Clinical features and distribution of the APC variant in duodenal and ampullary polyps in patients with familial adenomatous polyposis: a multicenter retrospective cohort study in Japan

Int J Clin Oncol. 2024 Feb;29(2):169-178. doi: 10.1007/s10147-023-02442-7. Epub 2023 Dec 24.

Abstract

Background: Management of duodenal or ampullary adenomas in patients with familial adenomatous polyposis (FAP) is a major challenge for clinicians. Insufficient data are available to evaluate the clinical manifestations and distribution of adenomatous polyposis coli (APC) variants in these patients.

Methods: We enrolled 451 patients with data regarding duodenal or ampullary polyps from 632 patients with FAP retrospectively registered in a nationwide Japanese multicenter study. Clinicopathological features and distribution of APC variants were compared between patients with and without duodenal or ampullary polyps.

Results: Duodenal and ampullary polyps were found in 59% and 18% of patients with FAP, respectively. The incidence of duodenal cancer was 4.7% in patients with duodenal polyps, and that of ampullary cancer was 18% in patients with ampullary polyps. Duodenal polyps were significantly associated with the presence of ampullary polyps and jejunal/ileal polyps. Duodenal polyps progressed in 35% of patients with a median follow-up of 776 days, mostly in those with early Spigelman stage lesions. Ampullary polyps progressed in 50% of patients with a follow-up of 1484 days. However, only one patient developed a malignancy. The proportion of patients with duodenal polyps was significantly higher among those with intermediate- or profuse-type APC variants than attenuated-type APC variants. The presence of duodenal polyps was significantly associated with ampullary and jejunal/ileal polyps in patients with intermediate- or profuse-type APC variants.

Conclusions: Periodic endoscopic surveillance of the papilla of Vater and small intestine should be planned for patients with FAP with duodenal polyps.

Keywords: APC pathogenic variant; Ampullary polyp; Duodenal polyp; Familial adenomatous polyposis; Spigelman stage.

Publication types

  • Multicenter Study

MeSH terms

  • Adenomatous Polyposis Coli* / genetics
  • Adenomatous Polyposis Coli* / pathology
  • Ampulla of Vater* / pathology
  • Common Bile Duct Neoplasms* / complications
  • Common Bile Duct Neoplasms* / genetics
  • Common Bile Duct Neoplasms* / pathology
  • Duodenal Neoplasms* / genetics
  • Humans
  • Intestinal Polyps
  • Japan
  • Retrospective Studies

Substances

  • APC protein, human