Langerhans cell histiocytosis: differences and similarities in long-term outcome of paediatric and adult patients at a single institutional centre

Hematology. 2015 Mar;20(2):83-92. doi: 10.1179/1607845414Y.0000000173. Epub 2014 May 29.

Abstract

Purpose: This study determined the frequency of clinical features, reactivations, sequelae, mortality, and overall survival (OS) and compared paediatric with adult Langerhans cell histiocytosis (LCH) patients.

Materials and methods: Ninety patients (60 paediatric and 30 adults) with LCH treated during 28 years were analysed retrospectively.

Results: Craniofacial lesion was the most frequent lesion at LCH presentation in children and adults. However, some differences were found. Orbital lesions were more frequent in paediatric than adult patients (P = 0.001). There was a tendency for mandible lesions to be more common in adults than the paediatric group (P = 0.0710). Mucocutaneous lesions were observed in a higher proportion in adults compared to paediatric patients (P = 0.0395). Reactivation episodes (36.8 versus 62.5%) and deaths (10.7 versus 24.0%) occurred in lower proportions in paediatric than adult patients, respectively. The probability of OS in 10 years for both groups was similar (P = 0.137).

Conclusion: The OS was similar in both groups despite clinical differences between paediatric and adult patients, and higher reactivation and death rates in adults.

Keywords: Adults; Children; Diabetes insipidus; Langerhans cells histiocytosis; Lytic bone lesions.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Adult
  • Aged
  • Anti-Inflammatory Agents / therapeutic use*
  • Child
  • Child, Preschool
  • Facial Bones / drug effects
  • Facial Bones / pathology
  • Facial Bones / surgery
  • Female
  • Gamma Rays / therapeutic use*
  • Histiocytosis, Langerhans-Cell / mortality
  • Histiocytosis, Langerhans-Cell / pathology
  • Histiocytosis, Langerhans-Cell / surgery*
  • Histiocytosis, Langerhans-Cell / therapy*
  • Humans
  • Infant
  • Lung / drug effects
  • Lung / pathology
  • Male
  • Middle Aged
  • Mucous Membrane / drug effects
  • Mucous Membrane / pathology
  • Mucous Membrane / surgery
  • Prednisone / therapeutic use*
  • Retrospective Studies
  • Survival Analysis

Substances

  • Anti-Inflammatory Agents
  • Prednisone