A nonrandom association of gastrointestinal stromal tumor (GIST) and desmoid tumor (deep fibromatosis): case series of 28 patients

Ann Oncol. 2012 May;23(5):1335-1340. doi: 10.1093/annonc/mdr442. Epub 2011 Oct 12.

Abstract

Background: Gastrointestinal stromal tumors (GISTs) and desmoid tumors (DTs) are two rare mesenchymal tumor. Anecdotal reports of individuals with both diseases led us to make the hypothesis that the association is a nonrandom event as the probability would be extremely low to observe such cases if they were independent events.

Patients and methods: We evaluated the existence of patients with GIST and DT in a large multicenter cohort at 10 institutions in the United States, Australia and Europe. Data on gender, age at diagnosis, KIT, PDGFRA, CTNNB1 mutation status and follow-up time after diagnosis were collected.

Results: We identified 28 patients diagnosed with both tumors. DT was diagnosed after GIST in 75% of patients and concomitantly in 21%. In only one case (4%), GIST was diagnosed after DT. KIT or PDGFRA mutations were detected in 12 of 14 GIST, 9 in KIT exon 11, 2 in KIT exon 9 and 1 in PDGFRA.

Conclusion: A statistical analysis of these 28 cases suggests a nonrandom association between GIST and DT. Further studies may be able to elucidate the underlying biology responsible for this association.

Publication types

  • Multicenter Study
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adult
  • Aged
  • Aged, 80 and over
  • Australia / epidemiology
  • Cohort Studies
  • Europe / epidemiology
  • Female
  • Fibromatosis, Aggressive / complications*
  • Fibromatosis, Aggressive / epidemiology*
  • Follow-Up Studies
  • Gastrointestinal Stromal Tumors / complications*
  • Gastrointestinal Stromal Tumors / epidemiology*
  • Humans
  • Incidence
  • Male
  • Middle Aged
  • Retrospective Studies
  • United States / epidemiology