Oromotor Deficits Research Articles

Exploring Motor Speech Disorders in Low and Minimally Verbal Autistic Individuals: An Auditory-Perceptual Analysis.

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. 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. 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.

Feeding Difficulties Among Children With Special Needs: A Cross-Sectional Study From India.

Deficits in feeding can lead to impairments in occupational performance for children with special needs. This correlational study assessed the relationship between oromotor deficits, behavior problems related to feeding, and caregiver perception of the behavior in children with special needs. We included children with neurodevelopmental disorders (n=79), between 2 and 12 years of chronological age, and their caregivers. Those fulfilling the selection criteria were administered the Behavioral Pediatric Feeding Assessment Scale (BPFAS) and Schedule for Oromotor Assessment (SOMA). More than half the sample had skill deficits and behavioral problems related to feeding. There was a statistically significant correlation of oromotor deficits with specific food consistencies and feeding-related behavior problems. Children with special needs have impaired participation in feeding. Deficits at the body system level are associated with parental and cultural factors, which would have to be mitigated to optimize performance.

Investigating Tongue Strength and Endurance in Children Aged 6 to 11 Years

Objective measures of tongue strength and endurance are used to assess lingual weakness and fatigue, and may have significant clinical value for dysphagia management. Recent studies investigating age and gender effects on tongue strength in children are limited by small sample sizes. The current study investigated age and gender effects on tongue strength with a larger sample size, and collected preliminary normative data for a paediatric population. This study also investigated the reliability of tongue endurance measures in children using a modified method, which has not previously been investigated. Using a cross-sectional design, this study examined tongue strength and endurance in 119 children aged 6 to 11years, with no history of speech sound disorders, oro-motor deficits, dysphagia or cognitive impairment. Measures were collected using the Iowa Oral Performance Instrument. Children participated in two sessions, 45min and 10min in duration. Tongue strength was found to significantly increase with age (p < 0.001), while no gender effects were found. Modified tongue endurance measures involved using only one measure of maximal tongue strength to set parameters for tongue endurance scores. Despite this modification, data did not reach acceptable test-retest reliability, ICC = 0.68, p < 0.001; however, reliability improved from previous studies. These findings provide normative data for tongue strength, as a basis to compare individuals, and highlights the need for more reliable protocols for measuring tongue endurance. Normative data was collected from city-dwelling Australian children.

Corticobulbar Tract Injury, Oromotor Impairment and Language Plasticity in Adolescents Born Preterm.

Children born preterm are at risk of impairments in oromotor control, with implications for early feeding and speech development. In this study, we aimed to identify (a) neuroanatomical markers of persistent oromotor deficits using diffusion-weighted imaging (DWI) tractography and (b) evidence of compensatory neuroplasticity using functional MRI (fMRI) during a language production task. In a cross-sectional study of 36 adolescents born very preterm (<33 weeks’ gestation) we identified persistent difficulties in oromotor control in 31% of cases, but no clinical diagnoses of speech-sound disorder (e.g., dysarthria, dyspraxia). We used DWI-tractography to examine the microstructure (fractional anisotropy, FA) of the corticospinal and corticobulbar tracts. Compared to the unimpaired group, the oromotor-impaired group showed (i) reduced FA within the dorsal portion of the left corticobulbar tract (containing fibres associated with movements of the lips, tongue, and larynx) and (ii) greater recruitment of right hemisphere language regions on fMRI. We conclude that, despite the development of apparently normal everyday speech, early injury to the corticobulbar tract leads to persistent subclinical problems with voluntary control of the face, lips, jaw, and tongue. Furthermore, we speculate that early speech problems may be ameliorated by cerebral plasticity – in particular, recruitment of right hemisphere language areas.

Characterization of oromotor and limb motor dysfunction in the DJ1 -/- model of Parkinson disease

Parkinson disease (PD) is devastating to sensorimotor function that includes cranial/oromotor and limb motor deficits. However, the onset, progression, and neural correlates of PD-related dysfunctions are poorly understood. To address this gap, we used a genetic rat model of PD, DJ1 -/-, and hypothesized that motor deficits would manifest early in the disease process, be progressive in nature, and be related to pathologies in brainstem structures associated with sensorimotor function. The present study compares homozygous DJ1 -/- male rats to age-matched wild type controls. Progressive cranial sensorimotor function (ultrasonic vocalizations and tongue motor performance) and limb motor function (tapered balance beam) was analyzed at 2, 4, 6, and 8 months of age. Additionally, tyrosine hydroxylase cell counts were performed in the locus coeruleus and correlated to behavioral measures. We found that compared to wild type controls, DJ1 -/- show deficits in ultrasonic vocalizations as well as oromotor (tongue) deficits that were progressive. Overtime, DJ1 -/- rats cross a tapered balance beam with significantly decreased speed of traversal. Additionally, in the DJ1 -/-, tyrosine hydroxylase positive cells in the locus coeruleus are significantly reduced and are negatively correlated to oromotor behaviors. Characterizing the DJ1 -/- model of PD provides important foundational work necessary to define behavioral and early-onset biomarkers that parallels early-stage PD pathology in humans.

The Implication of the Thalamus in Speech Disturbance in a Boy with Rolandic Epilepsy

Transient oromotor deficits in children with rolandic epilepsy (RE) have been described in literature. It has been reported that thalamic aphasia can be caused by thalamic infarction or hemorrhage; however, it has never been reported that thalamic aphasia can be caused by epileptic disorders. Here, we describe a 6-year-old right-handed boy who showed transient oromotor deficits and speech disturbance with worsening of both epileptic seizure and paroxysmal discharges on electroencephalogram. He had paucity of spontaneous speech, feeble speech, arrest of speech, and anomia, but his repetition skills were intact. A single-photon emission computed tomography (SPECT) scan using technetium-99m hexamethyl propylene amine oxime (99mTc-HMPAO) to measure regional cerebral blood flow showed a reduction in flow in the left thalamus and ipsilateral frontotemporal areas, which appeared concurrently with the speech dysfunction. We suggest that speech disturbance such as thalamic aphasia, which is similar to the symptoms of Landau-Kleffner syndrome, may occur due to epileptic disorders such as RE. It may be possible to miss speech disturbance implicated in the thalamus in cases complicated by oromotor dysfunction. It is important that speech disturbance implicated in the thalamus is not overlooked in patients with RE showing an atypical course.

Childhood apraxia of speech: A survey of praxis and typical speech characteristics

Purpose: The purpose of this study was to investigate current knowledge of the diagnosis childhood apraxia of speech (CAS) in Sweden and compare speech characteristics and symptoms to those of earlier survey findings in mainly English-speakers.Method: In a web-based questionnaire 178 Swedish speech–language pathologists (SLPs) anonymously answered questions about their perception of typical speech characteristics for CAS. They graded own assessment skills and estimated clinical occurrence.Results: The seven top speech characteristics reported as typical for children with CAS were: inconsistent speech production (85%), sequencing difficulties (71%), oro-motor deficits (63%), vowel errors (62%), voicing errors (61%), consonant cluster deletions (54%), and prosodic disturbance (53%). Motor-programming deficits described as lack of automatization of speech movements were perceived by 82%. All listed characteristics were consistent with the American Speech–Language–Hearing Association (ASHA) consensus-based features, Strand’s 10-point checklist, and the diagnostic model proposed by Ozanne. The mode for clinical occurrence was 5%. Number of suspected cases of CAS in the clinical caseload was approximately one new patient/year and SLP.Conclusions: The results support and add to findings from studies of CAS in English-speaking children with similar speech characteristics regarded as typical. Possibly, these findings could contribute to cross-linguistic consensus on CAS characteristics.

Evidence for early and progressive ultrasonic vocalization and oromotor deficits in a PINK1 gene knockout rat model of Parkinson's disease.

Parkinson's disease (PD) is a progressive neurodegenerative disease that leads to a wide range of motor and nonmotor deficits. Specifically, voice and swallow deficits manifest early, are devastating to quality of life, and are difficult to treat with standard medical therapies. The pathological hallmarks of PD include accumulation of the presynaptic protein α-synuclein (αSyn) as well as degeneration of substantia nigra dopaminergic neurons. However, there is no clear understanding of how or when this pathology contributes to voice and swallow dysfunction in PD. The present study evaluates the effect of loss of function of the phosphatase and tensin homolog-induced putative kinase 1 gene in rats (PINK1(-/-) ), a model of autosomal recessive PD in humans, on vocalization, oromotor and limb function, and neurodegenerative pathologies. Behavioral measures include ultrasonic vocalizations, tongue force, biting, and gross motor performance that are assayed at 2, 4, 6, and 8 months of age. Aggregated αSyn and tyrosine hydroxylase immunoreactivity (TH-ir) were measured at 8 months. We show that, compared with wild-type controls, PINK1(-/-) rats develop (1) early and progressive vocalization and oromotor deficits, (2) reduced TH-ir in the locus coeruleus that correlates with vocal loudness and tongue force, and (3) αSyn neuropathology in brain regions important for cranial sensorimotor control. This novel approach of characterizing a PINK1(-/-) genetic model of PD provides the foundational work required to define behavioral biomarkers for the development of disease-modifying therapeutics for PD patients.

Assessing the role of dopamine in limb and cranial-oromotor control in a rat model of Parkinson's disease

Parkinson's disease (PD) is a neurodegenerative disorder primarily characterized by sensorimotor dysfunction. The neuropathology of PD includes a loss of dopamine (DA) neurons of the nigrostriatal pathway. Classic signs of the disease include rigidity, bradykinesia, and postural instability. However, as many as 90% of patients also experience significant deficits in speech, swallowing (including mastication), and respiratory control. Oromotor deficits such as these are underappreciated, frequently emerging during the early, often hemi-Parkinson, stage of the disease. In this paper, we review tests commonly used in our labs to model early and hemi-Parkinson deficits in rodents. We have recently expanded our tests to include sensitive models of oromotor deficits. This paper discusses the most commonly used tests in our lab to model both limb and oromotor deficits, including tests of forelimb-use asymmetry, postural instability, vibrissae-evoked forelimb placing, single limb akinesia, dry pasta handling, sunflower seed shelling, and acoustic analyses of ultrasonic vocalizations and pasta biting strength. In particular, we lay new groundwork for developing methods for measuring abnormalities in the acoustic patterns during eating that indicate decreased biting strength and irregular intervals between bites in the hemi-Parkinson rat. Similar to limb motor deficits, oromotor deficits, at least to some degree, appear to be modulated by nigrostriatal DA. Finally, we briefly review the literature on targeted motor rehabilitation effects in PD models. Learning outcomes: Readers will: (a) understand how a unilateral lesion to the nigrostriatal pathway affects limb use, (b) understand how a unilateral lesion to the nigrostriatal pathway affects oromotor function, and (c) gain an understanding of how limb motor deficits and oromotor deficits appear to involve dopamine and are modulated by training.

Comprehensive motor testing in Fmr1-KO mice exposes temporal defects in oromotor coordination.

Fragile X syndrome (FXS; MIM #300624), a well-recognized form of inherited human mental retardation is caused, in most cases, by a CGG trinucleotide repeat expansion in the 5'-untranslated region of FMR1, resulting in reduced expression of the fragile X mental retardation protein (FMRP). Clinical features include macroorchidism, anxiety, mental retardation, motor coordination, and speech articulation deficits. The Fmr1 knockout (Fmr1-KO) mouse, a mouse model for FXS, has been shown to replicate the macroorchidism, cognitive deficits, and neuroanatomical abnormalities found in human FXS. Here we asked whether Fmr1-KO mice also display appendicular and oromotor deficits comparable to the ataxia and dysarthric speech seen in FXS patients. We employed standard motor tests for balance and appendicular motor coordination, and used a novel long-term fluid-licking assay to investigate oromotor function in Fmr1-KO mice and their wild-type (WT) littermates. Fmr1-KO mice performed equally well as their WT littermates on standard motor tests, with the exception of a raised-beam task. However, Fmr1-KO mice had a significantly slower licking rhythm than their WT littermates. Deficits in rhythmic fluid-licking in Fmr1-KO mice have been linked to cerebellar pathologies. It is believed that balance and motor coordination deficits in FXS patients are caused by cerebellar neurophathologies. The neuronal bases of speech articulation deficits in FXS patients are currently unknown. It is yet to be established whether similar neuronal circuits control rhythmic fluid-licking pattern in mice and speech articulation movement in humans.

A diagnostic challenge: Language difficulties and hearing impairment in a secondary-school student from a non-English-speaking background

Children who have sensory, cognitive or oromotor deficits, or come from a bilingual-speaking background are currently excluded from the diagnosis of specific language impairment (SLI). Emerging evidence, however, suggests that at least 7% of all children have language learning difficulties, irrespective of other diagnoses or language learning background. The aim of this study was to investigate the language abilities of an adolescent boy with pre-lingual, severe-profound hearing loss who comes from a non-English-speaking background. This student’s performance on standardized tests is described and qualitative language analyses examine his impairment. His abilities are compared with that of a matched control. The language difficulty identified could not be attributed to either hearing impairment or non-English-speaking background. The student’s data suggest the need to expand the concept of SLI to include children with other impairments and from different language backgrounds in order to provide equity of support services and access to the classroom curriculum.

Growth disturbance of frontal lobe in BCECTS presenting with frontal dysfunction

A considerable proportion of children with benign childhood epilepsy with centrotemporal spikes (BCECTS) have increasingly been found to show neuropsychiatric deficits such as cognitive impairment and impulsivity, possibly associated with frontal lobe dysfunctions. We performed 3-dimensional magnetic resonance imaging (MRI)-based brain volumetry to characterize serial changes in frontal and prefrontal lobe volumes and compare volumetric changes with clinical symptoms. Serial changes in regional cerebral volumes were measured in a 5-year-old boy with BCECTS. Seizures were not easily controlled, and he demonstrated oromotor deficits, cognitive impairments and behavioral problems. Cognitive and behavioral deficits persisted even after remission of seizure disorder and oromotor deficits. Two BCECTS patients (5–6 years old) without neuropsychological disorders served as typical BCECTS, and nine normal subjects (4–10 years old) served as controls. Volumes of the frontal and prefrontal lobes were determined using a workstation, and prefrontal to frontal lobe volume ratio was calculated. Frontal and prefrontal lobe volumes showed no growth even after remission of seizure disorders. Prefrontal to frontal lobe volume ratio reduced slightly even after remission of both seizure disorders and EEG abnormalities. The result suggests that BCECTS presenting with atypical course may be associated with frontal lobe dysfunction resulting in cognitive and behavioral deficits.

Early speech- and language-impaired children: linguistic, literacy, and social outcomes

The aim of this study was to follow-up prospectively a cohort of preschool children originally recruited from successive referrals to speech and language therapy community clinics and to investigate their linguistic, literacy, and social outcomes at 7 to 10 years of age. Three hundred and fifty children aged 84 to 113 months (mean age 99.9mo [SD 5.4mo]) were singletons from monolingual backgrounds where there was concern about their speech and language development. Children who had severe learning difficulties, autism, oromotor deficits, dysfluency, or dysphonia were excluded. Altogether 196 (56%), 134 males and 62 females, were seen at follow-up. A control group of children who had never been referred for speech and language therapy, 57% of whom were males, was also recruited (n=94; mean age 104.4mo [SD 6.8mo]). All children were assessed on standardized measures of speech, language, and literacy. Teachers and parents completed questionnaires on educational and social outcomes. In total, 139 children in the cohort were within the normal range on standardized language assessments. About 30% of the original cohort of children continue to struggle with language, literacy, and social difficulties. The study demonstrates the long-term nature of language impairment and reinforces the need for awareness among professionals in child development and education of the ongoing needs of this population of children.

Early speech- and language-impaired children: linguistic, literacy, and social outcomes

The aim of this study was to follow-up prospectively a cohort of preschool children originally recruited from successive referrals to speech and language therapy community clinics and to investigate their linguistic, literacy, and social outcomes at 7 to 10 years of age. Three hundred and fifty children aged 84 to 113 months (mean age 99.9mo [SD 5.4mo]) were singletons from monolingual backgrounds where there was concern about their speech and language development. Children who had severe learning difficulties, autism, oromotor deficits, dysfluency, or dysphonia were excluded. Altogether 196 (56%), 134 males and 62 females, were seen at follow-up. A control group of children who had never been referred for speech and language therapy, 57% of whom were males, was also recruited (n=94; mean age 104.4mo [SD 6.8mo]). All children were assessed on standardized measures of speech, language, and literacy. Teachers and parents completed questionnaires on educational and social outcomes. In total, 139 children in the cohort were within the normal range on standardized language assessments. About 30% of the original cohort of children continue to struggle with language, literacy, and social difficulties. The study demonstrates the long-term nature of language impairment and reinforces the need for awareness among professionals in child development and education of the ongoing needs of this population of children.

Case of epilepsy with speech disorders: EEG changes and magnetoencephalographic analysis

We report a 6-year-old girl who developed speech and oromotor deficits during the course of epilepsy. Before the onset of the speech disorders, EEG showed spikes over the bilateral frontotemporal region (predominantly on the left side) and occasionally over the bilateral central region. At the onset of the speech disorders, EEG showed background activity mixed with high-voltage slow waves over diffuse regions, predominantly over the left side, and multifocal spikes and slow spike and wave (SW) complexes. Magnetoencephalography (MEG) showed three dimensionally that the calculated foci of temporal spikes were especially located beneath the somatosensory area for the right thumb. The focal EEG abnormalities over the left temporal region and the abnormal background activity were considered to have become serious and to have caused the speech and oromotor deficits. Antiepileptic drug (AED) therapy led to marked improvement of EEG, and oral corticosteroid administration had a beneficial effect on the speech disorders. Some investigators believe that benign childhood epilepsy with centrotemporal spikes (BECTS) and acquired epileptic aphasia represent a spectrum. Our patient had some of the clinical manifestations and EEG findings of these two epileptic syndromes. Therefore, we suggest that the condition of our patient lies between these two syndromes

Speech and Oromotor Deficits of Epileptic Origin in Benign Partial Epilepsy of Childhood with Rolandic Spikes (BPERS)

The authors report three children who suffered temporary oromotor or speech disturbances as focal epileptic manifestations within the frame of benign partial epilepsy of childhood with rolandic spikes and review similar cases described in the literature. The deficit can occur as an initial symptom of the disorder without visible epileptic seizures and interferes in a variable way with simple voluntary oromotor functions or complex movements including speech production, depending on the exact location and spread of the discharging epileptic focus around the perisylvian region. The most severe deficit produces the anterior operculum syndrome. More subtle non-linguistic deficits such as intermittent drooling, oromotor apraxia or dysfluency, as well as linguistic ones involving phonologic production, can occur. The rapidity of onset, progression and recovery of the deficit is very variable as well as its duration and presumably reflects the degree of epileptic activity. In some cases, rapid improvement with antiepileptic medication occurs and coincidence between the paroxysmal EEG activity (which is usually bilateral) and the functional deficit is seen. The clinical and EEG profile of the seizures disorder and the dynamic of the deficit in these cases bear a strong resemblance to what is seen in the acquired epilepsy-aphasia syndrome (Landau and Kleffner). The variations in clinical symptoms appear more related to the main site, local extension and bilaterality of the epileptic foci rather than a basic difference in physiopathology.

A follow-up case study of transitory developmental apraxia of speech: 'L'enfant à voyelles'

It is well known that the acquisition of consonants can be a more difficult developmental process than of vowels. In this study the authors describe a case of 'transitory developmental apraxia of speech' (TDAS) in which a French-speaking child, although he acquired the whole vowel system, showed a significant delay in consonant acquisition and usage. The results of systematic observations and assessments of general cognitive and specific language functioning cover a period of 4 years (age 3:8 to 7:1l), from first clinical assessment to evidence of a good academic achievement. It is argued that this case demonstrates an unusual example of TDAS. The language deficits observed could not be attributed to oromotor, neurological, cognitive or intellectual deficits. The findings here also raise crucial questions concerning the origins of such a speech disorder and why it is transient in nature.

Verbal Auditory Agnosia in Children

Four (possibly five) boys are described with a profound comprehension deficit for acoustic language, leading to severe or complete abolition of expressive speech. One boy had presumed megalencephaly from birth but was of superior intelligence. He had a severe articulation deficit from early childhood, with delayed acquisition of speech. Another boy is thought to have a small angiomatous anomaly in the depth of the left parietal lobe. No brain lesions are known in the other three. Evidence for bilateral brain dysfunction consists of minor motor abnormalities in three boys, oromotor deficits in two boys, and bilaterally synchronous diffuse or independent focal paroxysmal discharges in the EEG of three, possibly four, of the boys. Seizures have occurred in only three boys, and have been easily controlled with anticonvulsants. One boy with a grossly abnormal EEG has had no clinical seizures to date and has not benefited frome one year of anticonvulsant therapy. Two of the boys are brothers, including the boy in whome the diagnosis is questionable since speech was never normal and since he has had neither seizures for an abnormal EEG. The severity of EEG abnormalities did not correlate closely with the course of the language deficit. The relationship of this syndrome to acquired aphasia in children, to Wernicke's aphasia and pure word deafness in adults, and to developmental lagnuage disability with predominantly receptive deficits, is discussed. One child illustrated the close association between writing and phonologic encoding and decoding operations, and two children the preservation of linguistic skills provided the acoustic channel was by-passed and language presented visually. This latter point has been emphasized because of its implications for the remedial education of children with this syndrome.