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Faisal Alghubaywi,1,2 Mohammed I AlJasser,1– 3 Majid Alfadhel,1,3,4 Hind M Almohanna5 1College of Medicine, King Saud bin Abdulaziz University for Health Sciences, Riyadh, Saudi Arabia; 2Division of Dermatology, King Abdulaziz Medical City, Ministry of National Guard Health Affairs, Riyadh, Saudi Arabia; 3King Abdullah International Medical Research Center, Riyadh, Saudi Arabia; 4Genetics and Precision Medicine Department, King Abdullah Specialized Children Hospital, Riyadh, Saudi Arabia; 5Department of Dermatology and Dermatologic Surgery, Prince Sultan Military Medical City, Riyadh, Saudi ArabiaCorrespondence: Faisal Alghubaywi, Division of Dermatology, King Abdulaziz Medical City, Ministry of National Guard Health Affairs, P.O. Box 3660, Riyadh, 11481, Saudi Arabia, Tel +966-11-801 1111 ext. 15626, Fax +966-11-801 1111 Ext. 80-14229, Email Faisalalghubaywi@gmail.comAbstract: The distribution of lentigines offers great insight into the multitude of potential-associated genetic or acquired conditions. In this report, we described a unique presentation of lentigines limited to the palms and soles in a healthy individual. Personal and familial history, clinical examination, serological testing, and whole-genome sequencing were all unremarkable. The benign clinical features and absence of associated medical conditions support the likely lentigo simplex with an isolated palmoplantar distribution. To date, no similar distribution has been reported. This case extends the breadth of view into all possible presentations of lentigines.Keywords: lentiginosis, palmoplantar, isolated, case report