Fifteen years of treatment with intravenous immunoglobulin in central nervous system Langerhans cell histiocytosis

Acta Paediatr. 2011 Jul;100(7):e36-9. doi: 10.1111/j.1651-2227.2010.02125.x. Epub 2011 Jan 11.

Abstract

Aim: There is currently no well-accepted therapy for central nervous system Langerhans cell histiocytosis (CNS-LCH), a neuroinflammatory disease clinically characterized by often progressive, neurological symptoms including ataxia, dysarthria, dysphagia, hypertonicity, intellectual impairment and behavioural abnormalities. We applied immunomodulative/anti-inflammatory treatment on a patient with progressive CNS-LCH disease.

Method: Intravenous immunoglobulin (IVIG) was administered monthly for 15 years to a patient with severe, image-verified neurodegenerative CNS-LCH.

Results: During the IVIG treatment, the neurological deterioration initially appeared to be haltered, but over time there was still some deterioration.

Conclusions: IVIG may be beneficial in partly haltering CNS-LCH neurodegeneration, but further studies are needed.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Central Nervous System Diseases / drug therapy*
  • Child
  • Child, Preschool
  • Disease Progression
  • Follow-Up Studies
  • Histiocytosis, Langerhans-Cell / drug therapy*
  • Humans
  • Immunoglobulins, Intravenous / therapeutic use*
  • Immunologic Factors / therapeutic use*
  • Infant
  • Male
  • Time Factors
  • Treatment Outcome

Substances

  • Immunoglobulins, Intravenous
  • Immunologic Factors