Title: Caregiver Ratings of Appropriateness of the Children’s Communication Checklist – 2 (CCC-2) in Youth with Down Syndrome

Authors: Rebekah Bosley1, Marie Moore Channell1, Alexandria Barkhimer1, Laura J. Mattie1, Amy P. Cohen1, Natasha Ludwig2, & Tracie C. Rosser3

Introduction: Youth with Down syndrome (DS) have a unique communication profile of expressive language delays (e.g., syntax, morphology) paired with relatively strong pragmatic language skills (Grieco et al., 2015). The Children’s Communication Checklist-2 (CCC-2; Bishop, 2006) is a standardized caregiver rating scale designed to examine different communication skills and identify youth with pragmatic language or other speech and language impairments. The CCC-2 is the only standardized caregiver report measure of broad language normed for older children and adolescents (4:0-16:11 years), but it is only validated for those who speak in simple sentences. However, many individuals with DS in this age range do not yet speak in sentences. Further, although some studies have provided initial evidence of the use of the CCC/CCC-2 in individuals with DS to examine their pragmatic and structural language profiles (de la Torre Carril et al., 2021; Laws & Bishop, 2004; Lee et al., 2017; Losh et al., 2012; Smith et al., 2017; Stephen et al., 2021), the extent to which this measure adequately captures their language and communication skills in authentic settings is not yet known. One approach is to ask caregivers to indicate the appropriateness, or applicability, of the questions for their youth with DS. Thus, the present study examined the use of the CCC-2 in a large sample of youth with DS, some of whom spoke in sentences and some of whom used single words or phrases. We aimed to (1) examine caregiver ratings of the appropriateness of the CCC-2 items across the sample, and (2) determine whether caregiver appropriateness ratings differed for those who speak in sentences vs. those who do not.

Method: Participants were 94 caregivers (94.7% mothers) of youth with DS (6-16 years; M = 11.5; SD = 3.2; 54.3% female; 79.8% White, 10.6% Multi-racial, 4.3% Black, 4.3% Asian, 1.1% Other; 7.4% Hispanic) who completed a large nationwide survey on autism characteristics and development in youth with DS. For the current study, only youth who used speech as a primary mode of communication were included. Via caregiver report, 54.3% (n = 51) spoke in single words or phrases, and 45.7% (n = 43) spoke using sentences. Additionally, 25.5% of the sample had uncorrected hearing loss. Caregivers completed the CCC-2, a 70-item questionnaire measuring language (i.e., speech, syntax, semantics, and coherence) and pragmatics (i.e., initiations, scripted language, context, nonverbal communication, social relations, and interests). At the end of the questionnaire, we asked caregivers if any of the CCC-2 items were inappropriate for or not applicable to their youth (yes/no).

Results: For Aim 1, in the full sample of youth with DS, 35.1% (n = 33) of caregivers indicated that some items were not appropriate or applicable for their youth (“Inappropriate”); 64.9% (n = 61) indicated that all items were appropriate (“Appropriate”). For Aim 2, there was a significant association between the youth’s verbal ability level (speaks in sentences vs. single words or phrases) and caregiver rating groups (Inappropriate vs. Appropriate), Χ2(1) = 19.18, p <.001. 15.2% (n = 5) of caregivers whose youth speak in sentences rated the CCC-2 as “Inappropriate,” whereas 84.8% (n = 28) whose youth speak in single words or phrases rated it as “Inappropriate.” There was no significant difference in the CCC-2 Global Communication Composite standard scores between the Inappropriate (M = 71.0, SD = 12.4) and Appropriate (M = 71.6, SD = 13.1) groups, t (92) = -.23, p = .82, Cohen’s d = -.05.

Discussion: Approximately one-third of caregivers stated that the CCC-2 included some items that were not appropriate for, or applicable to, their youth with DS. Significantly more caregivers whose youth do not yet speak in sentences rated the CCC-2 as including items that were inappropriate for their youth (compared to those whose youth speak in sentences). This is not surprising given that the CCC-2 was originally validated for children who speak in sentences. However, regardless of their youth’s verbal ability level, some caregivers reported that the CCC-2 included items that were not appropriate or applicable to their youth. Thus, it may not be a functional measure of communication, at least for some individuals with DS. Future research should evaluate the utility of the CCC-2 across the range of characteristics and communication profiles observed among individuals with DS. Despite caregiver ratings, across our sample, those who speak in simple sentences had higher standard scores on the CCC-2 than those who speak in single words or phrases, indicating the CCC-2 at least broadly differentiated language and communication skills in the expected direction.

Key References:

de la Torre Carril, A.; Durán-Bouza, M.; Pérez-Pereira, M. Capacity of the CCC-2 to Discriminate ASD from Other Neurodevelopmental Disorders. Children 2021, 8, 640. https://doi.org/10.3390/children8080640

Laws, G., & Bishop, D. V. (2004). Pragmatic language impairment and social deficits in Williams syndrome: a

comparison with Down's syndrome and specific language impairment. International Journal of Language & Communication Disorders, 39(1), 45-64. https://doi.org/10.1080/13682820310001615797

Lee, M., Bush, L., Martin, G. E., Barstein, J., Maltman, N., Klusek, J., & Losh, M. (2017). A multi-method investigation

of pragmatic development in individuals with Down syndrome. American journal on intellectual and developmental disabilities, 122(4), 289-309. https://doi.org/10.1352/1944-7558-122.4.289

Losh, M., Martin, G. E., Klusek, J., Hogan-Brown, A. L., & Sideris, J. (2012). Social communication and theory of mind

in boys with autism and fragile X syndrome. Frontiers in psychology, 3, 266. https://doi.org/10.3389/fpsyg.2012.00266

Smith, E., Næss, K.-A. B., & Jarrold, C. (2017). Assessing pragmatic communication in children with Down syndrome. Journal of Communication Disorders, 68, 10-23. https://doi.org/10.1016/j.jcomdis.2017.06.003

Stephan, C., Clasen, L., Adeyemi, E., & Lee, N. R. (2021). Speech Impairments Explain Unique Variance in Adaptive

Behavior Skills in Young People With Down Syndrome. American journal of speech-language pathology, 30(1), 253–259. https://doi.org/10.1044/2020_AJSLP-20-00054

1University of Illinois Urbana-Champaign, 2Kennedy Krieger Institute, 3Emory University

Funding support: NIH grant R21HD106125

1. Grieco, J., Pulsifer, M., Seligsohn, K., Skotko, B., & Schwartz, A. (2015). Down syndrome: Cognitive and behavioral functioning across the lifespan. American Journal of Medical Genetics, Part C: Seminars in Medical Genetics, 1

2. de la Torre Carril, A.; Durán-Bouza, M.; Pérez-Pereira, M. Capacity of the CCC-2 to Discriminate ASD from Other Neurodevelopmental Disorders. Children 2021, 8, 640. https://doi.org/10.3390/children8080640

3. Laws, G., & Bishop, D. V. (2004). Pragmatic language impairment and social deficits in Williams syndrome: a comparison with Down's syndrome and specific language impairment. International Journal of Language & Communication Disorders, 39(1), 45-64. https://doi.org/10.1080/13682820310001615797

4. Lee, M., Bush, L., Martin, G. E., Barstein, J., Maltman, N., Klusek, J., & Losh, M. (2017). A multi-method investigation of pragmatic development in individuals with Down syndrome. American journal on intellectual and developmental disabilities, 122(4), 289-309. https://doi.org/10.1352/1944-7558-122.4.289

5. Losh, M., Martin, G. E., Klusek, J., Hogan-Brown, A. L., & Sideris, J. (2012). Social communication and theory of mind in boys with autism and fragile X syndrome. Frontiers in psychology, 3, 266. https://doi.org/10.3389/fpsyg.2012.00266

6. Smith, E., Næss, K.-A. B., & Jarrold, C. (2017). Assessing pragmatic communication in children with Down syndrome. Journal of Communication Disorders, 68, 10-23. https://doi.org/10.1016/j.jcomdis.2017.06.003

7. Stephan, C., Clasen, L., Adeyemi, E., & Lee, N. R. (2021). Speech Impairments Explain Unique Variance in Adaptive Behavior Skills in Young People With Down Syndrome. American journal of speech-language pathology, 30(1), 253–259. https://doi.org/10.1044/2020_AJSLP-20-00054

8. Karmiloff-Smith, A., Al-Janabi, T., D'Souza, H., Groet, J., Massand, E., Mok, K., ... & Strydom, A. (2016). The importance of understanding individual differences in Down syndrome. F1000Research, 5. 

Background: Caregiver language facilitates development in children with developmental disabilities (Anderson et al., 2021). Research has examined caregiver-child interactions with preschool-age children with Down syndrome and found them more directive than caregivers of age-matched children with neurotypical development. A notable gap exists concerning caregiver input to school-age children with Down syndrome. Evidence indicates that children with Down syndrome continue to develop language throughout school-age, and caregivers report regularly reading with their school-age children with Down syndrome. However, until this study, no research had examined the nature of these interactions.

Research Question: What are the key features of maternal talk to their school-age children with Down syndrome during a wordless storybook interaction?

Methods: Mothers of 40 school-age children with Down syndrome (6-11 years) completed a wordless storybook task that was recorded and transcribed using Systematic Analysis of Language Transcripts software (Miller & Iglesias, 2012). Maternal talk was coded for communicative function: Prompting Communication (e.g., asking questions; encouraging child talk), Verbal Responding (e.g., answering questions; responding to child talk), and Directing Behavior (e.g., maintaining the interaction). The coding scheme was modified from prior literature (Hilvert et al., 2021; Sterling & Warren, 2014; 2018) to fit a wordless storybook.

Results: The rate of maternal talk (i.e., frequency of type of talk per minute) was highest for Prompting Communication (M = 3.17), then Verbal Responding (M = 0.92), and Directing Behavior (M = 0.32). Maternal talk that was off-task (M = 0.11) or storytelling (M = 4.13) was coded but excluded from analyses.

Conclusion: As the first study examining caregiver talk with school-age children with Down syndrome, caregivers actively provided opportunities for their children to engage in the storybook. Notably, caregivers were not as directive as compared to previous findings. Future research must replicate and extend these findings, including caregiver talk to school-age children in other communication contexts.

References:

• Anderson, N. J., Graham, S. A., Prime, H., Jenkins, J. M., & Madigan, S. (2021). Linking quality and quantity of parental linguistic input to child language skills: A meta‐analysis. Child Development, 92(2), 484-501.

• Hilvert, E., Lorang, E., & Sterling, A. (2021). Maternal use of decontextualized and contextualized talk: An in-depth investigation of early parent–child interactions in Down syndrome. American Journal of Speech-Language Pathology, 30(4), 1767-1780.

• Miller, J., & Iglesias, A. (2012). Systematic Analysis of Language Transcripts (SALT) (Research Version 2012).

• Sterling, A., & Warren, S. F. (2014). Maternal Responsivity in Mothers of Young Children with Down Syndrome. Developmental Neurorehabilitation, 17(5), 306–317. https://doi.org/10.1038/jid.2014.371

• Sterling, A., & Warren, S. F. (2018). Parenting of children with Down syndrome compared to fragile X syndrome. Developmental Neurorehabilitation, 21(1), 64–67. https://doi.org/10.1080/17518423.2016.1259274