Cost of Illness and Health-Related Quality of Life for Stuttering: Two Systematic Reviews.

PURPOSE: For those who stutter, verbal communication is typically compromised in social situations. This may attract negative responses from listeners and stigmatization by society. These have the potential to impair health-related quality of life across a range of domains, including qualitative and quantitative impacts on speech output, mental health issues, and failure to attain educational and occupational potential. These systematic reviews were designed to explore this matter using traditional health economics perspectives of utility measures and cost of illness. METHOD: Studies were included if they involved children, adolescents, or adults with stuttering as a primary diagnosis. The quality of life search strategy identified 2,607 reports, of which three were included in the quality of life analysis. The cost of illness search strategy identified 3,778 reports, of which 39 were included in the cost of illness analysis. RESULTS: Two of the three studies included in the quality of life analysis had a high risk of bias. When measured using utility scores, quality of life for people who stutter was in the range of those reported for chronic health conditions such as diabetes mellitus, cardiovascular disease, and cancer. However, there is little such evidence of quality of life impairment during the preschool years. Studies included in the cost of illness analysis carried considerable risk of bias overall. CONCLUSIONS: For people who stutter, there are substantive direct and indirect costs of illness. These include impairment, challenges, and distress across many domains throughout life, including income, education, employment, and social functioning. Evidence of quality of life impairment using utility measures is extremely limited. If this situation is not remedied, the lifetime impairment, challenges, and distress experienced by those who stutter cannot be documented in a form that can be used to influence health policy and health care spending. SUPPLEMENTAL MATERIAL: https://doi.org/10.23641/asha.24168201.

Lidcombe Program translation to community clinics in Australia and England.

BACKGROUND: Early intervention is essential healthcare for stuttering, and the translation of research findings to community settings is a potential roadblock to it. AIMS: This study was designed to replicate and extend the Lidcombe Program community translation findings of O'Brian et al. (2013) but with larger participant numbers, incorporating clinicians (speech pathologists/speech anlanguage therapists) and their clients from Australia and England. METHODS & PROCEDURES: Participants were 51 clinicians working in public and private clinics across Australia (n = 36) and England (n = 15), and 121 of their young stuttering clients and their families. Outcome measures were percentage of syllables stuttered (%SS), parent severity ratings at 9 months post-recruitment, number of clinic visits to complete Stage 1 of the Lidcombe Program, and therapist drift. OUTCOMES & RESULTS: Community clinicians in both countries achieved similar outcomes to those from randomized controlled trials. Therapist drift emerged as an issue with community translation. Speech and language therapists in England attained outcomes 1.0%SS above the speech pathologists in Australia, although their scores were within the range attained in randomized trials. CONCLUSIONS & IMPLICATIONS: Community clinicians from Australia and England can attain Lidcombe Program outcome benchmarks established in randomized trials. This finding is reassuring in light of the controlled conditions in clinical trials of the Lidcombe Program compared with its conduct in community practice. The long-term impact of therapist drift in community clinical practice with the Lidcombe Program has yet to be determined. WHAT THIS PAPER ADDS: What is already known on the subject The Lidcombe Program is an efficacious early stuttering intervention. Translation to clinical communities has been studied with one Australian cohort. What this paper adds to existing knowledge A larger translation cohort is studied, comprising community clinicians and children in Australia and England. What are the potential or actual clinical implications of this work? Community clinicians from Australia and England can attain Lidcombe Program outcome benchmarks established in randomized trials. This finding is reassuring in light of the controlled conditions in clinical trials of the Lidcombe Program compared with its conduct in community practice.

Searching for Lidcombe Program mechanisms of action: Inter-turn speaker latency.

The Lidcombe Program is a well-established and efficacious treatment for early stuttering, but little is currently known about its mechanisms of action. The present report explores the possibility that inter-turn speaker latency might be associated with such mechanisms of action. Inter-turn speaker latency was measured in audio recordings of children, parents, and clinicians conversing, taken during Lidcombe Program treatment consultations. Five clinicians reduced their inter-turn speaker latencies during clinical consultations when they were speaking to children, in comparison with when they were speaking to parents. It is possible that inter-turn speaker latency is associated with the Lidcombe Program treatment process vicariously, and this possibility requires further research.

Stuttering, family history and counselling: A contemporary database.

BACKGROUND: Information about genetic influence is useful to when counselling parents or caregivers who have infants and children at risk for stuttering. Yet, the most comprehensive family aggregate database to inform that counselling is nearly four decades old (Andrews et al., 1983). Consequently, the present study was designed to provide a contemporary exploration of the relationship between stuttering and family history. METHODS: Data were sourced from the Australian Stuttering Research Centre, comprising 739 participants who presented for assessment, treatment, or investigation of stuttering. Reported family history data were acquired from pedigrees collected during assessment. We sought to establish the relation of the following variables to family history of stuttering: incidence, proband sex, parent sex, stuttering severity, age, reported age of stuttering onset, and impact of stuttering. Data were analysed with chi-square tests for independence, logistic and linear regression models. RESULTS: Results were broadly consistent with existing data, but the following findings were novel. Males and females who stutter have the same increased odds of having a father who stutters relative to a mother who stutters. Males had later stuttering onset than females, with genetic involvement in this effect. There was a greater impact of stuttering for females than males with a family history of stuttering. CONCLUSION: These findings have clinical applications. Speech-language pathologists may have infant or child clients known to them who are at risk of beginning to stutter. Information from the present study can be applied to counselling parents or caregivers of such children about stuttering and family history.

The Complexity of Stuttering Behavior in Adults and Adolescents: Relationship to Age, Severity, Mental Health, Impact of Stuttering, and Behavioral Treatment Outcome.

PURPOSE: This study investigated the complexity of stuttering behavior. It described and classified the complexity of stuttering behavior in relation to age, behavioral treatment outcomes, stuttering severity, anxiety-related mental health, impact of stuttering, and gender. METHOD: For this study, a taxonomy was developed-LBDL-C7-which was based on the Lidcombe Behavioral Data Language of stuttering. It was used by five experienced judges to analyze the complexity of stuttering behavior for 84 adults and adolescents before and after speech restructuring treatment. Data were 3,100 stuttering moments, which were analyzed with nominal logistic regression. RESULTS: The complexity of stuttering behavior appears not to change as a result of treatment, but it does appear to change with advancing age. Complexity of stuttering behavior was found to be independently associated with clinician stuttering severity scores but not with percentage of syllables stuttered or self-reported stuttering severity. Complexity of stuttering behavior was not associated with gender, anxiety, or impact of stuttering. CONCLUSION: Clinical and research applications of these findings are discussed.

White matter connectivity in neonates at risk of stuttering: Preliminary data.

BACKGROUND: Developmental stuttering is thought to be underpinned by structural impairments in the brain. The only way to support the claim that these are causal is to determine if they are present before onset. MATERIALS AND METHODS: Magnetic resonance imaging (MRI) was conducted on 18 neonates, aged 8-18 weeks, 6 of whom were determined to be genetically at risk of stuttering. RESULTS: With tract-based spatial statistics (TBSS) analysis, no statistically significant differences were found between the at-risk group and the control group. However, fractional anisotropy (FA), mean diffusivity (MD), and radial diffusivity (RD) in the corpus callosum of the at-risk group were lower (uncorrected) than in the control group. Automated Fiber Quantification (AFQ) yielded lower FA in the at-risk group than in the control group in the medial section of the callosum forceps minor. DISCUSSION: The findings, albeit with a small number of participants, support the proposition that reduced integrity of white matter in the corpus callosum has a causal role in developmental stuttering. Longitudinal research to determine if children with this impairment at birth later start to stutter is needed to confirm this. The left arcuate fasciculus is thought to develop as speech develops, which likely explains why there were no abnormal findings in this area in our at-risk neonates so soon after birth. This is the first study to investigate the brains of children before the onset of stuttering, and the findings warrant further research.

Comparison of Stuttering Severity and Anxiety During Standard and Challenge Phone Calls.

PURPOSE: This study was designed to answer three questions. (a) Does percentage of syllables stuttered (%SS) differ between standard and challenge phone calls. (b) Does anxiety differ between standard and challenge phone calls. (c) Is there a relationship between %SS and anxiety during standard and challenge phone calls? METHOD: Participants were 230 adults diagnosed with stuttering, who were participants from five clinical trials. Each participant received two 10-min phone calls at pretreatment and a further two phone calls 6 months or 20 weeks postrandomization. One phone call was standard, and the other presented challenge: occasionally disagreeing with, interrupting, and talking over participants, or asking for clarification of their views. RESULTS: Statistically significant, but clinically minor, increases of %SS and anxiety occurred during the challenge phone calls. There was a statistically significant association between %SS and anxiety. CONCLUSIONS: Variable phone call procedures to assess stuttering severity in clinical trials are not likely to spuriously inflate or deflate treatment outcomes to a clinically important extent. Regardless, the present results suggest that there is statistical merit in controlling the nature of phone calls during clinical trials with the simple and replicable method developed in this report. Additionally, there is procedural merit in the challenge phone call procedure; it is a more valid representation of the challenges of everyday speech than the standard procedure. However, a disadvantage of the challenge phone call procedure is the practical issues associated with its use. The clinical and theoretical applications of the results are discussed.

Conversational speech of school-age children after syllable-timed speech treatment for stuttering.

Purpose: The purpose of this laboratory study was to investigate whether rhythmic speech was primarily responsible for stuttering reductions in four school-aged children after the instatement stage of the Westmead Program of syllable-timed speech (STS) intervention. The study was designed to inform further development of the program. Reduction in variability of vowel duration is a marker of STS, and it was predicted that this would be present in the children's conversational speech after Stage 1 of the program if they were using STS. To strengthen such a finding, it was also predicted that there would be no reduction in articulation rate, sentence complexity, and utterance length after treatment, as there is evidence that reductions in these can reduce stuttering. Perceptual judgments of speech quality after treatment were also made by independent listeners.Method: Participants were four children, ages 8-11 years, who completed Stage 1 of an STS program and whose stuttering had reduced significantly. Pre-treatment (PRE) and post-treatment (POST) within-clinic audio-visual recordings of conversational speech were analysed for percentage of syllables stuttered, variability of vowel duration, articulation rate, and length and complexity of utterance. Four blinded listeners made perceptual judgments of speech quality in the POST recordings.Result: Recordings of all children showed that variability of vowel duration clearly reduced from the PRE to POST speech samples. Importantly, articulation rate and language use were not compromised. Some possible indicators of rhythmicity were identified in one child in the perceptual study.Conclusion: The findings suggest that STS was primarily responsible for the clinically significant reductions in stuttering after Stage 1 of the program. There is an urgent need for more evidence-based interventions for stuttering in this age group and further development of STS interventions is warranted.

Contemporary issues with stuttering: The Fourth Croatia Stuttering Symposium.

PURPOSE: During the 2019 Fourth Croatia Clinical Symposium, speech-language pathologists (SLPs), scholars, and researchers from 29 countries discussed speech-language pathology and psychological practices for the management of early and persistent stuttering. This paper documents what those at the Symposium considered to be the key contemporary clinical issues for early and persistent stuttering. METHODS: The authors prepared a written record of the discussion of Symposium topics, taking care to ensure that the content of the Symposium was faithfully reproduced in written form. RESULTS: Seven contemporary issues for our field emerged from the Symposium. CONCLUSION: Effective early intervention is fundamental to proper health care for the disorder. However, as yet, there is no consensus about the timing of early intervention and how it should be managed. Currently, clinical translation is a barrier to evidence-based practice with early stuttering, and proactive strategies were suggested for junior SLPs. Apprehension emerged among some discussants that treatment of early stuttering may cause anxiety. For persistent stuttering, assessment procedures were recommended, as were strategies for dealing with childhood bullying. There was agreement that SLPs are the ideal professionals to provide basic cognitive-behavior therapy for clients with persistent stuttering. Questions were raised about our discipline standards for basic professional preparation programs for stuttering management.

Measures of Psychological Impacts of Stuttering in Young School-Age Children: A Systematic Review.

Purpose Recent research has shown that some school-age children who stutter may have speech-related anxiety. Given this, speech-language pathologists require robust measures to assess the psychological effects of stuttering during the school-age years. Accordingly, this systematic review aimed to explore available measures for assessing the psychological impacts of stuttering in young school-age children and to examine their measurement properties. Method The systematic search protocol was registered with PROSPERO (ID: 163181). Seven online databases, in addition to manual searching and screening of reference lists, were used to identify appropriate measures for the population of children who stutter aged 7-12 years. The first two authors independently assessed the measures using the quality appraisal tool described by Terwee et al. (2007). Results Despite the comprehensive search strategy, only six measures were identified for quality appraisal. No assessment tool was found to possess adequate measurement properties for the eight assessed domains: content validity, internal consistency, construct validity, reproducibility, reliability, responsiveness, floor and ceiling effects, and interpretability. No measure had clear evidence of responsiveness to clinical change. Based on the criterion defined by the Terwee et al. (2007) appraisal tool, the Communication Attitude Test and the Overall Assessment of the Speaker's Experience of Stuttering for School-Age Children received the highest number of ratings in support of their measurement properties. Conclusions The results highlight a lack of available measures in this domain and poor practices in developing and testing measurement instruments. To ensure that clinicians and researchers are equipped with sound measures to meet the mental health needs of this vulnerable population, further research to establish resources is needed.

Psychological characteristics of early stuttering.

PURPOSE: The purpose of this study was to use psychological measures of pre-schoolers who stutter and their parents to inform causal theory development and influence clinical practices. This was done using data from a substantive clinical cohort of children who received early stuttering treatment. METHOD: The cohort (N = 427) comprised parents and their children who were treated with the Lidcombe Program, the Westmead Program, and the Oakville Program. The study incorporated demographic information, stuttering severity, and child and parent psychological measures prior to treatment. RESULT: The cohort revealed nothing unusual about behavioural and emotional functioning, or the temperaments, of pre-school children that would influence treatment, be targeted during treatment, or influence causal theory development. However, a third of parents were experiencing moderate to high life stressors at the time of seeking treatment, and half the parents failed first-stage screening for Anankastic Personality Disorder. CONCLUSION: The present results are consistent with a number of previous reports that showed that the population of pre-schoolers who stutter have no unusual psychological profiles. Hence, these results suggest that the association between mental health and stuttering later in life is a consequence of the disorder rather than being a part of its cause. The finding of the life stress of parents who seek stuttering treatment for pre-school children has potential clinical importance and warrants further investigation. Further psychological research is required about parents of pre-school children who stutter, because half the parents in the cohort failed the screener for Anankastic Personality Disorder. This is of interest because a previous study associated screening failure for another personality disorder (Impulsive Personality Disorder) with treatment dropout for early childhood stuttering.

Validity of telephone calls to assess percentage of syllables stuttered with adolescents in clinical research.

PURPOSE: Karimi, O'Brian, Onslow, and Jones (2013) reported, for adults, no systematic differences between percentage of syllables stuttered (%SS) scores during a 12-hour day and 10-minute phone calls. The present study replicated that finding with adolescents, using valid methods for the age group. The present study also extended that initial report by determining whether the gender of the caller influenced %SS scores. METHOD: Participants were 17 adolescents with stuttering. Percentage of syllables stuttered scores were obtained from a 12-hour day of the adolescents' lives, and two 10-minute unscheduled phone calls made before and after that day. One phone call was from a male caller and the other from a female caller. RESULTS: For adolescents, analysis of covariance (ANCOVA) and intraclass correlations (ICC) replicated the overall Karimi, O'Brian, Onslow, and Jones (2013) finding. No significant differences were found between the %SS scores of the three speech samples, and these %SS scores were found to be highly correlated. However, in contrast to the Karimi, O'Brian, Onslow, and Jones (2013) finding with adults, Bland-Altman plot results revealed a caveat to this finding when applied to individual adolescents. Additionally, there was no effect due to the gender of the caller. CONCLUSION: A 10-minute phone call can be used confidently to assess group mean %SS scores during stuttering research with adolescents. However, a 10-minute phone call cannot be used confidently to assess %SS scores of individual adolescent participants. For the latter context, such as with data-based case studies and single-subject experimentation, we recommend supplementing %SS scores with self-reported severity scores.

Speech and Anxiety Management With Persistent Stuttering: Current Status and Essential Research.

Purpose The purpose of this review article is to provide an overview of the current evidence base for the behavioral management of stuttering and associated social anxiety. Method We overview recent research about stuttering and social anxiety in the context of contemporary cognitive models of social anxiety disorder. That emerging evidence for self-focused attention and safety behavior use with those who stutter is considered in relation to current treatment approaches for stuttering: speech restructuring and social anxiety management. Results The emerging information about social anxiety and stuttering suggests a conflict between the two clinical approaches. For those clients who wish to control their stuttering and where speech restructuring is deemed the most suitable approach, it is possible that speech restructuring may (a) induce or increase self-focused attention, (b) promote the use of safety behaviors, and (c) become a safety behavior itself. This conflict needs to be explored further within clinical and research contexts. Conclusions The issues raised in this review article are complex. It appears that evidence-based speech treatment procedures are in conflict with current best-practice treatment procedures that deal with social anxiety. In this review article, we propose directions for future research to inform the development of improved treatments for those who stutter and recommendations for interim clinical management of stuttering.

Natural recovery from stuttering for a clinical cohort of pre-school children who received no treatment.

PURPOSE: The natural stuttering recovery rate by adulthood is high. Community cohort studies suggest a much lower rate during the first 18 months after onset, but this may be different for clinical cohorts of pre-school aged children. The present research and case presentations add to data reported by Franken et al. by investigating early natural recovery for a clinical cohort. METHOD: Participants were 16 pre-school children presenting to a clinic with stuttering onset 1-15 months earlier. The children were studied for a mean of 19.4 months (84.3 weeks) using parent report and clinician identification of stuttering from recorded conversations. The children received no treatment during the study. Data were obtained for each participant and are presented graphically. RESULT: Experienced speech-language pathologists detected stuttering in the recordings of 3 of 4 children identified as recovered by their parents. Only 1 of the 16 children (6.3%) was confirmed as recovered. CONCLUSION: There is no reason to believe that the early natural recovery rate for clinically presenting children is different from community cohorts. Parent report of natural recovery during the pre-school years needs to be confirmed by clinician observation of the child's speech; otherwise, there is risk of harmful false negative identification. The present data support the Yairi et al. different recovery pathways for children who stutter.

Predictors of Lidcombe Program treatment dropout and outcome for early stuttering.

BACKGROUND: Information is available about what predicts Lidcombe Program treatment time, but nothing is known about what predicts treatment prognosis. AIMS: To investigate the predictors of treatment dropout and treatment outcome for children who were treated for early stuttering with the Lidcombe Program (N = 277). METHODS & PROCEDURES: A total of 32 variables were used as predictors in regression analyses of short- and medium-term Lidcombe Program outcome, and of treatment dropout. OUTCOMES & RESULTS: Regression analyses associated children who have better language skills and easy temperament with better treatment outcome, although only a small portion of the variance of treatment outcome was accounted for by these variables. There was an association between treatment dropout and parental scores on a personality screening tool relating to their impulsivity. CONCLUSIONS & IMPLICATIONS: Variables identified as predictors of Lidcombe Program treatment outcome were statistically significant, but not clinically significant. They did not account for a clinically substantive portion of treatment outcomes. Findings about parental impulsivity and their relationship with intervention drop-out require replication with prospective methods and comprehensive assessment of parent psychological status. This is particularly important because parents are involved in conducting all early interventions. What this paper adds What is already known on the subject Information is available about what predicts Lidcombe Program treatment time, but nothing is known about what predicts Lidcombe Program treatment outcome. What this paper adds to existing knowledge There are predictors of Lidcombe Program treatment outcome that are statistically significant, but none are clinically significant. What are the potential or actual clinical implications of this work? Clinicians can tell parents that nothing has been found that can assist with making prognostic indications about treatment outcome for their children.

Verbal Contingencies in the Lidcombe Program: A Noninferiority Trial.

Purpose The Lidcombe Program is an efficacious and effective intervention for early stuttering. The treatment is based on parent verbal response contingent stimulation procedures, which are assumed to be responsible for treatment effect. The present trial tested this assumption. Method The design was a parallel, open plan, noninferiority randomized controlled trial. In the experimental arm, the five Lidcombe Program verbal contingencies were removed from parent instruction. The primary outcome was beyond-clinic percentage syllables stuttered at 18-month follow-up. Seventy-four children and their parents were randomized to one of the two treatment arms. Results Findings of noninferiority were inconclusive for the primary outcome of stuttering severity, based on a margin of 1.0 percentage syllables stuttered. Conclusions The inconclusive finding of noninferiority means it is possible that verbal contingencies make some contribution to the Lidcombe Program treatment effect. However, considering all primary and secondary outcomes, an overriding impression from the trial is a similarity of outcomes between the control and experimental arms. The clinical applications of the trial are discussed, along with further research that is needed.

Clinical Trials of Adult Stuttering Treatment: Comparison of Percentage Syllables Stuttered With Self-Reported Stuttering Severity as Primary Outcomes.

Purpose In a companion paper, we found no statistical reason to favor percentage syllables stuttered (%SS) over parent-reported stuttering severity as a primary outcome measure for clinical trials of early stuttering. Hence, considering the logistical advantages of the latter measure, we recommended parent-reported stuttering severity for use as an outcome measure. The present report extends the prior analysis to a comparison of %SS with self-reported stuttering severity (SRSS) for use as an outcome measure in clinical trials of stuttering treatments for adults. Method We analyzed data from four randomized clinical trials for adults that incorporated %SS and SRSS data at prerandomization and at 6 months post randomization. We analyzed the distributions associated with the two measures, their agreement, and their estimates of effect sizes. Results The positively skewed distribution of %SS warrants much reservation about its value as a clinical trial outcome measure. This skew causes inherent instability because of spurious data associated with low scores, which occur commonly at the low end of such a distribution. This inherent instability is compounded by inherent problems with absolute reliability of %SS measures. These problems are reduced with the much more normal distribution of SRSS. Conclusions The logistical arguments in favor of SRSS apply similarly to adults as they do when parents report the stuttering severity of their children. However, there are statistical reasons to favor SRSS over %SS measures as a primary outcome of clinical trials with adult participants: SRSS has acceptable discriminant validity and a normal distribution, and it is less error prone than %SS. We recommend SRSS as a primary outcome for clinical trials of adults with stuttering.

Phase I trial of a standalone internet social anxiety treatment for adolescents who stutter: iBroadway.

BACKGROUND: iGlebe is a fully automated internet treatment program for adults who stutter that has been shown, in some cases, to reduce anxiety and effectively manage social anxiety disorder for many participants. No such automated internet treatment program exists for adolescents who stutter. AIMS: The present paper reports a Phase I trial of an adolescent version of the adult program: iBroadway. METHODS & PROCEDURES: Participants were 29 adolescents in the age range 12-17 years who were seeking cognitive-behaviour therapy (CBT) for anxiety associated with stuttering. The design was a non-randomized Phase I trial with outcome assessments at pre-treatment and immediately post-treatment after 5 months of access to the program. No contact by a clinical psychologist occurred during participant use of the program. Outcomes were a range of psychological, quality-of-life and stuttering severity measures. OUTCOMES & RESULTS: The compliance rate for the seven iBroadway modules over 5 months was extremely favourable for internet CBT, at 52.4%. There was evidence of treatment effects for (1) the number of DSM-IV mental health diagnoses with the Diagnostic Interview Schedule for Children; (2) the Unhelpful Thoughts and Beliefs About Stuttering scale; (3) the Subjective Units of Distress Scale; and (4) parent-reported speech satisfaction. CONCLUSIONS & IMPLICATIONS: Further development of iBroadway, the adolescent version of iGlebe, with Phase II trialling is warranted.

Three Lidcombe program clinic visit options: a phase II trial.

PURPOSE: Weekly clinic visits are recommended in the Lidcombe Program Treatment Guide (Packman et al., 2015). That specification is based on traditional speech-language pathology practices rather than empirical research, and two studies have suggested that such a format does not always occur in clinical communities. This research was conducted to determine the relative efficacy of different Lidcombe Program models of clinic visits. METHOD: Thirty-one children were randomized to three different service delivery models: twice-weekly, weekly and fortnightly (once every two weeks) clinic visits. All children were treated with the Lidcombe Program following manualised procedures. Measures of percentage syllables stuttered were obtained from beyond clinic audio recordings pre- and post-randomization. RESULTS: Results showed that the twice-weekly and fortnightly treatment formats were not suitable for all families. However, the fortnightly outcomes at 9 months post-randomization were comparable with those attained during weekly clinic visits. CONCLUSIONS: These results justify further, large-scale clinical trialling to compare weekly Lidcombe Program clinic visits with schedules involving less frequent clinic visits.