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Assessment of speech in early-onset ataxia : A pilot study

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. (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.

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publishing date
type
Contribution to journal
publication status
published
subject
in
Developmental Medicine and Child Neurology
volume
56
issue
12
pages
5 pages
publisher
John Wiley and Sons Inc.
external identifiers
  • pmid:24942085
  • scopus:84911378489
ISSN
0012-1622
DOI
10.1111/dmcn.12517
language
English
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no
id
8debe29f-cbc0-415e-bc8b-0b2b5f6c9605
date added to LUP
2020-02-26 10:07:44
date last changed
2020-04-07 05:44:03
@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&lt;0.05) and with ataxia scores (ICARS: r=0.552; SARA: r=0.336; p&lt;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&lt;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 and 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},
}