Proptosis secondary to bilateral extraocular muscle enlargement in Noonan syndrome with hypertrophic cardiomyopathy: A case report

Eur J Ophthalmol. 2023 Sep;33(5):NP67-NP70. doi: 10.1177/11206721221125035. Epub 2022 Sep 5.

Abstract

Purpose: To report and investigate proptosis in a young girl with Noonan syndrome.

Methods: Observational case report.

Results: A 16-year-old girl affected by Noonan syndrome underwent a complete ophthalmological examination showing bilateral proptosis with hypofunction of lateral rectus and superior oblique muscles. Visual acuity, color discrimination and fundus examination were unremarkable. The orbital MRI showed bilateral proptosis and symmetrical enlargement of extraocular muscles, with bellies thickening and tendon sparing. The young patient also complained restrictive hypertrophic cardiomyopathy.

Conclusions: Proptosis is an uncommon ocular manifestation of Noonan syndrome and its pathophysiology has never been clarified. The MRI evidence of extraocular muscles enlargement associated with hypertrophic cardiomyopathy, led us to hypothesize a common altered pathway beneath these features, more specifically the MAP kinase pathway, since extraocular and cardiac muscles share a mesenchymal embryological origin.

Keywords: Noonan syndrome; diagnostic studies; extraocular muscles enlargement; genetic disease/congenital abnormalities; neuro imaging; ocular motility disorders; orbital disease; pediatric ophthalmology; proptosis; restrictive hypertrophic cardiomyopathy.

Publication types

  • Observational Study
  • Case Reports

MeSH terms

  • Adolescent
  • Cardiomyopathy, Hypertrophic* / complications
  • Cardiomyopathy, Hypertrophic* / diagnosis
  • Exophthalmos* / diagnosis
  • Exophthalmos* / etiology
  • Eye
  • Female
  • Humans
  • Noonan Syndrome* / complications
  • Noonan Syndrome* / diagnosis
  • Oculomotor Muscles / diagnostic imaging