Chordoma Occurs in Young Children With Tuberous Sclerosis

J Neuropathol Exp Neurol. 2017 Jun 1;76(6):418-423. doi: 10.1093/jnen/nlx032.

Abstract

Chordomas are rare bony neoplasms usually unassociated with a familial tumor predisposition syndrome. The peak incidence of this midline axial skeletal tumor is in adulthood but when very young children are affected, consideration should be given to occurrence within the tuberous sclerosis (TS) complex, especially when presenting in neonates <3 months of age. To call attention to this association, we present a brachyury-immunopositive chordoma occurring in the skull base of a 2-month-old male infant who was later realized to have metastases to the subcutaneous tissues and lungs, as well as rhabdomyoma of the heart and renal cysts/angiomyolipomas, that is, characteristic features of the TS complex. We review the limited literature on this topic.

Keywords: Angiomyolipoma; Brachyury; Chordoma; Neonatal; Pediatric neoplasm; Rhabdomyoma; Tuberous sclerosis.

Publication types

  • Case Reports

MeSH terms

  • Angiomyolipoma / diagnostic imaging
  • Angiomyolipoma / pathology
  • Chordoma / diagnostic imaging
  • Chordoma / genetics
  • Chordoma / pathology*
  • Heart Neoplasms / diagnostic imaging
  • Heart Neoplasms / secondary
  • Humans
  • Infant
  • Kidney Neoplasms / diagnostic imaging
  • Kidney Neoplasms / secondary
  • Lung Neoplasms / diagnostic imaging
  • Lung Neoplasms / secondary
  • Magnetic Resonance Imaging
  • Male
  • Neoplasm Metastasis
  • Rhabdomyoma / pathology
  • Skin Neoplasms / diagnostic imaging
  • Skin Neoplasms / pathology
  • Skin Neoplasms / secondary
  • Skull Base Neoplasms / diagnostic imaging
  • Skull Base Neoplasms / genetics
  • Skull Base Neoplasms / pathology*
  • Tuberous Sclerosis / diagnostic imaging
  • Tuberous Sclerosis / pathology*
  • Tuberous Sclerosis Complex 1 Protein
  • Tumor Suppressor Proteins / genetics

Substances

  • Tuberous Sclerosis Complex 1 Protein
  • Tumor Suppressor Proteins