Punctate palmoplantar keratoderma is a rare hereditary palmoplantar keratoderma. Herein we report a 59-year-old male, otherwise healthy, who presented with a 25-year history of asymptomatic persistent slowly progressing skin lesions on both hands. The parents are non-consanguineous and none of his family members had similar lesions. Skin examination revealed multiple tiny keratotic pits on both palms. Punch skin biopsy from the palmar lesion revealed epidermal depression with an overlying column of compact orthokeratosis. Based on the above clinicopathological findings, a diagnosis of punctate palmoplantar keratoderma type 1 was made. The patient was started on 40% urea and 20% salicylic acid ointment for months but with little improvement.
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