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aDepartment of Paediatrics, fSchool of Behavioural Sciences, University of Melbourne, Melbourne, Australia; bRoyal Childrens Hospital, Melbourne, Victoria, Australia;
cMurdoch Childrens Research Institute, Melbourne, Victoria, Australia; dAustralian Stuttering Research Centre, University of Sydney, Sydney, Australia; eSchool of
Psychological Science, La Trobe University, Melbourne, Victoria, Australia
The authors have indicated they have no nancial relationships relevant to this article to disclose.
Stuttering involves repeated movements and xed postures of the speech mechanism. The cumulative incidence of stuttering onset by age 3 years was higher than previ-
Stuttering may severely impair communication, leading to speech-related social anxiety ously reported. Risk factors for stuttering onset explained little of the variation in
and social phobia and failure to attain occupational potential. Although stuttering is a stuttering onset. Language delay and shyness were not associated with stuttering
common disorder, the cause remains unknown. onset.
ABSTRACT
OBJECTIVES. Our goals were to document (1) the onset of stuttering and (2) whether
specific child, family, or environmental factors predict stuttering onset in children
aged up to 3 years. www.pediatrics.org/cgi/doi/10.1542/
peds.2007-3219
disfiguring. School-aged children who stutter suffer bullying and teasing. In later life, mcri.edu.au
stuttering can lead to speech-related social anxiety and social phobia, failure to attain PEDIATRICS (ISSN Numbers: Print, 0031-4005;
Online, 1098-4275). Copyright 2009 by the
occupational potential, and severely impaired communication.2 American Academy of Pediatrics
The cause is unknown, although it is commonly believed to be a disorder of the
neural processing underpinning spoken language, with genetic and environmental
influences.3 It is generally accepted that stuttering typically begins between 2 and 4 years of age and coincides with
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advances in language development, particularly the de- ories and to underpin stuttering interventions during the
velopment of 2- to 3-word phrases. There are many preschool years.18,19 Prospective information about stut-
causal theories, but for the most part they remain un- tering incidence, antecedents and predictors, and natural
tested.4 recovery rates is urgently required to inform treatment
To date, there have been 2 prospective, community practices with preschoolers. Particularly pressing is the
studies of incidence and natural recovery. Stuttering was need to identify children in whom stuttering is and is not
examined in the 1000-family study conducted in New- likely to persist, so that differing advice (watchful wait-
castle-Upon-Tyne, England,5,6 with all children born in ing versus recommending treatment) can be correctly
that city in May and June 1947 assessed over a number targeted.
of years. Cumulative incidence up to 5 years old (ie, the This report aims to begin to address some of these
proportion of children experiencing onset by age 5) was methodologic issues by prospectively documenting the
3.5%, and 18 (42%) of 43 children recovered naturally onset of stuttering in children up to 3 years of age in a
by 6 years.6 Shortcomings of these data7 include the fact large, community-ascertained cohort of Australian chil-
that stuttering was identified by health visitors rather dren. Specifically, we aimed to describe the cumulative
than by speech pathologists, and it was not clear how incidence, age, and characteristics of stuttering at onset,
long after stuttering onset the children were assessed. and to determine any child, family, or environmental
The cumulative incidence of stuttering in 1042 pre- factors that might predict stuttering onset.
schoolers up to 3 years old born between 1990 and 1991
on the Danish island of Bonholm was 5%.8 By 5 years of METHOD
age, 71% were reported to have naturally recovered.
Limitations of this study included the fact that the chil- Overview of the Early Language in Victoria Study
dren were not assessed until their third birthday, and the The children in this study were participants in a larger
presence of stuttering was not established before the age study of early language development, the Early Lan-
of 3 years. guage in Victoria Study (ELVS), conducted in Mel-
Many antecedents and risk factors have been thought bourne, Australia (population: 3.6 million), and de-
to be implicated in stuttering onset. There is ongoing scribed in detail elsewhere.20,21 Briefly, ELVS is a
debate about the relationship between language and prospective observational longitudinal study commenc-
stuttering.9 Whereas some studies revealed that lan- ing at 8 months, followed by repeated measures at each
guage learning difficulties were not associated with stut- birthday throughout the preschool and into the primary
tering,10 others demonstrated that children who stutter school years. Infants were recruited from 6 of metropol-
have lower language proficiency than children who do itan Melbournes 31 local government areas (LGAs). The
not stutter.11 In addition, they are reported to differ on Disadvantage Index derived from the census-based So-
various temperament characteristics,12 including shy- cio-economic Indexes for Areas (SEIFA),22 represents
ness,13 sensitivity,14 adaptability,14 and vulnerability.15 attributes such as low income, low educational attain-
Some have proposed that these temperament character- ment, and high unemployment. The index was used to
istics play a role in the etiology of stuttering.13 To date, stratify the 31 LGAs into 3 tiers, after which 2 noncon-
however, no study we are aware of has measured either tiguous LGAs were selected from each tier to ensure the
language or temperament before stuttering onset. There- study sampled from geographic areas across the spec-
fore, it is not clear whether these factors are associated trum of disadvantage/advantage.
with stuttering onset and, if they are, whether they Between September 2003 and April 2004, maternal
precede or develop as a result of the stuttering. and child health nurses were asked to consecutively
Our review of the literature also identified several approach parents of all infants at their universal
other potential variables thought to be implicated in 8-month visit (attended by over 80% of all Melbourne
stuttering onset. Included were prenatal and perinatal infants), followed by a formal recruitment process. Chil-
factors, birth order, socioeconomic status, parent-child dren were excluded if they had an obvious congenital
relationships, traumatic experiences in early childhood, (eg, Down Syndrome) or developmental (eg, cerebral
and growth and neurologic development.5,16 Again, few palsy) problem or other serious intellectual or physical
of these factors have been measured before stuttering disability diagnosed by 8 months of age. Participants
onset, and the reported associations are therefore not were also excluded if their parents did not speak and
well supported. We identified 1 exception, a study of 93 understand English sufficiently for recruitment and
children in the Netherlands17 in which language devel- completion of questionnaires. The final sample was 1911
opment measured before any of the children started to children. Parents reported on early communication and
stutter did not predict stuttering onset. The only predic- language development at each annual wave, and a broad
tors associated with onset were increased child speech range of other child, family, and environmental mea-
rate and reduced maternal language complexity. Chil- sures was also collected.20,21
dren in the Dutch study were selected on the basis that
at least 1 parent stuttered, and the results may not Procedure
therefore generalize to the large numbers of children ELVS participants were eligible for inclusion in this stut-
who stutter without an overt family history. tering study once they had returned their 2-year-old
Existing knowledge about stuttering onset has been questionnaires. Parents were then sent a letter of invi-
used to inform the development of causal pathway the- tation explaining the aims and procedures of the ELVS
Predictor Measures
RESULTS
Predictor measures were largely drawn from the ques-
tionnaire completed by parents as children turned 2 The Sample
years of age. Raw (quantitative) vocabulary scores were Figure 1 summarizes the participant flow between ELVS
calculated from the MacArthur-Bates Communicative and the Stuttering Study. At 2 years of age, 57 of the
Development Inventories (CDI), with children below the original 1911 ELVS participants were unavailable (with-
10th percentile for vocabulary production identified as drawn, lost contact, or moved out of Victoria), and an
late talkers.24,25 The development of communicative be- additional 235 parents contacted us and opted not to
haviors up to 24 months of age was summarized using participate in the study, leaving 1619 participants (85%
the Communication and Symbolic Behavior Scales of the original sample). Table 1 compares the study
(CSBS) Infant-Toddler Checklist26; standardized Total participants with the nonparticipants, with larger pro-
Scores (normative mean: 100, SD: 15) were calculated portions of children whose mothers had a degree or
according to the manual. Parents completed the 5-item postgraduate qualification among the participants than
Approach/Withdrawal scale, 1 of the 5 temperament the nonparticipants.
dimensions that comprise the Australian Temperament A total of 158 (9.8%) of 1619 parents called to report
Project-Short form suitable for toddlers aged 1 to 3 years the onset of stuttering by 3 years of age. The presence of
(Toddler version).27 Each item was rated on a scale from stuttering was then confirmed in 137 children (8.5%
1 (almost never) to 6 (almost always) and summed to [95% confidence interval (CI): 7.2%9.9%]) by a
provide a total score (maximum score: 30).28 Shy/with- trained research assistant during the initial home visit.
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ELVS cohort
1911 subjects
Stuttering-study
FIGURE 1 Lost to follow-up participants Opted out
Flowchart of ELVS participants and relationship to the stuttering 57 subjects 1619 subjects 235 subjects
study.
Gender
Proportion
Male, n (%) 824 (50.9) 142 (48.6) 0.06
Female, n (%) 795 (49.1) 150 (51.4)
Twin birth, n (%) 42 (2.6) 11 (3.8) 0.04
Premature birth, n (%) 47 (2.9) 12 (4.1)
Birth weight, mean (SD), kg 3.4 (0.5) 3.4 (0.6)
0.02
Birth order
First child, n (%) 820 (50.8) 134 (46.5)
Second child or more, n (%) 795 (49.2) 154 (53.5) 0.00
Family history 0 10 20 30 36
Age, mo
No problem, n (%) 1213 (74.9) 222 (76.0)
Speech/language/reading problems 312 (19.3) 60 (20.5) FIGURE 2
only, n (%) Proportion of study participants who stuttered by a given age. Data shown for children
Stuttering problem, n (%)c 94 (5.8) 10 (3.4) up to 3 years of age.
SEIFA disadvantage score, mean (SD) 1037 (61) 1033 (60)
Mothers education level
Did not complete year 12, n (%) 359 (22.4) 72 (25.2) when the child was stringing 3 or more words together
Completed year 12, n (%) 628 (39.2) 130 (45.5) (133 of 137 [97.1%]). In more than half the children,
Degree/postgraduate qualication, n (%) 615 (38.4) 84 (29.4) stuttering started suddenly; 51 parents (37.2%) reported
Maternal mental health score, mean (SD) 3.2 (2.9) 3.1 (2.9)
stuttering commenced during a period of 1 day, and 17
a The number of participants ranged from 1469 to 1619.
b The
parents (12.4%) over 2 to 3 days. In some children,
number of nonparticipants ranged from 242 to 292.
c Children with a family history of stuttering may also have reported family histories of speech/ stuttering was reported to emerge more slowly: 1 to 2
language and/or reading problems. weeks (37 [27.0%]), 3 to 4 weeks (19 [13.9%]), and
during a period of 5 weeks in 8 children (5.8%). Four
parents (2.9%) were unsure about the time it took for
The panel agreed that there were 21 borderline chil- stuttering to emerge. For 1 case, the data were missing.
dren in whom stuttering reports were ambiguous, and Family histories were obtained from all the children
these children were classified as nonstuttering for the in the ELVS cohort (Table 1). A more detailed family
main analyses. The median age of onset was 29.9 history of stuttering and related communication prob-
months (interquartile range [IQR]: 27.0 33.1; range: lems was elicited after onset from the families of children
12.0 36.9) and of parent telephone to report stuttering who stuttered, which revealed a specific family history
onset was 31.8 months (IQR: 29.0 34.9; range: 24.9 of stuttering in 71 (51.8%) parents of children who
42.4). The median period from reported onset to tele- started to stutter. Between 20% and 30% reported family
phone contact was 1.4 months (IQR: 0.6 3.2; range: histories of other related conditions including speech prob-
0.113.1), and between reported onset and the home lems, problems at school, difficulty with math, and diffi-
visit was 2.1 months (IQR: 1.0 3.9; range: 0.218.1). culty with reading and/or writing. Less than 20% reported
Figure 2 plots the proportion of the ELVS cohort who problems in a variety of domains including epilepsy, intel-
had stuttered by any age up to 3 years. The constant lectual impairment, language problems, motor difficulties,
slope from 25 months emphasizes that there was no and attention and concentration problems.
obvious shorter age window during which stuttering The majority of parents described the nature of their
characteristically commenced. Eleven parents reported childs stuttering as episodic (125 [91.2%]) rather than
that their children started to stutter before 2 years of age. continuous. The most commonly reported stuttering be-
Most parents reported that they first noticed stuttering havior was whole word repetition (97 [71%]). Table 2
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To our knowledge, this is the largest epidemiological majority of those with a family history of stuttering were
study of early stuttering onset to date, and the first to unaware of it (ie, this could not have been the reason for
recruit a cohort and measure putative risk factors before self-selection into either ELVS or the stuttering study).
onset and to incorporate reliable, expert diagnoses. Finally, the study successfully captured families from
Some of our findings confirm previous reports about across the entire socioeconomic spectrum to quantify
early stuttering. For example, boys were more likely to internal predictor-outcome relationships, for which a
stutter than girls. In addition, we confirmed the reported representative sample is not essential.
rapid onset of stuttering, its episodic nature, and the This report includes onset only up to 3 years. The
prominence of repeated words at onset. Consistent with cumulative incidence of stuttering onset by 4 years will
existing retrospective evidence, the majority of parents be higher. We may have missed some cases because
reported that stuttering onset occurred during the 2- or parents elected not to call or stuttering duration was so
3-word stage of language development. brief that it did not elicit a parent report. As we continue
Reports of a family history of stuttering varied accord- our observation of this cohort into the preschool years,
ing to when the information was obtained. Before stut- we will be able to ascertain any missed cases of persistent
tering onset, 12 (8.8%) of 137 families with stuttering stuttering that were not notified by parents.
children reported a family history of stuttering. When Four factors were significantly associated with stut-
family history data were collected after stuttering onset, tering onset at the 5% level. The association with gender
many more of these families recalled a family history (71 is the best-recognized factor in the literature, but the
of 137 [51.8%]). Similar findings have been reported in associations with both twinning and maternal education
studies involving screening for hearing impairment,34 were surprising and, as noted earlier, should be inter-
and the finding may simply reflect parents being un- preted cautiously. Mothers with a degree or postgradu-
aware of the risk factor at the time of questioning (eg, ate qualification were more likely to have children that
before onset). The finding, however, does raise concern stuttered by 3 years. It could be that well-educated
about using a potential risk factor such as family history mothers were more likely to be aware of (and therefore
to identify children at risk of stuttering onset. to report) stuttering onset. It is also possible that the
The high cumulative incidence of stuttering onset of association with maternal education may be an artifact;
8.5% by 3 years old may seem surprising. We are con- stuttering onset typically occurs with the development of
fident about the estimate, because the 95% CIs were 3-word combinations, and such combinations may sim-
narrow (7.2%9.9%). The response rate was high (85% ply occur earlier in children of more highly educated
of parents in the original ELVS cohort), and stuttering mothers. Against this, vocabulary scores at 2 years were
was rigorously verified by experts using strict criteria, so significantly associated with onset of stuttering but not
we can be reasonably confident that both the majority of with maternal education,21 and clearly this unexpected
children in the population who started to stutter before association requires additional elucidation as the chil-
age 3 were identified and that the high rate was not dren mature. Our results do not support the premise that
because of false-positives. Finally, the high incidence is language problems are associated with stuttering onset
plausible: it is likely we have identified stuttering that but that communication skills and vocabulary are more
would have been missed in previous studies, either be- highly developed in children who start to stutter by 3
cause it was mild or because it was short-lived. In the years of age.
Newcastle-Upon-Tyne study,5 for example, stuttering Of clinical and theoretical interest is our finding that
sometimes seemed to last only a few months. the dimension of temperament we measured did not
Whenever a major study reports more extreme predict stuttering onset children who started to stutter
findings than previous research, the possibility of bias were neither more shy nor more outgoing than their
must be considered. We acknowledge that our cumula- peers who did not stutter. Importantly, our measures
tive incidence estimate may be somewhat inflated, be- were taken before stuttering onset by using a well-vali-
cause disadvantaged parents and infants were underrep- dated parent report instrument normed on Australian
resented in this cohort (a common limitation in children.27 Temperament is regarded as a relatively sta-
longitudinal population studies), whereas children of ble trait, but is nonetheless receptive to environmental
highly educated mothers were both more likely to stut- experiences.27,35,36 It is possible that when children begin
ter and to be included in the study. However, this does to stutter they become more vigilant and more reticent,
not alter our conclusions. Australian census data on as described by Anderson et al.12 Alternatively, it may be
mothers of children born in 2001 indicate that 43.8% that parents, who are the ones that typically fill out
did not complete school, compared with only 22.4% in temperament questionnaires, perceive their children to
our stuttering study. When we applied inverse probabil- be so simply because they stutter. Regardless, our results
ity weights accordingly as a sensitivity check, the esti- do not suggest that innate shyness has a causal role in
mated cumulative incidence fell from 8.5% to 7.5% stuttering.
(95% CI: 6.2% 8.8%), which is still considerably higher Additional study of this cohort will enable estimation
than previously reported. Other contributors to selection of the cumulative incidence of stuttering onset during
bias seem unlikely: the ELVS cohort was not originally the preschool years and identify whether stronger pre-
recruited to a study of stuttering, recruitment occurred dictors emerge with stuttering onset after 3 years of age.
well before any concern about stuttering onset might We will also be able to estimate the natural recovery rate
have arisen, and our findings indicate that the great during the preschool years and provide a much-needed
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Development Inventories: Users Guide and Technical Manual. San Equations: an orientation. Am J Epidemiol. 2003;157(4):
Diego, CA: Singular Publishing Group; 1993 364 375
25. Fenson L, Pethick SJ, Renda C, Cox JL, Dale PS, Reznick JS. 31. Hosmer D, Lemeshow S. Applied Logistic Regression. New York,
Short-form versions of the MacArthur Communicative De- NY: Wiley; 2000
velopment Inventories. Appl Psycholinguistics. 2000;21(1): 32. Stata Corporation. Stata Statistical Software [computer program].
95116 Release 9.2. College Station, TX: Stata Corporation; 2005
26. Wetherby A, Prizant B. Communication and Symbolic Behaviour 33. Royston P, Ambler G, Sauerbrei W. The use of fractional poly-
Scales. Baltimore, MD: Paul H. Brookes Publishing Co; 2002 nomials to model continuous risk variables in epidemiology.
27. Prior M, Sanson A, Oberklaid F. The Australian Temperament Int J Epidemiol. 1999;28(5):964 974
Project. In: Kohnstamm GA, Bates JE, Rothbart MK, eds. Tem- 34. Russ R, Rickards F, Poulaki Z, Barke M, Saunders K, Wake M. Six
perament in Childhood. Chichester, Sussex, United Kingdom: year effectiveness of a population based two tier infant hearing
Wiley; 1989:537554 screening programme. Arch Dis Child. 2002;86(4):245250
28. Prior M, Sanson A, Smart D, Oberklaid F. Pathways From 35. Sanson A, Hemphill S, Smart D. Connections between temper-
Infancy to Adolescence: Australian Temperament Project ament and social development: a review. Soc Dev. 2004;13(1):
19832000. Melbourne, Australia: Australian Institute of 142170
Family Studies; 2000 36. van den Boom D. Do first-year intervention effects endure?
29. Kessler R, Mroczek D. Final Version of Our Non-specific Psycho- Follow-up during toddlerhood of a sample of Dutch irritable
logical Distress Scale [memo]. Ann Arbor, MI: Institute for Social infants. Child Dev. 1995;66(6):1798 1816
Research; 1994 37. Craig A, Hancock K, Tran Y, Craig M, Peters K. Epidemiology
30. Hanley JA, Negassa A, Edwardes MD, Forrester JE. Statistical of stuttering in the community across the entire life span. J
analysis of correlated data using Generalized Estimating Speech Lang Hear Res. 2002;45(6):10971105
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