The aim of this study was to investigate a possible correlation between handedness and laterality of hearing impairment due to otosclerosis. All patients operated for otosclerosis between August 2008 and February 2014 were queried about their handedness. The study group consisted of 218 right-handed and 21 left-handed (8.9%) patients [139 female (58.2%) and 100 male] with an age range of 18-75 years (mean 46.1 years). One-hundred and fifty-seven patients had a bilateral otosclerosis (BO) and 82 (34.3%) had a unilateral otosclerosis (UO). There were 11 left-handed male and 10 left-handed female (11% vs. 7.2 %, p = 0.305). In patients with UO, the left ear (LE) was affected in 6/11 (58.3%) left handed ones, and the right ear (RE) in 41/71 (57.7%) right handed ones (p = 0.842). In patients with BO, the LE was more affected in the left-handed ones, and the RE in the right-handed ones (7/10, 70% and 87/147, 59.2 %, respectively, p = 0.5). Overall, 13/21 (61.9%) left-handed patients presented with only/mostly left-sided otosclerosis, while 128/218 (58.7%) right-handed patients presented with only/mostly right-sided otosclerosis (p = 0.584). Clinical relevance of presented findings is unclear yet nevertheless current study may contribute one more element in the multifactorial process of otosclerosis-related hearing loss.