Characteristics of Fluency and Speech in Two Families With High Incidences of Stuttering Purpose This study presents data from 2 families with high incidence of stuttering, comparing methods of phenotype assignment and exploring the presence of other fluency disorders and corresponding speech characteristics. Method Three methods for assigning phenotype of stuttering were used: self-identification, family identification, and expert identification. Agreement on ... Research Article
Research Article  |   October 01, 2015
Characteristics of Fluency and Speech in Two Families With High Incidences of Stuttering
 
Author Affiliations & Notes
  • Sheila V. Stager
    National Institute on Deafness and Other Communication Disorders, National Institutes of Health, Bethesda, MD
  • Frances J. Freeman
    Stephen F. Austin State University, Nacogdoches, TX
  • Allen Braun
    National Institute on Deafness and Other Communication Disorders, National Institutes of Health, Bethesda, MD
  • Disclosure: The authors have declared that no competing interests existed at the time of publication.
    Disclosure: The authors have declared that no competing interests existed at the time of publication. ×
  • Correspondence to Sheila V. Stager: sstager@mfa.gwu.ed
  • Editor: Jody Kreiman
    Editor: Jody Kreiman×
  • Associate Editor: Hans-Georg Bosshardt
    Associate Editor: Hans-Georg Bosshardt×
Article Information
Speech, Voice & Prosodic Disorders / Fluency Disorders / Speech, Voice & Prosody / Speech / Research Articles
Research Article   |   October 01, 2015
Characteristics of Fluency and Speech in Two Families With High Incidences of Stuttering
Journal of Speech, Language, and Hearing Research, October 2015, Vol. 58, 1440-1451. doi:10.1044/2015_JSLHR-S-14-0080
History: Received March 10, 2014 , Revised August 19, 2014 , Accepted June 24, 2015
 
Journal of Speech, Language, and Hearing Research, October 2015, Vol. 58, 1440-1451. doi:10.1044/2015_JSLHR-S-14-0080
History: Received March 10, 2014; Revised August 19, 2014; Accepted June 24, 2015

Purpose This study presents data from 2 families with high incidence of stuttering, comparing methods of phenotype assignment and exploring the presence of other fluency disorders and corresponding speech characteristics.

Method Three methods for assigning phenotype of stuttering were used: self-identification, family identification, and expert identification. Agreement on which individuals were assigned by each of these methods was studied. Multiple measures of fluency and speech production were obtained.

Results Self-reports and descriptions of blocking rather than self-identification as a person who stutters demonstrated the best agreement with expert identification of stuttering. Family identification showed poor agreement with both expert and self-identification of stuttering. Using binary categories of fluent or stuttering, 90% of individuals in 1 family were classified by expert consensus. Only 70% of the other family could be similarly categorized. Experts required 2 other categories, cluttering and other fluency disorders, to fully characterize dysfluency within this family. These 2 families also demonstrated differences in speech production.

Conclusion Some families with high incidence of stuttering may also have high incidence of other fluency disorders and other speech-production difficulties. This finding may have ramifications for genetic studies, including criteria for defining phenotype and collapsing data across multiple families.

Acknowledgments
This research was funded by the Intramural Division of the National Institute on Deafness and Other Communications, Protocol #97-DC-0002, titled “Genetic Linkage Analysis in Developmental Stuttering: Gene Mapping in Extended Kindreds and Candidate Gene Analyses” (Principal Investigator: Sheila Stager).
We would like to acknowledge the contribution of those who joined the authors in serving as experts on our panel: Edward Conture, Barry Guitar, Tom Gurrister, Peter Ramig, and Ben Watson. They gave significantly of their time in reviewing the patient tapes, filling out the forms, and coming to Bethesda for the consensus conference. The analyses could not have been completed without their clinical acumen. We would also like to acknowledge Klaas Bakker and Florence Myers, who reviewed a subset of the subjects following the consensus conference. Again, they gave significantly of their time in reviewing patient tapes and their efforts helped to clarify the classification of those individuals for whom the panel could not achieve consensus. We would also like to acknowledge the willingness, time, and patience of the family members who participated in this study.
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