Language

English

Publication Date

5-1-2024

Journal

British Journal of Haematology

DOI

10.1111/bjh.19380

PMID

38501390

PMCID

PMC12700493

PubMedCentral® Posted Date

12-13-2025

PubMedCentral® Full Text Version

Author MSS

Abstract

Optimal therapeutic approaches for advanced Langerhans cell histiocytosis (LCH) are not known. We assessed the safety and efficacy of combined chemotherapy with MAPK pathway inhibition in 10 patients with refractory systemic disease and/or LCH-associated neurodegeneration. Overall response rate was 9/10 (90%) for the entire cohort: 5/5 (100%) for patients with systemic disease and 6/7 (86%) for patients with central nervous system disease. BRAFV600E+ peripheral blood fraction decreased in 5/6 (83%). Toxicities included fever, skin rash, myalgias, neuropathy, cytopenias and hypocalcaemia. Prospective trials are required to optimize combination strategies, determine potential to achieve cure and compare outcomes to chemotherapy or MAPK inhibitor monotherapy.

Keywords

Female, Humans, Male, Antineoplastic Combined Chemotherapy Protocols, Histiocytosis, Langerhans-Cell, MAP Kinase Signaling System, Protein Kinase Inhibitors, Proto-Oncogene Proteins B-raf, Recurrence, Treatment Outcome

Published Open-Access

yes

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