Mothers and fathers of young Dutch adolescents with Down syndrome: Health related quality of life and family functioning

doi:10.1016/j.ridd.2016.09.014

Research in Developmental Disabilities

Volume 59, December 2016, Pages 359-369

https://doi.org/10.1016/j.ridd.2016.09.014 Get rights and content

Highlights

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We studied HRQoL and family functioning in parents of young adolescents with Down syndrome.
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We studied change in parental HRQoL from children’s ages 6–8 to 11–13 years.
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Mothers showed lower HRQoL than reference mothers in one domain, fathers in none.
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Mothers and fathers both reported poorer family functioning than reference parents.
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Parental HRQoL did not change significantly from children’s ages 6–8 to 11–13 years.

Abstract

Background

Like any child, children with Down syndrome (DS) affect the lives of their families. Most studies focus on the adaptation of parents and families of young children with DS, while relatively few studies include the perspective of fathers.

Aims

To determine 1) whether mothers and fathers of 11 to 13-year-olds with DS differ from reference parents in health related quality of life (HRQoL) and family functioning, and 2) whether HRQoL in parents of children with DS changes over time, from when the child was 6–8 years old to when the child was 11–13 years old.

Methods

80 mothers and 44 fathers completed HRQoL and family functioning questionnaires. 58 parents (53 mothers) had completed the HRQoL-questionnaire in a previous study.

Results

Mothers differed from reference mothers in one HRQoL-domain (Sexuality), while fathers’ HRQoL did not significantly differ from reference fathers. Both mothers and fathers scored in the (sub)clinical range more frequently than reference parents in Total family functioning, and in the domains Partner relation and Social network. Furthermore, fathers scored in the (sub)clinical range more frequently than reference parents in Responsiveness and Organization. HRQoL showed no significant change over time.

Conclusions and implications

Our findings indicate frequent family functioning problems but few HRQoL problems in parents and families of children with DS. In offering care, a family based approach with special attention for partner relation and social functioning is needed.

Section snippets

What this paper adds

This study contributes to the body of knowledge on the adaptation of mothers, fathers and families of young adolescents with DS growing up in today’s society. Furthermore, this paper is the first to report on longitudinal health related quality of life (HRQoL) in parents of children with DS.

In contrast with recent literature, we found that mothers and fathers showed only few statistically significant differences in their HRQoL as compared with reference parents. In family functioning, on the

Recruitment

The recruitment for this study is presented in Fig. 1. For the first aim of this study we assessed HRQoL and family functioning in mothers and fathers of 11 to 13-year-olds with DS. These mothers and fathers were recruited among parents of 123 children with DS who had previously participated in a follow-up study of an RCT, during which they had given permission to be invited for future studies. The RCT and follow-up study aimed to establish effects of thyroxine treatment during the first two

Respondents

Recruitment of respondents is presented in Fig. 1. Sociodemographics of responding mothers (N = 80) and fathers (N = 44) in the current study, and their children with DS (N = 88) are presented in Table 1. Results of the preparatory analyses, which we performed to detect possible selection bias, revealed no significant differences in sociodemographics of responding and non-responding families (data not shown). Comparing parents of children with DS with the TAAQOL reference parents revealed that

Discussion

In the current study we focused on parental HRQoL and family functioning in families of 11–13 year old children with DS and studied change in parental HRQoL since their child’s age 6–8 years. We found few significant differences in HRQoL of parents of 11 to 13-year-olds with DS compared with reference parents. Mothers of children with DS showed poorer HRQoL in one domain (sexuality), while fathers showed no statistically significant differences compared with reference fathers. In family

Conclusion

Where previous studies generally found poorer parental well-being but normal family functioning in families of children with DS, we found few statistically significant differences in parental HRQoL compared with parents of children without conditions, although there appeared to be relevant tendencies towards lower HRQoL in mothers and fathers of children with DS. Predominantly, though, we found poorer family functioning outcomes. This implies that in clinical practice a family-based approach is

References (51)

S.E. Althof et al.
Psychological and interpersonal dimensions of sexual function and dysfunction
Journal of Sexual Medicine
(2005)
J. Cantwell et al.
Social support and mastery influence the association between stress and poor physical health in parents caring for children with developmental disabilities
Research in Developmental Disabilities
(2014)
K. Crnic et al.
Mothers and fathers together: Contrasts in parenting across preschool to early school age in children with developmental delays
International Review of Research in Mental Retardation
(2009)
S.L. Hartley et al.
Marital quality and families of children with developmental disabilities
International Review of Research in Developmental Disability
(2011)
Jan Pieter Marchal et al.
Health related quality of life in parents of six to eight year old children with Down syndrome
Research in Developmental Disabilities
(2013)
M. Van Riper
Families of children with Down syndrome: Responding to a change in plans with resilience
Journal of Pediatric Nursing
(2007)
J. Bourke et al.
Physical and mental health in mothers of children with Down syndrome
Journal of Pediatrics
(2008)
J. Bruil et al.
TAAQOL manual
(2004)
J. Cohen
Statistical power analysis for the behavioral sciences
(1988)
C. Cunningham
Families of children with Down syndrome
Down Syndrome Research and Practice
(1996)

M. Cuskelly et al.

Families of children with Down syndrome: What we know and what we need to know

Down Syndrome Research and Practice

(2008)

A. Dabrowska et al.

Parenting stress and coping styles in mothers and fathers of pre-school children with autism and Down syndrome

Journal of Intellectual Disability Research

(2010)

J.E. Dumas et al.

Parenting stress, child behavior problems, and dysphoria in parents of children with autism, Down syndrome, behavior disorders and normal development

Exceptionality

(1991)

D.J. Fidler et al.

Stress in families of young children with Down syndrome, Williams syndrome: And Smith-Magenis syndrome

Early Education & Development

(2000)

S. Gavidia-Payne et al.

Parental resilience: A neglected construct in resilience research

Clinical Psychologist

(2015)

L. Gilmore et al.

Parenting satisfaction and self-efficacy: A longitudinal study of mothers of children with Down syndrome

Journal of Family Studies

(2012)

S. Hamlyn-Wright et al.

Locus of control fails to mediate between stress and anxiety and depression in parents of children with a developmental disorder

Autism

(2007)

A. Harper et al.

Respite care, marital quality, and stress in parents of children with autism spectrum disorders

Journal of Autism and Developmental Disorders

(2013)

J. Hatzmann et al.

Hidden consequences of success in pediatrics: Parental health-related quality of life–results from the care project

Pediatrics

(2008)

P. Hauser-Cram et al.

Children with disabilities: A longitudinal study of child development and parent well-being

Monographs of the Society for Research in Child Development

(2001)

L. Haverman et al.

Implementation of electronic patient reported outcomes in pediatric daily clinical practice: The KLIK experience

Clinical Practice in Pediatric Psychology

(2014)

G. Hedov et al.

Self-perceived health in Swedish parents of children with Down’s syndrome

Quality of Life Research

(2000)

G. Hedov et al.

Swedish parents of children with Down’s syndrome

Scandinavian Journal of Caring Sciences

(2002)

R.M. Hodapp et al.

Less stress, more rewarding: Parenting children with Down syndrome

Parenting-Science and Practice

(2001)

R.M. Hodapp et al.

The effects of the child with Down syndrome on maternal stress

British Journal of Developmental Psychology

(2003)

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The quality of life of parents of children with down syndrome in a tertiary care hospital: A qualitative research study at Saudi Arabia
2022, Annals of Medicine and Surgery
Citation Excerpt :
Moreover, many complications can be associated with the disease, including hearing loss, heart defects, ear infections, eye diseases, and obstructive sleep apnea [3–5]. In addition to the significant morbidity that DS has on the affected children, it can also affect their caregivers's quality of life (QoL) [6–9]. The term QoL is a broad one that is usually used to assess the different aspects of life.
As children with down syndrome (DS) usually have significant morbidities, they can also represent a significant burden on their caregivers and impact their quality of life (QoL). We conducted this study to investigate whether or not having DS children can impact the different domains of the QoL of their caregivers in Saudi Arabia.
This is a cross-sectional phenomenological qualitative research study that was conducted in a tertiary care hospital, Riyadh, Saudi Arabia. To assess the study outcomes, we used the WHOQOL-BREF to assess the different domains of the QoL.
We have included 261 caregivers to DS children that responded to our questionnaire. The mean (SD) scores for the WHOQOL-BREF domains were 84 (±15), 88 (±15), 41 (±10), and 105 (±24), including the physical, psychological, social relations, and environmental domains, respectively. There was a significant difference between all of the scores that have been reported for these domains (P-value <0.001). Furthermore, educational level and the number of children were significantly associated with the psychological and physical domains, while the number of children was the only significant variable with the social relation. Finally, educational level, number of children, and average monthly income were all significantly correlated with the environmental domain.
Our study indicates that the QoL of caregivers to DS children is significantly impacted in the different domains, indicating the urgent need to apply adequate interventions.
Variables related to the quality of life of families that have a child with severe to profound intellectual disabilities: A systematic review
2021, Heliyon
Family quality of life (FQoL) of families that have a child with severe to profound intellectual disabilities (SPID) is an important and emerging concept, however, related variables are inconclusive.
To gain a better understanding of variables related to the FQoL of families that have a child with SPID, variables related to the FQoL of families that have a child with intellectual disabilities (ID) were systematically reviewed.
A search strategy was performed in five databases. Critical appraisal tools were employed to evaluate the quality of both quantitative and qualitative studies. Data extraction and synthesis occurred to establish general study characteristics, variables, and theoretical concepts. Variables were categorised into four key concepts of the FQoL: systemic concepts, performance concepts, family-unit concepts and individual-member concepts.
A total of 40 studies were retrieved with 98 variables. Quality scores ranged from 7 to 13 (quantitative) and 5 to 13 (qualitative) out of 13 and 14 points, respectively. Five out of the 40 studies (13%) focused on individuals with SPID. Variables related positively or negatively to the FQoL, and were categorised within systemic concepts (n = 3); performance concepts (n = 11); family-unit concepts (n = 26); and individual-member concepts (n = 58).
Several variables were found to be (inter)related to the FQoL of families that have a child with ID. A contrasting picture emerged regarding the impact of a disability in relation to transitional phases. However, studies which include families of children with SPID were minimal, therefore, it remained ambiguous to what extent the identified variables apply to these families.
Cost-Effectiveness Analysis of the Surgical Management of Infants Less than One Year of Age with Feeding Difficulties
2020, Journal of Pediatric Surgery
Citation Excerpt :
Our data show that caring for a sick child has a direct impact on the caregiver's health state. Caring for a sick child has been reported to have a multitude of psychosocial impacts including parental distress, less leisure time, worse partner relations, sleep disturbances, and social isolation [19–22]. Parents of children with chronic conditions score lower on health-related quality of life [23].
We compared the cost-effectiveness of the common surgical strategies for the management of infants with feeding difficulty.
Infants with feeding difficulty undergoing gastrostomy alone (GT), GT and fundoplication, or gastrojejunostomy (GJ) tube were enrolled between 2/2017 and 2/2018. A validated GERD symptom severity questionnaire (GSQ) and visual analog scale (VAS) to assess quality of life (QOL) were administered at baseline, 1 month, and every 6 months. Data collected included demographics, resource utilization, diagnostic studies, and costs. VAS scores were converted to quality adjusted life months (QALMs), and costs per QALM were compared using a decision tree model.
Fifty patients initially had a GT alone (71% laparoscopically), and one had a primary GJ. Median age was 4 months (IQR 3–8 months). Median follow-up was 11 months (IQR 5–13 months). Forty-three did well with GT alone. Six (12%) required conversion from GT to GJ tube, and one required a fundoplication. Of those with GT alone, six (14%) improved significantly so that their GT was removed after a mean of 7 ± 3 months. Overall, the median GSQ score improved from 173 at baseline to 18 after 1 year (p < 0.001). VAS scores also improved from 70/100 at baseline to 85/100 at 1 year (p < 0.001). ED visits (59%), readmissions (47%), and clinic visits (88%) cost $58,091, $1,442,139, and $216,739, respectively. GJ tube had significantly higher costs for diagnostic testing compared to GT (median $8768 vs. $1007, p < 0.001). Conversion to GJ tube resulted in costs of $68,241 per QALM gained compared to GT only.
Most patients improved with GT alone without needing GJ tube or fundoplication. GT and GJ tube were associated with improvement in symptoms and QOL. GJ tube patients reported greater gains in QALMS but incurred higher costs. Further analysis of willingness to pay for each additional QALM will help determine the value of care.
Cost-effectiveness study, Level II.
Health-related quality of life, anxiety, depression and distress of mothers and fathers of children on Home parenteral nutrition
2019, Clinical Nutrition
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Although HPN parents in this study did not report more problems with their (ex)partner than reference parents, and the vast majority of the parents is married/living together, it is important to pay attention to the quality of the partner relation in clinical practice. This is shown in research within parents of children with Down's syndrome, where fathers also did not report more problems with their (ex)partner, measured with the same DT-P questionnaire [28], but the same fathers did indicate a worse partner relation (marital satisfaction, conflict-management, support and trust) on another questionnaire [30]. Furthermore, our findings reflect the results of a small survey [31], which indicated that almost all HPN families experienced great difficulty trying to find a person to look after their child and going out.
Parents of children with intestinal failure, dependent on Home Parenteral Nutrition (HPN), may experience psychosocial problems due to the illness and intensive treatment of their child. Literature concerning psychosocial problems is scarce. Therefore, we aimed to investigate Health-Related Quality of Life (HRQOL), levels of anxiety, depression, distress and everyday problems of these mothers and fathers.
A multicenter study was conducted among 37 mothers and 25 fathers of 37 children on HPN (response-rate 37/49 = 76%, mean age children = 5.1 years, SD = 4.6). Parents completed three questionnaires to measure different outcomes on the KLIK website (www.hetklikt.nu): the TNO-AZL QOL Questionnaire (TAAQOL) to measure HRQOL, the Hospital Anxiety and Depression Scale (HADS) to measure anxiety and depression, and the Distress Thermometer for Parents (DT-P) to measure distress. Scores were compared to Dutch reference mothers and fathers using Mann–Whitney U-tests.
No differences were found in HRQOL, measured by the TAAQOL, between HPN parents compared to the reference groups, except for the subscale ‘depressive emotions’ for mothers (p = .01) and ‘daily activities’ for fathers (p = .04). HPN mothers reported higher levels of depression compared to reference mothers (p = .001). In addition, HPN mothers and fathers reported higher levels of distress than reference mothers (p = .001) and fathers (p = .03). HPN mothers reported significantly more problems in the practical, emotional, cognitive and parenting domains, fathers in the social, emotional and parenting domains.
On HRQOL, anxiety and depression, HPN parents generally did not show much differences compared to reference parents. However, when asked about parental distress and everyday problems, HPN treatment of their child seems highly stressful for some parents and influences daily functioning. Therefore, structural screening for parental psychosocial problems in clinical practice, e.g. using the DT-P, is necessary in order to improve the well-being of both these parents and their children dependent on HPN.
Social support and post-crisis growth among mothers of children with autism spectrum disorder and mothers of children with down syndrome
2019, Research in Developmental Disabilities
Citation Excerpt :
Additionally, mothering a child with ASD is depicted as challenging in terms of the behavioral difficulties facing these children, such as unanticipated and uncontrollable tantrums and aggression that lead to increased stress and difficult daily life (Fletcher, Markoulakis, & Bryden, 2012; Myers, Mackintosh, & Goin-Kochel, 2009). Mothers of children with DS also face a variety of meaningful challenges in the raising of their children, and often experience greater stress than mothers of typically developing children (e.g., Dabrowska & Pisula, 2010; Marchal, Maurice-Stam, van Trotsenburg, & Grootenhuis, 2016; Phillips, Conners, & Curtner-Smith, 2017). However, studies have found that compared to families of children with other cognitive disabilities or ASD, the effects of raising a child with DS seem milder (Dabrowska & Pisula, 2010; Hodapp, Ly, Fidler, & Ricci, 2001; Lanfranchi & Vianello, 2012).
Raising a child with special needs challenges mothers in complicated ways, yet, alongside these difficulties, there is evidence for maternal post-crisis growth. Social support is one element that may contribute to growth.
This study explores the relationship between social support and post-crisis growth, examines type of disability as a mediating variable between support and growth, and, looks at the relations between subtypes of support and growth.
Participants included 99 mothers of children with Autism Spectrum Disorder (ASD) and 119 mothers of children with Down Syndrome (DS). Mothers completed three self-report questionnaires: demographic, Multidimensional Scale of Perceived Social Support, and the Stress-Related Growth Scale.
Social support was found to predict maternal post-crisis growth with type of disability serving as a mediating variable between them, such that social support contributes to post-crisis growth only among mothers of children with ASD. In addition, results revealed various correlations between types of support and types of growth.
The findings indicate that compared to DS, characteristics of ASD may contribute to less maternal post-crisis growth, and that social support serves as an important predictor for growth in this group. Finding ways to increase social support for mothers of children with ASD thus gains additional importance.
Parent-Reported Contribution of Family Variables to the Quality of Life in Children with Down Syndrome: Report From an International Study
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Citation Excerpt :
Although the life expectancy for this population has increased remarkably owing to improvements in medical technology and care provided (Wu & Morris, 2013), the quality of life (QoL) of children with DS has received limited attention (Murphy et al., 2017; Shields, Leonard, Munteanu, Bourke, Lim, Taylor, & Downs, 2018; Xanthopoulos et al., 2017). Most prior studies have focused on QoL of parents or caregivers of children with DS (e.g., Choi, Lee, Lee, & Jung, 2019; Marchal, Maurice-Stam, Van Trotsenburg, & Grootenhuis, 2016; Senses Dinc, Cop, Tos, Sari, & Senel, 2019) rather than the children themselves. Based on the conceptualization derived from studies about individuals with intellectual disabilities (IDs), QoL is a multi-dimensional phenomenon comprised of key domains that constitute personal well-being (Schalock, Verdugo, Gomez, & Reinders, 2016) (Table 1).
The intent of this study was to determine parents' views of the contribution of family variables to the quality of life (QoL) of children with Down Syndrome (DS). Based on prior research, we hypothesized that parents would report that family variables reflecting positive aspects of family functioning contributed to better QoL; and family variables reflecting the negative aspects of family functioning contributed to poorer QoL.
A cross-sectional study was carried out. Invitation letters were sent to DS support groups. Upon agreement, the parents of the children were sent a link to an online survey that included a consent form, demographic questionnaire, and child and family measures. Results: Results demonstrated the contribution of family demands to children's interpersonal relations. Family appraisals concerning the child and the family's ability predicted the children's physical and material well-being, personal development, self-determination, social inclusion, interpersonal relations, and rights. Family problem-solving served as a prominent predictor for children's physical and emotional well-being, personal development, interpersonal relations, and rights. Also, family resources predicted various aspects of children's QoL including physical, emotional, and material well-being, self-determination, social inclusion, interpersonal relations, and rights.
Results confirmed the significant relationship between family and children's QoL variables. Family appraisal and family problem-solving were especially identified as significant predictors of children's QoL that can be targeted for family interventions, since the family variables are modifiable aspects of family life.
Nurses can use current findings to develop interventions to enhance QoL of children with DS and families.

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Mothers and fathers of young Dutch adolescents with Down syndrome: Health related quality of life and family functioning

Highlights

Abstract

Background

Aims

Methods

Results

Conclusions and implications

Section snippets

What this paper adds

Recruitment

Respondents

Discussion

Conclusion

Journal of Sexual Medicine

Research in Developmental Disabilities

International Review of Research in Mental Retardation

International Review of Research in Developmental Disability

Research in Developmental Disabilities

Journal of Pediatric Nursing

Physical and mental health in mothers of children with Down syndrome

Journal of Pediatrics

TAAQOL manual

Statistical power analysis for the behavioral sciences

Families of children with Down syndrome

Down Syndrome Research and Practice

Families of children with Down syndrome: What we know and what we need to know

Down Syndrome Research and Practice

Parenting stress and coping styles in mothers and fathers of pre-school children with autism and Down syndrome

Journal of Intellectual Disability Research

Parenting stress, child behavior problems, and dysphoria in parents of children with autism, Down syndrome, behavior disorders and normal development

Exceptionality

Stress in families of young children with Down syndrome, Williams syndrome: And Smith-Magenis syndrome

Early Education & Development

Parental resilience: A neglected construct in resilience research

Clinical Psychologist

Parenting satisfaction and self-efficacy: A longitudinal study of mothers of children with Down syndrome

Journal of Family Studies

Locus of control fails to mediate between stress and anxiety and depression in parents of children with a developmental disorder

Autism

Respite care, marital quality, and stress in parents of children with autism spectrum disorders

Journal of Autism and Developmental Disorders

Hidden consequences of success in pediatrics: Parental health-related quality of life–results from the care project

Pediatrics

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Monographs of the Society for Research in Child Development

Implementation of electronic patient reported outcomes in pediatric daily clinical practice: The KLIK experience

Clinical Practice in Pediatric Psychology

Self-perceived health in Swedish parents of children with Down’s syndrome

Quality of Life Research

Swedish parents of children with Down’s syndrome

Scandinavian Journal of Caring Sciences

Less stress, more rewarding: Parenting children with Down syndrome

Parenting-Science and Practice

The effects of the child with Down syndrome on maternal stress

British Journal of Developmental Psychology