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Journal of Speech, Language, and Hearing Research : JSLHR logoLink to Journal of Speech, Language, and Hearing Research : JSLHR
. 2020 Dec 29;64(1):218–229. doi: 10.1044/2020_JSLHR-20-00342

Family-Level Executive Functioning and At-Risk Pediatric Hearing Loss Outcomes

Andrew Blank a,✉, Rachael Frush Holt a, David B Pisoni b,c, William G Kronenberger c,d
PMCID: PMC8608142  PMID: 33375824

Abstract

Purpose

Using a new measure of family-level executive functioning (EF; the Family Characteristics Scale [FCS]), we investigated associations between family-level EF, spoken language, and neurocognitive skills in children with hearing loss (HL), compared to children with normal hearing.

Method

Parents of children with HL (n = 61) or children with normal hearing (n = 65) completed the FCS-Parent, and clinicians evaluated families using the FCS-Examiner. Children completed an age-appropriate version of the Concepts and Following Directions subtest of the Clinical Evaluation of Language Fundamentals and the Peabody Picture Vocabulary Test–Fourth Edition. Child EF was assessed via the parent report Behavior Rating Inventory of Executive Function.

Results

Two higher order components were derived from FCS subscales: Family Inhibition and Family Organization. For both samples, Family Inhibition was positively associated with child inhibition, child shifting, and child language comprehension skills. Family Organization was differentially associated with child inhibition, working memory, and planning/organization skills across the samples. Additionally, Family Inhibition was associated with child planning and organization skills for children with HL.

Conclusions

Results support the FCS as a measure of family-level EF. Family-level inhibition related to better child inhibition, flexibility/shifting, and language comprehension across both samples and to better planning and organization skills in children with HL. As children with HL experienced greater difficulties in EF, families demonstrated greater organization, possibly as a compensatory measure. Results suggest that inhibition and organization at a family level may be important targets for the development of novel interventions to promote EF and language outcomes for children with HL.

Considerable unexplained variability in spoken language and neurocognitive outcomes exists for children with hearing aids (HAs; Khan et al., 2005; Stiles et al., 2012) and cochlear implants (CIs; Beer et al., 2014; Niparko et al., 2010). Outcomes research on spoken language and neurocognitive skills of children with hearing loss (HL) has evaluated how audiological, medical, and parenting characteristics contribute to individual differences in these at-risk developmental domains. Indeed, studies examining these contributing factors have revealed important processes that underlie spoken language and neurocognitive development for this clinical population. However, traditional approaches to examining pediatric HL outcomes treat children as “closed-loop” systems largely existing in isolation from the influences of and experiences with their environment (see von Bertalanffy, 1968, for a review of general systems theory). When environmental experiences are considered, research has generally investigated the contributions of individual parental and parenting attributes (typically deriving from mothers). Thus, conventional approaches to outcomes research have yet to integrate the entire scope of a child's environmental experiences as factors that contribute to variability in at-risk spoken language and neurocognitive skills.

Emerging evidence suggests that whole-family factors account for a significant proportion of outcomes variability for this pediatric population (Holt et al., 2013, 2012, 2019). However, the role of family-level functional and organizational dimensions remains largely unexplored. In particular, the role of family-level environmental factors in shaping the development of child executive functioning (EF; cognitive, behavioral, and emotional self-regulation in the service of achieving goals) has received little investigation to date, despite evidence that child EF is at risk for delay in a significant subgroup of children with HL (Kronenberger & Pisoni, 2018). In contrast to individual EF skills, family-level EF reflects the shared modeling, teaching, and behavioral regulation of EF skills and resources in the family environment. Given the enormous influence of modeling, teaching, and reinforcement on child development, it is likely that these family-level EF influences may have a profound impact on child EF development and on the development of other neurocognitive and spoken language skills. Thus, the purpose of this investigation was to investigate differential associations between family-level EF and at-risk spoken language and neurocognitive skills in children with HL, compared to children with normal hearing (NH).

Ecological Perspectives on Development

Child development occurs in the context of environmental experiences and processes (Bradley et al., 1979, 1988). From family systems and ecological systems perspectives of development, children are situated within multiple open-loop ecosystems, ranging from proximally oriented (e.g., home, school) to more distally oriented (e.g., local community, media) microsystems. Proximal influences pertain to a child's interactions and experiences with their immediate environment (Bronfenbrenner, 1979; Bronfenbrenner & Ceci, 1994). The direct and indirect influences of individual ecosystems change over time, but the most proximal and most impactful ecosystem for child development is the family (Belsky, 1981, 2005; Bronfenbrenner, 1977; Kazak, 1992; Sameroff, 2010). Although often accounted for in the developmental sciences, investigating the structural and functional aspects of the primary ecosystems of children with HL offers a novel approach to inform the hearing sciences of how family dynamics and processes impact at-risk outcomes.

The social–behavioral risk (S-B-R) model of pediatric HL proposed by Holt et al. (2019) draws heavily on ecological and systems perspectives on child development to explain variability in pediatric HL outcomes. The S-B-R model recognizes that development occurs within the context of multiple ecosystems (Bronfenbrenner, 1977) and that experiences and activities between children and family members are transactional (Sameroff, 1975, 2010). Most children with HL (96%) have parents with typical hearing (Mitchell & Karchmer, 2004). Thus, a communication mismatch exists in hearing families of children with HL, challenging the organization of family–child interactions (Moeller & Tomblin, 2015). Child language learning most often occurs through proximal parent–child interactions, but pediatric HL interferes with language-learning mechanisms. Thus, pediatric HL introduces greater risk for poorer outcomes in specific domains of development, particularly spoken language and neurocognition (Cruz et al., 2013; Geers et al., 2011; Kronenberger & Pisoni, 2018; Kronenberger et al., 2013; Niparko et al., 2010).

Earlier work on relations between pediatric hearing outcomes and proximal environmental dimensions has identified several family-level influences on spoken language and neurocognitive abilities. Using a sample of children with CIs, Holt et al. (2012) found that children with higher receptive vocabulary had families reporting fewer controlling and inflexible behaviors among family members. Additionally, children exhibiting fewer difficulties with at-risk neurocognitive skills also had families emphasizing greater organizational structure and achievement. In a follow-up study, Holt et al. (2013) found that dimensions of the family environment differed between preschool- and school-age children with CIs and differentially contributed to language and neurocognitive abilities. Using a combined sample of children with HAs and CIs and a control sample of children with NH, Holt et al. (2019) compared parental reports of family environment between the two groups. Although dimensions of the family environment were broadly similar between the groups, dimensions of family environment contributed differently to spoken language versus neurocognitive skills in the CI sample compared to the control sample.

The studies described in the previous paragraph investigated broad, social climate characteristics of the family, such as relationship dimensions (e.g., family cohesion or conflict), personal growth dimensions (e.g., achievement, intellectual–cultural, active–recreational, and moral–religious orientations), and system maintenance dimensions (e.g., family organization and control), using the parent report Family Environment Scale (FES; Moos & Moos, 2009). However, whereas the family dimensions assessed by the FES characterize important components of family climate, other family dimensions not captured by the FES may be specifically relevant for development of spoken language and neurocognitive skills in children with HL. As a result, it is still unclear how family dimensions pertaining directly to shaping and teaching language and neurocognitive development in the families of children with HL might contribute to their outcomes in these at-risk domains. Investigating the dimensions of family functioning that reflect and model specific at-risk neurocognitive skills through social learning experiences could provide additional insight into how the family environment contributes to individual differences in foundational pediatric HL outcomes, including language and neurocognitive development.

Family-Level Executive Function and Child Development

Consistent with ecological and transactional perspectives on child development, early environmental and familial experiences contribute to brain development (Gunnar et al., 2006; Nelson, 2000). Of note, EF comprises a suite of higher order neurocognitive processes that underpin goal-oriented behavior and that emerge with protracted maturational changes occurring in the prefrontal cortex (Gogtay et al., 2004; Moriguchi & Hiraki, 2011). The family environment can influence this early neurocognitive development by acting as a teacher and regulator of emerging self-regulatory skills (Bernier et al., 2010; Clark et al., 2013; Hofer, 1995; Kopp, 1982).

Family-level EF represents the components of the family system that model and encourage shared regulation and organization among family members. Higher levels of structured, goal-directed family behaviors (e.g., following planned schedules, collective behavioral control) are associated with better child EF and self-regulation skills in nonclinical pediatric populations (McCarty et al., 2005; Schroeder & Kelley, 2010). Additionally, families employing consistent and predictable routines in which members systematically follow-through with daily and weekly activities beneficially impact child EF skills (Adam et al., 2007; Barker et al., 2014). Conversely, chaotic and unpredictable environments or inflexibly structured daily schedules are associated with poorer child EF (Barker et al., 2014; Vernon-Feagans et al. 2016). Children with HL are at greater risk for weaknesses in EF following language delay, but families that demonstrate well-controlled behaviors and maintain organized home environments and schedules could represent proximal sources of external regulation to scaffold language and neurocognitive development for this pediatric population.

This Study

Previous work on family-level dimensions used the FES, a parent report measure of important aspects of the home environment that does not target EF specifically (Holt et al., 2013, 2012, 2019). This study extends that previous body of work by introducing the Family Characteristics Scale (FCS), a parent report measure that assesses specific dimensions of family-level EF. The purpose of this investigation was to examine the extent to which family-level EF is associated with spoken language and neurocognitive skills in children with HL relative to children with NH.

Method

Participants

Children, ages 3–8 years (inclusive), and their parents participated in the Families and Hearing Study, a longitudinal project examining family and environmental contributions to pediatric HL outcomes (e.g., Blank et al., 2020; Holt et al., 2019). The present analyses comprise data collected at the first assessment. In total, 61 families of children with HL and a control group of 65 families of children with NH provided data at this time point. Regardless of hearing status, all children had English-speaking caregivers and received educational instruction in spoken English. All children scored 2 SDs (T score > 30) below the mean or higher on a subtest measuring nonverbal intelligence (the Picture Similarities subtest of the Differential Ability Scales–Second Edition [DAS-II; Elliot, 2007]). Children with HL were recruited from local medical centers, were referred from local or state-wide service providers, or were from answered local advertisements.

Families of 27 children fit with HAs with mild-to-severe sensorineural HL (M age = 6.38 years, SD = 1.71 years) and families of 34 children fit with CIs with severe-to-profound sensorineural HL (M age = 6.36 years, SD = 1.53 years) comprised the HL group. Children in this group were diagnosed with bilateral sensorineural HL and began intervention by 2 years of age. Children with CIs were implanted by 3.5 years of age and had at least 6 months of experience with their devices at the time the study was carried out. Children with HL and their families were excluded from participation if the child was diagnosed with auditory neuropathy spectrum disorder, if the child had other neurodevelopmental disabilities or delays not closely associated with HL or if HL diagnosis and first HA fitting occurred after the age of 2 years and/or cochlear implantation occurred after 3.5 years of age. One primary caregiver was enrolled per family, including 58 mothers and three fathers of children with HL.

A control group of 65 NH children (M age = 5.78 years, SD = 1.63 years) and their families were also enrolled. Parents of NH children reported no concerns regarding their child's speech, language, motor, sensory, or cognitive development. All NH children passed a behavioral hearing screening bilaterally at 25 dB HL at 500, 1000, 2000, and 4000 Hz (American National Standards Institute, 2004, 2010) at the time of assessment. One primary caregiver was enrolled per family, including 61 mothers and four fathers of NH children.

Table 1 displays a summary of the demographic characteristics of the two samples. Independent-samples t tests and Pearson chi-square analyses were carried out to compare the demographic characteristics of the two groups. Children with HL were significantly older than children with NH, t(124) = −2.05, Cohen's d = −0.36, p = .042. However, there were no significant differences between groups in annual family income, Mann–Whitney U = 816.00, η2 = .004, p = .566; parent education level, Mann–Whitney U = 700.50, η2 = .03, p = .111; child gender, χ2(1) = 1.13, p = .287; number of children in the household, t(124) = 0.05, η2 = .001, p = .962; and number of household family members, t(124) = −0.34, η2 = −.06, p = .736.

Table 1.

Family and child demographics and hearing history.

Participant characteristics NH HL (CI & HA) CI HA
n 65 61 34 27
No. of male/female 36/29 28/33 16/18 12/15
No. of 2-parent/1-parent households 60/5 57/4 33/1 24/3
No. of children in household a 2.37 (1.0) 2.36 (1.0) 2.32 (1.1) 2.41 (0.9)
Household size 4.29 (1.1) 4.36 (1.2) 4.38 (1.2) 4.33 (1.1)
Average caregiver education b 5.15 (1.2) 4.39 (1.4) 4.26 (1.5) 4.56 (1.4)
Average annual family income c 8.86 (1.5) 7.90 (2.5) 7.41 (2.8) 8.52 (2.1)
Chronological age, child (years) 5.78* (1.6) 6.37* (1.6) 6.36 (1.5) 6.38 (1.7)
Age range, child (years) 3.01–8.74 3.52–8.97 3.52–8.97 3.77–8.94
Audiological characteristics
 Hearing age (years) d NA 5.08 (1.8) 4.76 (1.7) 5.49 (1.8)
 Aided 4-frequency PTA e (dB HL) NA 23.83 (6.4) 24.97 (3.7) 21.00 (10.3)
 Unaided 4-frequency PTA e , f (dB HL) NA 72.83 (26.9) 94.00** (12.9) 46.79** (12.2)
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Note. NH = normal hearing; HL = hearing loss; CI = cochlear implant; HA = hearing aid; PTA = pure-tone average re: American National Standards Institute (2004); NA = not applicable.

a

Includes child enrolled in study and their siblings living in the household.

b

Parental education was coded based on levels of formal education: 1 = some high school, 2 = high school diploma, 3 = some college, 4 = associate degree, 5 = bachelor's degree, 6 = master's degree, 7 = doctorate degree.

c

Parents indicated their annual income from the following income brackets: 1 = under $5,000, 2 = $5,000–$9,999, 3 = $10,000–$14,999, 4 = $15,000–$24,999, 5 = $25,000–$34,999, 6 = $35,000–$49,999, 7 = $50,000–$64,999, 8 = $65,000–$79,999, 9 = $80,000–$94,999, 10 = $95,000 and over.

d

Hearing age was calculated by subtracting chronological age at time of assessment from age at which the child was fit with hearing aids (HA group) or fit with cochlear implants (CI group).

e

Calculated at 0.5, 1, 2, and 4 kHz.

f

At time of HL diagnosis.

*

p < .05.

**

p < .01.

Analyses were also conducted to compare characteristics of the HA and CI groups. Although the HA and CI groups differed on pure-tone average (average auditory thresholds for the better ear at 500, 1000, 2000, and 4000 Hz) at the time of HL diagnosis, t(27) = 9.99, Cohen's d = 3.74, p < .0001, there were no significant differences between the HA and CI groups on annual family income, Mann–Whitney U = 356.00, η2 = .04, p = .120; parent education level, Mann–Whitney U = 398.50, η2 = .01, p = .364; child age, t(59) = 0.59, Cohen's d = 0.015, p = .950; aided pure-tone average at most recent evaluation, t(33) = 1.70, Cohen's d = 0.511, p = .100; child gender, χ2(1) = 0.04, p = .840; number of children in the household, t(59) = −0.32, η2 = −.08, p = .752; and number of household family members, t(59) = −0.16, η2 = .04, p = .871. Therefore, we combined the HA and CI groups into a single HL group to increase statistical power.

Materials

Family Questionnaires

Family-level executive function

Parents completed the FCS-Parent, a 28-item parent report survey evaluating seven total dimensions of the family system with four items per dimension (developed for this study by the last author). The FCS-Parent was designed to assess six dimensions of family-level EF: (a) Family Organization in Time (Cronbach's α = .82), (b) Family Organization in Space (Cronbach's α = .89), (c) Family Inhibition (Cronbach's α = .83), (d) Family Flexibility (Cronbach's α = .80), (e) Family Focus (Cronbach's α = .79), and (f) Family Emotional Control (Cronbach's α = .93). A seventh dimension unrelated to family-level EF, Family Support, also contributes to the FCS-Parent (Cronbach's α = .9). Items belonging to Family Organization in Time evaluate planning, expectations, and regularity of daily activities within the family unit (example item: Family members plan their day carefully). The Family Organization in Space dimension assesses the degree to which living and working spaces are kept organized and well maintained (example item: We keep things in their places at home). Family Inhibition items query how family members demonstrate thoughtful/controlled (vs. impulsive) behaviors when acting or making decisions (example item: Family members tend to act first and think later [note that this item is reverse-scored]). Items belonging to Family Flexibility assess the degree to which family members adapt to unexpected changes in plans or their environment (example item: Family members cope well with unexpected changes). Family Focus items evaluate how well family members sustain attention and resist distraction during daily activities (example item: Family members are distracted away from their work). Items related to Family Emotional Control measure the degree to which family members regulate emotions and react to emotionally provocative stimuli (example item: Family members have trouble controlling their emotions [note that this item is reverse-scored]). Family Support, a seventh FCS-Parent scale that assesses how family members demonstrate care and affection for each other, was excluded from analyses because it is not an EF-related dimension (example item: We care about each other in our family). Parents were asked to consider the extent to which each item described the overall function or environment of their family. Items were evaluated on a 5-point Likert scale (1 = very untrue of our family [we are almost never like this], 2 = not true of our family [we are typically not like this], 3 = neither true nor untrue of our family [we are sometimes like this and sometimes not like this], 4 = true of our family [we are typically like this], 5 = very true of our family [we are almost always like this]) and were reverse-scored if necessary, such that higher ratings indicated better family-level EF.

The FCS-Parent was developed by the senior author of the study based on research and clinical experience in using assessment measures of the family environment and executive function. The goal for the scale's development was to create a brief, parent report questionnaire that would provide information about family characteristics relevant to EF. To keep the scale as brief as possible (e.g., under 30 items and thus requiring less than 5 min to complete), a preliminary set of 28 FCS-Parent items (seven subscales, with four items per subscale) was identified. Items were presented to a team consisting of two psychologists, two speech-language pathologists, and one research coordinator, all of whom have extensive experience working with families of children with HL and weaknesses in EF. FCS-Parent items, instructions, and rating scales were refined prior to the administration in questionnaire format in the current research study, but the same structure of four items for each of the seven subscales was retained for the Families and Hearing Study. Given the strong internal consistency for all FCS subscales and the brevity of the FCS, no items were excluded from the final scale. The FCS-Parent is available from the last author upon request.

Trained clinical researchers interviewed the parent in the family's home and completed an examiner-rated version of the FCS-Examiner. The FCS-Examiner evaluates the same seven dimensions as of the FCS-Parent. Examiners use a subset of open-ended questions and observations of family members and the home environment to provide a rating of each dimension of family-level EF. Using interview responses to queries (e.g., Family Organization in Time: “Do family members talk or review their plans and schedules? When and how often?”) and observations (e.g., Family Focus: Do family members seem to have problems sustaining attention to tasks), the examiner assigns a single numerical rating for each EF dimension based on specific criteria from a coding manual that is available from the last author upon request, using the a single rating from the same 1–5 scale as the FCS-Parent. Higher ratings reflect greater family-level EF. As with the FCS-Parent, the Family Support subscale is also represented on the FCS-Examiner but was excluded from further analysis because it is not an EF-specific subscale.

The FCS-Examiner was developed in the months following the start of the Families and Hearing Study. Members of our research team involved on home visits indicated that they could provide additional information about family-level EF based on observations made during their visits. The FCS-Examiner was developed with the FCS-Parent questionnaire content, constructs, and rating scale to assess family-level EF from the perspective of trained researchers. Specific open-ended questions and observations were identified for each dimension, and specific criteria were developed for the numeric value of the rating scale (1–5). Following the creation of the coding manual and team-based review of the examiner items, the last author held training sessions at both study sites. Because the FCS-Examiner was developed after the start of the Families and Hearing Study, not all families received FCS-Examiner scores. There were 33 families of children with HL (n = 14 families of children with HA, n = 19 families of children with CI) and 40 families of children with NH with FCS-Examiner ratings.

Child Assessments

Child executive function

Parents of children between 3;0 and 5;11 (years;months) completed the Behavior Rating Inventory of Executive Function–Preschool Version (BRIEF-P; Gioia et al., 2003), a 64-item parent report measure evaluating everyday EF behaviors for preschool-age children. For children 6;0 and older, parents completed the Behavior Rating Inventory of Executive Function–Second Edition (BRIEF2; Gioia et al., 2015), an 86-item parent report measure assessing real-world EF behaviors of school-age children. Subscales common to both the BRIEF-P and the BRIEF2 are Inhibit, Working Memory, Plan/Organize, Shift, and Emotional Control. BRIEF-P/BRIEF2 subscale scores are converted to T scores using age- and gender-specific norms. Higher T scores indicate greater dysfunction for a given EF domain.

Receptive vocabulary

Receptive vocabulary skills were assessed using the Peabody Picture Vocabulary Test–Fourth Edition (PPVT-4; Dunn & Dunn, 2007). The PPVT-4 is a norm-referenced and psychometrically robust assessment of single-word receptive vocabulary.

Language comprehension

Children ages 5;11 and younger were administered the Concepts and Following Directions (C&FD) subtest of the Clinical Evaluation of Language Fundamentals Preschool–Second Edition (CELF Preschool-2; Wiig et al., 2004). Children ages 6;0 and older were administered the Following Directions subtest of the Clinical Evaluation of Language Fundamentals–Fifth Edition (CELF-5; Semel et al., 2013). Both the Following Directions (CELF-5) and C&FD (CELF Preschool-2) subtests evaluate the comprehension of spoken utterances that progressively increase in linguistic complexity. One child with HAs did not complete the CELF Preschool-2 C&FD subtest due to testing fatigue.

Procedure

Parents completed research questionnaires (including the BRIEF2/BRIEF-P and the FCS-Parent) prior to a scheduled home visit. All in-person testing (including the FCS-Examiner, DAS-II, PPVT-4, and CELF-5/CELF Preschool-2) was completed at the home visit by a pair of trained clinical researchers with extensive backgrounds in speech-language pathology, audiology, and/or pediatric HL. Home visits were conducted at a time convenient for the parent and child enrolled in the study (e.g., after school, weekends) and generally took 2–3 hr to complete. The clinical researchers observed parent–child behaviors, viewed and queried aspects of the home environment (e.g., organizational status of common and living areas; presence or absence of toys, books, and common household items), and interviewed parents using inventories that quantify dimensions of the home environment and the family system (e.g., FCS-Examiner, Home Observation for Measurement of the Environment; Caldwell & Bradley, 2003). All data collection was obtained in accordance with procedures of the local institutional review board, and consent was obtained prior to study participation.

Data Analysis Strategy

First, the FCS-Parent and FCS-Examiner were analyzed using principal components analysis (PCA) to determine higher-order components of family-level EF. Next, multiple linear regression estimating individual child EF and spoken language skills was performed in two blocks. The first regression block was conducted using forced variable entry to estimate child outcomes from child hearing status and the identified higher-order components of family-level EF from the FCS. A second regression block was conducted using stepwise entry to determine the presence of differential associations between family-level EF and child outcomes based on hearing status using product variables. The inclusion of interactions (e.g., the product variable of hearing status × FCS component score) was only added to the first block regression model if significant (p < .05).

Results

PCA of FCS

PCAs with Promax rotation were performed to extract higher order components for family-level EF from the subscales of the parent and examiner versions of the FCS. Higher order components were extracted based on eigenvalues approximately > 1 as well as inspection of scree plots. An initial set of PCA analyses were performed with the six FCS EF subscales. However, because the Family Flexibility subscale showed poor associations with the other subscales, that subscale was omitted, and a second set of PCA analyses were conducted on the five remaining FCS subscales.

Table 2 displays the final two-component pattern matrix for the FCS-Parent (Columns 1 and 2) and FCS-Examiner (final two columns). For both the FCS-Parent and FCS-Examiner, Family Emotional Control, Family Focus, and Family Impulsivity yielded loadings of > 0.5 on the first component, which was named Family Inhibition, drawing from FCS scales that examine the extent to which families demonstrate thoughtful, focused, and well-controlled behaviors. For both the FCS-Parent and FCS-Examiner, Organization in Space and Organization in Time subscales yielded factor loadings of > 0.8 on the second component, which was named Family Organization, drawing from FCS scales reflecting organization in time and space. Raw scores for constituent subscales from the PCA two-component models were averaged to produce composite indices of Family Inhibition and Family Organization for the FCS-Parent and the FCS-Examiner separately.

Table 2.

Principal components analysis (PCA), promax rotation: pattern matrix.

FCS subscale Parent ratings/responses
 Examiner ratings/responses
FCS-Parent Factor 1: Family Inhibition FCS-Parent Factor 2: Family Organization FCS-Examiner Factor 1: Family Inhibition FCS-Examiner Factor 2: Family Organization
Family Focus 0.97 −0.19 0.86 −0.76
Family Emotional Control 0.52 0.46 0.87 −0.04
Family Impulsivity 0.81 0.07 0.77 0.14
Family Organization Time −0.20 0.96 0.26 0.84
Family Organization in Space 0.14 0.74 −0.34 0.87
Component Eigenvalue 2.73 0.93 2.52 1.08
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Note. Subscale loadings greater than .50 are bolded. PCA yielded the same two higher order factors for both the FCS-Parent and the FCS-Examiner: Family Inhibition and Family Organization. Two component models accounted for 73% and 72% of the variance of the FCS-Parent and FCS-Examiner, respectively. Analyses excluded FCS Family Flexibility subscale to improve PCA solutions. Constituent subscales from the PCA models were summed and averaged to produce an index of Family Inhibition and Family Organization for the FCS-Parent and the FCS-Examiner separately. FCS = Family Characteristics Scale.

Comparative Analyses

Table 3 displays means and standard deviations for the FCS-Parent and FCS-Examiner as well as the child EF and language measures. Paired-samples t tests using the entire sample revealed no significant differences between FCS-Parent and FCS-Examiner Family Inhibition scores, t(97) = 1.73, p = .87, or Family Organization scores, t(97) = 0.44, p = .660. Parents of children in the HL sample reported higher levels of Family Organization compared to parents of children in the NH sample, t(124) = −2.11, p = .036. On the other hand, examiners rated families in the NH sample as showing higher levels of inhibition than families in the HL sample, t(71) = 2.56, p = .013.

Table 3.

Means and standard deviations of family and child measures.

Measure NH HL (CI & HA) CI HA
Family measures
 FCS-Parent Family Inhibition (SD) 3.55 (0.59) 3.77 (0.67) 3.75 (0.65) 3.72 (0.71)
 FCS-Parent Family Organization (SD) 3.77 (0.58) 3.99* (0.59) 4.04 (0.61) 3.93 (0.57)
 FCS-Examiner Family Inhibition (SD) 3.73* (0.44) 3.42 (0.59) 3.51 (0.45) 3.31 (0.74)
 FCS-Examiner Family Organization (SD) 3.74 (0.63) 3.88 (0.69) 4.13* (0.50) 3.54 (0.77)
Child measures
 BRIEF Inhibit (SD) 50.91 (8.85) 54.21 (11.66) 57.24** (10.88) 50.41 (11.67)
 BRIEF Working Memory (SD) 51.66 (9.37) 55.07 (10.52) 56.74 (8.34) 52.96 (12.60)
 BRIEF Plan/Organize (SD) 49.29 (8.10) 51.20 (9.17) 52.29 (7.15) 49.81 (11.21)
 BRIEF Shift (SD) 50.65 (9.67) 51.23 (10.70) 52.06 (10.47) 50.19 (11.07)
 BRIEF Emotional Control (SD) 50.92 (8.58) 52.66 (11.86) 53.32 (10.40) 51.81 (13.65)
 PPVT-4 (SD) 116.54** (10.56) 96.26 (17.44) 91.15 (17.04) 102.7** (16.00)
 CELF a (SD) 10.69** (2.80) 8.28 (3.45) 8.92 (3.50) 7.79 (3.38)
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Note. NH = normal hearing; HL = hearing loss; CI = cochlear implant; HA = hearing aid; FCS = Family Characteristics Scale; BRIEF = Behavior Rating Inventory of Executive Function; PPVT-4 = Peabody Picture Vocabulary Test–Fourth Edition; CELF = Clinical Evaluation of Language Fundamentals.

a

Mean scaled scores were collapsed across the Following Directions subtest from the CELF-5 and the Concepts and Following Directions subtest from the CELF Preschool-2.

*

p < .05.

**

p < .01.

As expected, the NH sample had better language comprehension and receptive vocabulary scores than the HL sample (see Table 3). Although the HL sample scored numerically higher on all BRIEF EF subscales compared to the NH sample (e.g., exhibiting more EF domain difficulty), none of these differences reached statistical significance.

Multiple Regression and Moderation Analyses

Multiple regression analyses were performed in two blocks to estimate child performance on EF and spoken language outcome measures. The individually estimated outcome measures were BRIEF Inhibit, BRIEF Working Memory, BRIEF Plan/Organize, BRIEF Shift, BRIEF Emotional Control, PPVT-4, and CELF C&FD. In the first regression block, each outcome measure was estimated from group hearing status (0 = NH; 1 = HL) and standardized FCS-Parent (Family Inhibition and Family Organization) and FCS-Examiner (Family Inhibition and Family Organization) scores. Thus, both parent- and examiner-rated FCS dimensions were included in first block analyses. Table 4 displays regression coefficients from statistically significant first block multiple regression models (BRIEF Plan/Organize, Working Memory, and Emotional Control are omitted from that table because these models were not significant and/or had no significant regressors).

Table 4.

First block regression analyses.

Independent variable Executive function
 Language
BRIEF Inhibit
T score, b 		BRIEF Shift
T score, b 		PPVT-4
Standard score, b 		CELF C&FD
Scaled score, b
Intercept 49.88 51.65 115.05 9.92
Hearing status (0 = NH, 1 = HL) 5.33 1.44 −16.59** −1.24
Family Inhibition, Examiner −2.02 1.26 3.36 1.35*
Family Organization, Examiner 0.65 0.45 0.29 −0.197
Family Inhibition, Parent −3.56* −3.06* 0.79 0.04
Family Organization, Parent 1.02 0.63 −1.71 0.04
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Note. First block regression analyses predicting child inhibition, shifting, receptive vocabulary, and language comprehension skills from child hearing status and FCS-Parent and FCS-Examiner higher order factors. Family inhibition was significantly associated with improved performance on each outcome measure for both groups.

BRIEF = Behavior Rating Inventory of Executive Function; PPVT-4 = Peabody Picture Vocabulary Test–Fourth Edition; CELF = Clinical Evaluation of Language Fundamentals; C&FD = Concepts and Following Directions; NH = normal hearing; HL = hearing loss; FCS = Family Characteristics Scale.

*

p < .05.

**

p < .001.

First Block Regression Analyses

First block regression models estimating child EF

First block regression analyses revealed a significant multiple regression model estimating BRIEF Inhibit T scores, R 2 = .19, F(5, 67) = 3.07, p = .015. BRIEF Inhibit performance was negatively associated with FCS-Parent Family Inhibition (see Table 4), indicating that greater family inhibition was related to better child inhibition skills (lower BRIEF scores reflect better EF). No other first block regression models were significant for child EF, although BRIEF Shift T scores were negatively associated with FCS-Parent Family Inhibition.

First block regression models estimating child spoken language

First block regression analyses revealed a significant multiple regression model estimating PPVT-4 standard scores, R 2 = .31, F(5, 67) = 6.05, p < .001, indicating that group hearing status was significantly associated with PPVT-4 performance. The first block multiple regression model estimating CELF C&FD scaled scores was also significant, R 2 = .25, F(5, 67) = 4.05, p = .001, and CELF C&FD scaled scores were significantly positively associated with FCS-Examiner Family Inhibition.

Second Block Regression Analyses

Following the first block of regression analyses, the estimation of BRIEF Inhibit, BRIEF Working Memory, BRIEF Plan/Organize, BRIEF Shift, BRIEF Emotional Control, PPVT-4, and CELF C&FD was performed by testing (using a stepwise method with retention of terms significant at p < .05) Group × Family Inhibition and/or Group × Family Organization interactions from the FCS-Parent and/or FCS-Examiner. Thus, the interaction between parent- and examiner-rated FCS scores and hearing status was only included in the analyses if significant. Figure 1 displays the significant Group × Family-Level EF interactions and child outcomes. Each panel in Figure 1 shows the relation between FCS higher order factors and child outcome performance separately for children with HL and NH and were generated using PROCESS, an SPSS macroinstruction that uses ordinary least squares regression for mediation, moderation, and conditional path analyses (Hayes, 2017).

Figure 1.

Figure 1.

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Group × FCS-Examiner Family Organization interactions for child EF outcomes (A–C). Group × FCS-Parent Family Inhibition interaction for child EF outcomes (D). The x-axis for each panel shows standardized FCS scores. (A) Parent-reported problems with inhibition increased in children with HL and decreased in children with NH as family-level organization increased, b = 6.73, p = .008. (B) Problems with working memory skills increased in children with HL and decreased in children with NH as family-level organization increased, b = 6.46, p = .005. (C) Problems with planning and organization skills decreased in children with NH only as family-level organization increased, b = 4.41, p = .027. (D) Problems with planning and organization skills decreased for children with HL as family-level inhibition increased, b = −4.69, p = .015. FCS = Family Characteristics Scale; BRIEF = Behavior Rating Inventory of Executive Function; EF = executive functioning; HL = hearing loss; NH = normal hearing.

Group × FCS-Examiner interactions estimating child EF

The multiple regression model estimating BRIEF Inhibit T scores remained significant following the inclusion of a Group × FCS-Examiner Family Organization product term, b = 6.73, ΔR 2 = .08, p = .008 (see Figure 1A). Specifically, for families of children with HL, there was positive association between examiner-rated Family Organization and parent-reported difficulties with child inhibition skills. Conversely, for families of children with NH, there was a negative association between examiner-rated Family Organization and parent-reported difficulties with child inhibition.

Although the original first block multiple regression model estimating child BRIEF Working Memory performance was not significant, second block analyses revealed that the model estimating BRIEF Working Memory was significant following the inclusion of a Group × FCS-Examiner Family Organization product term, R 2 = .21, F(6, 66) = 2.86, p = .015, b = 6.46, ΔR 2 = .10, p = .005 (see Figure 1B). For families of children with HL, there was a positive association between examiner-rated Family Organization and parent-reported difficulties with child working memory. For families of children with NH, there was a negative association between examiner-rated Family Organization and parent-reported difficulties with child working memory.

The original first block multiple regression model estimating child BRIEF Plan/Organize performance was also not significant. However, second block analyses revealed that the model estimating BRIEF Plan/Organize was significant following the inclusion of hearing status as a Group × FCS-Examiner Family Organization product term, R 2 = .18, F(6, 66) = 2.40, p = .037, b = 4.41, ΔR 2 = .063, p = .027 (see Figure 1C). For families of children with NH, there was a negative association between examiner-rated Family Organization and parent-reported difficulties with child planning/organization skills. There was no apparent association for families of children with HL.

Group × FCS-Parent interactions estimating child EF

Another stepwise regression block conducted on the model estimating BRIEF Plan/Organize T scores found a second significant Group × FCS-Parent Inhibition product term, R 2 = .25, F(7, 65) = 3.10, p = .007, b = −4.69, ΔR 2 = .07, p = .015 (see Figure 1D). For families of children with HL, there was a negative association between parent-reported Family Inhibition and parent-reported child difficulties with planning/organization skills. There was no association observed for families of children with NH.

Discussion

The objective of this study was to compare associations between family-level EF and neurocognitive and spoken language skills in families of children with HL to families of children with NH. Previous research carried out by Holt et al. (2013, 2012, 2019) found that dimensions of the home environment were related to pediatric HL outcomes. This study extended that prior work by examining family-level EF to assess relations between the shared regulation of behaviors among family members and the EF and language skills in an at-risk clinical population.

The FCS as a Novel Measure of Family-Level EF

Investigating family-level EF first required the development of a method for quantifying family-level EF. This study used both parent- and examiner-rated versions of the FCS—a new measure designed to assess dimensions of family-level EF. Parent report FCS subscales showed strong internal consistency, and results from a PCA of FCS subscales yielded the same two higher-order components for both the FCS-Parent and the FCS-Examiner. The first higher-order component, family-level inhibition, was composed of FCS subscales assessing regulatory behaviors within the family system. The second higher-order component, family-level organization, was derived from FCS scales assessing the consistency of routines and regular maintenance of the family's home environment. Given the similar component loadings for both the FCS-Parent and the FCS-Examiner, as well as the high internal consistency for each of the FCS-Parent scales, this study presented preliminary evidence that both FCS versions can be used to assess family-level EF.

Differences in Family-Level EF Between Families of Children With HL Versus NH Children

Comparisons of EF between families of children with HL and families of NH children revealed several significant differences between the two groups on FCS-assessed Family Inhibition and Family Organization, depending on whether the rater was a parent or an examiner. First, families of children with NH had significantly higher examiner-rated (but not parent-rated) family-level inhibition compared to families of children with HL. This discrepancy in ratings of Family Inhibition depending on the rater indicates that external examiners are seeing lower levels of Family Inhibition in families of children with HL (compared to families of children with NH), but no such difference is being seen by the parents themselves. This finding suggests that parents of children with HL have less recognition of (or more tolerance for) limitations in focus, emotional control, and behavioral inhibition, relative to ratings provided by an external observer. This hypothesis is supported by the finding that parent-rated Family Inhibition (but not examiner-rated inhibition) was associated with better parent-rated child inhibition (see Table 4) and with better parent-rated child planning and organization behaviors in the HL sample (see Figure 1). Such an association between parent ratings of family-level and child-level EF could be, in part, a result of a tendency of some parents to be more accepting of EF delays, not seeing them as problematic or unusual at either a child or family level. Alternatively, the examiner raters might be interviewing and observing families of children with HL during a period of time (i.e., the study visit) when less family-level inhibition is present, causing them to see lower levels of Family Inhibition than parents themselves, who are providing ratings based over a longer time frame and larger sample of the relevant behaviors. Additional research is recommended to better understand the discrepancy between parent and examiner reports.

In contrast to findings for family-level inhibition, parents in families of children with HL rated their families as more organized than did parents in families of children with NH, while no differences were found in Family Organization based on examiner ratings. This result may reflect the greater level of organizational effort and awareness required in families of children with HL, in order to address challenges related to communication, education, behavior, and treatment, compared to families of children with NH. Parents of children with HL may be particularly aware of the extra effort involved in family organization, even though the end point manifestation of this organization is less apparent to an external observer such as an examiner, or even an experienced and highly trained clinician. Furthermore, the additional organizational effort needed to meet the challenges of having a child with HL, while necessary to address challenges, may not be consistently sufficient to have a marked impact on child EF. This explanation is supported by both sets of findings that parent-rated Family Organization is unrelated to child outcomes and that examiner-rated Family Organization is unrelated to child outcomes, specifically in the sample with HL.

Associations of Family-Level EF With Outcomes

Specific domains of child spoken language and neurocognitive outcomes were found to be associated with specific domains of family-level EF, and in some cases, these associations were different depending on whether the child had HL. Multiple regression analyses demonstrated significant associations between parent-rated Family Inhibition and child EF skills (BRIEF Inhibit and Shift subscales) as well as between examiner-rated inhibition and higher-order language (CELF C&FD subtest), even after accounting for hearing status. Although only parent-rated Family Inhibition predicted child EF skills in the regression, post hoc zero-order Pearson correlations also demonstrated a statistically significant relationship between examiner-rated Family Inhibition and child inhibition (for BRIEF Inhibit, r = −.27, p < .05). Thus, both parent- and examiner-rated Family Inhibition were associated with child inhibition, but entering both in the regression model resulted in an attenuation of the term for examiner-rated Family Inhibition because of overlap between examiner- and parent-rated Family Inhibition and child inhibition. This result strongly supports an association between family-based inhibition and child inhibition, because similar findings were identified across different raters.

For language outcomes, examiner ratings of Family Inhibition predicted higher-order language comprehension in regression equations controlling for hearing status. Although the term for examiner-rated Family Inhibition did not reach significance in regressions predicting receptive vocabulary (PPVT-4; r = −.86, p = .340), the zero-order correlation between examiner-rated Family Inhibition and receptive vocabulary scores was statistically significant (r = .31, p = .007). Based on these findings, family-level inhibition also appears to be an important predictor of language development in children, consistent with prior research in NH samples (Evans, 2006; Vernon-Feagans et al., 2012).

For children with HL specifically (contrasting with children with NH), there were unexpected associations between family-level EF and several child neurocognitive domains. Specifically, in contrast to findings for children with NH, greater amounts of parent-reported difficulty with child inhibition and working memory on the BRIEF was associated with greater examiner-rated family-level organization for children with HL, while Family Organization was unrelated to BRIEF Plan/Organize scores in the sample of children with HL. Because children with HL are at greater risk for delays in inhibition and working memory compared to NH children (Kronenberger & Pisoni, 2018), greater organization in families of children with HL might reflect compensatory efforts to strengthen the structure and consistency of daily routines and the organizational behaviors of family members in response to the risk and/or challenges of poorer child EF. Additionally, parents of children with HL who are more organized might also be less tolerant and more cognizant of EF-related child difficulties, leading to greater report of EF problems on the BRIEF-P/BRIEF2. On the other hand, families with reduced organizational attributes might be more tolerant (or less aware) of poorer child EF, corresponding with fewer reported EF difficulties.

Family Inhibition (parent-rated) on the other hand, was found to be related to fewer planning and organization problems in children with HL, suggesting a positive role for family-level inhibition for the development of that EF domain in children with HL. It is likely that children, regardless of hearing status, benefit from the collective modeling of higher level self-regulatory behaviors among family members. Given that children with HL are already at increased risk for weaknesses in EF domains such as planning and organization, greater family-level inhibition could represent a critically important attribute of the family system that could offset this at-risk outcome.

Study Limitations

There were several limitations to the study. The first limitation was that the sample size of children with HL was small, precluding the separation of children with HAs and CIs into groups while maintaining adequate statistical power. As families continue to be enrolled in the Families and Hearing Study, future analyses will investigate the role of family-level EF in families of children with HAs and families of children with CIs separately once sufficient power has been achieved to evaluate each group individually. A second limitation concerns the FCS-Examiner ratings. Research clinicians were well trained on the administration of the FCS-Examiner, integrating parental reports and direct observations of the home environment when determining scale ratings. However, no interrater reliability exists for this version of the FCS. Correlations between the FCS-Parent and FCS-Examiner were modest for family-level inhibition (r = .22, p < .07) and family-level organization (r = .37, p < .001), but interrater reliability is still unknown for this particular measure of EF. A third limitation is the correlational/cross-sectional nature of this study, which precludes conclusions about the causal direction of results. Future research should address these limitations in order to better understand the role of family-level EF in child outcomes after interventions to improve HL. A fourth limitation and potential area of future research is distinguishing the role of family-level EF within families with multiple children. Previous work found that having fewer family members was associated with better language outcomes for children with HL (Geers et al., 2003). It is possible that the number of children within the family system could impact how EF is modeled between siblings. Furthermore, family EF levels might be modeled similarly between children with HL and any NH siblings, but the EF dimensions might still be differentially related to outcomes within the same family system. Although outside the scope of this study's objectives, a related future direction would also be to investigate longitudinal relations between family EF and pediatric HL outcomes to evaluate directional relations between family and child EF. Results could support potential family-based targets for intervention.

Conclusions

The investigation of whole-family contributions to at-risk outcomes is a novel direction to pediatric HL research that offers the potential for a more sophisticated biopsychosocial systems understanding of child development following use of HAs or CIs. The measurement of higher-level family attributes such as family-level EF can help elucidate contributions to neurocognitive and spoken language outcomes. Results using a novel measure of family-level EF, the FCS, extend previous work examining relations between proximal environmental factors and pediatric HL outcomes, providing additional support for the S-B-R model of development for children with HL. Specifically, inhibition and organization expressed at the family level were found to be significantly associated with child EF and higher-order language development. As a result, these family-level EF domains could have important implications for the development of novel targeted family-based interventions and individualized counseling for families of children with HL, particularly families who have a child that displays poor speech-language outcomes with their HA or CI.

Acknowledgments

This study was supported by the National Institutes of Health (Grant R01DC014956 awarded to Holt and Pisoni, MPIs).

Funding Statement

This study was supported by the National Institutes of Health (Grant R01DC014956 awarded to Holt and Pisoni, MPIs).

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