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Giant Brunner’s Gland Hamartoma of the Duodenal Bulb Presenting with Upper Gastrointestinal Bleeding and Obstruction
Ju Hyoung Lee, Kyeong Min Jo, Tae Oh Kim, Jong Ha Park, Seung Hyun Park, Jae Won Jung, So Chong Hur, Sung Yeun Yang
Clin Endosc 2016;49(6):570-574.   Published online October 13, 2016
DOI: https://doi.org/10.5946/ce.2016.022
AbstractAbstract PDFPubReaderePub
Brunner’s gland hamartomas are small benign lesions that are most commonly found in the bulb of the duodenum. They are very uncommon, and most are found incidentally during upper gastrointestinal series or esophagogastroduodenoscopy. The lesions tend to be asymptomatic, but patients may present with symptoms of duodenal obstruction or hemorrhage secondary to ulceration. Histologically, a Brunner's gland hamartoma consists of the components of Brunner's gland cells, as well as glandular, adipose and muscle cells. In this study, we report the case of a 30-year-old man who presented with upper gastrointestinal bleeding and obstructive symptoms due to a giant Brunner's gland hamartoma in the duodenal bulb. The hamartoma was successfully removed by endoscopic resection. No significant complications were observed. Microscopically, the lesion was found to be entirely composed of variable Brunner's glands and adipocytes.

Citations

Citations to this article as recorded by  
  • Brunner’s gland hamartomas: Not always benign
    Manar Shmais, Ahmad Mousawi, Fadi Mourad, Ala I. Sharara
    Arab Journal of Gastroenterology.2024; 25(1): 70.     CrossRef
  • The Characteristics and Treatment Outcomes of 71 Duodenal Brunner’s Gland Adenomas with Endoscopic Submucosal Dissection
    Ying Xiang, Jinyan Liu, Nan ya Wang, Dehua Tang, Lei Wang, Ping xiao Zou, Guifang Xu, Qin Huang
    Digestive Diseases.2023; 41(6): 852.     CrossRef
  • Giant brunner gland hamartoma: An unusual cause of upper gastrointestinal bleed
    Bershic Valantine, ManjunathB Venkatapur, Sultan Nawahirsha, SBabu Kumar
    Gastroenterology, Hepatology and Endoscopy Practice.2023; 3(3): 90.     CrossRef
  • A giant Brunner’s gland hamartoma being treated as a pedunculated polyp: a case report
    Lizhi Yi, Zhengyu Cheng, Huarong Qiu, Jianjun Yang, Tao Wang, Ke Liu
    BMC Gastroenterology.2019;[Epub]     CrossRef
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Autosomal Dominant Inherited Cowden's Disease in a Family
Jun-Wook Ha
Clin Endosc 2013;46(1):85-90.   Published online January 31, 2013
DOI: https://doi.org/10.5946/ce.2013.46.1.85
AbstractAbstract PDFPubReaderePub

Cowden's disease, also known as a kind of phosphatase and tensin homolog (PTEN) hamartoma tumor syndrome, is an uncommon autosomal dominant inherited complex disorder with various hamartomatous growths of multiple organs involving all three germ cell layers. It usually manifests with polyps throughout the gastrointestinal tract, ranging anywhere from 30% to 85%, and more common extra intestinal findings. Mucocutaneous lesions like facial trichilemmomas, acral keratoses, papillomatous papules and macrocephaly, and malignancies including breast, thyroid and endometrial carcinoma are the hallmark of the disease. Here we report on familial Cowden's diseases case of a 52-year-old male proband with mucocutaneous lesions and mutation on the PTEN gene obtained by extrapolating from gastrointestinal polyposis as a starter and his daughter who developed thyroid cancer.

Citations

Citations to this article as recorded by  
  • Cowden Syndrome: A Review
    Ye-Ryeong Jung, Ji-Young Jun, Seeyoun Lee, Kum Hei Ryu, Dong Ock Lee, Yoon-Jung Chang, So-Youn Jung, Sun-Young Kong
    Laboratory Medicine Online.2023; 13(3): 165.     CrossRef
  • Colorectal polyposis as a clue to the diagnosis of Cowden syndrome: Report of two cases and literature review
    Giovanni Innella, Sara Miccoli, Dora Colussi, Laura Maria Pradella, Laura Benedetta Amato, Roberta Zuntini, Nunzio Cosimo Mario Salfi, Guido Collina, Francesco Ferrara, Luigi Ricciardiello, Daniela Turchetti
    Pathology - Research and Practice.2021; 218: 153339.     CrossRef
  • Clinical manifestations of gastrointestinal diseases in the oral cavity
    Mohammad S. Al-Zahrani, Ahmed A. Alhassani, Khalid H. Zawawi
    The Saudi Dental Journal.2021; 33(8): 835.     CrossRef
  • Hereditary Cancer Syndromes and Risk Assessment

    Obstetrics & Gynecology.2019; 134(6): e143.     CrossRef
  • Syndrome In Question
    Gabriela Maldonado, Juliano Peruzzo, Mariana Quirino Tubone, Clarissa Prieto Herman Reinehr, Gabriela Fortes Escobar
    Anais Brasileiros de Dermatologia.2015; 90(1): 131.     CrossRef
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  • 6 Web of Science
  • 5 Crossref
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A Case of Hamartomatous Polyp without Peutz-Jeghers Syndrome Arising from Appendix
Wee Sik Sohn, M.D., Ju Sang Park, M.D., Ji Eun Kim, M.D., Bong Hwan Kim, M.D., Seung Hee Yoo, M.D. and Eun Mee Han, M.D.*
Korean J Gastrointest Endosc 2010;41(1):36-40.   Published online July 31, 2010
AbstractAbstract PDF
Peutz-Jeghers syndrome is a familial syndrome consisting of mucocutaneous pigmentation and gastrointestinal polyposis and appears to be inherited as a single pleiotropic autosomal dominant gene with variable and incomplete penetrance. Cases of hamartomatous polyps of the Peutz-Jeghers type without Peutz-Jeghers syndrome have only rarely been reported. Moreover, only one case of a Peutz-Jeghers polyp at the appendix has been reported; it was resected by appendectomy. We report here on a case of a 45 year old man who had a hamartomatous polyp of the Peutz-Jeghers type arising from the appendix. The polyp was successfully removed by endoscopic polypectomy. To our knowledge, this is the first case of a hamartomatous polyp of the Peutz-Jeghers type that originated from the appendix and that was resected endoscopically. (Korean J Gastrointest Endosc 2010;41:36-40)
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Clinical Characteristics of Large Brunner's Gland Tumors in Korea
Ji Hun Kang, M.D., Yun Jeong Lim, M.D., Suk Jae Hahn, M.D., Jong Sun Choi, M.D.*, Moon Soo Koh, M.D. and Jin Ho Lee, M.D
Korean J Gastrointest Endosc 2010;40(5):297-302.   Published online May 30, 2010
AbstractAbstract PDF
Background
/Aims: Brunner's gland tumor is a common benign tumor noted in duodenum and might be designated as Brunner's gland hamartoma, adenoma or hyperplasia. But, a large duodenal polyp (over 1 cm in diameter) pathologically proven as a tumor of Brunner's gland origin is rare.
Methods
We analyzed a total of 39 cases including our direct experience of two cases and reports of 37 cases to clarify the clinical features of large Brunner's gland tumors.
Results
This tumor tends to present predominantly after the fourth decade: Median age is 52.6 years. Neither gender showed predominance. The size of the tumor ranged from 1 to 8 cm and the mean diameter was 2.9 cm. The most common location was the bulb. A pedunculated polyp was the common appearance. Epigastric pain was the most common symptom. Brunner's gland tumor can cause gastrointestinal hemorrhage or obstruction. Most of the polyps had non-diagnostic pathologic yield at pinch biopsy and only one case was diagnosed as focal adenocarcinoma. Endoscopic or surgical removal was undertaken for treatment. Most cases had no recurrence after removal. Snare polypectomy or submucosal dissection very safe and useful treatment modalities.
Conclusions
Large Brunner's gland tumors are mostly found during a check up or epigastric pain. Anemia, melena and obstruction often develop. Endoscopic polypectomy is recommended as a first line treatment. (Korean J Gastrointest Endosc 2010;40:297-302)
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Successful Treatment of Gastric Hamartomatous Polyp with Hemorrhage by Endoscopic Mucosal Resection
Jung Min Lee, M.D., Ji Hoon Baek, M.D., Hye Mi Kang, M.D., Byeng Do Min, M.D., Sang Hoon Park, M.D., Ji Woong Kim, M.D., Jin Woong Cho, M.D. and Myoung Jin Ju, M.D.*
Korean J Gastrointest Endosc 2009;39(5):300-303.   Published online November 30, 2009
AbstractAbstract PDF
Hamartomatous polyp is an uncommon lesion. Most of the gastric hamartomatous polyps occur in patients in their 40s because of polyposis coli. Gastric hamartomaotus polyps are usually asymptomatic, but they occasionally manifest with hematemesis or anemia. If they are congenital, then sessile polyps in children should be observed with some measurable frequency. However, many reports have revealed that if the polyp size is big and the risk of rebleeding is high, then ESD or surgical resection can be used to completely remove them. An inverted gastric hamartomatous polyp may have an early gastric cancer component. We report here on an unusual polyp in a 14-years male who presented with hematemesis. Those were about two 3 cm sized large polyps at the proximal antrum and mid body of the stomach, respectively. The polyps were successfully removed by an endoscopic mucosal resection and the lesions were diagnosed as gastric harmatomas. (Korean J Gastrointest Endosc 2009;39:300-303)
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A Case of Giant Brunner's Gland Hamartoma Presenting as Gastrointestinal Hemorrhage
Sang Yong Lee, M.D., Jung Hyun Lee, M.D., Hyo Jin Jung, M.D., Joo Ho Lee, M.D., Tae Oh Kim, M.D., Dae Hwan Kang, M.D., Geun Am Song, M.D. and Mong Cho, M.D.
Korean J Gastrointest Endosc 2007;34(1):47-50.   Published online January 30, 2007
AbstractAbstract PDF
Brunner's gland hamartoma is also called Brunner's gland adenoma and Brunner's gland hyperplasia of Brunneroma, and it is a rare tumor of the duodenum. This tumor is caused by a benign proliferation of Brunner's glands, and the disease is normally present on the duodenum. These tumors arise mainly in the duodenal bulb and only rarely in the jejunum and proximal ileum. The clinical manifestations are nonspecific gastrointestinal complaints such as bloating or epigastric pain, and the tumor often gives rise to melena or obstruction. We report here on a case of a giant Brunner's gland hamartoma in a 48 year-old woman, and it was treated by endoscopic polypectomy. We also include a review of the related literatures.
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A Case of a Giant Brunner's Gland Hamartoma Originating from the Pyloric Ring
Yon Soo Jeong, M.D., Jun Pyo Chung, M.D., Dok Yong Lee, M.D., Young Kim, M.D., Hee Jung Yoon, M.D., Sang Won Ji, M.D., Yong-Han Paik, M.D., Se Joon Lee, M.D., Kwan Sik Lee, M.D., Sang In Lee, M.D. and Chanil Park, M.D.*
Korean J Gastrointest Endosc 2003;27(6):531-535.   Published online December 30, 2003
AbstractAbstract PDF
Brunner's gland hamartoma, also called as Brunner's gland adenoma or Brunner's gland hyperplasia, is a relatively rare disease that results from benign proliferation of the Brunner's gland normally present in the duodenum. It is mostly located at the duodenal bulb, occasionally second or third portion, but is rarely found at the pyloric ring, jejunum or proximal ileum. In Korea, total 27 cases of Brunner's gland hamartoma have been reported, but none of them had their origin at the pyloric ring only. We report a case of Brunner's gland hamartoma, found incidentally, originating from the pyloric ring in a 54-year-old woman, which was resected endoscopically after retracting the tumor into the stomach. (Korean J Gastrointest Endosc 2003;27:531⁣535)
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혈관종 또는 간세포암과 감별이 어려웠던 단일 결절형 담도과오종 1 예
Korean J Gastrointest Endosc 2001;23(5):417-417.   Published online November 30, 2000
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증례 / 점막하 종양양상의 위 과오종 1예 ( A Case of Gastric Hamartoma Presenting as a Submucosal Tumor )
Korean J Gastrointest Endosc 2001;22(3):169-173.   Published online November 30, 2000
AbstractAbstract PDF
Hamartomatous polyps in the stomach have been described as gastric lesions of familial polyposis coli or not associated with polyposis coli. However, submucosal tumor-like lesion of the gastric hamartoma is very rare. We have experienced an unusual hamartoma in the stomach in a 69-year-old man. He was hospitalized with epigastric discomfort. Endoscopy revealed a submucosal mass at the greater curvature of the high body of the stomach. The resected mass measured 3 X 2 X 1.5 cm and was characterized by cystic dilation of glandular structures, The glandular structures consisted of various types of lining cells, including surface foveolar cell types, pyloric cell types and parietal-like cells, and irregularly arranged smooth muscle bundles and collagen fibers were noted. We report this unusual gastric hamartoma presenting as a submucosal tumor with a review of literatures. (Korean J Gastrointest Endosc 2001;22:169 - 173)
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점막하 종양소견을 보인 위 과오종
Korean J Gastrointest Endosc 2000;20(5):391-391.   Published online November 30, 1999
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유문륜으로 탈출된 과오종성 위 용종 1예 ( A Case of Gastric Hamartoma Prolapsing Through the Pylorus )
Korean J Gastrointest Endosc 1999;19(6):941-944.   Published online November 30, 1998
AbstractAbstract PDF
Gastric hamartoma is usually an asymptomatic, small, sessile polyp, located in the body and fundus of the stomach. A case was experienced involving a solitary large hamartomatous polyp prolapsing through the pylorus in a 63-year old female presented with melena. A gastroendoscopy determined a pedunculated gastric polyp prolapsing through the pylorus with fresh blood oozing and congestion. A UGI series revealed that a 5 cm sized lobulated mass with a long stalk attached at the greater curvature side of the fundus. The polyp was removed by an endoscopic snare polypectomy and diagnosed as a gastric hamartoma. (Korean J Gastrointest Endosc 19: 941∼944, 1999)
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증례 : 위장관 ; 장중첩증과 심한 빈혈을 일으킨 Brunner 선 과오종 1예 ( Case Reports : Stomach & Intestine ; A Case of Brunner's Gland Hamartoma with Severe Anemia and Intussusception )
Korean J Gastrointest Endosc 1997;17(5):691-695.   Published online November 30, 1996
AbstractAbstract PDF
Brunner's gland hamartomas are rare duodenal tumors with characteristic pathologic featmes. The usual clinical presentation is nonspecific symptoms, obstructive symptoms, or intestinal bleeding. The majority of these tumors are less than 3 cm in diameter. In cases of larger size, the manifestations are usually intestinal obstruction or intestinal bleeding. The cases with massive gastrointestinal bleeding and severe anemia, requiring transfusion are rare. The diagnosis is made by radiologic studies and gastroduodenoscopy. The treatment of Brunner's gland hamartomas should be conservative, since they are not premalignant, However, the lesions originate in the submucosa, so the confimative diagnosis usually cannot be made by endoscopie biopey. For definitive diagnosis and relief of symptoms, the lesions must be removed surgically or endoscopically. Endoscopic excision is indicated if the tumar is pedunculated. We recently experienced a case of Brunner's gland hamartoma of about 5.5 cm in diameter with intestinal bleeding, requiring transfusion and intussusception. Preoperative diagnosis was submucosal tumor, such as lymphoma, with duodeno-duodenal intussusception. After surgical removal, the resected specimen showed the histologic features of Bruaner's gland hamartoma. (Korean J Gastrointest Endosc 17: 691-695, 1997)
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원저 : 식도 위장관 ; 위저선 폴립의 임상 및 병리학적 분석 : 가족성 선종성 폴립증에 동반된 다발성 위저선 폴립과 산발적으로 발생된 위저선 폴립의 비교 ( Original Articles : Esophagus , Stomach & Intestine ; Fundic Gland Polyps : A Clinical and Pathologic Analysis with Special Reference to Familial Adenomatous Polyposis )
Korean J Gastrointest Endosc 1997;17(2):133-141.   Published online November 30, 1996
AbstractAbstract PDF
Background
/Aims: The aims of this study are to clarify the morphology of fundic gland polyp (FGP) and to compare the features of FGP between familial adenomatous polyposis-associated group and sporadic development group. Methods: A total of 15 endo- scopic biopsy specimens of FGP from 13 patients were divided into three groups; Group A(3 cases; familial adenomatous polyposis family, multiple FGPs), Group B(3 cases; sporadic development, multiple FGPs) and Group C(7 cases; sporadic development, single FGP), and their endoseopic /microscopic features including mucin histochemistry and immunohistoc- hemistty(for PCNA) were compared. Results: FGPs were confined to the gastric body and fundus in all 3 groups, and measured 2-8 mm. Their numbers varied even in Group A and Group B, The difference was observed in their median age: 26 years in Group A and 55 years in Group B, respectively, but there were no differences in endoscopic, histologic, mucin histochemical and immunohistochemical(for PCNA) features. Micro-scopically, all FGPs were composed of fundic glands and scattered microcysts with a spectrum of disordered glandular architecture which ranged from convoluted gland to Y-shaped gland, to stellateshaped gland, and to irregular tortuous glancl with dilated lumen. Conclusions: We assume that diversity af morphologic features of FGP may develop from progression of hyperplastic/hamartomatous fundic glandular proliferation which may end up with microcyst formation as an evolutional change. Familial adenomatous polyosis-associated FGPs were not endoscopically and histologically distingishable from sporadic deveoped FGPs. (Korean J Gastrointest Endosc 17: 133-139, 1997)
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증례 : 식도 위장관 ; 대장용종증 동반이 없는 위에서 발견된 과오종 4예 ( Case Reports : Esophagus , Stomach & Intestine ; Four Cases of Gastric Hamartomatous Polyps without Polyposis Coli )
Korean J Gastrointest Endosc 1996;16(6):994-999.   Published online November 30, 1995
AbstractAbstract PDF
Hamartoma is a very rare congenital tumor. A few cases of hamartomatous pol yps in the stomach were reported. Most of harrurtomatous polyps in the stomach occur in patients of polyposis coli. We have experienced four cases of gastric hamartomatous polyps without polyposis coli. There are some differences between previously reported cases and ours. In our casee, the distribution of age is from fourth to ninth decade, men and women ratio is 1: 1. The number of polyps are one in three cases and two in one case. The size of polyps is from 5mm to 12 mm. The location of polyps are various, but most common in antrum. We have experienced four cases which were different from previously reported ones and report them with a brief review of literatures. (Korean J Gastrointest Endosc 16: 994-998, 1996)
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원저 : Brunner 선의 과오종성 증식을 시사하는 6예의 임상 - 병리학적 분석 ( Original Articles : Brunner's Gland Hamartoma. A Clinicopathologic Analysis of Six Cases )
Korean J Gastrointest Endosc 1994;14(1):19-23.   Published online November 30, 1993
AbstractAbstract PDF
Pathological proliferation of Brgnners gland is rare, and its wide range of morphologieal variations have led to confusing the terminology with considerable lack of agreement. Six cases of duodenal nodular lesions which consisted of pathological proliferation of Brunners glands were examined by light microscopy. Polypectomy was made in two cases, and the remaining 4 cases were examined with endoscopic biopsy materials. Two polypectomy specimens, 2 cm and 3 cm each in great diameters, consisted of protruded ovoid mass with broad and short stalks. Microacopically, all of 6 cases revealed thin strands of fibrous connective tissue which separated the normal-looking Brunners glands in lobules. Individual lobules were composed of groups of acini formed by cuboidal cells admixed with occasional endocrine cells. Presence of glandular and ductal configuration of the Brunners gland aside from thick, randomly arranged bundles of smooth muscle were featured without distinct relation to interlobular septa. Also, one polypectomy case disclosed the nests of fat cells interspersed with the glandular acini. One mucosal biopsy case contained not only the smooth muscle bands but also the intimate mixture of both acini and ducts. We conclude that some of heterogeneous composition of duodenal nodular proliferation of Brunners gland is indicative of a hamartomatous growth.
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