Cicatricial upper eyelid retraction in encephalocraniocutaneous lipomatosis: a report of two cases and review of literature

Ophthalmic Plast Reconstr Surg. 2002 Mar;18(2):151-5. doi: 10.1097/00002341-200203000-00012.

Abstract

Purpose: To describe two patients with encephalocraniocutaneous lipomatosis (ECCL) and to review the literature on this disorder.

Methods: Brain and orbit CT scans were performed on two patients with ECCL. Both patients were examined by the same ophthalmologist and neurologist during at least a 2-year follow-up period.

Results: Unilateral skull hamartomas, intracranial abnormalities, epibulbar choristomas, and ocular adnexal changes including a specific form of cicatricial upper eyelid retraction were present in both patients.

Conclusions: ECCL is a special form of oculocerebrocutaneous disease that has significant adnexal findings that are essential for the diagnosis of this rare disorder.

Publication types

  • Case Reports
  • Review

MeSH terms

  • Brain / diagnostic imaging
  • Child, Preschool
  • Cicatrix / diagnostic imaging
  • Cicatrix / etiology*
  • Eyelid Diseases / diagnostic imaging
  • Eyelid Diseases / etiology*
  • Female
  • Humans
  • Infant
  • Lipomatosis / complications*
  • Lipomatosis / diagnostic imaging
  • Male
  • Neurocutaneous Syndromes / complications*
  • Neurocutaneous Syndromes / diagnostic imaging
  • Orbit / diagnostic imaging
  • Tomography, X-Ray Computed