Assessment of dysarthria with Frenchay dysarthria assessment (FDA-2) in patients with Duchenne muscular dystrophy

Abstract

Purpose

The purpose of this study was to test the psychometric properties of the Japanese version of Frenchay Dysarthria Assessment (FDA-2) and to use this tool to describe the features of speech in patients with Duchenne muscular dystrophy (DMD).

Materials and methods

The Japanese version of FDA-2 was culturally adapted, and reliability and validity were examined in 22 and 50 patients, respectively. The Japanese version of FDA-2 was administered to 51 patients with DMD. Multiple regression analysis was performed to identify factors related to FDA-2 scores.

Results

Inter-/intra-rater reliabilities (ICCs) and internal consistency (Cronbach’s α) for total scores were 0.76, 0.97, and 0.94 respectively. For construct validity, two-way ANOVA showed a significant interaction between the disorders and FDA-2 sections (p < 0.05). In DMD patients, the item of tongue at rest was most severely affected, reflecting tongue hypertrophy. Multiple regression analysis identified age, swallowing status, and ventilator use as significantly related.

Conclusions

The results showed that the Japanese version of FDA-2 has satisfactory reliability and validity. The present study demonstrated the features of dysarthria and related factors in patients with DMD.

Implications for rehabilitation

• In Duchenne muscular dystrophy (DMD), an absent or defective dystrophin protein causes progressive weakness of respiratory and oropharyngeal muscles, both of which are crucial contributors to speech production.

• This study shows that the Japanese version of FDA-2 has satisfactory reliability and validity compared to original version.

• The Japanese version of FDA-2 characterizes dysarthria in patients with DMD in this cohort.

Keywords:

• Analysis
• dysarthria
• language
• neurology
• rehabilitation
• speech

Acknowledgements

The authors thank the study participants for their time and effort. They also thank Aki Ito and Yuko Nomoto for the data acquisition. Finally, they would like to express the deepest appreciation to Emeritus Prof Pamela Enderby at University of Sheffield for her assistance on the project.

Disclosure statement

MK and ML are founding scientists of the startup company Connect Inc for the social implementation of university research results.

Additional information

Funding

This research received no specific grant from any funding agency in the public, commercial, or not-for-profit sectors.