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Abstract

PURPOSE:

Psychometric data are reported for a new Global Family Quality of Life Scale (G-FQOLS) (3-items) evaluating family members, parents and adolescent/young adults (AYA).

METHODS:

Families ( $N=$ 209) were interviewed in a study addressing secondary conditions and adaptation in families of AYA with and without spina bifida (SB). Principal component factor analysis with Varimax rotation, Cronbach alpha, and Intraclass correlation (ICC) for parent-AYA agreement of FQOL assessment were conducted.

RESULTS:

A single factor with an eigenvalue greater than 1 was identified. Factor loadings were 0.79–0.94. Internal reliabilities were strong ( $\alpha=$ 0.86–0.90). ICC coefficients between parent and AYA ratings were 0.38–0.48.

CONCLUSION:

This study provides preliminary support for the G-FQOLS. This global appraisal of Family Quality of Life (FQOL) reflects a participant’s personal weighting of domains important to him/her.

Keywords

Family quality of life parents adolescent spina bifida

1. Background

An emerging concept, Family Quality of Life (FQOL), is useful to understand adaptation outcomes of families. Appraising the family, extends the meaning of individual quality of life as defined by the WHO (1997) “an individual’s perception of the position in life in the context of the culture and value systems in which they live, and in relation to their goals, expectations, and concerns” [1, p. 1]. Similar to individual quality of life (QOL) which captures the lived experience of the individual, FQOL can be multidimensional or an overall global perception of FQOL. The aim of this article is to detail psychometric data from parent and adolescent/young adult (AYA) in a report for a new Global Family Quality of Life Scale (G-FQOLS), specifically, construct validity and internal reliability. This scale measures FQOL, which has emerged from the perspective of families with children.

Three authors have conceptually defined FQOL. The earliest definition proposed by Poston [2] is:

Family quality of life can be defined as the conditions, consistent with the family’s values where the family’s needs are met (i.e., daily family life, emotional well-being, financial well-being, physical environment, health, parenting, advocacy); family members enjoy their life together as a family (i.e., family interaction); and family members have the opportunities to do things that are important to them (i.e., social well-being and productivity) [2, p. 346].

Brown et al. conceptualize FQOL as “…the degree to which family quality of life is enjoyable, meaningful, and supported by the types of resources that are important to family members, as well as the struggles faced by families [3, p. 3]. Thereafter, Zuna et al. defined FQOL as “a dynamic sense of well-being of the family, collectively and subjectively defined and informed by its members, in which individual and family-level needs interact” [4, p. 262].

Two of these definitions [2, 3] suggest a multi-dimensional concept, such as an individual’s perspective of components of family life while the third proposed by Zuna et al. is defined as a collective overall wellbeing [4]. The two multi-dimensional definitions have similar domains including (a) family life that is meaningful or consistent with family values, (b) enjoyment, and (c) resources. A reflection of family struggle is a unique aspect of Brown et al.’s conceptualization [3]. These multi-dimensional definitions are limited by the a-priori delineation of specific domains that are important to the family. In contrast, Zuna et al.’s approach is a collective conceptualization based on family needs; however, this conceptualization may or may not be reflective of the beliefs in a variety of families [4]. Important aspects of family life may vary between members. A common limitation of all these definitions is that none includes the family member’s ability to differentially prioritize domains of FQOL important to him/her. These multiple conceptualizations of FQOL have led to several measures of the construct.

It is important to differentiate FQOL, which focuses on a sense of well-being of the family, from a related concept of family functioning. Family functioning is defined as the attributes of a family system that characterize how they operate or behave [5]. It includes attributes such as family cohesiveness, satisfaction, mastery, hardiness, or resourcefulness. While family functioning and FQOL are related empirically ( $r=$ 0.34–0.60) [6, 7], they are not the same concept. Family functioning is associated with both individual and family outcomes in families with children with chronic conditions [8]. Family processes are modifiable and may be protective for families. In families with AYA with a complex chronic condition [spina bifida (SB)], family satisfaction and parent depressive symptoms predicted FQOL, but family cohesion or resources did not [6]. In families with adolescents with SB, family cohesion is relatively stable when observed over time and family conflict does not increase as dramatically when compared to families of children without SB [9].

In addition, resilience or protective factors in AYA with SB such as communication self-efficacy (confidence in communication), future expectations, and coping support may themselves be modifiable and were also related to family factors [10]. Certain family factors may be important correlates to FQOL while protective factors in AYA may relate to family factors and/or to FQOL. Therefore, FQOL becomes an important construct to be able to assess.

For this study, FQOL was defined as an appraisal of the domains of life important to the family [11]. The construct FQOL can be operationalized as a family outcome or result of the efforts of families to balance those interactions and relationships to stabilize the family and environment on a continuum which is dynamic and salient to the family. The items of the FQOL Scale (rate your; your teen’s/parent’s; or your family’s QOL) were expected to load on a single factor due to their strong interrelationships in a previous study [10].

1.1 Overview of FQOL measurement

In the individual and FQOL literature, there are two approaches to measurement. One is domain-specific and the other is overall QOL. The first uses established measures based on domains found important to the population of individuals at large or with certain conditions at large. The second obtains a summative overall score influenced by the domains and priorities of the respondent. In this latter approach, follow-up questions are needed to understand which domains are pertinent to the respondent. Many instruments measure both domain-specific and overall QOL. An overview of existing domain-specific and overall measures is described to provide a review of the current measurement of FQOL. See Table 1 for summary of measures of FQOL.

1.2 Domain-specific measures

1.2.1 Beach FQOL Scale

The Beach FQOL Scale was developed in 2006 to assess family outcomes in families of children with developmental or intellectual disabilities, birth to age 21. The Beach overall FQOL value is a mean score of the total number of items [12]. Hoffman et al. [12] found evidence of validity using confirmatory factor ana- lysis and convergent validity for two of five subscales

Table 1
Summary of Domain-Specific and Overall measures of FQOL

Author	Domains-specific measures	Overall measures		Sample/summary of psychometric properties
	Domains	Overall sum	Overall
		of domains	global
Beach Scale/ Hoffman et al., 2006 [12]	Physical/Material well-being Family interaction Parenting Disability-related support Emotional well-being	X		Two hundred and eighty families with children (0–12 years old) with disabilities (developmental delay, autism spectrum disorder, attention deficit disorder/attention deficit with hyperactivity disorder, mental retardation, emotional disorders, learning disabilities, and other physical and health disabilities.). Location of the sample was in the following US States: Washington, Kansas, Michigan, Indiana, Pennsylvania, North Carolina and Texas. Child gender, 64% male. Race/ethnicity was 78.9% White non-Hispanic. Mild level of severity 51%. The 25-item scale measures satisfaction in five domains. Good internal reliability reported for the five subscales ( $\alpha=$ 0.70–0.90) and total scale ( $\alpha=$ 0.88). Response pattern was 1 (very dissatisfied) to 5 (very satisfied). Confirmatory factor analysis supported a good fit for a model with five subscales and a second order overall FQOL factor ( $\chi^{2}$ (270) $=$ 439.24, $p<$ 001, CFI $=$ 0.92, RMSEA $=$ 0.05). Convergent validity with 2 domain subscales-The Family Interaction subscale of the Beach Scale related to Family APGAR [22], ( $r=$ 0.68). The Family Resource Scale [23] related to Physical/Material Well-being subscale ( $r=$ 0.60). Test-retest reliability for satisfaction subscales across domains showed significant correlations between time points ( $r=$ 0.60–0.77), time between test and retest was 3 months [12].
FQOL-2006 Survey Isaacs et al., 2012 [17]	Family Relationships Influence of values Health Careers Community Support from services Support from others Leisure Finances	X	X	Four hundred and two families and family members (across country sub-samples mean age 11.5–20.1 years) with disabilities (intellectual disability, autistic spectrum disorder/pervasive developmental disorder, down syndrome, cerebral palsy, other). Location of the sample was from four countries (Australia, Canada, Nigeria, and US). Child gender across country sub-samples ranged 60–69% male [17]. 54-item survey. Overall global items are “Overall, how would you describe your family’s quality of life?” and “Overall, how satisfied are you with your family’s quality of life?” [3] Response pattern for overall global item was 5 (excellent) to 1 (poor). Response pattern for satisfaction question was 5 (very satisfied) to 1 (very dissatisfied). Confirmatory factor analysis supported a good fit for a model with nine domains (Health, Finances, Family, Others, Services, Values, Careers, Leisure, Community) and overall latent FQOL factor in 3-countries data (Australia, Canada, US), not Nigeria. When error terms for highly correlated domains were allowed to enter the analysis, the Nigerian sample also showed good fit for model with nine domains and an overall latent FQOL factor. Several factor loadings for the domains were low (0.29–0.40) in Nigerian sample and most paths had moderate loadings in 3-countries sample (0.41–0.64) [19]. Internal reliability for each domain was good in families in Nigeria ( $\alpha=$ 0.73–0.83) and fair to good in 3-countries sample ( $\alpha=$ 0.53–0.83) [17]. Although several domains had problems (health $\alpha=$ 0.53; support from service $\alpha=$ 0.67), an overall latent FQOL factor was supported in this analysis. Additionally test-retest and convergent validity were not reported [17].
Single item/Sawin et al., 2002 [10]			X	Sixty families with children with spina bifida (parent and AYA). Location of the sample was in US Midwestern state from a single clinical setting. Child mean age 16.2 years; range 12–21; AYA gender 60% female; 80% had shunts. Single item was “How would you rate your family’s quality of life?" The response pattern was zero (poor) to 100 (excellent) [10].

Table 2

Summary of studies using FQOL measure

Author/ Year	Sample size/ Location	Condition/ disability	Sample characteristics	FQOL measure	Strength/limitation
Sawin 2002 [10]	60 AYA & parents US Midwest	Spina bifida	Mean age 16.2 of AYA with Spina bifida; range 12–21; AYA gender 60% female; 80% had shunts; parents 73% married; single clinical setting	Single item measure	Report on individual and FQOL single items. Small convenience sample, single site
Summers 2007 [14]	180 parents US Midwest	Developmental/ intellectual disability	Age range of child with disability birth to 5 years; child gender 63% male; 46% had mild level of severity of disability; families of children who receive early childhood services	Beach FQOL Scale	19% response rate; respondents who completed FQOL scale with highest possible ratings were excluded from the sample
Davis 2009 [28]	64 parents Australia	Developmental/ intellectual disability	Mean age 51.98 months (9.65), range 36–72 months, child gender 43 males/21 females; received services in early intervention program between 2–60 months; diagnoses Autism (34), Speech/language Impairment (28), Developmental Disability (19), Physical Disability/Cerebral Palsy (9), Down Syndrome (1), Fragile X (1), Dandy-Walker (1), Dravet Syndrome (1); 48% described severity of delay as moderate	Beach FQOL Scale	16% response rate; sample of families of children with different disabilities was not examined by sub-samples of conditions
Werner 2009 [24]	35 family members Toronto Canada	Developmental/ intellectual disability	Mean age 25.43 (14.58); range 3–59; majority families with member with autism; 60% lived in residential group homes, 40% lived with family; 24 mothers, 7 fathers, 3 siblings, 1 mother and sister participated together; 26 families were 2 parent homes	FQOL-2006	Small sample, sample recruited from 2 sites, which differed in age and living situation (residential placement or home) of participants; low internal consistency on dimensions (importance, opportunities, attainment, and satisfaction)
Jackson 2010 [16]	207 family members US – 42 states	Hearing impaired	Mean age 44 months (SD 16.58); range 2–72 months (6 years); child with deaf or hard of hearing and receiving services	Beach FQOL Scale	The instrument was modified by omitting question related to adult with disabilities; modification included impact of deafness on family life, child outcomes and desired family support
Eskow 2011 [29]	228 parents in waiver group; 627 parents registry group US – Maryland	Developmental/ intellectual disability	Parents of child or adult with Autism ages 3 years – 18 and older enrolled or wait-listed (registry) for state Medicaid Home and Community-Based Services waiver program	Beach FQOL Scale	28.8% response rate; 80% male and 20% female; children in waiver group were older
Neikrug 2011 [30]	103 family members Israel	Developmental/ intellectual disability	Mean age 10.86 of child with Developmental Disability, range 1–31 years old; 81% mothers 4% fathers other unknown; 7 single parent homes (others 2-parent); child gender 70% male; 19% Developmental Disability, 3% Cerebral Palsy, 32% Pervasive Developmental Disorder, 8.7% Down Syndrome, 3.9% Rett Syndrome, 28% other; convenience sample	FQOL-2006	Translated to Hebrew by professional translator not part of research team pretested for modifications; not random sample, did not report qualitative findings of instrument
Ajuwon 2012 [31]	80 family members Nigeria	Developmental/ intellectual disability	Mean age 12.3 (7.85); main caregivers of school-aged children & youth with ID; 82% 2 parent home; 78% children lived with family; 35% unknown diagnosis, 30% Cerebral Palsy, 15% Down Syndrome, 12.5% Autism	FQOL-2006	Qualitative findings add context to family experience beyond questions of instrument/sample included those receiving services

Table 2, continued
Author/ Year	Sample size/ Location	Condition/ disability	Sample characteristics	FQOL measure	Strength/limitation
Clark 2012 [32]	52 family members Malaysia	Developmental/ intellectual disability	Mean age 7.54 (3.99) range from 2–18 years; 43 respondents were mothers; 33 were in 2 parent families children had Developmental disability/Intellectual disability, 2 families had 2 children with disability, 49 of 54 children lived at home, diagnoses were Intellectual Disability, Down Syndrome, Cerebral Palsy, Autism, & others; random selection receiving services	FQOL-2006	Survey instrument translated and back translation done (details of changes not available); short form did not allow for data to add meaning or context to responses; sample gender of child not accounted for 38 boys and 16 girls; Eighteen of the 52 families in the current study reported that they had live-in paid caregivers or extended family members that provided care and support for their family member with a disability, reducing responsibility left to the primary caregiver
Hu 2012 [13]	442 family members China	Developmental/ intellectual disability	Age of child 0 $->$ 18 with majority between 7–17 years old; child with Intellectual Disability living in urban and suburban Beijing; stratified sampling method	Beach FQOL Scale	Return rate of 89.1% father and mother respondents, initial response rate 72%/skewed distribution of family income (low income); no data of family dynamics, family support services and family coping
Rillotta 2012 [33]	150 family members South Australia	Developmental/ intellectual disability	Age range 2–46 years with Intellectual Disability or Autism; mean 17.3 years; 64.3% male, 35.7% female; 2 parent home 66.7%, randomly selected from South Australian Government’s disability service provider client database	FQOL-2006	Response rate 18%; low to moderate internal consistency across dimensions (importance, stability, initiative)

Note. AYA $=$ adolescents and young adults.

(Physical/material well-being and the Family Interaction subscales) (see Table 1). Internal reliability for the total and five subscales was $\alpha=$ 0.70–0.90 [12]. Good reliability was also found in two other studies ( $\alpha=$ 0.73–0.92) [13, 14]. These study samples were not diverse, therefore, validity in subgroups of race/ethnicity and those with a chronic condition without intellectual/developmental disability is limited. The Beach Scale was modified by Zuna et al. by deleting the disability support subscale in families with children who do not have a disability [15]. A factor analysis of the latter modified scale yielded a four-factor solution comprising Family Interaction, Parenting, Emotional Well-Being and Physical/Material Well-Being. These subscales and the total scale had high reliability ( $\alpha=$ 0.77–0.92) [15]. Jackson et al. [16] modified the scale for use in families with children who have a hearing impairment. They added a subscale on the impact of deafness for each FQOL item, a subscale for satisfaction with a child’s progress on communication outcomes, and an open ended section for comments about desired supports [16]. No additional factor analyses or reliability analyses were reported for Jackson et al.’s modification [16].

1.2.2 The FQOL-2006 Survey

The FQOL-2006 Survey [3] measures FQOL in families who have a member with intellectual or developmental disability. Mean scale scores are calculated for each domain. The survey contains two sections for each domain, (1) quantitative and qualitative items about characteristics of the domain, and (2) six items for each dimension and one open ended question for each domain, “What things do you think could improve your family quality of life?”. Additionally, two global items were included which are described below. Isaacs et al. tested the factor structure of the FQOL-2006 Survey in a large sample across four countries (Nigeria, Australia, Canada, US) in families with a member with an intellectual disability ( $n=$ 402) and found the importance dimension ratings across the nine domains were skewed (all domains rated high) [17]. The stability dimension across the nine domains was not supported due to low factor loading leading to a final factor structure of 4 dimensions: opportunity, initiative, attainment, and satisfaction [17]. Confirmatory factor analysis supported a good fit for a model with nine domains (Health, Finances, Family, Others, Services, Values, Careers, Leisure, Community) and an overall latent FQOL factor. Although an overall latent FQOL factor was supported in this analysis, it has not been used in the literature by other investigators [17].

1.3 Global measures of overall FQOL

Single item individual QOL measures have been validated and supported [10, 18]. Some researchers feel that the overall global perception of QOL that reflects the individual’s emphasis on important domains may be useful as an outcome [10, 19, 20]. Similarly, a global concept of a family member’s perception of FQOL can include the domains important to the family.

Two global items at the end of the FQOL-2006 Survey address FQOL: “Overall, how would you describe your family’s quality of life?” and “Overall, how satisfied are you with your family’s quality of life?” [3]. Response pattern for the overall global item was 5 (excellent) to 1 (poor). Response pattern for the satisfaction question was 5 (very satisfied) to 1 (very dissatisfied). Only the global satisfaction item has been reported in the literature. In addition to the two global measures of overall FQOL described, a single item measure, “How would you rate your family’s quality of life?” was used by Sawin, Brei, Buran, and Fastenau to capture FQOL in families who had an adolescent with SB [10]. The response pattern was zero (poor) to 100 (excellent).

1.4 Summary of studies using FQOL measures

A small number of studies have reported measurement of FQOL in a variety of ways, mostly in correlational descriptive studies. While assessment of FQOL has been conducted since 2002, 12 studies have examined FQOL in families with children with either chronic health conditions or intellectual and developmental disabilities [11]. Only two studies examined families with children with and without chronic health conditions (CHC) [6] and without disabilities [15]. A majority of studies were conducted in families of children with intellectual and developmental disabilities. See Table 2 for a summary of studies using FQOL measures.

2. Methods

This psychometric analysis used data from a larger study on secondary conditions and adaptation in adolescents with SB. This study included 209 families who had an AYA with and without SB. Participants (parents and AYA) were asked to rate their own, their parent (or AYA) and their family’s QOL.

Figure 1.

Items for Global Family Quality of Life Scale parent version. For teen version, items are reordered: (1) Your quality of life; (2) Your family’s quality of life; (3) Your parent’s quality of life.

Table 3

Characteristics of the sample

	Total		Subsample		Comparison
			with SB		subsample
Variable	$N$	%	$n$	%	$n$	%
Group			112	54	97	46
AYA age
12–15 years	121	58	67	60	54	56
16–18 years	56	27	29	26	27	28
19–25 years	32	15	16	14	16	17
Gender (child)
Female	113	54	57	51	55	43
Male	97	46	55	49	42	57
Combined family income ${}^{*}$
Less than $20,000	24	12	20	18	4	4
$20,000–$50,000	57	27	35	31	22	23
$50,000 or over	126	60	56	50	70	72
Gender (parent)
Female	196	94	105	94	90	93
Race (parent)
Black	22	11	6	5.4	16	17
Caucasian	179	86	101	90.2	78	80
Other	7	3.5	4	3.6	3	3.1
Ethnicity
Hispanic	6	3	4	3.6	2	2.1

Note. Demographic variables were tested for significant differences between subsamples using Chi Square statistic. Income significantly different by subsample. ${}^{*}\chi^{2}$ (207) $=$ 16.67, $p<$ 0.001.

2.1 Sample

Participants were 209 parents of AYA (112 SB group; 97 comparison) and 197 AYA (103 SB group; 94 comparison) from a total of 209 families. Parents and AYA were recruited for a larger study of secondary conditions and adaptation in AYA with SB from four childrens’ hospital spina bifida programs in the Midwest and the Eastern United States (US). Comparison families were recruited by referral from participants with SB, primary care providers, and advertisement in each hospital. Inclusion criteria were English speaking families with AYA 12 to 25 years of age and without diagnoses of moderate or severe intellectual disabilities. AYA had no major medical conditions unrelated to their SB. Parents and AYA with complete data on the FQOL measure were included in the psychometric evaluation. IRB approval was obtained for both the original AYA adaptation study and secondary analysis. Data were collected by telephone interviews [6, 7].

2.2 Instrument

Three global items, your, your teen’s/parent’s, or your family’s quality of life were rated on a scale from zero (poor) to 100 (excellent) (see Fig. 1). Depending upon who is responding, either AYA or parent, FQOL reflect the respondents’ report of their family’s and/or their family member’s quality of life. The three items measure individual’s perceptions and when combined by respondent measures FQOL.

Table 4
Summary of Factor Analysis, Reliability, and Parent-AYA Agreement for 3-item FQOL Scale

Item	1	Communalities	1	Communalities	$\alpha$	ICC ${}^{*}$	95% CI
	Factor loading		Factor loading		Parent/AYA
Total sample	Parents ( $n=$ 209)		AYA ( $n=$ 197)		0.88/	0.41 ${}^{*}$	0.22–0.56
1 Family’s QOL	0.94	0.89	0.94	0.88	0.86
2 Your QOL	0.90	0.80	0.81	0.66
3 Teen’s/Parent’s QOL	0.86	0.73	0.89	0.80
Eigenvalue	2.43		2.34
% of variance	80.83		77.95
Group with SB	Parents ( $n=$ 112)		AYA ( $n=$ 103)		0.90/	0.38 ${}^{*}$	0.10–0.58
1 Family’s QOL	0.94	0.89	0.94	0.89	0.86
2 Your QOL	0.91	0.84	0.79	0.63
3 Teen’s/Parent’s QOL	0.87	0.76	0.91	0.83
Eigenvalue	2.49		2.35
% of variance	82.99		78.19
Comparison group	Parents ( $n=$ 97)		AYA ( $n=$ 94)		0.86/	0.48 ${}^{*}$	0.22–0.65
1 Family’s QOL	0.94	0.88	0.93	0.87	0.87
2 Your QOL	0.89	0.79	0.87	0.76
3 Teen’s/Parent’s QOL	0.86	0.74	0.86	0.75
Eigenvalue	2.41		2.38
% of variance	80.25		79.17

Note. Principal Component Analysis with Varimax Rotation; ${}^{*}$ Intraclass correlation for assessment of parent and AYA agreement significant $p<$ 0.05.

2.3 Procedure and data analysis

Principal component analysis using Varimax rotation was conducted. A significant Bartlett test and KMO statistic above 0.60 were used to determine if the analysis supported factorability. Eigenvalues greater than 1 and scree plot patterns were used to identify the number of factors. Intraclass correlation coefficients (ICC) were computed. Guidelines for interpretation of ICC are 0.90 excellent, 0.80 very good, and 0.70 adequate [21, p. 92]. Further, two additional measures of family functioning, the Family APGAR [22] (family satisfaction) and the Family Mastery and Health subscale of the Family Inventory of Resources for Management [23] (family resources) were used for concurrent validity analysis. Family APGAR reflects the parent or AYA satisfaction with family adaptation, partnership, growth, affection, and resolve [22]. Family resources, reported by the parent, were an indicator of mastery over family events, family support resources, family esteem, and communication [5]. For all scales, higher scores reflect greater family satisfaction and family support resources. Data from each respondent (parent and AYA) were analyzed separately using total samples with and without SB and then by subsamples. Principal component factor analysis using Varimax rotation was conducted to assess factor structure. Cronbach alpha was used to assess internal reliability. Frequencies were used to describe the scale scores for all measures. T-tests evaluated the differences in FQOL between parent groups (e.g., parents with and without an AYA with SB) and AYA groups (e.g., those with and without SB). The relationships of FQOL to measures of family function were evaluated using Pearson Correlations. FQOL scores between parents and AYA pairs were evaluated using ICC.

3. Results

Parents were primarily female (94%), Caucasian (86%), well-educated either attending or completed college/vocational training/graduate program (71%) and married (74%) (see Table 3 for sample characteristics). Combined family income was significantly lower for families with AYA with SB, 18% earned less than $20,000 and 50% earned greater than $50,000. In comparison group, 4% earned less than $20,000 and 73% earned greater than $50,000. About half of the AYA were female (54%) with a mean age of 15 years (range 12–25). Eighty-four percent of AYA with SB had a shunt and half used a wheelchair (50%). AYA with SB had a broad range of functional motor levels, high lumbar (19%), mid lumbar (33%), low lumbar (35%) and sacral (8%).

Principal component analysis of these parent and AYA data and subsamples (with or without SB) with Varimax rotation of the 3-item scale of FQOL items identified that the items load on one factor. In the total and subsamples with parent or AYA ratings (with and without SB), the Bartlett tests were significant and KMOs were 0.654–0.692 indicating factorability. In each analysis, the scree plot test suggested 1 factor and the loading on to a single factor ranged from 0.79–0.94. The amount of variance explained by a single factor ranged from 78–83% (see Table 4).

Table 5
Descriptive statistics for QOL and overall FQOL

	Total			Subsample with SB			Comparison subsample
	$M$	SD	Range	$M$	SD	Range	$M$	SD	Range
AYA
Own QOL	87.57	17.44	15–100	86.05	20.90	15–100	89.03	12.90	50–100
Family QOL	88.06	16.83	0–100	88.31	19.77	0–100	87.61	13.25	50–100
Parent QOL	86.82	17.52	0–100	87.68	19.56	0–100	85.73	15.06	50–100
AYA Overall FQOL Scale	87.40	15.28	8–100	87.35	17.68	8–100	87.46	12.21	50–100
Parent
AYA QOL	85.69	14.79	25–100	79.79	16.42	25–100	92.49	8.66	50–100
Own QOL	84.73	15.00	20–100	83.54	15.72	20–100	86.10	14.09	30–100
Family QOL	86.44	14.40	20–100	84.08	16.52	20–100	89.15	10.94	30–100
Parent Overall FQOL Scale	85.62	13.23	27–100	82.47	14.77	27–100	89.25	10.10	47–100

Note. Total parent sample $N=$ 209; Subsample with SB $n=$ 112; Comparison Subsample $n=$ 97; Total AYA sample $N=$ 197; Subsample with SB $n=$ 103; Comparison subsample $n=$ 94.

Parents reported high FQOL (parent report SB group $m=$ 82.47, $\sigma=$ 14.77; comparison group, $m=$ 89.25, $\sigma=$ 10.10). Parents with AYA with SB had slightly lower FQOL with greater variance. This difference was significant ( $t=-$ 3.915; $p<$ 0.001) but relatively small (e.g., less than 7 points on the 100-point scale). The AYA with and without SB also reported high FQOL and there was no significant difference between groups ( $t=-$ 0.052; $p=$ 0.959) (see Table 5 for descriptive statistics for QOL and overall FQOL). Internal consistency reliability for the scale in total sample and subsamples was strong ( $\alpha=$ 0.86–0.90) for parents and AYA.

Parent report of FQOL was moderately correlated with their report of family satisfaction and resources ( $r=$ 0.51–0.57) and AYA report of FQOL was correlated with AYA report of family satisfaction ( $r=$ 0.39). These moderate correlations support FQOL as a related but different construct than family functioning measured by family satisfaction and resources.

The relationship of FQOL in parent and AYA pairs was poor to moderate with wide confidence intervals indicating poor agreement and variability between parent and AYA report of FQOL scale scores for the total and sub-samples: ICC 0.41, 95% CI [0.22–0.56] for total sample; ICC 0.38, 95% CI [0.10–0.58], for SB group; ICC 0.48, 95% CI [0.22–0.65] for comparison group. Agreement between parents and AYA was highest in the group without SB.

4. Discussion

Two approaches to FQOL measurement have been used in the literature: first, measures that tap domain-specific aspects of family life; and second measures that assess overall perspective of FQOL. The G-FQOLS, an example of the second, is a weighted perception of the domains important to the reporter about the family as a whole, which in this case is, an average of a family member’s perspectives of the individual, the child or parent, and their family’s quality of life. The nature of FQOL is a dynamic approach which facilitates parents’ and AYA’s report of important domains of life which can vary across time. A strength of the G-FQOLS is its focus on perception versus function.

In the current overall G-FQOLS instrument, perception of individual, other family members (either parent or child) and FQOL loaded onto one factor. Investigators might wonder about the close relationship of the three items. However, strong relationship between these items ( $r=$ 0.60–0.76) has been reported previously [10] and there is evidence that family is the predominate concept when evaluating both AYA and parent individual quality of life. In this study, when queried about the meaning of individual quality of life, family was the concept most commonly nominated by all participant groups (AYA with and without SB and their parents) (unpublished data). The “core” contribution of family in individual QOL supports the factor loadings for the G-FQOLS three items. It is interesting to note that family is rarely a domain included in individual QOL measures, especially those based on function.

In one study that used the FQOL-2006 Survey with both overall and domain-specific measures, the global single FQOL item was related ( $r=$ 0.62, $p<$ 0.001) to the composite domain-specific score of FQOL [24]. The overall shorter measure is highly related to domain-specific total score. Therefore, using single items (or in this case 3 items) may be an effective screening tool. Conducting clinical research with instruments measuring predetermined domains is an efficient way to identify need for specific support services. Assessing overall FQOL may help us understand factors that contribute to FQOL. If family member reports a relatively poor FQOL, then further assessment of factors contributing to their perception would be warranted. It is important to distinguish between this measure of global FQOL and family functioning, the latter being a process measure of how families operate rather than a measure of a rating from the perspective of a family member of their overall FQOL. Factors related to FQOL may vary in different environments or conditions. The G-FQOLS, as a potential outcome measure, provides a way to go beyond what has been identified as relevant, since these domains may change because of varied needs and resources over the life course. This approach facilitates the identification of modifiable factors to inform interventions for individuals and families.

Poor agreement between parent and AYA report of FQOL indicates measure is sensitive to differences in perspectives by reporter. Findings support importance of obtaining AYA appraisal from AYA, rather than by parent proxy. This instrument offers the capacity to assess discrepancies between family members which may identify a meaningful target for intervention. Further assessment of the meaning of FQOL for both parents and AYA can determine if family members have the same meaning of FQOL or prioritize the domains similarly. If AYA had moderate to severe intellectual disabilities, they may not have the capacity to appraise their own FQOL. In this instance, ratings by parent proxy might be used to assess FQOL.

Differences between groups with and without SB were evident in parents, but not in AYA data. Although differences were detected in parent report, those differences were small. The lack of difference by group with and without SB in AYA may indicate that the meaning of FQOL for AYA differs from that of parents. Parents may place more weight on aspects of life related to their experiences as caregivers. In families of children with moderate to severe intellectual disabilities, parents’ appraisal of FQOL may be influenced by continued responsibility for care as their AYA transitions to adulthood.

Another explanation for the lack of difference between the report of FQOL in AYA’s with and without SB may be due to the “disability paradox” seen when adults with disabilities report a high QOL despite limitations in activities of daily living, difficulty in social roles, and experience with persistent discrimination [25]. The perception of AYA with SB may be based on meaning and values different from individuals without disability. While professionals might expect lower QOL in youth with SB, there is conflicting evidence about QOL in youth with SB in other than the physical domain [10, 26]. The universal finding of lower physical QOL is based on tools that use function such as items about walking or running to assess QOL/health-related QOL(HRQOL). Murray et al. acknowledge their findings of lower HRQOL could be a result of the measure they used where physical domain was measured by function [27]. Among QOL/HRQOL researchers studying individuals with disabilities, there is strong support for measurement of HRQOL based on perception (the WHO definition) rather than function.

Either a comprehensive FQOL instrument, which addresses domain-specific issues in a family who has a member with a disability, or a brief measure of overall perception can be useful in different research or clinical settings, depending on the purpose of the project. Future research may use rigorous mixed methods to develop/refine an item pool and test content validity for a domain-specific measure of FQOL in SB. This would help researchers/clinicians identify domain-specific targets for intervention. Additionally, item response theory may be applied to extend our measure of FQOL to predict expected ratings given specified parameters or our measure may be converted to a Patient-Reported Outcomes Measurement Information System (PROMIS ${}^{\@setsize{\scriptsize}{8pt}{\viipt}{\@viipt}\textregistered}$ ) metric to enable comparisons to other global health or well-being measures across studies. This study supported the use of a new brief summative instrument, the G-FQOLS. The analysis of total and subsamples provides evidence of construct validity and internal reliability of the G-FQOLS. This global appraisal of FQOL reflects parent’s and AYA’s personal weighting of domains important to them. Although domains or their weight may change over time, the overall rating is an indicator of the family’s overall quality of life.

5. Limitations

As a self-report measure, there is potential for social desirability response bias. Previous research using similar items in the SB population found scores of QOL/FQOL single item means between 72–77 [10], slightly lower than our current findings. The G-FQOLS items were administered in a set of 10 single-items including measures of stress, health and responsibility for care which might have facilitated a response bias. However, all items had a broad response range, supporting respondents’ ability to determine differences between items. For example, the mean stress of everyday life for parents of AYA with and without SB specifically was 53.33 (SD 26.32) [6]. However, in future testing, to protect against any response bias, FQOL items should be interspersed with items using a different response pattern. Test-retest reliability and sensitivity to change have not been determined and are needed prior to use in intervention studies. Another limitation is the lack of diversity in the sample tested. This sample was mostly Caucasian and resided primarily in Midwest (Wisconsin, Indiana, Ohio, and Virginia). Evaluation in a more diverse sample is indicated.

6. Implications for practice and research

This measure may be useful in primary or specialty care settings to identify families at-risk who need further assessment and intervention. Evaluation of families can begin with a preliminary screening of G-FQOLS. Although there is not enough evidence to determine cut points using the G-FQOLS, clinicians can either use the tool as a screening tool or as an entry to discussion. Further assessment and discussion might be indicated when the G-FQOLS score is 2.0 standard deviations below the mean of the evidence to date (e.g. values of 56.84–59.16 using data from this study). Alternatively, when used primarily as an entry to discussion, probes used could include: “What does FQOL mean to you? What are the barriers to achieving your FQOL goals? What would make your FQOL better?”. Additional evaluation could follow with a standardized assessment for area(s) of concern such as depressive symptoms, pain, or other issues.

Measuring FQOL over time would enable clinicians and researchers to understand developmental influences on FQOL. While most positive appraisals may occur in the younger years when discrepancies for development between themselves and peers are smallest, parents’ future expectations for their family members may influence their FQOL. Families with difficulty in typical tasks of family life in the context of chronic illness may be identified with a substantial decrease in score or low G-FQOLS score. This should lead to follow-up assessment. FQOL measurement may be particularly helpful during times of transition when additional independence is expected (e.g., transitioning to adolescence or emerging adulthood). Changes in G-FQOLS scores over time, when experiencing acute illnesses, secondary complications, or hospitalizations would be indications for specific assessment. Understanding perceptions of FQOL may help develop targeted family interventions over the life course.

Further research is needed to establish G-FQOL test-retest reliability and sensitivity to change. If established, the instrument can be used to monitor and evaluate future family-centered interventions in research and practice. Research to date has used the measure to quantify FQOL from a family member’s perspective. Future research should also address the use of the instrument with multiple family members.

7. Conclusion

This study provides preliminary evidence that supports use of a simple 3-item global measure of FQOL in research and practice. The G-FQOLS rating may be used for additional assessment of the components of FQOL in parents and AYA. If G-FQOLS is low, it may prompt referral to support services or screening for mental health concerns in both pediatric and specialty care settings.

Footnotes

Acknowledgments

The original study was supported by a grant from the Association of University Centers on Disabilities (AUCD) in cooperative agreement with the Centers for Disease Control and Prevention (CDC)’s National Center on Birth Defects and Developmental Disabilities (NCBDDD). In addition, the authors thank the families for participation.

Conflict of interest

The authors have no conflict of interest to report.

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A Global Family Quality of Life Scale: Preliminary psychometric evidence

Abstract

PURPOSE:

METHODS:

RESULTS:

CONCLUSION:

Keywords

1. Background

1.1 Overview of FQOL measurement

1.2 Domain-specific measures

1.2.1 Beach FQOL Scale

Table 1 Summary of Domain-Specific and Overall measures of FQOL

1.3 Global measures of overall FQOL

1.4 Summary of studies using FQOL measures

2. Methods

2.2 Instrument

Table 4 Summary of Factor Analysis, Reliability, and Parent-AYA Agreement for 3-item FQOL Scale

3. Results

Table 5 Descriptive statistics for QOL and overall FQOL

5. Limitations

6. Implications for practice and research

7. Conclusion

Footnotes

Acknowledgments

Conflict of interest

References

Table 1
Summary of Domain-Specific and Overall measures of FQOL

Table 4
Summary of Factor Analysis, Reliability, and Parent-AYA Agreement for 3-item FQOL Scale

Table 5
Descriptive statistics for QOL and overall FQOL