Analysis of five cases with neurogenic stuttering following brain injury in the basal ganglia

This study examined stuttering patterns in five patients with basal ganglia injury. None of the patients had a history of developmental stuttering. Four patients were right-handed; one patient was ambidextrous. Stuttering tests administered to patients assessed sentence repetition, reading aloud, explanations of a comic strip, and conversation. Accessory behaviors such as facial grimaces, associated movements of the limbs, and avoidance behaviors were observed. The results of this study differ from those of previous studies of neurogenic stuttering in several respects: (1) blocks were frequently observed. (2) Adaptation was observed. (3) Almost all stuttering occurred at the initiation of words. (4) Across patients, stuttering frequency did not vary in a consistent manner with speaking task. New speech characteristics for neurogenic stuttering without aphasia following injury to the basal ganglia are described.Educational objectives: After reading this text, the reader will be able to: (1) provide characteristics of neurogenic stuttering after the basal ganglia in patients without aphasia; (2) discuss the difference of the features and characteristics of stuttering between previously reported patients and present patients.     

Motor sequence learning in children with recovered and persistent developmental stuttering: preliminary findings

PurposePrevious studies have associated developmental stuttering with difficulty learning new motor skills. We investigated non-speech motor sequence learning in children with persistent developmental stuttering (CWS), children who have recovered from developmental stuttering (CRS) and typically developing controls (CON).MethodsOver the course of two days, participants completed the Multi-Finger Sequencing Task, consisting of repeated trials of a10-element sequence, interspersed with trials of random sequences of the same length. We evaluated motor sequence learning using accuracy and response synchrony, a timing measure for evaluation of sequencing timing. We examined error types as well as recognition and recall of the repeated sequences.ResultsCWS demonstrated lower performance accuracy than CON and CRS on the first day of the finger tapping experiment but improved to the performance level of CON and CRS on the second day. Response synchrony showed no overall difference among CWS, CRS and CON.Learning scores of repeated sequences did not differ from learning scores of random sequences in CWS, CRS and CON. CON and CRS demonstrated an adaptive strategy to response errors, whereas CWS maintained a high percentage of corrected errors for both days.ConclusionsOur study examined non-speech sequence learning across CWS, CRS and CON. Our preliminary findings support the idea that developmental stuttering is not associated with sequence learning per se but rather with general fine motor performance difficulties.     

Some correlates of stuttering severity judgments

Thirteen adult male stutterers recorded the Amplifier Passage on high-quality audiotape. One sentence from each recording was paired with one sentence from each of the remaining 12 recordings. The resulting 78 sentence pairs were presented to eight speech-language pathologists who were asked to judge which stutterer in each pair was more severe and to rate the confidence of each of these judgements. The confidence ratings were analyzed by means of the individual differences model of multidimensional scaling. The results suggested that reading rate and the number of intrasentence pauses were important parameters in determining the severity of stuttering.     

fMRI of developmental stuttering: a pilot study

The purpose of this investigation was to explore the feasibility of fMRI in the study of developmental stuttering. Speech contrasts (loud versus silent reading) and language contrasts (reading of semantically meaningful text versus nonsense words) of six developmental stutterers and six nonstutterers were compared using a commercial 1 Tesla MR-Scanner (Siemens Expert). Results indicate that mapping cortical function in persons who stutter is indeed feasible, even with a 1TMR-system. Compared to normals the stutterers seemed to employ different and particularly less differentiated auditory and motor feedback strategies in speech. They apparently rely on auditory processing and on cerebellar contribution as much during silent reading as during reading aloud. Moreover, they showed a greater involvement of the right hemisphere in language processing, activating not only the typical language areas on the left but also and with equal magnitude the right side homologues of these areas. In spite of the promising results, at present several practical problems such as possible movement artifacts and possible masking through scanner noise still hamper a more straightforward use of fMRI in the study of developmental stuttering.     

Multidimensional sequence learning in patients with focal basal ganglia lesions

Parkinson's patients have been found to be impaired in learning movement sequences. In the current study, patients with unilateral basal ganglia lesions due to stroke were tested on a serial reaction time task in which responses were based on the spatial location of each stimulus. The spatial locations either followed a fixed sequence or were selected at random, with learning operationalized as the difference in reaction time between these two conditions. In addition, three response-to-stimulus intervals were used, and these either followed a fixed sequence or were randomized. Compared to control participants, the patients showed normal learning of the spatial and temporal sequences, as well as normal cross-dimensional learning. This was true for performance with either the contralesional or ipsilesional hand. Sequence learning was not correlated with maximum tapping rate, a simple measure of motor impairment. These results raise questions concerning the use of Parkinson's disease as a model for studying basal ganglia dysfunction.     

Motor functions of the basal ganglia

Summary A study of movement disorders such as Parkinson's disease and Huntington's disease can provide an indication of the motor functions of the basal ganglia. Basal-ganglia diseases affect voluntary movement and can cause involuntary movement. Deficits are often manifested during the coordination of fine multi-joint movements (e. g., handwriting). The disturbances of motor control (e.g. akinesia, bradykinesia) caused by basal-ganglia disorders are illustrated. Data suggest that the basal ganglia play an important role in the automatic execution of serially ordered complex movements.     

Parent perceptions of an integrated stuttering treatment and behavioral self-regulation program for early developmental stuttering

PurposeRecent research has identified approximately half of children who stutter present with self-regulation challenges. These manifest in elevated inattentive and/or impulsive behaviours, aligned with attention deficit hyperactivity disorder (ADHD) symptoms. These symptoms have been found to influence the child’s responsiveness to their stuttering treatment, and may exacerbate the psychosocial consequences of stuttering for them and their families. Early stuttering intervention identifies parents as key agents of change in the management of their children’s stuttering. This study sought feedback from parents regarding their experiences with an integrated stuttering treatment and behavioral self-regulation program for early developmental stuttering, addressing the child’s self-regulation challenges.MethodEight parents of children who stutter who had co-occurring self-regulation challenges completed the integrated program. This incorporated the Triple P--Positive Parenting Program adapted for the developmental stuttering population, and the Curtin University Stuttering Program (CUSP). Semi-structured qualitative interviews were conducted to capture parents’ reflections on, and experiences with, the integrated program.ResultsThematic analysis identified several major themes regarding the parents’ experiences with the integrated program: emotional impact on parents, child self-regulation, link between stuttering and behaviour, parent self-regulation, impact on family dynamics, and overall positive perceptions of the integrated program. All of the parents indicated they would recommend the program to future parents of children who stutter.ConclusionThis study provides insights into parents’ perceptions regarding an integrated intervention approach for early stuttering and behavior management. It also indicates how adopting a holistic approach to stuttering intervention is positive and has social validity.     

Towards a functional neural systems model of developmental stuttering

This paper overviews recent developments in an ongoing program of brain imaging research on developmental stuttering that is being conducted at the University of Texas Health Science Center, San Antonio. This program has primarily used H₂¹⁵O PET imaging of different speaking tasks by right-handed adult male and female persistent stutterers, recovered stutterers and controls in order to isolate the neural regions that are functionally associated with stuttered speech. The principal findings have emerged from studies using condition contrasts and performance correlation techniques. The emerging findings from these studies are reviewed and referenced to a neural model of normal speech production recently proposed by Jürgens [Neurosci. Biobehav. Rev. 26 (2002) 235]. This paper will report (1) the reconfiguration of previous findings within the Jürgens Model; (2) preliminary findings of an investigation with late recovered stutterers; (3) an investigation of neural activations during a treatment procedure designed to produce a sustained improvement in fluency; and (4) an across-studies comparison that seeks to isolate neural regions within the Jürgens Model that are consistently associated with stuttering. Two regions appear to meet this criterion: right anterior insula (activated) and anterior middle and superior temporal gyri (deactivated) mainly in right hemisphere. The implications of these findings and the direction of future imaging investigations are discussed.

Educational objectives: The reader will learn about (1) recent uses of H₂¹⁵O PET imaging in stuttering research; (2) the use of a new neurological model of speech production in imaging research on stuttering; and (3) initial findings from PET imaging investigations of treated and recovered stutterers.     

A case of persistent neurogenic stuttering following a missile wound

Observational criteria that have been proposed to distinguish developmental from neurogenic stuttering are critically reviewed. It is concluded that both conditions may closely resemble each other. As an example, a case of neurogenic stuttering following a missile would is reported.     

The differential role of premotor frontal cortex and basal ganglia in motor sequence learning: evidence from focal basal ganglia lesions

There has been a growing interest in the differential role of various neural structures in implicit learning processes. The goal of our study was to clarify how focal lesions restricted to the basal ganglia interfere with different aspects of implicit visuo-motor sequence learning. A version of the Serial Reaction Time Task (SRTT) of Nissen and Bullemer using a 12-trial sequence was administered. A total of 20 subjects with focal basal ganglia lesions caused by ischemic or hemorrhagic infarction and 20 matched control subjects participated in this study. The results indicate that subjects with focal basal ganglia lesions showed unimpaired implicit learning of a 12-item motor sequence. Subjects with basal ganglia lesions, however, had more difficulties improving their general proficiency with the reaction-time task independent of sequence-specific learning. We observed a tendency toward smaller regional volumes in the cerebellum and left pre-supplementary motor area (pre-SMA) of subjects with basal ganglia lesions. Smaller cerebellar and pre-SMA volumes were related to lower implicit learning performance in the lesion group. The size of lesions in the basal ganglia was not related to sequence-specific implicit learning but had a significant influence on subjects' general proficiency for execution of the reaction-time task. We propose that implicit learning is achieved by a distributed network of cortical and subcortical structures. The basal ganglia seem to be responsible for adjusting to the general requirements of a task rather than for learning specific associations between stimuli that might be accomplished by premotor frontal areas and the cerebellum instead.     

Severity of sleep problems in children with severe learning difficulties: Description and correlates

In this study we have examined the percentage occurrence of settling and waking problems and of sleeping in the parents' bed in a sample of children with sleep problems. We found that about two-thirds had settling problems, nearly nine-tenths had waking problems, and one-third slept regularly in their parents' bed. Parents of children with sleep problems were more likely to experience a range of family problems than were parents of children without problems, and they were more likely to report a range of symptoms of stress. Most children with settling problems took at least two hours to settle to sleep at night. This behaviour was mainly tolerated or accepted by the parents. Parents gave a variety of suggestions as to triggers for settling problems, from being upset during the day to fears, illness and alterations in routine. Most parents thought the underlying explanation was the child's intellectual impairment. Over half the children with waking problems woke up every night, while 85% woke up at least three nights a week. One-third woke up three or more times a night. About two-thirds took up to half an hour to resettle, with one-third taking up to an hour. Parents (mainly mothers) tended to get up to attend to the waking child. About half of the children were thought to need attention because of physical disabilities. Illness, alteration in routine, becoming ‘overtired’ and ‘upset’ were thought to trigger waking problems. Intellectual impairment was again seen as the more general underlying reason for waking problems. Very few children were receiving treatment for their problems—7% were receiving medication and one was receiving a behavioural programme. Few parents had a systematic bedtime routine for their child and few had a consistent agreed response to settling or waking problems. When we examined the relationship between certain family variables and severity of sleep problems a number of relationships were found. Sleep problem severity was related to maternal responsiveness, maternal stress, impact of the child on the family, maternal behaviour towards the child, maternal attitudes towards the child and marital satisfaction. Severity of sleep problems was also related to certain child characteristics. Children with more severe problems tended to present more problems in their daytime behaviour. Sleep problem severity was also found to be related to child communication skills. These findings, together with those from other pilot studies, suggest that using behavioural methods of managing sleep problems might prove a promising way forward.     

Impairment of temporal organization of speech in basal ganglia diseases.

Absolute and relative speech timing were examined in patients suffering from Parkinson's, Huntington's, and Wilson's disease. The task was to speak a standard sentence 10 times, first slowly, and then successively faster up to maximum rate. All patient groups had low maximal speech rates and showed decreased variability of speech rate. The duration of pauses between words was the same as in normals and the relative time structure of the test sentence was basically preserved. For comparison, two cases with nonfluent aphasia had even slower speech rates, large increases in pause duration, and major changes in relative speech timing. The results show the same type of alterations of the temporal organization of speech as those characteristic for rapid alternating limb movements in such patients. They support the view that the speech and skeletomotor systems share common neural control modes despite fundamental biomechanical differences. The common denominator between the speech and the skeletomotor disturbances in basal ganglia diseases may be the undamping and slowing of a fast central oscillator.     

Two syndromes of aphasia occurring with ischemic lesions involving the left basal ganglia

Seven aphasic patients with circumscribed left basal ganglia infarctions were investigated within the first 15 days after their strokes. Five showed transcortical motor aphasia initially. Two patients suffered from anterior chorioideal artery infarction. As this vessel does not contribute to cortical supply, cortical malfunction probably cannot account for the language deficits. Patients with infarctions in the supply area of anterior lenticulostriate arteries became fluent with frequent phonemic and semantic paraphasias resembling Wernicke's aphasia. Three of four patients showed transiently more pronounced deficits in auditory than in written-language comprehension.     

Neurocognitive outcomes in children with unilateral basal ganglia arterial ischemic stroke and secondary hemidystonia

Dystonia is a movement disorder that involves excessive, involuntary muscle contractions resulting in repetitive movements and/or abnormal posturing. One common cause of unilateral dystonia in childhood is ischemic stroke involving the basal ganglia and/or thalamus. Virtually nothing is known about neuropsychological outcomes in children who have dystonia following basal ganglia stroke. The present study explored whether or not children with secondary dystonia experience additional cognitive challenges when compared to children with similar patterns of brain injury, but no dystonia. We examined intellectual function, academics, and several aspects of executive function in children with unilateral basal ganglia stroke during childhood, comparing those with dystonia and those without. Although groups did not differ in terms of lesion size, we found significantly lower performance on measures of verbal and nonverbal reasoning, inhibitory control, and academic ability in children with secondary dystonia compared to those without. In contrast, there were no significant group differences on parent ratings of their child’s executive function in daily life. These findings suggest that maladaptive reorganization following basal ganglia stroke may contribute to the development of secondary dystonia and also to poor intellectual and academic outcomes in this group.     

Behavioral correlates of developmental expressive language disorder

The association of behavior problems with preschool language disorders has been documented extensively. However, researchers have typically failed to differentiate subgroups of language-impaired children, to use observational data in documenting the behavior disorders, or to study children at the youngest ages. Using a multimodal assessment, this study examined parent-child interaction and behavior problems in a clearly defined subgroup of language-impaired children, those with developmental expressive language disorder (ELD). These children exhibit a delay in expressive language compared with receptive language and nonverbal cognitive skills. Subjects were identified and studied at the youngest age at which the disorder can be assessed. A group of ELD children, averaging 27 months of age, was contrasted with a group of normally developing children, matched for age, sex, and receptive language ability. Groups were compared on observed parent-child interactions as well as maternal responses on the Parenting Stress Index, the Eyberg Child Behavior Inventory, and a behavior-related structured interview. ELD children, when compared with normally developing children, exhibited higher levels of negative behavior and were perceived as different by their parents.Portions of these data were presented at the biennial meeting of the Society for Research in Child Development, Baltimore, April 1987, and at the annual meeting of the Society for Pediatrie Research, Anaheim, California, April 1987. This work has been supported by NIMH grant no. 1 R03 MH41603 to author Fischel, and by NICHD grant no. 1 ROI HD19245 to authors Whitehurst and Fischel. It has also been supported by grants of equipment from Commodore Business Machines, Inc., Koala Corporation, and NEC Telephones. We thank the Department of Pediatrics at the Nassau County Medical Center for the use of their facilities.     

Electromyographic indications of laryngeal-area activity during stuttering expectancy

Using surface electrodes, mean microvolt values of laryngeal-area muscle activity were determined for 13 male and 6 female stutterers during 10-sec periods in which single words were silently read and stuttering expectancy was indicated. No significant differences in levels of laryngeal-area muscle activity were found between periods of fluency expectancy and stuttering expectancy preceding both fluent and stuttered speech. The results were interpreted as being nonsupportive of the hypothesis that a breakdown in stutterers' laryngeal-area muscle activity occurs during moments of stuttering expectancy. In addition, the results suggest that the most common rationale for the utilization of laryngeal biofeedback procedures to enhance fluency should be questioned.     

Effects of focal basal ganglia lesions on timing and force control

Studies of basal ganglia dysfunction in humans have generally involved patients with degenerative disorders, notably Parkinson's disease. In many instances, the performance of these patients is compared to that of patients with focal lesions of other brain structures such as the cerebellum. In the present report, we studied the performance of patients with focal basal ganglia lesions on three fundamental motor tasks. The patients all had suffered unilateral damage in the striatum and were tested in the chronic state. The first task required the participants to tap with their index finger as fast as possible; this test provided a simple assessment of motor competence. Compared to controls, the maximum tapping rate was lower for the patients when tapping with their contralesional limb, although the deficit was not severe. The second and third tasks were designed to assess timing and force control, two functions that have been associated with basal ganglia function. The patients performed similar to controls on both tasks and showed no evidence of impairment when using their contralesional limb compared to their ipsilesional limb. The results indicate that unilateral basal ganglia lesions tend to produce minor motor problems in force control, and fail to support the hypothesized role of the basal ganglia in timing.