IgG4-Related Sialoadenitis with a Skin Lesion and Multiple Mononeuropathies Suggesting Coexistent Cryoglobulinemic Vasculitis

Intern Med. 2016;55(10):1355-61. doi: 10.2169/internalmedicine.55.5332. Epub 2016 May 15.

Abstract

A 68-year-old man was admitted because of weakness of the left leg, dysesthesiae of the extremities and bilateral lower extremity purpura. A neurological examination showed mononeuritis multiplex with laboratory evidence of hypocomplementemia, cryoglobulinemia and leukocytoclastic vasculitis in the biopsy of a skin specimen. The patient also exhibited bilateral submandibular gland swelling, elevated serum IgG4 levels and infiltration of a large number of IgG4-positive plasma cells in the submandibular glands. These findings were consistent with both cryoglobulinemic vasculitis and IgG4-related disease. The administration of oral prednisolone (1 mg/kg/day) resolved the neurological manifestations and the swelling of the submandibular glands and cryoglobulinemia.

MeSH terms

  • Aged
  • Cryoglobulinemia / complications*
  • Cryoglobulinemia / drug therapy
  • Humans
  • Immunoglobulin G / blood*
  • Male
  • Mononeuropathies / complications*
  • Plasma Cells / pathology
  • Prednisolone / therapeutic use
  • Sialadenitis / complications*
  • Submandibular Gland / pathology
  • Vasculitis / complications*
  • Vasculitis / drug therapy
  • Vasculitis, Leukocytoclastic, Cutaneous

Substances

  • Immunoglobulin G
  • Prednisolone

Supplementary concepts

  • Erythema elevatum diutinum