Fatal B-cell lymphoma following chronic lymphocytic inflammation with pontine perivascular enhancement responsive to steroids

JAMA Neurol. 2013 Jul;70(7):915-8. doi: 10.1001/jamaneurol.2013.2016.

Abstract

Importance: Recent reports on chronic lymphocytic inflammation with pontine perivascular enhancement responsive to steroids (CLIPPERS) suggest that patients who have a relapse respond very well and that disease progression can be avoided if timely corticosteroid therapy is started. We report on a well-documented patient who presented with clinical, radiological, and pathological characteristics of CLIPPERS and who had an unfavorable outcome.

Observations: We present the clinical, imaging, laboratory, brain biopsy, and autopsy findings of a 57-year-old male patient with CLIPPERS who repeatedly responded well to high-dose corticosteroids. During follow-up, however, treatment failed, and he had a biopsy-confirmed diagnosis of lymphomatoid granulomatosis that evolved into fatal B-cell lymphoma of the central nervous system.

Conclusions and relevance: The clinical and imaging features of CLIPPERS include an abundance of differential diagnoses, and the follow-up periods of the described cases classified as CLIPPERS have been limited. Therefore, the question remains whether CLIPPERS is an actual new disease entity or represents a syndrome that includes different overlapping diseases and their prestages. Our case report shows that a typical presentation of CLIPPERS does not uniformly imply a favorable outcome, even when timely treatment regimens have been given.

Publication types

  • Case Reports

MeSH terms

  • Fatal Outcome
  • Humans
  • Lymphoma, B-Cell / drug therapy
  • Lymphoma, B-Cell / mortality
  • Lymphoma, B-Cell / pathology*
  • Lymphomatoid Granulomatosis / drug therapy
  • Lymphomatoid Granulomatosis / mortality
  • Lymphomatoid Granulomatosis / pathology*
  • Magnetic Resonance Imaging
  • Male
  • Middle Aged
  • Pons / metabolism
  • Pons / pathology*
  • Syndrome