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Evidence-Based Treatment and Stuttering—Historical Perspective



To illustrate the way in which both fluency shaping (FS) and stuttering management (SM) treatments for developmental stuttering in adults are evidence based. A brief review of the history and development of FS and SM is provided. It illustrates that both can be justified as evidence-based treatments, each treatment seeking evidence of a different kind: FS seeks evidence concerning treatment outcome, and SM seeks evidence concerning the nature of the stutter event. Although outcome evidence provides the principal support for FS, support for SM comes principally from a cognitive learning model of defensive behavior as applied to the nature of the stutter event. Neither approach can claim anything like uniform success with adults who stutter. However, self-management and modeling are strategies common to both approaches and have shown consistently positive effects on outcome. It is argued that both strategies merit additional treatment efficacy study. Cognitive behavior theory may provide a useful framework for this research.
Evidence-Based Treatment and
StutteringHistorical Perspective
Purpose: To illustrate the way in which both fluency shaping (FS) and stuttering
management (SM) treatments for developmental stuttering in adults are evidence
Method: A brief review of the history and development of FS and SM is provided. It
illustrates that both can be justified as evidence-based treatments, each treatment
seeking evidence of a different kind: FS seeks evidence concerning treatment outcome,
and SM seeks evidence concerning the nature of the stutter event.
Conclusion: Although outcome evidence provides the principal support for FS, support
for SM comes principally from a cognitive learning model of defensive behavior as
applied to the nature of the stutter event. Neither approach can claim anything like
uniform success with adults who stutter. However, self-management and modeling are
strategies common to both approaches and have shown consistently positive effects on
outcome. It is argued that both strategies merit additional treatment efficacy study.
Cognitive behavior theory may provide a useful framework for this research.
KEY WORDS: stuttering, treatment, evidence-based practice
he purpose of this article is to illustrate, through a historical pers-
pective, how both stuttering management (SM) and fluency shaping
(FS) are evidence-based treatments. They share treatment proce-
dures that not only have shown consistent positive effects on outcome but
that suggest the causal processes involved and a direction for future treat-
ment efficacy studies.
The modern era for research and treatment of stuttering began about
1925 with the establishment at the University of Iowa of the first truly
academic program for the study of speech disorders (Johnson, 1955b).
Since that time, two basic behavioral management approaches for treat-
ing older children and adults who stutter have been practiced. Both focus
on altering behavior associated with the occurrence of stutter events
but in quite different ways. One has come to be identified as fluency shap-
ing (FS). It seeks to reduce or eliminate the occurrence of stutter events.
The other, usually identified as stuttering management (SM), seeks to nor-
malizereactionstotheoccurrence of those events.
More specifically, FS teaches the person who stutters to produce
fluent speech in a manner that prevents the fluency disruptions that
trigger stuttering reactions. Its goal is stutter-free speech. SM, on the other
hand, teaches the person who stutters to react to fluency disruptions
calmly, without unnecessary tension or struggle. Rather than stutter-free
speech, its goal is speech that, although disfluent, is free of unnecessary
David Prins
University of Washington
Roger J. Ingham
University of California, Santa Barbara
Journal of Speech, Language, and Hearing Research Vol. 52 254263 February 2009 D American Speech-Language-Hearing Association
effort. It is not uncommon for treatment programs to
combine the two approaches (Guitar, 2006).
FS and SM have come to be distinguished in the lit-
erature as evidence-based (FS) and assertion-based (SM)
treatments (see Blomgren, Roy, Callister, & Merrill, 2005;
Onslow, 2003; Ryan, 2006).
The effect o f th is distinc-
tion clouds the essence of the two treatments as well as
a meaningful discussion of e ach in relation to the na-
ture of, and future research concerning, evidence-based
practice (EBP).
FS and SM are essentially products of the eras in
which they gained stature and of learning theories then
in vogue. From that perspective, both conform to Sacket,
Rosenberg, Gray, Haynes, and Richardsons (1996) def-
inition of EBP: Evidenced based medicine is the con-
scientious, explicit, and judicious use of current best
evidence in making decisions about the care of individ-
ual patients. The practice of evidence based medicine
means integrating individual clinical expertise with the
best available external clinical evidence from systematic
research (p. 71).
Each approach, pertinent to its concepts of nature
and treatment of stuttering, has sought support from a
different kind of evidence. FS, an outcome-driven treat-
ment, has sought outcome evidence even as it eschewed
theory. SM, a theory-driven treatment, has done the op-
posite. Accordingly, FS has asked what procedures re-
duce the occurrence of stutter events, whereas SM comes
from a tradition that has asked what procedures are jus-
tified by the nature of those events. A closer look at each
reveals these characteristics.
Stuttering Management (SM)
Nature of Stutter Events
For about 20 years beginning in the 1930s, theory of
learning was dominated by the ideas of Clark Hull and
Kenneth Spence (see Hergenhan & Olson, 2004). From
their research, they concluded that behavior was rein-
forced due to drive reduction (i.e., need satisfaction) and,
further, that anxiety reduction, in particular, served as a
powerful reinforcer. This was the climate into which
Wendell Johnson introduced ideas about stuttering that
were, quite simply, revolutionary. Among them was his
concept of the moment of stuttering”—what we speak of
today as the stutter event (Johnson, 1933; Johnson &
Knott, 1936).
According to Johnson (1955b), the concept of the
moment of stuttering almost immediately suggested the
feasibility of measuring the amount of stuttering and
the variations in it by the rather obvious means of count-
ing the moments of stuttering in systematically obtained
samples of speech (p.14).Thenatureof thethe moment,
he believed, would reveal the nature of the disorder and
the pathway to appropriate treatment. Thereupon, he
and his colleagues launched an unprecedented program
of research; make no mistake about itJohnson was a
data collector (see Johnson & Leutenegger, 1955).
The research consisted, to a large extent, of studies
concerning the variation in stutter event occurrence un-
der different experimental conditions involving oral read-
ing and spontaneous speech. Results led to discoveries
concerning stutter event distribution in sentences and on
different parts of speech; the increase in their occurrence
in response to threat of punishment; and the adapta-
tion, spontaneous recovery, and consistency phenomena
(Johnson & Leutenegger, 1955).
From this work, Johnson (1955b) found that stut-
teringI behaves like a response made to identifiable
stimuli or cues with a very considerable degree of con-
sistency and predictability (p. 16). He concluded that
such cues I function as reminders, and so as storm sig-
nals, warning of danger ahead (p. 23). In other words,
stuttering was a reaction to cues contained in the act of
speechcues that, as a result of environmental conse-
quences, had acquired the capacity to threaten.
Influenced by, and in harmony with, the ideas of
Hull and Spence, Johnson (1955b) came to view stut-
tering as an anxiety-motivated avoidant response that
becomes conditioned to the cues or stimuli associated
with its occurrence (p. 23). He maintained that what
these stimuli meant to the speaker, the threat of stut-
tering to follow, was the key to sustaining the behavior.
In Johnsons words, there is something peculiarly lack-
ing in those concepts of habit or conditioning which fail
to stress dulyIthe meaningful aspects of the stimuli
(Johnson, 1955a, p. 441).
Johnson believed that neither of the traditional
theories of stutteringone which attributed it to a phys-
ical fault and the other which viewed it as a symptom of
an unstable or turbulent personalitywas based on suf-
ficient information about the ways in which stuttering
behaves, prompting him to regard stuttering as learned
behavior, and to investigate it, theorize about it, and treat
it clinically, as such (Johnson, 1955b, p. 18). With that
conclusion, Johnson had contributed the first true behav-
ioral model of stutteringa model, according to Bandura
(1969), that defines behavior as the result of t he way
the individual has learned to cope with environmental
and self-imposed demands (p. 10).
During the Johnson era, research concerning stut-
tering was dominated by interest in theory and, more
specifically, in his ideas concerning the nature of the
stutter event. Even studies of treatment were concerned
Stuttering management has sometimes been referred to as tradition-
based or the Iowa tradition, and fluency shaping as research-based or the
behavioral tradition (Bothe, 2003).
Prins et al.:
Stuttering Treatment History 255
less about outcome, per se, than about whether outcome
evidence supported theoryand, hence, provided justi-
fication for the SM approach to treatment (see, e.g., Oxtoby ,
1955; Sheehan, 1951; Sheehan & Voas, 1954). Sheehan and
Voass conclusion from an experiment comparing treat-
ment techniques is typical: I the obtained experimental
data support the theory of stuttering as an approach-
avoidance conflict (Sheehan & Voas, 1954, p. 279).
Likewise, Johnson (1939) was more concerned about
the ideas of treatment, those based on the nature of the
stutter event, than he was about the techniques for
changing speaking performance: One need not be too
concerned over the question of how one is to change overt
behavior, he said (p. 172); and later, he said, It is as a
listener, a perceiver, an evaluator, quite as much
probably, in fact, far more thanas a speaker that the
person who stutters is to be treated (Johnson, 1957,
p. 913). In that sense, Johnson anticipated a tenet of
cognitive behavior therapy: [O]utcomes change behav-
ior in humans largely through the intervening influence
of thought (Bandura, 1977b, p. 18).
To Johnson, the critical factor in stuttering was not
what he regarded as normal disruptions of fluency, but
what these disruptions had come to mean to the speaker
that, in turn, motivated his reactions (i.e., stuttering)
to the expectation or experience of their occurrence. With
this idea, Johnson gave birth to the two-component view
of stutter events: (a) an anticipation or experience of
fluency disruption and (b) the speakersreaction.Al-
though the sources of fluency disruption have been a
matter of research a nd spec ulation sin ce th e Johnson
era (see, e.g., Brown, Ingham, Ingham, Laird, & Fox,
2005; Ludlow & Loucks, 2003; Perkins, Kent, & Curlee,
1991; Postma & Kolk,1993; Smith & Kelly, 1997;
Wingate, 1988; Zimmermann, 1980), it remains widely
believed that what we observe as stuttering results
from the nature of speaker reactions to the sensation of
disrupted fluency (Bloodstein, 1997; Nudelman, Herbrich,
Hoyt, & Rosenfield, 1991; Perkins et al., 1991; Postma &
Kolk, 1993; Prins, 1997; Van Riper, 1990; Wingate, 1988,
Zimmermann, 1980).
Evidence gradually accumulated that did not sup-
port a relationship between anxiety and stuttering (see,
e.g., Gray & Brutten, 1965; Reed & Lingwall, 1976;
Ritterman & Reidenbach, 1975), and Oliver Bloodstein,
taking this into account, modified the two-component
concept. He defined stuttering as an anticipatory strug-
gle reaction (Bloodstein, 1995, p. 404). The anticipatory
struggle, according to Bloodstein, is a response to cues
that have come to arouse the expectation of fluency
failurein other words, a response to what the cues mean
(Bloodstein, 1997). The struggle reaction is learned and
may occur in response to fluency failures that arise from
a variety of different sources, but, in contrast to findings
by Johnson, does not require anxiety and is not nec-
essarily an avoidance act. As viewed by Bloodstein, the
observable reactions of stuttering are essentially what
contemporary cognitive learning theory calls defensive
behavior—“activated by events which become threatening
through association with painful experiences (Bandura,
1977b, p. 60). What the cues mean motivat es and sustains
defensive reactions. Reflex-like in their quickness, they
may come to seem involuntary (Bandura, 1977b, 1986).
Bloodsteins version of the two-component explanation
of stutter events preserves Johnsons idea about fluency
disruptions as storm signalswarning of danger ahead,
and it undergirds SM treatments today (Prins, 1984, 1997).
Nature of Treatment
It remained for Van Riper, based on a revised con-
cept of the first component of the stutter event, to de-
velop the treatment procedures we now associate with
SM. Although Johnson maintained that stuttering speak-
ers learned to react abnormally to the kind of fluency dis-
ruptions all speakers experience, Van Riper believed the
fluency disruptions that preceded stuttering reactions
resulted from a breakdown of the speech motor system.
In his words, a stuttering behavior consists of a word
improperly patterned in time and the speakers reaction
thereto (Van Riper, 1971, p. 15).
Van Riper shunned the idea of treatments based on
fluency enhancement procedures that measure success
in terms of the decreased number of spasms in the stut-
terers speech (Van Riper, 1937b, p. 149). Such treat-
ment, he maintained, fails to target what accounts for
most of the abnormality in stuttering, the learned and
modifiable struggle or avoidance behavior. Moreover,
fluency disruptions, though they might be masked tem-
porarily by fluency-inducing procedures, are not (because
they result from inherent failures of the speech motor
system) amenable to permanent elimination (Van Riper,
1937b, 1957, 1990). In a similar vein, Johnson decried the
notion of teaching the stutterer to talk without stutter-
ing, or to speak perfectly, believing that this would lead
to speech that was grotesque due to its artificiality
(Johnson, 1946).
From the beginning, Van Riper based SM procedures
quite specifically on findings from his own research:
(a) anticipatory breathing patterns, rehearsal movements,
and levels of tension appeared to pre-model behavior
during actual stutter events (Van Riper, 1936); ( b) threat
of penalty for stuttering increased its occurrence (Van
Riper, 1937a); (c) stuttering speakers could predict the
duration of stutter events (Milisen & Van Riper, 1934);
and (d) they could perform new preparatory sets at the
outset of stutter events that seemed to generalize to
other similar, though not identical, occurrences (Van Riper,
256 Journal of Speech, Language, and Hearing Research Vol. 52 254263 February 2009
1937b). In fact, a new preparatory set became the goal
for Van Ripers SM approach. It was to replace old stut-
tering reactions with a new response, one that would
start from a state of quiescence, initiate air flow sim-
ultaneously with the speech attempt, and produce the
first sound with movement leading directly into the
succeeding soundI a speech attempt such as a nonstut-
terer might make (Van Riper, 1947, p. 359).
For more than half a century, Van Riper worked to
develop, evaluate, and justify a system of procedures to
teach adults who stutter to explore, calm, and modify
their abnormal reactions to fluency failure (see Van Riper,
1937b, 1947, 1954, 1957, 1958, 1973, 1990). More so than
Johnson, Van Riper emphasized performance change
through the mastery of motor reactions during the stutter
event. In turn, this would lead to necessary changes in
attitude and beliefs. In this way, Van Riper presaged the
view of cognitive behavior therapy: Change is mediated
through cognitive processes, but the cognitive events are
induced and altered most readily by experiences of mas-
tery arising from successful performance (Bandura,
1977b, p. 79).
In all this time Van Riper, and others who espoused
SM treatment, paid little attention to developing ex-
plicitly replicable procedures or quantitative measures
of outcome (Van Riper, 1973). In fact, as noted, the very
nature of SM caused its proponents to distrust outcome
evidence based on stuttering frequency counts.
Fluency Shaping (FS)
Nature of Stutter Events
As the 1950s drew to a close, Hull and Spences ideas
about learning fell out of favor. They were replaced by
the ideas of B. F. Skinner that would become the founda-
tion for designing and interpreting behavioral experiments
during the next decade. According to Skinner , It is possi-
ble that the most rapid progress toward an understanding
of learning may be made by research that is not de-
signed to test theories. An adequate impetus is supplied by
the inclination to obtain data showing orderly changes char-
acteristic of the learning process (Skinner, 1950, p. 215).
Precisely in this spirit, Flanagan, Goldiamond, and
Azrin (1958, 1959) conducted and interpreted two ex-
periments in which an aversive stimulus ( loud noise or
shock), or its termination, was a consequence for stut-
tering or nonfluency . They concluded that breaks, pauses,
repetitions and other nonfluencies can be considered op-
erant responses I controlled by ensuing consequences
(1959, pp. 979980). In this paradigm, there was no need
for theory concerning the nature of stutter eventsit was
enough to show that their occurrence could be brought
under stimulus control. These early studies provided,
though indirectly, the foundation for FS as it is practiced
today (see Ingham, 1984). For stuttering, a new behav-
ioral revolution was on its way: the era of radical be-
haviorism (Bandura, 1977b).
Nature of Treatment
After Flanagan, Goldiamond, and Azrin (1958, 1959),
the occurrence of stutter events was considered sufficient
to define the disorder, and elimination of those events
the treatment goal of stutter-free speechwas validated.
Outcome evidence, not evidence to support theory, would
justify FS treatments. If certain procedures reliably re-
duced or eliminated the occurrence of stuttering in the
laboratory, they were legitimate candidates for clinical
application. Accordingly, scores of studies followed to eval-
uate the effects on stuttering frequency of consequential
stimuli (Ingham, 1984). Far more important, however,
was the shift toward applying the principles of scientific
method to treatmentthe development of replicable pro-
cedures that could be tested for their efficacy using re-
liable and valid measures of the problem behavior. An
additional and important contribution was the use of
experimental time-series treatment formats that lent
themselves to ongoingeven real-timetreatment eval-
uation (see Yates, 1970).
In a pivotal study, Goldiamond (1965b) discovered,
almost by accident, that prolonged, stutter-free speech
was a natural accompaniment to delayed auditory feed-
back (DAF) and, further, that the prolonged speech pat-
tern, while remaining stutter-free, could be shaped into
more normal-sounding fluency. As Goldiamond (1965b)
described it, Where the new prolonged behavior is main-
tained without the delayed feedback, the reading rate
may now be speeded up, by machine control of the pre-
sentations, through appropriately programmed steps to
normal and supernormal rates (p. 142). Goldiamonds
laboratory studies appeared to give scientific credibility
to an age-old fluency-inducing technique, variations of
which have become the cornerstone of FS treatments.
Referred to collectively as prolonged speech, these tech-
niques include, among others, prolonged, continuous
phonation, slow articulatory movements, easy voice, and
articulatory onsets. However, what then emerged from
this pivotal study was unfortunate: There appeared a
virtual plethora of variants of prolonged speech strat-
egies that were mainly unreplicable and were subjected
to questionable investigations for their efficacy (see
Ingham, 1984, 1990). Remarkably, only recently have
serious attempts been made to translate the hypothesized
critical components of prolonged speech into replicable
and testable therapy procedures (Hillis, 1993; Hillis &
McHugh, 1998; Ingham et al., 2001).
Goldiamonds (1965b) study was noteworthy in an-
other way: It was the first to introduce a self-control (or
Prins et al.: Stuttering Treatment History 257
self-management) procedure to stuttering treatment.
This consists in training S to recognize those behaviors
of his which he wants to modify. Rather than telling him
to modify themI he is trained in the experimental ana-
lysis of behavior, and also in the variables which main-
tain it, or which he can recruit to modify it (Goldiamond,
1965b, p. 153). As we point out in the paragraphs that fol-
low, Goldiamonds introduction of self-management an-
ticipated the discovery of what we are coming to view as a
strategy that might be critical to the success of both FS
and SM approaches.
The ultimate concern of FS was with process and out-
come, not theory. As a consequence, FS has been charac-
terized by a clearly stated treatment goalstutter-free,
natural-sounding speech; carefully described and repli-
cable procedures; performance-contingent progress during
treatment; and the quantitative measurement of process
and outcome. Like SM, FS grew from laboratory studies of
the effect on stuttering frequency of different experimental
conditions. But, unlike SM, it was based on an approach
to treatment that relied on the application of experimen-
tal method to the treatment process (Ingham & Andrews,
1973a). This important paradigm shift placed new empha-
sis on the development of measurement and measurement
procedures (Cordes & Ingham, 1994). These characteris-
tics were a perfect fit for the age of accountabilityanat-
ural outgrowth of the new behaviorism.
FS and SM Today
FS and SM remain the principal behavioral ap-
proaches for the treatment of stuttering. Both are evi-
dence basedone on treatment process and outcome, the
other on a behavioral model for the nature of the stutter
event. Each relies primarily on the evidence it has always
sought and accepted. What does that evidence show?
In comparison with SM, outcome studies of FS treat-
ments (including those based on response-contingent
stimulation principles) abound in the literature (Bothe,
Davidow, Bramlett, & Ingham, 2006). On balance, they
show that FS, although it may be an effective treatment
for some adults who stutter, is clearly not for all. The age-
old problems re main : post-t reatment relapse and high
treatment dropout rates (Bothe et al., 2006; Perkins,
1992). At the same time, though, it has revolutionized
treatments for children who stutter. In this respect, it has
replaced one of the sad legacies of Johnsons commitment
to theorythat direct treatment of stuttering in children
was to be discouraged.
SM, on the other hand, offers scant outcome evi-
dence to support its effectiveness (Blomgren et al., 2005;
Bothe et al., 2006) that must be adduced largely from
studies of defensive behavior treatments reported in the
cognitive learning literature (Bandura, 1977a, 1986). As
a consequence, SMs justification relies today, as it has
since the beginning, on the widely accepted two-component
model of the stutter event and the belief that modifica-
tion of second-component behavior, the defensive reac-
tions to perceived fluency disruption, is the appropriate
objective of treatment (Prins, 1997). It is in this sense
that SM conforms to Banduras (1969) dictum: The de-
velopment of principles and procedures of behavioral
change is largely determined by the model of causality to
which one subscribes (p. 1).
Van Ripers (1990) final words on the subject not
only capture the essential difference between SM and FS
treatments but restate a hypothesis that he held from
the beginning: The goal of therapy for the confirmed
stutterer should not be a reduction in the number of dys-
fluencies or zero stuttering. Fluency-enhancing proce-
dures can easily result in stutter-free speech temporarily
but maintaining it is almost impossible (Van Riper, 1990,
p. 318). So is it better, as Van Riper opined, to accept the
inherent inevitability of fluency disruptions that trig-
ger stutter reactions and teach the speaker to react
normally (SM) or to try to prevent their occurrence al-
together ( F S)? Frankly, we dont know the answerhis
hypothesis has never been tested.
We do know, however, that two treatment strategies,
modeling and self-management, are being used increas-
ingly as adjuvants for SM (Guitar, 2006; Prins, 1997) and
FS (OBrian, Onslow, Cream, & Packman, 2003). Both
strategies have been shown repeatedly, in the laboratory
and in the clinic, to have a positive influence on the gen-
eralization and maintenance of behavior change, two of
the most vexing problems that plague both SM and FS
Modeling and Self-Management
Modeling is a procedure for learning by observing an-
other personsorones own performance. It is assumed
that individuals will acquire a desired behavior without
having to perform it. In his excellent review of the history
of behavior modification, Kazdin (1978) notes that early
therapeutic applications of modeling were essentially fo-
cused on fear reduction. Evidence accrued that the most
powerful effects relied on similarity between the model
and the observer, competence, prestige, and status of the
model, the number of models, characteristics of the ob-
server, and consequences following emulated behavior
(p. 227). Ultimately, the procedure evolved into covert
or imagined modeling and, later, carefully structured
It was experimen tal treatment evidence that directly challenged the retro-
grade effects of that theory s stranglehold on clinical practice (Martin, Kuhl,
& Haroldson, 1972; Reed & Godden, 1977) and ushered in the principal cur-
rent direct therapies for stuttering in children (Onslow, Packman, & Harrison,
2003; Prins & Ingham, 1983).
258 Journal of Speech, Language, and Hearing Research Vol. 52 254263 February 2009
imitation strategies that focus on much more than just
fear reduction (Miller, 2006).
Beginning in the early 1960s, experiments in the be-
havior modification literature had already demonstrat-
ed clearly that observation of models could induce new
responses that were not previously in a participants rep-
ertoire, strengthen or weaken inhibitory responses, and
elicit previously learned responses in new environments
(Bandura, 1965). More than a decade later, Martin and
Haroldson (1977) reported their landmark experiment
concerning application of modeling to the treatment of
stuttering. Twenty stutterers showed a significant de-
crease in stuttering frequency as a result o f observing
another client responding dramatically to a treatment
procedure (p.25).Despitethisfindingandthefactthat
Van Riper had informally employed modeling as an in-
tegral part of his SM program (Van Riper, 1958, 1973),
experimental evaluation of its effects on the treatment of
stuttering languished following Martin and Haroldsons
report. In fact, almost 20 years passed before recent
studies by Bray and Kehle (1996, 1998, 2001) showed the
positive influence of self-modeling on the generalization
and maintenance of treatment effects. Since then, we
have only the report by OBrian et al. (2003) suggesting
how modeling might be used in treatment to induce pro-
longed speech.
In the meantime, modeling of a target behavior had
been a well-established clinical strategy within many
areas of speech-language pathology. Indeed, tape-recorded
target models of prolonged speech were used in early FS
programs (e.g., Ingham & Andrews, 1973b). Yet there have
been no recent experimental investigations into how such
model recordingsof client or cliniciancould be used
in a systematic way to increase the probability of gener-
alization and maintenance.
Self-management procedures are believed to influ-
ence the modification of behavior because they help to
establish the self-regulatory systems that provide the
very basis for purposeful action (Bandura, 1991, p. 248).
Through self-observation, self-judgment, and self-reaction,
people learn to exercise and maintain control over their
behavior. Further, according to social cognitive theory,
self-regulatory systems are ultimately enabled by the es-
tablishment of self-efficacy, peoples beliefs about their
capabilities to exercise control over their own level of func-
tioning and over events that affect their lives (Bandura,
1991, p. 257).
Concerning stuttering, as noted earlier, pioneering
work in self-management was done by Goldiamond
(1965a), who advocated using self-control strategies
in conjunction with prolonged speech treatment
(Goldiamond, 1965b). Later, a single-subject study by
La Croix (1973) drew attention to the positive reactivity
of self-recording on stuttering. However, it was two
groundbreaking experimental studiesJames (1981a)
and Martin and Haroldson (1982)that had important
implications for the application of self-management pro-
cedures in treatment. James (1981a) clarified the ben-
eficial effects of self-monitoring on stuttering as well as
the latitude of monitoring accuracy within the procedure.
And then Martin and Haroldson (1982) showed that par-
ticipants who provided their own time-out for stuttering
events had less recovery of stuttering frequency fol-
lowing the time-out condition and, even more important,
showed significantly greater generalization to an un-
treated telephone situation than participants who re-
ceived experimenter-delivered time-out.
Additional clinical investigations by James (1981b)
and Ingham (1982) showed that self-managed time-out
and self-evaluation of performance are capable of produc-
ing sustained reductions in stuttering in young adults
arguably the most relapse-prone type of patient. James
and colleagues (James, 1983; James, Ricciardelli, Rogers,
& Hunter, 1989) then went on to demonstrate that self-
managed time-out could be employed to increase the ef-
ficacy of FS treatments with adolescents and adults who
One later extension of those findings to stutter-
ing therapy occurred in a study reported by the second
author (Ingham et al., 2001). This showed that training
adult stuttering speakers to self-manage reduced frequen-
cies of short phonated int ervals ( PIs), whereas speaking
resulted in sustained reductions in stuttering, and that
this reduction in PIs tended to generalize across a variety
of speaking tasks.
There are covert aspects of SM and FS treatments
that lend themselves to systematic investigations into
how they might best be combined with self-management
strategies. In the case of SM, the focus is often directed
toward reducing sensations of apprehension of pending
stuttering. This type of sensation lends itself to quanti-
fication, if only via self-monitoring for frequency of oc-
currence in order to determine if those sensations during
speech are reduced by self-recording. In the case of FS, it
is already apparent that the quantification of the most
problematic features of prolonged speech might be ame-
nable to self-management strategies. For instance, stud-
ies have shown that speech naturalness can be measured
and modified using self-judgments (Finn & Ingham, 1994).
Those same self-judgments of naturalness are now being
Recent claims made by Hewat, Onslow, Packman, and OBrian (2006) to
have developed a new treatment program for persistent developmental
stuttering in adolescents and adults using self-delivered timeout turns
out to describe a program that is almost identical to that developed more
than 20 years earlier by James (see James, 2007). Such claims serve to
illustrate not only the importance of just attribution but also the importance
of highlighting the discipline s extant knowledge baseone purpose of this
Currently, this treatment is being investigated within the framework
of neuroscience in order to determine the extent to which this strategy
promotes neural plasticity (Ingham et al., 2007).
Prins et al.:
Stuttering Treatment History 259
used as performance criteria in a program-based PI mod-
ification (Ingham et al., 2001). Similarly, the private sen-
sation of speech effort, one of the critical dimensions of
normally fluent speech, is also being measured by self-
ratings (Ingham, Warner, Byrd, & Cotton, 2006). And in
turn, the ratings may be used as performance criteria for
progress in FS treatments. These are but some of the
potentially rich areas of clinical research made possible
by investigating how self-management strategies can be
employed to maximize the durability of changes that are
a consequence of the core components of SM and FS.
Self-management in its p urest sens e implies no
clinician-directed constraints on the process, which of
course is essentially never true in practice. Nevertheless,
it is possible to consider levels of self-managed control
over the treatment process as a worthy topic of research
and to test whether self-established targets for treatment
are more effectivethat is, show more durable change
than clinician-established targets.
The Internet and other technical advances now offer
clinicians and researchers a powerful medium for de-
livering clinical services, including strategies for aiding
generalization and maintenance. There is evidence that
many Web sites are taking advantage of this, but there is
little evidence of research into methods that might be
particularly amenable to Web use. Relevant here are
ways in which the Web makes it possible to implement
modeling and self-management strategies. For example,
self-recorded audiovisual speech samples during treat-
ment can be used to make performance comparisons with
target models of normally fluent speech produced by the
participant. Similarly, the Web can provide access for
observing others using methods that help them with SM
or FS techniques. The new generation of cell phones makes
it is possible to produce and deliver performance tasks to
select sites for both self and clinician evaluation. The
possibilities for such research seem endless.
Finally, there is a rather obvious link between
(a) the concepts of modeling and self-management and
(b) the concepts of efference copy (Blakemore, Frith,
& Wolpert, 2001; von Holst & Mittelstaedt, 1950) and
self-efficacy (Bandura, 1977a). As mentioned earlier,
modeling has gradually evolved into imitation and an as-
sociation with neural structures that support an efference
copy of the neural system prior to behavior performance
(see Hurley & Chater, 2005). In turn, this relates to stud-
ies with direct connections to self-efficacy theory show-
ing that imagining behaviors prior to their performance
may increase the probability that the behavior will be
performed successfully (Gentili, Papaxthansis, & Pozzo,
2006; Mulder, Zijlstra, Zijlstra, & Hochstenbach, 2004).
Evidence shows that the aberrant neural region activa-
tions associated with both real and imagined stuttered
speech are normalized when adults who stutter imagine
speaking fluently (Ingham, Fox, Ingham, & Zamarripa,
2000). This process, too, might be investigated for its
therapy potential.
The time is ripe for testing the effects of modeling
and self-management on FS and SM procedures. It is a
time to ask efficacy questionsto learn whether model-
ing and self-management are differentially effective when
applied to FS versus SM approaches. It is a time to deter-
mine which behavior associated with the occurrence of
stutter events, the fluency disruptions or the reactions to
them, is more amenable to permanent change. It is quite
possible, of course, that what is crucial to treatments
long-term effectiveness has nothing to do with the FS or
SM approach, per se. Rather, successful outcome may be
a function of the employment of self-management, mod-
eling, or other strategies not yet identified. Efficacy stud-
ies will be required to provide the answerstudies that
isolate treatment parameters in order to illustrate their
specific effects.
These potentially rich treatment parameters may
be identified in both behavioral and cognitive domains
by carefully elucidating the numerous strategies that
those who recover from stuttering in adulthood have found
to be effective (see Finn, 2004). Recent trends in cogni-
tive neuroscience are also fertile sources. The role played
by mirror neurons in modeling is especially interesting
given recent findings suggesting that it might be pos-
sible to control their role in learning a new behavior (see
Catmur, Walsh, & Heyes, 2007). Another interesting
possibility is the investigation of self-management through
the use of real-time fMRI (de Charms et al., 2005); self-
control over feedback from regional activity may increase
the probability of neuroplasticity in regions known to be
functionally related to reduced stuttering using FS pro-
cedures (see Ingham, Finn, & Bothe, 2005).
It is also a time to re-evaluate the role of theory in
treatment and ensure that we have theories that are
able to drive the development of more effective thera-
pies. The understanding and treatment of stuttering
need not await the appearance of an all-encompassing
theoryit is certainly not clear that past theories of
stuttering have done much to advance its treatment. Some
of the great advances in science have emerged from the
judicious use of less encompassing theory/models that
build on parameters that are related to powerful inter-
vention effects. For instance, in a recent review of the
enormous contributions made by the late Nobel Prize
winner, Pierre-Gilles de Gennes (Brochard-Wyart, 2007,
p. 149), it was noted that many were derived by isolat-
ing the few essential parameters to derive minimalist
260 Journal of Speech, Language, and Hearing Research Vol. 52 254263 February 2009
theories that could be used and tested (p. 149). Iso-
lating the essential parameters that control stuttering
behaviorsuch as may be the case within modeling and
self-management methodsmay well provide the small-
scale theory/models that can be used to advance treat-
ment. An d, as m entione d above, such mod els may help
build a stronger link with current advances in cognitive
neuroscience, especially with respect to neuroplasti-
the therapy arena (Ingham,Cykowski,Ingham,&Fox,
These observations on historic trends in stuttering
therapy are offered by two individuals with very differ-
ent backgrounds. Our hope is that they encourage fresh
approaches to stuttering treatment research.
Parts of this article were written with the support Grant
R01 DC007893 from the National Institute on Deafness and
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Received May 23, 2007
Revision received October 2, 2007
Accepted April 7, 2008
DOI: 10.1044/1092-4388(2008/07-0111)
Contact author: Roger J. Ingham, Department of Speech and
Hearing Sciences, University of California, Santa Barbara,
Santa Barbara, CA 93106.
Prins et al.: Stuttering Treatment History 263
... Different treatment approaches for stuttering have shown to be most amenable for younger aged individuals compared with individuals in adulthood who stutter. This may be related to the greater degree of neuronal plasticity in individuals of a younger age compared with the less tractable and far less responsive to treatment neurons of adults, for whom it has often been a long-term problem (27,(33)(34)(35). ...
... The severity of stuttering was significantly reduced to a mild status amongst adolescents who suffered from MS. Approximately 4-5% preadolescents suffer from some degree of stuttering compared with ≤1% in adults (9,33,35), and this has compounding effects on the behavioral, cognitive, psychological and social aspects for a patient (2)(3)(4). In addition, more severe negative psychological states are significantly more commonly reported in adolescents and adults who stutter, particularly in relation to mental health (31,32,39). ...
Full-text available
Green tea and its polyphenolic compounds have been shown to exert positive effects in individuals with psychological disorders. The protective role of green tea against stuttering or its related consequences, depression, anxiety and stress, were evaluated in adolescents with moderate stuttering (MS). A total of 60 adolescents aged (12-18) years old were enrolled in this study. Patients were classified according to standardized test material Stuttering Severity Instrument, 4th Edition was used to estimate the severity of stuttering; participants were classified into two groups: a normal healthy group (n=30) and a MS group (n=30). The Depression Anxiety Stress Scale and General Health Questionnaire were used to estimate the degree of depression, anxiety and stress as well as general mental health. The physiological profile of stress hormones, as a measure of the response to green tea response, was also measured amongst participants. Adrenal stress hormones cortisol, dehydroepiandrosterone (DHEA), acetylcholine (ACTH), corticosterone and the cortisol:DHEA ratio were assayed. In addition, the constituent green tea polyphenols and their quantities were determined using liquid chromatography analysis. Decaffeinated green tea was administered six cups/day for 6 weeks, and this significantly improved the depression, anxiety, stress and mental health consequences associated with stuttering in adolescents. In addition, increased consumption of green tea significantly reduced elevated levels of adrenal stress hormones; cortisol, DHEA, ACTH and corticosterone, and increased the cortisol:DHEA ratio in the control and adolescents who stuttered. The data showed that drinking six cups of decaffeinated green tea, which is enriched in catechins (1,580 mg) and other related polyphenols, was sufficient to improve the consequences of mental health associated with stuttering in younger aged individuals.
... En esta misma línea Johnson (1957), señala que el problema central del tartamudeo es la anticipación de la situación de tartamudeo por parte del sujeto estas expectativas y el resultado de esa situación, es decir, su reacción ante la misma, anticipando así la terapia cognitivo conductual. Por su parte, Prins & Ingham (2009), establecen que, en la actualidad, el manejo del tartamudeo y el modelado de la fluidez siguen siendo los métodos de intervención más utilizados en el tratamiento de la tartamudez. Aún con ello, es cierto que no funcionan en todos los casos en adultos y pueden detectarse ciertas recaídas. ...
La tartamudez se define como un trastorno de la fluidez del habla que afecta a la comunicación. Este trastorno, resulta complejo y puede suponer un importante desafío para muchos logopedas y otros profesionales. Hasta el momento, no abundan las investigaciones focalizadas acerca de cómo evaluar e intervenir en la tartamudez desde la perspectiva de los profesionales en logopedia. Precisamente por ello, el objetivo del presente estudio es conocer cuáles son los principales métodos de evaluación e intervención para la tartamudez que utilizan los logopedas en el contexto español y la satisfacción que tienen con éstos. Para ello, se administró una encuesta en la que participaron un total de 123 logopedas y, posteriormente, se efectuó un análisis cuantitativo de los datos obtenidos. Los resultados permitieron conocer los diferentes métodos de evaluación e intervención más ampliamente utilizados en el tratamiento de la tartamudez, aunque se han observado ciertas dificultades para alcanzar un consenso en la utilización de los mismos. Además, los logopedas reclaman la necesidad de confeccionar nuevos instrumentos de evaluación y de una mayor colaboración por parte de los centros escolares. Por tanto, resultaría primordial adoptar un enfoque holístico tratando de enseñar a los sujetos con tartamudez estrategias para afrontar la fluidez y, a la vez, aprender a convivir y manejar el trastorno.
... Historically, two behavioral approaches for stuttering therapy have been practiced; namely, fluency shaping and stuttering management (or modification) [15]. The focus of fluency shaping is on the reduction or elimination of all disfluencies by systematically reducing the rate of speech. ...
Purpose: This study estimated the treatment outcomes of a behavioral stuttering therapy program that blended a combination of intensive face-to-face therapy with telepractice-based follow-up therapy. Method: A total of 17 participants (mean age = 22 years) who stutter participated in the program, preceded by an extended baseline period. The participants completed a series of assessments conducted over multiple time points, spanning a total of 42 weeks. Growth curve modeling was used to analyze the changes participants experienced in the frequency of stuttering, stuttering severity, communication attitudes, and quality of life. Results: The participants demonstrated stability throughout the extended baseline period, and experienced positive outcomes from the intensive program and the gains in communication attitudes and quality of life were largely maintained with weekly follow-up telepractice sessions. However, stuttering frequency and severity increased when the telepractice follow-up session frequency transitioned to a biweekly basis. Neither gender nor age group predicted the treatment outcomes for frequency or severity of stuttering. Gender-based differences were found for the treatment outcomes of specific self-report measures, with male participants having demonstrated a greater proportional decline on their standard scores, relative to female participants. Outcomes were similar for both adolescents and adults. Conclusions: Participants attending the intensive stuttering therapy program experienced positive and significant changes in their speech, attitudes toward communication, and overall quality of life, which were maintained over time with structured, weekly telepractice follow-up sessions.
... Numerous approaches in stuttering management have been proposed to date and are based on different ideologies and theoretical principles. The focus of these approaches or treatment programs aim at enhancing the fluency and improving the overall communication skills of PWS (Prins & Ingham, 2009). The selection of the most appropriate method would pose challenges to speech therapists and require them to have in-depth knowledge about the theoretical principles behind the treatment approach (Ramig, 1993). ...
The current study was carried out with the aim of investigating the effect of maximally relaxed lying posture on disfluencies in young adults who stutter. A total of 24 participants (17 males, seven females; mean age = 24.9 ± 6.2 years) with developmental stuttering were a part of the study. The participants were asked to perform spontaneous speaking and reading aloud tasks in standard sitting and maximally relaxed lying postures. The severity of stuttering for the studied postures was estimated by using the Stuttering Severity Instrument. The results on the Stuttering Severity Instrument showed that stuttering parameters improved during the maximally relaxed lying posture compared with the standard sitting position. The results are discussed in the light of motor control concepts. It is concluded that the maximally relaxed lying posture can facilitate improvement in stuttering scores during spontaneous speaking as well as reading aloud in young adults who stutter. Reduced stuttering scores in the maximally relaxed lying posture suggest that speech therapists can position participants in this position while treating people who stutter.
... Behavioral stuttering therapy incorporates either fluency shaping or stuttering modification/management strategies, or a combination of both to help AWS increase fluency and reduce stuttering severity [35]. Use of real-time feedback based on facial AUs can help with the identification stage of stuttering modification and also aid in providing real-time feedback to help reduce facial tension to decrease stuttering severity and increase overall fluency, similar to the effect seen from using facial EMG [36]. ...
Speech disorders such as stuttering disrupt the normal fluency of speech by involuntary repetitions, prolongations and blocking of sounds and syllables. In addition to these disruptions to speech fluency, most adults who stutter (AWS) also experience numerous observable secondary behaviors before, during, and after a stuttering moment, often involving the facial muscles. Recent studies have explored automatic detection of stuttering using Artificial Intelligence (AI) based algorithm from respiratory rate, audio, etc. during speech utterance. However, most methods require controlled environments and/or invasive wearable sensors, and are unable explain why a decision (fluent vs stuttered) was made. We hypothesize that pre-speech facial activity in AWS, which can be captured non-invasively, contains enough information to accurately classify the upcoming utterance as either fluent or stuttered. Towards this end, this paper proposes a novel explainable AI (XAI) assisted convolutional neural network (CNN) classifier to predict near future stuttering by learning temporal facial muscle movement patterns of AWS and explains the important facial muscles and actions involved. Statistical analyses reveal significantly high prevalence of cheek muscles (p<0.005) and lip muscles (p<0.005) to predict stuttering and shows a behavior conducive of arousal and anticipation to speak. The temporal study of these upper and lower facial muscles may facilitate early detection of stuttering, promote automated assessment of stuttering and have application in behavioral therapies by providing automatic non-invasive feedback in realtime.
... There are different treatment approaches for children and adults who stutter since strategies that may work well for preschool children may be of little use for adults [8]. Some therapeutic approaches and treatment programs may allow a person with stuttering (PWS) to improve speech fluency and communicate in an effective way [9]. Alternative approaches may instead focus on cognitive, behavioral, or psychological therapy or use prosthetic devices that deliver altered auditory feedback [10,11]. ...
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Objective: The current study aimed to investigate the effects of body position on the level and severity of stuttering in young adults with developmental stuttering. Methods: A total of 24 subjects (male: 17; female: 7; mean age: 24.9 ± 6.2 years) with developmental stuttering participated. The participants were asked to perform oral reading and spontaneous monologue-speaking tasks in different body postures while their speech was recorded. During reading and speaking tasks, the Stuttering Severity Instrument was used to quantify the severity of stuttering. The effects of different body postures on stuttering severity, reading task, and speaking task scores were analyzed. Results: Significant differences in stuttering severity, reading task, and speaking task scores were found for different body postures. Post hoc analyses revealed a significant difference in stuttering severity, reading task, and speaking task scores when subjects were sitting on a chair with no arm support compared to lying down (p<0.05). Similarly, there were significant differences for two sitting positions (sitting on a chair with no arm support vs sitting on a chair with arm support (p<0.05)). Conclusions: Body postures or body segment positions that relax and facilitate the muscles of the neck and shoulders may potentially improve speech fluency in young adults with developmental stuttering.
... Subjects ranged in age from 16 to 55, and all reported a family history of stuttering. Therapy for all subjects was based on a fluency shaping approach (Blomgren, 2010;Prins & Ingham, 2009) and was performed in a standardized, intensive program taking place over 12 successive days at the Hollins Communications Research Institute, Roanoke, VA (Webster, 1980). In this program, individuals use computer-and therapist-assisted methods to learn to focus on speech fluency targets to reduce overt stuttering. ...
Purpose: We investigated whether outcomes of therapy for persistent developmental stuttering differ in individuals who carry a mutation in one of the known genes associated with stuttering compared to individuals without such mutations. Method: We studied outcomes of an intensive fluency shaping-based therapy program in individuals with persistent developmental stuttering. We evaluated a cohort of 51 stuttering individuals with who carried a mutation in either the GNPTAB, GNPTG, NAGPA, or AP4E1 gene. We compared therapy outcomes in these individuals with outcomes in 51 individuals matched for age, gender, and ethnicity, who stutter and underwent the same therapy program, and did not carry a mutation in any of these genes. Fluency pre- and post-therapy was evaluated using blinded observer-based quantitative stuttering dysfluency measures (Dysfluent Words Score, DWS), and by subjects' self-reported measures of struggle, avoidance and expectancy behavior associated with speaking (Perceptions of Stuttering Inventory, PSI). The difference between pre- and post-therapy fluency scores was taken as the measure of near-term therapy efficacy. Results: Comparison of fluency measures showed a strong effect of therapy overall. Mutation carriers achieved significantly less resolution in PSI following therapy, with PSI scores showing significantly less improvement in individuals who carry a mutation (p = 0.0157, RR = 1.75, OR = 2.92) while the group difference in DWS between carriers and non-carriers was statistically not significant in the present study, the trend observed in the results warrants further research focused on this important issue. Conclusions: These results suggest stuttering is more resistant to therapy in individuals who carry a mutation in one of the genes known to be associated with stuttering.
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Previous studies suggest that excessive attention to speech motor control may lead to inappropriate coping behaviors and clinical distress for adults who stutter. However, when combined with psychological-based approaches, such as cognitive behavioral therapy (CBT), it is possible that these behaviors and distress could be effectively alleviated. This study investigated whether group CBT, with a specific focus on decreasing the excessive attention to speech motor control, could reduce stuttering frequency as well as the psychological and behavioral difficulties associated with stuttering. A group CBT training program for adolescents and adults who stutter was conducted over the course of five weeks. Forty-eight adolescents and adults who stutter participated in eleven groups. Changes in stuttering frequency and self-reported outcomes (questionnaire scores) from the baseline (pre-intervention) were investigated immediately post-intervention and six months post-intervention. Results show that stuttering frequency was significantly reduced after interven- tion. Additionally, some questionnaire scores, including those on overall impact of stuttering, social anxiety, and coping behaviors, showed significant improvement both immediately post-intervention and six months post-intervention. Therefore, it is suggested that group CBT focusing on the reduction of excessive attention to speech motor control is effective in reducing stuttering frequency as well as the psychological and behavioral difficulties associated with stuttering. 吃音のある成人では,発話運動制御に過度に注意が向くことが不適切な対処行動を引き起こし,臨床的な困難につながるという仮説があり,認知行動療法などの心理・行動面での支援が効果的であると考えられる。本研究は,発話運動制御に対する過度な注意を弱める認知行動療法が,吃音中核症状頻度,心理・行動面での困難の緩和につながるかを調べることを目的とした。吃音のある成人48名が参加し,介入前・後での吃音頻度(吃音検査法・フリースピーチ; 主要アウトカム)・質問紙得点(副次アウトカム)の変化と,介入6ヶ月後での質問紙得点の変化を調べた。その結果,吃音頻度は,吃音検査法とフリースピーチ共に有意に減少し,一部の質問紙得点は,介入前に比べて介入6ヶ月後でも有意な改善を示していた。発話運動制御に対する過度な注意を弱める認知行動療法は,吃音頻度の減少に加え,長期的な心理・行動面の困難の緩和にもつながり得ると考えられた。
Es wird ein kurzer historischer Überblick über die Therapie des Stotterns gegeben. Anschließend werden die beiden Hauptansätze, die Stottermodifikation und das Fluency Shaping, beschrieben. Auf die Therapie von Kindern, die medikamentöse Behandlung und die Bedeutung von Selbsthilfegruppen wird gesondert eingegangen. Abschließend wird die Effektivität von Stottertherapien diskutiert.
Presents an integrative theoretical framework to explain and to predict psychological changes achieved by different modes of treatment. This theory states that psychological procedures, whatever their form, alter the level and strength of self-efficacy. It is hypothesized that expectations of personal efficacy determine whether coping behavior will be initiated, how much effort will be expended, and how long it will be sustained in the face of obstacles and aversive experiences. Persistence in activities that are subjectively threatening but in fact relatively safe produces, through experiences of mastery, further enhancement of self-efficacy and corresponding reductions in defensive behavior. In the proposed model, expectations of personal efficacy are derived from 4 principal sources of information: performance accomplishments, vicarious experience, verbal persuasion, and physiological states. Factors influencing the cognitive processing of efficacy information arise from enactive, vicarious, exhortative, and emotive sources. The differential power of diverse therapeutic procedures is analyzed in terms of the postulated cognitive mechanism of operation. Findings are reported from microanalyses of enactive, vicarious, and emotive modes of treatment that support the hypothesized relationship between perceived self-efficacy and behavioral changes. (21/2 p ref)
Stuttering is probably the best known and most re-searched speech disorder; but it also ranks among the most difficult to define, plausibly explain, or, especially in adults, treat effectively. In the recent edition of the Diagnostic and Statistical Manual of Mental Disorders (DSM-III-R) (American Psychiatric Association, 1987), the “essential features” of the disorder are described as “frequent repetitions or prolongations of sound or syllables. Various other types of speech dysfluencies may also be involved, including blocking of sounds or interjection of words or sounds” (American Psychiatric Association, 1987, p. 86). However, this description neither defines the frequency of these behaviors nor distinguishes then from normal disfluencies. Nevertheless, few observers have difficulty in recognizing the features of chronic stuttering. The behaviors that listeners judge as stutterings appear to vary in their frequency across speaking situations and are dramatically reduced during certain speaking conditions or with the use of certain speech patterns. Chronic stuttering usually begins in early childhood, although adult onset of the disorder occasionally occurs, usually in conjunction with brain damage. Stuttering appears to occur in all nationalities with an incidence of approximately 1% and a prevalence of 4% to 5%.