Multifocal nerve lesions and LZTR1 germline mutations in segmental schwannomatosis

Ann Neurol. 2016 Oct;80(4):625-8. doi: 10.1002/ana.24753. Epub 2016 Aug 13.

Abstract

Schwannomatosis is a genetic disorder characterized by the occurrence of multiple peripheral schwannomas. Segmental schwannomatosis is diagnosed when schwannomas are restricted to 1 extremity and is thought to be caused by genetic mosaicism. We studied 5 patients with segmental schwannomatosis through microstructural magnetic resonance neurography and mutation analysis of NF2, SMARCB1, and LZTR1. In 4 of 5 patients, subtle fascicular nerve lesions were detected in clinically unaffected extremities. Two patients exhibited LZTR1 germline mutations. This appears contrary to a simple concept of genetic mosaicism and suggests more complex and heterogeneous mechanisms underlying the phenotype of segmental schwannomatosis than previously thought. Ann Neurol 2016;80:625-628.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adult
  • DNA Mutational Analysis
  • Female
  • Germ-Line Mutation
  • Humans
  • Lower Extremity / diagnostic imaging*
  • Lower Extremity / innervation
  • Lumbosacral Plexus / diagnostic imaging
  • Magnetic Resonance Imaging
  • Male
  • Middle Aged
  • Neurilemmoma / diagnostic imaging*
  • Neurilemmoma / genetics*
  • Neurofibromatoses / diagnostic imaging*
  • Neurofibromatoses / genetics*
  • Peripheral Nerves / diagnostic imaging*
  • Skin Neoplasms / diagnostic imaging*
  • Skin Neoplasms / genetics*
  • Transcription Factors / genetics*
  • Upper Extremity / diagnostic imaging*
  • Upper Extremity / innervation

Substances

  • LZTR1 protein, human
  • Transcription Factors

Supplementary concepts

  • Schwannomatosis