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High-speed Video Microscopy Analysis for First-line Diagnosis of Primary Ciliary Dyskinesia
Published on: January 19, 2022
A longitudinal evaluation of hearing and ventilation tube insertion in patients with primary ciliary dyskinesia
Tobias Nicolai Andersen1, Mikkel Christian Alanin1, Christian von Buchwald1
1Department of Otorhinolaryngology, Head and Neck Surgery and Audiology, Copenhagen University Hospital, Rigshospitalet, Denmark.
Insights
Hearing improves with age in primary ciliary dyskinesia (PCD) patients. Ventilation tube insertion also aids hearing but may lead to prolonged otorrhoea in nearly half of cases.
Area of Science:
- Otolaryngology
- Genetics
- Pediatrics
Background:
- Primary ciliary dyskinesia (PCD) is a genetic disorder causing oto-sino-pulmonary symptoms.
- Otitis media with effusion (OME) is a frequent childhood complication in PCD patients.
- Management strategies for OME in PCD lack consensus.
Purpose of the Study:
- To assess long-term hearing outcomes in patients with PCD.
- To investigate the impact of ventilation tube (VT) insertion on hearing in PCD.
- To determine the incidence of postoperative otorrhoea following VT insertion.
Main Methods:
- Retrospective analysis of longitudinal data from 57 PCD patients over 30 years.
- Evaluation of pure tone average (PTA) and tympanometry from 278 audiometries.
- Review of medical records for VT insertion and duration of postoperative otorrhoea.
Main Results:
- Hearing, measured by mean PTA, significantly improved with age in PCD patients (p < 0.0001).
- Ventilation tube insertion demonstrated a positive effect on hearing levels.
- Prolonged otorrhoea (≥4 weeks) occurred in 48% of patients after VT insertion.
Conclusions:
- Hearing in PCD patients naturally improves with age.
- Ventilation tube insertion is beneficial for hearing in PCD.
- A significant proportion of PCD patients experience prolonged otorrhoea post-VT insertion, indicating a trade-off.
Introduction:
Primary ciliary dyskinesia (PCD) is an autosomal recessive genetic disease, which primarily manifests with oto-sino-pulmonary symptoms. Otitis media with effusion (OME) is common from early childhood. The existing literature on OME management in PCD is conflicting. The goals of the present study were firstly to evaluate the long-term hearing in PCD patients and secondly to determine the influence of ventilation tube (VT) insertion on hearing and postoperative otorrhoea.
Methods:
A longitudinal retrospective study extracting the hearing level (pure tone average (0.5, 1, 2, 4 kHz, PTA)) and tympanometry from the medical records. Furthermore, the patient files were reviewed for VT insertion and postoperative otorrhoea. Postoperative otorrhoea was defined prolonged when it lasted for four weeks or longer.
Results:
Fifty-seven patients were identified in a 30-year period, age 2-72 years, and 278 audiometries were evaluated. The median number of audiometries per patient was 3 (range 1-29) and the median follow-up was 60 months (range 0-351 months). The mean PTA was 34 dB HL in patients below six years of age and improved significantly (p < 0.0001) with age. VT insertion improved hearing; however, 48% of patients with VT insertion experienced prolonged otorrhoea.
Conclusion:
In this study of PCD patients the hearing improved as a function of age as well as following VT insertion. However, VT insertion was also associated with prolonged otorrhoea.
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