Off-label use is common in dermatology, and is inevitable for rare cutaneous diseases such as perforating dermatosis. Allopurinol is traditionally considered to be a drug for hyperuricemia only, but the recent demonstration of its efficacy in congestive heart failure has spurred renewed interest in its application in other clinical specialties. In dermatology, allopurinol is best known for its severe cutaneous adverse reactions. Recent genomic studies conducted in Taiwan have discovered useful HLA markers for determining the susceptibility of Stevens-Johnson syndrome and toxic epidermal necrolysis associated with allopurinol. Allopurinol has also been used in a number of dermatologic disorders including acquired reactive perforating collagenosis, sarcoidosis, psoriasis and granulomas caused by methacrylate microspheres, silicon and tattoos. Allopurinol may express its therapeutic effects via its antioxidation or anti-inflammatory properties, or its ability to improve vascular function.