Early onset drusen and RPE dysfunction in a patient with NLRP3-AID

Ocul Immunol Inflamm. 2023 Nov;31(9):1877-1880. doi: 10.1080/09273948.2022.2113804. Epub 2022 Nov 17.

Abstract

Retinal pigment epithelium (RPE) dysfunction, manifested as drusen formation and RPE mottling, is a characteristic lesion of aging. The mechanism of RPE dysfunction remains unknown. Previous animal studies have proven that the activation of NLRP3 inflammasome in RPE leads to apoptosis and pyroptosis, which may play a very important role in the development of age-related macular degeneration (AMD). However, there is a lack of clinical evidence to support the above hypothesis. Herein, we report a 38-year-old Chinese Han woman who had NLRP3-associated autoinflammatory disease (NLRP3-AID) with widely scattered drusen at the posterior pole in both eyes. NLRP3-AID is a rare disease caused by mutations of the NLRP3 gene, leading to NLRP3 inflammasome activation. This report of early-onset drusen provides clinical evidence that the NLRP3 inflammasome might contribute to the occurrence of RPE dysfunction and is a potential cause of age-related macular degeneration (AMD).

Keywords: Age-related macular degeneration; NLRP3-AID; RPE dysfunction; drusen; inflammasome.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Female
  • Humans
  • Inflammasomes
  • Macular Degeneration* / diagnosis
  • Macular Degeneration* / genetics
  • NLR Family, Pyrin Domain-Containing 3 Protein / genetics
  • Retina / pathology
  • Retinal Drusen* / etiology
  • Retinal Drusen* / genetics
  • Retinal Pigment Epithelium / pathology

Substances

  • Inflammasomes
  • NLR Family, Pyrin Domain-Containing 3 Protein