Unusual presentation Of Sjögren-associated neuropathy with plasma cell-rich infiltrate

Muscle Nerve. 2017 Apr;55(4):605-608. doi: 10.1002/mus.25475. Epub 2016 Nov 28.

Abstract

Introduction: Sjögren syndrome is thought to be a lymphocyte-driven process. Peripheral nervous system involvement occurs in about 20%-25% of patients. A sensory-predominant, large-fiber peripheral neuropathy is most common, and it is usually associated with a subacute to chronic presentation.

Methods: We report a rare case of an acute Sjögren-associated, sensory predominant, length-dependent peripheral neuropathy mimicking Guillain-Barré syndrome. The patient presented with sensory ataxia preceded by fever and polyarthralgia. She gave a history of years of dry eyes and dry mouth.

Results: She had a positive Shirmer test, abnormal salivary gland scan, and positive SS-A and SS-B antibodies. A sural nerve biopsy showed an unusual, dense, non-IgG4, polyclonal, plasma-cell perivascular infiltrate. The patient responded to treatment with weekly pulse intravenous methylprednisolone.

Conclusions: Sjögren syndrome can present with acute-onset, sensory predominant peripheral neuropathy. The role of plasma cells in Sjögren syndrome is unexplored and deserves further study. Muscle Nerve 55: 605-608, 2017.

Keywords: GBS mimic; Sjögren syndrome; acute axonal neuropathy; acute sensory neuropathy; plasma cell infiltrate; sicca symptoms.

MeSH terms

  • Administration, Intravenous
  • Aged
  • Anti-Inflammatory Agents / administration & dosage
  • Antigens, CD / metabolism
  • Female
  • Humans
  • Magnetic Resonance Imaging
  • Methylprednisolone / administration & dosage
  • Neurologic Examination
  • Neutrophil Infiltration / physiology*
  • Peripheral Nervous System Diseases / complications*
  • Peripheral Nervous System Diseases / diagnostic imaging
  • Plasma Cells / pathology*
  • Sjogren-Larsson Syndrome / blood*
  • Sjogren-Larsson Syndrome / complications*
  • Sjogren-Larsson Syndrome / diagnostic imaging
  • Sjogren-Larsson Syndrome / drug therapy
  • Sural Nerve / pathology
  • Sural Nerve / ultrastructure

Substances

  • Anti-Inflammatory Agents
  • Antigens, CD
  • Methylprednisolone