Somatic DICER1 gene mutation in sporadic intraocular medulloepithelioma without pleuropulmonary blastoma syndrome

Hum Pathol. 2015 May;46(5):783-7. doi: 10.1016/j.humpath.2015.01.020. Epub 2015 Feb 19.

Abstract

Germline DICER1 gene mutation has been described in ocular medulloepithelioma associated with pleuropulmonary blastoma family tumor and dysplasia syndrome. We present a case of sporadic ocular medulloepithelioma in an 18-year-old woman with D1709N somatic mutation in DICER1 gene, which has not been previously described. This case highlights the potential use of DICER1 gene sequencing to resolve the diagnostic challenge in recurrent and metastatic malignant medulloepithelioma, when morphology and immunohistochemistry are inconclusive. Further studies in larger series of this type of tumor are needed to confirm the relevance of this molecular abnormality in the tumorigenesis of this embryonic-type ocular tumor.

Keywords: Ciliary body; DICER1; Medulloepithelioma; Ocular neoplasm; Pleuropulmonary blastoma family tumor.

Publication types

  • Case Reports

MeSH terms

  • Adolescent
  • DEAD-box RNA Helicases / genetics*
  • Female
  • Germ-Line Mutation* / genetics
  • Humans
  • Immunohistochemistry / methods
  • Lung Neoplasms / genetics*
  • Lung Neoplasms / pathology
  • Neoplasms, Germ Cell and Embryonal / diagnosis
  • Neoplasms, Germ Cell and Embryonal / genetics*
  • Neoplasms, Germ Cell and Embryonal / pathology
  • Neuroectodermal Tumors, Primitive / diagnosis
  • Neuroectodermal Tumors, Primitive / genetics*
  • Neuroectodermal Tumors, Primitive / pathology
  • Pulmonary Blastoma / diagnosis
  • Pulmonary Blastoma / genetics
  • Ribonuclease III / genetics*

Substances

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

Supplementary concepts

  • Pleuropulmonary blastoma