Xanthinuria is a very rare disease in cats. Its etiology may have a genetic origin or may be due to an iatrogenic xanthine-dehydrogenase inhibition that finally results in urolithiasis. The present work reports two cases of xanthine urolithiasis in European Shorthair unrelated male and female cats. Both uroliths were analyzed by stereoscopic microscopy, infrared spectroscopy and scanning electron microscopy. Besides the report of these two clinical cases, a detailed pathophysiologic review and some updated recommendations for diagnosis and treatment for this condition were done.