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Huntington's disease (HD) is a neurodegenerative disorder characterized by motor, cognitive, and psychiatric symptoms and by a progressive degeneration of neurons in basal ganglia and in brain cortex. Brain‐derived neurotrophic factor (BDNF) is a pro‐survival factor for striatal neurons. Some evidence implicates a brain BDNF deficiency, related to mutated huntingtin expression, in the selective vulnerability of striatal neurons in HD. We compared BDNF serum levels in 42 patients with HD (range 28–72 years, mean age 51.9 ± 11.5), and 42 age‐matched healthy subjects (range 25–68 years, mean age 48.2 ± 12.5). We evaluated the potential relationship between BDNF serum levels, CAG repeat number (range 40–54, mean 44.8 ± 3.4) and duration of illness (range 6–228 months, mean 103.6 ± 62.1). Serum BDNF levels were significantly lower in patients than in age‐matched healthy subjects. Lower …