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Purpose The aim of this longitudinal study was to describe developmental courses of childhood dysarthria against the background of typical speech motor development by collecting auditory-perceptual data. Method Fourteen children (four girls, 10 boys; 5;1–8;4 [years;months] at Time 1) with neurological conditions (CNC) and 14 typically developing children (CTD) matched for age and gender were assessed at three points in time over an 18-month period. Speech samples were collected using the Bogenhausener Dysarthrie Skalen–Kindliche Dysarthrien (BoDyS-KiD; in English: Bogenhausen Dysarthria Scales–Childhood Dysarthria), a German tool for the assessment of childhood dysarthria, and analyzed by means of nine perceptual scales covering all clinically relevant speech components. Age normalization was performed according to a method published recently. Data from the matched controls were used to estimate whether the gradients of the CNC group's developmental trajectories exceeded those of typical development. Results The children with neurological conditions presented heterogeneous speech profiles with a wide range of severity. At the group level, relatively stable trajectories of the age-normalized dysarthria total score were found over the observation period. The nine perceptual scales showed more or less parallel developments. All patients except two followed the growth curve describing the developmental course of the typically developing children. Conclusions Most children took advantage of the developmental dynamics as they developed parallel to the age norm. With its comprehensive description of the developmental courses of 14 children with neurological conditions, this study may contribute to a more valid, statistically verified clinical assessment of the course of childhood dysarthria.
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JSLHR
Clinical Focus
Developmental Courses in Childhood
Dysarthria: Longitudinal Analyses of
Auditory-Perceptual Parameters
Elisabet Haas,
a
Wolfram Ziegler,
a
and Theresa Schölderle
a
Purpose: The aim of this longitudinal study was to describe
developmental courses of childhood dysarthria against
the background of typical speech motor development by
collecting auditory-perceptual data.
Method: Fourteen children (four girls, 10 boys; 5;18;4 [years;
months] at Time 1) with neurological conditions (CNC) and
14 typically developing children (CTD) matched for age
and gender were assessed at three points in time over an
18-month period. Speech samples were collected using
the Bogenhausener Dysarthrie SkalenKindliche Dysarthrien
(BoDyS-KiD; in English: Bogenhausen Dysarthria Scales
Childhood Dysarthria), a German tool for the assessment
of childhood dysarthria, and analyzed by means of nine
perceptual scales covering all clinically relevant speech
components. Age normalization was performed according
to a method published recently. Data from the matched
controls were used to estimate whether the gradients of the
CNC groups developmental trajectories exceeded those
of typical development.
Results: The children with neurological conditions presented
heterogeneous speech profiles with a wide range of severity.
At the group level, relatively stable trajectories of the age-
normalized dysarthria total score were found over the
observation period. The nine perceptual scales showed
more or less parallel developments. All patients except two
followed the growth curve describing the developmental
course of the typically developing children.
Conclusions: Most children took advantage of the
developmental dynamics as they developed parallel to
the age norm. With its comprehensive description of the
developmental courses of 14 children with neurological
conditions, this study may contribute to a more valid,
statistically verified clinical assessment of the course of
childhood dysarthria.
Many children with neurological conditions show
symptoms of dysarthria. The most relevant
cause of childhood dysarthria is cerebral palsy
(CP), with up to 90% of the affected children experiencing
dysarthria (Bax et al., 2006; Mei et al., 2014; Nordberg
et al., 2013). Furthermore, other neurological conditions
can also cause dysarthria, such as cerebellar or brainstem
tumors (Murdoch & Hudson-Tennent, 1994; Richter et al.,
2005; van Mourik et al., 1998) and traumatic brain injury
(Cahill et al., 2002, 2003, 2005). Dysarthria usually manifests
itself on all functionally relevant components of speech,
that is, speech breathing, voice production, articulation/
resonance, and prosody. As a result, perceptual deviations
of speech features can occur to varying degrees in any one
or several of these components (Duffy, 2013; Workinger &
Kent, 1991).
Dysarthric impairment in most cases interferes with
the childrens ability to communicate, compromising, for
instance, intelligibility and naturalness of speech (Hustad
et al., 2018, 2012; Patel et al., 2012; Pennington & Mc-
Conachie, 2001). As a consequence, research indicates that
the childrens social and educational participation is often
restricted, and their quality of life can be negatively af-
fected (Dickinson et al., 2007; Fauconnier et al., 2009;
Mei et al., 2014; Pennington & McConachie, 2001). This
is especially burdening as children with dysarthriadue
to the early onset of the disorderare confronted with
these limitations throughout their entire lives. An early
diagnosis and detailed characterization of the speech dis-
order of affected children are therefore essential to ensure
adequate treatment at an early stage in order to achieve
maximum treatment outcomes (Romski & Sevcik, 2005;
Sigurdardottir & Vik, 2011).
a
Clinical Neuropsychology Research Group, Institute of Phonetics
and Speech Processing, LMU Munich, Germany
Correspondence to Elisabet Haas: elisabet.haas@ekn-muenchen.de
Editor-in-Chief: Bharath Chandrasekaran
Editor: Stephanie Borrie
Received August 19, 2020
Revision received October 30, 2020
Accepted December 15, 2020
https://doi.org/10.1044/2020_JSLHR-20-00492
Disclosure: The authors have declared that no competing interests existed at the time
of publication.
Journal of Speech, Language, and Hearing Research Vol. 64 14211435 May 2021 Copyright © 2021 American Speech-Language-Hearing Association 1421
... This study was approved by the Medical Faculty's Ethics Committee of the Ludwig-Maximilians-Universität München (LMU Munich). As there is some overlap with previous publications, details on participants, assessment materials, and auditory-perceptual analyses beyond the information provided herein can be obtained from Schölderle et al. (2020Schölderle et al. ( , 2021Schölderle et al. ( , 2022Schölderle et al. ( , 2023 as well as Haas et al. (2021Haas et al. ( , 2022. The present article refers to age and gender estimates in CTD and in CWD collected in a comprehensive listening experiment with adult listeners. ...
... Several children from both the CTD (14 of 144) and CWD groups (11 of 25) participated in a longitudinal study, with three or four assessments in 9-month intervals (see Haas et al., 2021). This results in a total of 175 sets of recordings of CTD and 47 of CWD. ...
... for Childhood Dysarthria (BoDyS-KiD; seeHaas et al., 2020Haas et al., , 2021Schölderle et al., 2020). The BoDyS-KiD assessment tool builds upon the BoDyS proposed byZiegler et al. in 2018 as an approach established ...
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Purpose The purposes of this study were (a) to investigate adult listeners' perceptions of age and gender in typically developing children and children with dysarthria and (b) to identify predictors of their estimates among auditory-perceptual parameters and an acoustic measure of vocal pitch (F0). We aimed to evaluate the influence of dysarthria on the listeners' impressions of age and gender against the background of typical developmental processes. Method In a listening experiment, adult listeners completed age and gender estimates of 144 typically developing children (3–9 years of age) and 25 children with dysarthria (5–9 years of age). The Bogenhausen Dysarthria Scales for Childhood Dysarthria (BoDyS-KiD) were applied to record speech samples and to complete auditory-perceptual judgments covering all speech subsystems. Furthermore, each child's mean F0 was determined from samples of four BoDyS-KiD sentences. Results Age estimates for the typically developing children showed a regression to the mean, whereas children with dysarthria were systematically underestimated in their age. The estimates of all children were predicted by developmental speech features; for the children with dysarthria, specific dysarthria symptoms had an additional effect. We found a significantly higher accuracy of gender attribution in the typically developing children than in the children with dysarthria. The prediction accuracy of the listeners' gender attribution in the preadolescent children by the included speech characteristics was limited. Conclusions Children with dysarthria are more difficult to estimate for their age and gender than their typically developing peers. Dysarthria thus alters the auditory-perceptual impression of indexical speech features in children, which must be considered another facet of the communication disorder associated with childhood dysarthria.
... Dysarthria research is traditionally focused on adults; the topic of childhood dysarthria is relatively new. Knowledge about the clinical picture and prognosis of childhood dysarthria is still emerging, with the majority of articles on which current knowledge is based being published over the last 10 years (e.g., Allison & Hustad, 2018;Boliek & Fox, 2017;Haas et al., 2021;Hustad et al., 2019;Kuschmann & Lowit, 2021;Levy et al., 2021;Mei et al., 2014;Nordberg et al., 2013;Pennington, Roelant, et al., 2013;Schölderle et al., 2022). In addition, as some of the most common textbooks on dysarthria do not yet consider children as a client group (English: Duffy, 2020;German: Ziegler & Vogel, 2010), it can be challenging for SLPs to acquire knowledge specific to childhood dysarthria. ...
... However, results showed that the presence or absence of professional training in childhood dysarthria did not affect how many assessment instruments respondents used for the purposes of dysarthria assessment. In this context, it is important to note that many of the instruments (e.g., BoDyS-KiD [see Haas et al., 2021], the Intelligibility in Context Scale-German version [see Neumann et al., 2017]) have only recently become available or been translated into German, respectively. They may therefore not have been covered as part of their professional education at the time they were in training. ...
Article
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Purpose This survey study aimed to establish current clinical practices of German-speaking speech-language pathologists (SLPs) regarding their assessment and treatment of communication disorders in children with neurological conditions, with a particular focus on the management of childhood dysarthria. Method A 23-question cross-sectional online survey was disseminated to practicing SLPs in Germany, Austria, and Switzerland via relevant professional bodies. SLPs were invited to provide information on their current assessment and treatment practices. Demographic data including case load and clinical settings were also gathered to contextualize practices. Results One hundred two SLPs responded to the survey, of which 68 valid responses were analyzed. German-speaking SLPs comprehensively assess and treat various aspects of overall communication, language, and swallowing functions in children with neurological conditions. Speech motor aspects did not represent a main intervention focus. In cases where the dysarthric component was targeted, specific approaches for childhood dysarthria were rarely used. Instead, SLPs reported using approaches developed for speech disorders other than dysarthria. Conclusions German-speaking SLPs working with children with neurological conditions use various assessment and treatment methods to support children's communication. However, dysarthria-specific approaches were not an established part of clinical practice. Results of the survey highlight the need for access to relevant developments in German and for evaluation of current curricula for speech-language pathology students and continuing education opportunities for practicing clinicians.
... Notably, the BoDyS-KiD total scores and the intelligibility scores (see section on Listening Experiment for details) show that a substantial proportion of the CNC sample had severe speech deficits and intelligibility restrictions, respectively. On the other hand, the agenormalized BoDyS-KiD total score (see Haas et al., 2021) • • • demonstrated that three children from the CNC group ranged within their age norm and, hence, were not classified as dysarthric (i.e., CNC05, CNC08, and CNC09). They were included as part of the CNC sample in the following analyses. ...
... Standard speech samples were obtained using the protocol of the BoDyS-KiD (see Haas et al., 2021;Schölderle et al., 2020). This tool builds upon the BoDyS, proposed by Ziegler et al. (2018) as an approach established for adults with dysarthria. ...
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Purpose This study investigated perceived speech naturalness estimated by adult listeners in typically developing children and children with dysarthria. We aimed to identify predictors of naturalness among auditory-perceptual parameters and to evaluate the concept of naturalness as a clinical marker of childhood dysarthria. Method In a listening experiment, naive adult listeners rated speech naturalness of 144 typically developing children (3–9 years old) and 28 children with neurological conditions (5–9 years old) on a visual analog scale. Speech samples were recorded using the materials of the Bogenhausen Dysarthria Scales–Childhood Dysarthria, which also provides for auditory-perceptual judgments covering all speech subsystems. Results Children with dysarthria obtained significantly lower naturalness ratings compared to typically developing children. However, there was a substantial age effect observable in the typically developing children; that is, younger typically developing children were also perceived as somewhat unnatural. The ratings of the typically developing children were influenced by the occurrence of developmental speech features; for the children with neurological conditions, specific symptoms of dysarthria had an additional effect. In both groups, the perception of naturalness was predominantly determined by the children's articulation and intelligibility. Conclusions Both symptoms of childhood dysarthria and developmental speech features (e.g., regarding articulation and intelligibility) were associated to some extent with unnatural speech by the listeners. Thus, perceived speech naturalness appears less suitable as a marker of dysarthria in children than in adults.
... CD may result from dysfunction of the respiratory system, larynx, and/or articulatory organs, resulting in difficulties producing clear speech. The characteristics of dysarthria in children may include distorted speech sounds, irregular speech rhythm, changes in voice quality (such as breathiness, hoarseness, or hypernasality), and difficulties with speech volume, pitch, and rate (Haas et al., 2021;Yorkston et al., 2010). The exact speech signs of CD and their severity vary widely among individuals, depending on the underlying cause and the specific muscles affected. ...
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Purpose Motor deficits are widely documented among autistic individuals, and speech characteristics consistent with a motor speech disorder have been reported in prior literature. We conducted an auditory-perceptual analysis of speech production skills in low and minimally verbal autistic individuals as a step toward clarifying the nature of speech production impairments in this population and the potential link between oromotor functioning and language development. Method Fifty-four low or minimally verbal autistic individuals aged 4–18 years were video-recorded performing nonspeech oromotor tasks and producing phonemes, syllables, and words in imitation. Three trained speech-language pathologists provided auditory perceptual ratings of 11 speech features reflecting speech subsystem performance and overall speech production ability. The presence, attributes, and severity of signs of oromotor dysfunction were analyzed, as were relative performance on nonspeech and speech tasks and correlations between perceptual speech features and language skills. Results and Conclusions Our findings provide evidence of a motor speech disorder in this population, characterized by perceptual speech features including reduced intelligibility, decreased consonant and vowel precision, and impairments of speech coordination and consistency. Speech deficits were more associated with articulation than with other speech subsystems. Speech production was more impaired than nonspeech oromotor abilities in a subgroup of the sample. Oromotor deficits were significantly associated with expressive and receptive language skills. Findings are interpreted in the context of known characteristics of the pediatric motor speech disorders childhood apraxia of speech and childhood dysarthria. These results, if replicated in future studies, have significant potential to improve the early detection of language impairments, inform the development of speech and language interventions, and aid in the identification of neurobiological mechanisms influencing communication development.
... The respiratory subsystem provides the driving pressure required to produce speech. Respiratory physiological impairments contribute to auditory-perceptual features of speech motor involvement such as inappropriate phrasing, reduced stress, voice quality changes, and difficulties regulating loudness, all of which are common in children with CP (Haas et al., 2021;Workinger & Kent, 1991). Respiration for speech, or speech breathing, is a carefully regulated process. ...
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Purpose The purpose of this longitudinal study was to examine the effect of time and sentence length on speech rate and its characteristics, articulation rate and pauses, within 2 groups of children with cerebral palsy (CP). Method Thirty-four children with CP, 18 with no speech motor involvement and 16 with speech motor involvement, produced sentences of varying lengths at 3 time points that were 1 year apart (mean age = 56 months at first time point). Dependent measures included speech rate, articulation rate, proportion of time spent pausing, and average number and duration of pauses. Results There were no significant effects of time. For children with no speech motor involvement, speech rate increased with longer sentences due to increased articulation rate. For children with speech motor involvement, speech rate did not change with sentence length due to significant increases in the proportion of time spent pausing and average number of pauses in longer sentences. Conclusions There were no significant age-related differences in speech rate in children with CP regardless of group membership. Sentence length differentially impacted speech rate and its characteristics in both groups of children with CP. This may be due to cognitive–linguistic and/or speech motor control factors.
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en Aim We examined receptive language developmental trajectories between 18 months and 54 months for three clinical speech‐language profile groups of children with cerebral palsy (those with speech motor involvement, without speech motor involvement, and with anarthria) and quantified differences from age‐level expectations. We identified latent classes of comprehension development, related these classes to clinical profile groups, and examined how well early receptive language predicted outcomes. Method We used a prospective longitudinal design. Eighty‐five children with cerebral palsy (43 females, 42 males) were followed longitudinally from 18 to 54 months of age. Children were seen two to eight times (322 data points). Children were classified into clinical profile groups. Language comprehension age‐equivalent scores were the primary measures of interest. Results Children with anarthria had significant language delays, limited developmental change over time, and comprised their own latent class. Children with speech motor impairment had slight receptive language delays over time. Children with no speech motor impairment had age‐appropriate receptive language over time. Early language comprehension scores were highly predictive of later latent profile group membership. Interpretation Early language comprehension abilities are highly predictive of language comprehension growth trajectory and suggest that children with early language delay, particularly those who are non‐speaking, should receive language intervention to support development. What this paper adds • There are two growth trajectories for language comprehension among children with cerebral palsy. • Children with speech motor impairment had a constant 6‐month receptive language delay. • Children without speech motor impairment had age‐appropriate receptive language. • Non‐speaking children had significant receptive language delay. • Early language comprehension change was highly predictive of later trajectory group. Resumen es Crecimiento longitudinal del lenguaje receptivo en niños con parálisis cerebral entre los 18 y 54 meses de edad Objetivo Examinamos las trayectorias del desarrollo del lenguaje receptivo de tres grupos clínicos entre 18 y 54 meses, según el perfil del habla y el lenguaje de niños con parálisis cerebral (aquellos con afectación motora, sin afectación motora del habla y con anartria) y diferencias cuantificadas de las expectativas del nivel de edad. Identificamos tipos de desarrollo de comprensión latentes, relacionando estas clases con grupos de perfiles clínicos y examinamos qué tan bien pronosticaron los resultados del lenguaje receptivo temprano. Metodo Usamos un diseño longitudinal prospectivo. Ochenta y cinco niños con parálisis cerebral (43 mujeres, 42 varones) fueron seguidos longitudinalmente desde los 18 a los 54 meses de edad. Los niños fueron vistos de dos a ocho veces (322 puntos de datos). Los niños fueron clasificados en grupos de perfiles clínicos. Los puntajes de la comprensión del lenguaje equivalentes a la edad fueron las principales medidas de interés. Resultados Los niños con anartria tuvieron retrasos significativos en el lenguaje, con un cambio limitado en el desarrollo a lo largo del tiempo y conformaron su propio tipo latente. Los niños con discapacidad motora del habla tuvieron retrasos leves en el lenguaje receptivo al pasar el tiempo. Los niños sin discapacidad motora del habla tenían un lenguaje receptivo apropiado para su edad a lo largo del tiempo. Las puntuaciones de comprensión del lenguaje tempranas fueron altamente predictivas de la membresía al grupo latente según el perfil posterior. Interpretacion Las habilidades tempranas de comprensión del lenguaje son altamente predictivas de la trayectoria de crecimiento de la comprensión del lenguaje y sugieren que los niños con retraso temprano del lenguaje, particularmente aquellos que no hablan, deben recibir intervención del lenguaje para apoyar el desarrollo. Resumo pt Desenvolvimento longitudinal da linguagem receptiva em crianças com paralisia cerebral entre 18 e 54 meses de idade Objetivo Nós examinamos as trajetórias do desenvolvimento da linguagem receptiva entre 18 e 54 meses para grupos de crianças com paralisia cerebral com três perfis clínicos de linguagem receptiva (aqueles com envolvimento motor da fala, sem envolvimento motor da fala, e com anartria) Identificamos as classes latentes de desenvolvimento da compreensão, relacionadas a estes perfis clínicos, e examinamos quão bem a linguagem receptiva precoce foi preditiva dos resultados. Método Usamos um desenho prospectivo longitudinal. Oitenta e cinco crianças com paralisia cerebral (43 do sexo feminino, 42 do sexo masculino) foram acompanhadas longitudinalmente dos 18 aos 54 meses de idade. As crianças foram vistas de duas a oito vezes (322 pontos de dados) e foram classificadas nos grupos de acordo com o perfil clínico. Os escores equivalentes à idade para compreensão da linguagem foram as medidas primárias de interesse. Resultados Crianças com anartria tinham atraso significativo na linguagem, mudança desenvolvimental limitada com o passar do tempo, e formaram sua própria classe latente. Crianças com envolvimento motor da fala tinha leve atraso na linguagem receptiva com o tempo. Crianças sem comprometimento motor da fala tinham linguagem receptiva adequada para a idade com o passar do tempo. Os escores precoces de compreensão da fala foram altamente preditivos do pertencimento ao grupo de perfil latente posteriormente. Interpretação Habilidades precoces de compreensão da linguagem são altamente preditivas da trajetória de desenvolvimento da comrpeensão da linguagem, e sugerem que crianças com atraso precoce, particularmente as que não falam, devem receber intervenção de linguagem para favorecer seu desenvolvimento.
Article
Kindliche Dysarthrien stellen ein häufiges Störungsbild dar. Trotzdem existieren kaum geeignete Untersuchungsmethoden. Dieser Beitrag erläutert spezifische Herausforderungen in der Diagnostik kindlicher Dysarthrien und stellt ein neues Untersuchungsmaterial vor. Dieses ermöglicht es, kindgerecht und motivierend standardisierte Sprechproben von Kindern mit Dysarthrie zu elizitieren und stellt so die Basis für umfassende auditive Analysen dar. Childhood dysarthria is a common disorder. However, specific assessment tools are scarce. The current paper illustrates challenges related to the assessment of childhood dysarthria and introduces assessment materials that allow for the elicitation of standardized speech samples from children with dysarthria in a child-oriented and motivating way. Thus, it provides the basis for comprehensive auditory-perceptual analyses.