Double Abcx Model of Family Stress Pile Up

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• Published: 09 September 2021

Perceived mental health in parents of children with rare congenital surgical diseases: a double ABCX model considering gender

• Holger Zapf^{1  na1},
• Mareike Fuerboeter²,
• Rojin Nazarian^two,
• Konrad Reinshagen²,
• Silke Wiegand-Grefe^{1  na1} &
• Michael Boettcher^{2  na1}

Orphanet Journal of Rare Diseases book sixteen, Article number:384 (2021) Cite this article

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Abstract

Background

Previous inquiry has supported the utility of the Double ABCX model of family adaptation for parents in diverse diseases. Withal, information technology remains unclear how raising a child with rare built surgical diseases impacts the mental health of both mothers and fathers.

Methods

The potential predictors of maternal and paternal mental health in a German language sample of 210 parents of children with rare congenital surgical diseases were investigated. Parents were investigated cross-sectionally utilizing standardized psychometric questionnaires that assessed factors attributed to parental adaptation within the Double ABCX model.

Results

Stressor pile-up, family functioning, perceived stress, and mental health were positively associated with mothers and fathers. Withal, further analyses revealed that family functioning, social support, and perceived stress fully mediated the positive association between stressor pile-up and mental health in mothers, but not fathers.

Conclusion

Our findings suggest that parental accommodation to a rare congenital surgical illness in their children may be improved past increased intra- and extrafamilial resources and decreased perceived family-related stress in mothers, but not fathers. Our results may aid to place gender-specific factors that may guide clinicians and future interventions.

Background

Rare diseases are defined by a prevalence of less than ane:2000 in the European Union [1]. It is estimated that more than 7000 different rare diseases affect well-nigh xiii.5–25 million people in Europe [2]. Even though parents of children with rare diseases evidence a reduced Quality of Life [3] and increased mental health symptoms [iv,5,vi], in general, those parents have received little attending from the inquiry and medical customs [seven].

A specific subgroup of rare diseases requires surgical treatment early on on. These rare diseases include anorectal malformation, biliary atresia, congenital diaphragmatic hernia, esophageal atresia, and Hirschsprung's disease. Caused by the enormous heterogeneity and sustainable variation in severity, some defects tin be easily repaired, merely others require multiple surgeries, which potentially proceed with life-long illness and chronic inability [viii]. Following the diagnosis of rare congenital surgical diseases, parents engage in a continuous adaptation process throughout their child's evolution [9]. Similarly, parents of children with other rare diseases tend to show reduced mental health [10].

One model that tin can account for cumulative stressors such as unlike degrees of disease severity and time since diagnosis is the Double ABCX model of family stress and adaptation [11]. The respective model represents an expansion of the ABCX Model developed by Hill (1958) [12], which includes innovations in theory development and statistical modeling, opening up new pathways for research in family unit stress and coping [thirteen]. The double ABCX model describes the process of accommodation to stressful events, including the following iii variables are included in the double ABCX model, which is fed past the crunch issue (x) [14]: The stressor pile-up (aA) represents aggregated demands resulting from the crisis upshot (eastward.one thousand., special intendance needs of the kid) and additional stressors occurring with time (e.g., unemployment) [11]. The intermediary factors include existing adaptive resources (bB) of the family unit (e.g., social support) and perception and coherence (cC) of the crisis within the family (e.thou., evaluation of the stressor every bit a danger or a challenge). The product of the interactions between these variables yields whether a family unit and its members adapt (20) positively or negatively to a crisis (Fig. i).

In the context of rare congenital surgical diseases, a crisis outcome (x) is the diagnosis of a rare disease and, as given in this study, the circumstances acquired by the COVID-19 pandemic. In recent inquiry on families of children with a chronic status, dissimilar indicators accept been used to operationalize the cumulative demands forming stressor pile-up (aA), among them scales assessing stressful life events of the family [xv], life stress [16], disease severity, behavioral problems of the child [17], or unmarried items such equally parental educational attainment, marital condition [xviii], and income [19]. Adaptive resources (bB) were assessed as social or family back up and family unit functioning or family environment [16, 18, 20]. The operationalization of perception and coherence (cC) has also varied across studies, including self-reports of sense of coherence [20], spiritual wellbeing, and perceived parental/family stress [15, eighteen]. Mental health, depression, and health-related quality of life [xix] were used every bit adaptation indicators (xX).

Recent studies indicated that different outcomes in fathers and mothers of children with rare congenital surgical diseases are expected. Mothers showed worse weather condition regarding mental wellness and quality of life, even though the begetter's quality of life has been constitute to be reduced in comparing to normative samples [21, 22].

The objective of the current cross-sectional study was to investigate factors inside the Double ABCX model to explain parental mental health when raising a child with a rare built surgical affliction. Our specific aim was to explore the fit of the ABCX model concerning the mothers and fathers in the sample. In line with previous enquiry, we specified adaptation (twenty) as mental health, resource (bB) as family functioning and social support, perception, and coherence (cC) equally perceived family stress, and stressor pile-up (aA) as the cumulative demands the parents confront. Co-ordinate to the model, we expected that the clan between stressor pile-upwardly (aA) and accommodation (xX) diminishes relevantly when resources (bB), as well equally perception and coherence (cC), are integrated into the model. Concerning the parents' sex, we assumed that models would differ for mothers and fathers in accordance with previous findings.

Methods

Study blueprint

Parents of children and adolescents with rare built surgical diseases were investigated in a quantitative-based cross-sectional study between March 2020 to April 2021 by means of standardized psychometric questionnaires. The study received ethical approving from the Medical Bedroom Hamburg (PV7161) and was preregistered in ClinicalTrials.gov (NCT04382820).

Measures

Stressor pile-up (aA factor)

The items for stressor pile-up were selected based on previous studies [8,ix,10] and discussions of expected stressors with clinicians. Every bit in previous inquiry, dichotomous variables were summed together [18]. Also, in line with previous research and due to a lack of information on the touch of the dissimilar aspects, no weights were assigned to the single variables. The stressor pile-up count was based on the summary of the post-obit nine dichotomized variables: (one) altitude from the last surgery less than 1.5 years (due north[yeah] = 56/114, 49.i%); (2) presence of level of care of the affected child (n[yes] = 43/109, 39.4%); (3) presence of physical comorbidities of the affected child (north[yes] = 49/114, 43.0%); (four) parents' non-high schoolhouse teaching attainment in mothers (n[yes] = 32/101, 31.7%) and fathers (northward[yeah] = 42/96, 43.8%); (5) parents' unemployment in the last 12 months in mothers (n[yes] = ix/107, 8.4%) and fathers (n[yes] = ii/100, 2.0%); (6) presence of a physical disease in mothers (n[yep] = 23/109, 21.1%) and fathers (northward[yes] = 20/100, 20.0%); (7) presence of COVID-nineteen lockdown measures during the survey (n[yep] = 43/114, 37.7%); (viii) parent-rated psychological distress of the affected children (SDQ total score \(\ge\) 13) by mothers (north[yes] = 29/96, 30.2%) and fathers (n[aye] = 28/ninety, 31.1%); (9) cocky-reported psychological distress of the partner (BSI GSI Score \(\ge\) 63) in mothers (n[yes] = 34/104, 33.iii%) and fathers (n[yeah] = eleven/93, 11.eight%).

Parental resources (bB factor)

The German version of the Family Assessment Mensurate (FAM; Cierpka & Frevert, 1994) was used to assess the family unit functioning of the family unit as a whole. It consists of xl items, which are answered on a four-point rating scale. In improver to the vii subscales task accomplishment, office performance, communication, emotionality, affective involvement, control, and values and norms, the FAM contains the two control scales, social desirability, and defensiveness. We solely used the total FAM score of the general scale to capture the resources and problems of the families by summing upwards all values. Higher scores indicate worse family functioning. The FAM has shown acceptable psychometric properties [31].

The Oslo-Social Support Scale [23] is a cursory checklist measuring social support. Information technology consists of three items asking virtually the number of people one can rely on for personal problems, the evaluation of tertiary parties' involvement in oneself, and the possibility of getting applied back up from neighbors and friends. The higher the sum score, the stronger the social back up. The German language version of the OSSS-3 has shown skillful psychometric properties [24].

Parental perception and coherence (cC cistron)

The German version of the Bear on on Family Calibration (IFS) every bit a cocky-report instrument was used to measure the family-related stress due to the chronic condition and disability in babyhood of the kid [25]. The German language scale includes 33 items on five subscales, including daily/social bear upon, personal bear on/worries about the future, financial impact, bear upon on coping, and touch on siblings [26]. The items were answered on a four-point scale with higher values indicating a higher level of family-related stress. We solely used the total score that includes all items except for the dimension impact on siblings. The psychometric properties of the IFS are considered to be acceptable [26].

Parental adaptation (xX cistron)

The Cursory Symptom Inventory (BSI) was used to measure parental mental health bug [27]. The BSI includes 53 items covering somatization, compulsivity, interpersonal sensitivity, low, anxiety, hostility, phobic fear, paranoid thinking, and psychoticism. We used the Global Severity Index (GSI) as a global alphabetize of psychological distress. The GSI is calculated using the sums for the ix symptom dimensions plus iv boosted items not included in any of the dimension scores and dividing past the total number of items to which the individual responded. Higher BSI scores indicate greater mental health. The German version of the BSI has been found to have good psychometric backdrop [28].

Socio-demographic and clinical variables Parents completed a study-specific socio-demographic questionnaire virtually their sex, age, and socioeconomic status. Clinical variables of the children included the clinical diagnoses, time since final surgery, level of care, and physical comorbidities (atopic eczema, bronchial asthma, cardiac anomalies, celiac disease, chronic cholecystitis, chronic reflux esophagitis, cleft lip and palate, clubfeet, disturbance of claret coagulation, duodenal stenosis, epilepsy, failure to thrive in obesity, renal anomalies, scoliosis, tracheobronchomalacia, urogenital anomalies, VACTERL-association). Psychosocial variables of the parents included the presence of a physical disease, the parent-rated psychological distress of affected child measured with the Force and Difficulties Questionnaire (SDQ) total score amidst children anile 3–16 years, and psychological distress of the partner measures with the BSI Global Severity Index. Moreover, the presence of COVID-nineteen lockdown measures during the survey was included.

Sample

Inclusion criteria for the families were equally follows: (1) having a child under 21 years of age, (2) with a diagnosed rare congenital surgical affliction, including anorectal malformation, biliary atresia, built diaphragmatic hernia, esophageal atresia, or Hirschsprung's disease were included in the report. In addition, astringent concrete, mental, or cerebral impairments were gear up every bit exclusion criteria, making participation impossible or unreasonable. Signed informed consent was given by the parents. The parents were immune to withdraw from the study at any given time.

Three hundred xl-two families with children and adolescents with rare built surgical diseases were identified between 2012 and 2020 in the operative registry from the Clinic of Pediatric Surgery of the Academy Medical Center Hamburg Eppendorf. Families were excluded due to lack of consent to participate (north = 3), lack of German language skills (due north = thirteen), lack of contact data (n = 62), or not meeting the inclusion criteria (northward = 56). Questionnaires were handed out to 208 families.

The response charge per unit was 54.3% for families of rare diseased children. Finally, written consent from 113 families was obtained. A total number of 210 parents completed the questionnaires nigh parental mental health, consisting of 109 mothers and 101 fathers. 96 (84.2%) of all parent ratings were answered by both parents. All participating children met the definition of a rare affliction according to the European Commission [1], and all diagnoses were verified by medical personnel.

Statistics

Data analysis was performed using descriptive statistics (frequencies, ways, and standard deviations) and bivariate tests (chi-square tests). Associations between variables were analyzed with Spearman correlations. Three variables were tested every bit a possible mediator of the relations between stressor pile-up and parental mental wellness. Multiple mediation was tested post-obit the procedure from Preacher and Hayes (2008) [29]. The multiple mediation analyses were conducted using the PROCESS macro [30], with 10,000 bootstrapping resamples, and bias-corrected 95% confidence intervals were calculated. Equally an indication of effect size, R ^two was used. Statistical significance was ready at p < 0.05 (two-tailed). Statistical analyses were conducted using SPSS Statistics 26.

Results

Characteristics of the report population

Table 1 shows the main sociodemographic and affliction characteristics. The questionnaires were completed by 210 parents. For two mothers and four fathers, items from a specific measure were missing. The final sample for analysis therefore comprised of 204 parents. Regarding the child's age, at that place was a pregnant difference between participants (M = 4.two, 95% CI [3.6, 4.eight], SD = 3.33) and not-participants (G = vi.v, 95% CI [half dozen.0, 7.1], SD = iii.96). Yet, the child's gender between participants (female = 44, male = 70) and non-participants (female person = 123, male = 107) did non differ. At last, no deviation between participants (anorectal malformation = xxx, biliary atresia = xiv, congenital diaphragmatic hernia = xiv, esophageal atresia = 27, Hirschsprung's affliction = 29) and not-participants (anorectal malformation = 65, biliary atresia = 20, congenital diaphragmatic hernia = 27, esophageal atresia = xl, Hirschsprung'due south affliction = 76) could be found regarding the disease-groups.

Tabular array 1 Sociodemographic and illness characteristics of the respondent

Full size table

Correlation analyses

Tabular array ii shows the Pearson correlation coefficients and level of possible associations between variables of the ABCX-Model separated for gender. Pregnant bivariate associations were establish for mothers for all variables and stressor pile-up, except for family functioning. In contrast, meaning bivariate associations were found for fathers all variables and stressor pile-up, except for social back up. No pregnant associations were constitute for family functioning, social support, and perceived stress for both mothers and fathers.

Tabular array 2 Pearson correlation between predictor and upshot parameters (n = 204)

Full size tabular array

Mediation analyses

Table 3 and Fig. 2 show the multiple mediation models for mothers and fathers. Results showed pregnant indirect effects of stressor pile-up on mental health through family performance, social support, and perceived stress in mothers (Model A; run across Fig. 2). While the total effect of stressor pile-up on mental wellness was significant (b = 0.069, p = 0.003, R ² = 0.084), the direct effect was not statistically significant after including family performance, social support, and perceived stress. The pattern of direct, indirect, and total effects suggests that family unit functioning, social support, and perceived stress fully mediated the association between stressor pile-up and mental health in mothers of children with rare built surgical diseases. The multiple mediator model almost explained forty pct of the variance in perceived mental wellness in mothers.

Table iii Multiple mediator models predicting parental mental health in mothers (n = 107) and fathers (due north = 97)

Full size table

Fig. i

Adapted from McCubbin & Patterson (1982)

Double ABCX model.

Full size paradigm

Fig. 2

Parallel mediation of mothers (Model A) and fathers (Model B) of children with rare built surgical diseases. Note. The Path a represents the effect of stressor pile-up on each mediator; Path b represents the combined effects of each mediator on parental mental health; the direct event represents the outcome of the stressor pile-up on parental mental health while keeping levels of the mediators abiding; the indirect effect represents the combined effect of path a and path b and therefore the mediation. The total effect represents the combined indirect and straight effects. Significance inferences at the α = .05 for effects are based upon the notion of whether conviction intervals include goose egg. FAM: Family Assessment Measure; OSSS: Oslo Social Support Calibration; IFS: Impact on Family Calibration; BSI GSI: Brief Symptom Inventory Global Severity Index

Full size prototype

Regarding fathers, results showed no significant indirect furnishings of stressor pile-up on mental health through family functioning, social support, and perceived stress in fathers (Model B; come across Fig. two). While the total effect of stressor pile-upwards on mental health was significant (b = 0.025, p = 0.002, R ² = 0.099), the directly effect was not statistically significant subsequently including the respective mediator variables. The pattern of direct, indirect, and total furnishings suggests that family operation, social support, and perceived stress all do not mediate the association between stressor pile-up and mental health in fathers of children with rare congenital surgical diseases. Withal, nearly i-4th of the variance in perceived mental wellness in fathers was explained by the multiple mediator model.

Discussion

The nowadays study used the double ABCX model as a theoretical framework to investigate the associations between stressor pile-up, resources, and mental health in mothers and fathers of children with rare congenital surgical diseases. For affected mothers, our results were consistent with previous research on chronic childhood conditions, demonstrating a pregnant clan between parental stressor pile-up and adaptation, and a decrease of this clan past appropriate internal and external resources, and perceived power to effectively deal with stress [15,16,17,18,xix,20].

Most importantly, our findings contribute to agreement the clan between parental stressors predominantly due to the child'south illness and the current mental health, emphasizing the mediating role of family unit-related resources and perceived stress. Our multiple mediation supports this conclusion in mothers, but not fathers. Family functioning, social support, and perceived stress were all found to fully mediate the association betwixt parental stressor pile-up and mental health in mothers, whereas, in fathers, the results point a direct-only non-arbitration [32]. These findings support the proposition that when mothers experience increased exposure to stressors, they may respond adequately to these crisis events through available internal and external resources likewise equally the appraisal of the situation [33]. Therefore, the arroyo of using the ABCX model in the context of parents of children with rare congenital surgical disease seems to be confirmed for affected mothers but non for fathers.

It is likewise of import to note that information technology was constitute that mothers of children with rare congenital surgical diseases reported higher levels of overall psychological distress compared to fathers, which is in line with previous inquiry on parents of children with chronic conditions [10, 34] and the general adult populations [35, 36]. In addition, mothers, compared to fathers, are typically more involved in taking care of their children. Therefore, information technology may be that mothers feel more than responsible for the illness, and consequently, the caregiving burden increases, which could explicate boosted increased parental psychosocial impairment in mothers compared to fathers.

In this study, responding parents had a higher education primarily and were married; thus, parents in these families may have had an additional protective factor of financial resources and having a stable primary source of social back up in contrast to the full general population [33].

In particular, our results ostend the importance of intervening factors in explaining parental stress in fathers and mothers caring for a kid with rare congenital surgical diseases. Although the framework within the ABCX model could not be confirmed for fathers, preliminary correlation assay showed that there were associations among the included variables. Additional factors would accept to exist investigated in future studies to determine more relevant variables for paternal mental health within the ABCX model. The identification of potential protective and risk factor such equally marital satisfaction and support from the partner may exist more suitable variables in the theoretical framework, as previous qualitative research suggests, and related to this, father-friendly programs may exist enhanced through the focus on the parent's partnerships [37].

Study limitations

In the present study, several limitations take to be recognized. First, the representativeness of the sample may exist somewhat limited regarding the historic period of the affected children. Since participating families had considerably younger children than non-participating families, the possibility of non-response bias cannot be ruled out. Thus, the psychosocial impairment of affected parents may be overestimated, as information technology could be argued that parents of older children are less impaired. Yet, the participation charge per unit was similar to previous research on child health in western countries [38, 39]. Second, our cross-exclusive multiple arbitration analyses do not allow for causal interpretations, even if the pile-upward of the dissimilar components of the stressor may exist justified as a causal factor. 3rd, any determination cannot exist fatigued on clinical groups other than the rare congenital surgical diseases presented in this report. Fourth, the psychometric instruments used were not validated on populations such as rare diseases, so this must be considered when interpreting the results. Lastly, although rare congenital surgical weather condition are present inside the studied sample and parents may suit better over time, these rare atmospheric condition may increase in babyhood due to comorbidities and complications and atomic number 82 to a renewed increase in psychological distress for parents. Unfortunately, information on these factors was non collected in the questionnaires because of the cantankerous-sectional report design. Therefore, further detailed inquiry is needed using longitudinal designs in which comprehensive disease-specific and psychosocial outcomes are collected prospectively from large, multi-center samples using rigorous analyses. This is the case in a recent multi-center study examining ii psychosocial interventions within Germany [forty].

Conclusion

To our knowledge, the Double ABCX model has not been used to investigate the mental health of parents caring for children with rare congenital rare diseases, even if this has already been investigated in the context of children's chronic diseases [fifteen,sixteen,17,18, 20]. Our findings contribute to the literature on parental adaptation to their children'due south rare congenital surgical disease past describing the gender-specific functioning of the afflicted parents. Potential areas for family intervention can thus be identified to promote resilience in afflicted parents. Future research should comprise prospective, longitudinal studies to further describe the qualities that contribute to parental adjustment to rare congenital surgical affliction and so that within specific family-oriented interventions, these qualities can exist promoted.

Availability of data and materials

The datasets generated during the current study are bachelor from the corresponding author on reasonable request.

Abbreviations

BSI:

Brief symptom inventory

FAM:

Family assessment mensurate

GSI:

Global severity index

OSSS:

Oslo social back up scale

SDQ:

Strength and difficulties questionnaire

IFS:

Bear on on family calibration

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Acknowledgements

A particular thank you goes to all the families who participated. Special thank goes to Antonia Zapf, whose statistical expertise was invaluable during the assay and interpretation of the data.

Funding

Open Admission funding enabled and organized past Projekt Bargain. The inquiry leading to these results received funding by the German Federal Joint Committee (Grand-BA) nether Grant Number 01NVF17028. The funders had no role in the pattern of the report; the collection, analyses, or interpretation of data; the writing of the manuscript, or the conclusion to publish the results.

Author information

Author notes

1. Johannes Boettcher, Holger Zapf, Silke Wiegand-Grefe, Michael Boettcher are share the offset and last authorship.

Affiliations

1. Department of Child and Adolescent Psychiatry, Psychosomatics and Psychotherapy, Academy Medical Heart Hamburg-Eppendorf, Martinistrasse 52, 20246, Hamburg, Germany

   Johannes Boettcher, Holger Zapf & Silke Wiegand-Grefe

2. Department of Pediatric Surgery, Academy Medical Center Hamburg-Eppendorf, Martinistrasse 52, 20246, Hamburg, Germany

   Mareike Fuerboeter, Rojin Nazarian, Konrad Reinshagen & Michael Boettcher

Contributions

MB and KR are the master investigators of the study. JB, KR, SWG, MB developed the study concept and the design. JB and SWG developed the report materials, and MF and RN acquired the data. JB, HZ, AZ, and MB analyzed and interpreted the data. JB wrote the showtime draft of the manuscript. JB, HZ, MF, RN, SWG, KR, and MB revised the start typhoon. All authors have revised the subsequent drafts critically, canonical the final manuscript to exist published, and agreed to be answerable for all aspects of the work. All authors read and canonical the final manuscript.

Corresponding author

Correspondence to Johannes Boettcher.

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Ethics approving and consent to participate

The study was carried out according to the Declaration of Helsinki and was approved past the Medical Chamber Hamburg (PV7161) and was preregistered at ClinicalTrials.gov (NCT04382820).

Consent for publication

Not applicable.

Competing interests

All authors declare that they have no competing interests.

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Boettcher, J., Zapf, H., Fuerboeter, M. et al. Perceived mental wellness in parents of children with rare congenital surgical diseases: a double ABCX model considering gender. Orphanet J Rare Dis 16, 384 (2021). https://doi.org/x.1186/s13023-021-01998-9

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• Received: 05 July 2021

• Accepted: 24 August 2021

• Published: 09 September 2021

• DOI : https://doi.org/x.1186/s13023-021-01998-nine

Keywords

• Mental health
• Rare diseases
• Parents
• Pediatric surgery
• Double ABCX model

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Source: https://ojrd.biomedcentral.com/articles/10.1186/s13023-021-01998-9

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