Non‐pharmacological interventions for stuttering in children six years and younger

Background Stuttering, or stammering as it is referred to in some countries, affects a child's ability to speak fluently. It is a common communication disorder, affecting 11% of children by four years of age. Stuttering can be characterized by sound, part word or whole word repetitions, sound prolongations, or blocking of sounds or airflow. Moments of stuttering can also be accompanied by non‐verbal behaviours, including visible tension in the speaker's face, eye blinks or head nods. Stuttering can also negatively affect behavioural, social and emotional functioning. Objectives Primary objective To assess the immediate and long‐term effects of non‐pharmacological interventions for stuttering on speech outcomes, communication attitudes, quality of life and potential adverse effects in children aged six years and younger. Secondary objective To describe the relationship between intervention effects and participant characteristics (i.e. child age, IQ, severity, sex and time since stuttering onset) at pretest. Search methods We searched CENTRAL, MEDLINE, Embase, PsycINFO, nine other databases and two trial registers on 16 September 2020, and Open Grey on 20 October 2020. There were no limits in regards to language, year of publication or type of publication. We also searched the reference lists of included studies and requested data on unpublished trials from authors of published studies. We handsearched conference proceedings and programmes from relevant conferences. Selection criteria We included randomized controlled trials (RCTs) and quasi‐RCTs that assessed non‐pharmacological interventions for stuttering in young children aged six years and younger. Eligible comparators were no intervention, wait list or management as usual. Data collection and analysis We used standard methodological procedures expected by Cochrane. Main results We identified four eligible RCTs, all of which compared the Lidcombe Program to a wait‐list control group. In total, 151 children aged between two and six years participated in the four included studies. In the Lidcombe Program, the parent and their child visit a speech and language therapist (SLT) in a clinic. One study conducted clinic visits by telephone. In each clinic visit, parents were taught how to conduct treatment at home. Two studies took place in Australia, one in New Zealand and one in Germany. Two studies were conducted for nine months, one for 16 weeks and one for 12 weeks. The frequency of clinic visits and practice sessions at home varied within the programme. One study was partially funded by the Rotary Club, Wiesbaden, Germany; and one was funded by the National Health and Medical Research Council of Australia. One study did not report funding sources and another reported that they did not receive any funding for the trial. All four studies reported the outcome of stuttering frequency. One study also reported on speech efficiency, defined as articulation rate. No studies reported the other predetermined outcomes of this review, namely stuttering severity; communication attitudes; emotional, cognitive or psychosocial domains; or adverse effects. The Lidcombe Program resulted in a lower stuttering frequency percentage syllables stuttered (% SS) than a wait‐list control group at post‐test, 12 weeks, 16 weeks and nine months postrandomization (mean difference (MD) −2.16, 95% confidence interval (CI) −3.48 to −0.84, 4 studies, 151 participants; P = 0.001; very low‐certainty evidence). However, as the Lidcombe Program is designed to take one to two years to complete, none of the participants in these studies had finished the complete intervention programme at any of the data collection points. We assessed stuttering frequency to have a high risk of overall bias due to high risk of bias in at least one domain within three of four included studies, and to have some concern of overall bias in the fourth, due to some concern in at least one domain. We found moderate‐certainty evidence from one study showing that the Lidcombe Program may i crease speech efficiency in young children. Only one study reported outcomes at long‐term follow‐up. The long‐term effect of intervention could not be summarized, as the results for most of the children in the control group were missing. However, a within‐group comparison was performed between the mean % SS at randomization and the mean % SS at the time of extended follow‐up, and showed a significant reduction in frequency of stuttering. Authors' conclusions This systematic review indicates that the Lidcombe Program may result in lower stuttering frequency and higher speech efficiency than a wait‐list control group in children aged up to six years at post‐test. However, these results should be interpreted with caution due to the very low and moderate certainty of the evidence and the high risk of bias identified in the included studies. Thus, there is a need for further studies from independent researchers, to evaluate the immediate and long‐term effects of other non‐pharmacological interventions for stuttering compared to no intervention or a wait‐list control group. Plain language summary Medicine‐free treatment for stuttering in children aged six years and younger What was the aim of this review? The aim of this review was to find out if medicine‐free treatment for stuttering can improve speech fluency, children's communication attitudes and the impact on the child's quality of life, and potential harmful effects in children aged six years and younger, both in the short‐ and long‐term. We collected and analyzed all relevant studies to answer this question and found four studies. Key messages The Lidcombe Program may result in lower stuttering frequency and higher speech efficiency (i.e. number of words or syllables spoken per minute) for young children after receiving the amount of treatment included in the studies. We do not yet know the impact of the programme delivered in its entirety, as no study reported outcomes for children who had completed the programme, which is designed to last for one to two years. Only one study reported how treatment worked in the long run, but the effect of treatment could not be summarized, as the results for most children in the control group were missing. We require more high‐quality studies assessing stuttering treatments for young children, including studies that report on a broader range of outcomes and that assess treatments other than the Lidcombe Program. What did the review study? Stuttering, or stammering as it is sometimes called, is a common communication disorder, that usually begins when children are between two and four years of age. Stuttering can be characterized by repetitions of individual speech sounds, parts of words or whole words; involuntary lengthening of speech sounds; or speech blockages. Moments of stuttering can also be accompanied by visible tension in the speaker's face or voice, eye blinks or head nods. Stuttering can have a negative effect on how people feel about themselves and how they live their lives. Therefore, it is important to identify effective treatments for stuttering in young children to reduce the chance that they will experience these negative effects. What were the main results of the review? We found four studies, all of which compared young children who received the Lidcombe Program to young children on a wait‐list. In three studies, the children on the wait‐list did not receive any treatment until after the study was finished. In the fourth, the children on the wait‐list could receive treatment from their local speech language therapist (SLT) if they wanted to. At the end of the study, parents of seven children (35%) in the control group reported their child had received some treatment, either the Lidcombe Program or another treatment programme called "Easy Does It", while on the wait‐list. There were 151 children aged between two and six years in the four studies. In the Lidcombe Program, an SLT conducted clinic visits with the child and parent in person in a clinic or spoke to them by telephone During these visits, parents were taught to conduct treatment at home during 10‐ to 15‐minute daily practice sessions. Two studies were conducted in Australia, one in New Zealand and one in Germany. Two studies were conducted for nine months, one for 16 weeks and one for 12 weeks. One study was partially funded by the Rotary Club, Wiesbaden, Germany; and one was funded by the National Health and Medical Research Council of Australia. One study did not report any funding sources and another reported that they did not receive any funding for the trial. All four studies reported the effects of treatment on stuttering frequency. One study also reported on speech efficiency. We found no studies that looked at the effects of treatments for stuttering on stuttering severity; communication attitudes; emotional (how a child recognizes, expresses, and manages feelings), cognitive (how a child thinks, explores and works problems out) or psychosocial (how a child's individual needs link to the needs or demands of society) development; or side effects. The included studies suggest that the Lidcombe Program may reduce stuttering frequency in young children compared to a wait‐list control group (very low‐quality evidence). One study also reported that the Lidcombe Program may increase speech efficiency in young children compared to a wait‐list control group (moderate‐quality evidence). Only one study followed the children up to five years after treatment started, and at that time the results for most of the children in the control group were missing. Therefore, we do not know if the benefits of the treatment lasted over time. How up‐to‐date was this review? We searched for studies that had been published up to 16 September 2020. One additional database was searched on 20 October 2020. How reliable was the evidence generated by this review? The quality of the results related to stuttering frequency was very low and the results related to speech efficiency was moderate. This means that we are uncertain that the treatment effect would stay the same if we added more studies to the review. More studies comparing different treatments for stuttering to a wait‐list control group are needed to know the effect of treatment for stuttering with greater certainty.