Drs. Curlee and Yairi s (1997) recent

Second Opinion Treatment Decisions for Young Children Who Stutter: Further Concerns and Complexities Roger J. Ingham University of California, Santa Barbara Anne K. Cordes The University of Georgia, Athens Ingham Cordes Drs. Curlee and Yairi s (1997) recent paper provided some interesting and provocative comments regarding treatment for children who have been stuttering for less than approximately 2 years. Their paper raised several issues that were important, complex, and fully deserving of critical assessment. It was accompanied by two Second Opinion commentaries (Bernstein Ratner, 1997; Zebrowski, 1997), each of which addressed some additional pieces of this complex area, and each of which essentially agreed with Curlee and Yairi on two general points: that some children who stutter may not need clinical services, and that we have reason to be dissatisfied with the available evidence about treatment effectiveness for children who stutter. We do not entirely disagree, but the purpose of this response is to present an opinion about Curlee and Yairi s paper that challenges their premises and their conclusions in several areas. These areas include the incidence and prevalence of stuttering, and whether those data provide evidence of high recovery rates; several issues related to determining whether treatments are effective and whether they should be recommended; and several issues related to the experimental evaluation of treatment effectiveness. Incidence, Prevalence, and Mathematically Necessary Recovery Curlee and Yairi (1997) began with the premise that there is considerable disagreement about the necessity of treating every child who stutters as soon after onset as possible and about the risks involved if their treatment is postponed for a while (p. 8). Some of this disagreement, according to Curlee and Yairi, stems from the differences in the prevalence and the lifetime incidence of stuttering and from our knowledge about the spontaneous remission of stuttering. They wrote that the lifetime incidence of stuttering in the United States and western Europe is generally believed to approximate 4 to 5% whereas its prevalence [ranges] from 0.5 to 1% (1997, p. 8). If these numbers are correct, wrote Curlee and Yairi, then it is mathematically transparent that 75% to 90% of all who begin (p. 9) to stutter will not continue to do so. In addition, between 32% and over 80% (p. 9) of these recoveries are said to be spontaneous or unaided (p. 9). Curlee and Yairi are not the first to summarize the prevalence and incidence literature as showing a 75 90% recovery rate, but the literature is not entirely consistent with such an interpretation. Table 1 summarizes the studies listed by Bloodstein (1995, his Tables 3 and 6) that have been conducted since 1950. 1 Estimates of the lifetime incidence of stuttering range from 0.70% to 15.40% across these studies, whereas estimates of prevalence range from 0.30% to 2.12%. These differences clearly reflect substantial and significant variability across studies, and they also argue against Curlee and Yairi s (1997, p. 9) claim that it is mathematically transparent that 75% to 90% of all who begin to stutter will recover: At the extreme, these numbers actually suggest that the lifetime incidence of stuttering is only 0.7% (Culton, 1986) at the same time that its prevalence is somehow as high as 2.12% in juniorhigh and high-school students (Gillespie & Cooper, 1973). It is certainly not mathematically simple to translate such data into a 75 90% recovery rate. Recommending Treatments for Stuttering The next section of Curlee and Yairi s paper argued that it may be appropriate to withhold treatment for young children not only because of the supposedly high rate of natural recovery but also because the efficacy of early interventions with children soon after stuttering onset is 1 An additional nine studies in Bloodstein s Table 3, and three studies in his Table 6, were conducted between 1893 and 1942. Inclusion of these studies would only lengthen our Table 1 without changing the conclusions drawn, as readers may verify. 10 American American Journal of Journal of Speech-Language Pathology Pathology Vol. Vol. 7 7 No. 1058-0360/98/0703-0010 3 American Speech-Language-Hearing Association August 1998

TABLE 1. Summary of incidence and prevalence studies of stuttering reported since 1950, selected from those summarized by Bloodstein (1995, Tables 3 and 6). Study N Population % Prevalence studies Schindler (1955) 22,976 Grades 1 12 0.55 Hull (1969) 6,287 Grades 1 12 0.30 Gillespie & Cooper (1973) 5,054 Grades 7 12 2.12 Leavitt (1974) 10,445 Grades 1 6 0.84 Leavitt (1974) 10,449 Grades 1 6 1.50 Brady & Hall (1976) 187,420 Grades K 12 0.35 Hull et al. (1976) 38,802 Grades 1 12 0.80 Leske (1981) 7,119 Grades 1 6 2.00 Incidence studies Glasner & Rosenthal (1957) 996 First grade 15.4 Andrews & Harris (1964) 1,000 Birth 16 4.9 Andrews & Harris (1964) 206 Adults 4.8 Sheehan & Martyn (1970) 5,138 Freshmen/graduates 2.9 Dickson (1971) 3,923 K 12 9.4 Cooper (1972) 5,054 High school 3.7 Porfert & Rosenfield (1978) 2,107 University students 5.5 Seider, Gladstien, & Kidd (1983) 1,857 Stutterers relatives 13.9 Culton (1986) 30,586 University freshmen 0.7 essentially unknown (p. 10). In expanding on this idea, Curlee and Yairi drew on a conference presentation by one of us (Ingham, 1996) that has since become a book chapter by both of us (Ingham & Cordes, in press). In this chapter, we criticized current stuttering treatment research, asserting that our discipline and our clients are poorly served when researchers promote treatments that are supported by inadequate treatment outcome data. We provided specific examples of articles that recommended treatment procedures that were undocumented, at best, and shown to be ineffective, at worst. We also pointed out that there is, in fact, an established and growing body of treatment research studies that do satisfy a relatively standard evaluation framework, providing repeated speech measures from before, during, and after treatment and from both within- and beyond-clinic conditions (e.g., Craig et al., 1996; Kully & Boberg, 1991; Lincoln, Onslow, Lewis, & Wilson, 1996; Martin, Kuhl, & Haroldson, 1972). These studies consistently report clinically significant benefits relative to untreated control conditions, some for very young children, and these benefits may also be evaluated through comparisons with other reports of children who have not received professional treatment. Finally, we presented a reanalysis of available data that we believed strongly suggested two conclusions: that treatment should be offered to young children who stutter, and that delaying treatment, even for as little as 15 months after onset, may diminish the chances that children will recover. 2 Curlee and Yairi (1997), on the other hand, concluded that treatment may not need to be offered to young children and that delaying treatment will have no deleterious effects: Our contention is that active monitoring of young preschoolers during the first 2 years of stuttering rather than intervening actively, permits the unaided remission of stuttering for most of them and does not adversely affect later treatment of stuttering for those who do not stop (Curlee & Yairi, 1997, p. 12). Clearly, Ingham and Cordes (in press) and Curlee and Yairi (1997) have drawn diametrically opposed conclusions from the same research and clinical literature, making opposite recommendations for young children who stutter. There would appear to be several factors underlying this disagreement, several of which we explore below. Distinguishing Between Disfluency and Stuttering One of the first issues that deserves further consideration is as simple as the basic fact that 2 The more important implication of these findings, actually, is that parents should be given information about the benefits that may occur if they do try to intervene in their child s stuttering. Studies reported by Onslow and colleagues, and others, strongly suggest that children s stuttering can be reduced if parents employ relatively mild verbal contingencies for occasions of stuttering and for periods of fluent speech production. There is, on the contrary, absolutely no evidence that such procedures will cause stuttering to increase or become chronic. Ingham Cordes 11

a disfluency is not necessarily a stuttering. Measuring disfluencies cannot be equated with measuring stuttering, because measuring all disfluencies would include measuring both normal and stuttered disfluencies. Systems that distinguish between stuttered disfluencies and nonstuttered disfluencies (e.g., Conture, 1990; Ryan, 1974; Wingate, 1964) also fail, because it is consistently possible to find examples of speech behaviors that cannot be appropriately classified under such systems (the attempt to define all between-word disfluencies as normal provides one clear example; Cordes & Ingham, 1995). This problem has led some researchers to measure stutter-type (Meyers, 1986) or stutter-like (Yairi & Ambrose, 1992) disfluencies, an equivocating approach that does not solve the problem of determining whether the reported data are meant to represent stuttering or not. Much other current research about stuttering is conducted simply in terms of disfluencies, with no attempt to provide data about stutterings at all. The implications of such basic definitional problems are enormous, especially for very young children. They lead to diagnostic criteria that label children as stuttering if their speech includes 10% or more total disfluency (Gregory & Hill, 1993, p. 28), whether or not that speech includes any atypical, abnormal, or stuttered disfluencies. They also lead to important difficulties in evaluating the effects of treatment: A report that disfluencies were reduced is not necessarily a report that stuttering was reduced and should not be interpreted as such. Given these complexities, any meaningful discussion about whether children are stuttering, or about whether children need treatment for stuttering, should begin by affirming that normal disfluency will not be confused with stuttering and by documenting that the children in question were, in fact, stuttering. Curlee and Yairi (1997) did not address this seemingly basic issue of defining their terms, a problem that may have led them to some questionable conclusions. They wrote, for example, that a variety of different treatment procedures may be capable of eliminating or significantly reducing almost all young children s stuttering (p. 10), basing this statement on the evidence that a number of different intervention procedures are reported to have high rates (e.g., >85%) of success or recovery (Fosnot, 1993; Martin, Kuhl, & Haroldson, 1972; Starkweather, Gottwald, & Halfond, 1990) (pp. 9 10). The definitional problem here is that some of these papers did not report data in terms of children s stuttering: Fosnot (1993) discussed disfluencies and stutter-type disfluencies, so her data may or may not be related to changes in stuttering, and Starkweather et al. (1990) discussed very little data at all (as Curlee and Yairi pointed out). Distinguishing Among General Approaches to Treatment The determination of what constitutes treatment is equally critical to a meaningful discussion about whether to provide treatment for children who stutter. Three related issues are important here: whether treatment must be administered by a professional, whether treatment includes direct or indirect procedures, and whether a procedure should be referred to as a treatment if there is little or no evidence that it will be effective. The question of whether treatment must be administered by a professional was raised by Curlee and Yairi in the context of spontaneous recovery, and it is related to previous suggestions that parent- or self-initiated procedures might be a relevant factor in recoveries that appear to occur without professional intervention (Finn, 1996; Ingham, 1983). Curlee and Yairi argued that most advocates of this view do not address the apparent failure of caregivers interventions (p. 11) when stuttering persists. In fact, Ingham (1983) addressed this very issue: there is also no evidence that these procedures are necessarily effective in reducing stuttering in all children. All that we have to look to is some evidence that they may be effective with some children who stutter (p. 123). The importance of this point is highlighted by the fact that Curlee and Yairi used it to support another suggestion that early intervention for stuttering may be unnecessary: If intervention or treatment is restricted to activities that are undertaken by or under the supervision of certified clinicians trying to remediate stuttering, current evidence indicates that 60% to 70% of preschool age children who begin to stutter stop within the first 2 years of onset without having received any kind of such professionally directed intervention (p. 11). The premises underlying this statement deserve some thoughtful scrutiny. First, we see no reason to reserve the label treatment for cases where a professional was involved. Adults often provide treatments for themselves or for their children, for everything from headaches to behavioral, emotional, or learning problems. Second, we know of no data to suggest that 60% to 70% of all children who begin to stutter receive no professional intervention at all. Third, there is evidence that some children who recover without professional intervention are exposed to procedures that are known to reduce stuttering (Finn, 1996; Ingham, 1976, 1983; Martin & Lindamood, 1986). In addition, 12 American Journal of Speech-Language Pathology Vol. 7 No. 3 August 1998

Ingham (1983) and Martin and Lindamood (1986) showed that direct parent intervention was generally reported more frequently among recovered than among nonrecovered stutterers. The conclusion that a group of children stopped stuttering without professional intervention, in other words, should not be equated with the conclusion that they all stopped stuttering in the absence of actively administered procedures that are known to reduce stuttering in some children that is, depending on how the word is defined, without treatment. This discussion is also related to the difference between direct and indirect treatments for children s stuttering. Direct treatments, in this context, are usually defined as those that target the child s speech behaviors (whether through the parent or through a clinician); indirect treatments are usually defined as those that target only environmental or parental behaviors. Curlee and Yairi did not distinguish between the two, but Zebrowski (1997) made several recommendations specifically for indirect treatment. Bernstein Ratner (1997) also noted that indirect treatments are undoubtedly preferred to direct treatment by clinicians (p. 30) for early stuttering. She suggested several possible reasons for this preference, including the comment that it is unclear to me whether most of the major tasks of direct stuttering treatment can be performed by the average child as young as 2;6 years for the simple reason that they are not metalinguistically capable of many of the behavioral features of these programs (p. 30). There are at least two large classes of direct treatments, however, that are ideally suited to very young children, that require no metalinguistic awareness, and that have been shown to be effective at reducing young children s stuttering. These include punishment of undesired responses combined with reinforcement of desired responses, which was discussed above (Martin et al., 1972; Onslow, Andrews, & Lincoln, 1994), and procedures that depend on controlling the length or complexity of the child s utterances (by controlling the speaking situation; no metalinguistic awareness is required) (Costello, 1983; Ryan, 1974). The distinctions between professionally directed and parent-directed treatment, and between direct and indirect treatment strategies, are both related to the question of whether a treatment should be referred to as a treatment if it has not been demonstrated to be effective. Bernstein Ratner (1997), for example, recommended some indirect home management strategies, because some controlled experimental data (Stephenson-Opsal & Bernstein Ratner, 1988) as well as observational data (Winslow & Guitar, 1994), validate the palliative effects of some types of modifications to the home communication environment (p. 30). The problem with this suggestion is that these studies simply do not provide experimentally compelling evidence that the procedures mentioned can improve children s stuttering. The Stephenson-Opsal and Bernstein Ratner study, for example, employed an AB treatment design, with no experimental control. The subjects were 2 boys who stuttered: one aged 3;3 who may have confounded the results by being concurrently enrolled in another behavior management program (1988, p. 50) and one aged 6;2. They were recorded for unspecified amounts of time in mother-child dyads, and the palliative effects were a mean 28% and 45% decrease (respectively) in the children s mean percent time disfluent. No evidence was presented to show that this decrease was stable, that it exceeded base-rate variability, that it reflected beyond-clinic performance, or that it reflected changes in the children s stuttering (as opposed to normal disfluency; see above). In short, this study provided no evidence that the intervention techniques can show relatively immediate reductions in stutter frequency that are time-linked to the intervention manipulation (Bernstein Ratner, 1997, p. 32). Winslow and Guitar (1994) obtained similar results. One boy who stuttered, aged 5;9, completed an ABAB experimental design, where the A condition was routine interaction and the B condition was structured turn taking during conversations at meal time in the child s home. Results showed, at best, a 50% reduction in stuttering-type disfluencies during the structured turn-taking condition in the home (from a mean of 7 20 stuttering-type disfluencies per 300 words to a mean of 5 10 stuttering-type disfluencies per 300 words), with no data from other settings, no follow-up data, and no evidence that the child s stuttering (not stutteringtype disfluencies) was decreasing. This change might be palliative, but it cannot be justified as an experimentally verified and effective treatment for children s stuttering, as we would define those terms. Zebrowski s (1997) response to Curlee and Yairi was similar to Bernstein Ratner s in this respect and raises similar issues. Zebrowski provided a series of decision streams, including suggesting that clinicians could introduce indirect treatments for certain children. Suggested procedures included modeling a reduced speaking rate and increased turn-switching pause duration, allowing the child to finish his conversational turn without interruption, and so forth (1997, p. 25), methods that Zebrowski justified by reference to publications by Kelly (1993, 1995) and Zebrowski, Weiss, Savelkoul, and Hammer Ingham Cordes 13

(1996). Kelly offered less critical reviews of this literature than Nippold and Rudzinski (1995), who could find little supporting evidence for these procedures. Zebrowski et al. (1996) did offer some data on the effects of slowing the mother s speech rate, from a study of 5 children who stuttered (aged 2;10 7;5) and their mothers, but those data showed only variable and nonsignificant effects on the children s disfluencies in a clinic setting. In other words, as Nippold and Rudzinski (1995) concluded previously, not one of the indirect treatment strategies recommended by either Bernstein Ratner (1997) or Zebrowski (1997) can be justified on the grounds of data that should be interpreted as showing clinically significant reductions in children s stuttering. The Desirable Outcomes for Children Who Stutter Arguments about whether treatment is desirable or necessary for young children who stutter are further complicated by arguments about the desired treatment outcome. Whether administered by a professional or not, whether direct or indirect, whether administered immediately or delayed, we assume that the goals of treatment for a child who stutters are twofold: to eliminate the stuttering and to allow the child to grow up without any of the social or emotional consequences of living with a communication disorder. We also believe that accomplishing the former is among the most straightforward ways to achieve the latter (Lincoln & Onslow, 1997). One of the many complications in this area involves determining whether stuttering has been eliminated. Clearly, this determination cannot be made from brief speech samples recorded within a single treatment environment; there are sufficient reports of stuttering varying across time and place, and of clinicbound fluency, that this point should be selfevident (Andrews & Ingham, 1972; J. Ingham, 1989; Ingham & Packman, 1977; Lincoln & Onslow, 1997; Lincoln et al., 1996; Martin et al., 1972; Onslow, Costa, & Rue, 1990; Onslow et al., 1994; Reed & Godden, 1977; Ryan, 1971, 1974; Ryan & Van Kirk Ryan, 1983). Equally, this determination cannot be made if data are reported only in terms of disfluencies, rather than in terms that make it clear whether the child is stuttering or not, as discussed above. The longitudinal study of Yairi and colleagues, referred to by Curlee and Yairi (1997), exemplifies some of the difficulties in this area and is directly relevant to Curlee and Yairi s recommendations about delaying treatment. First, the performance criteria for defining children as recovered in the longitudinal study have varied from publication to publication (Onslow & Packman, in press). Yairi and Ambrose (1992) required, among other criteria, that a child display not more than 2.99 SLDs per 100 words to be classified as recovered. Later studies, however, have allowed as many as 4 SLDs (see Paden & Yairi, 1996, p. 983; Watkins & Yairi, 1997, pp. 387 388; Ambrose, Cox, & Yairi, 1997, p. 569). At the same time, the opposite problem is also present: In Yairi and Ambrose s (1992, 1996) data, 4 of the 8 continuing stutterers displayed less than 2.99 SLDs at Visit 5 in the clinic but were still regarded as nonrecovered (presumably because of parental reports that these children still stuttered outside the clinic). These and other complexities in the data from Yairi and colleagues would be unremarkable were it not for the fact that these are the numbers used by Curlee and Yairi (1997, p. 10) to claim that the rate of recovery in untreated stutterers within the longitudinal study is as high as 89%. In a more general sense, these conflicting reports simply demonstrate the importance of establishing and following clear criteria for determining when a child will be said to have recovered from stuttering, as well as the difficulties inherent in doing so. 3 The Effects of Delaying Treatment As Curlee and Yairi and others have observed, there is unquestionably some number of children who stutter for several months and then stop stuttering without formal treatment. For example, of the 43 children who ever stuttered in Andrew and Harris s 1964 longitudinal study, approximately 18 children stopped stuttering within 6 months. 4 They labeled most of these children Transient Nonfluent, but such 3 The issue is complicated by the fact that Yairi and Ambrose (1996) published a correction to the Yairi and Ambrose (1992) data that, itself, may require correction. In a personal communication to one of us (R. I.) regarding the original correction, Ambrose (personal communication, April 22, 1996) reported that by Visit 5 there were 13 Recovered and 8 Continuing stutterers. The mean SLD scores for both groups of subjects, as shown in the 1996 Erratum, correspond precisely to that number of subjects within each category. Yairi and Ambrose (1996) also reported, however, that at Visit 5 there were 14 Recovered and 7 Continuing stutterers. 4 Readers should be aware that the Andrews and Harris study was not without its problems (Ingham, 1976). The data from this study were first displayed in a figure (Andrews & Harris, 1964, p. 31) that has been reproduced in various editions of Bloodstein s A Handbook on Stuttering. In 1984, however, Andrews (1984, p. 4) made unexplained changes to the years of stuttering onset and recovery for 10 of the 43 children. A comparison between the original table (Bloodstein, 1981, p. 84) and the revised table (Bloodstein, 1987, p. 94) shows several changes in the longitudinal data for which no explanation has ever been provided. 14 American Journal of Speech-Language Pathology Vol. 7 No. 3 August 1998

children may display both speech behaviors and emotional and social reactions that are indistinguishable from those exhibited by older children or adults who stutter. Given that these data do suggest that some children may stutter for something less than 6 months and then stop, we would agree with the principle of waiting to initiate treatment to this extent: It certainly does make sense for clinicians to consider waiting perhaps 3 to 6 months from the time of stuttering onset before initiating intervention, if waiting does not introduce social or emotional pressures for the child or for the parents and if there is some indication that the individual child in question is improving without treatment. One issue that Curlee and Yairi did not mention, however, is that 3 to 6 months or more has often passed before parents bring a child to a speech-language pathologist in the first place, making the question of a 6-month waiting period essentially moot in many cases. We emphasize here that the most effective use of a waiting period would not be simply to wait in the hope or expectation that the child will recover spontaneously. Instead, the parents and the clinician could collect recorded base-rate speech samples, at least monthly and in multiple speaking situations, to provide a rational and data-based source for deciding whether stuttering is increasing or decreasing for a particular child. Again, Curlee and Yairi (1997) reported data from Yairi and colleagues longitudinal study that are relevant to this point and that we would interpret as potentially confirming the need for treatment within 12 months after onset. Repeated measurements of children in the longitudinal study showed that percentages of SLDs declined over the first 12 months in those children who stopped stuttering within 12 months, whereas the percentage of SLDs either increased or remained at relatively stable levels throughout this period among those whose stuttering persisted (Curlee & Yairi, 1997, p. 12). We would argue that these data provided ample justification for introducing treatment for the children whose stuttering was persisting, especially if the failure to provide treatment was related to these children s increased risk of stuttering for another 2 years or longer (Curlee & Yairi, 1997, p. 12). Curlee and Yairi noted that older children most likely constitute a subgroup of stuttering children who have passed through the primary remission period of the stuttering population (1997, p. 10). They continued, however, with the statement that these older children are, therefore, much more likely to possess characteristics or predispositions that result in chronic stuttering (1997, p. 10). We would argue that providing treatment for younger children is preferable to withholding treatment from children who are clearly not recovering and then concluding, when they are older, that untreated and unrecovered children must have had a predisposition to display chronic stuttering. Both Zebrowski (1997) and Bernstein Ratner (1997) also made recommendations that are relevant to decisions about when to introduce treatment. Zebrowski presented five potential treatment plans, each based on a combination of suggested risk factors. It is only the last of these, Plan E, that includes any mention of direct treatment, designed to decrease the child s stuttering, and the child must be 24 36 months postonset (p. 26) to qualify. Children in Plan C are allowed to stutter for up to 24 months without direct treatment; parents here can be informed about changes in their speech that might have an indirect fluency-inducing effect on the child (p. 26). Children in Plan D have also been stuttering for up to 24 months, and their parents are often beginning to suspect that the child may not grow out of it (p. 26). We simply see no justification for suggesting that a child who is clearly not growing out of it should be required to wait for 2 full years before being given access to treatments that are known to be more effective if used within 15 months of stuttering onset. Again, as we interpret the literature, there is compelling evidence that the probability of successful treatment is lower for older children, and the decision to delay stuttering treatment should be made with full awareness of this evidence. The comparisons that we presented in the Ingham and Cordes (in press) paper showed that 81.8% of preschoolers who received treatment met our criteria for satisfactory outcome, but only 54.2% of school-age children who received treatment met these criteria, for treatment studies that provided data from treatment evaluation procedures. Similar comparisons showed that 85.7% of children who received treatment within 15 months of stuttering onset met our recovery criteria (7/9 boys and 5/5 girls), whereas only 59.4% of children who received treatment starting more than 15 months after stuttering onset did so (16/28 boys and 3/4 girls). Other evidence about the importance of early treatment can be found in the reports of Onslow and colleagues, for example, who have reported comparatively better results at longterm follow-up for children aged 2 4 years (Onslow et al., 1994) than for children aged 7 12 years (Lincoln et al., 1996) who received essentially the same treatment. Curlee and Yairi argued, in commenting on these comparisons, that there are a number of risks involved in combining data from different studies [including] such factors as subjects Ingham Cordes 15

ages, time since onset, sex, and family history (p. 10). 5 We agree, but the comparisons presented by Ingham and Cordes (in press) were, in fact, completed separately for boys and girls of different ages (preschoolers and school-age) and of different times since onset [more or less than 15 months, a dividing line chosen based on Yairi and Ambrose s suggestion that chances for chronicity increase approximately 15 months after onset (1996, p. 73)]. The claim that a particular treatment was solely and directly responsible for the recovery of the children who received it is different from the claim that the children who received a treatment recovered, and this is an important distinction. Nevertheless, and even given the many terminological difficulties that we have raised here, our comparisons consistently showed that children who received their treatment relatively early displayed more substantial treatment benefits than children who received their treatment relatively late, even when late treatment was defined as only 15 months postonset. Nothing in Curlee and Yairi s comments on this issue alters this conclusion. Treatment Research Methodology Curlee and Yairi s final section on Research Needs and Ethical Issues begins by acknowledging that treatment for very young children who stutter can be effective, and that some interventions can be causally related to the changes reported in some children s stuttering (p. 14). Nevertheless, they still hesitate to recommend treatment for young children, because of the absence of scientifically credible treatment outcome data (p. 15). We do agree, as discussed above, that there is an absence of credible data to support many of the procedures that are recommended as treatments for children who stutter, but these issues also deserve to be more carefully addressed. Curlee and Yairi suggested that the use of randomly assigned, untreated control groups has long been viewed as essential for evaluating treatment effectiveness (p. 15) and that it is critically important that such important scientific standards not be abandoned until there is sufficient evidence available (p. 15). Untreated control groups are by no means universally accepted, however, for two distinct but overlapping 5 They also rightly observed that one of the studies included in our comparisons (Ramig s 1993 historical study of untreated children) was a study of older children, not of very young preschoolers. But Ramig s study also included 8 children, including 2 girls, who were assessed within 15 months of reported onset and of those 8, only 1 boy recovered. reasons (both of which Curlee and Yairi also recognized): an untreated control group may be scientifically unnecessary, and an untreated control group may be unethical (Lewin, 1995). These issues are complex, in part because the overriding principle of treatment research should be to provide the best possible treatment to all participants (World Medical Assembly, 1989). If it truly is not known whether the treatment in question will be any better than no treatment at all, then a notreatment control group can provide a scientifically relevant and entirely ethical comparison. This is not the only option, however; investigators can compare different components or levels of a treatment, or compare newer and older treatments, both procedures that have been used effectively in evaluating treatments for stuttering in adults (e.g., Ingham, Andrews, & Winkler, 1972; Perkins, Rudas, Johnson, Michael, & Curlee, 1974). Large-scale treatment trials may also be appropriately abandoned when treatment effects become so obvious that it is no longer defensible to deny those benefits to the control group. Such decisions are often made using the same logic that Sidman (1960) employed to determine the number of subjects that are needed before researchers will accept that a powerful treatment has been identified. If a treatment is introduced, for example, to a relatively small number of similarly afflicted patients, all of whom had shown stable (or deteriorating) base-rate levels, if all immediately reduce their disorder when treatment is introduced, and if this effect is replicated across clinics or laboratories, then it is not necessary to repeat the procedure with too many other patients before clinical scientists should accept that the treatment will most probably have the same positive effects on the next patient. The possibility certainly does exist that the next subject might respond differently, but similar problems of external validity are evident in the assumption that averaged findings from a large group are applicable to any one particular subject. More importantly, sequential analysis (Wald, 1947) can establish the probability that a sequence of consistently positive (or negative) treatment responses could not have occurred by chance (Bross, 1952), without the need for the sampling-theory assumptions of large-group research. Curlee and Yairi did suggest that the studies needed to provide credible treatment outcome data could begin with single-subject experimental designs to assess the effects of different treatment procedures in an unequivocal manner (p. 16). At least eight studies over the past two decades have done precisely that, and these were cited by Curlee and Yairi. They then 16 American Journal of Speech-Language Pathology Vol. 7 No. 3 August 1998

proceeded to suggest, however, not only that this information is not sufficient without the support of group studies, but also that satisfactory treatment designs would require controlgroup subjects to receive no treatment for 2 years. It is critically important to recognize, in addition to recognizing that a no-treatment control group might not be necessary at all, that the standard pretest-posttest control-group design does not require the control group to remain in no-treatment conditions for the entire duration of any possible untreated recovery period. In fact, the design is meant precisely to allow for untreated recovery, and improvement in the no-treatment group does not, by any means, prevent a meaningful comparison from being made: The question is whether, during the time period covered by the study, the treatment group shows more improvement than the no-treatment group (Schiavetti & Metz, 1997). There is absolutely no reason to accept that experimentally sound stuttering treatment research would require large groups of children to remain as untreated controls for 24 full months postonset, as Curlee and Yairi (1997, p. 16) would have us believe. Conclusions Curlee and Yairi s (1997) article was published in the Second Opinion section of this journal. Zebrowski (1997) and Bernstein Ratner (1997) presented second and third opinions, both of which concurred with some of Curlee and Yairi s major premises. The purpose of our response has been to present another opinion that has diverged sharply in several places from those presented in the original three papers. We also suggested that many of the issues raised by Curlee and Yairi are more complex and more important than the original papers appeared to recognize. Most importantly, where Curlee and Yairi argued that our discipline s data base and our discipline s professional and research ethics support the idea of waiting to provide treatment to some children who stutter, we have argued essentially the opposite. If our discipline chooses to advocate withholding treatment from children who may not recover without it or chooses to advocate the use of indirect treatments or treatments that cannot be shown to be clinically effective, then we should not be surprised to find ourselves in the position of needing to affirm our role as the experts in the identification and management of stuttering (Zebrowski, 1997, p. 21). We suggest only that the experts on the management of any disorder might reasonably be expected to base their treatment decisions on critical analyses of all available data and might reasonably be expected to insist on treatments that have been demonstrated to be effective in well-designed experimental analyses. One of the greatest strengths of Curlee and Yairi s paper, in this light, may be that they have delivered a much-needed reality check about the status of stuttering treatment research, and we thank them for their thought-provoking contributions. Author Note Authorship is equal. Preparation of this paper was supported in part by research grant number 5 R01 DC 00060 from the National Institute of Deafness and Other Communication Disorders, National Institutes of Health, awarded to the first author. References Ambrose, N. G., Cox, N. J., & Yairi, E. (1997). The genetic basis of persistence and recovery in stuttering. Journal of Speech, Language, and Hearing Research, 40, 567 580. Andrews, G. (1984). The epidemiology of stuttering. In R. Curlee & W. H. Perkins (Eds.), Nature and treatment of stuttering: New directions (pp. 1 12). San Diego, CA: College-Hill. Andrews, G., & Harris, M. (1964). The syndrome of stuttering. London: Heinemann. Andrews, G., & Ingham, R. J. (1972). 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