TY - JOUR T1 - CiliarMove: new software for evaluating ciliary beat frequency helps find novel mutations by a Portuguese multidisciplinary team on primary ciliary dyskinesia JF - ERJ Open Research JO - erjor DO - 10.1183/23120541.00792-2020 VL - 7 IS - 1 SP - 00792-2020 AU - Pedro Sampaio AU - Mónica Ferro da Silva AU - Inês Vale AU - Mónica Roxo-Rosa AU - Andreia Pinto AU - Carolina Constant AU - Luisa Pereira AU - Carla M. Quintão AU - Susana S. Lopes Y1 - 2021/01/01 UR - http://openres.ersjournals.com/content/7/1/00792-2020.abstract N2 - Evaluation of ciliary beat frequency (CBF) performed by high-speed videomicroscopy analysis (HVMA) is one of the techniques required for the correct diagnosis of primary ciliary dyskinesia (PCD). Currently, due to lack of open-source software, this technique is widely performed by visually counting the ciliary beatings per a given time-window. Our aim was to generate open-source, fast and intuitive software for evaluating CBF, validated in Portuguese PCD patients and healthy volunteers.Nasal brushings collected from 17 adult healthy volunteers and 34 PCD-referred subjects were recorded using HVMA. Evaluation of CBF was compared by two different methodologies: the new semi-automated computer software CiliarMove and the manual observation method using slow-motion movies. Clinical history, nasal nitric oxide and transmission electron microscopy were performed for diagnosis of PCD in the patient group. Genetic analysis was performed in a subset (n=8) of suspected PCD patients.The correlation coefficient between the two methods was R2=0.9895. The interval of CBF values obtained from the healthy control group (n=17) was 6.18–9.17 Hz at 25°C. In the PCD-excluded group (n=16), CBF ranged from 6.84 to 10.93 Hz and in the PCD group (n=18), CBF ranged from 0 to 14.30 Hz.We offer an automated open-source programme named CiliarMove, validated by the manual observation method in a healthy volunteer control group, a PCD-excluded group and a PCD-confirmed group. In our hands, comparisons between CBF intervals alone could discern between healthy and PCD groups in 78% of the cases.New software for easy and faster evaluation of ciliary beat frequency in an unbiased manner validated in three groups: healthy volunteers; patients with confirmed primary ciliary dyskinesia (PCD); and patients with PCD excluded https://bit.ly/2Kvwwes ER -