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Purpose: We sought to determine whether speech perception parent-reported executive functioning. Open-set word
and language skills measured early after cochlear implantation recognition was correlated with early speech perception
in children who are deaf, and early postimplant growth in and language skills and long-term speech perception and
speech perception and language skills, predict long-term language outcomes. Hierarchical regressions showed that
speech perception, language, and neurocognitive outcomes. early speech perception and language skills at 6 months
Method: Thirty-six long-term users of cochlear implants, postimplantation and growth in these skills from 6 to 18 months
implanted at an average age of 3.4 years, completed both accounted for substantial variance in long-term outcomes
measures of speech perception, language, and executive for language and verbal working memory that was not
functioning an average of 14.4 years postimplantation. explained by conventional demographic and hearing
Speech perception and language skills measured in the factors.
1st and 2nd years postimplantation and open-set word Conclusion: Speech perception and language skills
recognition measured in the 3rd and 4th years postimplantation measured very early postimplantation, and early postimplant
were obtained from a research database in order to assess growth in speech perception and language, may be clinically
predictive relations with long-term outcomes. relevant markers of long-term language and neurocognitive
Results: Speech perception and language skills at 6 and outcomes in users of cochlear implants.
18 months postimplantation were correlated with long-term Supplemental materials: https://doi.org/10.23641/
outcomes for language, verbal working memory, and asha.5216200
C
ochlear implants (CIs) provide children who have Miyamoto, 1998). By elementary school, more than half of
prelingual deafness with access to sound that is pediatric CI users can be expected to score in the average
sufficient in many cases to support the acquisition range on assessments of spoken language skills (Geers,
of spoken language (Geers, Brenner, & Davidson, 2003; Brenner, & Tobey, 2011; Geers, Nicholas, & Sedey, 2003),
Geers, Nicholas, & Sedey, 2003; Meyer, Svirsky, Kirk, & and this percentage increases with more experience by high
school age (Geers & Sedey, 2011). However, the effective-
ness of CIs in supporting speech and language development
a
is highly variable. Although variability in outcomes has
Speech Research Laboratory, Department of Psychological and
been identified as a major target for research by the National
Brain Sciences, Indiana University, Bloomington
b
Riley Child and Adolescent Psychiatry Clinic, Department of
Institutes of Health, individual outcomes remain extremely
Psychiatry, Indiana University School of Medicine, Indianapolis difficult to predict (National Institutes of Health, 1995;
c
DeVault Otologic Research Laboratory, Department of Niparko et al., 2010; N. R. Peterson, Pisoni, & Miyamoto,
Otolaryngology—Head and Neck Surgery, Indiana University School 2010; Pisoni et al., 2008; Svirsky, Teoh, & Neuburger, 2004).
of Medicine, Indianapolis Individual outcomes after several years of implant experience
d
Department of Otolaryngology—Head and Neck Surgery, The Ohio range from minimal benefit and awareness of environmental
State University, Columbus sounds to robust speech perception and language (SP-L)
Correspondence to Cynthia R. Hunter: cynthunt@indiana.edu skills that are on par with peers with normal hearing (NH;
Editor: Frederick Gallun Barnard et al., 2015; Cooper, 2006; Gantz, Woodworth,
Associate Editor: Amy Lederberg Fryauf-Bertschy, Kelsay, & Tyler, 1997; Geers, Brenner, &
Received April 13, 2016
Revision received August 24, 2016
Accepted February 21, 2017 Disclosure: The authors have declared that no competing interests existed at the time
https://doi.org/10.1044/2017_JSLHR-H-16-0152 of publication.
Journal of Speech, Language, and Hearing Research • Vol. 60 • 2321–2336 • August 2017 • Copyright © 2017 American Speech-Language-Hearing Association 2321
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Davidson, 2003). The identification of children who are at during their first years of exposure to sound. Gains in
risk for a poor outcome is a high research priority, because speech and language skills following cochlear implantation
early identification may inform clinical decisions about are measurable on a variety of assessments very early
habilitation and intervention, potentially increasing the postimplantation, following CI use of less than 1 year
benefits children receive from their CIs. (Nikolopoulos, Archbold, & Gregory, 2005; Niparko et al.,
To date, the relations of speech and language mea- 2010; Svirsky et al., 2004; Svirsky, Robbins, Kirk, Pisoni,
sures taken in the first few years of implant use to long-term & Miyamoto, 2000). To date, practically no studies have
outcomes after a decade or more of CI use have been exam- investigated whether speech and language skills after less
ined by only a small handful of studies, some of which than 1 year of CI use predict long-term outcomes. The
have used data from a larger study of long-term outcomes major aim of the current study was to address this gap in
at the Indiana University School of Medicine (Castellanos our knowledge by examining the relations of pediatric CI
et al., 2014; Castellanos, Pisoni, Kronenberger, & Beer, 2016; users’ SP-L skills, measured as soon as possible following
Hay-McCutcheon, Kirk, Henning, Gao, & Qi, 2008). Hay- cochlear implantation, to long-term outcomes in speech
McCutcheon et al. found that receptive language scores perception, language, and neurocognitive domains.
obtained when children were in preschool and kindergarten Both for children who use CIs and for children with
were significantly correlated with language scores in the NH, language development is closely linked to executive
teenage years. The observed relations between early recep- functioning (EF), an umbrella term for the neurocognitive
tive language and long-term language outcomes are similar skills that are involved in the self-regulation of thoughts
to those found by Geers and colleagues in a separate, large- and behaviors (Astington & Jenkins, 1999; de Rosnay &
scale study of long-term CI users (Geers & Sedey, 2011; Hughes, 2006; Figueras, Edwards, & Langdon, 2008; C. C.
Tobey, Geers, Sundarrajan, & Lane, 2011). They observed Peterson, 2004). Pediatric CI users tend to score below
that language scores measured in elementary school after their peers with NH on several domains of EF, and these
several years of CI use were highly predictive of language group differences may be evident as early as preschool
scores in high school. Low receptive and productive lan- age (Beer et al., 2014; Kronenberger, Pisoni, Henning, &
guage scores of CI users in preschool have also been asso- Colson, 2013). In addition, speech and language abilities
ciated with persistent language delays from elementary are more strongly associated with EF processes related
through the high school years (Geers, Nicholas, Tobey, & to verbal working memory and fluency-speed for CI users
Davidson, 2016). These findings suggest that the relation who received prelingual implantation than for their peers
between early and long-term language outcomes is present with NH (Cleary, Pisoni, & Kirk, 2000; Kronenberger,
as early as the preschool period. Colson, Henning, & Pisoni, 2014; Pisoni, Kronenberger,
A more recent study by Castellanos et al. (2014) pro- Roman, & Geers, 2011). Thus, examination of outcomes
vides support for this conclusion. That study examined in domains such as neurocognitive functioning in addition
whether speech intelligibility and receptive vocabulary sam- to language is important for providing converging infor-
pled in preschool after participants had used their implant mation about individual differences in benefit from cochlear
for nearly 2 years on average would predict long-term out- implantation.
comes for speech perception, language, and verbal work- One recent study has provided evidence that long-
ing memory. The authors found that CI users whose speech term neurocognitive outcomes can be predicted from
was more intelligible in preschool scored higher on assess- speech and language skills after an average of 1 year of
ments of speech perception, language, and verbal working- CI experience. Castellanos et al. (2016) examined the rela-
memory capacity taken approximately a decade later. tion between expressive language skills as measured by the
Children who scored higher on vocabulary in preschool MacArthur–Bates Communicative Development Invento-
also had significantly better long-term outcomes for SP-L. ries (Fenson et al., 2006) after an average of 1 year of
The relations between early language and long-term out- CI use and long-term neurocognitive outcomes an average
comes were not accounted for by conventional demographic of 11 years later. The MacArthur–Bates Communicative
and hearing-history variables, although age at onset of Development Inventories are a parent-completed inven-
deafness and age at implantation were related to several tory of a child’s earliest words and sentences. Castellanos
long-term outcomes. These findings indicate that CI users et al. (2016) controlled for nonverbal intelligence and
who are at risk for suboptimal long-term outcomes for demographic factors, and found strong relations between
speech, language, and verbal working memory may be early expressive language scores on the instrument and
identified during the preschool period. long-term neurocognitive outcomes. The neurocognitive
Current clinical guidelines recommend cochlear im- outcomes that were related to early expressive language
plantation in children who are deaf by 12 months of age included verbal working memory, fluency and speed of
(Boons et al., 2012; Dettman, Pinder, Briggs, Dowell, & information processing, and parent ratings of everyday EF
Leigh, 2007). Children who receive implants according to and learning. These findings suggest that an inventory of
these guidelines will have had years of experience with a child’s earliest expressive language taken after an average
their implants by preschool. It follows that identification of 1 year of CI experience may be clinically useful for
of risk for a poor outcome in the preschool period will not identifying pediatric CI users who may be at high risk of
be early enough to target interventions for many children poor long-term neurocognitive outcomes.
2322 Journal of Speech, Language, and Hearing Research • Vol. 60 • 2321–2336 • August 2017
Note. Em dashes indicate data not obtained. CI = cochlear implant; PTA = pure-tone average.
a
Unaided PTA (in dB HL) in the better ear for 500, 1000, and 2000 Hz. bExpressed as t scores (M = 50, SD =
10) from the Wechsler Abbreviated Scale of Intelligence Matrix Reasoning Subtest for Nonverbal Intelligence.
On average, our sample of CI users had nonverbal IQ scores within the normal range, although individual
scores ranged between more than 1 SD above and below the normed mean. cCoded on a scale from 1 (under
$5,000) to 10 ($95,000 and over).
2324 Journal of Speech, Language, and Hearing Research • Vol. 60 • 2321–2336 • August 2017
2326 Journal of Speech, Language, and Hearing Research • Vol. 60 • 2321–2336 • August 2017
Note. HINT-C = Hearing in Noise Test for Children; PPVT-4 = Peabody Picture Vocabulary Test–
Fourth Edition; CELF–Core = Clinical Evaluation of Language Fundamentals–Fourth Edition, Core
Language; WISC-III = Wechsler Intelligence Scale for Children–Third Edition; WISC-IV-I = Wechsler
Intelligence Scale for Children–Fourth Edition Integrated; WJ-III = Woodcock–Johnson Tests of
Cognitive Abilities, Third Edition; TOVA = Test of Variables of Attention; LEAF = Learning, Executive,
and Attention Functioning scale.
a
Raw scores. bStandard scores (M = 100, SD = 15). cScaled scores (M = 10, SD = 3).
to age norms. Mean scores on the CELF-4 and PPVT-4 than 1 SD below to more than 2 SDs above the normed
measures of language were approximately 1 SD below the mean. Mean scores on Fluency-Speed were within the
norm-referenced mean score of 100, and ranged from clinical normal range, but individual scores ranged more than 2 SDs
deficits of more than 3 SDs below the norm-referenced above and below the normed mean. Mean scores on the
mean score to above average relative to norms. Performance Test of Variables of Attention subtests for Inhibition-
on the HINT-C measure of speech perception in quiet, which Concentration were approximately 1 SD below the normed
is not scaled to age norms but uses a vocabulary suitable mean, and ranged from more than 3 SDs below to 1 SD
for children, ranged from 4% to 100% correct. Scores on the above the normed mean. At long-term follow-up, the par-
WISC-III digit-span tasks in the current sample of long-term ticipants from Table 1 who continued to use total commu-
CI users were lower on average than the age-normed mean nication had z-standardized composite scores of speech
score of 10, and the mean score for Digit Span Forward was perception that were more than 2 SDs below the mean for
more than 1 SD below the normed mean. Scores on the the current sample (M = −2.05, range = −2.72 to −1.09),
digit-span tasks ranged from 1 to 14. Mean scores on the language scores less than 1 SD below the mean for the sam-
assessments of Visual-Spatial Working Memory were within ple (M = −.57, range = −1.91 to 0.45), verbal working-
the normal range, but individual scores ranged from more memory and fluency-speed scores less than 0.5 SD below the
2328 Journal of Speech, Language, and Hearing Research • Vol. 60 • 2321–2336 • August 2017
Table 4. Partial correlations between early speech and language predictors and long-term performance outcomes, controlling for age at early test.
Cognitive subscales
Comprehension and Conceptual Learning −.54** −.28 −.50*
Factual Memory −.45* −.29 −.37
Attention −.51** −.54* −.53*
Processing Speed −.47* −.20 −.61**
Visual-Spatial Organization −.28 −.28 −.42
Sustained Sequential Processing −.46* −.43 −.58*
Working Memory −.44* −.27 −.53*
Novel Problem Solving −.43* −.15 −.44
Academic subscales
Mathematics Skills −.28 .06 −.33
Basic Reading Skills −.53** −.46 −.56*
Written Expression Skills −.44* −.37 −.61**
Note. Higher scores on the Learning, Executive, and Attention Functioning scale (LEAF) indicate greater
problems with executive functioning or related learning (see text for details).
a
Includes scores on the Reynell Developmental Language Scales II, the Pediatric Speech Intelligibility
Test with sentence and word materials, and the Mr. Potato Head task (see text for details). bYear 1 data
including only the matched set of participants who had data for Year 2.
*p < .05. **p < .01.
The amount of variance accounted for increased by 14% subsample (n = 18) and excluding children implanted after
when growth from Year 1 to Year 2 was added in Block 3, age 4 years (n = 7). The pattern of significant results was
with the full model accounting for 46% of the total vari- the same as in the original analysis.
ance in long-term language outcome. For long-term verbal
working memory, adding early SP-L scores for Year 1 in
the second block resulted in a significant 21% increase in Discussion
the variance accounted for by the model. Residual hearing The current study departs from earlier work on CI
also became a significant predictor when SP-L at Year 1 outcomes in three major respects. First, we focused on early
was added to the model. The amount of variance in verbal postimplant performance measures of children’s speech
working memory accounted for increased by 30% when and language skills to predict outcomes rather than con-
SP-L growth from Year 1 to Year 2 was added in Block 3. ventional demographic and hearing-history factors, and we
Nonverbal IQ and household income at long-term follow-up obtained these predictors earlier postimplant than did any
also became significant predictors when language growth prior study of long-term outcomes. Second, we examined
was added to the model in Block 3. The full model accounted long-term neurocognitive outcomes in addition to language
for 71% of the total variance in long-term verbal working- outcomes. Last, we compared SP-L skills measured very early
memory outcome (see Table 6). postimplant as well as growth in the following year as pre-
In summary, both long-term language and verbal dictors of long-term language and neurocognitive outcomes.
working memory were predicted by SP-L skills measured The first major finding was that SP-L skills measured
an average of 6 months after implantation. Further, the as early as 6 months after cochlear implantation predicted
addition to the model of growth in SP-L skills from Year 1 long-term language and neurocognitive outcomes. Although
to Year 2 predicted an additional 13% to 28% of variance the correlations between SP-L skills and long-term out-
in long-term language and verbal working-memory out- comes were stronger at 18 months than 6 months post-
comes, respectively. These predictive relationships could implantation when compared in the same set of participants,
not be accounted for by conventional demographic or both the correlation and regression analyses indicated that
hearing-history factors. SP-L skills after 6 months of implant use predicted substan-
Last, given that there are different expectations for tial variance in long-term language and neurocognitive out-
early postimplant performance for children who receive comes after a decade and a half of CI experience. Prior work
an implant before and after the age of 4 years (Robbins, has indicated stability between long-term language and
2007), we examined whether our results were representa- neurocognitive outcomes and speech and language skills
tive of the participants in our sample who were implanted as early as 1 to 2 years after implantation (Castellanos et al.,
younger than age 4 years. To this end, the regression 2014, 2016). The current results extend these earlier findings
analyses for language and verbal working-memory out- by documenting that stability in SP-L skills is observable
comes were rerun including only participants from this age using conventional clinical assessments as early as 6 months
2330 Journal of Speech, Language, and Hearing Research • Vol. 60 • 2321–2336 • August 2017
Note. Analysis includes participants with data for both Year 1 and Year 2 (n = 25 for all composite scores). The standardized coefficients
(β) and t values are provided for each predictor. The overall R2 is provided for each block. Only demographic and hearing-history variables
significant at the p < .05 level are displayed. Composite outcome measures include language (Peabody Picture Vocabulary Test–Fourth
Edition and Clinical Evaluation of Language Fundamentals–Fourth Edition, Core Language) and verbal working memory (Wechsler Intelligence
Scale for Children–Third Edition Digit Span Forward and Digit Span Backward subtests and Wechsler Intelligence Scale for Children–Fourth
Edition Integrated Visual Digit Span subtest).
a
Best preimplant pure-tone average (PTA) hearing threshold. bWechsler Abbreviated Scale of Intelligence Matrix Reasoning subtest at long-term
follow up. cYear 1 composite measure of speech-language. dDifference between Year 2 and Year 1 composite speech-language measures.
*p < .05. **p < .01. ***p < .001.
postimplantation and that very early postimplant SP-L skills this study may have utility for the assessment of risk after
predict long-term neurocognitive outcomes as well as long- 6 months of implant use in children who receive their im-
term language outcomes. Thus, SP-L skills measured after plants younger than age 4 years.
only 6 months of access to sound from a CI appear to be re- The second major finding of the current study is that
liable markers of long-term language and neurocognitive growth in SP-L skills from 6 to 18 months postimplantation
outcomes, and may have important clinical utility in identi- predicted long-term language and verbal working-memory
fying children who may be at high risk for poor outcomes outcomes. It has been known that very early experiences
following cochlear implantation. with a CI promote language growth and development. For
Assessment of at-risk status very early postimplant is example, Svirsky et al. (2000) reported gains of approxi-
in line with recommendations that speech-language pathol- mately 6 months of language age for each 6-month period
ogists should closely monitor speech and language progress from 0 to 30 months following cochlear implantation. Al-
within the first year after implantation for “red flags” of a though growth in the speech and language skills of CI users
poor outcome (Bradham & Houston, 2014; Nikolopoulos who received implantation prelingually has been docu-
et al., 2005; Robbins, 2007; Osberger, Robbins, & Trautwein, mented over the long term (e.g., Dunn et al., 2014), no
2006). Robbins (2007) provides different guidelines for prior study has examined whether early growth in speech
assessing progress in the first year after implantation for and language skills is a reliable predictor of long-term lan-
children who receive an implant before and after the age of guage and neurocognitive outcomes. The significant vari-
4 years. We found that the pattern of significant results for re- ance accounted for in the regression analyses by early growth
gression analysis that included only the subset of children in in SP-L skills indicates that long-term language and verbal
our sample who were implanted before the age of 4 years was working-memory outcomes following cochlear implanta-
the same as in the main analysis, which suggests that the con- tion are not only a function of demographics, hearing his-
ventional clinical assessments of SP-L skills that we used in tory, and SP-L ability in the first year postimplantation,
2332 Journal of Speech, Language, and Hearing Research • Vol. 60 • 2321–2336 • August 2017
2334 Journal of Speech, Language, and Hearing Research • Vol. 60 • 2321–2336 • August 2017
2336 Journal of Speech, Language, and Hearing Research • Vol. 60 • 2321–2336 • August 2017