Childhood Apraxia of Speech: Experience Dependent Changes Induced by Treatment

Study Overview
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Brief Summary
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Childhood Apraxia of Speech (CAS) is a severe speech-language disorder whose aetiological, neuroanatomical correlates are largely unknown. Furthermore, little is known about the neuroplastic effects induced by different treatment approaches and their relationships with the potential changes in the speech behavioural features that express the core deficit of CAS. Twenty four children with idiopathic CAS will be enrolled in a multidisciplinary study aimed at analysing the behavioural and neuroanatomical effects of a specific rehabilitative approach, PROMPT (PROMPTs for Restructuring Oral Muscular Phonetic Targets), that employs tactile-kinesthetic-proprioceptive cues vs a traditional speech-language treatment. The children will be allocated in two arms, one receiving a seven month cycle of individual PROMPT treatment, the other a traditional speech and language treatment for the same amount of time.The pre- and post-treatment speech and language performances and DTI and volumetric MR data will be compared in the two groups.

Detailed Description
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Background: Childhood Apraxia of Speech (CAS) is a severe speech-language disorder whose etiological, neuroanatomical and genetic correlates are largely unknown. In this trial the effects of an innovative rehabilitative approach, PROMPT (PROMPTS for Restructuring Oral Muscular Phonetic Targets) will be evaluated from a behavioral and neuroanatomical point of view. Objective: the current study is aimed to assess speech-language and white matter microstructure and volumetric changes induced by the PROMPT treatment. Participants: Twenty-two Children with CAS aged 4 -12 years are enrolled and assigned to a PROMPT treatment (PROMPT-t ) or a Language and Non-Speech Oral Motor-treatment group (LNSOM-t). Methods: All children with CAS will undergo a comprehensive clinical, neurological and speech-language assessment. The following procedures will be applied: a) anamnestic interview b) oral non verbal and verbal movements eveluation c) phonetic inventory, accuracy and consistency of speech d) DDK (Diadochokinetic rate, receptive and expressive vocabulary and grammar tests; f) spontaneous language analysis. These multiple measurements were aggregated and converted into a speech and language composite score. MRI data will be acquired using a 1.5 T MR scanner. Structural images obtained with a 3D isotropic T1 weighted sequence will be analysed to evaluate volumetric alterations, to investigate cortical thickness and automatically delineate various Regions of Interest (ROIs). HARDI images will be acquired using 32 gradient directions and will be processed using tools available in FSL and MRtrix. Tract-based spatial statistics (TBSS) will be used to statistically analyse images of FA and mean diffusivity. Fiber tracts will be calculated using constrained spherical deconvolution and probabilistic tractography. Connectomes will be calculated by combining ROIs obtained from the structural images with tractography. Network-based analysis will be used to statistically analyse connectomes. Speech and language assessment and brain MRI VBM and DTI analysis will be performed at baseline and at the end of the treatment. Statistical analysis: Paired t-test on pre and post treatment behavioural and MRI/DTI measures will be performed to assess the changes within each of the two groups. Moreover, comparisons between the two groups at T0 and T1 will be performed by means of non parametric tests. Evaluation of potentially training-induced changes of brain structural connectivity may provide further support to the hypothesis that CAS is due to a disruption of networks subserving the speech production system.

Official Title
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Childhood Apraxia of Speech: Neurobiological and Behavioural Markers and Experience Dependent Changes of Neural Connectivity Induced by Treatment

Conditions
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Childhood Apraxia of Speech

Intervention / Treatment
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* Behavioral: PROMPT- treatment
* Behavioral: LNSOM-treatment


Participation Criteria
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Eligibility Criteria
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Inclusion Criteria: Diagnosis of idiopathic CAS based on the presence of specific diagnostic features of apraxia of speech (see ASHA and Strand et al's criteria) and on a comprehensive clinical and neurological assessment. Non-verbal IQ within the normal range at standardized tests of intelligence. Normal structural brain MRI. Acquisition of parents' informed consent to the execution of behavoiural and neuroradiological assessment at baseline and to perform speech/language treatment. Exclusion Criteria: Orofacial structural abnormalities. Known pathologies of neurological, neurometabolical and genetic etiologies. Audiological deficits. Epilepsy. Intellectual disability. Autism spectrum disorder.

Ages Eligible for Study
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Minimum Age: 4 Years
Maximum Age: 16 Years

Sexes Eligible for Study
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All

Study Plan
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How is the study designed?
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Arms and Interventions

| Participant Group/Arm | Intervention/Treatment |
| --- | --- |
| Group 1<br>The PROMPT-treated group will include 12 children with idiopathic CAS. The pre-treatment assessment is aimed at evaluating the baseline speech and language level and at treatment planning. During a PROMPT session tactile-kinesthetic-proprioceptive inputs are consistently provided in order to shape speech movements, to give information on sequencing and timing, and to introduce constraints for the reduction of the degrees of freedom at the articulators' level in favour of motor control. In a PROMPT session the syllables, words and phrases are produced within a communicative context in play. Speech motor goals are, as soon as possible, integrated in goals for language and functional communication. | Behavioral: PROMPT- treatment<br>* PROMPTs for Restructuring Oral Muscolar Phonetic Targets<br>|
| GROUP 2<br>The LNSOM-treated group will include 12 children with idiopathic CAS. The pre-treatment assessment is aimed at evaluating the baseline speech and language level and at the treatment planning. None of the SLTs treating this group are PROMPT trained. According to the standard care methods used in Italy, the intervention consists of a linguistic and articulatory approach that includes auditory discrimination of phonemic categories at the syllable and word level and non-speech oral motor exercises. Receptive and expressive lexicon and morphosyntax are targeted depending on the children's linguistic profile.Differently from the PROMPT, selection of speech sounds to be targeted, is based on developmental speech sounds acquisition rather than motor criteria. According to this treatment approach, the motor goal is usually identified with the placement of the main articulator involved in the production of a speech sound. | Behavioral: LNSOM-treatment<br>* Language Non-Speech Oral Motor Treatment<br>|

What is the study measuring?
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Primary Outcome Measures

| Outcome Measure | Measure Description | Time Frame |
| --- | --- | --- |
| Speech Composite Severity Score changes after 7 months of treatment | Speech Severity Score aggregates measures of DDK, inaccuracy, inconsistency, Phonetic inventory, Syllable Omissions. Score ranges form: 0 (normal) to 5 (severely impaired) | At baselin and at 7 months of individual speech and language training | 

Secondary Outcome Measures

| Outcome Measure | Measure Description | Time Frame |
| --- | --- | --- |
| Post treatment Diffusion Weighed Imaging (DWI) metrics | Fractional Anisotropy (FA) | At baseline and at 7 months of speech and language training | 
| Post treatment Diffusion Weighed Imaging (DWI) changes | MD (Mean Diffusivity) | At baseline and at 7 months of speech and language training | 
| Post treatment cortical volumes modifications | Cortical thickness | At baseline and at 7 months of speech and language training | 
| Post treatment cortical volumes changes | Volumes analysis | At baseline and at 7 months of speech and language training | 

 Terms related to the study
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Keywords Provided by Centre Hospitalier Valida
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Treatment induced speech changes, PROMPT, MRI, Neuroplasticity