Natural History of Stuttering to 4 Years of Age: A Prospective Community-Based Study

We were taken aback by the implication in the heading of Ms Fraser's letter that our paper is misleading. In no way do we say that early intervention for stuttering does not "pay off." In our paper we iterate the guidelines for intervention with the Lidcombe Program of Early Stuttering Intervention: namely, that waiting for a year after onset to see if the child recovers naturally is unlikely to decrease the child's responsiveness to the program but that the program should be implemented earlier that this if the child and/or family are distressed. These guidelines are based on extensive data from clinical trials and natural recovery research, whereas Ms Fraser's assertions on intervention for early stuttering are just that...assertions.

Conflict of Interest:

None declared

We have several concerns regarding the published study, Natural History of Stuttering to 4 Years of Age: A Prospective Community-Based Study. Our first concern is the data, which stops at age four just as one might begin to see some harmful effects from stuttering. While we understand the authors' intent is to continue to assess this cohort, the publication of intermediate findings is at best misleading and at worst irresponsible and could irreversibly discourage early intervention and parental education.

Second, the study includes only 142 children, which does not allow for credible extrapolation to the broader public.

Third, the authors recommend a "wait and see" approach for up to one year as "best practice," yet the caveats to such an approach include "unless the child is distressed, there is parental concern, or the child becomes reluctant to communicate." In our experience, which includes fielding and tracking more than 15,000 phone calls per year, these caveats - parental concern, children in distress, and trouble communicating - tend to be present in 99% of the cases, rendering the best practice recommendation moot.

Fourth, we have concerns with the conclusion, "These findings from a community-ascertained cohort refute long-held views suggesting that developmental stuttering is associated with a range of poorer outcomes. If anything, the reverse was true, with stuttering predicting subsequently better language, nonverbal skills, and psychosocial health-related quality of life at 4 years of age." If the authors found this conclusion to be new or unexpected, then they are not familiar with the existing literature which indicates that advanced language skills are in fact a risk factor for stuttering. 1,2

We have grave concerns about the way in which the children were recruited and the "priming effect," which would tend to distort the data.

Based upon current reliable research, the Stuttering Foundation sees no reason to alter its 60 year old recommendation to parents of children who stutter: early intervention is the key to preventing stuttering from becoming a lifelong problem for many young children.

1 Yairi, E. & Ambrose, N. (2005). Early Childhood Stuttering: For Clinicians by Clinicians. Pro-Ed, Austin, TX.

2 Guitar, B. & Conture, E. (2005). The Child Who Stutters: To the Pediatrician. 4th ed. Stuttering Foundation of America, Memphis TN.

Conflict of Interest:

Jane Fraser is president of the Stuttering Foundation of America.

Response to "Too much made of too little too soon" by Donaher & Kelly

Sheena Reillya,b, Mark Onslowc, Ann Packmanc, Eileen Cinia, Laura Conwaya, Obioha C Ukoumunned, Edith L Bavine, Margot Priorf, Patricia Eadieg, Susan Blockh, Melissa Wakea,b

a Murdoch Childrens Research Institute Melbourne, VIC 3052, Australia bThe University of Melbourne Department of Paediatrics at the Royal Children's Hospital, Melbourne, VIC 3052, Australia c Australian Stuttering Research Centre, The University of Sydney, Sydney, NSW 2141, Australia d PenCLAHRC, University of Exeter Medical School, University of Exeter, Devon EX2 4SG, UK e School of Psychological Science, La Trobe University, Melbourne, VIC 3086, Australia f School of Behavioural Sciences, The University of Melbourne and The Royal Children's Hospital, Melbourne, VIC 3052, Australia g Department of Audiology & Speech Pathology, The University of Melbourne, Melbourne, VIC 3052, Australia h School of Human Communication Sciences, La Trobe University, Melbourne, VIC 3086, Australia Correspondence to: Professor Sheena Reilly [email protected] Phone: + 61 3 9345 6813, Fax: + 61 3 9345 7041 Murdoch Childrens Research Institute Flemington Road, Parkville, Victoria, 3052 AUSTRALIA

Drs Donaher and Kelly (1) express concerns about the clinical implications of our recent report (2) on children who start to stutter during the pre-school years. We reported on an ongoing epidemiological study, which is based on children in the community rather than those who present for clinical assessment. Our intention is to capture rigorous prospective long-term information on all children who start to stutter in a community cohort. As we say in the report, there is a dire need to shed light on the natural history and incidence of stuttering in the preschool- aged population and to track its natural course. The data we present in our paper are for children up to age 4 years only and these data show that children who had started to stutter --as a group- -do not appear to suffer ill effects at this age. The issue that appears to be of concern to Drs Donaher and Kelly is our suggestion that this confirms that speech pathology intervention may be delayed for more than a year after onset. They say, "a wait-and-see approach is not warranted". However, our group study in no way contradicts the need for intervention soon after the onset of stuttering, if this is judged to be necessary for an individual child. We state clearly in our report that intervention should occur during the first year after onset if "the child is distressed, there is parental concern, or the child becomes reluctant to communicate" (p. 446). Drs Donaher and Kelly are correct that there is evidence that preschoolers can suffer social penalty because of their stuttering; indeed that study was conducted by two of the present authors and colleagues (3). The guideline that it is safe to wait for at least a year after onset if a child is not affected negatively by their stuttering is based on research conducted about the Lidcombe Program, which is the only speech-language pathology treatment for early stuttering that is supported by randomized controlled trials (4,5). The evidence base for this program indicates that it is efficacious and that waiting for a year after onset may actually improve a child's responsiveness to the treatment. This involves "watchful waiting", which is not the same as the "wait and see" approach suggested by Drs Donaher and Kelly. The program treatment guidelines say that intervention should occur earlier if there is concern for the well being of the child (6). We say again that the findings in our report in no way contradict current treatment guidelines, as applied to individual children. We look forward to presenting more findings from this ongoing study, once the children reach age 7 years.

References
1. Donaher JG, Kelly EM. Too much mad of too little too soon. Pediatrics. 2013;132(3). Available at: http://pediatrics.aappublications.org/content/132/3/460.short/reply#pediatrics_el_56223
2. Reilly S, Onslow M, Packman A. et al. Natural history of stuttering to 4 years of age: A prospective community-based study. Pediatrics. 2013;132(3):460-467
3. Langevin M, Packman A, Onslow M. Peer responses to stuttering in the preschool setting. Am J Speech Lang Pathol. 2009;18:264-276
4. Jones M, Onslow M, Packman A, et al, (2005). Randomised controlled trial of the Lidcombe Programme of early stuttering intervention. BMJ. 2005;331:659-661
5. Lewis C, Packman A, Onslow M, Simpson JA, Jones M. A Phase II trial of telehealth delivery of the Lidcombe Program of Early Stuttering Intervention. Am J Speech Lang Pathol. 2008;17:139-149
6. Packman A, Onslow M, Attanasio, J. The timing of early intervention with the Lidcombe Program. In M. Onslow, A. Packman, & E. Harrison (Eds.) 2003 The Lidcombe Program of early stuttering intervention: A clinician's guide. Austin, TX: Pro-Ed; 2003:41-55

Conflict of Interest:

None declared

Several clinical implications regarding, Natural History of Stuttering to 4 Years of Age: A Prospective Community-Based Study require review. Notably, the authors reported the cumulative stuttering incidence by 4 years of age was 11.2%. If the incidence is, as suggested, "more than twice that previously reported," the findings would support the allocation of increased resources and clinical training regarding young children who stutter. This is especially true given the widely held belief that intervening close to onset significantly improves clinical outcomes for children and their families. The authors' finding that only 9 of 142 children recovered within 12 months further amplifies the need for doing "something" rather than "nothing" to help children who will apparently stutter for a prolonged period.

Contrary to the literature on adults who stutter, children in this sample "showed little evidence of harm to their mental health, temperament, or psychosocial health-related quality of life". Methodological issues, including a lack of heterogeneity between groups and questionable validity/reliability and responsiveness to change of the selected instruments for use in very young children, call into question the interpretation of the data. Furthermore, the small effect sizes preclude the findings from being generalizable. It is premature to say that developmental stuttering is not "associated with a range of poorer outcomes," when evaluating a sample that has only reached 4 years of age. It is important to note that research has indicated negative responses and problematic "in-the-moment" reactions to stuttering in young children. Until it is conclusively determined that these responses have no long-term impact, such responses must be addressed clinically when they occur. By tracking this cohort until at least 8-9 years of age, past the point of natural recovery, the researchers may be better positioned to report on the onset/course of stuttering while possibly adding to the emerging literature on predictive factors for persistent stuttering. Until then, their conclusions regarding recovery are speculative at best and the existing research base on the contributions of family history, genetics, acoustic properties of stuttering behaviors, subtle differences in linguistic abilities, impact on communication, temperamental contributions, and neurological structuring should not be ignored.

A multitude of individual factors must be judiciously considered when determining risk and making recommendations for therapy. When a parent's level of concern results in seeking professional guidance, or when a child reacts to difficulties experienced while trying to speak by refusing to talk, covering his mouth, or asking why he can't speak, it suggests a deviation from the typical pattern of communicative development. Logically, then, a wait-and-see approach is not warranted and the appropriate response would be to refer to a speech-language pathologist who is capable of making a developmentally informed decision. By assessing the child's communicative skills, emotional/social functioning and developmental history, in addition to gathering family input, providing resources, education and counseling, intervention has begun, and may be provided by means that are neither overly "intensive" nor "expensive". This step, regardless of whether direct therapeutic services, an indirect home program or continued monitoring is recommended, is "best practice."

Joseph Donaher, Ph.D., CCC/SLP Board Recognized Specialist - Fluency Disorders Academic and Research Program Director The Center for Childhood Communication The Children's Hospital of Philadelphia

Ellen M. Kelly, Ph.D., CCC-SLP Associate Professor Department Of Hearing and Speech Sciences Vanderbilt University Medical Center

Conflict of Interest:

None declared