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AJSLP

Review Article

Case History Risk Factors for Specific


Language Impairment: A Systematic
Review and Meta-Analysis
Johanna M. Rudolpha

Purpose: Research suggests that the best approach to Results: Eleven risk factors were found to be
early identification of children with specific language statistically significant predictors of SLI. Among
impairment (SLI) should include assessment of risk factors. these, maternal education level, 5-min Apgar score,
However, previous attempts to develop a list for this birth order, and biological sex met criteria for clinical
purpose have been unsuccessful. In this study, systematic significance.
review and meta-analytic procedures were used to determine Conclusions: At least 4 case history factors are as
whether any case history factors can be used to identify predictive as late talker status in the context of early
toddlers at risk of developing SLI. identification of toddlers at risk for SLI. The findings of this
Method: Epidemiological studies that examined the review highlight the importance of taking a child’s genetic
association between risk factors and SLI were identified. and environmental context into consideration when
Results across studies were aggregated to determine more deciding whether further evaluation and early intervention
precisely the strength of association between each risk factor services are warranted.
and the development of SLI. The clinical significance of these Supplemental Materials: https://doi.org/10.23641/
factors was established via comparison to late talker status. asha.5150122

S
pecific language impairment (SLI) affects ap- 2011; Hebbeler et al., 2007). As a result, recommended best
proximately 7.5% of kindergarten-age children practices for the population of children with SLI include
(Tomblin, Records, et al., 1997), which amounts early identification and early intervention to facilitate
to at least one and probably two children in every classroom the language learning process during the formative years
(RALLIcampaign, 2012). Individuals with SLI experience of communication development (Olswang, Rodriguez, &
significant difficulties learning language, which manifest as Timler, 1998).
deficits in language comprehension and/or language produc- There have been recent efforts in medical and clini-
tion. Because the ability to understand language and the cal communities to promote early identification of children
ability to communicate through language are so fundamental with language deficits through detection of early signs and
to human interaction and learning, such deficits place these symptoms of impairment (American Speech-Language-
individuals at great risk for poor social, emotional, aca- Hearing Association, n.d.; Hagan, Shaw, & Duncan,
demic, and vocational outcomes (Brinton, Fujiki, Spencer, 2008). However, many children who present with early
& Robinson, 1997; Catts, Fey, Tomblin, & Zhang, 2002; language delays spontaneously recover by the preschool
Conti-Ramsden & Botting, 2004; Hadley & Rice, 1991; or early school age years and, therefore, do not have SLI
Redmond, 2011; Young et al., 2002). It is widely believed (Ellis Weismer, 2007; Paul, 1996; Rescorla, 2002). Fur-
that the earlier that children with communication disorders thermore, many children with SLI may not exhibit early
receive services and supports, the more likely they are to language delays (Dale, Price, Bishop, & Plomin, 2003;
achieve successful language and learning skills (Guralnick, LaParo, Justice, Skibbe, & Pianta, 2004; Poll & Miller,
2013). These findings suggest that early language perfor-
a
mance alone is insufficient to predict which toddlers will
University of Texas at Dallas
experience significant and chronic difficulties with lan-
Correspondence to Johanna Rudolph: johanna.rudolph@utdallas.edu guage development.
Editor: Krista Wilkinson One potential path forward, suggested in a follow-up
Associate Editor: Laura DeThorne
to a systematic review for the U.S. Preventive Services
Received November 18, 2015
Revision received May 13, 2016
Accepted December 13, 2016 Disclosure: The author has declared that no competing interests existed at the time
https://doi.org/10.1044/2016_AJSLP-15-0181 of publication.

American Journal of Speech-Language Pathology • Vol. 26 • 991–1010 • August 2017 • Copyright © 2017 American Speech-Language-Hearing Association 991
Task Force, involves consideration of child, parent, or & Parsons, 2009), or some combination of language impair-
family characteristics that may place an individual at risk ments (Dale et al., 2003; Roulstone, Peters, Glogowska, &
for long-term impairments (i.e., risk factors; Berkman Enderby, 2003; Tomblin, Hardy, & Hein, 1991; Zambrana,
et al., 2015). The authors of the review suggested that the Pons, Eadie, & Ystrom, 2014). The deficits associated with
ability to stratify children according to risk might promote each label vary widely in severity and comorbidity. The pic-
efficient screening, thereby facilitating earlier identification. ture is further complicated by the fact that these studies
Indeed, some studies have found that significantly more have differed in sample size, sampling method, participant
children with SLI are correctly identified when risk factors ages, factors examined, and analyses used (Berkman et al.,
are considered alongside early language abilities than when 2015; Harrison & McLeod, 2010). Thus, attempts to develop
either risk factors or early language skills are considered a list of risk factors to guide professional practice have met
in isolation. For example, in a recent examination of the with a degree of heterogeneity that has precluded the pos-
American Academy of Pediatrics guidelines for early lan- sibility of meaningful aggregation. To avoid this dilemma
guage production milestones, Rudolph and Leonard (2016) in the current investigation, systematic review procedures
found that lack of word combinations at 24 months was were used to identify a cluster of studies matched on critical
a significant predictor of later language impairment; how- design features. In brief, this review focused on studies con-
ever, this criterion alone only identified about half of the taining epidemiological participant samples in which one
children with SLI. The authors then examined whether portion of the children presented with language impairment
more children with SLI would be correctly identified if risk and were characterized as exhibiting SLI whereas another
factors were considered in addition to word combining sta- portion of the children presented with no language impair-
tus. They found that adding the factor family history of ment and were characterized as typically developing (TD).
communication or reading disorders to the predictive model Furthermore, particular attention was paid to the types of
resulted in the identification of almost 90% of the children risk factors investigated to ensure that those included would
with SLI, significantly more than were identified by word be (a) representative of the range of genetic and environmen-
combining status alone. In a prospective, longitudinal, tal influences that might interact to promote or discourage
population-level study, Reilly et al. (2010) took the oppo- child language development (Rogers, Nulty, Betancourt, &
site approach by first identifying a set of predictive risk DeThorne, 2015) and (b) obtainable within the context of a
factors. When risk factors were considered in isolation, the routine pediatric screening. By focusing on studies that satis-
predictive model yielded only a moderate level of discrimi- fied these basic requirements, a more homogeneous group
nation between children developing typically and those of relatively high quality studies was expected to emerge.
with SLI. However, when late talker status was combined This homogeneity should allow for statistical aggregation of
with the risk factors, the discrimination of the model im- effect sizes across studies via meta-analysis and quantifica-
proved and, in the case of the expressive SLI group, the tion of the predictive strength of each risk factor.
predictive accuracy of the model increased significantly.
Together, the findings of these studies suggest that
the right combination of early behaviors and early risk fac- Clinical Significance
tors could yield a simple, efficient, and highly informative Bothe and Richardson (2011) emphasized the impor-
SLI screening tool appropriate for use in clinics and pedi- tance of looking beyond statistical and practical signifi-
atric practices. The first step, then, is to specify which risk cance when examining the results of an intervention study.
factors are the strongest and most clinically relevant pre- They argue that p values and effect sizes do not address
dictors of the disorder. The purpose of the current study the question of whether the improvement observed over the
was to synthesize the available epidemiological data on course of treatment reflects a meaningful level of change.
SLI risk factors and to determine which of the factors, if The concept of clinical significance has application beyond
any, are clinically significant predictors of SLI. The ulti- the realm of intervention research, however. In all areas of
mate goal of this work is to pave the way to earlier and clinical practice and investigation, it is valuable to know
more accurate identification of children with chronic and whether one’s professional approach is meaningful and ap-
debilitating language learning deficits. propriate. This is usually determined through comparison
to a gold standard—that is, a practice or approach that has
been found to be clinically reliable and is well established
The Importance of Homogeneity in the field. Within the context of the current review, the
Risk factor studies have sought to predict a wide question becomes how do we know that a given risk factor
range of language outcomes including expressive vocabu- provides valuable diagnostic information.
lary delay (Fischel, Whitehurst, Caulfield, & DeBaryshe, Because there is currently no gold standard against
1989; Henrichs et al., 2011; Horwitz et al., 2003; Whitehurst, which to compare a given risk factor’s predictive strength,
Smith, Fischel, Arnold, & Lonigan, 1991), late language clinical significance in the current review was judged
emergence (Zubrick, Taylor, Rice, & Slegers, 2007), phono- against a standard that is frequently used by pediatricians,
logical disorders (Campbell et al., 2003; Fox, Dodd, & child care providers, parents, and researchers to decide
Howard, 2002), low language performance (Choudhury & whether further investigation, professional evaluation, or
Benasich, 2003), vocabulary deficits (Law, Rush, Schoon, language services are warranted. This standard is delayed

992 American Journal of Speech-Language Pathology • Vol. 26 • 991–1010 • August 2017


development of expressive vocabulary skills (i.e., late talk- Hand Search
ing). Some consider late talking to be a prediagnosis that re- Secondary sources identified during the database
quires close monitoring and watchful waiting (Paul, 2000), searches were the origin of the references examined in the
whereas others view it as a clinical diagnosis that warrants hand search. All nonduplicate, English-language, journal
direct and immediate intervention (The Hanen Centre, article references were extracted from the reference lists of
2015). In spite of evidence that reveals the limitations of these secondary sources and entered into a Microsoft Excel
this construct as a predictor of SLI (Dollaghan, 2013), late workbook. An automated filter was applied to the titles
talking has become a diagnostic phenomenon due to its ac- to separate out those articles that focused on SLI from
cessibility and to the extensive research that has surrounded those that did not. The filter was comprised of 23 full or
it. Given its popularity as an early identification criterion, abbreviated terms that have been used to refer to SLI in
late talker status was judged to be an appropriate compari- previous research (see Supplemental Material S2). Next,
son standard for the purpose of the current review. Evidence the abstracts for all of the references that survived title
that any single case history risk factor is equally or more evaluation were collected and entered into the workbook.
predictive than late talker status holds important implica- A second automated filter was applied to the abstracts
tions for clinicians, researchers, and other professionals to distinguish those articles that focused on risk factors
interested in early identification of children with language from those that did not. This filter was comprised of
impairments. 18 full or abbreviated terms that had been used to refer
to risk factors in the abstracts of the articles identified dur-
Research Question ing the database searches (see Supplemental Material S2).
Last, the full texts of all remaining articles were obtained
In this study, the following question was addressed: and classified as primary or secondary sources. Primary
Can specific case history factors be used to identify toddlers studies were submitted to further evaluation.
in the general population who are at risk of developing
SLI? This question was examined in two phases. Phase I
focused on determining whether any of the factors examined Reference Evaluation Criteria
in previous research are statistically significant predictors The references were evaluated by title, abstract, and
of SLI. Phase II focused on assessing whether any statisti- full text. Criteria were established to yield a cluster of
cally significant predictors are also clinically significant. studies matched on certain critical design features. These
design features included: (a) focus on an epidemiological
sample, (b) inclusion of a group of participants consisting
Method exclusively of children with SLI, (c) examination of one or
Search Methods more case history risk factors, and (d) provision of odds
A three-step approach was used to identify relevant ratios (ORs) or enough data to allow for the calculation of
studies for this review. The first two steps involved data- ORs. These four features were selected to align with the
base searches; the initial search used broad risk factor ter- research question: Can specific case history factors be used
minology and the follow-up search used specific risk factor to identify toddlers in the general population who are at
terminology. The third step was a systematic hand search risk of developing SLI?
of references extracted from secondary sources identified
during the database searches. These steps are described Criterion 1: Epidemiological Sample
briefly below. Additional details can be found in Supple- The research question establishes the need to make
mental Materials S1 and S2. diagnostic distinctions among members of the general
population. Although sampling bias is a common and fre-
Database Searches quently cited barrier to external validity in all lines of
Five electronic databases were searched up to May human research, it is particularly problematic in studies
2015 for the initial database search and up to March 2016 that have population-wide decisional implications (Dollaghan,
for the follow-up database search. With the exception of 2015). Such bias can result in patterns of association be-
the search string, procedures were identical across the tween risk factors and outcomes that grossly overestimate
searches. No date range was specified to maximize returns. or underestimate true population-level associations (Fletcher,
English-language, peer-reviewed articles were the target of Fletcher, & Fletcher, 2014). Making a diagnostic decision
this review; therefore, books, book chapters, conference on the basis of skewed effects, even at the level of assigning
papers, dissertations, and articles not available in English risk, can have major repercussions for the individual, for
were excluded. Nonduplicate primary studies were submit- the family, and for society.
ted to further evaluation. The full texts of the primary In order to limit the influence of sampling bias in the
studies identified during the initial database search were current review, an effort was made to focus on epidemio-
examined for specific risk factor terms (e.g., family history, logical cohorts by evaluating two participant sample char-
maternal education, socioeconomic status [SES]), which acteristics. The first characteristic was whether participants
were used in the follow-up search. Reviews and other sec- were recruited using a one-gate or two-gate design (Bossuyt
ondary sources were dealt with separately. & Leeflang, 2008). In a one-gate design, the goal is generally

Rudolph: Case History Risk Factors for SLI 993


to obtain a sociodemographically representative sample of Criterion 3: Case History Risk Factors
participants by recruiting all of the individuals, for example, The research question specifies that case history risk
living within a certain geographic region, entering a certain factors are the exposure of interest. For the current review,
grade level, or born in a certain hospital in a given year. the term risk factor was defined as any prenatal, perina-
Some of these individuals, presumably a small proportion, tal, neonatal, child, parent, or family characteristic that
will present with disorders and some will not, but partici- might be associated with a child’s language learning abili-
pants are not selected on the basis of the presence or absence ties (Berkman et al., 2015; Harrison & McLeod, 2010;
of a diagnosis. In a two-gate design, on the other hand, the Nelson, Nygren, Walker, & Panoscha, 2006; Olswang
goal is to obtain two separate, and usually matched groups et al., 1998). The term case history was used to specify risk
of participants—one group exhibiting the disorder of inter- factor accessibility. Risk factors such as birth order and
est, the other developing typically. In general, the first group maternal education level, which can be collected via a
is obtained through clinical referral, and the second group is paper and pen questionnaire, are accessible by everyone
obtained through advertising in local newspapers or through and are, therefore, appropriate for routine early identifica-
fliers posted at preschools and daycare centers. Participant tion screening purposes. Risk factors such as microdele-
samples recruited by means of a two-gate design are not tions on certain genes and abnormal activity or unusual
intended to be representative of the general population and anatomical symmetry in specified brain regions are only
are, therefore, much more prone to sampling bias. Thus, accessible by those who have the equipment and training
one-gate studies were the focus of the current review. to obtain them. Thus, these risk factors are not appropriate
The second characteristic was whether participants for routine screening and were not included in the current
had been recruited from a special population or from the investigation. Furthermore, to ensure applicability to early
general population. It is possible that an investigator might identification practices, only risk factors that are observ-
use a one-gate design to selectively recruit, for example, able before the age of 3 years were considered. Note that
babies who were admitted to the neonatal intensive care early developing behaviors, such as babbling, gesture use,
unit, children of a multiple birth (e.g., twins), or individ- and vocabulary development did not fall under the scope
uals living in poverty. Selectively recruited samples are, by of this review.
definition, going to exhibit some level of sampling bias.
Furthermore, risk-outcome associations may be even more Criterion 4: ORs
skewed in these samples given that the participants are Last, the research question indicates that the goal is
often already at risk for language problems. Thus, studies to determine the level of risk associated with each factor.
that recruited from the general population were the focus The OR is an effect size that is designed to quantify risk. It
of the current review. By converging on one-gate, general is defined as a measure of association between a given ex-
population study samples, it was anticipated that epidemi- posure (e.g., later birth order) and an outcome (e.g., SLI).
ological cohorts would be identified. It is calculated as the odds that the outcome will occur
when an exposure is present over the odds that the outcome
Criterion 2: SLI will occur when an exposure is absent. If the odds of devel-
SLI is the outcome of interest specified in the research oping the disorder are the same whether or not an individ-
question. For the current review, SLI was broadly defined ual is exposed, the calculation yields an OR value of 1.0,
as a significant impairment in receptive and/or expressive suggesting that the exposure does not increase an individ-
grammar, morphology, or syntax in the absence of an obvi- ual’s risk of developing the disorder. If the odds of develop-
ous cause (e.g., hearing impairment, intellectual disability, ing the disorder are greater among those who are exposed,
autism spectrum disorder, etc.). This definition, therefore, the calculation yields an OR value greater than 1.0, sug-
excluded children with a primary diagnosis of speech sound gesting that the exposure increases the individual’s risk of
disorder, dyslexia, or pragmatic language impairment, al- developing the disorder. If the odds of developing the disor-
though children with SLI often have comorbid phonologi- der are greater among those who are not exposed, the cal-
cal, reading, and social difficulties (Leonard, 2014). Within culation yields an OR value less than 1.0. In this scenario,
individual studies, the particular method of diagnosis as the exposure is protective, which means it decreases an indi-
well as the quantity and stringency of SLI exclusionary cri- vidual’s risk of developing the disorder. Although the OR
teria were left free to vary. These variations were permitted value provides an estimate of the association between a risk
given that there is currently no standardized method for di- factor and an outcome for a particular group of partici-
agnosing SLI (Bishop, 2014); however, the age of diagnosis pants, the 95% confidence interval (CI) indicates what the
was strictly set at 4 years of age or older. This cut-off was OR might be within the general population. If the 95% CI
selected because language skills, particularly those aspects includes the value 1.0, this indicates that there may actually
of grammar most affected by SLI, are generally mastered be no association at the population level. For example, one
by this age (Rice, Wexler, & Cleave, 1995), making diagno- study found that the OR corresponding to the risk factor
sis of the condition more reliable. Many children diagnosed maternal smoking during pregnancy was 1.60 (Tomblin,
with language delays or deficits before the age of 4 years Hammer, & Zhang, 1998), suggesting that the odds of de-
have been found to spontaneously recover and therefore do veloping SLI among children whose mothers smoked while
not meet criteria for SLI (Leonard, 2013). pregnant are 1.6 times higher than the odds of developing

994 American Journal of Speech-Language Pathology • Vol. 26 • 991–1010 • August 2017


SLI for children whose mothers did not smoke. However, the definition of case history risk factor, and providing
the 95% CI ranged from 0.90 to 2.50, which includes the too little information to identify or calculate OR values.
value 1.0. It is necessary to take the 95% CI into account All primary studies that passed at the full text level were
when assessing statistical or practical significance (Bothe retained for the systematic review.
& Richardson, 2011). This review focused on studies that
either directly provided OR values and 95% CIs for the risk
Interrater Reliability
factors investigated, or provided enough information for
Two independent raters were trained on the criteria
OR values to be calculated. At a minimum, exposure data
and performed interrater reliability for all phases of the ref-
for a group with the outcome of interest and exposure data
erence evaluation process. Rating occurred consecutively—
for a comparison group are needed. For the sake of consis-
that is, the raters first performed reliability on a randomly
tency, TD children—that is, children developing language
selected set of 30% of the titles, then on a randomly se-
normally—were selected as the comparison group in the
lected set of 30% of the abstracts, and, finally, on a randomly
current review, so only studies that contained both an SLI
selected set of 20% of the full texts. The reliability work
group and a TD group were included.
was divided between the two raters. Reliability was calcu-
lated by dividing the total number of agreements by the
Reference Evaluation Procedures total number of opportunities for agreement. Agreement
These criteria were operationalized to enable exclu- for a given reference was defined as a mutual rating of IN
sion of irrelevant primary studies. Evaluation of titles and or OUT by both the author and the independent rater.
abstracts was undertaken to be highly inclusive, so that Across all three steps of the reference search (i.e., initial
only articles that explicitly addressed topics unrelated to database search, follow-up database search, hand search),
the research question were eliminated. Full text criteria total agreement was 95% for titles, 93% for abstracts, and
were highly stringent to ensure that the final article set sat- 97% for full texts. Out of 514 opportunities, there were a
isfied all of the criteria outlined above. See Supplemental total of 27 disagreements. Among these, eight were rated
Material S3 for an overview of the specific exclusions ap- as OUT by the author and IN by either the first or second
plied at each level. rater, indicating a potential data loss of only 1.6% across
all phases.
Title Level
Every reference retrieved from the database searches Data Extraction
and all references that survived the title filter (hand search
references only) were evaluated at the title level. It was Study Details
anticipated that information about the nature of the sam- The following information was extracted from each
ple (i.e., Criterion 1) and the statistics reported (i.e., Crite- qualifying study: (a) sample size, (b) number of children
rion 4) would not be available in the titles; therefore, the with SLI, (c) number of TD children, (d) participant ages,
title exclusions focused on Criteria 2 and 3. In brief, titles (e) participant races/ethnicities, (f ) geographical location of
were excluded if they indicated that the article focused the study population, (g) risk factor information source,
on a disorder other than SLI or on molecular genetics, (h) participant diagnostic information source, (i) diagnostic
neuroanatomy, or neurophysiology. measure(s) administered, ( j) cut-off score used to classify
children as SLI or TD, (k) naive status of individuals who
Abstract Level reported or collected risk factor or diagnostic information,
Every reference that passed at the title level and sur- and (l) chronology of risk factor report (i.e., prospective
vived the abstract filter (hand search references only) was or retrospective). Items (a) through (f ) were extracted for
evaluated at the abstract level. The same operationalized descriptive purposes and items (g) through (l) for the pur-
criteria served as the basis for abstract exclusion; however, pose of quality appraisal.
review of the abstracts revealed that several of the studies
focused on intervention approaches. As intervention does Risk Factors and OR Values
not satisfy the definition of a case history risk factor, ab- Risk factors and their corresponding effect sizes were
stracts were also excluded if they referred to intervention also extracted. OR values and 95% CIs were the effect
as the independent variable in the study. size of interest. Some studies reported adjusted OR values;
some reported unadjusted OR values; some reported fre-
Full Text Level quency data (for dichotomous and categorical risk factors)
Every reference that passed at the abstract level was or means and standard deviations (for continuous risk
evaluated at the full text level where all four criteria were factors), which could be used to calculate unadjusted OR
represented. In brief, full texts were excluded for using a values; and, finally, some studies reported a mixture of
two-gate recruitment approach, selectively recruiting from statistics. Adjusted and unadjusted OR values differ in the
a special population, not including a group of children way they are obtained and interpreted. Unadjusted OR
who were diagnosed with SLI after the age of 4 years, fo- values represent the association between a risk factor and
cusing on characteristics, abilities, or traits that fall outside outcome when no other factors are taken into consideration.

Rudolph: Case History Risk Factors for SLI 995


Adjusted OR values represent the association between the Quality Appraisal
risk factor and outcome when other potentially confound-
An adaptation of the Quality Assessment of Diag-
ing variables are controlled for. A confounding variable
nostic Accuracy Studies–2 (QUADAS-2; Whiting et al.,
is one that is associated with both the risk factor and the
2011) was used to appraise the quality of included studies.
outcome, but is not a mediator in the causal pathway be-
The QUADAS-2 is used to characterize the risk of bias
tween the two (Szklo & Nieto, 2007). Take the association
and applicability concerns that may exist in studies investi-
between maternal smoking and SLI. The OR value and
gating the diagnostic accuracy of new assessment instru-
95% CI mentioned previously are unadjusted (see Crite-
ments. Risk of bias results when there are threats to the
rion 4 above); however, the investigators also examined
internal validity of a study. An example is test adminis-
this association after taking parent education level into
tration by an individual who is not naive to participants’
consideration as a confounding variable. In order to per-
diagnostic status, which could bias the test results. Ap-
form this analysis, they first had to assume that parent
plicability concerns result when there are threats to the
education level is not a causal mediator between maternal
external validity of a study. An example is using a non-
smoking and SLI. This is reasonable given that an ex-
traditional diagnostic approach that could not be gener-
pected causal mediator might be something such as birth
alized to clinical practice. The QUADAS-2 focuses on
weight—that is, maternal smoking during pregnancy
five key domains: (a) participant selection, (b) index test,
might be causally associated with a child being born at a
(c) reference standard, (d) participant flow, and (e) timing.
low birth weight, which might then be causally associated
Domains (a) through (c) are assessed for both risk of
with SLI through a biological mechanism. In contrast,
bias and applicability concerns, whereas domains (d) and
maternal smoking during pregnancy does not cause less
(e) are assessed only for risk of bias. Studies can be rated
education, and, therefore, the association between mater-
as low, high, or unclear. A low rating is given when risk
nal smoking and SLI is unlikely to be causally mediated
of bias and/or applicability concerns are low. The more
by parent education level. It is interesting to note that
low ratings a study receives, the higher the quality. A high
when the investigators performed the analysis, the OR
rating is given when risk of bias and/or applicability con-
value was reduced and the 95% CI was shifted, suggesting
cerns are high. The more high ratings a study receives,
that parent education level was a confounding variable
the poorer the quality. An unclear rating is given when
in the association between maternal smoking and SLI. Al-
risk of bias and/or applicability concerns cannot be deter-
though focusing on adjusted OR values can be instructive,
mined on the basis of the information provided in the arti-
it can also be misleading if the variables that are chosen
cle. Unclear ratings indicate the need for more detailed
as confounders are, in fact, causal mediators. Because
disclosure of study features related to internal and external
of the high potential for causal mediation among the risk
validity.
factors investigated in this review, unadjusted OR values
were chosen as the primary statistic of interest. As such,
extraction of relevant statistics occurred in the following QUADAS-2 Domain 1: Participant Selection
sequence: unadjusted OR values, data that could be used In order to qualify for the present review, study par-
to calculate unadjusted OR values, and adjusted OR ticipants had to be recruited using a one-gate design and
values. Because some studies reported only adjusted not exclusively from a special population. As such, risk of
values, analyses were completed to determine whether their bias in the domain of participant selection would be likely
inclusion yielded significant differences in the obtained only in cases were inappropriate exclusions were applied.
results. Some inappropriate exclusions include eliminating all chil-
dren from a low-SES background or all those who exhib-
ited borderline language scores. Studies that used these
Interrater Reliability types of exclusions received a high risk of bias rating for
An independent observer, naive to the research this domain. Applicability concerns were expected only in
question, was trained on the procedures and performed cases where, as a result of errors in recruitment or diagnos-
reliability for 25% of calculated OR values. Four data tic procedures, the proportions of children in the TD or
points had to be extracted for each reliability item: SLI groups were substantially skewed. Examples of skewed
(a) number of children with SLI exposed to the risk fac- samples include those exhibiting an SLI prevalence rate
tor, (b) number of TD children exposed to the risk factor, below 3% or over 20% as accepted rates are between 5%
(c) number of children with SLI not exposed to the risk to 15%. Such disproportionality resulted in high applicabil-
factor, and (d) number of TD children not exposed to ity concern ratings in this QUADAS-2 domain.
the risk factor. Agreement was based on an exact match
between the author and independent observer on each QUADAS-2 Domain 2: Risk Factor Collection
of these four numerical values, and was calculated as For this review, the QUADAS-2 domain, “index
the total number of agreements over the total number test,” was adapted to apply to risk factor information
of opportunities for agreement, which came out to 94%. collection procedures. In this domain, risk of bias was
The disagreements yielded only slight differences, which associated with the naive status of the individuals reporting
had no effect on the final results of the meta-analyses. and collecting risk factor information. Studies received high

996 American Journal of Speech-Language Pathology • Vol. 26 • 991–1010 • August 2017


risk of bias ratings if neither the reporter nor the collector the final data are drawn. An example of systematic attri-
were naive to the participant’s diagnostic status. Judgment tion is discontinued participation of individuals from low
of applicability concerns focused on the feasibility of the socioeconomic backgrounds as a result of lack of resources
information collection methods. Studies received high rat- or transportation. If statistically significant differences in
ings for applicability concerns if, for example, family his- demographic, diagnostic, or risk factor characteristics were
tory information was collected through direct testing of found between participants in the initial cohort and the
all the participant’s family members because this is an ap- final sample, the study received a high risk of bias rating
proach that would be impractical to implement in a clinical in this domain. Those studies in which differences between
setting. the initial and final samples were not undertaken received
an unclear risk of bias rating.
QUADAS-2 Domain 3: Diagnostic Status
The QUADAS-2 domain, “reference standard,” was Data Analysis
adapted to apply to the diagnostic classification procedures
used within the risk factor studies. There were three main Phase I
considerations in the diagnostic status domain. The first The goal of Phase I was to determine which risk fac-
two were related to risk of bias and addressed, first, whether tors are statistically significant predictors of SLI. Meta-
the procedures were valid and likely to correctly classify analysis is a statistical method by which effect sizes, in this
children with the target condition and, second, whether the case OR values and 95% CIs, are aggregated across indi-
person administering the assessment or making the diag- vidual studies to produce an estimate that is representative
nostic decision was naive to the participant’s risk factors. of the range of effect sizes that may exist for a particular
Studies in which classification accuracy was dubious because factor. The aggregated OR value and 95% CI are generally
of the assessment methods used were rated as exhibiting high more precise than the values reported in any single study,
risk of bias. In general, if the authors used a standardized and can therefore be used to make more accurate assess-
language test with an established cut-off value, risk of bias ments of a factor’s statistical significance. Thus, in the cur-
was considered low; however, if, for example, classification rent review, meta-analysis procedures were used to determine
was based on subjective observation of symptoms, risk of the statistical significance of each eligible risk factor.
bias was considered high. Note that there was no direct
consideration of the psychometric properties of the standard- Phase II
ized assessments used. The third consideration addressed The goal of Phase II was to determine which statisti-
whether the procedures were similar to those that would cally significant risk factors from Phase I are also clinically
be implemented in clinical practice. If the procedures were significant predictors of SLI. This was accomplished by
unlike typical assessment methods, for example, based comparing the 95% CIs calculated for each significant
solely on parent report without direct evaluation, the study risk factor to the 95% CI associated with late talker status.
received a high rating for applicability concerns. Thus, late talker status was the standard against which the
clinical significance of each risk factor was judged.
QUADAS-2 Domain 4: Timing
For the current review, the “timing” domain was Results
used to refer to the timing of the risk factor report. Infor-
mation could be reported prospectively, meaning that risk Search Results
factors such as birth weight, gestational age, and Apgar Figure 1 provides a flowchart of the reference identi-
score were reported as soon as the information became fication and evaluation process combining the results
available (i.e., birth) and the children were followed for- from the initial database search, the follow-up database
ward in time. However, the information could be reported search, and the hand search. Ten studies were identified;
retrospectively, meaning that caregivers were asked to re- however, six of these studies focused on the same popula-
call events in their child’s life that occurred in the past. tion of children—that is, the participants from the National
Retrospective reporting can be easily biased by the inaccu- Institute of Deafness and Other Communication Disorders
racy of human memory, especially if parents are looking epidemiological study examining the prevalence of SLI in
for a cause or reason for their child’s difficulties. As a kindergarten-age children, hereafter, the EpiSLI cohort
result, studies that collected risk factor information retro- (Fey, Catts, Proctor-Williams, Tomblin, & Zhang, 2004;
spectively received a high risk of bias rating in the timing Hammer, Tomblin, Zhang, & Weiss, 2001; McGregor,
domain. Oleson, Bahnsen, & Duff, 2013; Tomblin et al., 1998;
Tomblin, Records, et al., 1997; Tomblin, Smith, et al.,
QUADAS-2 Domain 5: Participant Flow 1997). Examination of these articles revealed that the two
This last domain focused on the loss of participants most recent studies in the series (Fey et al., 2004; McGregor
from initial recruitment to final analysis. Attrition is com- et al., 2013) provided risk factor information that entirely
mon in prospective longitudinal studies, but systematic overlapped with the information reported in several of the
attrition can result in significant differences between the earlier studies; however, the samples in the recent studies
initial cohort of participants and the sample from which were more restricted because of missing data or loss from

Rudolph: Case History Risk Factors for SLI 997


Figure 1. Flowchart of the three-step reference search and evaluation process.

attrition as the participants were followed longitudinally. Ing, & Newnham, 2014) leaving a total of eight studies to
Because these studies provided no new information and include in the meta-analyses.
represented reduced subsets of the original epidemiological
sample, they were not included in the review. The four
remaining studies in this group each contained some novel, Data Extraction
but also some overlapping risk factor data. This was Table 1 provides details pertaining to the partici-
addressed during Phase I of data analysis. The four stu- pants in the eight included studies. A total of 212,984 chil-
dies that did not focus on the EpiSLI cohort contained dren, age 4 to 8 years, participated in these studies, 6,275
completely independent samples (Merricks, Stott, Goodyer, with SLI and 206,709 developing language normally.
& Bolton, 2004; Reilly et al., 2010; Stanton-Chapman, Note that these values took into consideration the fact that
Chapman, Bainbridge, & Scott, 2002; Whitehouse, Shelton, four of the studies focused on the EpiSLI cohort—that is,

998 American Journal of Speech-Language Pathology • Vol. 26 • 991–1010 • August 2017


Table 1. Study population characteristics.

Study N SLI TD Ages Race/ethnicity Location

Hammer et al. (2001)a 1,057 171 886 6 years Unavailable USA


Merricks et al. (2004) 98 33 65 8 years Unavailable UK
Reilly et al. (2010) 1,360 138 1,222 4 years Unavailable Australia
Stanton-Chapman et al. (2002) 207,692 5,862 201,830 6–7 years Unavailable USA
Tomblin, Records, et al. (1997)a 2,009 216 1,793 6 years 83.6% White USA
13.6% African American
2.8% Other
Tomblin, Smith, et al. (1997)a 1,102 177 925 6 years Unavailable USA
Tomblin et al. (1998)a 1,102 177 925 6 years 91.2% White USA
5.1% African American
1.6% Hispanic
2.1% Other
Whitehouse et al. (2014) 1,825 26 1,799 5 years Unavailable Australia

Note. SLI = specific language impairment; TD = typically developing.


a
These studies focused on the National Institute of Deafness and Other Communication Disorders EpiSLI sample.

the totals were calculated including only the sample from that risk of bias due to timing of risk factor report and
the original study (Tomblin, Records, et al., 1997), and participant flow was likely to threaten the internal validity
excluding the samples from the remaining three studies of many studies, whereas under- or overrepresentation of
(Hammer et al., 2001; Tomblin et al., 1998; Tomblin, children with SLI in the participant samples of a few stud-
Smith, et al., 1997). Only two studies reported participant ies could threaten their external validity.
race and ethnicity characteristics, and none of the studies
reported dialect variations. All studies took place in
Phase I Data Analysis
English-speaking countries, five in the United States, in-
cluding the four EpiSLI cohort studies, two in Australia, The goal of Phase I was to perform a separate meta-
and one in the United Kingdom. analysis for each eligible risk factor and to determine
which among them met criteria for statistical significance
(i.e., 95% CI lower limit > 1.0). An overview of this pro-
Quality Appraisal cess is provided in Figure 3. A total of 162 OR values and
Table 2 provides the details concerning research de- 95% CIs distributed across 122 different risk factors were
sign features related to the quality of evidence from each extracted or calculated from the eight included studies (see
study. Figure 2 summarizes the results of the quality ap- Supplemental Material S4). For the sake of consistency,
praisal assessment by domain from the modified version of values weighted protectively (i.e., OR values < 1.0) were
the QUADAS-2. Risk of bias was low for all of the stu- converted to risk values by means of inversion.
dies in the domain of participant selection, whereas both
risk of bias and applicability concerns were low for the Classification
vast majority of studies in the domains of risk factor col- To be eligible for meta-analysis, a risk factor must
lection and assignment of diagnostic status. In contrast, be represented by at least two effect sizes. If a factor is rep-
risk of bias was high in the domain of timing for half of resented by only one effect size, that effect size is elimi-
the studies (three of which focused on the EpiSLI cohort) nated from further analysis. As a result, 109 out of the
due to the high potential for erroneous reporting associ- 162 effect sizes extracted for the current review would have
ated with retrospective collection of risk factor informa- been excluded. To prevent major data loss, each effect
tion. Furthermore, the majority of studies were unclear or size (i.e., OR value and 95% CI) was classified into one of
exhibited high risk of bias in the domain of participant 25 risk categories. For example, effect sizes for long labor,
flow either because differences between the initial cohort delivery by forceps, induction, and emergency cesarean
and final participant sample were not analyzed (unclear) section were classified into the category perinatal event,
or differences were analyzed and found to be systematic whereas effect sizes for per capita income, percentage of
(high). Applicability concerns for the domain of partici- families below poverty, and income-to-poverty ratio were
pant selection were high for 35% of the studies due to no- classified into the category SES. The 25 risk categories
table disproportionality in the prevalence of SLI within were: biological sex, birth order, birth weight, childhood
the participant samples—that is, the prevalence was either chemical exposure, exposure to more than one language,
very high or very low compared with accepted population family history of communication or related disorders, be-
estimates (see Table 2). Overall, these results suggested ing formula fed, maternal age at child’s birth, maternal
that the quality of the set of included studies was high, but education level, paternal education level, maternal illness

Rudolph: Case History Risk Factors for SLI 999


Table 2. Quality characteristics.

Risk factor Diagnostic Diagnostic Cut-off Naive


Study SLI source source test score statusa Timing

Hammer et al. (2001)b 16.2% Parent report Direct assessment Batteryc −1.25 SDd Reporter Retrospective
Collector
Assessor
Merricks et al. (2004) 33.7% Parent report Direct assessment CELF-R −1.5 SD Retrospective
Reilly et al. (2010) 10.1% Parent report Direct assessment CELF-P2e −1.25 SD Reporter Prospective
Collector
Stanton-Chapman 2.8% Birth record School system N/A N/A Reporter Prospective
et al. (2002)
Tomblin, Records, 10.8% School record Direct assessment Battery c
−1.25 SD d
Reporter Concurrent
et al. (1997)b Collector
Tomblin, Smith, 16.1% Parent report Direct assessment Batteryc −1.25 SDd Reporter Retrospective
et al. (1997)b Collector
Assessor
Tomblin et al. 16.1% Parent report Direct assessment Batteryc −1.25 SDd Reporter Retrospective
(1998)b Collector
Assessor
Whitehouse et al. 1.4% Parent report and Parent report N/A N/A Reporter Prospective
(2014) medical record

Note. SLI = specific language impairment; CELF-R = Clinical Evaluation of Language Fundamentals–Revised (Semel, Wiig, & Secord, 1992);
CELF-P2 = Clinical Evaluation of Language Fundamentals–Preschool 2 (Wiig, Secord, & Semel, 2006).
a
Three types of naive status were possible given the nature of these studies: (a) naive status of the reporter—the individual reporting risk
factor information was naive to participants’ diagnostic status; (b) naive status of the collector—the individual collecting risk factor information
was naive to participants’ diagnostic status; (c) naive status of the assessor—the individual assigning diagnostic classifications was naive to
participants’ risk factor information. bThese studies focused on the National Institute of Deafness and Other Communication Disorders EpiSLI
sample. cBattery included subtests of the Test of Language Development–Second Edition (TOLD-II:P; Newcomer & Hammil, 1988) and a
narrative task. dOn two out of five tests in the battery. eAustralian adaptation.

during pregnancy, maternal mental health, maternal vo- of a congenital abnormality, presence of a newborn con-
cabulary, minority status, multiple birth, onset of prenatal dition, presence of a pregnancy condition, and SES. This
care, parent marital status, parenting behavior, perinatal approach substantially reduced the number of factors rep-
event, prematurity, prenatal chemical exposure, presence resented by only one effect size.

Figure 2. Quality appraisal results divided according to QUADAS-2 domain. Low ratings indicate good study quality. High ratings indicate
poor study quality. Unclear ratings were assigned when information was not provided and could not be determined.

1000 American Journal of Speech-Language Pathology • Vol. 26 • 991–1010 • August 2017


Figure 3. Overview of Phase I data analysis procedures. for several risk categories. For example, for the category
SES, four nearly identical effect sizes were reported for the
EpiSLI cohort, and two nearly identical effect sizes were
reported for the Reilly et al. cohort. For the EpiSLI cohort,
this phenomenon was the result of the same population
being the focus of four different studies (Hammer et al.,
2001; Tomblin et al., 1998; Tomblin, Records, et al., 1997;
Tomblin, Smith, et al., 1997). For the Reilly et al. cohort,
this phenomenon was the result of the authors reporting
effect sizes separately for overlapping samples (children
with expressive SLI and children with receptive SLI). Alto-
gether, these two studies yielded a total of 52 redundant
effect sizes. To eliminate redundancy in the EpiSLI data,
effect sizes from the oldest studies in the series were retained
for meta-analysis. This approach was taken because the
older studies had larger samples and examined the relevant
factors as they pertained to risk rather than simply report-
ing them as demographic characteristics. All other effect
sizes were excluded. To eliminate redundancy in the Reilly
et al. data, only the effect sizes for the receptive SLI group
were included because that sample was larger, allowing
for consideration of more factors. Together, these exclu-
sions resulted in the removal of 27 redundant effect sizes
leaving a total of 135 OR values and 95% CIs for further
evaluation.
Categorical risk factors. The second step involved re-
ducing categorical risk factors. For example, effect sizes
for maternal education were generally reported separately
for distinct education levels: less than a high school degree,
high school degree, some college, college degree, and ad-
vanced degree. As in this example, there might be up to
five effect sizes representing a categorical risk factor for a
single cohort. The decision of which effect size to select for
meta-analysis was based on the lower limits of the 95%
CIs. Lower limits indicate the minimum degree of associa-
tion between a risk factor and an outcome in the general
population, thus, the greater the lower limit, the greater
the association. For example, in the study by Stanton-
Chapman et al. (2002) the level “less than a high school
degree” was associated with a lower limit of 1.90, whereas
the level “high school degree” was associated with a lower
limit of 1.00. Because 1.90 is greater than 1.00, “less than
a high school degree” was the most predictive level in this
study, and was selected as the representative effect size for
this cohort. Reduction of categorical risk factors resulted
in the removal of 28 effect sizes, leaving 107 OR values
and 95% CIs for further evaluation.
Data Trimming Combined effect sizes. The third step involved combin-
Once the effect sizes were categorized, data trimming ing effect sizes for individual risk factors in each study that
was undertaken to ensure that the effect sizes falling into fell into the same risk category. For example, Whitehouse
each risk category were independent—that is, derived from et al. (2014) reported separate effect sizes for high maternal
independent participant samples. The three steps in this body mass index, hyperemesis, gestational diabetes, threat-
process included: (a) eliminating redundant effect sizes, ened abortion, hemorrhage, preeclampsia, and preterm
(b) reducing categorical risk factors, and (c) combining ef- hospital admission, all of which fell into the risk category
fect sizes for individual risk factors falling into the same pregnancy condition. Rather than select an individual risk
risk category. factor to represent the category, a combined effect size
Redundant effect sizes. First of all, the EpiSLI and was calculated using comprehensive meta-analysis (CMA,
Reilly et al. (2010) studies reported redundant effect sizes 2014). Combined OR values represent the average of the

Rudolph: Case History Risk Factors for SLI 1001


individual OR values, whereas combined 95% CIs take bias assessment involves plotting standard error, which is
into consideration the variance associated with and the a reflection of sample size, against OR value. Effect sizes
correlation among the individual OR values. Using this from studies with larger samples appear at the top of the
procedure, 13 combined effect sizes were calculated, which plot and tend to cluster closely around the mean OR value,
fell into seven unique risk categories (parenting behavior, whereas effect sizes from smaller studies appear at the
presence of a newborn condition, prenatal chemical expo- bottom and tend to be more widely dispersed. This distri-
sure, family history of communication or related disorders, bution of data points gives the impression of an upside
maternal illness during pregnancy, perinatal event, and down funnel (Light & Pillemer, 1984). If the funnel plot
presence of a pregnancy condition). Details are reported is symmetric around the mean OR value, this suggests that
in Table 3. the set of effect sizes represents an unbiased sample. If,
After data trimming was completed, the dataset was however, more effect sizes are distributed on one side of
reduced to 52 OR values and 95% CIs. Each of the 25 risk the mean than on the other, this suggests that some effect
categories was represented by one or more completely in- sizes are missing and that bias may exist.
dependent effect sizes. The funnel plot can be visually assessed for bias, but
a more objective method involves use of trim and fill im-
Finalizing the Risk Factor Set putation (Duval & Tweedie, 2000). In this iterative approach,
As mentioned above, risk categories represented by the most extreme effect sizes on either side of the funnel
only a single effect size were ineligible for meta-analysis. plot are systematically “trimmed” from the plot and the
Thus, once the effect sizes were categorized and trimmed, mean OR value is recalculated until the effect sizes are
11 risk categories were excluded. These were childhood symmetrically disbursed. During the “fill” phase of this
chemical exposure, exposure to more than one language, analysis, the original effect sizes are all replaced, and mirror
being formula fed, maternal mental health, maternal vo- effect sizes are imputed on the opposite side of the plot.
cabulary, minority status, multiple birth, onset of prenatal This allows for the calculation of a new 95% CI. The sever-
care, parenting behavior, paternal education level, and ity of the bias is estimated on the basis of the difference
presence of a congenital abnormality. After this procedure, between the original and imputed values. If the two values
41 OR values and 95% CIs distributed across 14 catego- lead to different conclusions, then the impact of bias is
ries remained. severe. If little to no difference is observed between the
values, the impact of bias is minimal. The results of the
Assessing Dataset Suitability trim and fill imputation procedure for the current review
Before meta-analyses were undertaken for the showed that no effect sizes needed to be trimmed from the
14 qualifying risk categories, the suitability of the dataset right side of the funnel plot, but 14 effect sizes needed to
was assessed. This process involved (a) examining the poten- be trimmed from the left side in order to form a symmetric
tial influence of adjusted effect sizes, (b) considering the distribution around the mean OR value. During the fill
possibility of publication bias, and (c) identifying hetero- phase, 14 mirror data points were imputed onto the right
geneity among the effect sizes. side of the graph. These findings were not surprising given
Adjustment. Two of the included studies (Reilly et al., they are consistent with the approach that was used to
2010; Tomblin, Smith, et al., 1997) reported only adjusted qualify studies for the current review. In particular, only
effect sizes for some or all of the investigated risk factors. one-gate designs with general population samples were
In order to examine the effect of adjustment on the meta- accepted, which, in effect, eliminated small case-control
analysis results, two meta-analyses were performed for each studies that may have yielded large positive effect sizes
of the associated risk categories. The first analysis contained (i.e., those in the lower right quadrant of the funnel
all of the reported effect sizes; the second contained only plot). The new mean OR value (OR = 1.44, 95% CI [1.33,
unadjusted effect sizes. There were no significant differences 1.56]) was larger than the original mean OR value (OR =
between the results of the two approaches; therefore, both 1.34, 95% CI [1.23, 1.46]); however, on the basis of the
adjusted and unadjusted effect sizes were included in all 95% CIs, both values overlapped substantially and were
remaining analyses. not significantly different from one another. Thus, the im-
Publication bias. Every effect size that was eligible pact of publication bias in the current set of studies was
for meta-analysis, whether independently or as part of a considered minimal (Rothstein et al., 2005).
combined effect size, was included in an assessment of pub- Heterogeneity. Heterogeneity statistics for the 14 eli-
lication bias. Publication bias occurs when the sample of gible risk categories were examined. If significant heteroge-
studies included in a meta-analysis is systematically unrep- neity exists among effect sizes extracted from individual
resentative of the range of studies that have investigated studies, it suggests that the association between the risk
the topic (Rothstein, Sutton, & Borenstein, 2005). Such factor and outcome varies substantially and nonrandomly
bias threatens a review’s validity and may lead to incorrect from study to study. As such, the aggregated effect size is
and sometimes detrimental conclusions—for example, that not meaningful. To assess heterogeneity, two statistics,
a particular treatment is clinically useful when it is actually Q and I2, were examined. Q is a chi-square statistic that
not. Results of a meta-analysis must be interpreted with describes the deviation of each individual OR value from
caution if bias is discovered. One approach to publication the mean OR value. The larger the Q value, the smaller

1002 American Journal of Speech-Language Pathology • Vol. 26 • 991–1010 • August 2017


Table 3. Combined effect sizes.

Study Risk category Individual risk factors Combined OR Combined 95% CI

Hammer et al. (2001) Parenting behavior Read to child 2.2 [1.15, 4.22]
Tell stories to child
Discuss daily activities
Routine wake time
Family meals
Stanton-Chapman et al. (2002) Newborn condition 5-min Apgar 1.87 [1.30, 2.70]
Anemia, assisted ventilation
Prenatal exposure Maternal tobacco use 1.2 [1.10, 1.32]
Maternal alcohol use
Tomblin, Smith et al. (1997) Family history Maternal 1.65 [1.06, 2.57]
Paternal
Maternal illness Kidney infection 1.09 [0.41, 2.87]
Thyroid issue
Sexually transmitted disease
Urinary tract infection
Perinatal event Cesarean section 0.85 [0.53, 1.38]
Induced labor
Forceps delivery
Labor duration
Birth complications
Pregnancy condition History of poor outcomes 1.07 [0.58, 1.95]
Gestational diabetes
Hypertension
Prenatal exposure Maternal smoking 1.1 [0.68, 1.78]
Paternal smoking
Maternal drug use
Paternal drug use
Maternal alcohol use
Paternal alcohol use
Occupational exposure
Whitehouse et al. (2014) Maternal illness Preexisting diabetes 0.96 [0.10, 9.24]
Kidney infection
Newborn condition Time to spontaneous respiration 1.72 [0.38, 7.84]
Resuscitation
Birth trauma
1-min Apgar
5-min Apgar
NICU placement
Poor sucking/feeding
Hypoglycemia
Jaundice
Anemia
Temperature maintenance
Perinatal event Breech 0.92 [0.24, 3.54]
Induced labor
Maternal fever
Abnormal fetal heart rate
Prostiglandins
Oxytocin
Narcotic analgesia
Epidural analgesia
Atypical placenta
Atypical umbilical cord
Elective cesarean section
Emergency cesarean section
Forceps/vacuum delivery
Labor duration
Expulsion
Pregnancy condition Maternal BMI > 30 0.97 [0.11, 8.99]
Hyperemesis
Gestational diabetes
Threatened abortion
Hemorrhage
Preeclampsia
Hospital admission (< 20 weeks)
Hospital admission (≥ 20 weeks)
(table continues)

Rudolph: Case History Risk Factors for SLI 1003


Table 3. (Continued).

Study Risk category Individual risk factors Combined OR Combined 95% CI


Prenatal exposure Maternal alcohol use 1.51 [0.45, 5.10]
Maternal smoking

Note. OR = odds ratio; CI = confidence interval; NICU = neonatal intensive care unit; BMI = body mass index.

the p value, and the greater the degree of heterogeneity were eliminated, leaving seven post hoc risk categories for
that exists. Because Q can be less sensitive as an indicator meta-analysis.
of heterogeneity when the number of included studies is
small, a second heterogeneity indicator was also consid- Final Meta-Analyses
ered. I2, or the inconsistency index, describes the percent- The aggregated OR values and 95% CIs for the
age of variation across effect sizes, which is due to systematic 18 risk categories (i.e., 11 original categories and seven
differences across studies as opposed to the variation that is post hoc categories) were calculated. Among the post hoc
due to random sampling error. For the current review, het- categories, 5-min Apgar score, maternal smoking during
erogeneity was considered significant either when p(Q) ≤ .10 pregnancy, and maternal alcohol consumption during
or when I2 > 50. Among the 14 categories that were eligible pregnancy were statistically significant. The original risk
for meta-analysis, significant heterogeneity was observed categories into which these had fallen, newborn condition
for birth weight (Q = 14.11, p = .003, I2 = 78.74), maternal and prenatal chemical exposure, were reanalyzed excluding
age (Q = 76.15, p = .000, I2 = 96.06), and SES (Q = 8.98, these three factors. This resulted in the elimination of prenatal
p = .011, I2 = 77.74). Because significant heterogeneity indi- chemical exposure from meta-analysis as it was represented
cates that these aggregated OR values cannot be interpreted by only one effect size when maternal smoking and maternal
with confidence, these three categories were eliminated, leav- alcohol consumption were treated as independent risk catego-
ing 11 risk categories for further consideration. ries, resulting in 10 original risk categories and seven post
hoc categories (n = 17). Table 4 reports the aggregated effect
Initial Meta-Analyses sizes and heterogeneity statistics following this modification.
Meta-analyses were performed for the 11 qualifying Eleven risk categories were statistically significant: maternal
risk categories using CMA. Random effects models were education level, family history, birth order, biological sex,
used for these and all remaining analyses. Nine of the cate- prematurity, presence of a newborn condition, presence
gories were statistically significant (i.e., 95% CI lower limit of a pregnancy condition, perinatal event, 5-min Apgar
> 1.0). Five out of these nine categories had been repre- score, maternal smoking during pregnancy, and maternal
sented by at least one combined effect size. These five cate- alcohol consumption during pregnancy. These risk catego-
gories were family history of communication or related ries were retained for Phase II of data analysis.
disorders, perinatal event, prenatal chemical exposure, pres-
ence of a newborn condition, and presence of a pregnancy Phase II Data Analysis
condition. This raised the question of whether the signifi-
cance of these categories might actually be due to the signif- To determine the OR value and 95% CI associated
icance of one or more of the individual risk factors that with late talker status, a group of studies from a recent
were combined to create them. To test this possibility, post review of the late talker literature (Dollaghan, 2013), as
hoc meta-analyses of the individual risk factors falling into well as more recently published studies that have examined
these five significant risk categories were performed. the association between early vocabulary skills and pre-
school or school age (i.e., 4 years or older) SLI, were iden-
tified (Ellis Weismer, 2007; Grimm & Schulz, 2014; Paul,
Post Hoc Risk Categories 2000; Poll & Miller, 2013; Rice, Taylor, & Zubrick, 2008;
Because at least two effect sizes are required to per- Rudolph & Leonard, 2016). An aggregated effect size for
form meta-analysis, post hoc meta-analyses could be com- late talker status was calculated using CMA. This yielded
pleted only for the nine individual risk factors that were an OR value of 2.29 and a 95% CI of [1.42, 3.70]. The ef-
examined in more than one study. These were 5-min Apgar fect sizes across the included studies were homogeneous
score, birth complications, cesarean section, delivery by (Q = 6.04, p = .302, I2 = 17.26), which indicates that the
forceps, gestational diabetes, induced labor, labor dura- aggregated OR value is a valid representation of the range
tion, maternal alcohol consumption during pregnancy, and of values associated with late talker status. Given these
maternal smoking during pregnancy. These will hereafter results, risk categories were considered to be clinically sig-
be referred to as post hoc risk categories. Heterogeneity nificant predictors of SLI if the lower limits of their associ-
statistics revealed significant heterogeneity for birth com- ated 95% CIs were ≥ 1.42. A forest plot was created for
plications (Q = 3.127, p = .077, I2 = 68.02) and labor dura- the 11 significant risk categories from Phase I (see Figure 4),
tion (Q = 2.90, p = .089, I2 = 65.48). These categories which were organized according to the magnitude of the

1004 American Journal of Speech-Language Pathology • Vol. 26 • 991–1010 • August 2017


Table 4. Aggregated odds ratios (ORs), confidence intervals (CIs), and heterogeneity statistics for original and post hoc risk categories.

Effect size Heterogeneity


Category N OR 95% CI Z p (Z ) Q df p (Q) I2

Risk
Maternal education 116,205 2.12 [1.95, 2.31] 17.22 0.000 3.04 3 0.386 1.26
Family history 2,308 1.77 [1.30, 2.40] 3.66 0.000 0.18 1 0.675 0.00
Birth order 162,380 1.70 [1.57, 1.86] 12.29 0.000 0.11 1 0.740 0.00
Biological sex 212,893 1.64 [1.43, 1.89] 7.13 0.000 5.00 4 0.288 19.96
Prematurity 201,939 1.49 [1.34, 1.67] 7.18 0.000 0.55 1 0.460 0.00
Newborn conditiona 195,389 1.41 [1.13, 1.74] 3.09 0.002 0.05 1 0.819 0.00
Parent marital status 195,375 1.40 [0.58, 3.34] 0.75 0.453 1.96 1 0.161 48.99
Pregnancy condition 196,491 1.20 [1.13, 1.27] 5.82 0.000 0.17 2 0.918 0.00
Perinatal event 196,477 1.09 [1.02, 1.17] 2.66 0.008 1.16 2 0.560 0.00
Maternal illness 2,912 1.07 [0.44, 2.16] 0.15 0.884 0.01 1 0.920 0.00
Post hoc risk
5-min Apgar 193,946 2.44 [1.59, 3.72] 4.11 0.000 0.06 1 0.800 0.00
Maternal smoking 196,384 1.28 [1.07, 1.52] 2.74 0.006 2.71 2 0.258 26.12
Maternal alcohol use 196,226 1.18 [1.05, 1.32] 2.84 0.005 1.15 2 0.563 0.00
Induced labor 2,711 1.02 [0.68, 1.52] 0.08 0.937 0.02 1 0.887 0.00
Gestational diabetes 2,911 1.00 [0.49, 2.04] −0.01 0.996 0.00 1 0.984 0.00
Cesarean section 2,913 0.72 [0.50, 1.06] −1.68 0.093 0.33 1 0.565 0.00
Forceps delivery 2,674 0.48 [0.28, 0.84] −2.57 0.010 0.08 1 0.777 0.00

Note. OR = odds ratio; CI = confidence interval. Significant factors (95% CI lower limit > 1.0) are bolded. Among the original 14 risk
categories, birth weight, maternal age, and socioeconomic status are not included due to significant heterogeneity. In addition, prenatal
chemical exposure is not included because it was no longer eligible for meta-analysis after maternal alcohol use and maternal smoking were
analyzed as independent risk factors. Among the nine post hoc risk factors, birth complications and labor duration are not included due to
significant heterogeneity.
a
Does not include 5-min Apgar score.

lower limit of their 95% CIs. The boundary of clinical sig- as the odds of developing SLI among children who are
nificance was demarcated with a dashed line. Maternal edu- late talkers. Of course, this does not imply that all children
cation level, 5-min Apgar score, birth order, and biological who fall into one of these risk categories should be imme-
sex fell entirely within the boundary of clinical significance. diately referred for further evaluation and services; none-
Family history and prematurity fell almost entirely within theless, this review brings clarity to the question of where
the boundary. Newborn condition and maternal smoking early identification efforts should be directed.
fell partially within the boundary. Pregnancy condition,
maternal alcohol consumption, and perinatal event all fell
entirely outside the boundary of clinical significance. Sources of Heterogeneity
Previous reviews have attempted to develop lists of
risk factors to guide professional decision making in the
Discussion context of early speech and language screening (Berkman
It is well established that early language performance et al., 2015; Nelson et al., 2006), but have been unable to
alone is insufficient to identify all of the children who will accomplish this goal because of the notable degree of hetero-
go on to suffer from severe and chronic difficulties with geneity encountered across the sets of studies examined. The
language (Dollaghan, 2013; Leonard, 2013). As noted in current systematic review differs from previous reviews in
previous studies (Reilly et al., 2010; Rudolph & Leonard, that several carefully defined exclusionary criteria were
2016), consideration of genetic and environmental factors applied to identify a cluster of studies that were likely to
may bolster the predictive accuracy of early behavioral yield homogeneous outcomes, thereby allowing for statisti-
indicators. The purpose of this systematic review was to cal aggregation of results. Although homogeneity was
synthesize the available data on case history risk factors achieved for the majority of factors, this was not the case
for SLI and to examine whether specific factors could be for birth weight, birth complications, labor duration, mater-
used to identify toddlers who are most at risk of develop- nal age, and SES. The observed heterogeneity was unlikely
ing the disorder. Eleven risk factors emerged as statistically to be due to differences in diagnosis, sample size, sampling
significant predictors of SLI, and four of these met cri- method, participant ages, or analyses used. Rather, the
teria for clinical significance. These results suggest that the heterogeneity was probably the result of discrepancies in
odds of developing SLI among children whose mothers how these particular risk factors were defined. In some cases,
have less than a high school degree or who are later born, studies assigned nearly opposing meanings to a given factor.
male, or scored very low on their 5-min Apgar are as high In other cases, differences in risk factor representation—that

Rudolph: Case History Risk Factors for SLI 1005


Figure 4. Forest plot identifying clinically significant risk factors. The dashed line indicates the lower limit for clinical significance (OR > 1.42).
Factors that fall entirely within the shaded area are clinically significant.

is, whether it was characterized dichotomously, catego- of clinical significance, indicating that these events are
rically, or continuously—contributed to the observed unlikely to be useful predictors in a clinical context.
disparities.
Differing Characterizations
Birth weight was defined continuously in one study,
Opposing Meanings categorically in a second, and dichotomously in two others.
Labor duration was defined as < 2 hr in one study Among categorical and dichotomous representations of
and as > 10 hr in another. The risks associated with a this factor, the limit of interest differed from very low birth
short labor are likely to be very different from the risks as- weight (< 1,500 g) to low birth weight (< 2,500 g) to small
sociated with a long labor, which could account for the for gestational age. The effect size associated with very low
variability in effect sizes for this risk factor. Neither study birth weight was high (OR = 2.20, 95% CI [1.80, 2.80]), but
found labor duration to be a significant risk factor for SLI, the other effect sizes were not significant. SES, in contrast,
and further exploration is not likely to yield different out- was defined continuously in two studies and dichotomously
comes. In a similar manner, maternal age was defined as in a third. The first two studies found this risk factor to be
< 18 years in one study and > 35 years in another. Again, significantly associated with SLI, whereas the last study did
the risks associated with having a very young mother are not. Further consideration of birth weight (categorically
likely to be quite different from the risks associated with defined) and SES (continuously defined) may be warranted.
having an older mother. In this case, the former study
found a significant association (OR = 1.60, 95% CI [1.50,
1.80]), suggesting that having a young mother may be a Clinical Significance
statistically and even clinically significant predictor of SLI, The 11 statistically significant factors identified in
whereas having an older mother is not. Studies examining this review include presence of a pregnancy condition, ma-
birth complications were, likewise, on opposite sides of ternal smoking during pregnancy, maternal alcohol con-
the spectrum. One study subsumed every sort of possible sumption during pregnancy, perinatal event, prematurity,
birth complication under this category (e.g., placenta pre- presence of a newborn condition, 5-min Apgar score, bio-
via, breech presentation, fetal distress, etc.), whereas birth logical sex, maternal education level, family history, and
complication was an exclusive category in the second study birth order. This suggests that a variety of factors spanning
and was used to characterize situations that did not involve the range from the earliest stages of fetal development to
cesarean section or labor induction. This factor was sig- the latest stages of language acquisition may have an im-
nificant in the first study, but not in the second, suggesting pact on a child’s language outcomes. Note, however, that
that some types of birth complications may, in fact, be only four out of the 11 factors met criteria for clinical
associated with SLI, but not others. Indeed, the risk cate- significance, indicating that not all of these predictors are
gory perinatal event was a statistically significant predictor reliable enough to be useful within the context of an SLI
of SLI; however, the category fell entirely below the boundary early identification screening protocol.

1006 American Journal of Speech-Language Pathology • Vol. 26 • 991–1010 • August 2017


The four factors that fell entirely within the range prospectively to eliminate bias associated with memory
of clinical significance, as well as the two factors that fell and outcome awareness; focused attempts should be
almost entirely within this range, could be quickly and eas- made to limit attrition of at risk populations; and, finally,
ily obtained through a parent questionnaire or review of the sample obtained should be assessed for population
a child’s medical chart. Furthermore, all of these risk fac- levels of prevalence to ensure that all children, and not
tors could be identified as early as the day a child is born. only those at the extremes, are represented and correctly
However, when applying the information from this review classified.
in a clinical or research setting, there are three consider-
ations that must be kept in mind. The first is that the four
clinically significant risk factors identified here are only Limitations
as informative in an early identification context as late This review is limited by the following internal con-
talker status and should not be given any more or any less straints. First, the primary statistical focus was unadjusted
predictive weight. Recall that late talker status, although OR values. This approach was taken because adjusted
widely associated with language impairment by pediatric values might provide inaccurate estimates of association in
professionals, childcare providers, and parents, is not a cases where causal mediators are treated as confounding
strong predictor of SLI (Dollaghan, 2013). Neither is any variables. Nonetheless, unadjusted values might also pro-
one of these risk factors by itself a strong predictor of SLI. vide inaccurate estimates of association if true confounding
Thus, making diagnostic decisions on the basis of a single variables do exist. Thus, it is possible that the values re-
factor or characteristic is never recommended. The second ported in this review are inflated. Note, however, that the
consideration is that not all risk factors should be weighted comparison standard, late talker status, was also repre-
equally. We do not yet know how much predictive power sented by an unadjusted OR value. The comparison is
to assign to the factors individually versus in different com- therefore equivalent, and the main results of the review are
binations. For example, what are the odds of developing unaffected by this limitation. An additional internal con-
SLI for a male child (biological sex) whose father has dys- straint involves the fact that the meta-analysis phase was
lexia (family history) and who is the fourth born in his confined to those risk factors that had been investigated in
family (birth order)? How does this child’s level of risk two or more studies. Among the 11 risk categories that
compare with that of another child with only one or two could not be included in meta-analysis, five met criteria for
of these risk factors? How does it compare with that of a clinical significance within the individual studies that inves-
child with a completely different set of risk factors? Fur- tigated them. These were childhood chemical exposure,
ther research is needed to answer these questions and to being formula fed, minority status, paternal education
provide a validated guide for clinical decision making. The level, and onset of prenatal care. It is possible that one or
final consideration, which is closely related to the previous more of these factors might be an important consideration
two, is that children’s early language skills should never in the process of determining an individual child’s level of
be considered in isolation from their language learning risk. A third limitation of this study was that aggregated
context. As evidenced in this review, there are a host of scores could not be reported for birth weight, birth com-
biological, genetic, and environmental factors that are as- plications, maternal age, SES, and labor duration as a result
sociated with and might contribute to a child’s language of significant heterogeneity. Results of individual studies
development (Rogers et al., 2015); every piece of the puzzle indicate that at least two and possibly three of these factors
is important when determining which children are most may be worthy of further investigation (see Sources of
likely to exhibit poor language outcomes. Heterogeneity above). Last, this review focused on studies
that compared children with SLI to children without lan-
guage impairment. The ability of the examined risk factors
Future Research to differentiate between children with different types of
The results of the study quality assessment suggest developmental disorders is unknown, although some stud-
particular considerations for research groups interested in ies have begun to explore this topic (Kim, Jeon, Park,
further exploration of SLI risk factors. The studies in the Chung, & Song, 2014).
current review, with few exceptions, provide excellent The review is also externally constrained by limita-
examples of high-quality research in the domains of par- tions of the individual studies. In particular, approximately
ticipant selection, risk factor collection, and assignment 75% of the studies suffered from risk of bias in the domain
of diagnostic status. However, retrospective reporting and of participant flow, and applicability concerns were ob-
high levels of systematic attrition could have compromised served for 35% of the participant samples. As such, it is
the internal validity of several studies and led to the wider possible that the populations examined in these studies
CIs obtained for some of the key risk factors. Further- were not truly representative, and that findings of this re-
more, remarkably high or low proportions of children with view do not accurately reflect the associations between case
SLI could have threatened the external validity of a few history risk factors and SLI as they exist in the general
investigations insofar as pockets of children with or with- population. In addition, although all of the children with
out the disorder might have been missed. Thus, in future SLI met basic criteria for the disorder, the types of diag-
research, risk factor information should be collected nostic procedures used and the stringency of exclusionary

Rudolph: Case History Risk Factors for SLI 1007


criteria varied across individual studies. Thus, a child who Bossuyt, P. M., & Leeflang, M. M. (2008). Developing criteria for
met criteria for SLI in one study may not have qualified as including studies. In J. J. Deeks, P. M. Bossuyt, & C. Gatsonis
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used to assign children to diagnostic categories might have clinical, and personal significance: Definitions and applications
worked well for monolingual mainstream English-speaking in speech-language pathology. American Journal of Speech-
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inherent in the assessment instruments that were used. Only https://doi.org/10.1044/jslhr.4005.1011
two studies reported information on race/ethnicity, and none Campbell, T. F., Dollaghan, C. A., Rockette, H. E., Paradise, J. L.,
of the studies reported using procedures that were sensitive Feldman, H. M., Shriberg, L. D., . . . Kurs-Lasky, M. (2003).
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1010 American Journal of Speech-Language Pathology • Vol. 26 • 991–1010 • August 2017


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