Bing-Neel syndrome: Two unexpected cases and a review of the literature

J Neurol Sci. 2015 Sep 15;356(1-2):19-26. doi: 10.1016/j.jns.2015.06.026. Epub 2015 Jun 16.

Abstract

Waldenström macroglobulinemia (WM) is a lymphoplasmacytic lymphoma characterized by the proliferation of small B-lymphocytes in the bone marrow that produce monoclonal immunoglobulin M (IgM). We describe two patients with WM who presented with neurological symptoms due to infiltration of lymphoplasmacytoid tumor cells in the central nervous system, a condition known as Bing-Neel syndrome. A literature review revealed that this syndrome is rare and commonly missed in clinical practice due to its variable presentation and a lack of awareness or knowledge. Brain and spinal magnetic resonance imaging may show a focal mass or diffuse infiltration. The diagnosis of Bing-Neel syndrome requires proof of IgM or lymphoplasmacytoid cells in cerebrospinal fluid or in a brain biopsy. Treatment with intravenous and/or intrathecal chemotherapy and cranial radiotherapy is described in literature with generally poor outcome, although a combination of these therapies seems to improve outcome. Nevertheless, insufficient data are currently available to make general treatment recommendations.

Keywords: Bing–Neel syndrome; Central nervous system lymphoma; Intrathecal chemotherapy; Lymphoplasmacytic lymphoma; MYD88; Radiotherapy; Waldenström macroglobulinemia.

Publication types

  • Case Reports
  • Review

MeSH terms

  • Aged
  • Aged, 80 and over
  • B-Lymphocytes / pathology
  • Bone Marrow / pathology
  • Cerebral Cortex / pathology
  • Drug Therapy
  • Female
  • Flow Cytometry
  • Humans
  • Hydrocephalus / etiology
  • Hydrocephalus / pathology
  • Immunoglobulin M / metabolism
  • Magnetic Resonance Imaging
  • Male
  • Mutation / genetics
  • Myeloid Differentiation Factor 88 / genetics
  • Myeloid Differentiation Factor 88 / metabolism
  • Radiotherapy
  • Spinal Cord / pathology
  • Waldenstrom Macroglobulinemia / diagnosis*
  • Waldenstrom Macroglobulinemia / genetics
  • Waldenstrom Macroglobulinemia / immunology*
  • Waldenstrom Macroglobulinemia / therapy*

Substances

  • Immunoglobulin M
  • MYD88 protein, human
  • Myeloid Differentiation Factor 88