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Keratoprosthesis in Ectodermal Dysplasia.

Rachel A F Wozniak1, Mithra Gonzalez, James V Aquavella

  • 1University of Rochester, Flaum Eye Institute, Rochester, NY.

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This study details complex surgeries and infliximab for a Boston type I keratoprosthesis (KPro) in a patient with ectodermal dysplasia and corneal necrosis. Novel approaches achieved visual rehabilitation despite chronic melting.

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Area of Science:

  • Ophthalmology
  • Regenerative Medicine

Background:

  • Ectodermal dysplasia presents complex challenges in corneal abnormalities and eyelid malformations.
  • Keratoprosthesis (KPro) implantation is crucial for visual restoration in severe corneal diseases.

Observation:

  • A monocular patient with ectodermal dysplasia experienced chronic, noninfectious corneal necrosis post-keratoprosthesis.
  • Progressive corneal melting and fistulization at the KPro optic border necessitated multiple surgical interventions.

Findings:

  • A multi-stage surgical strategy, including amniotic membrane graft, KPro replacement, conjunctival flap, corneal grafts, and muscle advancement, was employed.
  • The adjunctive use of infliximab significantly improved ocular surface stability.
  • The patient achieved and maintained a best-corrected visual acuity of 20/80 post-treatment.

Implications:

  • This case highlights the efficacy of innovative surgical techniques in managing complex KPro complications.
  • The off-label use of infliximab demonstrates a promising medical adjunctive therapy for sterile corneal melt.
  • Successful visual rehabilitation in ectodermal dysplasia underscores the importance of tailored, multidisciplinary treatment approaches.