Sclerouveitis as Part of Multiple Autoimmune Syndrome in a Patient with Alkaptonuria: A Case Report

Ocul Immunol Inflamm. 2024 Jul;32(5):758-762. doi: 10.1080/09273948.2023.2179498. Epub 2023 Feb 23.

Abstract

Purpose: To present a novel association of multiple autoimmune syndrome (MAS) in a patient with sclerouveitis and alkaptonuria.

Case report: A 68-year-old female with alkaptonuria, Hashimoto's thyroiditis, and familial autoimmunity presented with decreased VA, red eye, foreign body sensation, and ocular pain. Ophthalmological examination: OD conjunctival hyperemia, ochronosis, a reddish-violet scleral nodule, keratic precipitates, 2+ cells in the anterior chamber, 0.5+ vitreous cells, and mild vitreous haze. The patient was diagnosed with anterior uveitis and anterior nodular scleritis. Due to the associated sicca symptoms, a salivary gland biopsy was ordered, confirming Sjögren's syndrome. Then, MAS was diagnosed, and immunomodulatory medications were started; however, as she was refractory to more than two of them, it was suggested to start biological treatment.

Conclusion: We present a novel MAS-type 2 pattern consisting of Hashimoto's thyroiditis, sclerouveitis, and Sjögren's syndrome. Its diagnosis and management represent a challenge, so a multidisciplinary approach should be provided.

Keywords: Alkaptonuria; multiple autoimmune syndrome; polyautoimmunity; scleritis; sclerouveitis; uveitis.

Publication types

  • Case Reports

MeSH terms

  • Aged
  • Alkaptonuria* / complications
  • Alkaptonuria* / diagnosis
  • Autoimmune Diseases / complications
  • Autoimmune Diseases / diagnosis
  • Female
  • Hashimoto Disease / complications
  • Hashimoto Disease / diagnosis
  • Humans
  • Scleritis* / diagnosis
  • Scleritis* / drug therapy
  • Scleritis* / etiology
  • Sjogren's Syndrome* / complications
  • Sjogren's Syndrome* / diagnosis