Dysregulated TRK signalling has been proposed as a therapeutic target in CYLD defective tumours.2 Expression of TRKs is seen in the pulmonary cylindromas, supporting the consideration of emerging oral TRK inhibitors in the management of these tumours.15 In summary, we describe the pulmonary phenotype of this poorly studied rare disease, describe monitoring and management of pulmonary cylindromas and add to the debate on benign metastases. This evidence concerns the gene CYLD and neoplasm.