Non-paraneoplastic autoimmune retinopathy that developed in fellow eye 10 years after onset in first eye: a case report

BMC Ophthalmol. 2020 Apr 6;20(1):132. doi: 10.1186/s12886-020-01414-z.

Abstract

Background: Evidence-based criteria for the treatment of autoimmune retinopathy (AIR) have not been established. The pathology and clinical features of each antibody causing AIR, and its long-term course are still undetermined. We report our findings in a case of non-paraneoplastic AIR (npAIR) that developed in the fellow eye 10 years after the onset in the first eye.

Case presentation: Our patient had photophobia in both eyes and a rapidly progressing visual field defect in his right eye at the initial examination. He was diagnosed with non-paraneoplastic AIR based on the clinical findings and immunoblot analyses for anti-retinal antibodies, and he was treated with steroids. Ten years later, a visual field defect developed in the fellow eye, and a diagnosis of npAIR was made. Immunoblot analyses were positive for anti-α-enolase antibodies. He was treated with steroids, immunosuppressants, and plasma exchange. However, the response to the treatment was poor and both eyes eventually became blind.

Conclusions: As best we know, this is the first case report of npAIR that developed in the fellow eye over 10 years after the development in the first eye. Long-term follow-up and a search for tumor lesions are necessary in cases of npAIR. Further understanding of the long-term course of AIR can contribute to an understanding of the pathology and treatment of npAIR.

Keywords: Alpha-enolase; Autoimmune retinopathy; Non paraneoplastic autoimmune retinopathy.

Publication types

  • Case Reports

MeSH terms

  • Autoantibodies / blood
  • Autoimmune Diseases / diagnosis
  • Autoimmune Diseases / etiology*
  • Autoimmune Diseases / therapy
  • Blindness / etiology
  • Electroretinography
  • Fluorescein Angiography
  • Glucocorticoids / therapeutic use
  • Humans
  • Immunoblotting
  • Immunosuppressive Agents / therapeutic use
  • Male
  • Middle Aged
  • Paraneoplastic Syndromes, Ocular / diagnosis
  • Paraneoplastic Syndromes, Ocular / etiology*
  • Paraneoplastic Syndromes, Ocular / therapy
  • Phosphopyruvate Hydratase / immunology
  • Plasma Exchange
  • Retinal Diseases / diagnosis
  • Retinal Diseases / etiology*
  • Retinal Diseases / therapy
  • Time Factors
  • Tomography, Optical Coherence
  • Visual Acuity
  • Visual Field Tests

Substances

  • Autoantibodies
  • Glucocorticoids
  • Immunosuppressive Agents
  • Phosphopyruvate Hydratase