Filiform hyperkeratosis is a disease that often involves the palmoplantar region and rarely can involve other parts of the body. It is usually associated with malignancies or systemic diseases, but rarely it can also occur as idiopathic. An 18-year-old woman was admitted to our clinic with the complaints of dermatological skin extensions on her hands and feet for 6 months. She stated that there was no contact of any chemical agent and did not receive any topical or systemic treatment for these complaints. The patient was otherwise healthy and her past medical history and family history were non-contributory. The systemic examination was evaluated as normal. In dermatologic examination, painful, filiform papules were observed in both palmoplantar regions. Hyperkeratosis and focal regular acanthosis was detected in the skin biopsy from the palmar region of the patient. There was no individual with a similar lesion in in her family, and no pathological findings were found in biochemistry, hemogram and tumor markers. Patient was evaluated as idiopathic palmoplantar filiform like hyperkeratosis with these findings and 20 mg/day isotretinoin treatment was started. It was observed that the lesions almost completely retreat after 2 months of treatment.