[Multicentric Castleman disease-like case characterized by lymphadenopathy and polyclonal hypergammaglobulinemia associated with so-called Mikulicz disease]

Rinsho Ketsueki. 2007 Mar;48(3):229-34.
[Article in Japanese]

Abstract

A 73-year-old male was admitted in January 1999 with hyperimmunoglobulinemia with a serum IgG level of 6530 mg/dl, bilateral eyelid tumors, bilateral submandibular swelling, and swelling of the superficial lymph nodes. A left submandibular gland biopsy showed severe chronic sialoadenitis with fibrosis. A left cervical lymph node biopsy showed invasion by many mature lymphocytes and plasma cells, but no lymphoma cells. The patient was diagnosed as having so-called Mikulicz disease associated with a disease similar to multicentric Castleman disease (MCD) characterized by multicentric lymphadenopathy and polyclonal hyperimmunoglobulinemia. Steroid therapy (prednisolone, 20 mg/day) was effective in reducing the symptoms, the bilateral eyelid tumors and the swelling of the bilateral submandibular glands and superficial lymph nodes. The lack of any increase of serum IL-6 suggested that this case had a hitherto unknown etiology, other than MCD.

Publication types

  • Case Reports

MeSH terms

  • Aged
  • Castleman Disease
  • Diagnosis, Differential
  • Humans
  • Hypergammaglobulinemia / drug therapy
  • Hypergammaglobulinemia / etiology*
  • Immunoglobulin G / blood*
  • Interleukin-6 / blood
  • Lymphatic Diseases / drug therapy
  • Lymphatic Diseases / etiology*
  • Male
  • Mikulicz' Disease / complications*
  • Mikulicz' Disease / diagnosis*
  • Mikulicz' Disease / drug therapy
  • Prednisolone / therapeutic use
  • Treatment Outcome

Substances

  • Immunoglobulin G
  • Interleukin-6
  • Prednisolone