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<p>Esophageal submucosal gland duct adenoma (ESGDA) is an extremely rare benign tumor originating from the esophageal ducts, resembling sialadenoma papilliferum (SP) of the minor salivary glands. However, the clinicopathological and molecular features of ESGDA remain incompletely characterized. We performed a multicenter, retrospective study aimed to elucidate the clinicopathological and molecular&#xa0;mechanisms underlying ESGDA. From 2018 to 2024, 10 cases of ESGDA were identified across two institutions. The cohort included 2 females and 8 males, aged 51–75&#xa0;years (mean: 64.1 ± 8.56&#xa0;years). Comprehensive clinicopathological and molecular analyses were performed. Histologically, the tumors exhibited tubulopapillary and cystic architectures, with ducts lined by a double epithelial&#xa0;layer. Four cases predominantly consisted of large cystic structures mimicking esophageal cysts, while two cases displayed florid papillary and micropapillary patterns resembling adenocarcinomas. Interstitial lymphoid infiltrates and micropapillary/serrated structures were observed in all cases. Genomic analysis identified <i>BRAF V600E</i> mutations in 8 of the 10 tumors (80%). ESGDA is a rare benign tumor arising from the esophageal duct, characterized by multiple lobulated cystic proliferation.The key diagnositic&#xa0;point is the presence of double layers and micropapillary/serrated structures lining the cysts. We identified <i>BRAF V600E</i> gene mutations in ESGDA, providing further evidence that it is the esophageal counterpart of ductal tumors and expanding the morphologic spectrum of sialadenoma papilliferum&#xa0;(SP)-like lesions.</p>

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Submucosal gland duct adenoma of the esophagus: expanding the morphologic spectrum of a rare tumor entity

  • 期刊论文

  • 发表日期：

    2025年12月1日

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  • Hongjin Hua,
  • Ying Ding,
  • Jinxing Zhang,
  • Die Hu,
  • Yue Zhang,
  • Sha Zhao,
  • Xinmin Si,
  • Zhihong Zhang,
  • Hai Li

摘要

Esophageal submucosal gland duct adenoma (ESGDA) is an extremely rare benign tumor originating from the esophageal ducts, resembling sialadenoma papilliferum (SP) of the minor salivary glands. However, the clinicopathological and molecular features of ESGDA remain incompletely characterized. We performed a multicenter, retrospective study aimed to elucidate the clinicopathological and molecular mechanisms underlying ESGDA. From 2018 to 2024, 10 cases of ESGDA were identified across two institutions. The cohort included 2 females and 8 males, aged 51–75 years (mean: 64.1 ± 8.56 years). Comprehensive clinicopathological and molecular analyses were performed. Histologically, the tumors exhibited tubulopapillary and cystic architectures, with ducts lined by a double epithelial layer. Four cases predominantly consisted of large cystic structures mimicking esophageal cysts, while two cases displayed florid papillary and micropapillary patterns resembling adenocarcinomas. Interstitial lymphoid infiltrates and micropapillary/serrated structures were observed in all cases. Genomic analysis identified BRAF V600E mutations in 8 of the 10 tumors (80%). ESGDA is a rare benign tumor arising from the esophageal duct, characterized by multiple lobulated cystic proliferation.The key diagnositic point is the presence of double layers and micropapillary/serrated structures lining the cysts. We identified BRAF V600E gene mutations in ESGDA, providing further evidence that it is the esophageal counterpart of ductal tumors and expanding the morphologic spectrum of sialadenoma papilliferum (SP)-like lesions.