We aimed to assess anxiety of children with primary ciliary dyskinesia (PCD) and their primary caregivers at the beginning of Coronavirus disease-2019 (COVID-19) pandemic and change in levels of anxiety in first year with prolongation of pandemic. This was a two-step study; first step was questionnaire-based, conducted via teleconference. In first step, 29 patients and 105 healthy children and their mothers were participated; 25 children with PCD and their mothers were in second step. Demographic characteristics, clinical informations were recorded. Children’s and mothers’ state and trait anxiety levels were assessed and compared. Anxiety levels of mothers of patients were assessed according to clinical characteristics of children. Mothers’ knowledge of COVID-19 and effect of teleconference on their anxiety was evaluated. State anxiety levels in the first year of pandemic of children with PCD and their mothers were also compared. Compared to control group, state anxiety of children in 13-18 age group and trait anxiety of their mothers were lower (p