Neuro-Sjögren: A clinical-radiological paradox affecting the central nervous system

J Neuroimmunol. 2023 May 15:378:578085. doi: 10.1016/j.jneuroim.2023.578085. Epub 2023 Apr 7.

Abstract

Neurological manifestations can occur in up to 67% of patients with primary Sjögren's Syndrome, also known as Neuro-Sjogren's syndrome (NSS), and a 5% can present central nervous system involvement, with severe and possibly lethal consequences. We present the radiological follow-up of a patient with NSS who consulted for limb weakness and visual loss, and fourteen years later developed sicca symptoms. She was diagnosed with a saliva gland biopsy, and started treatment with steroids, cyclophosphamide, and then rituximab, achieving a favourable clinical response and stabilization of lesions. We discuss key aspects regarding the clinical presentation, diagnosis, imaging, and treatment of this elusive disease.

Keywords: Immunosuppressive agents; Magnetic resonance imaging; Nervous system diseases; Sjögren's syndrome.

MeSH terms

  • Central Nervous System / diagnostic imaging
  • Central Nervous System Diseases* / diagnostic imaging
  • Central Nervous System Diseases* / drug therapy
  • Cyclophosphamide / therapeutic use
  • Female
  • Humans
  • Rituximab / therapeutic use
  • Sjogren's Syndrome* / complications
  • Sjogren's Syndrome* / diagnostic imaging

Substances

  • Rituximab
  • Cyclophosphamide