Sign Language and Spoken Language for Children With Hearing Loss: A Systematic Review - American Academy of Pediatrics

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Sign Language and Spoken
                                       Language for Children With Hearing
                                       Loss: A Systematic Review
                                       Elizabeth M. Fitzpatrick, PhD,a,b Candyce Hamel, MSc,c Adrienne Stevens, MSc,c,d Misty Pratt, MES,c David
                                       Moher, PhD,c,e Suzanne P. Doucet, MEd,f Deirdre Neuss, PhD,b,g Anita Bernstein, MSc,h Eunjung Na, MSca,b

CONTEXT: Permanent hearing loss affects 1 to 3 per 1000 children and interferes with typical                                                                 abstract
communication development. Early detection through newborn hearing screening and
hearing technology provide most children with the option of spoken language acquisition.
However, no consensus exists on optimal interventions for spoken language development.
OBJECTIVE: To conduct a systematic review of the effectiveness of early sign and oral language
intervention compared with oral language intervention only for children with permanent
hearing loss.
DATA SOURCES: An a priori protocol was developed. Electronic databases (eg, Medline, Embase,
CINAHL) from 1995 to June 2013 and gray literature sources were searched. Studies in
English and French were included.
STUDY SELECTION: Two reviewers screened potentially relevant articles.

DATA EXTRACTION: Outcomes of interest were measures of auditory, vocabulary, language, and
speech production skills. All data collection and risk of bias assessments were completed
and then verified by a second person. Grades of Recommendation, Assessment, Development,
and Evaluation (GRADE) was used to judge the strength of evidence.
RESULTS: Eleven cohort studies metinclusion criteria, of which 8 included only children with
severe to profound hearing loss with cochlear implants. Language development was the
most frequently reported outcome. Other reported outcomes included speech and speech
perception.
LIMITATIONS: Several measures and metrics were reported across studies, and descriptions of
interventions were sometimes unclear.
CONCLUSIONS: Very limited, and hence insufficient, high-quality evidence exists to determine
whethersign language in combination with oral language is more effective than oral
language therapy alone. More research is needed to supplement the evidence base.

aFaculty of Health Sciences and eSchool of Epidemiology, Faculty of Medicine, University of Ottawa, Ottawa, Ontario, Canada; bChildren’s Hospital of Eastern Ontario Research Institute,

Ottawa, Ontario, Canada; cCentre for Practice-Changing Research, Ottawa Hospital Research Institute, Ottawa, Ontario, Canada; dTranslational Research in Biomedicine Graduate Program,
University of Split School of Medicine, Split, Croatia; fConsultant, Moncton, New Brunswick; gAudiology Clinic, Children’s Hospital of Eastern Ontario, Ottawa, Ontario, Canada; and hVoice for
Hearing-Impaired Children, Toronto, Ontario, Canada

Dr Fitzpatrick conceptualized the project, finalized the protocol, was involved in all stages of the analysis and interpretation, wrote the first draft of this manuscript,
and is the study guarantor; Ms Hamel and Ms Pratt were involved in screening articles and extracted data; Ms Hamel conducted the quality assessment; Ms Hamel,
Ms Pratt, Ms Stevens, and Dr Moher provided input into the final manuscript; Ms Stevens contributed to the development of the methods, oversaw the screening,

  To cite: Fitzpatrick EM, Hamel C, Stevens A, et al. Sign Language and Spoken Language for Children With Hearing Loss: A Systematic Review. Pediatrics.
  2016;137(1):e20151974

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PEDIATRICS Volume 137, number 1, January 2016:e20151974                                                                                                             REVIEW ARTICLE
Early detection of permanent               which might disrupt or delay spoken              better spoken language outcomes
childhood hearing loss through             language acquisition. However,                   when exposed to early intervention
population-based newborn screening         another body of research suggests                that uses signs to support language
has become standard care in much           that visual languages such as                    compared with oral language
of the world. Expectations are that        American Sign Language (ASL) are                 intervention without sign language?
early intervention through hearing         processed in the brain in the same
technology will improve spoken             manner as spoken languages and
language outcomes for children.1,2         are complementary to auditory                    METHODS
Childhood hearing loss is a relatively     stimulation, and therefore provide a
                                                                                            The protocol and report for this
frequent disorder, affecting               strong foundation for learning oral
                                                                                            review were prepared according
1 to 3 per 1000 live births,3–5            language.18,19 Therefore, adding
                                                                                            to Preferred Reporting Items for
that disrupts typical language             sign language may develop bilingual
                                                                                            Systematic Reviews and Meta-
acquisition, placing children at risk      skills and facilitate transition
                                                                                            Analyses for Protocols,20 and
for delays in language, literacy, and      to spoken language acquisition.
                                                                                            the protocol was published21
social development.6–8 There is            Anecdotal evidence supports various
                                                                                            and registered at PROSPERO
strong consensus that specialized          intervention options, but there is
                                                                                            (Registration #CRD42013005426),22
intervention must be combined              little scientifically based consensus.
                                                                                            an international register of
with early identification to develop       This information is essential to (1)
                                                                                            systematic review protocols. The
communication skills in these              guide families in making decisions
                                                                                            review was undertaken using an
children.1,9                               about care for their children in
                                                                                            integrated knowledge translation
                                           infancy, (2) inform clinicians so that
Historically, considerable debate                                                           approach involving knowledge-
                                           they can tailor treatment plans to
about optimal outcomes for children                                                         user clinicians from health and
                                           achieve the desired outcomes, and
with hearing loss have resulted                                                             education in the early stages and as
                                           (3) inform policy makers so that they
in the evolution of a plethora of                                                           required, to ensure relevance of the
                                           can make optimal investments in
intervention methods that constitute                                                        project for parents, clinicians, and
                                           early intervention services.
two broad but distinct philosophies.                                                        decision-makers.
The oral approach aims to facilitate
                                           This review is timely given the
spoken language and inclusion                                                               Literature Search
                                           worldwide proliferation of screening
with normal-hearing peers, and
                                           programs in the past 10 years. In                Ovid Medline In-Process & Other
the manual approach focuses on
                                           2008, the US Preventive Services                 Non-Indexed Citations and Ovid
visual communication systems
                                           Task Force recommendations                       Medline (1946 to June week 2,
(sign language) and a Deaf culture
                                           on newborn hearing screening                     2013), Embase (1974 to June 25,
identity.10,11 Although there is wide
                                           highlighted the need for further                 2013), PsycINFO (1806 to June week
recognition that various options
                                           research to demonstrate the                      3 2013), and Cochrane CENTRAL
should be available to families,
                                           effectiveness of the entire screening-           (through May 2013 issue) databases
2 events in the past two decades
                                           to-intervention process.2 New                    were searched using the Ovid
(newborn hearing screening and
                                           possibilities due to screening                   interface. CINAHL was searched on
cochlear implant technology) have
                                           and technology have reignited                    June 26, 2013, using the EbscoHost
made it possible for even children
                                           the discussion on best practices                 interface, and SpeechBITE was
with profound deafness to develop
                                           for children with hearing loss.                  searched on June 26, 2013. The
spoken language.7,12–14 Epidemiologic
                                           Accordingly, the primary purpose                 search strategy (Appendix 1)
data confirm that >90% of children
                                           of this research was to examine the              was developed in Medline by an
with hearing impairment are born to
                                           evidence for the effects of various              experienced information specialist,
parents with normal hearing.15
                                           intervention options for early-                  peer-reviewed using the Peer
Although there is substantial              identified children with hearing loss            Review of Electronic Strategies
evidence that children with hearing        when the desired outcome is spoken               (PRESS) standard,23 and adapted
loss can develop oral language             communication. Our interest was                  for the other databases. The search
skills,7,14,16 there is no consensus       in whether adding sign language                  was restricted to the pediatric age
about optimal interventions.17             facilitates spoken language, because             group,24 and the date was limited to
There is a common expectation that         of the increased focus on this                   material published in 1995 or later.
children receiving oral language           outcome since the advent of cochlear             A broad methodological filter was
intervention should develop better         implantation. Specifically, the review           applied, incorporating published
language skills than those who             addressed the following question:                filters for controlled trials and
are also exposed to sign language,         Do children with hearing loss have               other study designs.25,26 In addition,

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2                                                                                                                     FITZPATRICK et al
gray literature websites (A.G. Bell              Relevancy of papers was assessed                 screened all potentially relevant
Association for the Deaf, National               based on the components of the                   full-text articles. Disagreements
Acoustics Laboratory, National                   intervention described and not on                were resolved by consultation and
Health Services UK Newborn Hearing               the program label.                               consensus with a third researcher
Screening Program, CADTH Gray                                                                     and knowledge-user clinicians as
                                                 The comparator of interest was oral
Matters Checklist), conference                                                                    needed.
                                                 language intervention without sign
proceedings (Newborn Hearing
                                                 language.
Screening Conference Abstracts 2010                                                               Data Extraction
and 2012), two journals (Ear and                 Outcomes                                         A study-specific data extraction form,
Hearing and Journal of Deaf Studies                                                               finalized with input from knowledge-
                                                 Primary outcomes included all
and Deaf Education), and six books27–                                                             user clinicians, was developed in
33 were searched.                                measures of spoken language
                                                 including auditory, receptive, and               Distiller SR to extract predetermined
                                                 expressive language skills (eg,                  data variables. Items extracted
Eligibility Criteria
                                                 vocabulary), speech production, and              included (1) study characteristics
Study Designs                                                                                     (citation, year, setting, country,
                                                 intelligibility. These outcomes were
The following study designs were                 selected as clinically relevant based            language, publication status, and
included: randomized controlled                  on a large body of literature.7,33,34            source of funding or other potential
trials; controlled clinical trials and           Secondary outcomes included                      conflict of interest), (2) study design,
other quasi-experimental designs                 electrophysiologic outcomes (eg,                 (3) population characteristics (eg,
that include comparator groups; and              cortical responses). In addition, any            sample size, gender, ethnicity,
prospective and retrospective cohort             adverse outcomes (eg, parent stress)             etiology, age, severity of hearing
studies.                                         were noted.                                      loss, hearing technology, and time
                                                                                                  with hearing technology), (4)
Population                                       Time Frame                                       details (type) of intervention, (5)
Studies were eligible for inclusion if           We included studies from 1995                    details of comparison groups, and
they included children (1) with early-           onward to capture studies after                  (7) outcome data. One researcher
onset (before age 3 years) hearing               wide implementation of cochlear                  extracted data and a second
loss of any severity using hearing               implantation and newborn hearing                 researcher independently verified
aids and/or cochlear implants; (2)               screening. Earlier studies were                  data. Discrepant and unclear data
receiving early intervention (age                excluded, as previous generations of             were resolved through consensus
≤3 years); and (3)
bias, study design, confounders,          Evaluation (GRADE) working group38               in tabular and narrative form. A
blinding, data collection methods,        to rate the quality of the overall               meta-analysis was not possible due
withdrawals and dropouts, integrity       body of evidence for each outcome.               to heterogeneity in designs, methods,
of intervention, and study analysis.      Primary outcomes were assessed                   and outcome metrics. A narrative
One researcher conducted the quality      across the domains of risk of bias,              synthesis and table summary of
assessment after 2 studies were           consistency, directness, precision,              data were therefore completed. We
first assessed by 2 researchers, and      and publication bias. GRADE results              present continuous outcomes as
the second verified the remaining         in a rating of the quality of the body           mean differences (MDs) with 95%
assessments. A third researcher           of evidence as high (very confident              confidence intervals, where possible.
with content expertise rechecked all      that true effect is close to the effect          Planned subgroup analyses to
ratings and discussed any differences     estimate), moderate (moderately                  examine variables related to hearing
to reach consensus.                       confident), low (limited confidence),            loss severity, age of identification,
                                          or very low (little confidence).                 and hearing technologies could not
Grading Strength of Evidence                                                               be conducted owing to the lack of
                                          Data Synthesis                                   studies.
We applied methodology developed
by the Grades of Recommendation,          We summarized the characteristics of
Assessment, Development, and              study populations and interventions              RESULTS
                                                                                           We identified 432 records through
                                                                                           database searching and 113 records
                                                                                           through other sources, of which 470
                                                                                           were retained for screening after
                                                                                           duplicates were removed. Figure 1
                                                                                           outlines the flow of records through
                                                                                           the screening process. We assessed
                                                                                           352 full-text documents. As shown,
                                                                                           the primary reasons for exclusion
                                                                                           were (1) not a study design (58%)
                                                                                           and (2) not an intervention of
                                                                                           interest (32%). Eleven unique studies
                                                                                           were included.

                                                                                           Study Characteristics
                                                                                           Table 1 summarizes the study
                                                                                           characteristics. All were prospective
                                                                                           cohort studies published between
                                                                                           1999 and 2013, 4 since 2004. The
                                                                                           majority (n = 8) were conducted in
                                                                                           the United States,32,39–45 and one each
                                                                                           was conducted in Spain,46 the UK,47
                                                                                           and Denmark.48 Three studies44,46,47
                                                                                           were conducted specifically to
                                                                                           examine whether signs added to
                                                                                           spoken language intervention (sign
                                                                                           + oral) improved outcomes. The
                                                                                           remaining studies evaluated hearing
                                                                                           technology or provided data about
                                                                                           language intervention as 1 of several
                                                                                           predictor variables in the analysis.

                                                                                           Participants and Interventions
                                                                                           Study size for reported outcomes
FIGURE 1                                                                                   ranged from 13 to 90 participants.
Study selection.                                                                           It is not possible to rule out overlap

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4                                                                                                                     FITZPATRICK et al
TABLE 1 Characteristics of Included Studies
Study, Year,        Design   Main Study Objective and      Participants, Age, Hearing Loss, Other          Intervention              Comparator        Study Quality
Country                              Setting                          Characteristics
Miyamoto et al,     Cohort   To examine speech            n = 14 for review, NR by group; CI; only     TC: combined use         OC                    Weak
1999, US39                   perception, speech           children implanted
TABLE 1 Continued
Study, Year,         Design     Main Study Objective and         Participants, Age, Hearing Loss, Other              Intervention               Comparator             Study Quality
Country                                   Setting                             Characteristics
Nicholas and         Cohort     To examine whether              n = 76, 38 SC, 38 OC (OC from                   SC: emphasizes             OC: emphasizes            Moderate
Geers, 2003,                    deaf children who have          previously published study); age:               the combination of         the auditory signal
US44                            the addition of signs           18–54 mo; age tested: 18–54 mo; group           spoken language            with or without
                                show an early linguistic        scores reported at 18–30 mo (n = 16             with corresponding         visual cues from
                                advantage and greater           per intervention group) and 36–54 mo            manual signs               speaker’s lips
                                communicative function          (n = 22 per intervention group); HL:
                                maturity than those with        better-ear PTA threshold ≥80 dBHL,
                                spoken language input           103.21 vs 104.41 dBHL; HA: 27 vs 30; CI: 7
                                only; to examine whether        vs 5; tactile aid: 0 vs 3; no device: 4 vs 0;
                                deaf children who               age onset: birth (n = 34 vs 33) and 1–18
                                receive speech and sign         mo (n = 4 vs 5); age diagnosis: 10.42 vs
                                lag behind children in OC       12.03 mo; maternal education: 14.14 vs
                                in their use of speech in       14.71 y; duration CI: 7.5 (n = 7) vs. 3.8
                                preschool                       mo (n = 5)
Connor and           Cohort     To examine the effects          n = 91, 43 TC, 48 OC; 49.45% male; CI;          TC: use of sign            OC: use of spoken         Weak
Zwolan, 2004,                   of multiple variables           age: 11.16 ± 2.83 vs. 10.81 ± 2.53 y (for       language in                language only
US45                            (SDT, age, SES,                 reading evaluation); age pre–post               combination with
                                communication method,           vocabulary tests: NR; HL: >80 dBHL; age         spoken language;
                                pre- and postimplant            onset: 0.12 vs 0.19 y; age CI: 7.40 vs 6.23     sign systems based
                                vocabulary) on reading          y; duration CI: 3.82 ± 1.56 vs. 4.63 ± 2.51     on English, including
                                comprehensionb                  y; HL-SDT: 55.22 vs 51.72                       Signed English and
                                (pre- and postimplant                                                           Signing Exact English
                                vocabulary scores were
                                reported by group)
Jiménez et al,       Cohort     To compare speech/              n = 18, NR by group; 61.11% male; CI;           Bilingual: spoken +        Monolingual:              Moderate
2009, Spain46                   language development            age: mean 6.25 y, range 4 y 3 mo to             sign language              spoken language
                                after CI in children            8 y; HL: profound; groups similar for
                                educated using spoken           gender, age (avg 6.25 y), age diagnosis
                                language versus spoken          (avg 10 mo), age CI (avg 3.2 y), duration
                                and sign language               CI (avg 3.1 y) (P > .05 for all)
Nittrouer, 2010,     Cohort     To explain the                  n = 118, 44 sign support, 74 spoken             Spoken + sign              Spoken language           Moderate
US32                            contributions of various        language; 55% male; early-identified             support; 28 of 44
                                independent measures            (6        parents used ASL and
                                to developmental                mo) n = 15 vs 26; age: 12–48 mo; tested         16 a manually coded
                                outcomes for children           at 6-mo intervals; n varied per interval:       English system;
                                with hearing loss, 1 of         16 (12 mo) to 90 (48 mo); HL: moderate          parents reported
                                which was sign support          to profound; 38 HA, 80 CI by end of             using signs
of participants in some studies; for             therapy (Table 2). Most studies (n =             authors did not report scores
example, 339,42,43 from 1999 to 2002             10) reported multiple outcomes of                separately by group but concluded
included a subset of children from 1             interest, whereas 1 study41 reported             that not using total communication
US cochlear implant program. Eight               speech perception only (Table 3). As             was 1 factor associated with greater
studies included only children with              shown in Table 2, different metrics              odds of age-equivalent receptive
severe to profound deafness who had              (eg, standard score, raw score, age              vocabulary.
received cochlear implants. The 3                equivalency, language quotient) were
remaining studies included a mix of              reported in different studies. Studies           Three studies also reported results
children with respect to technology              also reported variations in test                 for expressive vocabulary. The
use.32,44,47 As shown in Table 1,                administration. In 1 study, Spanish              Danish study48 examined expressive
authors used various descriptions                test versions (which authors reported            vocabulary using a Danish norm–
and terms to describe both the                   as validated) were administered.46               referenced test but found no effect of
intervention (eg, sign, bilingual) and           Danish test adaptations48 were                   intervention mode. The US preschool
comparator (oral, spoken language)               used with American norms in                      study32 found that expressive
groups.                                          another study except for the                     vocabulary results favored oral
                                                 expressive vocabulary test, which                intervention only for late-identified
Quality Assessment                               was developed and standardized in                children (>12 months) and showed
Study quality and scores are shown               Danish. As noted in Table 1, tests               no significant effects of intervention
in Table 1. Studies were rated as                were administered in the child’s                 for early-identified children (
8
                                                                                                  TABLE 2 Summary of Evidence and GRADE Ratings
                                                                                                  Outcome Measure, Test, and Length of Follow-Up             Sign + Oral vs Oral Onlyb         Studies (Participants)    Effect Estimatec            Statistical Information           Quality of Evidencee
                                                                                                  or Test Agea                                                                                                                                       Provided by Authord
                                                                                                  Language
                                                                                                    PPVT39,42,46,48                                                                                                                                                                    Very low
                                                                                                      Unknown39,42f,g                                   In 2 studies, mean LQ 0.30–0.72 vs     2 (28)                    MD (no CI): −0.02 to 0.10   NS for both studies
                                                                                                                                                        0.32–0.62
                                                                                                       Mean 5.4 y46h                                    Mean standard score 66.9 vs 73.6       1 (18)                    MD (no CI): −6.7            NS (P > .05)
                                                                                                       Mean 3.2 y48h (range 6 to >36 mo)                Scores NR by group; reported as        1 (68)                    OR estimated to be          P = .012 (favors oral)
                                                                                                                                                        odds of having age-equivalent                                    infinity by authors
                                                                                                                                                        vocabulary
                                                                                                    Viborgmaterialet48 (Danish vocabulary)                                                                                                                                             Very low
                                                                                                       Mean 3.2 yh (range 6 to >36 mo)                  Scores NR by group; reported as        1 (49)                    OR not reported             NS
                                                                                                                                                        odds of having age-equivalent
                                                                                                                                                        vocabulary
                                                                                                    Picture Vocabulary Test/EOWPVT45i                                                                                                                                                  Very low
                                                                                                       Unknown45f,g                                     Pre- and postimplant scores            1 (91)                    MD (no CI): 7.48 to 13.46   NS preimplant (P > .05);
                                                                                                                                                        66.54–74.60 vs 53.08–67.12                                                                   postimplant NR
                                                                                                    EOWPVT32j                                                                                                                                                                          Very low
                                                                                                      3.5 y (early id), 2–3 y (late id)32k; test age:   Raw score: 32.89 ± 8.76 vs 32.18       1 (early: 52; late: 38)   MD-early (95% confidence     Early id: NS; late id: P = .002
                                                                                                  48 mo                                                 ± 10.81 (early id); 21.50 ± 13.40 vs                             interval): 0.71 (−4.72 to   (favors oral)
                                                                                                                                                        32.12 ± 7.86 (late id)                                           6.14); MD-late: −10.62
                                                                                                                                                                                                                         (−18.78 to −2.45)
                                                                                                    Reynell-R39,40,42,43,48                                                                                                                                                            Very low
                                                                                                      Unknown39,42,43g                                  Mean LQ 0.61–0.89 vs 0.64–0.91         3 (69)                    MD (no CI): −0.02 to        NS for all studies
                                                                                                                                                                                                                         −0.03
                                                                                                       Unknown40g                                       Mean 8- vs 9-mo increase in            1 (23)                    MD (no CI): −1-mo           NS
                                                                                                                                                        language age (no CIs)                                            language age
                                                                                                       Mean 3.2 y, unknown by group48                   Scores NR by group; odds of age-       1 (71)                    OR estimated to be          P = .013 (favors oral)
                                                                                                                                                        equivalent language                                              infinity by authors
                                                                                                    Reynell-E39,40,42,43                                                                                                                                                               Very low
                                                                                                      Unknown39,42,43g                                  Mean LQ 0.51–0.68 vs 0.69–0.93         3 (69)                    MD (no CI): −0.02 to        1 NS41; 1 NR42; 1 significant38
                                                                                                                                                                                                                         −0.26
                                                                                                       Unknown40g                                       Mean 7- vs 10-mo increase in           1 (23)                    MD (no CI): −3-mo           NS
                                                                                                                                                        language age                                                     language age
                                                                                                    ITPA (Spanish) Auditory Reception46                                                                                                                                                Very low
                                                                                                       5.4 y                                            Mean standard score 34.6 vs 56.0       1 (18)                    MD (no CI) −21.4            Significant

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                                                                                                    ITPA (Spanish) Auditory Association46                                                                                                                                              Very low
                                                                                                       5.4 y                                            Mean standard score 37.8 vs, 71.6      1 (18)                    MD (no CI) −33.8            Significant
                                                                                                    ITPA (Spanish) Auditory sequential memory46                                                                                                                                        Very low
                                                                                                       5.4 y                                            Mean standard score 44.8 vs 59.0       1 (18)                    MD (no CI)−14.2             NS
                                                                                                    ITPA (Spanish) Verbal Expression46                                                                                                                                                 Very low
                                                                                                       5.4 y                                            Mean standard score 52.5 vs 39.2       1 (18)                    MD (no CI 13.3              Significant
                                                                                                    ITPA (Spanish) Grammatical closure46                                                                                                                                               Very low
                                                                                                       5.4 y                                            Mean standard score 39.2 vs 71.3       1 (18)                    MD (no CI)−32.1             Significant
                                                                                                    ICAP (Spanish) Social communication46                                                                                                                                              Very low
                                                                                                       5.4 y                                            Mean standard score 38.75 vs 53.0      1 (18)                    MD (no CI): −14.25          NS
                                                                                                    Auditory Comprehension-PLS-432j                                                                                                                                                    Very low

FITZPATRICK et al
TABLE 2 Continued
                                                                                                                 Outcome Measure, Test, and Length of Follow-Up            Sign + Oral vs Oral Onlyb          Studies (Participants)    Effect Estimatec              Statistical Information           Quality of Evidencee
                                                                                                                 or Test Agea                                                                                                                                         Provided by Authord
                                                                                                                      3.5 y (early id), 2–3 y (late id)k; test age:   Mean raw score: 45.39 ± 8.82 vs         1 (early: 52; late: 38)   MD early id (95%              Early id: NS,j P = .011 (favors
                                                                                                                 48 mo                                                42.59 ± 10.02 (early id); 35.92 ±                                 confidence interval):          oral)
                                                                                                                                                                      12.80 vs 43.73 ± 8.59 (late id)                                   −2.80 (−5.48 to 5.08);
                                                                                                                                                                                                                                        MD late id: −7.81 (−15.76
                                                                                                                                                                                                                                        to 0.14)
                                                                                                                   Language Comprehension–SIB- R32j                                                                                                                                                     Very low
                                                                                                                     3.5 y (early id), 2–3 y (late id)k; test age:    Mean raw score: 19.78 ± 2.78 vs         1 (early: 52; late: 38)   MD early id (95%              Early id: NS; late id: NS
                                                                                                                 48 mo                                                19.15 ± 3.46 (early id); 19.83 ± 4.71                             confidence interval): 0.63
                                                                                                                                                                      vs 18.12 ± 3.43 (late id)                                         (−1.10 to 2.36); MD late
                                                                                                                                                                                                                                        id: 1.71 (−1.26 to 4.68)
                                                                                                                   Language Expression - SIB-R32j                                                                                                                                                       Very low

PEDIATRICS Volume 137, number 1, January 2016
                                                                                                                     3.5 y (early id), 2–3 y (late id)k; test age:    Mean raw score: 28.56 ± 1.89 vs         1(early: 52; late: 38)    MD early id (95%              Early id: NS; late id: P = .012
                                                                                                                 48 mo                                                27.03 ± 3.59 (early id); 23.42 ± 6.80                             confidence interval): 1.53     (favors oral)
                                                                                                                                                                      vs 26.88 ± 3.19 (late id)                                         (0.04 to 3.01); MD late id:
                                                                                                                                                                                                                                        −3.46 (−7.49 to 0.57)
                                                                                                                   Number of spoken words-LDS32j                                                                                                                                                        Very low
                                                                                                                      3.5 y - early-id; 2-3 y - late-id groupk Test   Mean: 185.79 ± 103.19 vs 157.08 ±       1 (early: 51 late: 16)    MD early id (95%              Early id: similar number of
                                                                                                                 age: 30 mo                                           104.56 (early id); 38.75 ± 29.64 vs                               confidence interval):          words; late id: sign had fewer
                                                                                                                                                                      122.25 ± 69.34 (late id)                                          28.71 (−34.98 to 92.40);      words (author report)
                                                                                                                                                                                                                                        MD late id: −83.5
                                                                                                                                                                                                                                        (−132.31 to −34.68)
                                                                                                                   BLADES47                                                                                                                                                                             Very low
                                                                                                                     Unknown                                          Delay 6–24 vs 12–24 mo                  1 (13)                    See next column               No difference between groups
                                                                                                                                                                                                                                                                      (author report)
                                                                                                                    Number of different words (sign or spoken)44                                                                                                                                        Very low
                                                                                                                      Rangel 7.6–19.6 mo (sign + oral), 6–18 mo       Mean 13.88 ± 17.55 vs 2.65 ± 5.87       1 (76)                    MD (95% confidence             NS
                                                                                                                 (oral); test age 18–30 mo                            words                                                             interval): 11.23 (5.35 to
                                                                                                                                                                                                                                        17.11)
                                                                                                                      Rangel 25.6–43.6 (sign + oral), 24–42 mo        Mean 45.50 ± 29.08 vs 38.90 ± 3.96      1 (76)                    MD (95% confidence             NS
                                                                                                                 (oral); test age: 36–54 mo                           words                                                             interval): 6.60 (−2.73 to
                                                                                                                                                                                                                                        15.93)
                                                                                                                   Words per utterance (sign or spoken)44                                                                                                                                               Very low
                                                                                                                     Test age 18–30 mo                                Mean 0.99 ± 0.44 vs 0.31 ± 0.5          1 (76)                    MD (95% confidence             Significant
                                                                                                                                                                      words                                                             interval): 0.68 (0.47 to

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                                                                                                                                                                                                                                        0.89)
                                                                                                                      Test age 36–54 mo                               Mean 1.25 ± 0.18 vs 1.33 ± 0.36         1 (76)                    MD (95% confidence             NS
                                                                                                                                                                      words                                                             interval): −0.08 (−0.21
                                                                                                                                                                                                                                        to 0.05)
                                                                                                                   Total number of spoken words44                                                                                                                                                       Very low
                                                                                                                     Test age 18–30 mo                                Mean 3.56 ± 5.19 vs 5.47 ± 14.1         1 (76)                    MD (95% confidence          NS
                                                                                                                                                                      words                                                             interval): −1.91 (−6.69
                                                                                                                                                                                                                                        to 2.87)
                                                                                                                      Test age 36–54 mo                               Mean 23.09 ± 33.03 vs 117.88 ±          1 (76)                    MD (95% confidence          NS
                                                                                                                                                                      207.75 words                                                      interval): −94.79 (−161.67
                                                                                                                                                                                                                                        to −27.91)

9
10
                                                                                                  TABLE 2 Continued
                                                                                                  Outcome Measure, Test, and Length of Follow-Up           Sign + Oral vs Oral Onlyb       Studies (Participants)   Effect Estimatec             Statistical Information             Quality of Evidencee
                                                                                                  or Test Agea                                                                                                                                   Provided by Authord
                                                                                                     Number of different words spoken44                                                                                                                                              Very low
                                                                                                       Test age 18–30 mo                              Mean 2.62 ± 3.59 vs 1.94 ± 4.29      1 (76)                   MD (95% confidence            NS
                                                                                                                                                      words                                                         interval): 0.68 (−1.10 to
                                                                                                                                                                                                                    2.46)
                                                                                                       Test age 36–54 mo                              Mean 12.91 ± 17.38 vs 38.67 ± 44     1 (76)                   MD (95% confidence            Significant
                                                                                                                                                      words                                                         interval): −25.76 (−40.80
                                                                                                                                                                                                                    to −10.72)
                                                                                                    PIHF44                                                                                                                                                                           Very low
                                                                                                       Test age 18–30 mo                              Mean 0.23 ± 0.2 vs 0.04 ± 0.12       1 (76)                   MD (95% confidence            Significant
                                                                                                                                                      words                                                         interval): 0.19 (0.12 to
                                                                                                                                                                                                                    0.21)
                                                                                                       Test age 36–54 mo                              Mean 0.46 ± 0.17 vs mean 0.34 ±      1 (76)                   MD (95% confidence            Significant
                                                                                                                                                      0.21 words                                                    interval): 0.12 (0.03 to
                                                                                                                                                                                                                    0.21)
                                                                                                    ICA44                                                                                                                                                                            Very low
                                                                                                       Test age 18–30 mo                              Mean 41.56 ± 35.71 vs 36.76 ±        1 (76)                   MD (95% confidence            NS
                                                                                                                                                      35.68 words                                                   interval): 4.80 (−11.27 to
                                                                                                                                                                                                                    20.87)
                                                                                                       Test age 36–54 mo                              Mean 121 ± 75.25 vs 131.67 ± 70.79   1 (76)                   MD (95% confidence            NS
                                                                                                                                                      words                                                         interval): −10.67 (−43.52
                                                                                                                                                                                                                    to 22.18)
                                                                                                  Speech
                                                                                                    BIT39                                                                                                                                                                            Very low
                                                                                                       Unknown                                        10.57% vs 30.62% correct words       1 (14)                   MD (no CI) −20.05            Significant
                                                                                                                                                      identified by listener
                                                                                                    CSIM32                                                                                                                                                                           Very low
                                                                                                      3.5 y (early id), 2–3 y (late id)k; test age:   Mean: 58% ± 19% vs 57% ± 17%         1 (early: 49 late: 36)   MD early id (95%             Early id: NS; late id: P < .001
                                                                                                  48 mo                                               (early id); 46% ± 25% vs 59 ± 16%                             confidence interval): 1.0     (favors oral)
                                                                                                                                                      (late id) words recognized by                                 (−9.78 to 11.78); MD late
                                                                                                                                                      listener                                                      id: −13.0 (−29.05 to 3.05)
                                                                                                    Induced Phonological Register (Spanish)46                                                                                                                                        Very low
                                                                                                       5.4 y                                          Mean standard score 30 vs 60         1 (18)                   MD (no CI): −30              Significant (P < .05) (favors
                                                                                                                                                                                                                                                 oral)
                                                                                                  Speech Perception

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                                                                                                    Mr. Potato Head-Words39,41–43                                                                                                                                                    Very low
                                                                                                      Unknown                                         Mean % correct 24%–56% vs            4 (105)                  MD (no CI): −9 to −39%       1 NS40; 1 NR42; 2 significant38,41
                                                                                                                                                      33%–95% (no CI)
                                                                                                    Mr. Potato Head – Sentences39                                                                                                                                                    Very low
                                                                                                      Unknown                                         Percent correct 39.76% vs 69.88%     1 (14)                   MD (no CI): −30.12%          Significant
                                                                                                    ESP-Low Verbal41                                                                                                                                                                 Very low
                                                                                                      Unknown                                         Percent correct 82% vs 90%           1 (36)                   MD (no CI): −8%              Significant
                                                                                                    GAEL-P Words recognized39,42                                                                                                                                                     Very low
                                                                                                      Unknown                                         Percent correct 46%–87.38% vs        2 (28)                   MD (no CI): −7% to −9.3%     Significant38,41
                                                                                                                                                      53%–96.68%
                                                                                                    GASP-W41                                                                                                                                                                         Very low

FITZPATRICK et al
TABLE 2 Continued
                                                                                                                 Outcome Measure, Test, and Length of Follow-Up                    Sign + Oral vs Oral Onlyb             Studies (Participants)                      Effect Estimatec                  Statistical Information                Quality of Evidencee
                                                                                                                 or Test Agea                                                                                                                                                                          Provided by Authord
                                                                                                                      Unknown                                               Percent correct 50% vs 63%                   1 (36)                                      MD (no CI): −13%                  Significant
                                                                                                                    PBK-Words41                                                                                                                                                                                                               Very low
                                                                                                                      Unknown                                               Percent correct 20% vs 24%                   1 (36)                                      MD (no CI): −4%                   Significant
                                                                                                                    PBK-Phonemes41                                                                                                                                                                                                            Very low
                                                                                                                      Unknown                                               Percent correct 29% vs 50%                   1 (36)                                      MD (no CI): −21%                  Significant
                                                                                                                 BIT, Beginner’s Intelligibility Test; BLADES, Bristol Language Developmental Scales; CI, confidence interval; CSIM, Children’s Speech Intelligibility Measure; EOWPVT, Expressive One Word Picture Vocabulary Test; ESP, Early Speech Perception; GAEL,
                                                                                                                 Grammatical Analysis of Elicited Language (used as closed-set speech perception measure); GASP, Glendonald Auditory Screening Procedure; ICA, intentionally communicative act; ICAP, Inventory for Client and Agency Planning; ITPA, Illinois Test of
                                                                                                                 Psycho-linguistic Abilities; LDS, Language Development Survey; id, identified; LQ, language quotient; MD, mean difference; NR, not reported; NS, not statistically significant; OR, odds ratio; PBK, Phonetically Balanced Kindergarten Test; PIHF, Proportion
                                                                                                                 of Informative or Heuristice Functions; PLS, Preschool Language Scale; PPVT, Peabody Picture Vocabulary Test; SIB-R, Scales of Independent Behavior-Revised
                                                                                                                 a Follow-up measured from time of intervention.
                                                                                                                 b Outcomes data; higher score = better performance.

PEDIATRICS Volume 137, number 1, January 2016
                                                                                                                 c Minus sign (−) indicates higher score in oral group.
                                                                                                                 d No meta-analysis due to insufficient data and different metrics reported; statistics reported are those from authors.
                                                                                                                 e Based on GRADE ratings.
                                                                                                                 f Length of follow-up (from diagnosis of hearing loss) with language intervention of interest is unknown. Studies39–43,45 report duration of cochlear implant use and age at assessment (details in Table 1).
                                                                                                                 g Authors stated that language measures were adapted and administered in child’s preferred communication modality. signed and spoken responses accepted,45 or instructions given nonverbally and certain vocabulary considered less likely to

                                                                                                                 be known by children using sign was removed.32
                                                                                                                 h Calculated based on information provided in article.
                                                                                                                 i Picture Vocabulary Test (PVT) of the Woodcock Johnson Test of Cognitive Ability or EOWPVT administered preimplant; PVT postimplant.
                                                                                                                 j Study reports 25 different language scores at 6-mo test intervals from 12 to 48 mo; table includes 6 clinically relevant test measures that reflect overall findings at last test age (5 standardized language measures and 1 speech intelligibility

                                                                                                                 measure). Remaining results (not shown in table) relate to specific aspects of language from language sample analysis (e.g., number of responses, number of pronouns): for early-identified children, 18 results showed no statistically significant
                                                                                                                 differences and 1 favored oral intervention; for late-identified children, 14 results favored oral intervention. Statistical information provided is based on authors’ report of simple effects analysis (signs, prosthesis, test age) for early- and late-
                                                                                                                 identified hearing loss.
                                                                                                                 k Calculated based on early group identified at 12 mo of age up to 24 mo of age. Results for last test age reported.
                                                                                                                 l Calculated from age of diagnosis and test age range; applies for all measures for study.44

                                                                                                                                                                                       test.

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                                                                                                                                                                                                                                                                                                                    comprehension only.

                                                                                                                                                                                                                                                                           statistical significance.
                                                                                                                                                                                                                                                                                                                    language intervention to be

                                                                                                                 subtest only. One additional
                                                                                                                 Various subtest scores were

                                                                                                                 version of the Illinois Test of

                                                                                                                 study from the UK47 (n = 13)
                                                                                                                                                                                       for 2 standardized measures
                                                                                                                                                                                       preschool study32 consistently
                                                                                                                                                                                       Three additional studies32,46,47
                                                                                                                                                                                                                                                                           language intervention reached
                                                                                                                                                                                                                                                                                                                    of intervention compared with

                                                                                                                                                                                       measures, all yielding different
                                                                                                                                                                                                                                                                                                                    American norms of the Reynell
                                                                                                                                                                                                                                                                                                                    using a Danish adaptation with

                                                                                                                 also reported in the study of 18
                                                                                                                                                                                                                                                                                                                    2002. According to the authors,

                                                                                                                                                                                       comprehension measure but no
                                                                                                                                                                                                                                                                           The same 4 US cochlear implant

                                                                                                                                                                                       intervention for early-identified

                                                                                                                                                                                       for late-identified children, with
                                                                                                                                                                                                                                                                                                                    there were no significant effects

                                                                                                                 children46 that used the Spanish
                                                                                                                                                                                                                                                                                                                    quotients, and 1 study, language

                                                                                                                                                                                       reported no significant effects of

                                                                                                                                                                                       children at 48 months. However,

                                                                                                                 Psycholinguistic Abilities (ITPA).
                                                                                                                                                                                       results. As shown in Table 1, a US

                                                                                                                                                                                       results favoring oral intervention
                                                                                                                                                                                                                                                                                                                    received cochlear implants before

                                                                                                                                                                                                                                                                           for expressive language measured

                                                                                                                 The authors reported significantly
                                                                                                                                                                                       receptive and expressive language
                                                                                                                                                                                                                                                                                                                    receptive subtest reported spoken

                                                                                                                 language–only group. Mean scores
                                                                                                                 higher standard scores in auditory

                                                                                                                 reported language outcomes using
                                                                                                                                                                                       the study produced mixed findings
                                                                                                                                                                                                                                                                           Only 1 study39 that favored spoken
                                                                                                                                                                                                                                                                           did not report expressive language.

                                                                                                                 grammatical closure for the spoken
                                                                                                                 reception, auditory association, and
                                                                                                                                                                                                                                                                                                                    statistically significant, for language
                                                                                                                                                                                                                                                                                                                    age scores). Only the Danish study48

                                                                                                                                                                                       on another language comprehension

                                                                                                                 were significantly higher for the sign
                                                                                                                                                                                                                                                                           by the Reynell test. The Danish study
                                                                                                                                                                                                                                                                           studies also reported similar findings

                                                                                                                 + oral group on the verbal expression
                                                                                                                                                                                       significant difference in interventions
                                                                                                                                                                                                                                                                                                                    controls (3 studies reported language

                                                                                                                                                                                       (expressive language) and 1 language
                                                                                                                                                                                       provided results for a variety of other

11
developmental scales to measure                                  development. A study by Nicholas                               sign + oral group at 18 to 30 months;
expressive language in the areas of                              and Geers44 reported findings for 2                            number of different words spoken,
pragmatics, semantics, and syntax.                               age groups, 18 to 30 months and 36                             which favored the oral group at 36
The authors reported delays ranging                              to 54 months, for 38 children who                              to 54 months; and the Proportion of
from 6 to 24 months in both groups                               received sign + oral intervention                              Informative or Heuristic Functions,
and concluded that intervention did                              and compared results to a historical                           which favored sign + oral in both age
not influence outcomes.                                          cohort of 38 children in oral language                         groups. Of the 19 natural language
                                                                 intervention. As shown in Table 2,                             results reported in the Nittrouer32
Language Results: Natural Language
                                                                 of 12 different results, statistically                         study, 18 showed no statistically
Sample Analysis
                                                                 significant differences were reported                          significant difference for early-
Finally, 2 studies reported scores for                           only for words per utterance (sign                             identified children (18 of 51 had sign
various aspects of communication                                 or spoken words), which favored the                            + oral), whereas 1 measure (number

TABLE 3 Overview of Outcome Measures for All Included Studies
Measure/tool                                                                            Study, reference no.                                                Sign + Oral          Oral
                                                 39         40          41        42         43         47        44       45      46      32       48
PPVT1                                              •                                          •                                     •               •                              1
Viborgmaterialeta                                                                                                                                   •
PVT/EOWPVT                                                                                                                  •               •                                     1b
Reynell Receptive                                  •         •                     •          •                                                     •                             1
Reynell Expressive                                 •         •                     •          •                                                                                   1
ITPAc Auditory Reception                                                                                                            •                                             1
ITPA Auditory Association                                                                                                           •                                             1
ITPA Auditory Sequential Memory                                                                                                     •
ITPA Verbal Expression                                                                                                              •                             1
ITPA Grammatical Closure                                                                                                            •                                              1
ICAP                                                                                                                                •
PLS-4 Auditory Comprehension                                                                                                                •                                     1b
Language Comprehension SIB-R                                                                                                                •
Language Comprehension SIB-R                                                                                                                •                                     1b
BLADES Expressive                                                                                        •
Number of different wordsd                                                                                         •                        •
Words per utteranced                                                                                               •                                              1
Total number of spoken wordsd                                                                                      •
Number of different words spokend                                                                                  •                                                               1
PIHFd                                                                                                              •                                              2
Intentionally communicative actsd                                                                                  •
Language-11 communication actse                                                                                                             •                                     7f
Language–5 grammar/syntaxe                                                                                                                  •                                     5b
Language-3 vocalizationse                                                                                                                   •                                     2b
Beginner’s Intelligibility Test                    •                                                                                                                               1
CSIM                                                                                                                                        •                                     1b
Induced Phonological Register                                                                                                       •                                              1
Mr. Potato Head—Words                              •                     •         •          •                                                                                    2
Mr. Potato Head—Sentences                          •                                                                                                                               1
ESP-Low Verbal                                                           •                                                                                                         1
GAEL-P Wordsg                                      •                                          •                                                                                    2
GASP—Words                                                               •                                                                                                         1
PBK—Words                                                                •                                                                                                         1
PBK—Phonemes                                                             •                                                                                                         1
Total                                                                                                                                                             4               36
Numbers in boxes refer to number of studies reporting statistical significance for sign + oral or oral group. BLADES, Bristol Language Developmental Scales; CSIM, Children’s Speech
Intelligibility Measure; ESP, Early Speech Perception; EOWPVT, Expressive One Word Picture Vocabulary Test; GAEL-P, Grammatical Analysis of Expressive Language: Presentence Level; GASP,
Glendonald Auditory Screening Procedure; ICAP, Inventory for Client and Agency Planning; Lang, language; PBK, Phonetically Balanced Kindergarten Test; PLS-4, Preschool Language Scale;
PVT, Picture Vocabulary Test of the Woodcock Johnson Test of Cognitive Ability; SIB-R, Scales of Independent Behavior-Revised.
a Danish version of test.
b Nittrouer32 reported significant difference for late-identified sign group only; results for early-identified group were not significant.
c Spanish version of ITPA for all subtests.
d Results for 2 age groups (18–30 mo and 36–54 mo for each measure in this study43).
e Various aspects of communication/language reported from analyses of language samples (e.g., number of responses, total words, number of pronouns, jargon).
f Both early- and late-identified groups showed significant difference for 1 communication act (number of inquiries); 6 others showed significance for late-identified group only.
g GAEL-P administered as closed-set speech perception test.

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12                                                                                                                                                                    FITZPATRICK et al
of inquiries made by child) favored              significant results for sign + oral              and expressive language outcomes.
oral intervention. However, results              intervention and 36 for oral language            Three studies addressing speech
were markedly different for the late-            only (9 related to speech perception,            significantly favored oral language
identified children (12 of 37 had sign           17 for late-identified group in US               therapy only (except 1 early-
+ oral), in whom 14 of 19 results                preschool study32).                              identified group32). Closed-set speech
favored oral intervention.                                                                        perception studies mostly favored
                                                                                                  spoken language only, whereas
We retrieved only 1 study that
                                                 DISCUSSION                                       open-set speech perception studies
addressed other aspects of language
                                                                                                  statistically favored spoken language
functioning considered relevant to               There continue to be questions about
                                                                                                  but with small clinical significance.
this review. Using the Inventory for             the advantages or disadvantages
                                                                                                  The majority of studies were
Client and Agency Planning, Jiménez              of adding sign language to
                                                                                                  methodologically weak, with a few
et al46 reported no significant                  interventions focused on spoken
                                                                                                  scoring a moderate rating. However,
differences between groups in social             language development in children
                                                                                                  when rated according to GRADE
communication skills.                            with hearing loss. This review found
                                                                                                  criteria, the overall quality of the
                                                 that few studies have systematically
Speech Results                                                                                    evidence per outcome is very low.
                                                 addressed the issue. Eight of the
Three studies addressed aspects                  11 studies in this review included               According to GRADE, cohort studies
of speech production, 2 related to               only children with severe to                     are initially considered as low quality
speech intelligibility32,39 and 1 to             profound deafness who were using                 before applying the assessment
phonological production.46 In 2                  cochlear implants. This is likely                criteria, as they are at a greater
studies,39,46 scores were significantly          because many children received                   risk of bias, generally. The risk of
better for the oral intervention                 preimplant intervention with a                   bias for the body of evidence for
groups based on author reports,                  signing component in addition                    outcomes was deemed to have
whereas in the US preschool study,32             to spoken language, owing to the                 serious limitations. The overall
scores were significantly better                 profound nature of hearing loss,                 quality of evidence for each outcome
for oral intervention in the late-               particularly in the 1990s when                   we assessed was limited because of
identified but not early-identified              cochlear implantation was a new                  the small number of included studies,
children.                                        option. Evidence to address the issue            which resulted in low ratings for the
                                                 for children with less than profound             precision of results and an inability
Speech Perception                                degrees of hearing loss is severely              to assess for consistency. There was
Four cochlear implant studies39,41–43            lacking, as this review retrieved only           also considerable variation in the
(1999 to 2002) specifically                      3 studies32,44,47 that included children         measurements of outcomes, which
investigated speech perception and               with hearing loss using hearing aids.            made comparison across studies
included 3 closed-set measures                                                                    difficult. Furthermore, the full
                                                 Measures of language development,
(response selected from defined                                                                   process of care from time of diagnosis
                                                 including receptive vocabulary
word set) and 2 open-set measures                                                                 is unknown, as is whether crossover
                                                 and receptive and expressive
(no word set available). All scores                                                               interventions occurred. Therefore,
                                                 language skills, contributed the most
were significantly better for the oral                                                            additional studies in contemporary
                                                 information to this review. When
group, with the exception of 1 closed-                                                            cohorts of children are needed to
                                                 comparing sign and oral language
set test (Mr. Potato Head, words) in                                                              supplement the evidence base. Most
                                                 intervention with oral language
141 (n = 36) of the 4 studies, which                                                              studies were conducted before
                                                 intervention alone, individual
was not statistically significant.                                                                2002 with children with severe to
                                                 studies showed no differences
However, for open-set monosyllabic                                                                profound deafness and therefore may
                                                 between groups for receptive and
words, the differences were clinically                                                            not reflect current practice, in which
                                                 expressive vocabulary outcomes,
small despite statistical significance                                                            children with congenital deafness
                                                 with the exception of 1 study’s32
(eg, 20% vs 24%). No speech                                                                       are likely to be identified and receive
                                                 results that favored oral intervention
recognition measures were reported                                                                early intervention well before 1 year
                                                 only for late-identified children
for children with hearing aids.                                                                   of age. Accordingly, new information
                                                 (n = 12 in sign group). However,
                                                                                                  about the effects of intervention
Table 3 provides an overview of                  a variety of measures and metrics
                                                                                                  in children across the spectrum
measures extracted in the included               were used across studies that limit
                                                                                                  of hearing loss severity has the
studies and the interventions that               the generalizability of the evidence.
                                                                                                  potential to change the conclusions.
showed statistically significant                 Overall, mixed results of statistical
differences. Of 61 total test scores             significance and no differences were             Since completing our review, we
reported, only 4 showed statistically            observed in studies for receptive                are aware of 1 recent retrospective

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PEDIATRICS Volume 137, number 1, January 2016                                                                                           13
cohort study conducted in Australia         that include comparator groups                   outcomes and reporting metrics
with young children who used                since previous reviews.51,52 Despite             would be very useful in comparing
cochlear implants that compared a           strong conclusions based on expert               future study results.
sign and oral language intervention         opinion and intervention results                 There is insufficient information to
group (n = 10) with 2 oral                  without comparator groups, this                  guide parents about contemporary
intervention groups (auditory-oral, n       review indicates that very limited,              cohorts of children who have
= 14, and auditory-verbal, n = 18).50       and hence insufficient, evidence                 benefited from early detection and
No significant differences in receptive     exists to determine whether adding               early access to hearing technology.
vocabulary, auditory comprehension,         sign language to spoken language is              The field would gain from carefully
or expressive language were found           more effective than spoken language              controlled prospective studies with
across the 3 groups after controlling       intervention alone to foster oral                today’s children.
for confounders such as age of              language acquisition.
hearing loss management and family
involvement in the intervention                                                              APPENDIX 1: MEDLINE SEARCH
program. These results are aligned          CONCLUSIONS                                      STRATEGY
with the overall findings of our                                                             MEDLINE
review and would not change the             This review showed that there
                                                                                              1. exp Hearing Loss/
conclusions.                                are important gaps in knowledge
                                            concerning the effectiveness of sign              2. (hearing adj (loss or impair$ or
Limitations
                                            and oral language intervention,                      disorder*)).tw.
Despite a comprehensive search              compared with oral language                       3. deaf$.tw.
by an information specialist, it is         intervention only, for children with
possible that some studies were                                                               4. (prelingual$ or pre-lingual$).tw.
                                            hearing loss when spoken language
not included owing to the range             is the intended outcome. To date,                 5. (sensorineural$ or sensori-
of descriptions of intervention             there is no evidence that adding                     neural$).tw.
methods. During study selection,            sign language facilitates spoken                  6. congenital.mp.
program descriptions were                   language acquisition. However, this
sometimes difficult to discern. For                                                           7. or/1-6
                                            review also found no conclusive
example, it was difficult to know           evidence that adding sign language                8. auditory verbal.tw.
whether interventions included the          interferes with spoken language                   9. ((speech or auditory) adj2
same level of emphasis on spoken            development. Overall, the literature                 feedback).tw.
language when sign language was             related to intervention methods
added. Although we contacted                                                                 10. cued speech.tw.
                                            for children with hearing loss lacks
authors for clarity of interventions        properly designed cohort studies                 11. (listen* and (spoken or speak*)).
and outcomes, we did not contact            of today’s generation of children.                   tw.
them for additional sample                  It will be important to conduct                  12. oral approach$.tw.
characteristics or statistical data. A      more research to supplement this
further important limitation is that 8      evidence base and to update this                 13. aural.tw.
of the 11 studies, although meeting         review as those studies become                   14. Lipreading/
inclusion criteria, were not designed       available. Given the current context             15. (lipread$ or lip read$ or
to directly examine the central             of widespread neonatal screening,                    speechread$ or speech read$).
question of this review.                    data can be collected prospectively                  tw.
                                            from diagnosis, allowing for more
Implications for Practice                                                                    16. or/8-15
                                            accurate data regarding onset,
Although the question of intervention       severity, and changes in hearing loss,           17. Manual Communication/ or Sign
effectiveness and specifically the          as well as specific characteristics and              Language/
contribution of sign language               changes related to intervention, than            18. (sign$ language or sign$ english).
combined with oral language is              was possible for study participants                  tw.
longstanding, there is strikingly           in this review. Interventions should
limited new research to guide               be reported with specific detail, and            19. ASL.tw.
clinical or policy decision-making.         if definitions can be developed and              20. visual language.tw.
The question of the contribution of         adopted on an international level,               21. (baby sign or infant sign).tw.
sign language to the development of         comparisons of similar programs
spoken language remains elusive, and        would be more feasible. In addition,             22. or/17-21
there has been little growth in studies     agreement on a common set of core                23. Communication Methods, Total/

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14                                                                                                                     FITZPATRICK et al
24. total communication.tw.                             39. ((consecutive or clinical) adj2                     45. 42 and (child* or adolescent or
25. simultaneous communication.tw.                          case$).tw.                                              infan*).mp.

26. (multilingual or multi-lingual).                    40. ((control$ or intervention or                       46. 44 or 45
    tw.                                                     evaluation or comparative or
                                                            effectiveness or evaluation or                      47. limit 46 to yr=”1985 -Current”
27. (bicultural or bi-cultural).tw.                         feasibility) adj3 (trial or studies
28. (bilingual or bi-lingual).tw.                           or study or program or design)).
29. bi bi.tw.                                               tw.                                                 ACKNOWLEDGMENTS
30. or/23-29                                            41. or/31-40                                            We thank Margaret Sampson, MLIS,
31. exp clinical trial/                                 42. 7 and ((16 and 22) or 30) and 41                    PhD, AHIP (Children’s Hospital of
                                                        43. 42 and ((Infan* or newborn*                         Eastern Ontario), for developing the
32. clinical trial.pt. or randomized.                                                                           electronic search strategies and Janet
    ti,ab. or placebo.ti,ab. or                             or new-born* or perinat* or
                                                            neonat* or baby or baby* or                         Joyce, MLIS, for peer review of the
    randomly.ti,ab. or trial.ti,ab. or                                                                          MEDLINE search strategy. We also
    groups.ti,ab.                                           babies or toddler* or minors
                                                            or minors* or boy or boys or                        thank Pauline Quach for assistance
33. ((control$ or clinical or                               boyfriend or boyhood or girl*                       with the quality assessment.
    comparative$) adj2 (trial$ or                           or kid or kids or child or child*
    stud$)).mp.                                             or children* or schoolchild*
                                                                                                                  ABBREVIATIONS
34. exp Epidemiologic studies/                              or schoolchild).mp. or school
    or Case-control studies/ or                             child.ti,ab. or school child*.ti,ab.                  ASL: American Sign Language
    Retrospective studies/ or Cohort                        or (adolescen* or juvenil* or                         GRADE: Grades of
    studies/ or Longitudinal studies/                       youth* or teen* or under*age*                                  Recommendation,
    or Cross-sectional studies/                             or pubescen*).mp. or exp                                       Assessment,
                                                            pediatrics/ or (pediatric* or                                  Development, and
35. between group design$.mp.
                                                            paediatric* or peadiatric*).mp.                                Evaluation
36. control group$.mp.                                      or school.ti,ab. or school*.ti,ab. or                 ITPA: Illinois Test of
37. (cohort stud$ or longitudinal).                         (prematur* or preterm*).mp.)                                Psycholinguistic Abilities
    mp.                                                                                                           MD: mean difference
                                                        44. limit 43 to (“in data review”
                                                                                                                  PPVT: Peabody Picture
38. (case adj2 (series or control$)).                       or in process or “pubmed not
                                                                                                                         Vocabulary Test
    mp.                                                     medline”)

was involved in extracting data and quality assessment, conducted the GRADE ratings, assisted with data interpretation; Dr Moher assisted with the development
of the study protocol and consulted on the quality assessment and data interpretation as required; Ms Doucet (a knowledge-user clinician), Dr Neuss (a
knowledge-user clinician), and Ms Bernstein (a knowledge-user clinician) provided input into the inclusion criteria and data abstraction form, helped clarify
data interpretation when needed, and commented on the manuscript; Ms Na verified and provided input into the synthesis tables and made comments on the
manuscript; and all authors approved the final manuscript.
This review is registered at PROSPERO: #CRD42013005426, http://www.crd.york.ac.uk/PROSPERO/display_record.asp?ID=CRD42013005426#.VWeaU-vqc6I.
DOI: 10.1542/peds.2015-1974
Accepted for publication Oct 7, 2015
Address correspondence to Elizabeth M. Fitzpatrick, Audiology and Speech-Language Pathology Program, Faculty of Health Sciences, University of Ottawa, 451
Smyth Road, Ottawa, Ontario, Canada K1H 8L1. E-mail: elizabeth.fitzpatrick@uottawa.ca
PEDIATRICS (ISSN Numbers: Print, 0031-4005; Online, 1098-4275).
Copyright © 2016 by the American Academy of Pediatrics
FINANCIAL DISCLOSURE: The authors have indicated they have no financial relationships relevant to this article to disclose.
FUNDING: This systematic review was funded by a Canadian Institutes of Health Research (CIHR) Knowledge Synthesis Grant (FRN-124600). The funding agency
was not involved in developing the protocol and was not involved in any aspect of the review (data collection, analysis, or interpretation) or publications. Dr
Fitzpatrick’s work is also supported by a CIHR New Investigator and a Canadian Child Health Clinician Scientist Program award. Dr Moher holds a University of
Ottawa Research Chair. All researchers are independent from the funding agencies.
POTENTIAL CONFLICT OF INTEREST: The authors have indicated they have no potential conflicts of interest to disclose.

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PEDIATRICS Volume 137, number 1, January 2016                                                                                                                     15
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