Aquagenic wrinkling of the palms (AWP) is a rare condition, defined clinically by the appearance or accentuation of an asymmetrical, translucent to white, papular eruption on the palms after immersion in water. It is associated with cystic fibrosis (CF), and approximately half of all reported cases occur in patients with documented CF. We report a case of AWP in a young woman with CF, where the AWP was related to treatment with the aminoglycoside antibiotic, tobramycin. Although the mechanism of AWP is unknown, influx of water across an osmotic gradient into eccrine ducts has been proposed. Aminoglycosides may affect AWP by blocking various cell surface channels and receptors, which may influence cell-volume regulation.