The Non-speech Oral Movement Assessment Children (NOMAC).

INTRODUCTION: Examination of oral movements is often part of an assessment undertaken by a speech and language therapist (SLT). Until now there have been no specific instruments or tests with reference values for typically developing children in Dutch that exclusively evaluate non-speech oral movements in young children. Therefore, a non-speech oral-motor observation list was designed to attempt to bridge this gap: The Non-speech Oral Movement Assessment Children (NOMAC). The aim of this study was to evaluate the psychometric properties of the NOMAC in terms of inter-rater reliability and its' construct validity. In addition, we aimed to collect reference values for the non-speech oral movements in children. METHODS: Data from typically developing Dutch children aged 2 to 8 years were collected. Inter-rater reliability was studied by estimating the intra-class correlation coefficient (ICC). Construct validity was investigated by assessing the effect of age group and gender on the mean execution score per item (general linear model). To present normative data the percentage of the children performing a normal oral - motor execution was calculated. RESULTS: The study includes a total of 318 children, divided into 9 age groups. The inter-rater reliability shows a [sufficient] to [good] ICC for most items. A significant effect of the factor age group for almost all items was seen, confirming a robust construct validity. Normative data are presented with the percentage of the children performing a normal oral movement execution. CONCLUSION: Non-speech oral movements can be assessed with the NOMAC in children between 2 to 8 years old and can be compared with values obtained from a normative group. It should be used as part of a clinical feeding and speech assessment. Despite the fact that current insights indicate that oral- motor training has no value for improving mastication, swallowing and speech, it is important to know the status of non-speech oral motor capabilities. With this assessment a complete profile of the child's oral-motor abilities can be achieved, supportive for clinical decision making in SLT.

Dysarthria in children and adults with ataxia telangiectasia.

AIM: To investigate the characteristics and severity of dysarthria in children and adults with ataxia telangiectasia. METHOD: All children and adults with ataxia telangiectasia who visited our multidisciplinary outpatient clinic for ataxia telangiectasia were asked to participate in this study, which took place in March 2019. To evaluate dysarthria, we used the Radboud Dysarthria Assessment in adults (older than 18y) and the paediatric Radboud Dysarthria Assessment in children (5-18y), including the observational tasks 'conversation' and 'reading', and the speech-related maximum performance tasks 'repetition rate', 'phonation time', 'fundamental frequency range', and 'phonation volume'. Speech intelligibility was measured using the Intelligibility in Context Scale. RESULTS: Twenty-two individuals (15 children [5-17y], seven adults [19-47y]; 14 males and eight females; mean age 19y, SD 15y 2mo) participated. Dysarthria was present in all participants and characterized by ataxic components in adults and similar uncontrolled movements in children. In most participants, speech was mildly to mildly/severely affected. Almost all participants had an abnormal score for at least one maximum performance task. INTERPRETATION: Dysarthria in ataxia telangiectasia is characterized by uncontrolled, ataxic, and involuntary movements, resulting in monotonous, unstable, slow, hypernasal, and chanted speech. WHAT THIS PAPER ADDS: Dysarthria in ataxia telangiectasia is characterized by uncontrolled, ataxic, and involuntary movements. Dysarthria in ataxia telangiectasia results in monotonous, unstable, slow, hypernasal, and chanted speech. Dysarthria in ataxia telangiectasia can be assessed using the Radboud Dysarthria Assessment and the paediatric Radboud Dysarthria Assessment.

Speech sound development in typically developing 2-7-year-old Dutch-speaking children: A normative cross-sectional study.

BACKGROUND: Dutch is a West-Germanic language spoken natively by around 24 million speakers. Although studies on typical Dutch speech sound development have been conducted, norms for phonetic and phonological characteristics of typical development in a large sample with a sufficient age range are lacking. AIM: To give a detailed description of the speech sound development of typically developing Dutch-speaking children from 2 to 7 years. METHODS & PROCEDURES: A total of 1503 typically developing children evenly distributed across the age range of 2;0-6;11 years participated in this normative cross-sectional study. The picture-naming task of the Computer Articulation Instrument (CAI) was used to collect speech samples. Speech development was described in terms of (1) percentage consonants correct-revised (PCC-R) and percentage vowels correct (PVC); (2) consonant, vowel and syllabic structure inventories; (3) degrees of complexity (phonemic feature hierarchy); and (4) phonological processes. OUTCOMES & RESULTS: A two-way mixed analysis of variance (ANOVA) confirmed a significant increase in the number of PCC-R and PVC between the ages of 2;0 and 6;11 years (p < 0.001). The consonant inventory was found to be complete at 3;7 years of age for the syllable-initial consonants, with the exception of the voiced fricatives /v/ and /z/, and the liquid /r/. All syllable-final consonants were acquired before age 4;4 years. At age 3;4 years, all children had acquired a complete vowel inventory, and at age 4;7 years they produced most syllable structures correctly, albeit that the syllable structure CCVCC was still developing. All phonological contrasts were produced correctly at 3;8 years of age. Children in the younger age groups used more phonological simplification processes than the older children, and by age 4;4 years, all had disappeared, except for the initial cluster reduction from three to two consonants and the final cluster reduction from two to one consonant. CONCLUSIONS & IMPLICATIONS: This paper describes a large normative cross-sectional study of Dutch speech sound development which, in clinical practice, can help Dutch speech-language pathologists to differentiate children with delayed or disordered speech development from typically developing children. What this paper adds What is already known on this subject In recent years many studies have been conducted worldwide to investigate speech sound development in different languages, including several that explored the typical speech sound development of Dutch-speaking children, but none of these latter studies explored both phonetic and phonological progress within a comprehensive age range and a large sample that is representative of the Dutch population. What this study adds to existing knowledge This study serves to fill this gap by providing normative cross-sectional results obtained in 1503 typically developing Dutch-speaking children aged between 2;0 and 6;11 years on informative parameters of speech development: PCC-R and PVC, consonant, vowel and syllabic structure inventories, degrees of complexity (phonemic feature hierarchy), and phonological simplification processes. What are the potential or actual clinical implications of this work? The detailed description of typical Dutch speech sound development provides speech-language pathologists with pertinent information to determine whether a child's speech development progresses typically or is delayed or disordered.

A Standardized Protocol for Maximum Repetition Rate Assessment in Children.

BACKGROUND/AIMS: Maximum repetition rate (MRR) is often used in the assessment of speech motor performance in older children and adults. The present study aimed to evaluate a standardized protocol for MRR assessment in young children in Dutch. METHODS: The sample included 1,524 children of 2-7 years old with no hearing difficulties and Dutch spoken in their nursery or primary school and was representative for children in the Netherlands. The MRR protocol featured mono-, tri-, and bisyllabic sequences and was computer-implemented to maximize standardization. RESULTS: Less than 50% of the 2-year-olds could produce >1 monosyllabic sequence correctly. Children who could not correctly produce ≥2 monosyllabic sequences could not produce any of the multisyllabic sequences. The effect of instruction ("faster" and "as fast as possible") was small, and multiple attempts yielded a faster MRR in only 20% of the cases. MRRs did not show clinically relevant differences when calculated over different numbers of repeated syllables. CONCLUSIONS: The MRR protocol is suitable for children of 3 years and older. If children cannot produce at least 2 of the monosyllabic sequences, the multisyllabic tasks should be omitted. Furthermore, all fast attempts of each sequence should be analyzed to determine the fastest MRR.

Feeding Problems in Patients with Noonan Syndrome: A Narrative Review.

Noonan syndrome (NS) belongs to the group of Noonan syndrome spectrum disorders (NSSD), which is a group of phenotypically related conditions. Feeding problems are often present not only in infancy but also in childhood, and even beyond that period. We describe the different aspects of feeding problems using a (theoretical) concept proposed in 2019. More than 50% of infants with NS develop feeding problems, and up to half of these infants will be tube-dependent for some time. Although, in general, there is a major improvement between the age of 1 and 2 years, with only a minority still having feeding problems after the age of 2 years, as long as the feeding problems continue, the impact on the quality of life of both NS infants and their caregivers may be significant. Feeding problems in general improve faster in children with a pathogenic PTPN11 or SOS1 variant. The mechanism of the feeding problems is complex, and may be due to medical causes (gastroesophageal reflux disease and delayed gastric emptying, cardiac disease and infections), feeding-skill dysfunction, nutritional dysfunction with increased energy demand, or primary or secondary psychosocial dysfunction. Many of the underlying mechanisms are still unknown. The treatment of the feeding problems may be a medical challenge, especially when the feeding problems are accompanied by feeding-skill dysfunction and psychosocial dysfunction. This warrants a multidisciplinary intervention including psychology, nutrition, medicine, speech language pathology and occupational therapy.

Process-Oriented Profiling of Speech Sound Disorders.

The differentiation between subtypes of speech sound disorder (SSD) and the involvement of possible underlying deficits is part of ongoing research and debate. The present study adopted a data-driven approach and aimed to identify and describe deficits and subgroups within a sample of 150 four to seven-year-old Dutch children with SSD. Data collection comprised a broad test battery including the Computer Articulation Instrument (CAI). Its tasks Picture Naming (PN), NonWord Imitation (NWI), Word and NonWord Repetition (WR; NWR) and Maximum Repetition Rate (MRR) each render a variety of parameters (e.g., percentage of consonants correct) that together provide a profile of strengths and weaknesses of different processes involved in speech production. Principal Component Analysis on the CAI parameters revealed three speech domains: (1) all PN parameters plus three parameters of NWI; (2) the remaining parameters of NWI plus WR and NWR; (3) MRR. A subsequent cluster analysis revealed three subgroups, which differed significantly on intelligibility, receptive vocabulary, and auditory discrimination but not on age, gender and SLPs diagnosis. The clusters could be typified as three specific profiles: (1) phonological deficit; (2) phonological deficit with motoric deficit; (3) severe phonological and motoric deficit. These results indicate that there are different profiles of SSD, which cover a spectrum of degrees of involvement of different underlying problems.

Maximum repetition rate in a large cross-sectional sample of typically developing Dutch-speaking children.

Purpose: The current study aims to provide normative data for the maximum repetition rate (MRR) development of Dutch-speaking children based on a large cross-sectional study using a standardised protocol.Method: A group of 1014 typically developing children aged 3;0 to 6;11 years performed the MRR task of the Computer Articulation Instrument (CAI). The number of syllables per second was calculated for mono-, bi-, and trisyllabic sequences (MRR-pa, MRR-ta, MRR-ka, MRR-pata, MRR-taka, MRR-pataka). A two-way mixed ANOVA was conducted to compare the effects of age and gender on MRR scores in different MRR sequences.Result: The data analysis showed that overall MRR scores were affected by age group, gender and MRR sequence. For all MRR sequences the MRR increased significantly with age. MRR-pa was the fastest sequence, followed by respectively MRR-ta, MRR-pata, MRR-taka, MRR-ka and MRR-pataka. Overall MRR scores were higher for boys than for girls, for all MRR sequences.Conclusion: This study presents normative data of MRR of Dutch-speaking children aged 3;0 to 6;11 years. These norms might be useful in clinical practice to differentiate children with speech sound disorders from typically developing children. More research on this topic is necessary. It is also suggested to collect normative data for other individual languages, using the same protocol.

Speech-language profiles in the context of cognitive and adaptive functioning in SATB2-associated syndrome.

SATB2-associated syndrome (SAS) is a neurodevelopmental disorder caused by heterozygous pathogenic variants in the SATB2 gene, and is typically characterized by intellectual disability and severely impaired communication skills. The goal of this study was to contribute to the understanding of speech and language impairments in SAS, in the context of general developmental skills and cognitive and adaptive functioning. We performed detailed oral motor, speech and language profiling in combination with neuropsychological assessments in 23 individuals with a molecularly confirmed SAS diagnosis: 11 primarily verbal individuals and 12 primarily nonverbal individuals, independent of their ages. All individuals had severe receptive language delays. For all verbal individuals, we were able to define underlying speech conditions. While childhood apraxia of speech was most prevalent, oral motor problems appeared frequent as well and were more present in the nonverbal group than in the verbal group. For seven individuals, age-appropriate Wechsler indices could be derived, showing that the level of intellectual functioning of these individuals varied from moderate-mild ID to mild ID-borderline intellectual functioning. Assessments of adaptive functioning with the Vineland Screener showed relatively high scores on the domain "daily functioning" and relatively low scores on the domain "communication" in most individuals. Altogether, this study provides a detailed delineation of oral motor, speech and language skills and neuropsychological functioning in individuals with SAS, and can provide families and caregivers with information to guide diagnosis, management and treatment approaches.

Clinical Reasoning for Speech Sound Disorders: Diagnosis and Intervention in Speech-Language Pathologists' Daily Practice.

Purpose This study aims to give an insight in clinical reasoning (diagnosis and intervention) of speech-language pathologists (SLPs) in the Netherlands for children with speech sound disorder (SSD). Method The study featured a mixed-method (qualitative and quantitative) design. Semistructured interviews containing nondirective, open-ended questions were conducted with 33 SLPs, which were analyzed using a constant comparative analysis. Other SLPs (137) filled out a questionnaire on the same topics. Multiple-choice questions were analyzed by descriptive frequencies, while open-ended questions were analyzed thematically. Results The results indicate that SLPs use a variety of assessments to diagnose SSD, complemented by observation and, often, case history. In total, 85 different diagnostic labels were reported. The choice of intervention is based on what is appealing to the child and what matches his or her age as well as on the specific diagnosis and severity. Interventions are used for multiple speech disorders, and according to SLPs, parents play a large role in diagnostics and intervention. Conclusion These results reveal the need for (a) a clear and consistent terminology of diagnoses in the field of pediatric SSD, (b) a fast and easy-to-administer comprehensive differential diagnostic instrument in combination with an instrument to assess participation in everyday life, and (c) a tool to conduct a case history online.

Profiling Speech Sound Disorders for Clinical Validation of the Computer Articulation Instrument.

Purpose The current article presents data from 2 studies on clinical groups of children referred for speech assessment. The aims of these studies are to validate the Computer Articulation Instrument (CAI) with the known-group validation method and to determine the differential diagnostic power of the resulting speech profiles. Method Study 1 examined known-group validity by comparing the scores of 93 children diagnosed with speech-language difficulties on the picture naming (PN) task of the CAI with intelligibility judgments given by speech-language pathologists. In Study 2, the speech profiles of 41 children diagnosed with speech sound disorders (SSDs), consisting of 4-6 factor scores extracted from the 4 tasks of the CAI, namely, PN, nonword imitation (NWI), word and nonword repetition, and maximum repetition rate (MRR), were validated against clinical judgments of severity of the SSD given by speech-language pathologists. Results In Study 1, a repeated-measures analysis of variance revealed a significant effect of intelligibility level on the PN performance of the CAI and there were highly significant correlations between intelligibility and PN performance in the expected direction. Neither intelligibility level nor PN performance was related to nonverbal intelligence and language scores. The analysis of variance and a series of t tests in Study 2 revealed significant differences between the moderate and severe groups for the CAI factors based on PN and NWI and the bisyllabic and trisyllabic sequences of MRR, but not for the factor word and nonword proportion of whole-word variability based on word and nonword repetition, and the monosyllabic sequences of MRR. These results suggest that, especially, the tasks PN, NWI, and the bisyllabic and trisyllabic sequences of MRR are most sensitive for diagnosing SSDs. Conclusions The findings of these 2 studies support the known-group validity of the CAI. Together with the results of a previous study of our group on reliability and validity ( van Haaften et al., 2019 ), we can conclude that the CAI is a reliable and valid tool for assessment of children with SSDs.

The Psychometric Evaluation of a Speech Production Test Battery for Children: The Reliability and Validity of the Computer Articulation Instrument.

Purpose The aims of this study were to assess the reliability and validity of the Computer Articulation Instrument (CAI), a speech production test battery assessing phonological and speech motor skills in 4 tasks: (1) picture naming, (2) nonword imitation, (3) word and nonword repetition, and (4) maximum repetition rate (MRR). Method Normative data were collected in 1,524 typically developing Dutch-speaking children (aged between 2;0 and 7;0 [years;months]). Parameters were extracted on segmental and syllabic accuracy (Tasks 1 and 2), consistency (Task 3), and syllables per second (Task 4). Interrater reliability and test-retest reliability were analyzed using subgroups of the normative sample and studied by estimating intraclass correlation coefficients (ICCs). Construct validity was investigated by determining age-related changes of test results and factor analyses of the extracted speech measures. Results ICCs for interrater reliability ranged from sufficient to good, except for percentage of vowels correct of picture naming and nonword imitation and for the MRRs for bisyllabic and trisyllabic items. The ICCs for test-retest reliability were sufficient (picture naming, nonword imitation) to insufficient (word and nonword repetition, MRR) due to larger-than-expected normal development and learning effects. Continuous norms showed developmental patterns for all CAI parameters. The factor analyses revealed 5 meaningful factors: all picture-naming parameters, the segmental parameters of nonword imitation, the syllabic structure parameters of nonword imitation, (non)word repetition consistency, and all MRR parameters. Conclusion Its overall sufficient to good psychometric properties indicate that the CAI is a reliable and valid instrument for the assessment of typical and delayed speech development in Dutch children in the ages of 2-7 years.

Development of oral motor behavior related to the skill assisted spoon feeding.

Milestones in the typical development of eating skills are considered to be nippling (breast or bottle), eating from a spoon, drinking from a cup, biting and chewing. The purpose of this research was to study the development and consolidation of oral motor behavior related to the skill assisted spoon feeding in young infants. The present study longitudinally investigated the development of this skill in 39 healthy children from the start of spoon feeding until the skill was acquired. The Observation List Spoon Feeding with 7 observation items for oral motor behavior and 6 items for abnormal behavior was used. Results showed that infants between 4 and 8 months of age needed 5.7 weeks (SD 2.1), with a range of 8 weeks (from 2 to 10 weeks) to acquire this skill. No significant correlation (p=.109) between age at start spoon feeding and weeks needed to develop the skill was found. During this period oral motor behavior consolidated and abnormal behavior diminished. With this study it is shown that the period in weeks needed to acquire the oral motor behavior for the skill assisted spoon feeding is important in case of feeding problems.