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Abstract We report a case of congenital multisystem Langerhans cell histiocytosis with cutaneous and hematopoietic involvement. After the failure of first‐line (vinblastine and prednisolone) and second‐line (vincristine and cytarabine) therapies, treatment with cobimetinib, a mitogen‐activated protein kinase (MEK) inhibitor, led to the remission of disease and a sustained response after 11 months of ongoing treatment. Protein kinase inhibitors targeting BRAF or MEK could represent a promising future therapeutic option, also in children with LCH.
Benjelloun et al. (Thu,) studied this question.