The Association Between Stuttering Burden and Psychosocial Aspects of Life in Adults.

PURPOSE: Stuttering is a speech condition that can have a major impact on a person's quality of life. This descriptive study aimed to identify subgroups of people who stutter (PWS) based on stuttering burden and to investigate differences between these subgroups on psychosocial aspects of life. METHOD: The study included 618 adult participants who stutter. They completed a detailed survey examining stuttering symptomatology, impact of stuttering on anxiety, education and employment, experience of stuttering, and levels of depression, anxiety, and stress. A two-step cluster analytic procedure was performed to identify subgroups of PWS, based on self-report of stuttering frequency, severity, affect, and anxiety, four measures that together inform about stuttering burden. RESULTS: We identified a high- (n = 230) and a low-burden subgroup (n = 372). The high-burden subgroup reported a significantly higher impact of stuttering on education and employment, and higher levels of general depression, anxiety, stress, and overall impact of stuttering. These participants also reported that they trialed more different stuttering therapies than those with lower burden. CONCLUSIONS: Our results emphasize the need to be attentive to the diverse experiences and needs of PWS, rather than treating them as a homogeneous group. Our findings also stress the importance of personalized therapeutic strategies for individuals with stuttering, considering all aspects that could influence their stuttering burden. People with high-burden stuttering might, for example, have a higher need for psychological therapy to reduce stuttering-related anxiety. People with less emotional reactions but severe speech distortions may also have a moderate to high burden, but they may have a higher need for speech techniques to communicate with more ease. Future research should give more insights into the therapeutic needs of people highly burdened by their stuttering. SUPPLEMENTAL MATERIAL: https://doi.org/10.23641/asha.25582980.

Early Speech Sound Production and Its Trajectories in Very Preterm Children From 2 to 4 Years of Age.

PURPOSE: Very preterm (VPT) children are at risk for speech and language problems throughout school age. However, little is known about early speech sound production in these children. This study aims to present a detailed description of early speech sound production and its trajectories in VPT children from 2 to 4 years of age. In addition, this study aimed to determine if early speech sound production is associated with speech production and expressive language function at 4 years of age. METHOD: In 63 VPT children (< 32 weeks of gestation, 41 boys, mean gestational age = 28.8 weeks, mean birth weight = 1,135 g), speech sound production was assessed by naturalistic speech analysis at 2 years of corrected age and speech and language function by standardized tests at 4 years of age. RESULTS: Speech sound production was found to be abnormal in 49% of the VPT children at 2 years of age and in 19% at 4 years of age. Four different speech production trajectories from 2 to 4 years of age could be identified: a normal trajectory, an abnormal trajectory, a catch-up trajectory, and a growing-into-deficit trajectory. Early speech production, defined by the number of acquired consonants at 2 years of age, significantly predicted the word production score at 4 years of age and the sentence production score at 4 years of age. CONCLUSIONS: Compared to the general population, an alarmingly high proportion of VPT children showed speech production problems at 2 years of age. About half of these children showed persistent speech problems at 4 years of age. Moreover, these problems were associated with expressive language problems at the age of 4 years. SUPPLEMENTAL MATERIAL: https://doi.org/10.23641/asha.19310822.

Language lateralization in very preterm children: associating dichotic listening to interhemispheric connectivity and language performance.

BACKGROUND: Language difficulties of very preterm (VPT) children might be related to weaker cerebral hemispheric lateralization of language. Language lateralization refers to the development of an expert region for language processing in the left hemisphere during the first years of life. Children born VPT might not develop such a dominant left hemisphere for language processing. A dichotic listening task may be a functional task to show the dominance of the left hemisphere during language processing. During this task, different acoustic events are simultaneously presented to both ears. Due to crossing fibers in the brain, right ear stimuli are transferred directly to the left hemisphere, and left ear stimuli are transferred first to the right hemisphere and then, through the corpus callosum (CC), to the left hemisphere. Dichotic listening typically shows a right ear advantage, assuming to reflect left hemispherical language dominance. The CC, in particular the splenium, is associated with auditory processing and is considered important for language lateralization. The objective of this work was to explore whether dichotic listening performance in school-aged VPT children are associated with language performance and interhemispheric connectivity. METHODS: This is a cross-sectional study of 58 VPT children and 30 full term controls at age 10 years. Language performance and dichotic digit test (DDT) were assessed. In 44 VPT children, additionally diffusion weighted imaging (DWI) was performed using a 3 T MRI scanner. Fractional anisotropy (FA) and mean diffusivity (MD) values of the splenium of the CC were extracted. RESULTS: Poorer right ear DDT scores were associated with poorer language performance in VPT children only (p = 0.015). Association between right ear DDT scores and MD of the splenium approached the level of significance (p = 0.051). CONCLUSIONS: These results support the hypothesis that poor language performance in VPT children may be a consequence of weaker lateralized language organization, due to a poorly developed splenium of the CC. Dichotic listening may reflect the level of language lateralization in VPT children. IMPACT: Poor language performance in VPT children may be a consequence of weaker lateralized language organization, due to a poorly developed splenium of the CC. Dichotic listening performance may reflect the level of language lateralization in VPT children and right ear scores of a dichotic listening task are associated with both the splenium of the corpus callosum and language performance. If our results could be validated in future research, it suggests that poor CC development may indicate VPT children at risk for long-term language problems.

[Formula: see text]Are multidisciplinary neurodevelopmental profiles of children born very preterm at age 2 relevant to their long-term development? A preliminary study.

To identify distinctive multidisciplinary neurodevelopmental profiles of relatively healthy children born very preterm (VPT) and describe the longitudinal course of these profiles up to age 10. At 2 years of corrected age, 84 children born VPT underwent standardized testing for cognitive, language, speech, motor, behavioral, and auditory nerve function. These data were submitted to factor and cluster analysis. Sixty-one of these children underwent cognitive, language, and behavioral assessment again at age 10. Descriptive statistics were used to analyze longitudinal trajectories for each profile. Four neurodevelopmental profiles were identified at age 2. Profile 1 children (n = 22/26%) had excellent cognitive-language-motor function, normal behavioral and auditory nerve function, but showed an unexpected severe decline up to age 10. Profile 2 children (n = 16/19%) had very low behavioral function, low cognitive-language-motor function, and accelerated auditory nerve function. Their scores remained low up until age 10. Profile 3 children (n = 17/20%) had delayed auditory nerve function, low behavioral function, and slightly lower cognitive-language-motor function. They showed the most increasing trajectory. Profile 4 children (n = 29/35%) had very low cognitive-language-motor function, normal behavioral and auditory nerve function, but showed wide variation in their trajectory. Our preliminary study showed that a multidisciplinary profile-oriented approach may be important in children born VPT to improve counseling and provide targeted treatment for at risk children. High performers at age 2 may not be expected to maintain their favorable development. Behavioral problems might negatively impact language development. Delayed auditory nerve function might represent a slow start and catch-up development.

Validity of an early parent-report questionnaire for language disorder in very preterm children from 2 to 10 years of age.

BACKGROUND: Language problems at an early age in very preterm (VP) children can have a detrimental effect on other developmental domains and often persist throughout childhood. The aim of this study was to examine the concurrent and predictive validity of an early language parent-report questionnaire for language disorder in VP children from 2 to 10 years of age. MATERIALS AND METHODS: In 80 VP children (<32 weeks' gestation) without major disabilities, a parent-questionnaire and formal language assessment, both normed for the general population, were administered at 2 years corrected age (CA). Of these infants, 62 were seen for follow-up formal language assessment at age 4 and 61 were seen at age 10. Sensitivity and specificity values were calculated. RESULTS: The Lexi-list showed acceptable concurrent validity for word production scores obtained at age 2 CA. The predictive validity was good for sentence production and acceptable for word production scores obtained at age 4, and low for language production scores obtained at age 10. A Lexi-list cut-off score of <85 (i.e., <-1 SD) was found optimal. INTERPRETATION: A norm-referenced parent-report questionnaire is a useful, first screening tool in a neonatal follow-up. It not only detected early language disorder at age 2 CA but also proved to be a good predictor for language disorder at age 4. However, it did not predict language disorder at age 10. Formal language assessment at age 4 would therefore be recommended for children with an abnormal parent-report language score at age 2 CA.

Cerebellar volumes and language functions in school-aged children born very preterm.

BACKGROUND: Volumes of cerebellar posterior lobes have been associated with cognitive skills, such as language functioning. Children born very preterm (VPT) often have language problems. However, only total cerebellar volume has been associated with language functioning, with contradicting results. The objective of this study was to ascertain whether total cerebellar structures or specific posterior lobular structures are associated with language ability of school-aged VPT children. METHODS: This is a prospective cohort study of 42 school-aged VPT children without major handicaps. Structural MRI was performed and the cerebellum segmentation pipeline was used for segmentation of separate lobules. Narrative retelling assessment was performed and language content and language structure scores were extracted. Linear regression analyses were used to associate language scores with whole gray matter (GM) cerebellar volume and right Crus I+II GM volume. RESULTS: Whole cerebellar GM volume was not significantly associated with language content nor with language structure; however, right Crus I+II GM volume was significantly associated with language content (ß = 0.192 (CI = 0.033, 0.351), p = 0.020). CONCLUSIONS: GM volume of Crus I+II appears to be associated with language functions in school-aged VPT children without major handicaps, while whole cerebellar volume is not. This study showed the importance of studying cerebellar lobules separately, rather than whole cerebellar volume only, in relation to VPT children's language functions. IMPACT: GM volume of Crus I+II is associated with semantic language functions in school-aged very preterm children without overt brain injury, whereas whole cerebellar volume is not. This study showed the importance of studying cerebellar lobules separately, rather than whole cerebellar volume only, in relation to very preterm children's language functions. This study might impact future research in very preterm children. Lobular structures rather than whole cerebellar structures should be the region of interest in relation to language functions.

Does a narrative retelling task improve the assessment of language proficiency in school-aged children born very preterm?

Almost half of the children born very preterm (VP) experience language difficulties at school-age, specifically with more complex language tasks. Narrative retelling is such a task. Therefore, we explored the value of narrative retelling assessment in school-aged children born VP, compared to item-based language assessment. In 63 children born VP and 30 age-matched full-term (FT) controls Renfrew's Bus Story Test and Clinical Evaluation of Language Fundamentals were assessed. The retelling of the Bus Story was transcribed and language complexity and content measures were analyzed with Computerised Language Analysis software. Narrative outcomes of the VP group were worse than that of the FT group. Group differences were significant for the language complexity measures, but not for the language content measures. However, the mean narrative composite score of the VP group was significantly better than their mean item-based language score, while in the FT group the narrative score was worse than the item-based score. Significant positive correlations between narrative and item-based language scores were found only in the VP group. In conclusion, in VP children narrative retelling appears to be less sensitive to detecting academic language problems than item-based language assessment. This might be related to the mediating role of attention in item-based tasks, that appears not to affect more spontaneous language tasks such as retelling. Therefore, in school-aged children born VP we recommend using narrative assessment, in addition to item-based assessments, because it is more related to spontaneous language and less sensitive to attention problems.

Language functions deserve more attention in follow-up of children born very preterm.

BACKGROUND: Language is a complex neurodevelopmental phenomenon. Approximately 45% of children born very preterm (VP) show mild-to-severe language problems throughout childhood. Nevertheless, in most hospitals in Europe language functions are not routinely assessed at follow-up. OBJECTIVE: To give clear indications for extensive language assessment in school-aged children born VP, based on routinely assessed intelligence and behavioral problems. METHOD: Language functions of 63 10-year-old children born VP (<32 weeks' gestation) without major handicaps were compared to their intellectual and executive functions and behavioral problems. Using multiple linear regression analyses, the predictive value of perinatal factors and the association with neurodevelopmental factors of low language were measured. RESULTS: The mean language score was significantly lower than the verbal intelligent quotient (VIQ; mean difference = 6.4, p < .001, d=.48) and the mean vocabulary knowledge (mean difference = 9.3, p < .001, d=.70). Besides, VIQ (ß = .649, p = .001) and performance IQ (PIQ; ß = .260, p = .035) were significantly associated with language scores. Significant predictors of language scores were number of days of assisted ventilation (ß = -.592, p = .015) and mother's vocabulary knowledge (ß =.473, p = .014), rather than mother's educational level (ß =.139, p = .956). CONCLUSIONS: Children born VP had language problems that were not expected from their significantly higher VIQ and vocabulary knowledge. Clinicians assessing these children should be aware of possible language problems, which cannot be detected with a simple vocabulary task. Our findings provide evidence of the need for adequate language assessments by a speech-language pathologist in children born VP, especially in those with VIQ scores in the low average range.

Association of Slight to Mild Hearing Loss With Behavioral Problems and School Performance in Children.

Importance: Children with severe hearing loss are known to have more behavioral problems and may perform worse at school than children without. Few large-scale studies of slight to mild hearing loss are available. Objective: To examine the relevance of slight to mild hearing loss by studying its association with behavioral problems and school performance. Design, Setting, and Participants: This cross-sectional study was performed within an ongoing prospective birth cohort study in Rotterdam, the Netherlands. Participants were part of a population-based sample of children. Between ages 9 and 11 years, 5355 children underwent audiometric and behavioral evaluations. Children were excluded if they had missing data for either audiometry or both outcomes. Data were collected from April 2012 through October 2015. Data were analyzed from March to June 2018. Exposures: Audiometric evaluation included pure-tone audiometry tests and speech-in-noise testing. Main Outcomes and Measures: Child behavior was rated by the primary caregiver using the Child Behavior Checklist at ages 9 to 11 years (n = 4471). School performance was measured with a standardized test at age 12 years (n = 2399). Results: The final sample included 4779 participants who were a mean (SD) age of 9.8 (0.3) years. The sample had nearly equal distribution between boys (n = 2200; 49.2%) and girls (n = 2271; 50.8%). Associations of hearing thresholds with behavioral problems differed between boys and girls. Among boys, higher pure-tone hearing thresholds at low frequencies were associated with higher total problem, social problem, and attention problem scores (total problems for the better-hearing ear: ß = 0.01; 95% CI, 0-0.02). Higher speech reception thresholds were associated with higher attention problem scores among girls (ß = 0.04; 95% CI, 0-0.08). Higher speech reception thresholds were associated with poorer school performance scores for both boys and girls (ß = -0.06; 95% CI, -0.10 to -0.02). Conclusions and Relevance: Higher hearing thresholds during pure-tone audiometric and speech-in-noise testing were associated with higher behavioral problem scores and poorer school performance. This supports the relevance of slight to mild hearing loss with these outcomes in school-aged children.

"Spontaneous" late recovery from stuttering: Dimensions of reported techniques and causal attributions.

PURPOSE: (1) To survey the employed techniques and the reasons/occasions which adults who had recovered from stuttering after age 11 without previous treatment reported as causal to overcome stuttering, (2) to investigate whether the techniques and causal attributions can be reduced to coherent (inherently consistent) dimensions, and (3) whether these dimensions reflect common therapy components. METHODS: 124 recovered persons from 8 countries responded by SurveyMonkey or paper-and-pencil to rating scale questions about 49 possible techniques and 15 causal attributions. RESULTS: A Principal Component Analysis of 110 questionnaires identified 6 components (dimensions) for self-assisted techniques (Speech Restructuring; Relaxed/Monitored Speech; Elocution; Stage Performance; Sought Speech Demands; Reassurance; 63.7% variance explained), and 3 components of perceived causal attributions of recovery (Life Change, Attitude Change, Social Support; 58.0% variance explained). DISCUSSION: Two components for self-assisted techniques (Speech Restructuring; Elocution) reflect treatment methods. Another component (Relaxed/Monitored Speech) consists mainly of items that reflect a common, non-professional understanding of effective management of stuttering. The components of the various perceived reasons for recovery reflect differing implicit theories of causes for recovery from stuttering. These theories are considered susceptible to various biases. This identification of components of reported techniques and of causal attributions is novel compared to previous studies who just list techniques and attributions. CONCLUSION: The identified dimensions of self-assisted techniques and causal attributions to reduce stuttering as extracted from self-reports of a large, international sample of recovered formerly stuttering adults may guide the application of behavioral stuttering therapies.

Recovery from stuttering in preschool-age children: 9 year outcomes in a clinical population.

PURPOSE: The first purpose was to define the recovery rate in children who stutter in a clinical sample, adding self-report to validate recovery status. The second purpose was to explore whether children who were judged to be recovered showed subjective experiences that might be interpreted as coping behaviors used to control speech fluency. METHODS: In this longitudinal study, preschool-age children whose parents consulted a speech-language pathologist about stuttering were followed for 9 years. At follow-up, children's self-reports on stuttering were reported, as well as traditional criteria on recovery (parental and expert judgment). The Overall Assessment of the Speaker's Experience of Stuttering (OASES) was used to collect children's experiences with speaking. RESULTS: Eleven of the 15 children (73%) were judged by parents and clinicians to have recovered from stuttering. However, when considering children's self-reports, 9 children (60%) might be considered to have recovered. In addition, 3 children who were judged to be recovered reported experiences with speaking that were uncommon, even compared to children who continued to stutter. CONCLUSION: In this exploratory study of a clinical population, the recovery rate in children that received treatment for stuttering appeared to be comparable to a non-clinical population. Considering self-reports can improve validity of assessing the "recovery rate". Moreover, recovery in children may not be effortless; instead, it may be the result of conscious or unconscious coping behavior. Future studies are recommended to consider self-reports to improve validity of recovery, and to document experiences with speaking to explore effortless, spontaneous fluency versus controlled fluency.

Correction: Language outcome related to brain structures in school-aged preterm children: A systematic review.

[This corrects the article DOI: 10.1371/journal.pone.0196607.].

Language outcome related to brain structures in school-aged preterm children: A systematic review.

Preterm children often have language problems. This atypical language development is probably due to atypical brain development. We conducted a systematic review to provide an overview of the extensive and diverse scientific literature on the relations between language outcome and underlying brain structures in school-aged preterm-born children. Embase, Medline Ovid, Web of Science, Cochrane central and Google scholar were searched for relevant studies. Inclusion criteria were: cases are school-aged preterm children; structural MRI (T1- and T2-weighted sequences) or DTI used in combination with a neurocognitive language test; publication in an English-language peer-reviewed journal. Correlational measures between language scores and brain volume or fractional anisotropy of a brain structure were extracted. 23 studies were included. The relations between oral language, verbal fluency and/or written language and MRI/DTI measurements of white matter, gray matter, cerebellum, corpus callosum and/or the fasciculi are presented. Oral language skills and verbal fluency appear to be related to the corpus callosum. Oral language skills are also related to the uncinate fasciculus. There seems to be no clear relation between cerebellar development and verbal fluency skills. Not one single brain area is responsible for atypical language development, but several brain areas and their connections are essential. For future research it is recommended to relate brain areas to oral language skills on a microstructural level in preterm children. We also recommend to use language tests in which it is possible to distinguish between several language domains, such as perceptive and expressive language.

Normal neonatal hearing screening did not preclude sensorineural hearing loss in two-year-old very preterm infants.

AIM: Very preterm infants are at risk of neonatal hearing loss. However, it is unknown whether infants with a normal neonatal hearing screening result risk sensorineural hearing loss (SNHL) at a later age. METHODS: This cohort study was conducted at the Erasmus Medical University Center Rotterdam, the Netherlands, on 77 very preterm infants born between October 2005 and September 2008. All infants underwent auditory brainstem response audiometry during neonatal hearing screening and at two years of corrected age. The frequency of SNHL in infants with a normal neonatal hearing screening was analysed and the risk factors associated with newly diagnosed SNHL in these infants were examined. RESULTS: We found that 3.9% (3/77) of the very preterm infants showed permanent hearing loss during their neonatal hearing screening. In addition, a relatively high prevalence of newly diagnosed SNHL (4.3%) was found in three of the 70 infants followed up at the age of two. The total prevalence rate of permanent hearing loss in the cohort was approximately 8%. CONCLUSION: A normal outcome of neonatal hearing screening did not guarantee normal hearing at two years of age in this very preterm cohort and paediatricians should be alert to the possibility of late-onset SNHL.

Development of emotional and behavioral problems in neurofibromatosis type 1 during young childhood.

This retrospective longitudinal study in young children with neurofibromatosis type 1 (NF1) aimed to identify if, and how early problems in behavior, intelligence, and language development are associated with later behavioral problems. At the first assessment at preschool age, we evaluated language skills, intelligence, and emotional and behavioral problems as reported by parents. The second assessment at school-age we evaluated intelligence, and emotional and behavioral problems as reported by parents and teachers. Association of baseline assessments with secondary assessment was evaluated using multivariable linear regression analysis. Of the 61 patients (25 males, 36 females; mean age 4;5 years [SD 1;1 years]) with NF1 who had a first assessment, 38 children (21 males, 17 females; mean age 7;11 years [SD 2;1 years]) had a second assessment after a mean period of 3;5 years. Longitudinal data on behavioral problems were collected for 23 of these children. Intelligence and language development were not associated with internalizing problems. Parent-rated internalizing behavioral problems significantly increased with age in this subgroup. Baseline internalizing problems predicted later internalizing problems (adjusted R2 = 0.33, p = 0.003). The presence of these problems at pre-school age may be predictive of internalizing problems at a later age.

Language functions in preterm-born children: a systematic review and meta-analysis.

BACKGROUND AND OBJECTIVE: Preterm-born children (<37 weeks' gestation) have higher rates of language function problems compared with term-born children. It is unknown whether these problems decrease, deteriorate, or remain stable over time. The goal of this research was to determine the developmental course of language functions in preterm-born children from 3 to 12 years of age. METHODS: Computerized databases Embase, PubMed, Web of Knowledge, and PsycInfo were searched for studies published between January 1995 and March 2011 reporting language functions in preterm-born children. Outcome measures were simple language function assessed by using the Peabody Picture Vocabulary Test and complex language function assessed by using the Clinical Evaluation of Language Fundamentals. Pooled effect sizes (in terms of Cohen's d) and 95% confidence intervals (CI) for simple and complex language functions were calculated by using random-effects models. Meta-regression was conducted with mean difference of effect size as the outcome variable and assessment age as the explanatory variable. RESULTS: Preterm-born children scored significantly lower compared with term-born children on simple (d = -0.45 [95% CI: -0.59 to -0.30]; P < .001) and on complex (d = -0.62 [95% CI: -0.82 to -0.43]; P < .001) language function tests, even in the absence of major disabilities and independent of social economic status. For complex language function (but not for simple language function), group differences between preterm- and term-born children increased significantly from 3 to 12 years of age (slope = -0.05; P = .03). CONCLUSIONS: While growing up, preterm-born children have increasing difficulties with complex language function.

Phonological development in very-low-birthweight children: an exploratory study.

AIM: Very-low-birthweight (VLBW; birthweight<1500 g and/or gestational age <32 wks) children are at risk for speech problems. However, there are few studies on speech development in VLBW children at an early age. The aim of this study was to investigate phonological development in 2-year-old VLBW children. METHOD: Twenty VLBW children without major neurosensory impairment (7 males, 13 females; mean birthweight 971 g, SD 315; mean gestational age 28 wks, SD 1.81) and 20 term children (7 males, 13 females; mean birthweight 3503 g, SD 416; mean gestational age 40 wks, SD 1.26) were compared on measures of phonological development derived from 20-minute spontaneous speech samples of standardized mother-child play interaction as well as on standardized tests of cognitive and psychomotor development, language, and behaviour. RESULTS: VLBW children had significantly fewer acquired consonants (median 9, p=0.02) and a significantly lower phonological mean length of utterance (pMLU; median 4.1, p<0.01) than term children (median acquired consonants 10, median pMLU 5.0). INTERPRETATION: This study provides evidence for poor phonological development in even healthy VLBW children, compared with term-matched children, independent of their cognitive, psychomotor, and language development, and their behavioural functioning.