|IMAGES AND VIDEOS
|Ahead of print publication
Jaundice and double-duct sign: Always cancer?
Diane Lorenzo1, Laurine Verset2, Jacques Devière1
1 Departement of Gastroenterology, Hepatopancreatology and Digestive Oncology, CUB Erasme Hospital, Free University of Brussels, Brussels, Belgium
2 Departement of Pathology, Bordet Institute, ULB, Brussels, Belgium
|Date of Submission||11-Nov-2020|
|Date of Acceptance||23-Mar-2021|
|Date of Web Publication||26-Jun-2021|
Department of Gastroenterology, Hepatopancreatology and Digestive Oncology, Erasme University Hospital, Free University of Brussels, Route de Lennik 808, 1070 Brussels
Source of Support: None, Conflict of Interest: None
Ampullary tumors manifest as symptoms of biliary (jaundice) and/or pancreatic (pancreatitis) obstruction, but incidental diagnoses are frequent. Endoscopically, there could present as a proliferative extra-ampullary tumor or more rarely as an intra-ampullary tumor (16% of patients). The intra-ampullary submucosal tumors are mainly adenoma/adenocarcinoma and neuroendocrine tumor. Rare cases of adenomyoma, intra-ampullary papillary–tubular neoplasm, gastrointestinal stromal tumor, lipoma, lymphangioma, and gangliocytic paraganglioma have been reported. Endoscopic resection is the first-line treatment when feasible. Depending on the histological analysis, additional surgical treatment may be necessary. Here, we report the first case described of an intra-ampullary Brunner's gland adenoma.
A 50-year-old woman presented with diffuse abdominal pain, finally related to sexually transmitted infection. She had a history of human immunodeficiency viruses and alcoholism. Blood test revealed a moderate cholestatic jaundice and important cytolysis (bilirubin: 1.4 mg/dL, alkaline phosphatase (PAL): 985 UI/L, gamma-glutamyl transferase: 3825 UI/L, alanine aminotransferase: 315 UI/L, and aspartate aminotransferase: 421 UI/L). A computed tomography scan and a magnetic resonance imaging showed double-duct dilation (common bile duct diameter at 14.5 mm and main pancreatic duct at 6.5 mm) and obstruction at the papilla without duodenal mass [Figure 1]. No lymph node invasion or metastasis was suspected. Endoscopic ultrasound confirmed a rounded little isoechoic homogeneous intra-ampullary tumor, responsible for double-duct dilation [Figure 1]. A papillectomy was performed without any further complication, and a protective pancreatic stent was easily positioned.
|Figure 1: Computed tomography scan, EUS, and endoscopic view showing little submucosal tumor responsible for double-duct dilation. The last image shows the papillectomy resection base|
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The histopathologic examination showed ampulla mucosa covered by flattened villi. In the submucosa, we observed a nodular proliferation of normal Brunner's glands. The lesion corresponded to an intra-ampullary Brunner's gland adenoma [Figure 2].
|Figure 2: (a) Duodenal mucosa covered by flattened villi. In the submucosa, we observed a nodular proliferation of normal Brunner's glands. (b) Black square of Figure 2a and only contains normal Brunner's glands|
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Brunner's glands are located mainly in the deep mucosa and submucosa of the duodenum (D1–D2). These glands secrete mucus which plays a role in neutralizing acidic gastric juice. Brunner's gland adenoma is a rare cause (less than 5% of benign duodenal tumors) of duodenal polyp with reported cases of bleeding and duodenal obstruction. The pathogenesis of Brunner's gland adenoma remains poorly understood. The risk of cancer remains debated, but cases of high-grade dysplasia and invasive cancer have been described.
Study concept and design: DL, JD; acquisition of data: DL, LV, JD; analysis and interpretation of data: DL, JD; drafting of the manuscript: DL, LV, JD; critical revision of the manuscript for important intellectual content: LV, JD; statistical analysis: none; obtained funding: none; technical, or material support: LV; study supervision: JD.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given her consent for her images and other clinical information to be reported in the journal. The patient understands that her name and initial will not be published, and due efforts will be made to conceal her identity, but anonymity cannot be guaranteed.
Financial support and sponsorship
Conflicts of interest
There are no conflicts of interest.
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[Figure 1], [Figure 2]