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Giant Brunner's gland hamartoma causing retrograde jejuno-duodenal intussusception: A case report.

ABSTRACT: Brunner's gland hamartoma is a rare benign duodenal tumor. It occurs in Brunner's glands, which are found in the duodenum and produce secretions that protect the duodenum from pancreatic enzymes, gastric acid, and other agents. Endoscopic or surgical resection is required for these hamartomas. Duodenal intussusception is a relatively rare condition, usually caused by the presence of benign tumors, such as fibroadenomas, lipomas, papillomas, or sometimes with malignant neoplasms. We report a case of giant Brunner's gland hamartoma in the duodenum causing antiperistaltic intussusception in a 45-year-old female patient. The patient reported a 3-year history of chronic anemia, and this mass was detected incidentally by computed tomography (CT) during investigations for chronic anemia and weight loss. Pre-operative abdominal and pelvis contrast revealed a sausage-shaped intraluminal structure with alternating fat planes and vessels distended in the third part of the duodenum up to the first part of the duodenum. Pancreas-sparing duodenectomy was performed. The patient recovered very slowly and was discharged on postoperative day 15 in good condition. Histology showed a large polypoid mass measuring 12.0?×?7.5?×?2.0 cm3, consistent with Brunner's gland hamartoma. Brunner's gland hamartoma can present with features of duodenal intussusception or ampullary obstruction but is rarely seen to cause retrograde jejuno-duodenal intussusception. Pancreas-sparing duodenectomy is the best surgical option in adult patients with intestinal intussusception associated with giant lesions close to the ampulla of Vater, especially in the presence of features of malignancy.

SUBMITTER: Aldossary MY

PROVIDER: S-EPMC6327855 | biostudies-other | 2019 Feb

REPOSITORIES: biostudies-other

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Giant Brunner's gland hamartoma causing retrograde jejuno-duodenal intussusception: A case report.

Aldossary Mohammed Yousef MY Alzahir Ali A AA Almulla Liqa A LA Alhajji Zahrah H ZH Alsaif Osama H OH

Annals of medicine and surgery (2012) 20190104

Brunner's gland hamartoma is a rare benign duodenal tumor. It occurs in Brunner's glands, which are found in the duodenum and produce secretions that protect the duodenum from pancreatic enzymes, gastric acid, and other agents. Endoscopic or surgical resection is required for these hamartomas. Duodenal intussusception is a relatively rare condition, usually caused by the presence of benign tumors, such as fibroadenomas, lipomas, papillomas, or sometimes with malignant neoplasms. We report a case ...[more]

PMID: 30671231

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Project description:Introductionand importance: Retrorectal cystic hamartoma (RCH) is a rare congenital lesion of the presacral space, which is part of the vestigial cystic tumors often benign and predominantly in women. Generally asymptomatic, the appearance of symptoms such as pain or neurological disorders should raise suspicion of degeneration.Case presentationWe report an unusual observation of a 62-year-old patient admitted for perineal pain evolving for 2 months associated with tenesma and chronic constipation. The digital rectal examination found a posterior bulge at 4 cm from the anal margin, without intraluminal lesion. Rectosigmoidoscopy had noted posterior extrinsic compression but no rectal tumor. Pelvic CT and MRI had shown a solidocystic formation of the retro-rectal and presacral spaces, related to an enteric cyst. The operation was performed by abdominal approach and the surgical exploration had found a bilobed cystic formation. The cystic mass was removed and the anatomopathological examination concluded that it was a cystic hamartoma with no sign of malignancy.Clinical discussionRetrorectal tumors develop in the space bounded anteriorly by the propria fascia of the rectum and posteriorly by the presacral fascia overlying the sacrum. Common in children and then often malignant, inversely, in adults, they are rare and most often benign tumors. They are generally asymptomatic with a predominance of females, unlike our observation where the patient was male with a symptomatology dominated by perineal pain and constipation. The discovery is incidental in the majority of cases, however, in some cases, these cysts may be revealed by complications. The lesion can be explored by transrectal or suprapubic ultrasound, MRI and CT scan. Rectoscopy and fistulography may complete the exploration in case of diagnostic doubt. The resection must be thorough and in monobloc because of the risk of recurrence and the approach depends on the location and the size of the lesion.ConclusionRCH is a rare benign lesion whose morphological characteristics seem quite stereotyped. A detailed postoperative anatomopathological examination allows the diagnosis to be made and, above all, to look for a site of malignant transformation. This is why a complete surgical removal is necessary to prevent recurrence.

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Giant Brunner's gland hamartoma causing retrograde jejuno-duodenal intussusception: A case report.

Publications

Giant Brunner's gland hamartoma causing retrograde jejuno-duodenal intussusception: A case report.

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