Judicious DICER1 testing and surveillance imaging facilitates early diagnosis and cure of pleuropulmonary blastoma

Pediatr Blood Cancer. 2014 Sep;61(9):1695-7. doi: 10.1002/pbc.25092. Epub 2014 May 13.

Abstract

Pleuropulmonary blastoma (PPB) and Sertoli-Leydig cell tumor (SLCT) are both associated with germline mutations in DICER1. In this brief report, a maternal history of SLCT led to identification of a deleterious DICER1 mutation in the patient and her asymptomatic infant. Radiographic screening revealed a large Type I PPB, which was completely resected. Identification of DICER1 mutation carriers and imaging of children at risk for PPB may allow detection of PPB in its earliest and most curable form, leading to increased likelihood of surgical cure and decreased risks of treatment-related late effects.

Keywords: DICER1; Sertoli-Leydig cell tumor; genetic testing; lung cysts; pleuropulmonary blastoma.

Publication types

  • Case Reports
  • Research Support, N.I.H., Extramural
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adult
  • Child, Preschool
  • DEAD-box RNA Helicases / genetics*
  • Early Diagnosis
  • Female
  • Germ-Line Mutation / genetics*
  • Humans
  • Infant
  • Male
  • Prognosis
  • Pulmonary Blastoma / diagnosis*
  • Pulmonary Blastoma / genetics
  • Pulmonary Blastoma / surgery*
  • Ribonuclease III / genetics*
  • Sertoli-Leydig Cell Tumor / pathology
  • Sertoli-Leydig Cell Tumor / surgery*
  • Tomography, X-Ray Computed

Substances

  • DICER1 protein, human
  • Ribonuclease III
  • DEAD-box RNA Helicases

Supplementary concepts

  • Pleuropulmonary blastoma