The novel triad of dorsal agenesis of the pancreas with concurrent pancreatic ductal adenocarcinoma and nonalcoholic chronic calcific pancreatitis: a case series and review of the literature

J Gastrointest Surg. 2011 Sep;15(9):1643-9. doi: 10.1007/s11605-011-1542-6. Epub 2011 Apr 22.

Abstract

Introduction: Dorsal agenesis of the pancreas (DAP) is a rare congenital anomaly, with only 44 cases having been reported in the English literature since 1966.

Materials and methods: A retrospective review of our IRB-approved pancreatic surgery database was performed from November 2005 to November 2010 searching for cases of DAP.

Discussion: Disorders in the retinoic acid (Raldh) and hedgehog (Hh) signaling pathways, which appear to play a role in the development of DAP, have been implicated in other diseases of the pancreas such as pancreatic ductal adenocarcinoma (PDA) and nonalcoholic chronic calcific pancreatitis (NCCP).

Conclusion: In this report, we describe three cases of DAP in the setting of PDA, two of which include the third component of NCCP. We provide a discussion of the clinical features of this novel triad and address the molecular pathways that relate to these respective diseases.

Publication types

  • Case Reports
  • Review

MeSH terms

  • Adenocarcinoma / complications*
  • Adenocarcinoma / diagnosis
  • Adenocarcinoma / genetics
  • Adenocarcinoma / surgery
  • Adult
  • Aged
  • Aldehyde Dehydrogenase 1 Family
  • Animals
  • Congenital Abnormalities / diagnosis*
  • Congenital Abnormalities / genetics
  • Female
  • Hedgehog Proteins / genetics
  • Humans
  • Male
  • Middle Aged
  • Pancreas / abnormalities
  • Pancreatic Neoplasms / complications*
  • Pancreatic Neoplasms / diagnosis
  • Pancreatic Neoplasms / genetics
  • Pancreatic Neoplasms / surgery
  • Pancreatitis, Chronic / complications*
  • Pancreatitis, Chronic / diagnosis
  • Pancreatitis, Chronic / genetics
  • Pancreatitis, Chronic / surgery
  • Retinal Dehydrogenase / genetics
  • Signal Transduction / genetics

Substances

  • Hedgehog Proteins
  • Aldehyde Dehydrogenase 1 Family
  • ALDH1A2 protein, human
  • Retinal Dehydrogenase

Supplementary concepts

  • Pancreas agenesis, dorsal