A 25-year-old female patient presented with palmoplantar keratoderma for 25 years and recurrent gingivitis and periodontitis for 20 years.The dermatological examination revealed slight bilateral palmoplantar keratoderma with desquamation and keratinizing erythema distributed in the fingertips,extensor aspects of metacarpophalangeal joints,and foot dorsum.The dental examination showed malocclusion,gingivitis,and periodontitis.The whole exome sequencing revealed a homozygous missense mutation in CTSC C.1015C>T.The patient was diagnosed with papillon-lefèvre syndrome and underwent orthodontic treatment without any additional treatment for palmoplantar keratoderma.