Egyptian experience in Langerhans cells histiocytosis: frequent multisystem affection and reactivation rates

Pediatr Hematol Oncol. 2020 Nov;37(8):696-706. doi: 10.1080/08880018.2020.1790703. Epub 2020 Jul 24.

Abstract

Background: Histiocytoses are unique disorders; their clinical presentations vary from self-healing lesions to life-threatening disseminated disease. Objectives: We aimed to evaluate the different clinical presentations, frequency of reactivations, and treatment outcome of Langerhans cell histiocytosis among Egyptian children. Methods: we restrospectively analyzed the data of 37 Langerhans cell histiocytosis patients (LCH) registered at Ain Shams University Children's Hospital for clinicopathological features, treatment modalities and their outcomes. Results: Twenty seven (73%) of the studied patients with LCH had multisystem disease (MS), 24 (88.9%) of them had risk organ involvement (MS RO+) and only 3 without risk organ (MS RO-). Most of the patients received LCH III protocols. Eleven patients (29.7%) had reactivations with median time till reactivation of 17 months (IQR 5-23).Reactivation rates were 40% and 50% in patients with no evidence of active disease (NAD) and those with active disease better (AD better) at week 6 evaluation respectively (p = 0.71).We report 9 deaths (all had MS RO+, two died after reactivation and 7 had progressive disease. The 5 years EFS and OS were 49.4% and 81.2% respectively. Risk stratification did not significantly affect the EFS or OS (p = 0.64 and p = 0.5 respectively). Conclusion: A high reactivation rate was encountered in children with LCH and MS-RO + irrespective of 6 weeks response to induction therapy. A high mortality in patients with progressive disease necessitates a possible earlier aggressive salvage in such group.

Keywords: Langerhans cells; reactivations; survival rates.

MeSH terms

  • Child
  • Child, Preschool
  • Cladribine / administration & dosage
  • Cyclosporine / administration & dosage
  • Disease Progression
  • Egypt
  • Female
  • Histiocytosis, Langerhans-Cell / drug therapy*
  • Histiocytosis, Langerhans-Cell / mortality
  • Histiocytosis, Langerhans-Cell / physiopathology
  • Humans
  • Infant
  • Langerhans Cells / pathology
  • Liver / drug effects
  • Liver / pathology
  • Lymph Nodes / drug effects
  • Lymph Nodes / pathology
  • Lymphohistiocytosis, Hemophagocytic / complications*
  • Lymphohistiocytosis, Hemophagocytic / drug therapy
  • Lymphohistiocytosis, Hemophagocytic / mortality
  • Lymphohistiocytosis, Hemophagocytic / physiopathology
  • Male
  • Recurrence
  • Retrospective Studies
  • Risk Factors
  • Survival Rate
  • Treatment Outcome

Substances

  • Cladribine
  • Cyclosporine