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  • Efficacy and improved tolerability of combination therapy with interleukin-1 blockade and MAPK pathway inhibitors for the treatment of Erdheim-Chester disease

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Correspondence
Efficacy and improved tolerability of combination therapy with interleukin-1 blockade and MAPK pathway inhibitors for the treatment of Erdheim-Chester disease
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  1. Corrado Campochiaro1,2,
  2. Giulio Cavalli1,2,
  3. Nicola Farina1,2,
  4. Alessandro Tomelleri1,2,
  5. Giacomo De Luca1,2,
  6. Lorenzo Dagna1,2
  1. 1 Unit of Immunology, Rheumatology, Allergy and Rare Diseases, IRCCS San Raffaele Hospital, Milan, Italy
  2. 2 Vita-Salute San Raffaele University, Milan, Italy
  1. Correspondence to Dr Corrado Campochiaro, Unit of Immunology, Rheumatology, Allergy and Rare Diseases, IRCCS San Raffaele Hospital, 20132 Milano, Italy; campochiaro.corrado{at}hsr.it

https://doi.org/10.1136/annrheumdis-2019-216610

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    We have read with interest the article by Cohen-Aubart et al 1 reporting the efficacy of infliximab in the treatment of Erdheim-Chester disease (ECD), a rare non-Langerhans histiocytosis. The anti-Tumor Necrosis Factor-α agent demonstrated a variable degree of efficacy, suggesting that infliximab might represent a therapeutic option for moderately severe ECD cases; similar results had been observed with anakinra.2 3 Nonetheless, following the identification of causative mutations along the mitogen-activated protein kinase(MAPK) pathway,4 severe forms of ECD are currently treated with targeted small molecule agents. Specifically, the BRAF inhibitor vemurafenib and the MAPK or extracellular-signal regulated kinase (MEK) inhibitor cobimetinib have been successfully used to treat life-threatening forms of ECD. Although life-saving, these drugs are associated with severe toxicity, which often mandates treatment discontinuation. …

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