Plexiform neurofibroma of the eye region occurring in patients without neurofibromatosis type 1

Ophthalmic Plast Reconstr Surg. 2012 Nov-Dec;28(6):413-5. doi: 10.1097/IOP.0b013e3182627ea1.

Abstract

Purpose: The purpose of the present study was to investigate the correlation between Plexiform Neurofibroma (PN) of the eye region and Neurofibromatosis type 1 (NF1). According to the textbooks of ophthalmology, PN and NF1 are very closely linked. Our clinical experience raised doubts about this, however.

Methods: All biopsy reports from the Eye Pathology Institute, Copenhagen, for the 10-year period from 1999 to 2008 with PN as the histological diagnosis were identified. From the pathology referral forms, we determined whether the patients had had a history of NF1, and the medical records were reviewed for symptoms and signs of NF1. Patients with no known NF1 were invited for a clinical examination and a blood test for mutation analysis.

Results: 13 patients had been given the histological diagnosis of PN of the eye region during the 10-year period. Of these, 10 patients fulfilled the National Institute of Health (NIH) diagnostic criteria for NF1, but 3 patients had PN of the eye region as the only sign, and thus did not have NF1. In 2 of these patients, mutation analysis was performed on blood, and was negative.

Conclusion: PN of the eye region is suggestive of NF1 but not pathognomonic of this disease.

MeSH terms

  • Adolescent
  • Adult
  • Child
  • Child, Preschool
  • DNA Mutational Analysis
  • DNA, Neoplasm / genetics
  • Eyelid Neoplasms / diagnosis*
  • Eyelid Neoplasms / surgery
  • Female
  • Humans
  • Male
  • Middle Aged
  • Neurofibroma, Plexiform / diagnosis*
  • Neurofibroma, Plexiform / surgery
  • Neurofibromatosis 1 / diagnosis*
  • Neurofibromatosis 1 / genetics
  • Neurofibromin 1 / genetics
  • Orbital Neoplasms / diagnosis*
  • Orbital Neoplasms / surgery
  • Young Adult

Substances

  • DNA, Neoplasm
  • Neurofibromin 1