Papillon‐Lefèvre syndrome (PLS) is a rare, autosomal recessive disorder of keratinization, characterized by palmoplantar keratoderma (PPK) and severe chronic periodontitis which leads to an early loss of primary and permanent teeth. Oral retinoids, such as etretinate, isotretinoin and acitretin, represent the most important therapeutic options for keratoderma of PLS, with varying results in terms of efficacy. We report here the first case of 55‐year‐old woman diagnosed with PLS treated with alitretinoin.
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