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Brain cavernomas associated with en coup de sabre linear scleroderma: Two case reports.

Emily T Fain1, Melissa Mannion, Elena Pope

  • 1Department of Pediatrics, Division of Rheumatology, University of Alabama at Birmingham, Birmingham, AL, USA. rcron@peds.uab.edu.

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Linear scleroderma en coup de sabre, a rare pediatric condition, is linked to brain cavernomas. Early neuroimaging is crucial for managing potential neurological complications in affected children.

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Area of Science:

  • Pediatric Rheumatology
  • Neurology
  • Dermatology

Background:

  • Linear scleroderma is a localized scleroderma primarily affecting children.
  • The en coup de sabre subtype, occurring on the scalp or forehead, has known extracutaneous associations, predominantly neurological.
  • Brain cavernomas are vascular malformations that can occur in the brain.

Purpose of the Study:

  • To report two pediatric cases of linear scleroderma en coup de sabre associated with ipsilateral brain cavernomas.
  • To highlight the importance of neuroimaging in patients with this condition.

Main Methods:

  • Case report and literature review.
  • Utilized a worldwide pediatric rheumatology electronic list-serve to identify additional cases.
  • Magnetic resonance imaging (MRI) was used for diagnosis.

Main Results:

  • Two pediatric patients with linear scleroderma en coup de sabre were identified.
  • Both patients presented with ipsilateral brain cavernomas confirmed by MRI.
  • This suggests a potential association between these two conditions.

Conclusions:

  • Linear scleroderma en coup de sabre may be associated with brain cavernomas.
  • Neuroimaging studies are recommended for patients with linear scleroderma en coup de sabre to detect potential neurological abnormalities.
  • Early detection and management of neurological complications are vital.