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Journal of Fluency Disorders
Critical review
Early stuttering, temperament and anxiety: Two hypotheses
Elaina Kefalianosa,b,!, Mark Onslowc, Susan Blockd, Ross Menziesc, Sheena Reillya,b,e
a Murdoch Childrens Research Institute, Australiab Department of Paediatrics, University of Melbourne, Australiac Australian Stuttering Research Centre, University of Sydney, Australiad School of Human Communication Sciences, La Trobe University, Australiae Speech Pathology Department, Royal Childrens Hospital, Australia
a r t i c l e i n f o
Article history:Received 25 October 2011Received in revised form 1 March 2012Accepted 3 March 2012Available online 29 March 2012
Keywords:StutteringTemperamentAnxietyChildren
a b s t r a c t
Purpose: The topic of temperament and early stuttering and the extent to which it involvesanxiety is theoretically and clinically relevant. The topic can contribute to theory develop-ment and clinical practices with early stuttering.Method: We present a review of the empirical literature for this area with a view to deter-mining which of two hypotheses might be true. The first is that, for the population of thosewho stutter, unusual temperament is a causal factor for the development of the disorderand its later association with anxiety. The second hypothesis is that for the populationof those who stutter the developmental manifestation of anxiety is an effect of stuttering.Both hypotheses attempt to account for the well-known association of anxiety with chronicstuttering.Results: A firm conclusion about the matter would be premature at present because theliterature involved is limited and contains some inconsistencies. We suggest some quan-titative and qualitative directions for future research. We argue also that the only way toresolve the matter is with longitudinal studies of cohorts ascertained prior to stutteringonset.Conclusion: Conclusive findings about the matter can only emerge when research extendsbeyond its modest scope of 10 interpretable publications.
Educational objectives: The reader will be able to: (a) describe temperament and the roleit plays during anxiety development, (b) explain two alternative hypotheses which impli-cate temperament in early childhood stuttering; (c) describe current knowledge regardingtemperament similarities and differences between stuttering and control children, and (d)discuss the theoretical and clinical implications of understand the relationship betweentemperament, anxiety and early stuttering.
Anxiety involves cognitive, behavioural and physiological components and is driven by threat expectation (Marks, 1987;Pahl, Barrett, & Gullo, 2012; Rapee, Schniering, & Hudson, 2009). Threat expectation refers to the way in which a personinterprets situations and environmental stimuli. Compared to others, anxious people process such stimuli in a threateningmanner. Anxious children are characterised by worry, fearfulness, excessive concern for themselves and others and tension.High levels of anxiety in children and adolescents can negatively impact on social and emotional development and academicachievements (Ameringen, Mancini, & Farvolden, 2003; Crawford & Manassis, 2011; Rapee et al., 2009). Trait anxiety isconsidered to be a stable predisposition to perceive environmental threats. It reflects temperament and environmentalexperiences such as socialisation and early parenting practices. In contrast, state anxiety is a temporary experience thatis situation specific, and its severity can fluctuate over time for an individual. Endler and Parker (1990) describe state andtrait anxiety in a multidimensional model whereby the state anxiety experienced by an individual is the result of interplaybetween particular aspects of their trait anxiety and the perceived threat of a situation. Researchers have examined thepotential role of both of these types of anxiety during stuttering.
2. Stuttering and anxiety
There is extensive literature that establishes a link between stuttering and anxiety (for a review, see Iverach, Menzies,O’Brian, Packman, & Onslow, 2011). Stuttering adults have repeatedly been demonstrated to have higher trait anxiety (Alm &Risberg, 2007; Craig, 1990, 2003; Ezrati-Vinacour & Levin, 2004) and state anxiety (Craig, 1990; Gabel, Colcord, & Petrosino,2002; Peters & Hulstijn, 1984) levels than controls. Using a social anxiety inventory, Kraaimaat et al. (2002) reported thatstuttering adults exhibit heightened levels of emotional distress in social settings. Tran, Blumgart, and Craig (2011) confirmedthis finding and additionally reported that stuttering adults exhibit more negative moods compared to controls. Similarly,stuttering adolescents have been reported to exhibit higher levels of state, trait and social anxiety compared to controls(Blood, Blood, Maloney, Meyer, & Qualls, 2007; Mulcahy, Hennessey, Beilby, & Byrnes, 2008).
Compared to controls, clinical populations of stuttering adults have been found to have a significantly heightened risk ofsocial phobia (Blumgart, Tran, & Craig, 2010; Iverach et al., 2009a; Stein, Baird, & Walker, 1996). Social phobia is defined as“marked and persistent fear of one or more social or performance situations in which the person is exposed to unfamiliarpeople or to possible scrutiny by others” (American Psychiatric Association, 1994, p. 416). Those with social phobia may avoidsocial situations or experience them with extreme levels of distress. The threat of social humiliation and embarrassmentis irrational; the perceived threat is excessive in light of the actual threat. Social phobia can cause social isolation andoccupational impairment. Additionally, clinical populations of those who stutter are at increased risk of anxiety-relatedmood and personality disorders compared to controls (Iverach et al., 2009b; Iverach et al., 2010).
3. Development of anxiety
A range of risk factors may predispose an individual to develop anxiety. A link between anxiety and gender has beenreported, with women having approximately twice the risk of men for having an anxiety disorder (Costello, Mustillo, Erkanli,Keeler, & Angold, 2003; Essau, Conradt, & Petermann, 2000; Lewinsohn, Zinbarg, Lewinsohn, & Sack, 1997). However, some
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reports failed to find such an effect (Canino, Shrout, Rubio-Stipec, Bird, & Bravo, 2004; Ford, Goodman, & Meltzer, 2003).Additionally, there is evidence to suggest that anxiety disorders are hereditary (Ehringer, Rhee, Young, Corley, & Hewitt,2006; Hettema, Neale, & Kendler, 2001; Last, Hersen, Kazdin, Francis, & Grubb, 1987; Lieb et al., 2000; Rapee et al., 2009;Stevenson, Batten, & Cherner, 1992; Thapar & McGuffin, 1995; Topolski et al., 1997).
Children with communication difficulties have a heightened risk of developing mental health disorders later in life.Baker and Cantwell (1987) examined a large cohort of children with communication difficulties and reported a heightenedprevalence of psychiatric disorders five years after the initial assessment. Similarly, Beitchman et al. (2001) conducted a 14year longitudinal study in which they concluded that children with speech and/or language difficulties had a significantlyhigher incidence of anxiety disorders, particularly social phobia, in adolescence compared to those who had no history ofspeech or language deficits.
3.1. Temperament as a risk factor for anxiety
Childhood temperament has been identified as a risk factor for development of anxiety disorders later in life. Temper-ament is an innate tendency to react to or interact with daily environments in a certain fashion. Thomas and Chess (1977)described nine dimensions of temperament which have been used for the design of the Behavioural Style Questionnaire(McDevitt & Carey, 1978), which is the tool most commonly used in studies of childhood temperament.
Thomas and Chess’ (1977) nine dimensions of temperament are: (1) activity level, referring to the amount of physicalactivity in a child’s behaviour, (2) rhythmicity, referring to the regularity of physiologic functions such as sleeping, feed-ing, or toileting, (3) adaptability, referring to the capacity to alter behaviour in a desired manner with ease, (4) attentionspan/persistence, referring to how long a child will engage in a particular activity and capacity to persist with that attentionwhen presented with distractions, (5) threshold of responsiveness, referring to how much stimulation is need to evoke aresponse, (6) intensity of reaction, referring to the energy a child exerts in response to a stimulus irrespective of the magni-tude or nature of the reaction, (7) quality of mood, referring to the extent to which a child expresses positive and negativeemotions, (8) distractibility, referring to how easily a child’s attention can be diverted from a task to an irrelevant stimulus,and (9) approach/withdrawal, referring to how children respond to new situations.
Temperament is a stable and genetically determined construct (Buss & Plomin, 1984; Costa & McCrae, 2001; Goldsmith,1996; Rothbart, Derryberry, & Hershey, 2000; Saudino, 2005). However the expression of temperament traits can varymarkedly from person to person as personality traits. Between 20 and 60% of the personality differences among peoplecan be accounted for by the biological underpinnings of temperament, including genetic predisposition (Goldsmith, Buss, &Lemery, 1997; Loehlin, 1992; McCrae et al., 2000; Saudino, 2005). These individual differences, however, cannot be accountedfor by genetics alone.
Environmental factors can also strongly influence expression of specific temperament dimensions as personality traits(Anderson, Pellowski, Conture, & Kelly, 2003; Buss & Plomin, 1984; Caspi, 1998; Costa & McCrae, 2001; Kagan, Reznick,Clarke, Snidman, & Garcia-Coll, 1984; Rothbart & Bates, 1998; Rothbart & Derryberry, 1981). Saudino (2005) purported thatenvironment and experiences are responsible for between 40 and 80% of individual personality variation.
Thomas and Chess’ (1977) approach/withdrawal temperament dimension (see earlier Section 3.2) has been linked todevelopment of anxiety disorders. Behavioural inhibition, characterised by reluctance to explore and an avoidance of new andunusual experiences, has been identified as the main temperament characteristic involved in their development (Chorpita &Barlow, 1998; Hayward, Killen, Kraemer, & Taylor, 1998; Hudson & Rapee, 2004; Kagan, Reznick, & Gibbons, 1989; Manassis& Bradley, 1994; Prior, Sanson, Smart, & Oberklaid, 2000; Prior, Smart, Sanson, & Oberklaid, 2000; Rapee, 2002; Rapeeet al., 2009; Rapee & Szollos, 2003; Schwartz, Snidman, & Kagan, 1999). Withdrawal has also been highlighted as a typicalbehavioural characteristic of anxious children and adolescents (Hayward et al., 1998; Rapee & Szollos, 2003).
In addition to temperament characteristics which may act as precursors of anxiety, it has also been reported that childrenwho exhibit an overall “difficult” temperament have a heightened risk of developing an anxiety disorder later in life. Coteet al. (2009), with a large cohort, reported that this effect begins during the preschool years. They examined the developmentof depressive and anxiety symptoms during the first five years of life and identified risk factors for such symptoms. A sampleof 1759 children were followed annually from 5 to 60 months of age. Maternal interviews were conducted each year tocollect information about parent and child behaviours and family characteristics. Each child’s temperament was calculatedby asking mothers to assign a score from 0 to 2 to indicate how frequently they observed their child exhibit each of thefollowing attributes: (1) “nervous, high strung or tense,” (2) “appears fearful or anxious,” (3) “appears worried,” (4) “not ashappy as other children,” and (5) “has difficulty having fun” (Cote et al., 2009, p. 1204). Those children who received higherscores were classified as exhibiting a more “difficult” temperament.
Cote et al. (2009) concluded that during the first five years of life, the frequency of depressive and anxiety symptomsgenerally increases without a gender effect. However, they identified “difficult” temperament as the “single most importantvulnerability factor” (Cote et al., 2009, p. 1206) for heightened depressive and anxiety symptoms. This finding supportedresults from previous studies that also found that depressive and anxiety symptoms may develop during early childhood(Briggs-Gowan, Carter, Skuban, & Howitz, 2001; Sterba, Egger, & Angold, 2007; Thompson et al., 1996; Wilens et al., 2002)and that having a difficult temperament increases the risk of depressive and anxiety symptoms (Bosquet & Egeland, 2006;Guerin, Allen, & Craig, 1997).
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3.2. Anxiety disorders during childhood
Anxiety disorders comprise generalised anxiety disorder, phobias, obsessive compulsive disorder, panic disorder, andposttraumatic stress syndrome (American Psychiatric Association, 1994). They are among the most common disorders ofadolescence and childhood and have the potential to significantly affect development (Rapee et al., 2009). Their prevalencehas been reported in the range 2.5–5.0% during childhood and early adolescence (Breton et al., 1999; Costello et al., 2003;Ford et al., 2003; Lewinsohn, Hops, Roberts, Seeley, & Andrews, 1993; Lewinsohn et al., 1997). Around two thirds of thosediagnosed with anxiety disorders during childhood or adolescence go on to exhibit the disorder during later years (Gregoryet al., 2007). Prevalence of social phobia during childhood and adolescence has been reported in the range 0.3–1.5% (Caninoet al., 2004; Ford et al., 2003; Lewinsohn et al., 1993). In order to be diagnosed with an anxiety disorder, the individual mustexhibit a level of anxiety which restricts and impairs their level of functioning. Given this, there is also a large proportion ofchildren in the population who exhibit trait anxiety but who do not meet criteria to be diagnosed with an anxiety disorder(see Rapee et al., 2009).
4. Early stuttering, temperament and anxiety: two hypotheses
In short, there is reason to believe that early development of anxiety symptoms may be associated with temperamentcharacteristics. Specifically, behavioural inhibition and a “difficult” temperament, which may emerge in the first years oflife, have been implicated as precursors of anxiety during personality development. Anxiety disorders are experienced bysome adults and adolescents who stutter and many other people who stutter experience heightened levels of anxiety, it isnot known when the initial symptoms of anxiety begin to occur in the development of the disorder. Hence, it is of interestto explore whether temperament research with stuttering children has identified anomalies during early stuttering duringthe preschool years, and whether any identified temperament anomalies potentially signal development of anxiety. Here,the preschool years are defined as younger than 6 years 0 months.
There are two possible research hypotheses about temperament differences between stuttering and control preschoolchildren and whether they may prompt anxiety as part of personality. The first is that, for the population of those whostutter, temperament is a causal factor for development of the disorder. In other words, certain temperament characteristicsmay increase risk of stuttering onset (Conture, 1991; Conture, 2001; Guitar, 1998; Riley & Riley, 1979; Riley & Riley, 2000;Sermas & Cox, 1982; Smith & Kelly, 1997; Starkweather & Gottwald, 1990; Zebrowski & Conture, 1998; Zebrowski & Schum,1993). Such a position is consistent with currently popular multifactorial theory of early stuttering (see Section 4.1). If thathypothesis is true, then temperament may be involved causally also in anxiety development for those who stutter.
A second research hypothesis is that, for the population of those who stutter, temperament differences are not a causalfactor for manifestation of anxiety within personality, but that such anxiety problems develop later during life and arean effect of stuttering. This hypothesis is tenable because awareness of stuttering develops in children from two years ofage (Ambrose & Yairi, 1994; Boey et al., 2009; Yairi, 1983) and negative evaluation of stuttering is exhibited by four yearold children (Ezrati-Vinacour, Platzky, & Yairi, 2001). Further, some stuttering preschool children experience negative peerresponses and social interactions with peers as a consequence of their stuttering (Bernstein Ratner, 1997; Langevin, Packman,& Onslow, 2009, 2010; Onslow, Attanasio, & Harrison, 2003; Yairi, 1983). It may be that children with a heightened “difficult”temperament with Thomas and Chess’ (1977) approach/withdrawal dimension (see above) are more susceptible than otherchildren to environmental influences that promote anxiety during subsequent personality development.
4.1. Theoretical and clinical importance of the topic
Multifactorial causal models of stuttering (for an overview, see Packman & Attanasio, 2004) are currently thought to beviable and are driving clinical trial development of early interventions based on these findings (Millard, Edwards, & Cook,2009; Millard, Nicholas, & Cook, 2008). All iterations of those models incorporate the notion of temperament, either as aninnate childhood development factor or as an environmental factor in the sense of the way a child reacts to or interactswith daily living environments. Theorists have hypothesised that the degree of emotional reactivity and regulation childrenpossess may interact with communication stressors in their environment and when coupled with functional brain deficitsresult in the child stuttering. The varying degree of emotional reactivity and regulation expressed by children may par-tially account for the phenomenon of natural recovery and the variability commonly observed in the severity of children’sstuttering.
Establishing which of the above hypotheses is true will contribute to evaluating the veracity of multifactorial models. Iftemperament is involved with the cause of stuttering, the models are confirmed, and if not they are partially refuted. Mul-tifactorial models of early stuttering stipulate that the physiological, emotional, linguistic and/or cognitive variables whichinfluence the onset and development of stuttering for one child are different to this for another child. As the factors impli-cated in the development of the disorder vary between individuals, it is not possible to completely refute the hypothesis thattemperament influences the onset and development of early stuttering. Comparison of individual temperament dimensionsbetween stuttering children and controls would however provide some indication as to whether particular temperamentcharacteristics are expressed differently for stuttering children. Further, a lack of significant findings may provide some
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evidence to partially refute the theory that temperament is involved causally in the manifestation of the disorder for thewider population of stuttering preschool children.
The topic of temperament is also clinically important. With emerging clinical trials data showing the efficacy of earlyintervention (for an overview, see Onslow, Jones, Menzies, O’Brian, & Packman, 2012), screening children for risk of stutteringonset will emerge as a clinical consideration. If hypothesis one is true, and it is the case that “difficult” temperament markersincrease risk of stuttering onset, that information will need to be incorporated within screening protocols. Concurrent withemerging knowledge of early intervention, knowledge about natural recovery is well established and is continuing to build,making the timing of early intervention a prime clinical issue. It is essential to find the optimal trade off between a “waitand see” strategy and an immediate treatment strategy. If hypothesis two is true, and it is the case that early stutteringultimately causes anxiety problems later in life, then that information will need to be taken into account when timingearly intervention. Such a consideration will be particularly salient if hypothesis one is true and “difficult” temperamentis associated causally with stuttering. In such a case, stuttering preschool children with such temperament markers willrequire earlier intervention than children without them.
While understanding the role of temperament during early stuttering has theoretical and clinical importance a limitedpeer review search has been published. During the past decade, ten studies have been published which have compared theexpression of temperament traits between stuttering and control children.
5. Early stuttering and the Thomas and Chess (1977) temperament dimensions
5.1. Activity level
Anderson et al. (2003) used the Behavioural Style Questionnaire (McDevitt & Carey, 1978) to measure activity levels of 31stuttering and 31 control children aged 3–5 years. The questionnaire contains 100 items which measure Thomas and Chess’(1977) nine temperament dimensions. Parents respond to the questionnaire by rating their child using a six-point scale.To measure the dimension activity level, parents were presented with questions such as “the child runs to where he/shewants to go.” No significant differences were detected between stuttering and control children’s activity levels. Howell et al.(2004) produced conflicting findings. They examined activity levels of 10 stuttering children and 10 age and gender matchedcontrols using the Behavioural Style Questionnaire (McDevitt & Carey, 1978), some of whom were younger than six years.Their age range was 3–7 years (mean 5 years 10 months). Howell et al. reported that the stuttering children were significantlymore active than controls. Both of these studies based findings on small cohorts of participants. This may partially account forthe conflicting findings reported. Further research needs to be conducted using a larger cohort before accurate conclusionscan be made.
5.2. Rhythmicity
Lewis and Golberg (1997) administered the Parent Childhood Temperament Questionnaire for 3–7 year olds (Thomas &Chess, 1977) to the parents of 11 children “at risk” for stuttering, aged between 3 and 5 years, and 11 age and gender matchedcontrols. The former group was recruited from a speech clinic at their initial consultation, which was prompted by parentconcern about fluency. The latter group was recruited from childcare centres. Temperament dimensions were examined toidentify which predicted stuttering onset for preschool children. Rhythmicity was found to be one of several (see Sections 5.3and 5.7) predictors of being “at risk” for stuttering, to the extent that those children showed more regularity of physiologicalfunctions than controls. A serious caveat to these findings is that children in the “at-risk” group were not confirmed tohave developed stuttering. Anderson et al. (2003) and Howell et al. (2004) (see Section 5.1) produced contrasting findings.Using the Behavioural Style Questionnaire (McDevitt & Carey, 1978), Anderson et al. reported the stuttering children to beless rhythmic than controls. In contrast, Howell et al. reported no significant differences between stuttering and controlchildren’s levels of rhythmicity.
5.3. Adaptability
Lewis and Golberg (1997) also reported that the children “at-risk” for stuttering were more adaptable than controls.However, two reports described above (Anderson et al., 2003; Howell et al., 2004) found stuttering children to be lessadaptable to changes in their environment. This finding was confirmed by another study using an alternative method of datacollection (Schwenk, Conture, & Walden, 2007). Schwenk et al. (2007) used observation of temperament-related behavioursrather than parent questionnaires to compare adaptability of 18 stuttering children and 18 age and gender matched controls,aged 3–5 years. The children were video recorded talking to their parents until a 300 word speech sample had been obtained.During each recording, the video camera was repeatedly moved with an audible sound. Adaptability was determined bymeasuring how long a child focused on each camera movement. Schwenk et al. reported that stuttering children were lessadaptable than controls.
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5.4. Attention span/persistency
Parent report data from Howell et al.’s (2004) study and observational data from Schwenk et al.’s (2007) study (seeSections 5.1 and 5.3) suggest preschool stuttering children have a shorter attention span and reduced ability to adapt tochanges in their environments compared to controls. In contrast, Anderson et al.’s (2003) study reviewed above reported nosignificant differences between stuttering and control children’s attention spans.
Karrass et al. (2006) departed from the usual method for using the Behavioural Style Questionnaire to explore temper-ament and early stuttering. Participants were 65 3–5-year-old stuttering children and 56 3–5-year-old control children.Karass et al. used expert consensus to group Behavioural Style Questionnaire responses into responses dealing with emo-tional reactivity, emotional regulation, and attention regulation. For the purpose of the present review, the latter categorywas considered the closest match for the Thomas and Chess (1977) attention span/persistency category. Karass et al. foundthat the stuttering children had significantly less capacity to regulate their attention than controls.
5.5. Threshold of responsiveness
Based on parent reports using the Behavioural Style Questionnaire, no significant differences for threshold of responsive-ness have been reported between stuttering and control children (Anderson et al., 2003; Howell et al., 2004).
5.6. Intensity of reaction
While some reports have found no significant differences between stuttering and control children’s reactivity to stimuli(Anderson et al., 2003; Howell et al., 2004) others have indicated that stuttering preschool children react more to environ-mental stimulation and have reduced capacity to regulate their emotions than controls (Karrass et al., 2006). Arnold, Conture,Key, and Walden (2011) studied the topic with a novel method and produced opposite results. They used electroencephalo-gram (EEG) recordings as measures of emotional reactivity for a clinical sample of nine stuttering and nine control 3–5year olds. EEG recordings have been shown to reliably measure emotional reactivity (for review, see Coan and Allen, 2004).Participants were presented with a sequence of pictures. Following this, a conversation depicting a happy, angry or neutralemotion was played in the background for the child to listen to. The conversation was played for 30 s. For the duration of theconversation, the researchers used EEG recordings to measure brain activity associated with emotional reactivity. Once thebackground conversation had finished, participants were presented with the picture sequences again and asked to generatea story about the pictures. This process was repeated three times so that the child was exposed to all three emotions in thebackground conversations. Results indicated no significant differences between stuttering and control participants for brainactivity related to emotional reactivity. Caveats to interpreting the Arnold et al. null result are that the laboratory protocolmay not have created the intended emotional reactions, and that the EEG measures may not have been valid markers ofemotional reactivity.
5.7. Quality of mood
The Lewis and Golberg (1997) study included a report that children “at risk” for stuttering showed more intense positivemood than the control group. However, as noted above, a caveat to that finding is that their stuttering status was unclear.In contrast, Anderson et al. (2003) (see Section 5.1) reported no significant differences between stuttering and control chil-dren’s display of positive or negative affect while the Howell et al. (2004) report with the Behavioural Style Questionnaire(see Section 5.1) showed that children of ages 3-7 years had a more negative affect than controls. This finding was supportedby Johnson, Walden, Conture, and Karrass (2010). These investigators compared emotional regulation of 16 stuttering and16 control children ages 3–5 years. Their expression of positive and negative emotions was measured with observationof children’s responses to a “disappointing gift” paradigm. They reported no significant difference in verbalisation of pos-itive emotions after receiving a desirable gift, but the stuttering children made more verbalisations of negative emotionalresponses after receiving an undesirable gift. Johnson et al. concluded that, compared to controls, stuttering preschoolchildren may have problematic regulation of emotion regulation in negative situations.
5.8. Distractibility
The Anderson et al. (2003) report (see Section 5.1) included comparison of stuttering and control preschool childrenmaintaining attention when presented with a distraction. Stuttering children were found to be less distractible than theirfluent peers. In Howell et al.’s (2004) study, no significant differences were reported between stuttering and control children’slevels of distractibility. Schwenk et al. (2007) generated contrasting findings using a direct observation paradigm wherechildren were distracted from a conversation with a parent by a sound in the room. They reported that 10 stuttering childrenages 3–5 years were more distractible than 10 control children. The conflicting conclusions may be due to the fact that theAnderson et al. and Howell et al. findings were based on parental responses to the Behavioural Style Questionnaire, butSchwenk et al. used a direct observation paradigm.
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Table 1Summary of findings about temperament and stuttering preschool children.
Higher Lower Equal
Thomas and Chess (1977) temperament dimensionsActivity level Behavioural Style Questionnaire Howell et al. (2004) Anderson et al. (2003)Rhythmicity Behavioural Style Questionnaire Anderson et al. (2003) Howell et al. (2004)Adaptability Behavioural Style Questionnaire Howell et al. (2004), Anderson et al. (2003)
Direct observation Schwenk et al. (2007)Attention span/persistency Behavioural Style Questionnaire Howell et al. (2004), Karrass et al. (2006) Anderson et al. (2003)
Direct observation Schwenk et al. (2007)Threshold of responsiveness Behavioural Style Questionnaire Howell et al. (2004), Anderson et al. (2003)Intensity of reaction Behavioural Style Questionnaire Karrass et al. (2006) Howell et al. (2004), Anderson et al. (2003)
EEG Arnold et al. (2011)Quality of mood Behavioural Style Questionnaire Howell et al. (2004) Anderson et al. (2003)
Disappointing gift paradigm Johnson et al. (2010)Distractibility Behavioural Style Questionnaire Anderson et al. (2003) Howell et al. (2004)
Direct observation Schwenk et al. (2007)Approach/withdrawal Short Temperament Scale Reilly et al. (2009)
Behavioural Style Questionnaire Howell et al. (2004), Anderson et al. (2003)
Rothbart and Derryberry (1981) Temperament ModelInhibitory control Children’s Behaviour Questionnaire Embrechts et al. (2000), Eggers et al. (2010)Attentional shifting Children’s Behaviour Questionnaire Eggers et al. (2010)Anger/frustration Children’s Behaviour Questionnaire Eggers et al. (2010) Embrechts et al. (2000)Approach Children’s Behaviour Questionnaire Eggers et al. (2010) Embrechts et al. (2000)Motor activation Children’s Behaviour Questionnaire Eggers et al. (2010)Impulsivity Children’s Behaviour Questionnaire Embrechts et al. (2000) Eggers et al. (2010)Activity level Children’s Behaviour Questionnaire Embrechts et al. (2000) Eggers et al. (2010)High intensity pleasure Children’s Behaviour Questionnaire Embrechts et al. (2000), Eggers et al. (2010)Shyness Children’s Behaviour Questionnaire Embrechts et al. (2000), Eggers et al. (2010)Smiling/laughter Children’s Behaviour Questionnaire Embrechts et al. (2000), Eggers et al. (2010)Discomfort Children’s Behaviour Questionnaire Embrechts et al. (2000), Eggers et al. (2010)Sadness Children’s Behaviour Questionnaire Embrechts et al. (2000), Eggers et al. (2010)Fear Children’s Behaviour Questionnaire Embrechts et al. (2000), Eggers et al. (2010)Falling reactivity and soothability Children’s Behaviour Questionnaire Embrechts et al. (2000), Eggers et al. (2010)Low intensity pleasure Children’s Behaviour Questionnaire Embrechts et al. (2000), Eggers et al. (2010)Perceptual sensitivity Children’s Behaviour Questionnaire Embrechts et al. (2000) Eggers et al. (2010)Attentional focusing Children’s Behaviour Questionnaire Embrechts et al. (2000) Eggers et al. (2010)Excitatory control Children’s Behaviour Questionnaire Eggers et al. (2010)
Note. Entries in the last three columns represent findings in relation to controls.
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5.9. Approach/withdrawal
As noted previously, of the nine Thomas and Chess (1977) dimensions of temperament, approach/withdrawal is the mostimplicated in development of anxiety during childhood, because it involves shyness, inhibition and withdrawal, which aretemperament precursors of anxiety.
Based on parent reports using the Behavioural Style Questionnaire, no significant differences have been found betweenstuttering and control children’s approach scores. Additionally, Reilly et al. (2009) reported a prospective, longitudinal studyof a community cohort of 1619 two-year-old children. The study was designed to predict stuttering onset for children upto three years of age with measures prior to stuttering onset. When the children were two years old, parents completed afive-item approach scale, which is one of the five temperament dimensions of the Australian Temperament Project-Shortform for children 1–3 years (Prior, Smart, et al., 2000; Prior, Sanson, et al., 2000). This is a well validated parent reportinstrument normed for Australian children. The dimension of approach did not predict stuttering onset for the 137 childrenin the cohort who began to stutter by age 3 years 0 months.
6. Early stuttering and temperament structure studied with the Children’s Behaviour Questionnaire
The Children’s Behaviour Questionnaire is based on an alternative temperament model to Thomas and Chess (1977). Itwas developed by Rothbart and Derryberry (1981). The Questionnaire has been shown to have various levels of reliabilityand validity depending on the temperament subscale being measured (Rothbart, Ahadi, Hershey, & Fisher, 2001). Table 1contains a list of the temperament dimensions measured with this instrument. Eggers, De Nil, and van den Bergh (2009) useda Dutch version of the questionnaire for factor analysis to show that stuttering, voice disordered and control children did notdiffer with temperament structure. Twenty-eight of the stuttering children and 78 of the control children were in the agerange 3–6 years. Subsequently, Eggers, De Nil, and van den Bergh (2010) reported a similar study with the same cohort. Fifty-eight stuttering children from the original cohort (mean age 5 years 11 months) were compared with 58 matched controlsfrom the original cohort (mean age 5 years 11 months). Significant differences between the stuttering and control groups onseveral temperament factors were reported. The stuttering group had lower levels of “inhibitory control” and “attentionalshifting,” and higher scores on measures of “anger/frustration,” “approach,” and “motor activation.” A caveat to interpretingthese findings is that the latter study used a subset of the original cohort of 69 stuttering and 149 control children, and it isnot clear how or why the 58 stuttering and control children were selected. Some of these findings supported earlier reportsfrom Embrechts, Ebben, Franke, and van de Poel’s (2000) study. Using the Children’s Behaviour Questionnaire (Rothbart& Bates, 1998) they examined individual temperament dimensions of 38 stuttering children and 38 controls, matched forage, gender and parent’s education level. Their age range was 3–7 years (mean age of 5.0 years). While Embrechts et al.found that stuttering children received a higher score for ‘activity level’ and ‘impulsivity’ and lower scores for ‘attentionalfocusing’ ‘perpetual sensitivity’ and ‘inhibitory control’ they did not report significant differences for any of the remainingtemperament dimensions, including ‘anger/frustration’ or ‘approach’.
Table 1 summarises the studies reviewed above that deal with temperament and stuttering preschoolers. Because of thecaveat interpreting the Lewis and Golberg (1997) study (see Section 5.2), it was not included in Table 1.
7. Conclusions
Using the guideline that independent replication of findings makes them trustworthy, there is an inevitable conclusionto this review. For stuttering children during the preschool years, there may be some association between temperamentand stuttering during the preschool years. The guarded nature of this statement arises because of the modest scope of theresearch on which it is based, amounting to 10 publications, and because of some inconsistencies with results, as outlinedbelow.
Using the Thomas and Chess (1977) temperament dimensions, Table 1 shows that there have been some independentreplications of findings. There have been three independent replications that they have (1) lower adaptability than controls,three independent replications that they have (2) lower attention span/persistency, and (3) two independent replications thatthey have more negative quality of mood than controls. For each of these findings, one of the replications involved a directobservation paradigm to complement the Behavioural Style Questionnaire method. Additionally, the replicated findingsabove about lower adaptability and attention span/persistency for stuttering children were replicated with the findings about“inhibitory control” and “attentional shifting” respectively using another instrument, the Rothbart and Derryberry (1981)Temperament Model. Finally, there have been two independent replications that stuttering children have higher activitylevels than controls using the Thomas and Chess (1977) and Rothbart and Derryberry (1981) Temperament Models.
At present, any firm conclusion about the two hypotheses presented in this paper would be premature. Woods, Shearsby,Onslow, and Burnham (2002) and van der Merwe, Robb, Lewis, and Ormond (2011) have produced some evidence thata causal hypothesis of temperament, anxiety and early stuttering may not be justifiable. The former study administeredthe Child Behaviour Checklist (Achenbach, 1988) to parents of eight preschool children aged 2–5 years seeking stutteringtreatment. This assessment measured participant’s anxiety levels pre treatment, during treatment and post treatment.Reduced levels of anxiety, withdrawal and depression were reported by parents post treatment which suggests that thesepsychological symptoms were an effect of stuttering. This finding was supported by van der Merwe et al. who compared state
E. Kefalianos et al. / Journal of Fluency Disorders 37 (2012) 151–163 159
and trait anxiety measures in seven stuttering children and seven age and gender matched controls. The Preschool AnxietyScale (Spence & Rapee, 1999), which measures specific anxiety types and an overall anxiety score was administered toparticipant’s parents. Participants completed the KiddyCAT (Vanryckeghem & Brutten, 2002) to determine their attitudes tocommunication. Additionally, participants provided saliva samples to measure their cortisol levels, a stress hormone whichindicates an individual’s anxiety levels in various situations. Parents of stuttering and control participants reported that theirchildren exhibited similar state and trait anxiety levels. Additionally, participants demonstrated identical communicationattitudes and no significant differences were detected regarding cortisol levels either pre- or post-conversation.
Conversely, using a large sample of 45 stuttering children, aged 3–6 years and 63 age-matched controls, Vanryckeghem,Brutten, and Hernandez (2005) measured participant’s responses to the KiddyCAT (Vanryckeghem & Brutten, 2002). Stut-tering children had more negative attitudes toward their speech than controls. Some of the test items of the KiddyCAT maybe interpreted as markers of anxiety. Using that assumption, these results may be interpreted as stuttering participantsreporting more anxious characteristics than controls.
It is far from clear whether temperament is a causal factor for early stuttering and its association with anxiety or whetherearly stuttering prompts development of such anxiety. In the first instance, the majority of studies reviewed did not studycommunity populations of stuttering preschoolers but clinical populations of preschoolers. Second, participants in all but oneof the studies reviewed recruited children subsequent to stuttering onset. To explore causality optimally, research shouldproceed with a community cohort of stuttering children who are ascertained prior to stuttering onset. There has only beenone such study, and no firm conclusions can be drawn from it. It is the case that a prospective, community ascertained cohortshowed that the “approach” dimension of the Short Temperament Scale (Reilly et al., 2009) did not predict stuttering onsetfor 137 children who began stuttering prior to three years of age. Yet that study was not dedicated to the research domainof temperament and included only that dimension.
There are caveats also about temperament and the development of anxiety during the preschool years for stuttering chil-dren. Certainly, some of the replicated findings of this review suggest the existence of markers of a “difficult” temperamentfor stuttering preschoolers might foreshadow anxiety disorders: lower attention span/persistency, and intensity of reaction.But on balance the literature reviewed might be construed as unsupportive of the existence of a “difficult” temperament forstuttering preschoolers. This is because an independent study conducted with the Australian Temperament Project’s revisedversion of the Thomas and Chess model found “approach” did not predict stuttering onset.
8. Needed research
Quantitative and qualitative research directions are suggested by the results of this review. Considering the theoretical andclinical importance of the relation between temperament and early stuttering, and a potential link between temperamentand anxiety development, it is clear that the body of research about the topic needs to expand beyond its current 10publications. When that occurs, the literature will be more easily interpretable as any consistently replicated positive or nullfindings emerge. Null findings for such research would be theoretically and clinically salient, because they would show norelation between temperament, stuttering and anxiety later during life. That being the case it will be important for journaleditors to be aware of the need to avoid the Rosenthal “file-drawer problem” (1979, p. 638) and publish such null findingsshould they emerge.
Qualitatively, we suggest that research on this topic could improve by uniting the two parent report measures in subse-quent research; the Thomas and Chess (1977) Model, and the Rothbart and Derryberry (1981) model. Should that integrationoccur in the future, some of the inconsistencies observed during this review across the two models might be resolved. Clearly,the validity of those parent report tools would be enhanced with objective paradigms for exploring temperament, such asthe “disappointing gift” and noise distraction methods reviewed above.
Arguably most importantly, the hypotheses in this paper can only be optimally examined with cohorts of stutteringchildren ascertained prior to onset. Those cohorts could be examined for temperament prior to stuttering onset, and tem-peraments of the stuttering and nonstuttering members of those cohorts could be examined with repeated, longitudinalassessments during childhood. According to the authors, these seem to be the only methods by which evidence might beestablished about whether temperament is causally related to stuttering or whether temperament changes occur system-atically as a result of stuttering. Although the cost and effort of such research is considerable, the theoretical and clinicalimportance of the topic justifies such effort.
CONTINUING EDUCATION
Early stuttering, temperament and anxiety: two hypotheses
QUESTIONS
1. Which of the following temperament traits are risk factors for development of anxiety disorders?
160 E. Kefalianos et al. / Journal of Fluency Disorders 37 (2012) 151–163
a) behavioural inhibition and ‘difficult’ temperamentb) rhythmicity and cooperationc) distractibility and reactivityd) persistence and cooperation
2. The majority of previous studies examining temperament and early stuttering have had similar limitations including:a) small sample sizesb) participants recruited from clinicsc) participants recruited after the onset of stutteringd) all of the above
3. Environmental factors influence the expression of temperament traits. What proportion of individual personality variationis accounted for by environmental influences?a) 50 percentb) More than 80 percentc) 40–80 percentd) Less than 30 percent
4. Which of the following questionnaires was devised based on Thomas and Chess (1977) temperament model:a) Children’s Behaviour Questionnaireb) KiddyCATc) Behavioural Style Questionnaired) Preschool Anxiety Scale
5. Which of the following statements is correct? The ‘approach’ dimension of temperament measures:a) How a child responds to new people and situationsb) The energy a child exerts in response to a stimulusc) The amount of physical activity in a child’s behaviourd) The extent to which a child expresses positive and negative emotions
Acknowledgement
This research was supported by the Victorian Government’s Operational Infrastructure Support Program.Supplementary data associated with this article can be found, in the online version, at doi:10.1016/j.jfludis.2012.03.002.
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Elaina Kefalianos is a PhD scholar at the University of Melbourne. She is based at the Murdoch Children’s Research Institute in Melbourne. Elaina isalso a clinical speech pathologist with a particular interest in childhood stuttering.
Mark Onslow is foundation Director of the Australian Stuttering Research Centre at the University of Sydney. He is a Principal Research Fellow ofthe National Health and Medical Research Council of Australia. His core research interests are clinical trials of innovative methods for treatment ofstuttering, treatment outcome measures for stuttering, and theoretical explanations for the onset and development of stuttering.
Susan Block is a Senior Lecturer at La Trobe University in Melbourne where she co-ordinates the academic and clinical stuttering programs. She holdshonorary positions at James Cook University in Townsville, the Murdoch Children’s Research Institute in Melbourne and the Australian StutteringResearch Centre at Sydney University.
Ross Menzies is located at the Australian Stuttering Research Centre at the University of Sydney. He is a clinical psychologist with an interest in theorigins and management of anxiety, and has a private practice in Sydney. He has developed cognitive behaviour therapy packages for the treatmentof obsessive compulsive disorders and published theories of the origins of phobias. He led the Australian Stuttering Research Centre team in clarifyingthe role of anxiety in stuttering and its deleterious effects on speech rehabilitation.
Sheena Reilly is Associate Director of Clinical and Public Health at the Murdoch Childrens Research Institute, Professor-Director of Speech Pathology(The Royal Children’s Hospital) and Professor of Paediatric Speech Pathology (University of Melbourne). Sheena’s research focuses on speech andlanguage difficulties in children. She has produced over 120 publications.
