Forging Successful Research Collaborations to Study Outcomes in Communication Disorders

1. Forging Successful Research Collaborations to Study Outcomes in Communication Disorders J Bruce Tomblin, University of Iowa Mary P Moeller, Boys Town National Research Hospital Supported by NIDCD R01 DC009560

2. Goals of Presentation • Discuss process of identifying gaps in research related to children with mild to severe hearing loss • Describe development of multi-center, multi-disciplinary grant to address gaps • Summarize progress of project to date • Review collaborative strategies leading to effective team function

3. Project Background • NIDCD working group (12/2006) • Identify research gaps (mild-severe HL) • Current State of Knowledge: • Research Methods (outcomes) • Language & Literacy • Psychosocial Development • Perceptual Processing • Speech Recognition & Production Ear & Hearing, December, 2007

4. Background • Most outcome studies focus on children who are deaf • Reduced body of literature concerning children with mild to severe HL • Sample sizes are small or mix D/HH children • Lack of control of amplification histories/audibility • Few studies attempted a population sample • Varied measurement strategies; earlier generation technologies • Need to understand sources of individual difference in outcomes

5. Summary of Research on Speech Outcomes • Delays in babble onset increase with increasing hearing loss (Carney, 1996) • Some children at risk for slow transitions from babble to word productions (Moeller, et al., 2007) • Consonant repertoire differences-e.g., fricatives (McGowan, et al, 2008; Moeller, et.al, 2007) • Generally intelligible speech as they mature (Wallace, et al, 2000) • Number & type of phoneme errors increase with increased severity of loss (Elfenbein, et al, 1994) • Substitution of fricatives & affricates most common

6. Summary of Research on Language Outcomes • Varied findings related to vocabulary development • Early lexicon delayed (Mayne, et al, 2000, Kiese-Himmel & Reeh, 2006) • Delayed at school age (Davis, et al, 1986; Wake, et al, 2004) • Not delayed at school age (Plapinger & Sikora, 1995; Wolgemuth, et al, 1998) • Bimodal distribution of performance (Gilbertson & Kamhi, 1995) • Less efficiency in Novel Word Learning (Pittman, et al, 2005) • Prolonged lexical access (Jerger et al, 2006) • Delays in morphology (McGukian & Henry, 2007; Brown, 1984; Norbury, et al, 2001) • 3 person singular, past tense markers delayed in preschoolers with mild-moderate HL • Grammar understanding comparable to peers with NH (Briscoe, et al, 2001; Gilbertson & Kamhi, 1995) • Few studies address narrative skills, discourse and verbal reasoning or intervention

7. Language for Social Purposes *Age when 2/3 of group passed item Unpublished data from Dr. A. Sedey (Colorado), analysis of 725 assessments from 352 children, ages 6 mo - 6 years, mild to profound

8. Importance of Classroom Social Experiences Academic Self Concept Lack of Peer Acceptance Academic Performance Internalizing Symptoms Bottom line: Promotion of positive relationships with peers contributes not only to social development, but emotional and cognitive (learning/academic) Flook, Repetti & Ullman, 2005

9. Psychosocial Effects on Learning • Quality of social relationships tied to academic progress (Flook, et al, 2005) • Learning with friend vs. classmate impacts (Newcomb & Bagwell, 1995): • Conflict management • Deductive reasoning • Problem solving • Exploration

10. Summary of Research on Psychosocial Outcomes • Some reports of difficulties participating in collaborative learning (Power & Hyde, 2002) • School age mainstreamed students (53/143 mild-mod) • 1/3 regarded as well-integrated with NH peers • 30% “go along with activities without significant role in planning or execution” • Impact on establishing friendships (Davis, et al, 1986) • School age mild to mod-severe (n = 40) • Half expressed concerns about making friends or social acceptance • Lower on expressing emotions/resolving conflict (Maxon et al, 1991)

11. Effects of Moderate HL on FQoL n = 223 * * * Hind & Davis, 2000 (ages 3 -11 yrs; X = 8 years)

12. Conclusions on Speech, Language, Psychosocial • Consistent findings that children with mild to severe HL are at risk for delays in phonology and morphology • Varied patterns of results for vocabulary • Limited understanding of factors contributing to individual differences in outcomes • Need for comprehensive study of outcomes in new generation of children to include psychosocial and family measures

13. Literacy Outcomes In Children with Mild to Severe Hearing Loss

14. Literacy • Literacy • Literacy refers to a socio-cultural activity of meaning construction using printed text. • Meaning construction occurs both during comprehension and writing . • Literacy provides the means to access and effect information sources via print. • Reading, spelling, punctuation, cursive, keyboarding are skills that serve literacy.

15. Literacy and Hearing Loss • Research on individual differences in reading development have demonstrated that spoken language skills are highly predictive of subsequent reading (literacy) skills. • Children with severe to profound hearing loss have very poor levels of reading and children with CI experience demonstrate much better performance. • We do not know how much hearing is necessary for adequate language and literacy development. • We can hypothesize that literacy skills may be limited in children with mild to moderate hearing to the extent that it impacts on language.

16. Model of Hearing and Reading Average Pure Tone Threshold Language (mediator) Reading Hearing Aid (moderator) Development

17. Reading • Gough: Reading is making sense of print through recognizing words. • Reading consists of getting words off the page and then understanding those words. • Simple view of reading: Reading involves decoding times comprehension. • Decoding: Going from orthographic form to lexical activation. • Comprehension: understanding the literal and implicit information in the passage (same as in listening).

18. World Knowledge Language Knowledge Text Sentence form & meaning Vocabulary Phonology Language Comprehension Reading Comprehension Decoding Skills Phonological Processing Naming Speed Phonics Orthographic knowledge Concepts about print Word Recognition

19. Component Literacy Skills • Foundations • Language • Phonological Processing • Phonological Awareness • Phonological Memory • Lexical Naming Speed • Key Literacy Skills • Reading • Spelling • Writing • Global Literacy Indicator • Academic Achievement

20. Research on Literacy Outcomes of Children with Mild to Severe Hearing Loss • Focus on literature that distinguishes between HH children with mild to severe losses and those who have severe to profound losses. • Most research aggregates HH and Deaf • ISI Web of Science Search • Reading or Literacy • Hard of Hearing or Hearing Impaired • Children • Not Deaf, Severe or Profound, Cochlear Implant • Reading or Literacy Outcome (not teaching) 181 39 9

21. Summary of Research on Literacy Outcomes • Phonological Processing • Gilbertson & Kamhi (1995) and Bristoe et al. (2001) showed poorer phonological awareness, phonological memory in children with HI than controls. Gibbs (2004) did not find differences. • Word Reading • Bess, Dodd-Murphy and Parker (1998) and Most et al. (2006) found poorer word reading in mild HI children. • Bristoe et al. (2001) and Gibbs (2004) did not find poorer word reading in HI children.

22. Summary of Research on Literacy Outcomes • Reading Comprehension • Davis, et al. (1986) and Blair et al. (1985) found HI children to be below hearing norms • Bristoe et al. (2001) found normal reading levels. • Academic Achievement • Kodman (1963), Blair et al. (1985), Davis et al. (1986), and Bess et al. (1998) reported poorer general academic achievement via test and/or parent report.

23. Conclusions on Literacy Literacy outcomes in mild to moderate HI children have been neglected There are good theoretical reasons to predict poorer performance in these children. The evidence is weighted in support of poorer performance particularly in PP and reading comprehension, but some of the best and more recent studies show less support. All of these studies do not take into account the child’s hearing aid fitting and use.

24. Outcomes Research in Children with Hearing Loss Bruce Tomblin University of Iowa

25. Taxonomy of Outcomes Research • Health Services Focused Outcomes Studies • Address questions concerning the services provided for a health condition. • Person/Disease Focused Outcomes Studies • Address questions concerning the individual or disease group including groups with risk exposures.

26. Health Services Outcomes Research in Medicine • “a term originally used to describe a particular line of health services research that focused on identifying variations in medical procedures and associated health outcomes.” Youngs MT, Wingerson L. The 1996 medical outcomes & guidelines sourcebook. New York (NY): Faulkner & Gray, Inc.; 1995. • Outcomes research in this context reserves the term for studies of efficacy, effectiveness, and negative consequences of clinical procedures.

27. Health Services Outcomes Designs • Randomized Clinical Trials (RCT) • Focus is on evaluation of new treatments. • Random assignment of cases to treatment and control arms. • Controls can be alternate treatments, placebo, “treatment as usual” or no treatment. • Efficacy and Effectiveness • Efficacy: treatment effect under ideal conditions • Effectiveness: treatment effect in real world conditions. • Outcome measures • Provider Oriented Outcomes: Laboratory markers of disease, complications and side effects. • Patient Oriented Outcomes: Functional Satisfaction, Quality of Life, Return to Work.

28. Health Services Outcomes Research (Patient Outcomes Research Teams or POTS) • Focused on evaluation of existing practices (preventive, diagnostic, and treatment). • Observational rather than experimental. • Systematic analysis of clinical records to document procedures, provider characteristics, patient characteristics. • Gather functional, perceived health, and quality of life outcomes data and associate these with practices as measures of effectiveness.

29. Person Oriented Outcomes Research • Outcomes research can be found in a wide variety of disciplines concerned with human development (psychology, education, sociology, child psychiatry, communication sciences and disorders, epidemiology) • The research questions in these studies are more concerned with issues having to do with what happens to cases (natural history) rather than on service provision: • Developmental features of the condition itself (course, recovery, stability). • Co-morbidities associated with developmental disorders. • Consequences of developmental or sensory disorders (education, occupation, quality of life).

30. Features of Person Centered Outcomes Research • Use of longitudinal designs. • Often a comparison group is incorporated. • Unaffected group - control • Population sample –cohort • Designs are all descriptive, correlational or quasi-experimental, thus there is no true independent variable. • Methods • Time A (diagnosis), Time B (Outcome) • Multiple observations • Growth curve analysis (HLM, latent growth analysis, growth mixture models) • Path and Structural Equation modeling • Hazard Survival analysis (time to event such as death)

31. Testing Causation in Longitudinal Designs • Person centered studies have come to focus on processes that contribute to outcomes. • Intervening variables between • Mediators – variables or conditions that are necessary aspects of the causal chain from initial condition to outcome • Moderators – variables that are not necessary causal elements but that interact with mediators. • Protective factors • Vulnerability factors • Questions: Risk-resilience in development • What factors contribute to “star” performance despite initial hearing status? • Methods of path analysis (regression) and structural equation models (latent variables) employed.

32. Path-Like Modeling Initial Health State Mediator Outcome Moderators Protective Vulnerability Development

33. Summary • The concept of what is an outcome study varies by discipline. • Outcome studies can be focused on clinical services and/or child characteristics. • Those concerned with clinical services may be wise to consider incorporating notions from person centered research such as moderators into their designs.

34. Putting together a collaboration Team Building

35. It Started with an NIH FOA Purpose. This Funding Opportunity Announcement (FOA) solicits longitudinal research applications from institutions/organizations to investigate outcomes in young children with mild to severe hearing loss. Applications must be multidisciplinary and have a primary focus on determining the impact of intervention, child, and family factors on communicative, educational and social development outcomes in young children with mild to severe hearing loss.

36. Challenge • Build an orchestra from scratch. • Where the orchestra writes the music as well as plays it. • Starting with a duet ?

37. Principles and Constraints for Team • Principles • Each team member contributes a particular skills to the team. • Some member’s roles involve more leadership than others, but every individual has an important contribution that needs to be respected. • Team members need to be able to work well with others. • Constraints • Multiple sites will be needed and therefore some redundancy in functions and roles will occur across sites. • One site will provide the general research infra-structure.

38. Skills Needed • Research Methods and Design • Health Outcomes Research Expertise • Longitudinal Research Expertise • Population/epidemiologic sampling • Measurement of pediatric hearing • Measurement of pediatric hearing management • Understanding of educational treatments and challenges of children and families with mild to moderate HI. • Measurement of outcomes • Hearing, speech, language, literacy, psycho-social, family

39. Skills Needed • Operational Infrastructure * • Project Manager *** • Budgets • Personnel • Day to day operations • Communications • IRB • Computer and Data Manager • Data Analysis • Recruitment and Retention • Front Line Research Assistants

40. Research Skills • Longitudinal Health Outcomes Research Expertise • BruceTomblin (Longitudinal Design, Outcomes of CD) • Jane Pendergast (Longitudinal Analysis, Epidemiology) • Jake Oleson (Biostatistics, Hearing Loss) • Measurement of Pediatric Hearing Loss and Management (Audiology) • Pat Stelmachowicz • Pat Roush • Ruth Bentler • Lenore Holte

41. Methods Skills • Outcomes Measurement • Mary Pat Moeller • Bruce Tomblin • Melody Harrison • Sandie Bass-Ringdhal • John Knutson (Psychology) • Educational Treatment and Family • Mary Pat Moeller • Melody Harrison • John Knutson

42. Operational Skills Infrastructure Front Line Data Collection • Audiology • Beth Walker (Iowa) • Meredith Spratford (Boys Town) • Shana Jacobs (UNC) • Speech, Language, Behavior • Thomas Page (UNC) • Lauren Berry (Boys Town) • Emilie Sweet (Boys Town) • Connie Ferguson • Marcia St. Clair • Project Manager • Marlea O’Brien • Data Base and Computer • Rick Arenas • Data Analysis • Jake Oleson

43. Challenges and Solutions • Compromising Personal Views • Allowing all to voice opinions and then arriving at a group decision. • Keeping goals of the research as the determining factor • Maintain honest open interchanges – no hidden agendas. • Communication • Video Conferences – monthly • Face-to-Face – yearly • Web based collaboration site (Share Point)

44. Challenges and Solutions • Restraining competition among subgroups and sites and minimizing personal egos • Overall strategy: Channel competition and egos toward the project. • Create a single identity that transcends sites • Attempt to build team interactions that cross-sites. • Make the goals of the project paramount. “Its not about you.” • Project leaders need to work to counter cliques and “back stabbing.”

45. Research Design & Methods

46. Aims • To describe the characteristics of: • children and families • intervention services • factors associated with service variations • To characterize: • developmental, behavioral and familial outcomes • compared to normally-hearing age mates with similar backgrounds • To explore: • how variations in child and family factors and in intervention characteristics relate to functional outcomes

47. Speaks English and has at least one primary caregiver that speaks English in the home Age at entry: 6 months to 6 years, followed annually Target Population • Mild to Severe Hearing Loss • PTA of 25-75 dB HL (500, 1k, 2k, 4 kHz) • Children who have one of the following types of HL: • High Frequency • Sensorineural • Permanent Conductive • Mixed • Auditory Neuropathy

48. Participants & General Design • 400 children with mild to severe hearing loss enrolled at age 6 mo to 6 years. • 150 children with normal hearing sampled from same communities. • Children followed for 3 years+. • Retrospective data prior to enrollment obtained by history. Accelerated Longitudinal Design

49. Recruitment • Sampling Frame • All children in Iowa, Nebraska, Eastern Kansas/Missouri and North Carolina with mild to severe permanent bilateral hearing loss • Sampling Method • Recruit children who have been identified via • Refer from Newborn Hearing Screening • Children identified in EHDI via follow up clinics • Children identified via audiology or medical service providers. • Children identified through school screening • Contact Method • Return of post card in flyer or telephone contact • Flyers will come to Iowa for processing • Telephone contact is made by the appropriate the regional research group

50. Phases of Enrollment