ANNOTATION Management of childhood stuttering Mark Onslow and Sue O’Brian Australian Stuttering Research Centre, The University of Sydney, Sydney, New South Wales, Australia Abstract: Stuttering is a speech disorder that begins during the first years of life and is among the most prevalent of developmental disorders. It appears to be a problem with neural processing of speech involving genetics. Onset typically occurs during the first years of life, shortly after language development begins. Clinical presentation during childhood is interrupted and effortful speech production, often with rapid onset. If not corrected during early childhood, it becomes intractable and can cause psychological, social, educational and occupational problems. There is evidence from replicated clinical trials to support early intervention during the pre-school years. Meta-analysis of studies indicates that children who receive early intervention during the pre-school years are 7.7 times more likely to have resolution of their stuttering. Early intervention is recommended with a speech pathologist. Some children who begin to stutter will recover without such intervention. However, the number of such recoveries is currently not known, and it is not possible to predict which children are likely to recover naturally. Consequently, the current best practice is for speech pathologists to monitor children for signs of natural recovery for up to 1 year before beginning treatment. Key words: diagnosis; management; paediatrics; stuttering; treatment. Stuttering Stuttering, also known as stammering in the United Kingdom, is a speech disorder that begins during the first years of life. A recent community cohort study of 1619 Australian children recruited at 8 months old found that 8.5% had begun to stutter by 3 years of age. 1 The shape of the cumulative inci- dence plot suggests that more cases will emerge as the cohort is studied further. Onset was found to be essentially unpre- dictable, with only 3.7% of cases explainable with case history variables such as advanced language development, twinning and maternal education level. A report of 3 to 17-year-olds derived from the United States National Health Interview Surveys (n = 95,132) showed stuttering to be the equal third most prevalent developmental disorder from among nine, which included attention-deficit/hyperactivity disorder, autism, cerebral palsy and learning disability. 2 The reported prevalence was 1.6%. The cause of stuttering is currently unknown; however, brain imaging data suggest that it involves a problem with neural processing of speech, 3 linked to structural and functional anomalies at brain sites responsible for spoken language. 4,5 As these anomalies have only been investigated with school chil- dren and adults, it is unclear whether they are a cause or an effect of the disorder. There is genetic involvement in stuttering, with clear evidence of vertical transmission within families. 6 Around two thirds of those affected, or their parents, report a family history. There is greater monozygotic concordance than dizygotic concordance, which offers a genetic account of around 70% of cases. 7 At present, genetic linkage studies can account for less than one tenth of cases; however, exome sequencing technology has yet to be applied to the study of the disorder. The clinical presentation of chronic stuttering in adolescence and adulthood is interrupted speech production. Symptoms include repetitions of sounds and words, periods when speech appears to be blocked, and excessive prolongation of sounds or words. These features are often accompanied by extraneous, effortful-sounding noises, and facial movements somewhat resembling tics. The latter extraneous movements during speech can extend to the arms and torso. The speech output of those affected is greatly reduced, with severe cases being able to say only a quarter as much as their peers or requiring four times as long to say as much as their peers. Chronic stuttering is associ- ated with clinical levels of social anxiety, with social phobia reported for 40–60% of clinical cases. 8–10 However, it is clear that anxiety does not cause the problem. A lifetime of stuttering can cause significant quality-of-life impairment. Adults with chronic stuttering often fail to attain occupational potential, 11 as employers can believe that those who stutter are less Key Points • Stuttering is a speech disorder with genetic involvement that begins during the first years of life and is among the most prevalent of developmental disorders. • Early intervention shortly after onset during the pre-school years is recommended. • If not corrected during early childhood, stuttering becomes intractable and can cause psychological, social, educational and occupational problems. Correspondence: Professor Mark Onslow, Faculty of Health Sciences, The University of Sydney, PO Box 170, Lidcombe, NSW 1825, Australia. Fax: +61 2 9351 9392; email: [email protected] Declaration of conflict of interest: None declared. Accepted for publication 1 May 2012. doi:10.1111/jpc.12034 Journal of Paediatrics and Child Health 49 (2013) E112–E115 © 2012 The Authors Journal of Paediatrics and Child Health © 2012 Paediatrics and Child Health Division (Royal Australasian College of Physicians) E112
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No Job NameManagement of childhood stuttering Mark Onslow and Sue O’Brian
Australian Stuttering Research Centre, The University of Sydney, Sydney, New South Wales, Australia
Abstract: Stuttering is a speech disorder that begins during the first years of life and is among the most prevalent of developmental disorders. It appears to be a problem with neural processing of speech involving genetics. Onset typically occurs during the first years of life, shortly after language development begins. Clinical presentation during childhood is interrupted and effortful speech production, often with rapid onset. If not corrected during early childhood, it becomes intractable and can cause psychological, social, educational and occupational problems. There is evidence from replicated clinical trials to support early intervention during the pre-school years. Meta-analysis of studies indicates that children who receive early intervention during the pre-school years are 7.7 times more likely to have resolution of their stuttering. Early intervention is recommended with a speech pathologist. Some children who begin to stutter will recover without such intervention. However, the number of such recoveries is currently not known, and it is not possible to predict which children are likely to recover naturally. Consequently, the current best practice is for speech pathologists to monitor children for signs of natural recovery for up to 1 year before beginning treatment.
Key words: diagnosis; management; paediatrics; stuttering; treatment.
Stuttering
Stuttering, also known as stammering in the United Kingdom, is a speech disorder that begins during the first years of life. A recent community cohort study of 1619 Australian children recruited at 8 months old found that 8.5% had begun to stutter by 3 years of age.1 The shape of the cumulative inci- dence plot suggests that more cases will emerge as the cohort is studied further. Onset was found to be essentially unpre- dictable, with only 3.7% of cases explainable with case history variables such as advanced language development, twinning and maternal education level. A report of 3 to 17-year-olds derived from the United States National Health Interview Surveys (n = 95,132) showed stuttering to be the equal third most prevalent developmental disorder from among nine, which included attention-deficit/hyperactivity
disorder, autism, cerebral palsy and learning disability.2 The reported prevalence was 1.6%.
The cause of stuttering is currently unknown; however, brain imaging data suggest that it involves a problem with neural processing of speech,3 linked to structural and functional anomalies at brain sites responsible for spoken language.4,5 As these anomalies have only been investigated with school chil- dren and adults, it is unclear whether they are a cause or an effect of the disorder. There is genetic involvement in stuttering, with clear evidence of vertical transmission within families.6
Around two thirds of those affected, or their parents, report a family history. There is greater monozygotic concordance than dizygotic concordance, which offers a genetic account of around 70% of cases.7 At present, genetic linkage studies can account for less than one tenth of cases; however, exome sequencing technology has yet to be applied to the study of the disorder.
The clinical presentation of chronic stuttering in adolescence and adulthood is interrupted speech production. Symptoms include repetitions of sounds and words, periods when speech appears to be blocked, and excessive prolongation of sounds or words. These features are often accompanied by extraneous, effortful-sounding noises, and facial movements somewhat resembling tics. The latter extraneous movements during speech can extend to the arms and torso. The speech output of those affected is greatly reduced, with severe cases being able to say only a quarter as much as their peers or requiring four times as long to say as much as their peers. Chronic stuttering is associ- ated with clinical levels of social anxiety, with social phobia reported for 40–60% of clinical cases.8–10 However, it is clear that anxiety does not cause the problem. A lifetime of stuttering can cause significant quality-of-life impairment. Adults with chronic stuttering often fail to attain occupational potential,11
as employers can believe that those who stutter are less
Key Points
• Stuttering is a speech disorder with genetic involvement that begins during the first years of life and is among the most prevalent of developmental disorders.
• Early intervention shortly after onset during the pre-school years is recommended.
• If not corrected during early childhood, stuttering becomes intractable and can cause psychological, social, educational and occupational problems.
Correspondence: Professor Mark Onslow, Faculty of Health Sciences, The University of Sydney, PO Box 170, Lidcombe, NSW 1825, Australia. Fax: +61 2 9351 9392; email: [email protected]
Declaration of conflict of interest: None declared.
Accepted for publication 1 May 2012.
doi:10.1111/jpc.12034
bs_bs_banner
Journal of Paediatrics and Child Health 49 (2013) E112–E115 © 2012 The Authors
Journal of Paediatrics and Child Health © 2012 Paediatrics and Child Health Division (Royal Australasian College of Physicians)
E112
Clinical Presentation During Early Childhood
Speech pathologists are responsible for the treatment of stut- tering. In North America, they are known as speech-language pathologists, and in the United Kingdom as speech and lan- guage therapists. The most common presentation of early stut- tering to a medical practitioner is soon after onset, most commonly when the child is aged between 2 and 5 years. That clinical presentation is often prompted by the distressing nature of early stuttering to parents. Unlike other speech problems, such as poor articulation or delayed language development, stuttering appears unexpectedly after a period of normal and uneventful speech development. It can start quite suddenly when children begin to form simple sentences. Around half of the cases appear within a period of 1–3 days and a third of the cases during a single day.1,14 The severe symptoms described previously can occur soon after onset.15 There is a general con- sensus that the most common early sign of stuttering is repeti- tions of sounds and words, followed by the development of more disabling symptoms.1,16
Diagnosis of early stuttering in pre-schoolers is rarely difficult. Its speech disturbances are distinctive from the normal dysflu- encies and hesitations of early language development. Parents typically initiate referrals and are rarely mistaken in their belief that a child has begun to stutter. A recent longitudinal study1
showed no evidence of co-morbid speech, language or reading problems close to onset. Rare diagnostic confusion may occur with tic syndromes of early childhood. In severe cases, early stuttering may appear to be neurological in nature. Cases of stuttering during the school years (7–12) are less likely to present to medical clinics, as parents typically seek speech pathology intervention rather than medical advice.
The Effects of Stuttering During Childhood
Stuttering may cause distress to young children shortly after onset.14 Peers recognise stuttered speech17 and may react nega- tively to it.18 Negative attitudes to communication have been measured in stuttering children as young as 3–6 years old.19
These signs of negative social conditioning are likely to be the origins of the relationship between stuttering and anxiety later in life. However, to date, no report has documented frank signs of clinical anxiety in pre-school children.
There is also limited evidence of clinical anxiety in school children, with only one report showing even a suggestion of heightened anxiety levels in stuttering children aged 9–12,20
while a report of 9 to 14-year-old children showed no effects at all.21 It is the case, however, that for these school children, the negative social conditioning connected with stuttering intensi- fies. From 7 years onwards, negative attitudes to communica- tion worsen for stuttering children.22,23 Bullying is associated with anxiety later in life, and school-age children who stutter are bullied more often than their peers.24 Their peers have a negative perception of them,25 and this appears linked to prob- lems forming relationships.26
Treatment of Childhood Stuttering
It is challenging to treat chronic stuttering during adolescence and adulthood. Speech rehabilitation at that time of life is labo- rious, costly and relapse prone. Additionally, concomitant treat- ment for anxiety is frequently necessary. Clearly, then, effective childhood intervention is desirable in order to obviate the need for speech or anxiety treatment later in life.
To date, the best evidence for childhood stuttering treatment lies with a conceptually simple operant procedure conducted by parents, with supervision from a speech pathologist. This treatment is known as the Lidcombe Program and is available world-wide.27 A treatment guide and brochures for parents are downloadable from the web site of the Australian Stuttering Research Centre.28 Parents use operant conditioning principles, such as praise for periods when their child does not stutter, and occasionally request their child to self-correct an utterance con- taining stuttering. Parents also measure the child’s stuttering severity each day with a simple scale to ensure that the child progresses to a target of no stuttering or almost no stuttering. When that target is attained, a maintenance phase of treatment for around 1 year is implemented to reduce the chance of relapse, which is known to occur.29
The efficacy of the Lidcombe Program has been demonstrated with a series of Phase I, Phase II and Phase III clinical trials.30
To date, there have been two successful Phase III randomised controlled trials of the treatment with a no-treatment control group: one with New Zealand pre-schoolers and one with German pre-schoolers.31,32 A meta-analysis (n = 136) of Lid- combe Program clinical trials and short exposure experiments showed an odds ratio of 7.7.30 There is evidence that children successfully treated with this method in clinical trials are able to produce speech that is perceptually normal.33 Of interest in cases where parents do not have access to standard speech pathology services is a randomised Phase II trial showing the treatment to be efficacious in a telehealth format.34 However, those results show that the treatment time to attain no stutter- ing or almost no stuttering with a low-tech telehealth format is much longer than the median of 16 h with the standard for- mat.35 At the time of writing, a clinical trial is under way to determine whether that problem can be solved with modern webcam technology.
There are two other treatments for early childhood stuttering that are in earlier phases of clinical trial development than is the Lidcombe Program. One of these, being developed in the United Kingdom, is a family-based treatment that seeks to alleviate stressors within the child’s daily environment that are thought to be responsible for a child’s continued stuttering. Such stres- sors include a generally hurried life-style, a rapid speech rate and having unrealistic developmental language expectations of the child. Two non-randomised Phase I trials have been pub- lished to date,36,37 with not particularly encouraging results, showing overall 65% stuttering reductions.
Another recent treatment development for early stuttering, occurring in Australia, is based on the well-known fact that when adults who stutter speak with each syllable in time to a rhythm, they stop stuttering, only to resume stuttering when they stop speaking in that manner. However, three non- randomised trials38–40 have shown that the effects of such
Management of childhood stutteringM Onslow and S O’Brian
Journal of Paediatrics and Child Health 49 (2013) E112–E115 © 2012 The Authors Journal of Paediatrics and Child Health © 2012 Paediatrics and Child Health Division (Royal Australasian College of Physicians)
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rhythmic speech may be more permanent with early stuttering. The latter of those reports suggested that stuttering reduction of 96% may be attainable with this method.
When to Treat Childhood Stuttering
Considering the problems with chronic stuttering later in life, early childhood is the best time for treatment. In light of the evidence outlined earlier that social anxiety problems with stut- tering may well begin during the school years, intervention shortly after onset before beginning school is recommended. That recommendation is supported by clinical trials data which suggest that stuttering is clinically less tractable and relapse following treatment is more likely during the school years than the pre-school years.41,42
A challenge for speech pathologists is that some pre-school children who begin to stutter will recover naturally by adult- hood without formal treatment. There are many methodologi- cal problems with estimating the number, but there is a prevailing belief that it is in the range of 70–80%. A critical review placed the natural recovery rate from childhood to ado- lescence at 30–50%.43 The challenge here for speech patholo- gists is to consider the need for early intervention against the chance of early natural recovery. The accepted best practice is for speech pathologists to monitor pre-school stuttering children for signs of natural recovery for no longer than 1 year before intervening. It appears that less than 5% of children will recover naturally during that period.44 Immediate treatment is recom- mended in cases where a child is showing signs of social distress or avoidance. This may occur in response to negative peer reactions to stuttering. An overarching clinical guideline is that treatment at least needs to have begun before the child reaches 5 years of age.
Summary
Stuttering is one of the most prevalent developmental disorders of early childhood. It can appear suddenly after a period of normal speech and language development. It is known to involve genetics and is currently thought to be a problem with neural processing of speech. Effective early intervention from a speech pathologist is critical to avoid long-term quality-of-life problems, which may include educational and occupational limitations and mental health problems. There is a good reason to believe that such mental health problems may begin shortly after onset during the pre-school years. There is replicated, randomised, clinical evidence for effective early intervention. Many children may recover without intervention, but the exact number is not known, and it is not possible to know which children will recover. The best practice is for speech pathologists to monitor children for signs of natural recovery for up to 1 year before beginning treatment.
Multiple Choice Questions
1. Stuttering is a A. Non-genetic disorder B. A disorder caused by psychological problems C. A speech disorder
D. Now known with certainty to be a problem with neural speech processing
E. A language disorder A is incorrect, because there is evidence of genetic involvement in stuttering. B is incorrect, because anxiety occurs after onset and does not cause stuttering. C is correct, because current evidence suggests it to be a problem with neural speech processing. D is incorrect, because current evidence suggests, rather than proves, that to be the case. E is incorrect, because stuttering is a speech disorder. 2. The recommended treatment practice is to
A. Always delay intervention for 1 year after onset, in the hope that natural recovery will occur
B. Intervene after monitoring for natural recovery for up to 1 year
C. Reassure parents that the disorder is innocuous D. Consider genetic testing E. Refer for psychological assessment
A is incorrect, because the accepted best practice is to delay intervention for up to 1 year after onset, unless the disorder is causing excessive distress to the child or family. B is correct, because this is the current recommended best practice. C is incorrect, because the disorder is not innocuous; if it persists and becomes intractable, it may impair the quality of life. D is incorrect, because genetic testing is not necessary for diagnosis. E is incorrect, because psychological problems develop only if stuttering persists during the pre-school years. 3. Diagnosis of stuttering in pre-school children
A. Can be difficult, because it is indistinguishable from tics B. Can be difficult, because it is difficult to distinguish from
the normal hesitations and dysfluencies of language development
C. Can be difficult, because it is a psychological problem D. Is rarely difficult, because parents are usually correct in
their report of stuttering onset E. Is rarely difficult, because early stuttering is always severe
soon after onset A is incorrect, because stuttering only somewhat resembles tics. B is incorrect, because it is easy to distinguish from these aspects of normal language development. C is incorrect, because it is a speech problem, and psychological problems do not emerge until later in life. D is correct, because parents are rarely mistaken about this. E is incorrect, because stuttering symptoms are severe soon after onset only in some cases.
References
1 Reilly S, Onslow M, Packman A et al. Predicting stuttering onset by the age of 3 years: a prospective, community cohort study. Pediatrics 2009; 123: 270–7.
2 Boulet SL, Boyle CA, Schieve LA. Health care use and health and functional impact of developmental disabilities among US children, 1997–2005. Arch. Pediatr. Adolesc. Med. 2009; 163: 19–26.
M Onslow and S O’BrianManagement of childhood stuttering
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Journal of Paediatrics and Child Health © 2012 Paediatrics and Child Health Division (Royal Australasian College of Physicians)
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3 Packman A, Onslow M. Searching for the cause of stuttering. Lancet 2002; 360: 655–6.
4 Sommer M, Koch MA, Paulus W, Weiller C, Buchel C. Disconnection of speech-relevant brain areas in persistent developmental stuttering. Lancet 2002; 360: 380–3.
5 Chang SE, Erickson KI, Ambrose NG, Hasegawa-Johnson MA, Ludlow CL. Brain anatomy differences in childhood stuttering. Neuroimage [Article] 2008; 39: 1333–44.
6 Bloodstein O, Bernstein Ratner N. A Handbook on Stuttering, 6th edn. Clifton Park, NY: Delmar, 2008.
7 Felsenfeld S, Kirk KM, Zhu G, Statham DJ, Neale MC, Martin NG. A study of the genetic and environmental etiology of stuttering in a selected twin sample. Behav. Genet. 2000; 30: 359–66.
8 Iverach L, O’Brian S, Jones M et al. Prevalence of anxiety disorders among adults seeking speech therapy for stuttering. J. Anxiety Disord. 2009; 23: 928–34.
9 Blumgart E, Tran Y, Craig A. Social anxiety disorder in adults who stutter. Depress. Anxiety 2010; 27: 687–92.
10 Stein MB, Baird A, Walker JR. Social phobia in adults with stuttering. Am. J. Psychiatry 1996; 153: 278–80.
11 Klein JF, Hood SB. The impact of stuttering on employment opportunities and job performance. J. Fluency Disord. 2004; 29: 255–73.
12 Hurst MI, Cooper EB. Employer attitudes toward stuttering. J. Fluency Disord. 1983; 8: 1–12.
13 O’Brian S, Jones M, Packman A, Menzies R, Onslow M. Stuttering severity and educational attainment. J. Fluency Disord. 2011; 36: 86–92.
14 Yairi E. The onset of stuttering in two- and three-year-old children: a preliminary report. J. Speech Hear. Disord. 1983; 48: 171–7.
15 Conture EG, Kelly EM. Young stutterers’ nonspeech behaviors during stuttering. J. Speech Hear. Res. 1991; 34: 1041–56.
16 Yairi E, Ambrose NG. Early Childhood Stuttering. Austin: Pro-Ed, 2005. 17 Ezrati-Vinacour R, Platzky R, Yairi E. The young child’s awareness of
stuttering-like disfluency. J. Speech Lang. Hear. Res. 2001; 44: 368–80.
18 Langevin M, Packman A, Onslow M. Peer responses to stuttering in the preschool setting. Am. J. Speech Lang. Pathol. 2009; 18: 264–76.
19 Vanryckeghem M, Brutten GJ, Hernandez LM. A comparative investigation of the speech-associated attitude of preschool and kindergarten children who do and do not stutter. J. Fluency Disord. 2005; 30: 307–18.
20 Blood GW, Blood IM. Preliminary study of self-reported experience of physical aggression and bullying of boys who stutter: relation to increased anxiety. Percept. Mot. Skills 2007; 104: 1060–6.
21 Craig A, Hancock K. Anxiety in children and young adolescents who stutter. Aust. J. Human Commun. Disord. 1996; 24: 28–38.
22 De Nil LF, Brutten GJ. Speech-associated attitudes of stuttering and nonstuttering children. J. Speech Hear. Res. 1991; 34: 60–66.
23 Vanryckeghem M, Brutten GJ. The speech-associated attitude of…
Australian Stuttering Research Centre, The University of Sydney, Sydney, New South Wales, Australia
Abstract: Stuttering is a speech disorder that begins during the first years of life and is among the most prevalent of developmental disorders. It appears to be a problem with neural processing of speech involving genetics. Onset typically occurs during the first years of life, shortly after language development begins. Clinical presentation during childhood is interrupted and effortful speech production, often with rapid onset. If not corrected during early childhood, it becomes intractable and can cause psychological, social, educational and occupational problems. There is evidence from replicated clinical trials to support early intervention during the pre-school years. Meta-analysis of studies indicates that children who receive early intervention during the pre-school years are 7.7 times more likely to have resolution of their stuttering. Early intervention is recommended with a speech pathologist. Some children who begin to stutter will recover without such intervention. However, the number of such recoveries is currently not known, and it is not possible to predict which children are likely to recover naturally. Consequently, the current best practice is for speech pathologists to monitor children for signs of natural recovery for up to 1 year before beginning treatment.
Key words: diagnosis; management; paediatrics; stuttering; treatment.
Stuttering
Stuttering, also known as stammering in the United Kingdom, is a speech disorder that begins during the first years of life. A recent community cohort study of 1619 Australian children recruited at 8 months old found that 8.5% had begun to stutter by 3 years of age.1 The shape of the cumulative inci- dence plot suggests that more cases will emerge as the cohort is studied further. Onset was found to be essentially unpre- dictable, with only 3.7% of cases explainable with case history variables such as advanced language development, twinning and maternal education level. A report of 3 to 17-year-olds derived from the United States National Health Interview Surveys (n = 95,132) showed stuttering to be the equal third most prevalent developmental disorder from among nine, which included attention-deficit/hyperactivity
disorder, autism, cerebral palsy and learning disability.2 The reported prevalence was 1.6%.
The cause of stuttering is currently unknown; however, brain imaging data suggest that it involves a problem with neural processing of speech,3 linked to structural and functional anomalies at brain sites responsible for spoken language.4,5 As these anomalies have only been investigated with school chil- dren and adults, it is unclear whether they are a cause or an effect of the disorder. There is genetic involvement in stuttering, with clear evidence of vertical transmission within families.6
Around two thirds of those affected, or their parents, report a family history. There is greater monozygotic concordance than dizygotic concordance, which offers a genetic account of around 70% of cases.7 At present, genetic linkage studies can account for less than one tenth of cases; however, exome sequencing technology has yet to be applied to the study of the disorder.
The clinical presentation of chronic stuttering in adolescence and adulthood is interrupted speech production. Symptoms include repetitions of sounds and words, periods when speech appears to be blocked, and excessive prolongation of sounds or words. These features are often accompanied by extraneous, effortful-sounding noises, and facial movements somewhat resembling tics. The latter extraneous movements during speech can extend to the arms and torso. The speech output of those affected is greatly reduced, with severe cases being able to say only a quarter as much as their peers or requiring four times as long to say as much as their peers. Chronic stuttering is associ- ated with clinical levels of social anxiety, with social phobia reported for 40–60% of clinical cases.8–10 However, it is clear that anxiety does not cause the problem. A lifetime of stuttering can cause significant quality-of-life impairment. Adults with chronic stuttering often fail to attain occupational potential,11
as employers can believe that those who stutter are less
Key Points
• Stuttering is a speech disorder with genetic involvement that begins during the first years of life and is among the most prevalent of developmental disorders.
• Early intervention shortly after onset during the pre-school years is recommended.
• If not corrected during early childhood, stuttering becomes intractable and can cause psychological, social, educational and occupational problems.
Correspondence: Professor Mark Onslow, Faculty of Health Sciences, The University of Sydney, PO Box 170, Lidcombe, NSW 1825, Australia. Fax: +61 2 9351 9392; email: [email protected]
Declaration of conflict of interest: None declared.
Accepted for publication 1 May 2012.
doi:10.1111/jpc.12034
bs_bs_banner
Journal of Paediatrics and Child Health 49 (2013) E112–E115 © 2012 The Authors
Journal of Paediatrics and Child Health © 2012 Paediatrics and Child Health Division (Royal Australasian College of Physicians)
E112
Clinical Presentation During Early Childhood
Speech pathologists are responsible for the treatment of stut- tering. In North America, they are known as speech-language pathologists, and in the United Kingdom as speech and lan- guage therapists. The most common presentation of early stut- tering to a medical practitioner is soon after onset, most commonly when the child is aged between 2 and 5 years. That clinical presentation is often prompted by the distressing nature of early stuttering to parents. Unlike other speech problems, such as poor articulation or delayed language development, stuttering appears unexpectedly after a period of normal and uneventful speech development. It can start quite suddenly when children begin to form simple sentences. Around half of the cases appear within a period of 1–3 days and a third of the cases during a single day.1,14 The severe symptoms described previously can occur soon after onset.15 There is a general con- sensus that the most common early sign of stuttering is repeti- tions of sounds and words, followed by the development of more disabling symptoms.1,16
Diagnosis of early stuttering in pre-schoolers is rarely difficult. Its speech disturbances are distinctive from the normal dysflu- encies and hesitations of early language development. Parents typically initiate referrals and are rarely mistaken in their belief that a child has begun to stutter. A recent longitudinal study1
showed no evidence of co-morbid speech, language or reading problems close to onset. Rare diagnostic confusion may occur with tic syndromes of early childhood. In severe cases, early stuttering may appear to be neurological in nature. Cases of stuttering during the school years (7–12) are less likely to present to medical clinics, as parents typically seek speech pathology intervention rather than medical advice.
The Effects of Stuttering During Childhood
Stuttering may cause distress to young children shortly after onset.14 Peers recognise stuttered speech17 and may react nega- tively to it.18 Negative attitudes to communication have been measured in stuttering children as young as 3–6 years old.19
These signs of negative social conditioning are likely to be the origins of the relationship between stuttering and anxiety later in life. However, to date, no report has documented frank signs of clinical anxiety in pre-school children.
There is also limited evidence of clinical anxiety in school children, with only one report showing even a suggestion of heightened anxiety levels in stuttering children aged 9–12,20
while a report of 9 to 14-year-old children showed no effects at all.21 It is the case, however, that for these school children, the negative social conditioning connected with stuttering intensi- fies. From 7 years onwards, negative attitudes to communica- tion worsen for stuttering children.22,23 Bullying is associated with anxiety later in life, and school-age children who stutter are bullied more often than their peers.24 Their peers have a negative perception of them,25 and this appears linked to prob- lems forming relationships.26
Treatment of Childhood Stuttering
It is challenging to treat chronic stuttering during adolescence and adulthood. Speech rehabilitation at that time of life is labo- rious, costly and relapse prone. Additionally, concomitant treat- ment for anxiety is frequently necessary. Clearly, then, effective childhood intervention is desirable in order to obviate the need for speech or anxiety treatment later in life.
To date, the best evidence for childhood stuttering treatment lies with a conceptually simple operant procedure conducted by parents, with supervision from a speech pathologist. This treatment is known as the Lidcombe Program and is available world-wide.27 A treatment guide and brochures for parents are downloadable from the web site of the Australian Stuttering Research Centre.28 Parents use operant conditioning principles, such as praise for periods when their child does not stutter, and occasionally request their child to self-correct an utterance con- taining stuttering. Parents also measure the child’s stuttering severity each day with a simple scale to ensure that the child progresses to a target of no stuttering or almost no stuttering. When that target is attained, a maintenance phase of treatment for around 1 year is implemented to reduce the chance of relapse, which is known to occur.29
The efficacy of the Lidcombe Program has been demonstrated with a series of Phase I, Phase II and Phase III clinical trials.30
To date, there have been two successful Phase III randomised controlled trials of the treatment with a no-treatment control group: one with New Zealand pre-schoolers and one with German pre-schoolers.31,32 A meta-analysis (n = 136) of Lid- combe Program clinical trials and short exposure experiments showed an odds ratio of 7.7.30 There is evidence that children successfully treated with this method in clinical trials are able to produce speech that is perceptually normal.33 Of interest in cases where parents do not have access to standard speech pathology services is a randomised Phase II trial showing the treatment to be efficacious in a telehealth format.34 However, those results show that the treatment time to attain no stutter- ing or almost no stuttering with a low-tech telehealth format is much longer than the median of 16 h with the standard for- mat.35 At the time of writing, a clinical trial is under way to determine whether that problem can be solved with modern webcam technology.
There are two other treatments for early childhood stuttering that are in earlier phases of clinical trial development than is the Lidcombe Program. One of these, being developed in the United Kingdom, is a family-based treatment that seeks to alleviate stressors within the child’s daily environment that are thought to be responsible for a child’s continued stuttering. Such stres- sors include a generally hurried life-style, a rapid speech rate and having unrealistic developmental language expectations of the child. Two non-randomised Phase I trials have been pub- lished to date,36,37 with not particularly encouraging results, showing overall 65% stuttering reductions.
Another recent treatment development for early stuttering, occurring in Australia, is based on the well-known fact that when adults who stutter speak with each syllable in time to a rhythm, they stop stuttering, only to resume stuttering when they stop speaking in that manner. However, three non- randomised trials38–40 have shown that the effects of such
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rhythmic speech may be more permanent with early stuttering. The latter of those reports suggested that stuttering reduction of 96% may be attainable with this method.
When to Treat Childhood Stuttering
Considering the problems with chronic stuttering later in life, early childhood is the best time for treatment. In light of the evidence outlined earlier that social anxiety problems with stut- tering may well begin during the school years, intervention shortly after onset before beginning school is recommended. That recommendation is supported by clinical trials data which suggest that stuttering is clinically less tractable and relapse following treatment is more likely during the school years than the pre-school years.41,42
A challenge for speech pathologists is that some pre-school children who begin to stutter will recover naturally by adult- hood without formal treatment. There are many methodologi- cal problems with estimating the number, but there is a prevailing belief that it is in the range of 70–80%. A critical review placed the natural recovery rate from childhood to ado- lescence at 30–50%.43 The challenge here for speech patholo- gists is to consider the need for early intervention against the chance of early natural recovery. The accepted best practice is for speech pathologists to monitor pre-school stuttering children for signs of natural recovery for no longer than 1 year before intervening. It appears that less than 5% of children will recover naturally during that period.44 Immediate treatment is recom- mended in cases where a child is showing signs of social distress or avoidance. This may occur in response to negative peer reactions to stuttering. An overarching clinical guideline is that treatment at least needs to have begun before the child reaches 5 years of age.
Summary
Stuttering is one of the most prevalent developmental disorders of early childhood. It can appear suddenly after a period of normal speech and language development. It is known to involve genetics and is currently thought to be a problem with neural processing of speech. Effective early intervention from a speech pathologist is critical to avoid long-term quality-of-life problems, which may include educational and occupational limitations and mental health problems. There is a good reason to believe that such mental health problems may begin shortly after onset during the pre-school years. There is replicated, randomised, clinical evidence for effective early intervention. Many children may recover without intervention, but the exact number is not known, and it is not possible to know which children will recover. The best practice is for speech pathologists to monitor children for signs of natural recovery for up to 1 year before beginning treatment.
Multiple Choice Questions
1. Stuttering is a A. Non-genetic disorder B. A disorder caused by psychological problems C. A speech disorder
D. Now known with certainty to be a problem with neural speech processing
E. A language disorder A is incorrect, because there is evidence of genetic involvement in stuttering. B is incorrect, because anxiety occurs after onset and does not cause stuttering. C is correct, because current evidence suggests it to be a problem with neural speech processing. D is incorrect, because current evidence suggests, rather than proves, that to be the case. E is incorrect, because stuttering is a speech disorder. 2. The recommended treatment practice is to
A. Always delay intervention for 1 year after onset, in the hope that natural recovery will occur
B. Intervene after monitoring for natural recovery for up to 1 year
C. Reassure parents that the disorder is innocuous D. Consider genetic testing E. Refer for psychological assessment
A is incorrect, because the accepted best practice is to delay intervention for up to 1 year after onset, unless the disorder is causing excessive distress to the child or family. B is correct, because this is the current recommended best practice. C is incorrect, because the disorder is not innocuous; if it persists and becomes intractable, it may impair the quality of life. D is incorrect, because genetic testing is not necessary for diagnosis. E is incorrect, because psychological problems develop only if stuttering persists during the pre-school years. 3. Diagnosis of stuttering in pre-school children
A. Can be difficult, because it is indistinguishable from tics B. Can be difficult, because it is difficult to distinguish from
the normal hesitations and dysfluencies of language development
C. Can be difficult, because it is a psychological problem D. Is rarely difficult, because parents are usually correct in
their report of stuttering onset E. Is rarely difficult, because early stuttering is always severe
soon after onset A is incorrect, because stuttering only somewhat resembles tics. B is incorrect, because it is easy to distinguish from these aspects of normal language development. C is incorrect, because it is a speech problem, and psychological problems do not emerge until later in life. D is correct, because parents are rarely mistaken about this. E is incorrect, because stuttering symptoms are severe soon after onset only in some cases.
References
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M Onslow and S O’BrianManagement of childhood stuttering
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