Toward a paradigm shift from deficit-based to proactive speech and language treatment: Randomized pilot trial of the Babble Boot Camp in infants with classic galactosemia

Beate Peter, Nancy Potter, Jennifer Davis, Inbal Donenfeld-Peled, Lizbeth Finestack, Carol Stoel-Gammon, Kari Lien, Laurel Bruce, Caitlin Vose, Linda Eng, Hanako Yokoyama, Daniel Olds, Mark VanDam, Beate Peter, Nancy Potter, Jennifer Davis, Inbal Donenfeld-Peled, Lizbeth Finestack, Carol Stoel-Gammon, Kari Lien, Laurel Bruce, Caitlin Vose, Linda Eng, Hanako Yokoyama, Daniel Olds, Mark VanDam

Abstract

Background: Speech and language therapy is typically initiated reactively after a child shows delays. Infants with classic galactosemia (CG), a metabolic disease with a known high risk for both speech and language disorders, hold the keys towards evaluating whether preventive treatment is effective when the risks are known at birth. We present pilot data from a randomized parallel trial of an innovative proactive speech and language intervention program, the Babble Boot Camp (BBC). Method: Five children with CG, otherwise healthy, participated in the study from approximately 2 to 24 months of age. One of these was randomly selected as control receiving conventional management, which typically starts at age 2-3 years. A pediatric speech-language pathologist met weekly via telepractice with the parents in the treatment cohort. Parents implemented the prespeech, speech, and language stimulation and expansion activities according to the intervention protocol. The control child was still too young for conventional treatment. Primary outcome measures were speech sound production complexity in babble and speech and expressive vocabulary size. Secondary outcome measures were vocalization rates and developmental milestones in communication, motor, and cognition. The trial is ongoing. Results: All four treated children had higher speech sound skills in babble, three had higher speech sound skills in meaningful speech, two had higher expressive vocabularies, three had higher global developmental scores, and two had higher vocalization rates, compared to the control child with CG. Discussion: Given the high risk for speech and language delays in children with CG, finding on-schedule abilities in two or more of the treated children but not the untreated child is unexpected under random conditions. The trends toward beneficial effects of the BBC on speech sound production, expressive language, and communication milestones warrant appropriately powered larger clinical trials with full randomization. Trial registration: ClinicalTrials.gov NCT03838016 (12 th February 2019).

Keywords: genetic risk; infant; language impairment; prevention; speech disorder; very early intervention.

Conflict of interest statement

No competing interests were disclosed.

Copyright: © 2020 Peter B et al.

Figures

Figure 1.. Mean Babbling Level (MBL) scores…
Figure 1.. Mean Babbling Level (MBL) scores for the four children with CG in the treatment cohort (blue symbols), the control child with CG (green symbols), and typically developing children (black symbols, archival data from ( Morris, 2010)).
Figure 2.. Syllable Structure Level (SSL) scores…
Figure 2.. Syllable Structure Level (SSL) scores for the four children with CG in the treatment cohort (blue symbols), the control child with CG (green symbols), and typically developing children (black symbols archival data from ( Morris, 2010)).
Figure 3.. Expressive vocabulary size for ages…
Figure 3.. Expressive vocabulary size for ages 12 through 24 months, shown as percentile rankings, for the four children with CG in the treatment cohort (blue symbols) and the control child with CG (green symbols).
Figure 4.. Receptive vocabulary size for ages…
Figure 4.. Receptive vocabulary size for ages 12 and 15 months, shown as percentile rankings, for the four children with CG in the treatment cohort (blue symbols) and the control child with CG (green symbols).
Figure 5.. Number of child vocalizations per…
Figure 5.. Number of child vocalizations per hour for ages 3 through 24 months for the four children with CG in the treatment cohort (blue symbols) and the control child with CG (green symbols).

References

    1. Akamoglu Y, Dinnebeil L: Coaching parents to use naturalistic language and communication strategies. Young Exceptional Children. 2017;20(1):41–50. 10.1177/1096250615598815
    1. American Speech-Language-Hearing Association: Childhood Apraxia of Speech [Position Statement].2007. 10.1044/policy.PS2007-00277
    1. Antshel KM, Epstein IO, Waisbren SE: Cognitive strengths and weaknesses in children and adolescents homozygous for the galactosemia Q188R mutation: a descriptive study. Neuropsychology. 2004;18(4):658–64. 10.1037/0894-4105.18.4.658
    1. Black LI, Vabratian A, Hoffman HJ: Communication disorders and use of intervention services among children aged 3-17 years: United States, 2012. NCHS Data Brief. 2015; (205):1–8.
    1. Cane J, O'Connor D, Michie S, et al. : Validation of the theoretical domains framework for use in behaviour change and implementation research. Implement Sci. 2012;7:37. 10.1186/1748-5908-7-37
    1. Cates CB, Dreyer BP, Berkule SB, et al. : Infant communication and subsequent language development in children from low-income families: the role of early cognitive stimulation. J Dev Behav Pediatr. 2012;33(7):577–85. 10.1097/DBP.0b013e318264c10f
    1. Chapman RS: Children's language learning: an interactionist perspective. J Child Psychol Psychiatry. 2000;41(1):33–54. 10.1111/1469-7610.00548
    1. Coss KP, Doran PP, Owoeye C, et al. : Classical Galactosaemia in Ireland: incidence, complications and outcomes of treatment. J Inherit Metab Dis. 2013;36(1):21–27. 10.1007/s10545-012-9507-9
    1. Dawson G, Rogers S, Munson J, et al. : Randomized, controlled trial of an intervention for toddlers with autism: the Early Start Denver Model. Pediatrics. 2010;125(1):e17–23. 10.1542/peds.2009-0958
    1. DePaolis RA, Vihman MM, Nakai S: The influence of babbling patterns on the processing of speech. Infant Behav Dev. 2013;36(4):642–9. 10.1016/j.infbeh.2013.06.007
    1. Dimitrova N, Ozcaliskan S, Adamson LB: Parents' Translations of Child Gesture Facilitate Word Learning in Children with Autism, Down Syndrome and Typical Development. J Autism Dev Disord. 2016;46(1):221–231. 10.1007/s10803-015-2566-7
    1. Fenson L, Marchman VA, Thal DJ, et al. : MacArthur-Bates Communicative Development Inventories: User's Guide and Technical Manual. (2nd Ed.). Brookes.2007.
    1. Goldman R, Fristoe M: Goldman-Fristoe Test of Articulation - 3. Pearson.2015.
    1. Gordon-Brannan ME, Weiss CE: Clinical management of articulatory and phonologic disorders. Lippincott Williams & Wilkins.2007.
    1. Guralnick MJ: Why Early Intervention Works: A Systems Perspective. Infants Young Child. 2011;24(1):6–28. 10.1097/IYC.0b013e3182002cfe
    1. Hassink JM, Leonard LB: Within-treatment factors as predictors of outcomes following conversational recasting. Am J Speech Lang Pathol. 2010;19(3):213–24. 10.1044/1058-0360(2010/09-0083)
    1. Hoffmann TC, Glasziou PP, Boutron I, et al. : Better Reporting of Interventions: Template for Intervention Description and Replication (TIDieR) Checklist and Guide. BMJ. 2014;348:168724. 10.1136/bmj.g1687
    1. Hughes J, Ryan S, Lambert D, et al. : Outcomes of siblings with classical galactosemia. J Pediatr. 2009;154(5):721–6. 10.1016/j.jpeds.2008.11.052
    1. Jumbo-Lucioni PP, Garber K, Kiel J, et al. : Diversity of approaches to classic galactosemia around the world: a comparison of diagnosis, intervention, and outcomes. J Inherit Metab Dis. 2012;35(6):1037–1049. 10.1007/s10545-012-9477-y
    1. Justice LM, Logan JR, Damschroder L: Designing Caregiver-Implemented Shared-Reading Interventions to Overcome Implementation Barriers. J Speech Lang Hear Res. 2015;58(6):S1851–1863. 10.1044/2015_JSLHR-L-14-0344
    1. Karadag N, Zenciroglu A, Eminoglu FT, et al. : Literature review and outcome of classic galactosemia diagnosed in the neonatal period. Clin Lab. 2013;59(9–10):1139–46. 10.7754/Clin.Lab.2013.121235
    1. Ludemann A, Power E, Hoffmann TC: Investigating the Adequacy of Intervention Descriptions in Recent Speech-Language Pathology Literature: Is Evidence From Randomized Trials Useable? Am J Speech Lang Pathol. 2017;26(2):443–455. 10.1044/2016_AJSLP-16-0035
    1. MacLeod A: The trajectory toward mastery of the phonological system. In: Peter B, MacLeod A, eds. Comprehensive perspectives on speech sound development and disorders: Pathways from linguistic theory to clinical practice New York: Nova.2013.
    1. McCune L, Vihman MM: Early phonetic and lexical development: a productivity approach. J Speech Lang Hear Res. 2001;44(3):670–84. 10.1044/1092-4388(2001/054)
    1. Morris SR: Clinical application of the mean babbling level and syllable structure level. Lang Speech Hear Serv Sch. 2010;41(2):223–30. 10.1044/0161-1461(2009/08-0076)
    1. Mundy P, Block J, Delgado C, et al. : Individual differences and the development of joint attention in infancy. Child Dev. 2007;78(3):938–54. 10.1111/j.1467-8624.2007.01042.x
    1. Oller DK, Eilers RE, Neal AR, et al. : Late onset canonical babbling: a possible early marker of abnormal development. Am J Ment Retard. 1998;103(3):249–63. 10.1352/0895-8017(1998)103<0249:LOCBAP>;2
    1. Oller DK, Eilers RE, Neal AR, et al. : Precursors to speech in infancy: the prediction of speech and language disorders. J Commun Disord. 1999;32(4):223–245. 10.1016/S0021-9924(99)00013-1
    1. Paul R, Jennings P: Phonological behavior in toddlers with slow expressive language development. J Speech Hear Res. 1992;35(1):99–107. 10.1044/jshr.3501.99
    1. Paul R, Norbury CF: Language disorders from infancy through adolescence.Elsevier.2012.
    1. Peter B: Babble Boot Camp. OSF. 2019. 10.17605/
    1. Potter NL, Lazarus JA, Johnson JM, et al. : Correlates of language impairment in children with galactosaemia. J Inherit Metab Dis. 2008;31(4):524–532. 10.1007/s10545-008-0877-y
    1. Potter NL, Nievergelt Y, Shriberg LD: Motor and speech disorders in classic galactosemia. JIMD Rep. 2013;11:31–41. 10.1007/8904_2013_219
    1. Roberts MY, Kaiser AP: The effectiveness of parent-implemented language interventions: a meta-analysis. Am J Speech Lang Pathol. 2011;20(3):180–199. 10.1044/1058-0360(2011/10-0055)
    1. Roberts MY, Kaiser AP, Wolfe CE, et al. : Effects of the teach-model-coach-review instructional approach on caregiver use of language support strategies and children's expressive language skills. J Speech Lang Hear Res. 2014;57(5):1851–1869. 10.1044/2014_JSLHR-L-13-0113
    1. Rogers SJ, Vismara L, Wagner AL, et al. : Autism treatment in the first year of life: a pilot study of infant start, a parent-implemented intervention for symptomatic infants. J Autism Dev Disord. 2014;44(12):2981–2995. 10.1007/s10803-014-2202-y
    1. Rowe ML, Goldin-Meadow S: Differences in early gesture explain SES disparities in child vocabulary size at school entry. Science. 2009;323(5916):951–953. 10.1126/science.1167025
    1. Schonhaut L, Armijo I, Schönstedt M, et al. : Validity of the ages and stages questionnaires in term and preterm infants. Pediatrics. 2013;131(5):e1468–1474. 10.1542/peds.2012-3313
    1. Semel E, Wiig E, Secord W: Clinical Evaluation of Language Fundamentals - Preschool - 2. Pearson. 2004.
    1. Shriberg LD, Tomblin JB, McSweeny JL: Prevalence of speech delay in 6-year-old children and comorbidity with language impairment. J Speech Lang Hear Res. 1999;42(6):1461–1481. 10.1044/jslhr.4206.1461
    1. Squires J, Bricker D: Ages & Stages Questionnaires. 3rd Ed. Brookes.2009.
    1. Stoel-Gammon C: Constraints on consonant-vowel sequences in early words. J Child Lang. 1983;10(2):455–457. 10.1017/S0305000900007893
    1. Stoel-Gammon C: Prespeech and early speech development of two late talkers. First Lang. 1989;9:207–223. 10.1177/014272378900900607
    1. Stoel-Gammon C: Relationships between lexical and phonological development in young children. J Child Lang. 2011;38(1):1–34. 10.1017/S0305000910000425
    1. Storkel HL, Komesidou R, Fleming KK, et al. : Interactive Book Reading to Accelerate Word Learning by Kindergarten Children With Specific Language Impairment: Identifying Adequate Progress and Successful Learning Patterns. Lang Speech Hear Serv Sch. 2017;48(2):108–124. 10.1044/2017_LSHSS-16-0058
    1. Tomblin JB, Records NL, Buckwalter P, et al. : Prevalence of specific language impairment in kindergarten children. J Speech Hear Res. 1997;40(6):1245–1260. 10.1044/jslhr.4006.1245
    1. Tomasello M, Farrar MJ: Joint attention and early language. Child Dev. 1986;57(6):1454–1463. 10.2307/1130423
    1. Tomasello M, Kruger AC: Joint attention on actions: acquiring verbs in ostensive and non-ostensive contexts. J Child Lang. 1992;19(2):311–333. 10.1017/S0305000900011430
    1. Vihman MM, Greenlee M: Individual differences in phonological development: ages one and three years. J Speech Hear Res. 1987;30(4):503–521. 10.1044/jshr.3004.503
    1. Vihman MM, Macken MA, Miller R, et al. : From Babbling to Speech - a Re-Assessment of the Continuity Issue. Language. 1985;61(2):397–445. 10.2307/414151
    1. von Hapsburg D, Davis BL: Auditory sensitivity and the prelinguistic vocalizations of early-amplified infants. J Speech Lang Hear Res. 2006;49(4):809–822. 10.1044/1092-4388(2006/057)
    1. Vygotsky LS: The Development of Higher Forms of Attention in Childhood. Sov Psychol. 1979;18(1):67–115. 10.2753/RPO1061-0405180167
    1. Waggoner DD, Buist NR, Donnell GN: Long-term prognosis in galactosaemia: results of a survey of 350 cases. J Inherit Metab Dis. 1990;13(6):802–818. 10.1007/BF01800204
    1. Wetherby AM, Guthrie W, Woods J, et al. : Parent-implemented social intervention for toddlers with autism: an RCT. Pediatrics. 2014;134(6):1084–1093. 10.1542/peds.2014-0757
    1. : Better reporting of interventions: template for intervention description and replication (TIDieR) checklist and guide. BMJ.2014;348: 10.1136/bmj.g1687 g1687 10.1136/bmj.g1687
    1. : Investigating the Adequacy of Intervention Descriptions in Recent Speech-Language Pathology Literature: Is Evidence From Randomized Trials Useable?. Am J Speech Lang Pathol.2017;26(2) : 10.1044/2016_AJSLP-16-0035 443-455 10.1044/2016_AJSLP-16-0035
    1. : Validation of the theoretical domains framework for use in behaviour change and implementation research. Implement Sci.2012;7: 10.1186/1748-5908-7-37 37 10.1186/1748-5908-7-37
    1. : Designing Caregiver-Implemented Shared-Reading Interventions to Overcome Implementation Barriers. J Speech Lang Hear Res.2015;58(6) : 10.1044/2015_JSLHR-L-14-0344 S1851-63 10.1044/2015_JSLHR-L-14-0344
    1. : How to assess and compare inter-rater reliability, agreement and correlation of ratings: an exemplary analysis of mother-father and parent-teacher expressive vocabulary rating pairs. Front Psychol.2014;5: 10.3389/fpsyg.2014.00509 509 10.3389/fpsyg.2014.00509
    1. : Joint attention and lexical acquisition style. First Language.1983;4(12) : 10.1177/014272378300401202 197-211 10.1177/014272378300401202
    1. : The Effectiveness of Parent-Implemented Language Interventions: A Meta-Analysis. American Journal of Speech-Language Pathology.2011;20(3) : 10.1044/1058-0360(2011/10-0055) 180-199 10.1044/1058-0360(2011/10-0055)
    1. : Effects of the teach-model-coach-review instructional approach on caregiver use of language support strategies and children's expressive language skills. J Speech Lang Hear Res.2014;57(5) : 10.1044/2014_JSLHR-L-13-0113 1851-69 10.1044/2014_JSLHR-L-13-0113
    1. : Early Intervention (Practice Portal). [accessed 28th April 2019].
    1. : DEC recommended practices in early intervention/early childhood special education 2014. [accessed 28th April 2019].
    1. : Coaching in Parent-Implemented Early Communication Interventions: Understanding and Overcoming Individual-Level Implementation Barriers. Perspectives of the ASHA Special Interest Groups.2016;1(1) : 10.1044/persp1.SIG1.144 144-153 10.1044/persp1.SIG1.144
    1. : Effects of a Triadic Parent-Implemented Home-Based Communication Intervention for Toddlers. Journal of Early Intervention.2015;37(1) : 10.1177/1053815115589350 44-68 10.1177/1053815115589350
    1. : Parent-Implemented Communication Intervention. Topics in Early Childhood Special Education.2016;36(2) : 10.1177/0271121416628200 115-124 10.1177/0271121416628200
    1. : Here's how to do early intervention for speech and language: Empowering parents. Plural Publishing.2012;
    1. : A primary-coach approach to teaming and supporting families in early childhood intervention. Working with families of young children with special needs.2010;175-202
    1. : Coaching parents to use naturalistic language and communication strategies. Young Exceptional Children.2017;20:41-50
    1. : Parent-implemented social intervention for toddlers with autism: an RCT. Pediatrics.2014;134(6) : 10.1542/peds.2014-0757 1084-93 10.1542/peds.2014-0757

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