Journal of Pediatric Psychology Advance Access published March 21, 2015
Journal of Pediatric Psychology, 2015, 1–9 doi: 10.1093/jpepsy/jsv009 Original Research Article
Children’s Food Allergies: Development of the Food Allergy Management and Adaptation Scale
Downloaded from http://jpepsy.oxfordjournals.org/ at Florida Atlantic University on May 24, 2015
Mary D. Klinnert,1,2 PHD, Elizabeth L. McQuaid,3,4 PHD, David A. Fedele,5 PHD, Anna Faino,1 MS, Matthew Strand,6,7 PHD, Jane Robinson,8,9 PHD, Dan Atkins,8,9 MD, David M. Fleischer,8,9 MD, Jonathan O’B. Hourihane,10 MD, Sophia Cohen,1 BA, and Hannah Fransen,11 MSW 1
Department of Pediatrics, National Jewish Health, 2Department of Psychiatry, University of Colorado School of Medicine, 3Department of Psychiatry and Human Behavior, Alpert Medical School, Brown University, 4Bradley/ Hasbro Children’s Research Center, 5Department of Clinical & Health Psychology, University of Florida, 6Division of Biostatistics & Bioinformatics, National Jewish Health, 7Department of Biostatistics & Informatics, University of Colorado School of Public Health, 8Department of Pediatrics, University of Colorado School of Medicine, 9Allergy Section, Children’s Hospital Colorado, 10Department of Paediatrics and Child Health, University College Cork, and 11 Graduate School of Social Work, University of Denver All correspondence concerning this article should be addressed to Mary D. Klinnert, PHD, National Jewish Health, 1400 Jackson St., Denver, CO 80206, USA. E-mail: [email protected] Received August 22, 2014; revisions received January 19, 2015; accepted January 21, 2015
Abstract Objective Develop a measure that evaluates effective pediatric food allergy (FA) management, child and parent FA anxiety, and integration of FA into family life. Methods A semistructured family interview was developed to evaluate FA management using a pilot sample (n ¼ 27). Rating scales evaluated eight dimensions of FA management (FAMComposite), child anxiety, parent anxiety, and overall balanced integration (BI). Families of children with IgE-mediated food allergies (n ¼ 60, child age: 6–12) were recruited for interview and rating scale validation. Results FAMComposite was correlated with physician ratings for families’ food avoidance and reaction response readiness. FA anxiety was correlated with general anxiety measures for children, but not parents. Parents’ FA anxiety was correlated with expectations of negative outcomes from FA. Low BI was associated with poor quality of life and negative impact on family functioning. Conclusions Preliminary analyses support Food Allergy Management and Adaptation Scale validity as a measure of family adaptation to pediatric FA. Key words: family adaptation; management; pediatric food allergy; psychosocial adjustment.
Children’s food allergies are increasingly common, with current U.S. prevalence estimates ranging up to 8% (Gupta et al., 2011). Food allergy (FA) reactions that are mediated by immunoglobulin E (IgE) can cause hives, breathing difficulties, and gastrointestinal symptoms. Reactions can progress to anaphylaxis, involving respiratory, cardiovascular, and/or gastrointestinal symptoms (Boyce et al., 2010). Although rare, anaphylactic reactions can result in death if not treated promptly and
effectively (Sicherer & Sampson, 2006). The negative impact of FA on families can be pervasive. Families of children diagnosed with FA report disruptions in daily activities, increased stress and symptoms of anxiety and depression, and lower quality of life (QoL) (Cummings, Knibb, King, & Lucas, 2010). Effective family management of food allergies and psychosocial adjustment to the chronic stresses of food allergies are key components of families’ adaptation.
C The Author 2015. Published by Oxford University Press on behalf of the Society of Pediatric Psychology. V
All rights reserved. For permissions, please e-mail: [email protected]
1
2
FA Management
Impact of FA on Children and Parents Parents and children with food allergies report lower QoL when compared with controls or other illness groups such as rheumatologic disease and insulin-dependent diabetes mellitus (Avery, King, Knight, & Hourihane, 2003; Primeau et al., 2000). A FA-specific questionnaire regarding parents’ QoL, the Food Allergy Quality of Life–Parent Burden (FAQL-PB), documented major impacts on parents owing to daily demands in areas such as meal preparation and activity planning, as well as extensive worry and anxiety about potentially life-threatening reactions (Cohen, Noone, Munoz-Furlong, & Sicherer, 2004). The parent-report Food Allergy Impact Scale (FAIS) revealed elevated stress for parents and children, multiple effects on daily activities such as meal preparation and eating out, and limitations on participation in activities not related to food (e.g., peer socialization, home schooling owing to FA) (Bollinger et al., 2006). Anxiety is a central feature of reduced QoL for parents and children with food allergies (DunnGalvin, Flokstra-de Blok, Burks, Dubois, & Hourihane, 2008). Parents’ outcome expectations for serious FA reactions or death were strongly correlated with poor QoL for parents (Cohen et al., 2004) and for children (DunnGalvin et al., 2008). Expectations for death are surprisingly common, possibly representing a misperception of the prevalence of FA fatalities among children (Umasunthar et al., 2013). Reports have been mixed regarding general anxiety levels among parents and children with food allergies (Cummings, Knibb, Erlewyn-Lajeunesse, et al., 2010; LeBovidge, Strauch, Kalish, & Schneider, 2009; Roy & Roberts, 2011), but also indicate that mothers’ anxiety and stress levels are correlated with ratings of poorer QoL (Roy & Roberts, 2011) and greater psychological distress for their children (LeBovidge et al., 2009). For a subset of families, anxiety relating to FA can be debilitating for both parents and children (Manassis, 2012; Mandell, Curtis, Gold, & Hardie, 2005; Roy & Roberts, 2011). Few studies have addressed how families cope with the daily burdens and emotions elicited by pediatric food allergies. The Food Allergy Parent Questionnaire was developed to assess psychosocial impact and coping strategies used by parents of children with FA (LeBovidge et al., 2006) but psychometric and conceptual shortcomings limited its usefulness as a measure of parents’ coping. It is unknown how families’ proficiency with FA management is related to their emotional and
behavioral adjustment or to their overall adaptation to children’s food allergies. To our knowledge, no studies have assessed the adequacy of families’ FA management skills in relation to the behavioral coping strategies required for positive adaptation to children’s food allergies.
Family Adaptation to FA Positive family adaptation to childhood FA requires achieving a balance between managing a potentially life-threatening condition that requires vigilance and preparedness, and simultaneously modulating anxiety levels and maintaining developmentally appropriate activities for children and families (Mandell et al., 2005). Poor family adaptation to food allergies can include taking excessive measures that limit appropriate developmental and social activities, or taking appropriate preventive measures, but becoming overwhelmed by the anxiety that accompanies constant vigilance (Klinnert & Robinson, 2008). Like other pediatric chronic illnesses, parents initially bear primary responsibility for FA management, but within the family system, each stage of children’s development is associated with greater responsibility and more FA self-management skills, learned through interactions and collaboration with parents as they manage and cope with the FA over time (Kazak, 1989). We propose that family adaptation to food allergies operates through multiple pathways (Figure 1). Child factors such as age and developmental level dictate the parental level of concern and involvement. Caregiver characteristics, such as age, marital status, and family socioeconomic status (SES), influence material and psychological resources available for effective illness management. Disease factors such as duration of illness, reaction history, and comorbid allergic conditions have shown mixed associations with FA anxiety and QoL (Roy & Roberts, 2011), while number of food allergies has consistently been associated with reduced parent QoL and negative family impact (Bollinger et al., 2006; Cohen et al., 2004). The relationship between parent and child FA management strategies and anxiety has rarely been investigated. We expected that variability within and interactions among these domains contribute to families’ levels of FA management and their maintenance of emotional equilibrium and normative child and family activities, and that positive adaptation to FA occurs when families are able to integrate the demands and stresses into their lives in a balanced manner, termed balanced integration (BI; McQuaid, Walders, Kopel, Fritz, & Klinnert, 2005).
Current Study To date, no measure exists that targets the complex interplay of FA management and psychosocial adjustment required for family adaptation to FA. The aims of this study were to develop and provide preliminary validation for an interview-based measure of family management of children’s food allergies that takes into account specific management strategies, including food avoidance and reaction response readiness, balanced with appropriate vigilance without excessive anxiety or unnecessary restrictions on child and family activities. Our measurement approach, modeled on a validated instrument for assessing families’ asthma management systems (McQuaid et al., 2005), used a semistructured interview and pertinent rating scales. Although potentially time intensive, semistructured family interviews have been shown to provide unique variance in explaining child health outcomes beyond self-report measures (McQuaid et al., 2005); this may be owing to the greater depth of information obtained and the potential to clarify discrepant perspectives among family members.
Downloaded from http://jpepsy.oxfordjournals.org/ at Florida Atlantic University on May 24, 2015
Appropriate management of a child’s food allergies necessitates food avoidance across home and social contexts, and response readiness, including recognizing symptoms of FA reactions and managing them with recommended medication (Sicherer & Sampson, 2006). FA management can be a complex process that entails lifestyle changes and poses multiple challenges for the family system. Parents often have difficulties correctly identifying causal foods in products (Joshi, Mofidi, & Sicherer, 2002) and may use inadequate strategies to guide their children’s food avoidance in activities outside of the home (Kapoor et al., 2004). Furthermore, parents often lack the skills to recognize symptoms of anaphylaxis, may not have medication readily available, and lack the knowledge or confidence to treat exposures appropriately (Arkwright & Farragher, 2006). Although several aspects of FA management have been evaluated among parents, no measure comprehensively evaluates the adequacy of parents’ FA management skills across requisite behavioral domains, and, to our knowledge, no measures have evaluated key dimensions of children’s FA self-management.
Klinnert et al.
Development of the FAMAS
3
Rating scales were constructed to capture a range of functioning for each of three constructs essential within our model of family adaptation to children’s food allergies: (1) multiple scales for assessment of FA management behavioral domains (Sicherer & Sampson, 2006), with child-specific scales for areas within children’s realm of responsibility (DunnGalvin, Gaffney, & Hourihane, 2009); (2) scales for assessment of FA anxiety for individual parents and children (Mandell et al., 2005); and (3) a global scale to assess how well families’ were balancing FA management demands with the psychological well-being and developmental needs of their children as well as themselves (McQuaid et al., 2005). We developed this measure for families of children aged 6–12 years with IgE-mediated food allergies because it is during the school-age years that children solidify self-management skills, develop specific styles for coping with food allergies, and accept or reject the incorporation of food allergies into their identity (DunnGalvin et al., 2009). Such an instrument could serve to identify families in need of clinical services owing to problems with FA adaptation, as well as relevant targets for future interventions. To test the validity of the Food Allergy Management and Adaptation Scale (FAMAS), we tested each of the three key constructs using multiple strategies. We hypothesized (1) the FA management component, composed of eight subscales, would (1a) demonstrate adequate internal consistency across the eight subscales; (1b) correlate with independent ratings of family FA management made by physicians who specialize in clinical care for children with FA; and (1c) be associated with an observational assessment of parents’ injectable epinephrine proficiency; (2) FA-specific anxiety would (2a) be moderately correlated with general anxiety measures for both parents and children; and (2b) mothers’ FA Anxiety would be correlated with outcome expectations for death; and (3) BI would be significantly correlated with (3a) lower FA family impact measured by the FAIS and (3b) better parent FA QoL, measured by the FAQL-PB.
Methods The FAMAS development and validation studies were conducted at a tertiary care center in a metropolitan setting and were approved by the institutional review board. Parental informed consent and child assent were obtained for both the pilot and the validation phases of the study.
FAMAS Interview and Scale Development Development of the FAMAS interview and rating scales was based on empirical reports regarding children and families’ experiences with FA, consultation with board-certified allergists regarding principles of FA management, pilot studies of parent FA QoL and outcome expectations (Klinnert & Robinson, 2008), and qualitative data obtained from focus groups (unpublished). The interview and rating scales were developed with reference to the constructs identified a priori as fundamental to FA adaptation. For each construct, we generated open-ended stem questions with follow-up probes designed to elicit meaningful behaviors and vignettes from children and parents regarding FA management (e.g., avoiding foods), subjective experience (e.g., emotional response to food allergies), objective experience (e.g., prior use of injectable epinephrine), and psychosocial adjustment (e.g., impact on family social activities). Based on clinical recommendations and effective behavioral strategies for FA management (Boyce et al., 2010), an initial series of 9point rating scales was developed for key constructs and behavioral domains, with the mid-point score (5) representing “adequate” functioning for respective domains. For each scale, core features were identified to consider when rating, and descriptive anchor points were developed. The pilot phase involved video-recorded interviews with 27 children with FA (M age ¼ 8.4, SD ¼ 1.8, range: 6–12 years) and their caregivers, who represented various levels of FA management and adaptation. Following each interview, each team member independently reviewed video recordings with attention to interview
Downloaded from http://jpepsy.oxfordjournals.org/ at Florida Atlantic University on May 24, 2015
Figure 1. Conceptual model of family adjustment to food allergies.
4
Klinnert et al.
Table I. Content and Scoring of the Food Allergy Management and Adaptation Scale (FAMAS) FAMAS subscales FA management composite FA knowledge
Symptom assessment
Family response readiness Child response readiness
Low, middle, and high anchor points
Knowledge of basic mechanisms of FA, including relevant organ systems, modes of exposure, risks for reaction or death, relationship with asthma, natural history. Knowledge of reaction symptoms, own child’s symptom pattern, gradation of symptom severity from mild to anaphylaxis; specific indications of anaphylaxis
1. Poor understanding 5. Basic elements, but one significant gap 9. Excellent grasp of FA 1. Limited awareness 5. Knows primary symptoms of FA reaction 9. Comprehensive understanding of FA reactions 1. Unprepared for reaction, minimization 5. Plan less than perfect, but could save child 9. Systematic, coherent plan 1. Denies or hides symptoms 5. Acknowledges symptoms/sufficient plan 9. Alert to symptoms, coherent/developmentally appropriate plan 1. Limited/hopeless regarding food avoidance 5. Adequate understanding, some inconsistencies 9. Coherent strategy across settings 1. No strategies/resistant to food avoidance 5. Good understandings of avoidance basics 9. Advocates for self in multiple settings 1. Not readily available 5. Generally on hand but have not practiced 9. Always at hand and have practiced 1. Do not inform or prepare 5. May be vague across alternate caregivers 9. Written and verbal reaction response plan 1. No evidence that FA produces anxiety 5. Moderate anxiety about FA/reactions 9. Clinically significant anxiety 1. Expresses no anxiety due to FA 5. Moderate anxiety about FA/reactions 9. Extreme avoidance/fear of dying 1. Imbalanced/child at risk/high FA burden 5. Adequate management/less burden 9. Balance between management/psychosocial adjustment
Family members’ preparation for managing reactions, including response appropriate to symptoms; quality and availability of action plan for future reactions. Child’s preparation for managing reactions, including response appropriate to symptoms (e.g. notifying adult, cooperating with treatment); quality and availability of action plan; developmentally appropriate.
Family food avoidance
Strategies for food avoidance at and away from home; awareness of potential for accidental exposure; knowledge and use of food labels; awareness of cross-contamination.
Child food avoidance
Developmentally appropriate strategies for food avoidance; awareness of potential accidental exposure; knowledge and use of food labels; awareness of cross-contamination. Have current, filled prescriptions for injectable epinephrine and antihistamines; medications stored appropriately, available at all times, and in all environments; availability of asthma medications if needed. All alternate caregivers are informed of child’s FA, food avoidance strategies, knowledge of symptoms and action plan, medication availability; preparedness to respond to symptoms, administer epinephrine. Anxiety levels self-reported by parent; anxiety revealed by responses regarding specific management strategies and experiences with reactions and by FA outcome expectations. Child’s self-reported anxiety level; anxiety revealed by responses regarding specific management strategies and experiences with reactions and by FA outcome expectations. Balance of attention to FA management and other developmental and family issues (e.g., emotional adjustment, participation in extracurricular and family activities).
Medication availability Alternate caregivers Parent anxiety
Child anxiety
Balanced integration
Note. FA ¼ food allergy. Each FAMAS subscale uses a 9-point scale, with higher scores indicating high (e.g., anxiety) or better (e.g., FA management scales); scores below the mid-point of 5 indicate inadequate FA management. Composites are a mean of the subsequently listed subscales.
structure and flow, and used preliminary rating scales to make initial ratings. Team meetings were then used to review team members’ ratings to (1) achieve consensus ratings consistent with the purpose of the scales, (2) use rating disagreements to identify unclear scale items or interview questions, (3) make indicated refinements to interview questions and follow-up probes and to rating scales. Finally, we discussed changes to improve the interview, such as rewording of questions, ordering of questions, and overall flow. The initial pilot phase ended when ratings were consistent and no further major refinements to the interview or rating scales were necessary to capture relevant content (Supplementary for FAMAS Interview). An abbreviated version of the final FAMAS 9-point rating scales is presented in Table I. Adequacy of FA management is assessed with eight dimensions that can be combined by taking the mean of the eight ratings to create an FA Management Composite (FAMComposite). Parent and Child FA Anxiety are assessed and rated for individual parents and children. Children’s responses are
rated separately for food avoidance, reaction response readiness, and FA-specific anxiety, with each child scale incorporating expectations consistent with child’s developmental level. BI represents a global judgment of how well the family has integrated requisite FA management behaviors and skills with other aspects of child and family life, avoiding unwarranted restrictions on family members’ behavior, and maintaining normative age- and role-appropriate functioning for family members.
Participants for FAMAS Validation Study A second, new sample of families was recruited via physician referrals from the hospital pediatric outpatient clinic, local private allergy practices, and mailings to members of a local FA support group. Eligibility for the study required that families have a child 6–12 years old with physician-documented FA, including a verified history of food reaction and positive FA testing within 18 months before the study visit. Children with unconfirmed FA, non-English-
Downloaded from http://jpepsy.oxfordjournals.org/ at Florida Atlantic University on May 24, 2015
Description/specifications
Development of the FAMAS speaking families, and children with severe developmental delay were excluded.
Study Procedures
Measures Physician Global Ratings of Family Food Avoidance and Reaction Response Readiness Two board certified allergists using clinical judgment, without review of the FAMAS rating scales, independently reviewed video recordings of interviews edited to include only information relevant to FA management. Physicians used 5-point scales to make global ratings of family food avoidance and family reaction response readiness (Supplementary for physician rating guidelines). For reliability assessment, both physicians rated the edited portion of 77% (n ¼ 46) of the interview recordings. Interrater reliability, computed as intraclass correlations (ICC), was .84 for reaction response readiness and .76 for family food avoidance. EPI-Demo The videotaped segments documenting the caregiver’s response to the epinephrine task were rated by two study personnel trained to reliably score whether the caregiver (1) knew the correct site to inject epinephrine, (2) removed the safety cap, (3) applied enough pressure to trigger the device, (4) left the needle in the skin for recommended time after the device was triggered, and (5) listed breathing problems or faintness as indications for use (Arkwright & Farragher, 2006). Analyses for the present study used the sum of correctly performed items, with higher scores corresponding to more appropriate device use, as well as a pass/fail score, with a pass based on correct demonstration of all five components. Interrater reliability was computed for both scores: sum correct (ICC ¼ .92) and pass/fail (ICC ¼ .93).
State-Trait Anxiety Inventory The State-Trait Anxiety Inventory (STAI) is a 40-item instrument that measures an individual’s stable trait and transient state anxiety. The STAI provided self-report data on general anxiety levels. Extensive validity testing suggests that the STAI has good concurrent, convergent, divergent, and construct validity (Speilberger, 1970; Spielberger, Gorsuch, Lushene, Vagg, & Jacobs, 1973). STAI State and Trait t scores were used in this study. The STAI demonstrated excellent internal consistency for mothers (n ¼ 57, Cronbach’s a ¼ .94 and .91), and for fathers (n ¼ 14, a ¼ .83 and .90) for the State and Trait subscales, respectively. State-Trait Anxiety Inventory for Children The STAI-C is a 40-item questionnaire for which children respond to statements that begin with “I feel . . . ” by choosing one of three answers reflecting varying levels of comfort (e.g., “very calm,” “calm,” “not calm”) (Spielberger et al., 1973). The STAI-C was used to provide data on the children’s general anxiety. STAI-C State and Trait t scores were used in this study. Adequate reliability and validity have been demonstrated (Spielberger et al., 1973). The STAI-C has been used with first and second graders by having it read to them and their response recorded (Papay & Hedl, 1978), a procedure that we followed. Internal consistency in the current study was good (a ¼ .86 and .83 for State and Trait subscales, respectively). Multidimensional Anxiety Scale for Children The MASC is a 39-item self-report clinical measure designed to assess a broad range of anxiety symptoms for children aged 8–18 years. Children are asked to rate items using a 4-point scale ranging from “never true” to “often true” (March, 1997). Greater MASC scores indicate higher levels of anxiety. The MASC has demonstrated acceptable psychometric properties (March, 1997). MASC Total Anxiety t scores were used in the current study. For the current study, internal consistency was examined separately for the younger children and those 8 years. Cronbach’s a for the younger children was .83, and .85 for the 8–12-year-olds. For the entire sample, a ¼ .85. Food Allergy Independent Measure The Food Allergy Independent Measure (FAIM) consists of four questions rated on 7-point Likert scales, and assesses the parent’s expectation of outcome for the child with FA (DunnGalvin et al., 2008; van der Velde et al., 2010). Parents rate for their child the chance of: (1) accidental exposure, (2) severe reaction when accidentally exposed, (3) dying when accidentally exposed, and (4) responding effectively when accidentally exposed. For the current study, ratings for question 3 concerning parent perception regarding chance of child’s death owing to FA was included as a concurrent measure of parent FA specific anxiety. Food Allergy Impact Scale The FAIS is a 32-item questionnaire with eight subscales that evaluate the caregiver’s perception of the impact of the child’s FA on aspects of daily family activities (e.g., meal preparation, family social activities); subscale scores were related to number of FA per child (Bollinger et al., 2006). For each item, caregivers rate the impact of the child’s FA using 7-point Likert scales, indicate if FA prevents child or family engagement in the queried activity (scored as 8), or mark that the item does not apply to their child (e.g., for FA impact on daycare, child does not attend daycare; scored as missing). FAIS
Downloaded from http://jpepsy.oxfordjournals.org/ at Florida Atlantic University on May 24, 2015
After parental informed consent and child assent were obtained, the parent(s) and child participated in an hour-long, video recorded interview conducted by one of two clinical psychologists. At the end of the interview, the child was separated from the parent(s) for questionnaire completion, supervised by research assistants (RAs). Children aged 8–12 completed questionnaires independently, while for 6- and 7-year-olds, RAs read the questions for the State-Trait Anxiety Inventory for Children (STAI-C) and Multidimensional Anxiety Scale for Children (MASC) questions and recorded the children’s responses. With the child absent, parents were asked additional questions privately that might have been uncomfortable in the presence of the child (e.g., “on a scale from 1 to 5 where 1 is ‘don’t worry at all’ and 5 is ‘get really scared,’ how worried or nervous do you get when your child is having FA symptoms?”) followed by open-ended questions to assess the parent’s emotions in response to FA reactions. An RA then videotaped the parent demonstrating the use of a trainer version of an auto-injectable epinephrine device (Epi-Demo procedure). Parents were allowed to choose among several devices to ensure they were using a familiar one. After their demonstration, parents were asked to describe indications for administering epinephrine to their child. Finally, parents completed questionnaires. Following study visits, all video-recorded interviews were viewed and rated independently using the scale guidelines by research team members. Raters were three pediatric psychologists (two of whom also conducted interviews) and two RAs with training in FA management and behavioral health. Ratings were recorded for reliability calculation before discussion of rater disagreements, and final consensus scores were generated.
5
6 subscale scores were calculated by taking the mean of item ratings. Responses were required for at least half of constituent subscale items; otherwise subscale scores were considered missing. Internal consistency for the FAIS was adequate for this sample, with seven of eight subscales exceeding Cronbach’s a of .60 for mothers (a ¼ .54–.96; caregiver-supervised social activities, a ¼ .54), and seven of eight subscales exceeding a of .85 for fathers (a ¼ .49–.99; family relations, a ¼ .49).
Overview of Statistical Analyses Interrater reliability was assessed with two-way, single-measures ICC based on consistency in response for Likert-scale items, and based on absolute agreement for dichotomous items. For FAMAS subscales, pairwise ICCs were calculated for each rater compared with consensus scores; the mean and range of raters’ ICCs were calculated for each scale. Internal consistency was determined using Cronbach’s alpha on standardized variables. FAMAS validity testing included a comparison of FAMComposite scores for parents who passed versus failed the Epi-Demo, using an independent samples t test with unequal variances, and Cohen’s d was calculated as a measure of effect size. Spearman correlation coefficients were initially used to assess relationships between FAMAS scales and validity measures with continuous distributions. Descriptive analyses revealed correlations among number of FAs, age, and scale scores. Multiple linear regressions were used to regress the FAMAS subscales on validity measures, adjusting for child age and total number of FA. The multiple scale scores of the FAIS, which were intercorrelated, were entered together in a model after multicollinearity was examined and found not to be a limiting factor. Analyses were performed using JMP 10.00, SPSS version 22 and SAS version 9.3.
Results The validation sample included 60 families with physiciandocumented FA (Table II). Twenty percent of children had one FA, 27% had two, and the remainder had three or more documented FAs. Most children (77%) were accompanied by mothers, 18% by both parents, and three children (5%) by their fathers. All mothers present for the interview identified themselves as the primary FA caregiver. Validity was examined for the 57 families with mothers present. Mean FAMAS subscale scores were generally high relative to the conceptually determined a priori scale midpoint (5 on a 9-point scale), particularly for the subscales comprising the FAMComposite (Table III). The mean score for the FAMComposite was 6.6 (SD ¼ 1.3). Mean scores for the anxiety subscales and BI fell between scale points 5 and 6. Interrater reliability was excellent for FAMAS subscales (ICC range: .91–.98; Table III).
Table II. Participant and Family Characteristics Variable
Mean 6 SD or N (%)
Child age Child gender Male Female Child race Asian Black White More than one race Child ethnicitya Hispanic or Latino Not Hispanic or Latino Child’s age at diagnosis (years) Number of food allergies Number of families reporting anaphylaxis Number of anaphylactic reactions Child diagnosed with other allergic disease Asthma Atopic dermatitis Caregiver(s) participating in study Mother only Father only Both parents Mother’s highest level of education Less than college degree College or graduate degree Father’s highest level of education Less than college degree College or graduate degree Parents’ marital status Married Not married
8.7 6 1.8 39 (65) 21 (35) 1 (2) 2 (3) 51 (85) 6 (10) 8 (14) 50 (86) 2.3 6 2.6 3.0 6 1.8 33 (55) 1.7 6 1 35 (58.3) 27 (45) 46 (77) 3 (5) 11 (18) 5 (8) 55 (92) 11 (18) 49 (82) 55 (92) 5 (8)
Note. aEthnicity data missing for two subjects.
Validity of the FAMAS Our first hypothesis regarding validity for the FAMComposite was supported. The FAMComposite had excellent internal consistency (a ¼ .91). FAMComposite scores differed for families who passed the Epi-Demo compared with those who failed, t(54) ¼ 3.73, p ¼ .0005 (t test based on unequal variances; Pass: M ¼ 7.31, SD ¼ 0.59; Fail: M ¼ 6.34, SD ¼ 1.33; Cohen’s d ¼ 1.13). Regression analyses showed that physician ratings of family Food Avoidance and of reaction Response Readiness as well as Epi-Demo scores were significantly related to FAMComposite after controlling for child age and number of FAs (Table IV). FAMAS ratings of Child FA Anxiety were significantly associated with STAI-C State and Trait scores and with MASC total anxiety t scores (Table IV); this supported Hypothesis 2, that child FA anxiety ratings were correlated with general anxiety scores. For mothers, Hypothesis 2 was not supported, as mother FA anxiety ratings were not significantly associated with STAI scores. However, mother FA anxiety was strongly associated with the FAIM query regarding expectation of death as FA outcome (Table IV). Finally, higher BI was significantly associated with better maternal FA QOL, measured with the FAQL-PB. Spearman’s raw correlations revealed relationships between higher BI and lower scores on a number of FAIS scales (Table IV). However, a regression model that included all FAIS scales showed that caregiver-supervised child social activities, family relations, and meal preparation were the significant predictors for BI scores.
Downloaded from http://jpepsy.oxfordjournals.org/ at Florida Atlantic University on May 24, 2015
Food Allergy Quality of Life-Parent Burden Questionnaire The FAQL-PB measures the impact of FA on health-related QoL of caregivers (Cohen et al., 2004). Using a 7-point scale, parents are asked to indicate how their child’s FA has limited or troubled them during activities of daily living (e.g., eating outside the home, sending child to camp). Higher scores indicate increasing caregiver burden. The FAQL-PB has demonstrated excellent internal consistency and test–retest reliability (Cohen et al., 2004). The FAQL-PB total score was used for the current study, and demonstrated excellent internal consistency for mothers, a ¼ .93 (n ¼ 57), and for fathers, a ¼ .95 (n ¼ 12).
Klinnert et al.
Development of the FAMAS
7
Table III. Descriptive Statistics and Interrater Reliability for Food Allergy Management and Adaptation Scale (FAMAS) Subscale Scores FAMAS subscales
Interrater reliabilitya
Scores: Entire sample (n ¼ 60)
Scores: Validation sample (n ¼ 57)
Mean
Mean (SD)
Median (Q1, Q3)
Mean (SD)
6.9 (1.3) 6.7 (1.1) 6.1 (1.7) 5.9 (1.7) 7.0 (1.4) 7.4 (1.6) 6.5 (2.1) 6.4 (1.9) 6.6 (1.3) 5.7 (1.9) 4.0 (1.4) 5.6 (1.6) 5.5 (1.8)
7.0 (6.0, 8.0) 7.0 (6.0, 7.0) 6.0 (5.0, 7.0) 6.0 (5.0, 7.0) 7.0 (6.5, 8.0) 8.0 (7.0, 8.5) 7.0 (5.5, 8.0) 7.0 (5.0, 8.0) 6.9 (5.9, 7.6) 6.0 (4.0, 7.0) 3.5 (3.0, 5.0) 5.0 (4.3, 7.0) 5.0 (5.0, 7.0)
Range
60 60 60 60 60 60 60 60
0.98 0.97 0.95 0.95 0.98 0.98 0.98 0.97
0.97–0.99 0.95–0.99 0.91–0.99 0.89–0.98 0.96–0.99 0.96–0.99 0.97–0.99 0.92–0.99
57 14 60 60
0.97 0.99 0.97 0.94
0.95–0.99 0.98–0.99 0.94–0.99 0.88–0.98
Median (Q1, Q3)
6.9 (1.3) 6.7 (1.1) 6.2 (1.6) 6.0 (1.6) 7.1 (1.4) 7.4 (1.5) 6.7 (2.1) 6.5 (1.9) 6.7 (1.2) 5.7 (1.9)
7.0 (6.0, 8.0) 7.0 (6.0, 7.0) 6.0 (5.0, 7.0) 6.0 (5.0, 7.0) 8.0 (7.0, 8.0) 8.0 (7.0, 9.0) 7.0 (6.0, 8.0) 7.0 (5.0, 8.0) 6.9 (6.1, 7.6) 6.0 (4.0, 7.0)
5.6 (1.7) 5.6 (1.8)
5.0 (5.0, 7.0) 5.0 (5.0, 7.0)
Note. FA ¼ food allergy. a Mean and range of pairwise intraclass correlations calculated for scores by each of five raters compared with consensus scores; number of raters varied for subscales. b Fathers’ FA anxiety scores not included in the validation sample owing to small sample size.
Table IV. FAMAS Concurrent Validity qa
bb
p
Construct
Validity measure
N
FA management composite
Physician global rating—food avoidance Physician global rating—response readiness EPI-demo: sum of passed items
57 57 57
0.75 0.69 0.43
1.407 1.039 0.565
Journal of Pediatric Psychology, 2015, 1–9 doi: 10.1093/jpepsy/jsv009 Original Research Article
Children’s Food Allergies: Development of the Food Allergy Management and Adaptation Scale
Downloaded from http://jpepsy.oxfordjournals.org/ at Florida Atlantic University on May 24, 2015
Mary D. Klinnert,1,2 PHD, Elizabeth L. McQuaid,3,4 PHD, David A. Fedele,5 PHD, Anna Faino,1 MS, Matthew Strand,6,7 PHD, Jane Robinson,8,9 PHD, Dan Atkins,8,9 MD, David M. Fleischer,8,9 MD, Jonathan O’B. Hourihane,10 MD, Sophia Cohen,1 BA, and Hannah Fransen,11 MSW 1
Department of Pediatrics, National Jewish Health, 2Department of Psychiatry, University of Colorado School of Medicine, 3Department of Psychiatry and Human Behavior, Alpert Medical School, Brown University, 4Bradley/ Hasbro Children’s Research Center, 5Department of Clinical & Health Psychology, University of Florida, 6Division of Biostatistics & Bioinformatics, National Jewish Health, 7Department of Biostatistics & Informatics, University of Colorado School of Public Health, 8Department of Pediatrics, University of Colorado School of Medicine, 9Allergy Section, Children’s Hospital Colorado, 10Department of Paediatrics and Child Health, University College Cork, and 11 Graduate School of Social Work, University of Denver All correspondence concerning this article should be addressed to Mary D. Klinnert, PHD, National Jewish Health, 1400 Jackson St., Denver, CO 80206, USA. E-mail: [email protected] Received August 22, 2014; revisions received January 19, 2015; accepted January 21, 2015
Abstract Objective Develop a measure that evaluates effective pediatric food allergy (FA) management, child and parent FA anxiety, and integration of FA into family life. Methods A semistructured family interview was developed to evaluate FA management using a pilot sample (n ¼ 27). Rating scales evaluated eight dimensions of FA management (FAMComposite), child anxiety, parent anxiety, and overall balanced integration (BI). Families of children with IgE-mediated food allergies (n ¼ 60, child age: 6–12) were recruited for interview and rating scale validation. Results FAMComposite was correlated with physician ratings for families’ food avoidance and reaction response readiness. FA anxiety was correlated with general anxiety measures for children, but not parents. Parents’ FA anxiety was correlated with expectations of negative outcomes from FA. Low BI was associated with poor quality of life and negative impact on family functioning. Conclusions Preliminary analyses support Food Allergy Management and Adaptation Scale validity as a measure of family adaptation to pediatric FA. Key words: family adaptation; management; pediatric food allergy; psychosocial adjustment.
Children’s food allergies are increasingly common, with current U.S. prevalence estimates ranging up to 8% (Gupta et al., 2011). Food allergy (FA) reactions that are mediated by immunoglobulin E (IgE) can cause hives, breathing difficulties, and gastrointestinal symptoms. Reactions can progress to anaphylaxis, involving respiratory, cardiovascular, and/or gastrointestinal symptoms (Boyce et al., 2010). Although rare, anaphylactic reactions can result in death if not treated promptly and
effectively (Sicherer & Sampson, 2006). The negative impact of FA on families can be pervasive. Families of children diagnosed with FA report disruptions in daily activities, increased stress and symptoms of anxiety and depression, and lower quality of life (QoL) (Cummings, Knibb, King, & Lucas, 2010). Effective family management of food allergies and psychosocial adjustment to the chronic stresses of food allergies are key components of families’ adaptation.
C The Author 2015. Published by Oxford University Press on behalf of the Society of Pediatric Psychology. V
All rights reserved. For permissions, please e-mail: [email protected]
1
2
FA Management
Impact of FA on Children and Parents Parents and children with food allergies report lower QoL when compared with controls or other illness groups such as rheumatologic disease and insulin-dependent diabetes mellitus (Avery, King, Knight, & Hourihane, 2003; Primeau et al., 2000). A FA-specific questionnaire regarding parents’ QoL, the Food Allergy Quality of Life–Parent Burden (FAQL-PB), documented major impacts on parents owing to daily demands in areas such as meal preparation and activity planning, as well as extensive worry and anxiety about potentially life-threatening reactions (Cohen, Noone, Munoz-Furlong, & Sicherer, 2004). The parent-report Food Allergy Impact Scale (FAIS) revealed elevated stress for parents and children, multiple effects on daily activities such as meal preparation and eating out, and limitations on participation in activities not related to food (e.g., peer socialization, home schooling owing to FA) (Bollinger et al., 2006). Anxiety is a central feature of reduced QoL for parents and children with food allergies (DunnGalvin, Flokstra-de Blok, Burks, Dubois, & Hourihane, 2008). Parents’ outcome expectations for serious FA reactions or death were strongly correlated with poor QoL for parents (Cohen et al., 2004) and for children (DunnGalvin et al., 2008). Expectations for death are surprisingly common, possibly representing a misperception of the prevalence of FA fatalities among children (Umasunthar et al., 2013). Reports have been mixed regarding general anxiety levels among parents and children with food allergies (Cummings, Knibb, Erlewyn-Lajeunesse, et al., 2010; LeBovidge, Strauch, Kalish, & Schneider, 2009; Roy & Roberts, 2011), but also indicate that mothers’ anxiety and stress levels are correlated with ratings of poorer QoL (Roy & Roberts, 2011) and greater psychological distress for their children (LeBovidge et al., 2009). For a subset of families, anxiety relating to FA can be debilitating for both parents and children (Manassis, 2012; Mandell, Curtis, Gold, & Hardie, 2005; Roy & Roberts, 2011). Few studies have addressed how families cope with the daily burdens and emotions elicited by pediatric food allergies. The Food Allergy Parent Questionnaire was developed to assess psychosocial impact and coping strategies used by parents of children with FA (LeBovidge et al., 2006) but psychometric and conceptual shortcomings limited its usefulness as a measure of parents’ coping. It is unknown how families’ proficiency with FA management is related to their emotional and
behavioral adjustment or to their overall adaptation to children’s food allergies. To our knowledge, no studies have assessed the adequacy of families’ FA management skills in relation to the behavioral coping strategies required for positive adaptation to children’s food allergies.
Family Adaptation to FA Positive family adaptation to childhood FA requires achieving a balance between managing a potentially life-threatening condition that requires vigilance and preparedness, and simultaneously modulating anxiety levels and maintaining developmentally appropriate activities for children and families (Mandell et al., 2005). Poor family adaptation to food allergies can include taking excessive measures that limit appropriate developmental and social activities, or taking appropriate preventive measures, but becoming overwhelmed by the anxiety that accompanies constant vigilance (Klinnert & Robinson, 2008). Like other pediatric chronic illnesses, parents initially bear primary responsibility for FA management, but within the family system, each stage of children’s development is associated with greater responsibility and more FA self-management skills, learned through interactions and collaboration with parents as they manage and cope with the FA over time (Kazak, 1989). We propose that family adaptation to food allergies operates through multiple pathways (Figure 1). Child factors such as age and developmental level dictate the parental level of concern and involvement. Caregiver characteristics, such as age, marital status, and family socioeconomic status (SES), influence material and psychological resources available for effective illness management. Disease factors such as duration of illness, reaction history, and comorbid allergic conditions have shown mixed associations with FA anxiety and QoL (Roy & Roberts, 2011), while number of food allergies has consistently been associated with reduced parent QoL and negative family impact (Bollinger et al., 2006; Cohen et al., 2004). The relationship between parent and child FA management strategies and anxiety has rarely been investigated. We expected that variability within and interactions among these domains contribute to families’ levels of FA management and their maintenance of emotional equilibrium and normative child and family activities, and that positive adaptation to FA occurs when families are able to integrate the demands and stresses into their lives in a balanced manner, termed balanced integration (BI; McQuaid, Walders, Kopel, Fritz, & Klinnert, 2005).
Current Study To date, no measure exists that targets the complex interplay of FA management and psychosocial adjustment required for family adaptation to FA. The aims of this study were to develop and provide preliminary validation for an interview-based measure of family management of children’s food allergies that takes into account specific management strategies, including food avoidance and reaction response readiness, balanced with appropriate vigilance without excessive anxiety or unnecessary restrictions on child and family activities. Our measurement approach, modeled on a validated instrument for assessing families’ asthma management systems (McQuaid et al., 2005), used a semistructured interview and pertinent rating scales. Although potentially time intensive, semistructured family interviews have been shown to provide unique variance in explaining child health outcomes beyond self-report measures (McQuaid et al., 2005); this may be owing to the greater depth of information obtained and the potential to clarify discrepant perspectives among family members.
Downloaded from http://jpepsy.oxfordjournals.org/ at Florida Atlantic University on May 24, 2015
Appropriate management of a child’s food allergies necessitates food avoidance across home and social contexts, and response readiness, including recognizing symptoms of FA reactions and managing them with recommended medication (Sicherer & Sampson, 2006). FA management can be a complex process that entails lifestyle changes and poses multiple challenges for the family system. Parents often have difficulties correctly identifying causal foods in products (Joshi, Mofidi, & Sicherer, 2002) and may use inadequate strategies to guide their children’s food avoidance in activities outside of the home (Kapoor et al., 2004). Furthermore, parents often lack the skills to recognize symptoms of anaphylaxis, may not have medication readily available, and lack the knowledge or confidence to treat exposures appropriately (Arkwright & Farragher, 2006). Although several aspects of FA management have been evaluated among parents, no measure comprehensively evaluates the adequacy of parents’ FA management skills across requisite behavioral domains, and, to our knowledge, no measures have evaluated key dimensions of children’s FA self-management.
Klinnert et al.
Development of the FAMAS
3
Rating scales were constructed to capture a range of functioning for each of three constructs essential within our model of family adaptation to children’s food allergies: (1) multiple scales for assessment of FA management behavioral domains (Sicherer & Sampson, 2006), with child-specific scales for areas within children’s realm of responsibility (DunnGalvin, Gaffney, & Hourihane, 2009); (2) scales for assessment of FA anxiety for individual parents and children (Mandell et al., 2005); and (3) a global scale to assess how well families’ were balancing FA management demands with the psychological well-being and developmental needs of their children as well as themselves (McQuaid et al., 2005). We developed this measure for families of children aged 6–12 years with IgE-mediated food allergies because it is during the school-age years that children solidify self-management skills, develop specific styles for coping with food allergies, and accept or reject the incorporation of food allergies into their identity (DunnGalvin et al., 2009). Such an instrument could serve to identify families in need of clinical services owing to problems with FA adaptation, as well as relevant targets for future interventions. To test the validity of the Food Allergy Management and Adaptation Scale (FAMAS), we tested each of the three key constructs using multiple strategies. We hypothesized (1) the FA management component, composed of eight subscales, would (1a) demonstrate adequate internal consistency across the eight subscales; (1b) correlate with independent ratings of family FA management made by physicians who specialize in clinical care for children with FA; and (1c) be associated with an observational assessment of parents’ injectable epinephrine proficiency; (2) FA-specific anxiety would (2a) be moderately correlated with general anxiety measures for both parents and children; and (2b) mothers’ FA Anxiety would be correlated with outcome expectations for death; and (3) BI would be significantly correlated with (3a) lower FA family impact measured by the FAIS and (3b) better parent FA QoL, measured by the FAQL-PB.
Methods The FAMAS development and validation studies were conducted at a tertiary care center in a metropolitan setting and were approved by the institutional review board. Parental informed consent and child assent were obtained for both the pilot and the validation phases of the study.
FAMAS Interview and Scale Development Development of the FAMAS interview and rating scales was based on empirical reports regarding children and families’ experiences with FA, consultation with board-certified allergists regarding principles of FA management, pilot studies of parent FA QoL and outcome expectations (Klinnert & Robinson, 2008), and qualitative data obtained from focus groups (unpublished). The interview and rating scales were developed with reference to the constructs identified a priori as fundamental to FA adaptation. For each construct, we generated open-ended stem questions with follow-up probes designed to elicit meaningful behaviors and vignettes from children and parents regarding FA management (e.g., avoiding foods), subjective experience (e.g., emotional response to food allergies), objective experience (e.g., prior use of injectable epinephrine), and psychosocial adjustment (e.g., impact on family social activities). Based on clinical recommendations and effective behavioral strategies for FA management (Boyce et al., 2010), an initial series of 9point rating scales was developed for key constructs and behavioral domains, with the mid-point score (5) representing “adequate” functioning for respective domains. For each scale, core features were identified to consider when rating, and descriptive anchor points were developed. The pilot phase involved video-recorded interviews with 27 children with FA (M age ¼ 8.4, SD ¼ 1.8, range: 6–12 years) and their caregivers, who represented various levels of FA management and adaptation. Following each interview, each team member independently reviewed video recordings with attention to interview
Downloaded from http://jpepsy.oxfordjournals.org/ at Florida Atlantic University on May 24, 2015
Figure 1. Conceptual model of family adjustment to food allergies.
4
Klinnert et al.
Table I. Content and Scoring of the Food Allergy Management and Adaptation Scale (FAMAS) FAMAS subscales FA management composite FA knowledge
Symptom assessment
Family response readiness Child response readiness
Low, middle, and high anchor points
Knowledge of basic mechanisms of FA, including relevant organ systems, modes of exposure, risks for reaction or death, relationship with asthma, natural history. Knowledge of reaction symptoms, own child’s symptom pattern, gradation of symptom severity from mild to anaphylaxis; specific indications of anaphylaxis
1. Poor understanding 5. Basic elements, but one significant gap 9. Excellent grasp of FA 1. Limited awareness 5. Knows primary symptoms of FA reaction 9. Comprehensive understanding of FA reactions 1. Unprepared for reaction, minimization 5. Plan less than perfect, but could save child 9. Systematic, coherent plan 1. Denies or hides symptoms 5. Acknowledges symptoms/sufficient plan 9. Alert to symptoms, coherent/developmentally appropriate plan 1. Limited/hopeless regarding food avoidance 5. Adequate understanding, some inconsistencies 9. Coherent strategy across settings 1. No strategies/resistant to food avoidance 5. Good understandings of avoidance basics 9. Advocates for self in multiple settings 1. Not readily available 5. Generally on hand but have not practiced 9. Always at hand and have practiced 1. Do not inform or prepare 5. May be vague across alternate caregivers 9. Written and verbal reaction response plan 1. No evidence that FA produces anxiety 5. Moderate anxiety about FA/reactions 9. Clinically significant anxiety 1. Expresses no anxiety due to FA 5. Moderate anxiety about FA/reactions 9. Extreme avoidance/fear of dying 1. Imbalanced/child at risk/high FA burden 5. Adequate management/less burden 9. Balance between management/psychosocial adjustment
Family members’ preparation for managing reactions, including response appropriate to symptoms; quality and availability of action plan for future reactions. Child’s preparation for managing reactions, including response appropriate to symptoms (e.g. notifying adult, cooperating with treatment); quality and availability of action plan; developmentally appropriate.
Family food avoidance
Strategies for food avoidance at and away from home; awareness of potential for accidental exposure; knowledge and use of food labels; awareness of cross-contamination.
Child food avoidance
Developmentally appropriate strategies for food avoidance; awareness of potential accidental exposure; knowledge and use of food labels; awareness of cross-contamination. Have current, filled prescriptions for injectable epinephrine and antihistamines; medications stored appropriately, available at all times, and in all environments; availability of asthma medications if needed. All alternate caregivers are informed of child’s FA, food avoidance strategies, knowledge of symptoms and action plan, medication availability; preparedness to respond to symptoms, administer epinephrine. Anxiety levels self-reported by parent; anxiety revealed by responses regarding specific management strategies and experiences with reactions and by FA outcome expectations. Child’s self-reported anxiety level; anxiety revealed by responses regarding specific management strategies and experiences with reactions and by FA outcome expectations. Balance of attention to FA management and other developmental and family issues (e.g., emotional adjustment, participation in extracurricular and family activities).
Medication availability Alternate caregivers Parent anxiety
Child anxiety
Balanced integration
Note. FA ¼ food allergy. Each FAMAS subscale uses a 9-point scale, with higher scores indicating high (e.g., anxiety) or better (e.g., FA management scales); scores below the mid-point of 5 indicate inadequate FA management. Composites are a mean of the subsequently listed subscales.
structure and flow, and used preliminary rating scales to make initial ratings. Team meetings were then used to review team members’ ratings to (1) achieve consensus ratings consistent with the purpose of the scales, (2) use rating disagreements to identify unclear scale items or interview questions, (3) make indicated refinements to interview questions and follow-up probes and to rating scales. Finally, we discussed changes to improve the interview, such as rewording of questions, ordering of questions, and overall flow. The initial pilot phase ended when ratings were consistent and no further major refinements to the interview or rating scales were necessary to capture relevant content (Supplementary for FAMAS Interview). An abbreviated version of the final FAMAS 9-point rating scales is presented in Table I. Adequacy of FA management is assessed with eight dimensions that can be combined by taking the mean of the eight ratings to create an FA Management Composite (FAMComposite). Parent and Child FA Anxiety are assessed and rated for individual parents and children. Children’s responses are
rated separately for food avoidance, reaction response readiness, and FA-specific anxiety, with each child scale incorporating expectations consistent with child’s developmental level. BI represents a global judgment of how well the family has integrated requisite FA management behaviors and skills with other aspects of child and family life, avoiding unwarranted restrictions on family members’ behavior, and maintaining normative age- and role-appropriate functioning for family members.
Participants for FAMAS Validation Study A second, new sample of families was recruited via physician referrals from the hospital pediatric outpatient clinic, local private allergy practices, and mailings to members of a local FA support group. Eligibility for the study required that families have a child 6–12 years old with physician-documented FA, including a verified history of food reaction and positive FA testing within 18 months before the study visit. Children with unconfirmed FA, non-English-
Downloaded from http://jpepsy.oxfordjournals.org/ at Florida Atlantic University on May 24, 2015
Description/specifications
Development of the FAMAS speaking families, and children with severe developmental delay were excluded.
Study Procedures
Measures Physician Global Ratings of Family Food Avoidance and Reaction Response Readiness Two board certified allergists using clinical judgment, without review of the FAMAS rating scales, independently reviewed video recordings of interviews edited to include only information relevant to FA management. Physicians used 5-point scales to make global ratings of family food avoidance and family reaction response readiness (Supplementary for physician rating guidelines). For reliability assessment, both physicians rated the edited portion of 77% (n ¼ 46) of the interview recordings. Interrater reliability, computed as intraclass correlations (ICC), was .84 for reaction response readiness and .76 for family food avoidance. EPI-Demo The videotaped segments documenting the caregiver’s response to the epinephrine task were rated by two study personnel trained to reliably score whether the caregiver (1) knew the correct site to inject epinephrine, (2) removed the safety cap, (3) applied enough pressure to trigger the device, (4) left the needle in the skin for recommended time after the device was triggered, and (5) listed breathing problems or faintness as indications for use (Arkwright & Farragher, 2006). Analyses for the present study used the sum of correctly performed items, with higher scores corresponding to more appropriate device use, as well as a pass/fail score, with a pass based on correct demonstration of all five components. Interrater reliability was computed for both scores: sum correct (ICC ¼ .92) and pass/fail (ICC ¼ .93).
State-Trait Anxiety Inventory The State-Trait Anxiety Inventory (STAI) is a 40-item instrument that measures an individual’s stable trait and transient state anxiety. The STAI provided self-report data on general anxiety levels. Extensive validity testing suggests that the STAI has good concurrent, convergent, divergent, and construct validity (Speilberger, 1970; Spielberger, Gorsuch, Lushene, Vagg, & Jacobs, 1973). STAI State and Trait t scores were used in this study. The STAI demonstrated excellent internal consistency for mothers (n ¼ 57, Cronbach’s a ¼ .94 and .91), and for fathers (n ¼ 14, a ¼ .83 and .90) for the State and Trait subscales, respectively. State-Trait Anxiety Inventory for Children The STAI-C is a 40-item questionnaire for which children respond to statements that begin with “I feel . . . ” by choosing one of three answers reflecting varying levels of comfort (e.g., “very calm,” “calm,” “not calm”) (Spielberger et al., 1973). The STAI-C was used to provide data on the children’s general anxiety. STAI-C State and Trait t scores were used in this study. Adequate reliability and validity have been demonstrated (Spielberger et al., 1973). The STAI-C has been used with first and second graders by having it read to them and their response recorded (Papay & Hedl, 1978), a procedure that we followed. Internal consistency in the current study was good (a ¼ .86 and .83 for State and Trait subscales, respectively). Multidimensional Anxiety Scale for Children The MASC is a 39-item self-report clinical measure designed to assess a broad range of anxiety symptoms for children aged 8–18 years. Children are asked to rate items using a 4-point scale ranging from “never true” to “often true” (March, 1997). Greater MASC scores indicate higher levels of anxiety. The MASC has demonstrated acceptable psychometric properties (March, 1997). MASC Total Anxiety t scores were used in the current study. For the current study, internal consistency was examined separately for the younger children and those 8 years. Cronbach’s a for the younger children was .83, and .85 for the 8–12-year-olds. For the entire sample, a ¼ .85. Food Allergy Independent Measure The Food Allergy Independent Measure (FAIM) consists of four questions rated on 7-point Likert scales, and assesses the parent’s expectation of outcome for the child with FA (DunnGalvin et al., 2008; van der Velde et al., 2010). Parents rate for their child the chance of: (1) accidental exposure, (2) severe reaction when accidentally exposed, (3) dying when accidentally exposed, and (4) responding effectively when accidentally exposed. For the current study, ratings for question 3 concerning parent perception regarding chance of child’s death owing to FA was included as a concurrent measure of parent FA specific anxiety. Food Allergy Impact Scale The FAIS is a 32-item questionnaire with eight subscales that evaluate the caregiver’s perception of the impact of the child’s FA on aspects of daily family activities (e.g., meal preparation, family social activities); subscale scores were related to number of FA per child (Bollinger et al., 2006). For each item, caregivers rate the impact of the child’s FA using 7-point Likert scales, indicate if FA prevents child or family engagement in the queried activity (scored as 8), or mark that the item does not apply to their child (e.g., for FA impact on daycare, child does not attend daycare; scored as missing). FAIS
Downloaded from http://jpepsy.oxfordjournals.org/ at Florida Atlantic University on May 24, 2015
After parental informed consent and child assent were obtained, the parent(s) and child participated in an hour-long, video recorded interview conducted by one of two clinical psychologists. At the end of the interview, the child was separated from the parent(s) for questionnaire completion, supervised by research assistants (RAs). Children aged 8–12 completed questionnaires independently, while for 6- and 7-year-olds, RAs read the questions for the State-Trait Anxiety Inventory for Children (STAI-C) and Multidimensional Anxiety Scale for Children (MASC) questions and recorded the children’s responses. With the child absent, parents were asked additional questions privately that might have been uncomfortable in the presence of the child (e.g., “on a scale from 1 to 5 where 1 is ‘don’t worry at all’ and 5 is ‘get really scared,’ how worried or nervous do you get when your child is having FA symptoms?”) followed by open-ended questions to assess the parent’s emotions in response to FA reactions. An RA then videotaped the parent demonstrating the use of a trainer version of an auto-injectable epinephrine device (Epi-Demo procedure). Parents were allowed to choose among several devices to ensure they were using a familiar one. After their demonstration, parents were asked to describe indications for administering epinephrine to their child. Finally, parents completed questionnaires. Following study visits, all video-recorded interviews were viewed and rated independently using the scale guidelines by research team members. Raters were three pediatric psychologists (two of whom also conducted interviews) and two RAs with training in FA management and behavioral health. Ratings were recorded for reliability calculation before discussion of rater disagreements, and final consensus scores were generated.
5
6 subscale scores were calculated by taking the mean of item ratings. Responses were required for at least half of constituent subscale items; otherwise subscale scores were considered missing. Internal consistency for the FAIS was adequate for this sample, with seven of eight subscales exceeding Cronbach’s a of .60 for mothers (a ¼ .54–.96; caregiver-supervised social activities, a ¼ .54), and seven of eight subscales exceeding a of .85 for fathers (a ¼ .49–.99; family relations, a ¼ .49).
Overview of Statistical Analyses Interrater reliability was assessed with two-way, single-measures ICC based on consistency in response for Likert-scale items, and based on absolute agreement for dichotomous items. For FAMAS subscales, pairwise ICCs were calculated for each rater compared with consensus scores; the mean and range of raters’ ICCs were calculated for each scale. Internal consistency was determined using Cronbach’s alpha on standardized variables. FAMAS validity testing included a comparison of FAMComposite scores for parents who passed versus failed the Epi-Demo, using an independent samples t test with unequal variances, and Cohen’s d was calculated as a measure of effect size. Spearman correlation coefficients were initially used to assess relationships between FAMAS scales and validity measures with continuous distributions. Descriptive analyses revealed correlations among number of FAs, age, and scale scores. Multiple linear regressions were used to regress the FAMAS subscales on validity measures, adjusting for child age and total number of FA. The multiple scale scores of the FAIS, which were intercorrelated, were entered together in a model after multicollinearity was examined and found not to be a limiting factor. Analyses were performed using JMP 10.00, SPSS version 22 and SAS version 9.3.
Results The validation sample included 60 families with physiciandocumented FA (Table II). Twenty percent of children had one FA, 27% had two, and the remainder had three or more documented FAs. Most children (77%) were accompanied by mothers, 18% by both parents, and three children (5%) by their fathers. All mothers present for the interview identified themselves as the primary FA caregiver. Validity was examined for the 57 families with mothers present. Mean FAMAS subscale scores were generally high relative to the conceptually determined a priori scale midpoint (5 on a 9-point scale), particularly for the subscales comprising the FAMComposite (Table III). The mean score for the FAMComposite was 6.6 (SD ¼ 1.3). Mean scores for the anxiety subscales and BI fell between scale points 5 and 6. Interrater reliability was excellent for FAMAS subscales (ICC range: .91–.98; Table III).
Table II. Participant and Family Characteristics Variable
Mean 6 SD or N (%)
Child age Child gender Male Female Child race Asian Black White More than one race Child ethnicitya Hispanic or Latino Not Hispanic or Latino Child’s age at diagnosis (years) Number of food allergies Number of families reporting anaphylaxis Number of anaphylactic reactions Child diagnosed with other allergic disease Asthma Atopic dermatitis Caregiver(s) participating in study Mother only Father only Both parents Mother’s highest level of education Less than college degree College or graduate degree Father’s highest level of education Less than college degree College or graduate degree Parents’ marital status Married Not married
8.7 6 1.8 39 (65) 21 (35) 1 (2) 2 (3) 51 (85) 6 (10) 8 (14) 50 (86) 2.3 6 2.6 3.0 6 1.8 33 (55) 1.7 6 1 35 (58.3) 27 (45) 46 (77) 3 (5) 11 (18) 5 (8) 55 (92) 11 (18) 49 (82) 55 (92) 5 (8)
Note. aEthnicity data missing for two subjects.
Validity of the FAMAS Our first hypothesis regarding validity for the FAMComposite was supported. The FAMComposite had excellent internal consistency (a ¼ .91). FAMComposite scores differed for families who passed the Epi-Demo compared with those who failed, t(54) ¼ 3.73, p ¼ .0005 (t test based on unequal variances; Pass: M ¼ 7.31, SD ¼ 0.59; Fail: M ¼ 6.34, SD ¼ 1.33; Cohen’s d ¼ 1.13). Regression analyses showed that physician ratings of family Food Avoidance and of reaction Response Readiness as well as Epi-Demo scores were significantly related to FAMComposite after controlling for child age and number of FAs (Table IV). FAMAS ratings of Child FA Anxiety were significantly associated with STAI-C State and Trait scores and with MASC total anxiety t scores (Table IV); this supported Hypothesis 2, that child FA anxiety ratings were correlated with general anxiety scores. For mothers, Hypothesis 2 was not supported, as mother FA anxiety ratings were not significantly associated with STAI scores. However, mother FA anxiety was strongly associated with the FAIM query regarding expectation of death as FA outcome (Table IV). Finally, higher BI was significantly associated with better maternal FA QOL, measured with the FAQL-PB. Spearman’s raw correlations revealed relationships between higher BI and lower scores on a number of FAIS scales (Table IV). However, a regression model that included all FAIS scales showed that caregiver-supervised child social activities, family relations, and meal preparation were the significant predictors for BI scores.
Downloaded from http://jpepsy.oxfordjournals.org/ at Florida Atlantic University on May 24, 2015
Food Allergy Quality of Life-Parent Burden Questionnaire The FAQL-PB measures the impact of FA on health-related QoL of caregivers (Cohen et al., 2004). Using a 7-point scale, parents are asked to indicate how their child’s FA has limited or troubled them during activities of daily living (e.g., eating outside the home, sending child to camp). Higher scores indicate increasing caregiver burden. The FAQL-PB has demonstrated excellent internal consistency and test–retest reliability (Cohen et al., 2004). The FAQL-PB total score was used for the current study, and demonstrated excellent internal consistency for mothers, a ¼ .93 (n ¼ 57), and for fathers, a ¼ .95 (n ¼ 12).
Klinnert et al.
Development of the FAMAS
7
Table III. Descriptive Statistics and Interrater Reliability for Food Allergy Management and Adaptation Scale (FAMAS) Subscale Scores FAMAS subscales
Interrater reliabilitya
Scores: Entire sample (n ¼ 60)
Scores: Validation sample (n ¼ 57)
Mean
Mean (SD)
Median (Q1, Q3)
Mean (SD)
6.9 (1.3) 6.7 (1.1) 6.1 (1.7) 5.9 (1.7) 7.0 (1.4) 7.4 (1.6) 6.5 (2.1) 6.4 (1.9) 6.6 (1.3) 5.7 (1.9) 4.0 (1.4) 5.6 (1.6) 5.5 (1.8)
7.0 (6.0, 8.0) 7.0 (6.0, 7.0) 6.0 (5.0, 7.0) 6.0 (5.0, 7.0) 7.0 (6.5, 8.0) 8.0 (7.0, 8.5) 7.0 (5.5, 8.0) 7.0 (5.0, 8.0) 6.9 (5.9, 7.6) 6.0 (4.0, 7.0) 3.5 (3.0, 5.0) 5.0 (4.3, 7.0) 5.0 (5.0, 7.0)
Range
60 60 60 60 60 60 60 60
0.98 0.97 0.95 0.95 0.98 0.98 0.98 0.97
0.97–0.99 0.95–0.99 0.91–0.99 0.89–0.98 0.96–0.99 0.96–0.99 0.97–0.99 0.92–0.99
57 14 60 60
0.97 0.99 0.97 0.94
0.95–0.99 0.98–0.99 0.94–0.99 0.88–0.98
Median (Q1, Q3)
6.9 (1.3) 6.7 (1.1) 6.2 (1.6) 6.0 (1.6) 7.1 (1.4) 7.4 (1.5) 6.7 (2.1) 6.5 (1.9) 6.7 (1.2) 5.7 (1.9)
7.0 (6.0, 8.0) 7.0 (6.0, 7.0) 6.0 (5.0, 7.0) 6.0 (5.0, 7.0) 8.0 (7.0, 8.0) 8.0 (7.0, 9.0) 7.0 (6.0, 8.0) 7.0 (5.0, 8.0) 6.9 (6.1, 7.6) 6.0 (4.0, 7.0)
5.6 (1.7) 5.6 (1.8)
5.0 (5.0, 7.0) 5.0 (5.0, 7.0)
Note. FA ¼ food allergy. a Mean and range of pairwise intraclass correlations calculated for scores by each of five raters compared with consensus scores; number of raters varied for subscales. b Fathers’ FA anxiety scores not included in the validation sample owing to small sample size.
Table IV. FAMAS Concurrent Validity qa
bb
p
Construct
Validity measure
N
FA management composite
Physician global rating—food avoidance Physician global rating—response readiness EPI-demo: sum of passed items
57 57 57
0.75 0.69 0.43
1.407 1.039 0.565
