Paediatrics Publications

Title

Orofacial strength, dysarthria, and dysphagia in congenital myotonic dystrophy.

Document Type

Article

Publication Date

9-1-2018

Journal

Muscle & Nerve

Volume

58

Issue

3

First Page

413

Last Page

417

URL with Digital Object Identifier

https://doi.org/10.1002/mus.26176

Abstract

Introduction

Herein we present an exploratory study of orofacial function in children with congenital myotonic dystrophy (CDM) vs. healthy controls.

Methods

We evaluated 41 children with CDM and 29 healthy controls for speech and swallow function and for lingual and labial strength.

Results

The Iowa Oral Performance Instrument (IOPI), measuring tongue strength, and a lip force meter (LFM), measuring lip strength, had excellent interrater reliability with intraclass correlation coefficients (ICCs) of 0.75 (n = 19, P < 0.001) and 0.96 (n = 20, P < 0.001), respectively. Mean overall lingual strength was 3.5-fold less and labial strength was about 7-fold less in CDM patients than in healthy controls. Eighteen of 24 children with CDM demonstrated dysarthria and an additional 11 participants were nonverbal. Dysarthria correlated moderately with lingual strength, age, and dysphagia. Strength measures correlated moderately with dysphagia.

Discussion

Children with CDM have impaired orofacial functioning that affects communication and swallowing. Reliability of strength measures may be useful for future therapeutic trials. Muscle Nerve 58: 413-417, 2018.

Notes

Article available at Muscle & Nerve

https://doi.org/10.1002/mus.26176

© 2018 Wiley Periodicals, Inc.

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