Is familial adenomatous polyposis associated with sensorineural hearing loss?

Abstract

Abstract

The mutation causing familial adenomatous polyposis (FAP) affects the adenomatous polyposis coli (Apc) gene, which has a role in the cytoskeleton and has been shown to be important in the structure of supporting cells in the cochlea. One previous study suggested that FAP sufferers may have sensorineural hearing loss. In order to demonstrate whether this is the case we invited patients known to suffer from familial adenomatous polyposis to take part in our study. Audiograms were performed and compared to normal values for that patient's age and gender calculated using ISO standard data. Thirteen patients were included in the study analysis. No conductive hearing losses were identified. A statistically significant greater hearing loss was identified at 500 Hz (2.8 dBHL, P = 0.03) and 1000 Hz (2.5 dBHL, P = 0.05). No audiometric difference could be identified between the patients with FAP and attenuated FAP. A power calculation demonstrated that the study was of adequate size. This study did not demonstrate a clinically significant difference in hearing loss between the FAP group and the calculated normal values.

Sumario

La mutación que causa la poliposis adenomatosa familiar (FAP) afecta el gene polipomatosis adenomatosa coli (Apc), que tiene un papel cuya importancia en el citoesqueleto y en la estructura de las células de sostén de la cóclea se ha demostrado. Un estudio previo sugirió que quienes sufren la FAP pueden tener una pérdida auditiva sensorineural. Con objeto de demostrar si esto es así, invitamos a pacientes que (sabíamos) sufrían la enfermedad para tomar parte en este estudio. Los audiogramas que se practicaron fueron comparados con los valores normales de acuerdo con la edad y el género de los pacientes, usando los datos ISO estándar. Se incluyeron trece pacientes para el análisis del estudio. No se identificaron pérdidas auditivas conductivas. Si hubo una pérdida auditiva mayor, estadísticamente significativa en 500 Hz (2.8 dBHL, P = 0.03) y en 1000 Hz (2.5 dBHL, P = 0.05). No se pudo identificar una diferencia audiométrica entre los pacientes con FAP o con FAP atenuada. Un cálculo especial demostró que el estudio fue de un tamaño adecuado. Este estudio no mostró una diferencia clínicamente significativa de la pérdida auditiva entre el grupo con FAP y los valores calculados como normales.

Keywords:

• Adenomatous polyposis coli
• Hearing loss, Sensorineural
• Audiometry, Pure tone

Acknowledgements

This work was funded by the NHS Tayside Acute Division Grant Scheme.

Thanks also to Dr. J. Berg and Ms. J. Dunlop, of the Department of Genetics, Ninewells Hospital, for their assistance with identification of patients.

Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.