Stuttering treatment for a school-age child with Down syndrome: A descriptive case report

BackgroundLittle is known about optimal treatment approaches and stuttering treatment outcomes for children with Down syndrome.Aims and methodThe purpose of this study was to investigate outcomes for a child with Down syndrome who received a combination of fluency shaping therapy and parent delivered contingencies for normally fluent speech, prolonged speech, and stuttered speech.ResultsIn-clinic speech measures obtained at post-treatment and at 4 months follow-up reflected improvements in fluency of 89.0% and 98.6%, respectively. The participant's beyond-clinic follow-up sample reflected an improvement of 95.5%. Following treatment, the participant demonstrated improved self-confidence, self-esteem, and improved participation and functioning at school.ConclusionsFindings suggest that fluency shaping with parental contingencies may be a viable treatment approach to reduce stuttering in children with Down syndrome. Future research using an experimental research design is warranted.Educational objectives: Readers will be able to describe (a) prevalence estimates of stuttering in individuals with Down syndrome, (b) the main components of a fluency shaping program for a child with Down syndrome who stutters and has co-occurring speech and language delays, and (c) speech and parent-, teacher-, and self-report treatment outcomes.     

Speech and bimanual hand organization in adult stutterers and nonstutterers

A multivariate approach was used to investigate the relationship between speech fluency and bimanual handwriting in right-handed male stutterers and nonstutterers. Handedness was determined by the Harris Test of Lateral Dominance. Subjects were required to write the numbers 1 through 12 simultaneously, as quickly as possible. Speech measures were derived from the Stuttering Severity Instrument and from a reading passage. Stutterers performed more poorly than nonstutterers on all speech measures. There were no differences in bimanual handwriting performance for the dominant hand. However, stutterers performed more poorly than nonstutterers in nondominant handwriting. There were no relationships between speech measures and bimanual handwriting for stutterers or nonstutterers. Some stutterers performed better on the handwriting task than did some nonstutterers. It is suggested that disorganization in interhemispheric integration functions may be a more fundamental problem in stuttering than is incomplete cerebral dominance.     

The effect of fast speech rate on stuttering frequency during delayed auditory feedback

Delayed auditory feedback (DAF) has been documented to improve fluency in those who stutter. The increased fluency has been attributed to the slowed speech rate induced by DAF, but recent experiments have suggested that increasing the speech rate may also decrease stuttering under DAF. This investigation described the effect of combining a fast speech rate and DAF on the fluency of four people who stutter. Fluency of the two mildly dysfluent subjects was the same for both no DAF and DAF conditions at normal and at fast oral reading rates. In contrast, the two severely dysfluent subjects improved in fluency from the no DAF to the DAF conditions. They were found to be dysfluent at both normal and fast oral reading rates without DAF. The results of the study point to the need for further research on the relationship between speech rate and stuttering frequency under conditions of DAF and no DAF. EDUCATIONAL OBJECTIVES: Readers will learn about and be able to describe how the frequency of stuttering is affected by: (1) speech rates; (2) DAF; and (3) how stuttering severity influences such effects.     

Common features of fluency-evoking conditions studied in stuttering subjects and controls: an H2O PET study

We used H₂¹⁵O PET to characterize the common features of two successful but markedly different fluency-evoking conditions — paced speech and singing — in order to identify brain mechanisms that enable fluent speech in people who stutter. To do so, we compared responses under fluency-evoking conditions with responses elicited by tasks that typically elicit dysfluent speech (quantifying the degree of stuttering and using this measure as a confounding covariate in our analyses). We evaluated task-related activations in both stuttering subjects and age- and gender-matched controls.

Areas that were either uniquely activated during fluency-evoking conditions, or in which the magnitude of activation was significantly greater during fluency-evoking than dysfluency-evoking tasks included auditory association areas that process speech and voice and motor regions related to control of the larynx and oral articulators. This suggests that a common fluency-evoking mechanism might relate to more effective coupling of auditory and motor systems — that is, more efficient self-monitoring, allowing motor areas to more effectively modify speech.

These effects were seen in both PWS and controls, suggesting that they are due to the sensorimotor or cognitive demands of the fluency-evoking tasks themselves. While responses seen in both groups were bilateral, however, the fluency-evoking tasks elicited more robust activation of auditory and motor regions within the left hemisphere of stuttering subjects, suggesting a role for the left hemisphere in compensatory processes that enable fluency.

Educational objectives: The reader will learn about and be able to: (1) compare brain activation patterns under fluency- and dysfluency-evoking conditions in stuttering and control subjects; (2) appraise the common features, both central and peripheral, of fluency-evoking conditions; and (3) discuss ways in which neuroimaging methods can be used to understand the pathophysiology of stuttering.     

Fluency disorders in genetic syndromes

The characteristics of various genetic syndromes have included “stuttering” as a primary symptom associated with that syndrome. Specifically, Down syndrome, fragile X syndrome, Prader-Willi syndrome, Tourette syndrome, Neurofibromatosis type I, and Turner syndrome all list “stuttering” as a characteristic of that syndrome. An extensive review of these syndromes indicated clients diagnosed with these syndromes do show evidence of nonfluency patterns, but not all would be considered stuttering. Many of the syndromes are marked by degrees of mental retardation that probably contribute to a higher than average prevalence of stuttering, as well as a higher than average prevalence of other fluency disorders (when compared to the population at large).

An in-depth analysis of the available data indicates that some of these genetic syndromes show patterns of stuttering that may be indicative of only that syndrome (or similar syndromes) that can be differentially diagnosed from developmental stuttering. Among these patterns are the word-final nonfluencies noted in Prader-Willi syndrome; the presence of stuttering in the absence of secondary behaviors noted in Prader-Willi syndrome and; the presence of palilalia, word-final and word-medial nonfluencies, and word-medial and word-final nonfluencies in Tourette syndrome. Implications for future research are discussed in light of these findings.

Educational objectives: The reader will be able to: (1) describe the various different genetic syndromes that are associated with fluency disorders; (2) describe the types of nonfluencies that are associated with the major types of genetic syndromes that have fluency disorders; (3) describe the behaviors that may assist in differentially diagnosing different types of speech characteristics associated with various genetic syndromes.     

音乐训练促进诗句韵律整合加工的神经过程

本研究采用ERP技术, 考察音乐训练组和对照组完成诗句押韵判断任务时, 在绝句末对韵律信息(含声调和韵母两个维度)的整合加工过程。结果发现, 在100~300 ms, 仅音乐训练组在声调/韵母合适条件下, 对韵母/声调的一致性进行深入分析, 并诱发了更大的正波; 在韵母违反条件下, 声调违反相比声调合适诱发了更小的正波。在300~750 ms, 两组被试均在绝句末对诗句内出现的韵母和声调违反进行整合分析并诱发了广泛分布的负波。不过, 对照组仅在声调/韵母合适条件下进行, 而音乐训练组则在声调/韵母违反条件下完成此过程。综上, 音乐训练组和对照组均会在诗句末完成押韵信息的整合加工, 但是音乐训练组对韵律信息(尤其是声调)的加工更敏感和快速, 并且对不同类型的违反有更精细的差异性反应     

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.     

A positron emission tomography study of short- and long-term treatment effects on functional brain activation in adults who stutter

Previous studies have shown that fluency-inducing techniques, such as choral speech, result in changes in neural activation as measured by functional neuroimaging. In the present study, positron emission tomography was used to investigate the effects of intensive behavioural treatment, followed by a 1-year maintenance program, on the pattern of cortical and subcortical activation in stuttering adults during silent and oral reading of single words. The results indicate changes in activation lateralisation, as well as a general reduction in overactivation, especially in the motor cortex, following treatment. The results are discussed in light of previous functional imaging studies with stuttering adults.

Educational objectives: The reader will learn about and be able to describe the: (1) use of functional neuroimaging PET in the study of stuttering; (2) differences in neural activation between stuttering and non-stuttering adults; and (3) effects of behavioural fluency treatment on cortical and subcortical activations in stuttering speakers.     

Cortical plasticity associated with stuttering therapy

Neuroimaging studies have indicated that persistent developmental stuttering (PDS) may be associated both with an abnormality in white matter of left-hemispheric speech areas and a right-hemispheric hyperactivity. The latter may compensate for the deficient structural connectivity in the left hemisphere. To investigate the effects of stuttering therapy on brain activity nine male adults with PDS underwent functional magnetic resonance imaging (fMRI) before and within 12 weeks after fluency shaping therapy. Brain response differences during overt sentence reading before and after therapy were assessed by utilizing random effects analyses. After therapy, a more widespread activation was observed in frontal speech and language regions and temporal areas of both hemispheres, particularly and more pronounced on the left side. Interestingly, distinct posttreatment left-sided activation increases were located directly adjacent to a recently detected area of white matter anomaly [M. Sommer, M.A. Koch, W. Paulus, C. Weiller, C. Buchel (2002). Disconnection of speech-relevant brain areas in persistent developmental stuttering. The Lancet, 360, 380-383] suggesting that fluency shaping techniques reorganize neuronal communication between left-sided speech motor planning, motor execution, and temporal areas. Hence, a therapeutic mechanism can be assumed to remodel brain circuitry close to the source of the dysfunction instead of reinforcing compensation via homologous contralateral brain networks. EDUCATIONAL OBJECTIVES: The reader will learn about and be able to: (1) describe brain activation changes detected shortly after fluency-shaping therapy; (2) identify left-hemispheric regions where a (re)functionalization after fluency-shaping therapy seems to occur adjacent to a recently described abnormal white matter region in PDS subjects; and (3) discuss how an effective cerebral compensation mechanism for stuttering could work.     

Treatment with medications affecting dopaminergic and serotonergic mechanisms: effects on fluency and anxiety in persons who stutter

Medications with dopamine antagonist properties, such as haloperidol, and those with serotonin reuptake inhibitor properties, such as clomipramine, have been shown to improve fluency. To examine the degree to which each of these two pharmacological mechanisms might independently affect fluency, a selective serotonin reuptake inhibitor, paroxetine, and a selective dopamine (D-2) antagonist, pimozide, were evaluated. Both types of medications also affect mood and anxiety, factors that could influence fluency levels. Therefore, we also evaluated the medications’ effects on generalized and speech-related anxiety and the relationships between changes in anxiety and changes in fluency in 11 subjects with a history of developmental stuttering. The randomized, double blind, placebo-controlled crossover study that was designed had to be terminated prior to completion due to severe side effects following withdrawal from paroxetine. Even with a reduced sample size (n = 6), significant improvement in percent fluent speaking time (p = 0.02) was found using a telephone task between baseline and pimozide (n = 6), with average duration of dysfluencies significantly shorter (p = 0.04) but no significant difference in the estimated number of dysfluencies per minute. This significant improvement was associated with non-significant increases in generalized anxiety, but non-significant decreases in speech-related anxiety. No significant differences were found in fluency between baseline and paroxetine (n = 5). These preliminary results suggest that fluency improvement is more likely to be mediated by dopaminergic rather than serotonergic mechanisms. Due to its side effects, however, pimozide may be considered a risk for treatment of stuttering.

Educational objectives: As a result of reading this paper the reader will describe and explain: (1) how medications may affect fluency and the rationale for selecting medications for treatment trials; (2) the interrelationship between fluency and anxiety; and (3) factors important in developing clinical trials using medications.     

A comparison of the Probe technique and conventional therapy for young stutterers

This study contrasted the gains made in speech theraphy by 39 stutterers, 15 of whom were treated by conventional methods and 24 by the Probe technique. It was found that the Probe technique yielded substantially greater gains in fluency than did the conventional method, having more influence on final performance than factors such as initial fluency, time since onset of stuttering, or the presence of other unacceptable behaviors.     

BEHAVIORAL SELF-CONTROL OF STUTTERING USING TIME-OUT FROM SPEAKING

Research has shown that stuttering may be attenuated by a variety of response-contingent consequences. To date, however, few attempts have been made to develop comprehensive clinical procedures based on the operant manipulation of stuttering. The present research examined the efficacy of self-initiated response-contingent time-out from speaking in two exeperiments involving a single subject. Multiple baseline designs were used in which the subject's speech was monitored in a variety of laboratory and community settings. In Experiment 1, the time-out procedure produced reliable reductions in disfluency across all observational settings. Experiment 2 combined a response-cost contingency with time-out in an attempt to increase the reliability with which the subject timed himself out, and to increase further the effectiveness of the procedure. Increases in reliability were produced, and the associated improvements in fluency were maintained at 6- and 12-months' follow-up.     

The effects of differential vowel prolongations on perceptions of speech naturalness

Prolongation of speech sounds is currently used to modify stuttering and enhance fluency. Prolonged speech (PS) (e.g., prolonged vowels, prolongation throughout utterances) is, however, often perceived as unnatural by listeners. This study examined at which durations and in which contexts 52 college students (primary language was American English) perceived PS to be unnatural. Stimuli were limited to controlled variation in prolongation of the vowel in the middle single-syllable word of a carrier phrase (i.e., say word again). The prolongation was effected by digital waveform manipulation within the Kay Elemetrics Computerized Speech Laboratory (CSL). The listeners judged if they strongly agreed, agreed, or disagreed that the phrases sounded natural. Results indicated that the extent of vowel duration (and possibly context) does influence listener's perception of speech naturalness, findings which can be applied to facilitate fluency therapy.

Educational objectives: (1) The reader will learn about and be able to summarize the digital waveform manipulation procedure in the study. (2) The reader will learn about and be able to describe the effects of differential vowel prolongation on listener's perception of speech naturalness. (3) The reader will learn about and evaluate how differential vowel prolongation can be used to enhance fluency.     

Pseudofluency in adults who stutter: the illusory outcome of therapy

The majority of therapy programs for people who stutter are aimed at modifying the entire speech output by using techniques that reduce the overt signature events. Use of these techniques for extended time periods are thought to induce true fluency that is automatic, natural, and effortless. It is proposed that the present form of therapeutic intervention induces pseudofluency rather than true fluency. Pseudofluency is the speech posrtherapy of persons who stutter, free of the discrete signature events of stuttering, but replaced by cognitively mediated gestures that are embedded as continuous prolongation or masked stuttering events throughout the speech act. This may account for the high rate of relapse and the problems associated with the maintenance, stability, and naturalness of speech after stuttering therapy.     

The nature and treatment of stuttering as revealed by fMRI A within- and between-group comparison

This article reviews some of our recent functional magnetic resonance imaging (fMRI) studies of stuttering. Using event-related fMRI experiments, we investigated brain activation during speech production. Results of three studies comparing persons who stutter (PWS) and persons who do not stutter (PWNS) are outlined. Their findings point to a region in the right frontal operculum (RFO) that was consistently implicated in stuttering. During overt reading and before fluency shaping therapy, PWS showed higher and more distributed neuronal activation than PWNS. Immediately after therapy differential activations were even more distributed and left sided. They extended to frontal, temporal, and parietal regions, anterior cingulate, insula, and putamen. These over-activations were slightly reduced and again more right sided two years after therapy. Left frontal deactivations remained stable over two years of observation, and therefore possibly indicate a dysfunction. After therapy, we noted higher activations in persons who stutter moderately than in those who stutter severely. These activations might reflect patterns of compensation. We discuss why these findings suggest that fluency-inducing techniques might synchronize a disturbed signal transmission between auditory, speech motor planning, and motor areas.

Educational objectives: The reader will learn about and be able to: (1) identify regions of brain activations and deactivations specific for PWS; (2) describe brain activation changes induced by fluency shaping therapy; and (3) discuss the correlation between stuttering severity and brain activation.     

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.     

Semantic distinctions and memory for complex sentences

This investigation studied what people remember in recalling complex sentences, whether it is certain semantic distinctions or merely transformational markers. After short intervals 24 subjects tried to recall sentences of six kinds which formed paraphrase sets: S₁ before S₂, S₁ and then S₂, After S₁ S₂, S₂ after S₁, S₂ but first S₁, and Before S₂ S₁. (S₁ and S₂ denote first and second clauses in temporal, not linguistic, order.) Subjects remembered the underlying sense of sentences with S₁-S₂ clause ordering better than those with S₂-S₁ clause ordering, regardless of transformational complexity. Subjects also showed a response bias, hence better verbatim recall, for sentences with subordinate clause second and for sentences with S₁-S₂ clause ordering. Sentence confusions indicated that subjects remembered three semantic distinctions: the temporal order, order of mention, and main-subordinate relation of the two described events. A theory of memory for marked and unmarked semantic distinctions was used to account for the results.     

Stuttering: a dynamic motor control disorder

The purpose of this review is to determine what neural mechanisms may be dysfunctional in stuttering. Three sources of evidence are reviewed. First, studies of dynamic inter-relationships among brain regions during normal speech and in persons who stutter (PWS) suggest that the timing of neural activity in different regions may be abnormal in PWS. Second, the brain lesions associated with acquired stuttering are reviewed. These indicate that in a high percentage of cases, the primary speech and language regions are not affected but lesions involve other structures, such as the basal ganglia, which may modulate the primary speech and language regions. Third, to characterize the motor control disorder in stuttering, similarities and differences from focal dystonias such as spasmodic dysphonia (SD) and Tourette’s syndrome (TS) are reviewed. This review indicates that the central control abnormalities in stuttering are not due to disturbance in one particular brain region but rather a system dysfunction that interferes with rapid and dynamic speech processing for production.

Educational objectives: The reader will be able to describe: (1) the similarities and differences between stuttering and other speech motor control disorders, (2) which brain lesions are most likely to produce acquired stuttering in adults, and (3) what type of brain abnormality most likely underlies stuttering.     

Case descriptions of nonfluency and loci Initial and follow-up conversations with three preschool children

It has been hypothesized that fluency development may be influenced by linguistic uncertainty, slow speech milestones, and language delay. The purpose of this study was to describe the nonfluent speech characteristics of a stuttering child, a language-impaired child, and a nonstuttering child. Initial assessment results revealed that the stutterer produced more stuttering, the language-impaired child emitted more normal disfluency, and the nonstutterer had only a few normal disfluencies. Six months after therapy, the stutterer had decreased stuttering behavior but increased in normal disfluency. Six months after the initial evaluation and without fluency intervention, the language-impaired child showed an overall decrease in nonfluent behaviors, especially in part-word, whole-word, and phrase repetitions. Both the stutterer and the language-impaired children revised. Minimal nonfluency was observed in the nonstutterer during the initial and postobservation periods. All children produced more nonfluencies on conjunctions and pronouns. Implications for language therapy will be presented.