIERO NARILLI3, CLAUDIO DI CRISTOFANO4, VINCENZO PETROZZA4, FRANCESCO DE ANGELIS1, and GIUSEPPE RAGONA4 1

Department Department 3 Department 4 Department 2

of of of of

Surgery, University of Rome “La Sapienza”, Polo Pontino, Hospital “A. Fiorini”, Latina, Italy Surgery, Civil Hospital “P. Colombo”, Rome, Italy Surgery, General Hospital “Nuova Itor”, Rome, Italy Experimental Medicine and Pathology, University of Rome “La Sapienza”, Polo Pontino, “I.C.O.T.”, Latina, Italy

Abstract A 50-year-old woman was admitted because of severe sideropenic anemia. The gastrin levels were within normal ranges. Esophagogastroduodenoscopy showed diffuse gastric polyposis with signs of diffuse oozing. Colonoscopy showed the presence of a 3-cm wide pedunculated polyp of the ascending colon, which was removed by diathermy. The patient was treated by total gastrectomy with Roux-Y esophagojejunostomy. Histological examination showed the presence of diffuse gastric polyposis with the contemporary occurrence of hyperplastic polyps and mixed hyperplastic and adenomatous polyps, with a tubular pattern and the focal aspect of serrate adenoma. This is the first case report of sporadic diffuse hyperplastic and adenomatous polyposis of the stomach. Key words Stomach · Gastric polyposis · Surgery

Introduction Gastric hyperplastic polyposis has been defined as a syndrome comprising at least 50 hyperplastic polyps.1 Thus far, there have been only a few reports of gastric hyperplastic polyposis.1–7 Gastric adenomas are defined as circumscribed, polypoid lesions, composed of either tubular and/or villous structures, lined by dysplastic epithelium. Most of these adenomas are solitary; however, multiple adenomas have been reported in the context of familial adenomatous polyposis. We herein present a unique case of sporadic gastric polyposis with aspects of hyperplastic and adenomatous polyps.

Reprint requests to: M. Picchio, Via Giulio Cesare 58, 04100 Latina, Italy Received: August 24, 2010 / Accepted: October 13, 2010

Case Report A 50-year-old woman was admitted to our department because of severe sideropenic anemia. Physical examination showed pallor with no cutaneous abnormal pigmentation and/or onychodystrophy. Laboratory data revealed hemoglobin of 4.1 g/dl. On further investigation, the patient’s serum iron was observed to be low at 9 μg/dl, and her total iron-binding capacity was elevated at 450 μg/dl. Her transferrin saturation was low at 2.3%, while her serum ferritin was 3.8 μg/dl and reticulocyte count 2.9%. Gastrin levels were within the normal range. The patient was taking warfarin, since she had received aortic valve replacement 3 years before the present admission. Esophagogastroduodenoscopy showed diffuse gastric polyposis with signs of diffuse oozing (Fig. 1). According to histological examinations of the endoscopic biopsies, the polyps were either hyperplastic or adenomatous. The upper gastrointestinal series did not show any polyps in the small bowel. Colonoscopy showed the presence of a 3-cm wide pedunculated polyp of the ascending colon that was removed with diathermy. At histological examination, the polyp was tubulovillous with moderate dysplasia. A total body computed tomography scan showed no signs of lymph node enlargement and/or metastasis. The patient underwent a total gastrectomy with perigastric lymph node resection (D1 lymphadenectomy). Roux-Y esophagojejunostomy reconstruction was performed. The macroscopic appearance of the gastrectomy specimen is shown in Fig. 2. The postoperative course was uneventful. The histological examination of the specimen confirmed the presence of diffuse gastric polyposis with the contemporary occurrence of hyperplastic polyps, mixed hyperplastic and adenomatous polyps, and adenomatous polyps with a tubular pattern (Fig. 3). Some polyps had aspects of serrate adenoma (Fig. 4). Atrophic gas-

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Fig. 1. Endoscopic view of the gastric corpus showing multiple polyps with diffuse oozing

Fig. 2. The gastrectomy specimen, cut along the lesser curvature (white arrow, pylorus; black arrow, cardia). The entire mucosa of the stomach is involved by polyps

tritis with intestinal metaplasia was also present, with no morphological and histochemical evidence of Helicobacter pylori infection. At her 2-year follow-up, the patient was in good general condition with moderate anemia (red blood cells = 3 200 000/mm3, hemoglobin = 11.0 g/dl).

On searching the PubMed database (www.ncbi.nlm.nih. gov/PubMed) for gastric polyposis, we found no prior published cases of diffuse adenomatous and hyperplastic gastric polyposis, although there were various reports of diffuse gastric polyposis. Males and females are equally affected by diffuse gastric polyposis. The lesions usually occur in middle-aged or elderly patients. The genetic transmission of this syndrome is still controversial. According to other case reports, we found no significant family history.1,5,6 However, especially in young patients, it appears that these lesions may be associated with polyposis syndromes, such as familial adenomatous polyposis, Peutz–Jeghers syndrome, juvenile polyposis, Gardner syndrome, and Cowden disease. An association between hyperplastic gastric polyposis, gastric carcinomas, and colorectal neoplasms, both adenoma and carcinoma, has been described.1,5,8 In our case, a colonoscopy showed the presence of an adenoma of the ascending colon. A possible link between gastric hyperplastic polyposis and colorectal neoplasia was postulated to be hypergastrinemia. Gastrin has been reported to be a growth-promoting tumoral agent, and hypergastrinemia was found in all patients with gastric polyposis reported by Niv et al.1 Moreover, chronic gastritis and H. pylori infection were present in almost all of the patients with gastric polyposis.3,4 One can speculate that the inflammatory cell infiltration and acceleration of epithelial cell turnover induced by H. pylori infection contributes to the development and/or progression of hyperplastic polyps. Helicobacter pylori infection is also a well-recognized cause of hypergastrinemia in human beings.8 However, the data regarding the association of hypergastrinemia with risk of colon neoplasms remains controversial. In particular, a recent large prospective study showed no association between hypergastrinemia and colorectal adenoma formation.9 In the present case, the gastrin levels were within the normal range. Gastric adenoma is an uncommon neoplasm, with a prevalence ranging from 0.5% to 3.7% in Western countries, and the disease usually occurs in the context of atrophic gastritis with intestinal metaplasia.10 The tumors are generally solitary, while cases of multiple gastric adenomas are reported in the context of familial adenomatous polyposis. Our case represents a unique occurrence of sporadic multiple gastric adenomas. As was true in our case, anemia is the most frequent clinical manifestation of diffuse gastric polyposis.1,2,5–7 However, some patients experience vague epigastric discomfort, bloating, anorexia, nausea, vomiting, hematemesis, melena, or obstruction.11 A detailed family history regarding cancer, polyps, and congenital abnormalities should be obtained from every polyposis

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E. Spaziani et al.: Unique Gastric Polyposis

Fig. 3. The gastric wall with multiple gastric polyps with different morphological aspects. In the center, there is a hyperplasic polyp with a focal adenomatous area (hematoxylin–eosin, ×100)

plastic polyps.18 Gastric cancers arising in diffuse gastric polyposis are mostly limited to T1.19,20 Therefore, total gastrectomy with perigastric lymph node resection (D1 lymphadenectomy) seems to guarantee the same survival benefit with respect to extended D2 lymphadenectomy.21 In our case, a gastrectomy was also indicated because of the severe anemia with diffuse bleeding in the stomach. In conclusion, we have reported the first description of sporadic diffuse hyperplastic and adenomatous polyposis of the stomach. Gastrectomy is therefore the treatment of choice because of the relevant risk of malignancy of this syndrome. Fig. 4. The adenomatous area of a polyp with features of serrate adenoma (hematoxylin–eosin, ×200)

patient. Colonoscopy is mandatory because of the frequent involvement of the colon in familial polyposis. Moreover, in patients with gastric adenomas, a higher risk of colon adenoma is present.12 Barium studies may be useful to visualize intestinal polyps. The risk of malignancy in multiple hyperplastic lesions is as high as 3.6%.13 The overall prevalence of dysplasia in hyperplastic polyps is believed to be 2 cm).14,15 The malignant potential of gastric adenomas is higher, with cancer occurring in more than 50% of adenomatous polyps larger than 2 cm.16 Moreover, the risk of gastric cancer in other parts of the gastric mucosa in patients with adenoma has varied between 8% and 59%.16 Malignancy may also occur in patients with hamartomatous polyps occurring in those with Peutz–Jeghers syndrome, and in subjects with this very rare sporadic form.17 Gastrectomy is justified in patients with diffuse involvement of the stomach by polyps, which can make detection of a synchronous focus of cancer difficult. Endoscopic biopsies alone are insufficient to assess the extent of malignancy of both adenomatous and hyper-

Conflict of Interest Statement. None of the authors have any conflict of interest.

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