Assessment of speech in early-onset ataxia : A pilot study
(2014) In Developmental Medicine and Child Neurology 56(12). p.1202-1206- Abstract
Aim: The aim of the study was to determine whether paediatric ataxia speech subscores are reliably applicable for international early-onset ataxia (EOA) databases. If so, we reasoned that ataxia speech subscores should be associated with ataxia scores and involve high interobserver agreement, including those for internationally applicable Scale for Assessment and Rating of Ataxia (SARA) syllable repetition tasks (SARASRT). Method: Three independent paediatric neurologists and a speech therapist scored speech in 52 healthy children (mean age 10y, range 4-16y) and 40 individuals with EOA (mean age 15y, range 5-34y). We compared ataxia speech subscores for the association with age and ataxia scores as well as interobserver reliability.... (More)
Aim: The aim of the study was to determine whether paediatric ataxia speech subscores are reliably applicable for international early-onset ataxia (EOA) databases. If so, we reasoned that ataxia speech subscores should be associated with ataxia scores and involve high interobserver agreement, including those for internationally applicable Scale for Assessment and Rating of Ataxia (SARA) syllable repetition tasks (SARASRT). Method: Three independent paediatric neurologists and a speech therapist scored speech in 52 healthy children (mean age 10y, range 4-16y) and 40 individuals with EOA (mean age 15y, range 5-34y). We compared ataxia speech subscores for the association with age and ataxia scores as well as interobserver reliability. Results: In healthy children, ataxia speech subscores were moderately associated with age (International Cooperative Ataxia Rating Scale [ICARS]: r=-0.515; SARA: r=-0.321; p<0.05) and with ataxia scores (ICARS: r=0.552; SARA: r=0.336; p<0.05), and revealed slight to moderate interobserver agreement (ICARS-intraclass correlation coefficient [ICC]: 0.380; SARA-ICC: 0.185; SARASRT-ICC: 0.509). In EOA, speech subscores have a strong association with ataxia scores (ICARS: r=0.735; SARA: r=0.730; p<0.001) and revealed substantial to nearly perfect interobserver agreement (ICARS-ICC: 0.812; SARA-ICC: 0.854; SARASRT-ICC: 0.724). Interpretation: Early-onset ataxia speech subscores are associated with ataxia and also reveal high interobserver agreement, including those internationally applicable to SARASRT. We conclude that SARASRT appears to be applicable for EOA databases. However, before syllable repetition tasks are included, we would advise to wait for the results published by the international Childhood Ataxia and Cerebellar Group. What this paper adds: Healthy speech subscores are moderately associated with age. Early onset ataxia (EOA) speech subscores are clearly associated with ataxia. Early-onset ataxia speech subscores reveal high interobserver agreement. Scale for Assessment and Rating of Ataxia syllable repetition tasks appear applicable for EOA databases.
(Less)
- author
- Kuiper, Marieke J. ; Brandsma, Rick ; Lawerman, Tjitske F. ; Lunsing, Roelineke J. ; Keegstra, Anne L. ; Burger, Huibert ; De Koning, Tom J. LU ; Tijssen, Marina A.J. and Sival, Deborah A.
- publishing date
- 2014-12-01
- type
- Contribution to journal
- publication status
- published
- subject
- in
- Developmental Medicine and Child Neurology
- volume
- 56
- issue
- 12
- pages
- 5 pages
- publisher
- John Wiley & Sons Inc.
- external identifiers
-
- scopus:84911378489
- pmid:24942085
- ISSN
- 0012-1622
- DOI
- 10.1111/dmcn.12517
- language
- English
- LU publication?
- no
- id
- 8debe29f-cbc0-415e-bc8b-0b2b5f6c9605
- date added to LUP
- 2020-02-26 10:07:44
- date last changed
- 2024-03-04 15:06:48
@article{8debe29f-cbc0-415e-bc8b-0b2b5f6c9605,
abstract = {{<p>Aim: The aim of the study was to determine whether paediatric ataxia speech subscores are reliably applicable for international early-onset ataxia (EOA) databases. If so, we reasoned that ataxia speech subscores should be associated with ataxia scores and involve high interobserver agreement, including those for internationally applicable Scale for Assessment and Rating of Ataxia (SARA) syllable repetition tasks (SARASRT). Method: Three independent paediatric neurologists and a speech therapist scored speech in 52 healthy children (mean age 10y, range 4-16y) and 40 individuals with EOA (mean age 15y, range 5-34y). We compared ataxia speech subscores for the association with age and ataxia scores as well as interobserver reliability. Results: In healthy children, ataxia speech subscores were moderately associated with age (International Cooperative Ataxia Rating Scale [ICARS]: r=-0.515; SARA: r=-0.321; p<0.05) and with ataxia scores (ICARS: r=0.552; SARA: r=0.336; p<0.05), and revealed slight to moderate interobserver agreement (ICARS-intraclass correlation coefficient [ICC]: 0.380; SARA-ICC: 0.185; SARASRT-ICC: 0.509). In EOA, speech subscores have a strong association with ataxia scores (ICARS: r=0.735; SARA: r=0.730; p<0.001) and revealed substantial to nearly perfect interobserver agreement (ICARS-ICC: 0.812; SARA-ICC: 0.854; SARASRT-ICC: 0.724). Interpretation: Early-onset ataxia speech subscores are associated with ataxia and also reveal high interobserver agreement, including those internationally applicable to SARASRT. We conclude that SARASRT appears to be applicable for EOA databases. However, before syllable repetition tasks are included, we would advise to wait for the results published by the international Childhood Ataxia and Cerebellar Group. What this paper adds: Healthy speech subscores are moderately associated with age. Early onset ataxia (EOA) speech subscores are clearly associated with ataxia. Early-onset ataxia speech subscores reveal high interobserver agreement. Scale for Assessment and Rating of Ataxia syllable repetition tasks appear applicable for EOA databases.</p>}},
author = {{Kuiper, Marieke J. and Brandsma, Rick and Lawerman, Tjitske F. and Lunsing, Roelineke J. and Keegstra, Anne L. and Burger, Huibert and De Koning, Tom J. and Tijssen, Marina A.J. and Sival, Deborah A.}},
issn = {{0012-1622}},
language = {{eng}},
month = {{12}},
number = {{12}},
pages = {{1202--1206}},
publisher = {{John Wiley & Sons Inc.}},
series = {{Developmental Medicine and Child Neurology}},
title = {{Assessment of speech in early-onset ataxia : A pilot study}},
url = {{http://dx.doi.org/10.1111/dmcn.12517}},
doi = {{10.1111/dmcn.12517}},
volume = {{56}},
year = {{2014}},
}