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      Atypical presentation of myoepithelial hamartoma in the antrum of the stomach, mimicking a gastrointestinal stromal tumor: a case report

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          Abstract

          Introduction

          A myoepithelial hamartoma is a very uncommon submucosal tumor of the stomach. In an atypical presentation in our case, it mimicked the clinical presentation of a gastrointestinal stromal tumor. To the best of our knowledge, it is the first case of a hamartoma of the stomach reported from Bangladesh and one of few cases described in the literature.

          Case presentation

          We describe the case of a 35-year-old Bengali man with recurrent epigastric pain and occasional vomiting with radiographic findings of a gut mass. An upper gastrointestinal endoscopy revealed a healed duodenal ulcer, deformed ‘D’ bulb and a submucosal swelling in his antrum. Ultrasonography and a contrast-enhanced computed tomography scan confirmed the presence of a well-defined, oval gut mass in his upper abdomen, compressing his duodenum. The mass had a mixed density and was considered to probably be a gastrointestinal stromal tumor. Ultrasonography-guided fine needle aspiration cytology was inconclusive. After resection at laparotomy, a histopathological examination revealed a myoepithelial hamartoma. These tumors are characterized by hypertrophic smooth muscle bands surrounding varied epithelial elements, which may be arranged in diverse patterns such as simple glandular structure, Brunner’s gland, pancreatic ducts and sometimes pancreatic acini. This case report is complemented by a literature review relating to the atypical presentation.

          Conclusion

          Gut masses need to be investigated thoroughly and the possibility of rare tumors should not be excluded. Although the recommended treatment for such lesions is limited resection, radical procedures such as a pancreaticoduodenectomy are often performed when the lesion occurs in the periampullary area because of preoperative misdiagnosis as a carcinoma. Therefore, it is essential for clinicians to maintain current knowledge of the lesion to avoid inaccurate diagnosis and prevent unnecessary surgery.

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          Most cited references13

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          Gastric adenocarcinoma associated with adenomyoma of the stomach.

          We report a case of gastric adenomyoma contiguous with adenocarcinoma of the stomach. Although a similar association is well recognized in the duodenum, we believe this to be the first documented case in the stomach. We review the literature on adenomyomata and discuss the implications of this finding. It would appear that glandular and cystic intramural lesions of the stomach are a heterogeneous group, and while there is a well established association between gastric carcinoma and gastritis cystica profunda, the same does not apply to gastric adenomyoma.
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            Adenomyome des Pylorus

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              Adenomyoma of the small intestine.

              Adenomyoma of the gastrointestinal tract is a rare benign tumor-like lesion. The small intestine is the second most frequent location, usually in the periampullary area, but the lesion also occurs in the jejunum and ileum. While adenomyoma of the Vaterian system is primarily diagnosed in adults, more than half of reported cases of jejunal and ileal adenomyoma have been diagnosed in pediatric patients. Adenomyoma of the periampullary area usually presents with biliary obstruction or abdominal pain, whereas jejunal and ileal adenomyoma usually presents with intussusception or is incidentally discovered during surgery or autopsy. Since endoscopic and radiological examination yields uncharacteristic findings, histopathological evaluation is important in adenomyoma diagnosis. Pathologically, adenomyoma consists of glandular structures of various sizes and interlacing smooth muscle bundles that surround the glandular elements. The pathogenesis of adenomyoma is generally considered to be either a form of hamartoma or a pancreatic heterotopia. Although limited resection is considered the most effective treatment, pancreaticoduodenectomy is often performed when the lesion occurs in the periampullary area due to preoperative misdiagnosis as a carcinoma. It is, therefore, important that clinicians and pathologists maintain current knowledge of the disease to avoid inaccurate diagnosis, which could lead to unnecessary surgery.
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                Author and article information

                Contributors
                Journal
                J Med Case Rep
                J Med Case Rep
                Journal of Medical Case Reports
                BioMed Central
                1752-1947
                2012
                12 November 2012
                : 6
                : 382
                Affiliations
                [1 ]Department of General Surgery, Shaheed Suhrawardy Medical College and Hospital, Sher-e-Bangla Nagar, Dhaka, 1002, Bangladesh
                Article
                1752-1947-6-382
                10.1186/1752-1947-6-382
                3514267
                23146179
                71b1ed0a-ade2-4b7b-8b6a-6518d461079c
                Copyright ©2012 Nabi et al.; licensee BioMed Central Ltd.

                This is an Open Access article distributed under the terms of the Creative Commons Attribution License ( http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

                History
                : 28 May 2012
                : 3 October 2012
                Categories
                Case Report

                Medicine
                Medicine

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