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A woman in her 30s with a known case of polycythaemia vera [Janus Kinase 2 (JAK2) V617F-positive] presented to the clinic with progressive changes in skin and nail pigmentation. She had been receiving hydroxyurea 500 mg two times per day for the past 5 years following her diagnosis. Approximately 1 year after initiating therapy, she began noticing brownish discolouration of her face along with dark longitudinal streaks in her fingernails. On discontinuing or reducing the dose, she had a history of reduced pigmentations. Similarly, increasing the dose was associated with increased pigmentation. She denied taking any other medications, and her medical history was otherwise unremarkable.