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To describe a series of children with extensive PNF or treatment refractory PLGG treated on a compassionate basis with trametinib. We report on six patients with NF-1 treated with trametinib on a compassionate basis at British Columbia Children's Hospital since 2017. Data were collected retrospectively from the patient record. RAPNO and volumetric criteria were used to evaluate the response of intracranial and extracranial lesions, respectively. Subjects were 21months to 14years old at the time of initiation of trametinib therapy and 3/6