| Abstract|| |
The multi-dimensional impact on the quality of life (QOL) of families of children with the nephrotic syndrome (NS) has not been studied sufficiently in the literature. We aimed to study this aspect and the predictors of poor QOL among Indian families having children with NS. A cross-sectional study was conducted to compare the parents of children with chronic NS on treatment for at least one year with parents of a matched healthy control group. The parents of both groups were asked to complete the standard self-administered multi-dimensional questionnaire of Pediatric Quality of Life Inventory 4 (PedsQL TM ) Family Impact Module (FIM). Descriptive and analytical statistics were performed to compare scores between the two groups. Possible predictors of poor outcome in each of the summary scales among the cases were assessed by both univariate and multivariate analysis. The parents of 61 cases and 72 controls completed the PedsQL TM FIM questionnaire. The scores in each of the categories, namely FIM Total Scale Score, Parent HRQOL Summary Score, Family Functioning Summary Score and eight individual domains, were found to be significantly higher among controls. Female gender of the affected child was an independent risk factor for poor Family Functioning Summary Score. Also, presence of serious complications during the course of the disease independently predicted poor Total FIM and Parent HRQOL Summary Scores. Even a relatively benign and potentially curable chronic disorder in children, like the NS, can disturb the QOL of parents in multiple domains of functioning.
|How to cite this article:|
Mishra K, Ramachandran S, Firdaus S, Rath B. The impact of pediatric nephrotic syndrome on parents' health-related quality of life and family functioning: An assessment made by the PedsQL 4.0 family impact module. Saudi J Kidney Dis Transpl 2015;26:285-92
|How to cite this URL:|
Mishra K, Ramachandran S, Firdaus S, Rath B. The impact of pediatric nephrotic syndrome on parents' health-related quality of life and family functioning: An assessment made by the PedsQL 4.0 family impact module. Saudi J Kidney Dis Transpl [serial online] 2015 [cited 2021 May 18];26:285-92. Available from: https://www.sjkdt.org/text.asp?2015/26/2/285/152420
| Introduction|| |
Nephrotic syndrome (NS) is one of the common chronic diseases affecting children worldwide, with a higher incidence in South Asian children. , Although nearly 80% of these children respond to treatment with corticosteroids,  about half of them have a frequently relapsing or a steroid-dependent course,  necessitating prolonged follow-up at the healthcare facility and requiring constant alterations of drug dosage. Such a disease pattern and its complications undoubtedly would demand a rather high level of social, emotional and physical adjustment, not only from the affected child but also from the family members. Most of the earlier studies from India have assessed the behavioral pattern of children with NS. ,, However, the brunt on the Indian families and their coping strategies are largely un-researched. Considering that there are a substantial number of families in the country living with a child with the NS, there is a need to study this aspect. With this background, we aimed to study all aspects of health-related quality of life (HRQOL) of families of children with chronic NS and to determine the predictors of a poor HRQOL.
| Methods|| |
A cross-sectional study was conducted in the pediatric nephrology clinic of a tertiary care medical college and hospital in Northern India. Ethical clearance was obtained from the Institutional Review Board. Eligible subjects included all children with chronic NS belonging to any of the three categories: Frequent relapsers, steroid dependent and steroid resistant (defined as per the standard Indian Society of Paediatric Nephrology guidelines  ), on treatment for at least one year. They were identified from the clinic register and were contacted telephonically to appear for the interview. Those with secondary NS or other associated chronic illnesses and those who denied consent were excluded from the study. A control group was chosen consisting of parents of healthy subjects attending the immunization clinic. The control group was matched for age, sex, family size and socioeconomic status (including education, occupation and monthly family income). Because this was a cross-sectional study, all the eligible subjects were chosen from the clinic register at a single instance and were contacted telephonically to appear for the interview on different days as per their convenience over a two-month period. The controls were interviewed simultaneously during the same period.
Details of all the cases, including their age, gender, socioeconomic status, type of NS, duration of treatment and complications during the course, were obtained from the records.
- The parents of both groups were asked to complete the standard self-administered multidimensional questionnaire of Pediatric Quality of Life Inventory 4 (PedsQL™) Family Impact Module (FIM).  As most parents could read and understand the local Hindi language, the already existing Hindi translated version of the questionnaire was obtained with permission from the MAPI Research Trust, France, after signing the User Agreement Form. The PedsQL FIM consists of 36 questions grouped under eight domains related to physical functioning, emotional functioning, social functioning, cognitive functioning, communication, worry, daily activities and family relationships. Recall time was one month and a five-point response scale was used from zero (never a problem) to four (almost always a problem). The scores of each item were then reversed and linearly transformed to a 0-100 scale (0 = 100, 1 = 75, 2 = 50, 3 = 25, 4 = 0), with higher PedsQL scores indicating better QOL. The PedsQL FIM Total Scale Score was calculated as the sum of all 36 items divided by the number of items answered. The Parent HRQOL Summary Score (20 items) was derived from the sum of the items divided by the number of items answered in the physical, emotional, social and cognitive functioning scales. The Family Functioning Summary Score (eight items) was derived from the sum of the items divided by the number of items answered in the Daily Activities and Family Relationships Scales.  Since its development in 2004, the PedsQL FIM has been used to study the effects of childhood illness on parental HRQOL, and the reliability and validity of the questionnaire has been established in many studies. ,,,, The internal consistency reliability was determined by calculating Cronbach's Alfa Coefficient. Values ≥0.70 were considered acceptable for comparisons between groups. 
Construct validity was established using the "known groups method." In order to determine the magnitude of the differences between families, effect sizes were calculated by taking the difference between the means of the two groups divided by the pooled standard deviation. Effect sizes for differences in means were designated as small (0.20), medium (0.50) and large (0.80). ,,
Descriptive statistics were performed to calculate the median and inter-quartile range for continuous data and frequencies with proportions for categorical data. The FIM Total Scale Score, Parent HRQOL Summary Score, Family Functioning Summary Score and scores in individual domains were compared between the cases and controls by the Mann-Whitney U test (for continuous parameters) and Odd's ratio (for categorical parameters). Additionally, possible predictors for poor QOL among the cases were assessed by both univariate and multivariate logistic regression analyses. Model fitting for multivariate models was tested by the "Hosmer and Lemeshow Goodness of Fit Test." All statistical analyses were performed using SPSS (version 16; SPSS Inc., Chicago, IL, USA).
| Results|| |
A total of 132 children with the NS were identified from the nephrology clinic register. After excluding patients who had either secondary NS or had other associated chronic illnesses, and those who could not be contacted, a total of 69 parents reported for the study, of whom eight guardians did not give consent. Thus, parents of 61 children (35 males and 26 females) were asked to complete the self-administered questionnaire of PedsQL TM FIM. Details of their corresponding children regarding age, sex, type of NS, duration of therapy and complications were obtained from the records [Table 1]. A total of 72 age, sex and socioeconomic status-matched parents (controls) also completed the same questionnaire.
[Table 2] shows the alfa coefficients (the internal consistency reliability) and effect sizes (validity) for the PedsQL™ FIM sub-scales. The effect size was large in all the sub-scales. Among the cases, the values of the alfa coefficients were above the recommended cut-off of 0.7 in all the sub-scales. In the control group, the alfa coefficients achieved values above 0.7 in the three major summary sub-scales, namely PedsQL FIM Total Scale Score, Parent HRQOL Summary Score and the Family Functioning Summary Score, but the values were low in some sub-scales. As the Cronbach's alfa coefficients exceeded the recommended minimum of 0.70 for the FIM Total Scale Score and the major summary scores, it demonstrated adequate homogeneity of the scale.  However, heterogeneity was seen when assessing each domain separately. Hence, these individual domains like social functioning, cognitive functioning, communication, worry and daily activities have been used disjointedly only for descriptive analyses.
|Table 2: Reliability (by Chronbach's alfa) and validity (by effect size) of the PedsQL FIMa in the different summary and sub-scales.|
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The comparison of scores in each of the categories, namely FIM Total Scale Score, Parent HRQOL Summary Score, Family Functioning Summary Score and eight individual domains, showed significantly higher values in each category among the controls as compared with the cases [Table 3]. Higher scores among controls, in each category, were also found taking either gender separately. Parents of diseased children were found to have the lowest scores in two domains, namely "worry" and "daily activities." A poor score in a domain was defined as a score below the fifth percentile of the median score of controls in that domain. Accordingly, the cut-off values for poor score in FIM Total Scale Score, Parent HRQOL Summary Score and Family Functioning Summary Score were <74.3, <68.3 and <64.5, respectively. The number of parents having poor scores in these sub-scales was found to be significantly higher among the cases compared with the controls [Table 4].
|Table 3: Comparison of median (inter-quartile range) PedsQL FIM scores between cases and controls in different functioning categories.|
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|Table 4: Comparison of number of parents with poor scores in the FIMa Total Scale Score, Parent HRQOL Summary and Family Functioning Summary Score.|
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A few possible demographic and clinical predictors like age, gender, socioeconomic status (including education, occupation and per capita income of parents), duration of therapy, serious complications requiring hospitalization during the course of the disease and therapeutic response to steroids, were assessed for their effect on the scores of parents of diseased children. Univariate analysis [Table 5] showed that parents with female affected children had a 3.4-fold risk of poor Family Functioning summary score compared with those with male children. Also, the presence of serious complications during the course of disease predicted poor scores in Total FIM and Parent HRQOL categories, the Odd's ratio being 7.52 (P = 0.005) and 7.81 (P = 0.002), respectively. A multivariate regression analysis model was created for each of the three summary scales assessing the adjusted effect of three risk factors (gender, complications and steroid resistance) on poor score [Table 6]. These three risk factors gave the best fit to the multivariate model, as tested by "Hosmer and Lemeshow Goodness of Fit Test." It was found that the presence of complications during the course of the disease was an independent risk factor for poor parental Total FIM and parent HRQOL Summary Scores. Also, having a female affected child was an independent predictor of poor family functioning summary score.
|Table 5: Univariate analysis for predictors of poor scores in FIMa Total Scale Score, Parent HRQOLb Summary Score and Family Functioning Summary Score categories.|
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|Table 6: Multivariate analysis for independent predictors for poor FIM Total Scale Score, Parent HRQOL Summary Score and Family Functioning Summary Score categories.|
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| Discussion|| |
The impact of the prolonged relapsing and remitting course of NS on the family of the affected child has been sparsely reported in the literature. A number of studies have been performed assessing the QOL and behavioral pattern of children with NS and how a stressful family environment has a detrimental effect on the children. ,,, However, not much information is found regarding the effect the children with NS have on their parents. The few existing studies on care-givers of children with NS have mainly focused on the parental stress aspect. ,, Our study brings forth the multidimensional impact on the life of parents of children with the NS. By using the PedsQL TM FIM module, a holistic idea about different aspects of QOL of parents could be assessed.
The above study demonstrates that parents of children with the NS fare significantly worse than parents of healthy children in the FIM Total Scale Score, the two major sub-scales as well as in each of the eight different domains. Notably, the major brunt was on their daily activities, with the worst affected domain being "worry." Such restrictions of daily activities, pursuing hobbies and travel have also been reported by parents in earlier studies on children with chronic renal failure. , It has been seen that parents of children with the NS manifested increased introversion and neuroticism compared with controls. , The prevalence of probable depression has also been observed to be significantly higher among care-givers of children on chronic peritoneal dialysis when compared with those of healthy children. As in our study, "worry" has also been found to be the most affected sub-scale among care-givers of children with strabismus. The apprehension of most parents is about the long-term prognosis of their children.  The qualitative analysis by Soliday et al indicated that the nature of concern among parents varied with the age of the child. In their study, parents of children younger than eight years were worried about their acting-out behaviors and attention span, whereas those of older children expressed academic concerns.  Such age-specific behavioral changes were not included in our questionnaire. Our study displays a multi-domain impact on parental QOL, parallel with many studies on children with chronic disorders. ,,
Unlike our results, the family impact of children with the NS, which was assessed by Vance et al, based on 10 questions, showed that cases and controls were remarkably similar, with "limitations on travel" being the only problem significantly more frequently seen among cases. The authors explained that their essentially negative findings were because of using closely matched controls, taking into account most of the confounding factors.  Similar results were also obtained by Soliday et al in families of children with kidney diseases. They conclude that most families managing their children for more than a year stabilize and adapt to their children's illness. 
Most studies from India have described the behavioral profile of children with the NS, but little information is available from the country regarding the families of such children. ,,, Mehta et al studied the level of anxiety among mothers of children with the NS and found high neuroticism scores in 65.7% of them compared with 30.4% of mothers of healthy children.  Another study from the country assessing the mental stress of care-givers of children with the NS found significantly higher rates of moderate to severe depression among them. 
We studied some factors that could possibly influence the parental QOL. Female gender of the affected child and serious complications during the course of the disease were found to have an independent negative impact on summary scores. Not much has been studied regarding predictors of poor QOL among care-givers of children with kidney diseases. While one study reported that depression was inversely proportional to the socioeconomic status,  another study concluded that it was most affected by the recent health status of the care-givers.  Tsai et al observed that greater happiness perception and higher income were associated with higher scores in the domains of environmental quality, social relationships and psychological condition of the life of care-givers of children on chronic peritoneal dialysis.  It has also been evidenced that day-to-day family life disruption is exacerbated during hospital admissions, comparable with our findings.  In children with developmental delay, child's age has been found to be inversely related with family impact, which perhaps indicates the gradual adaptation of families with the growth of their affected child.  Although we assessed factors like age of the child, duration of therapy, socio-economic status and type of NS, none of them had any significant influence on parental QOL scores. This could partly be explained by the fact that most patients attending our government medical college belong to more or less similar socioeconomic class. Also, the type of NS, whether steroid sensitive or resistant, did not make any difference on parental QOL scores as most of the steroid-resistant children achieved remission with other drugs like cyclosporine.
In many of the earlier reports, investigators have assessed the financial burden on families due to their child's treatment. , Our hospital, being a Government hospital, offers treatment and drugs free of cost and, hence, studying the financial stress was irrelevant. Unlike other authors, the parent-sibling relationship and the effect on the marriage have not been evaluated in the present study.
| Conclusion|| |
This study shows that even a relatively benign and potentially curable chronic disorder in children like the NS can disturb the QOL of parents in multiple domains of functioning. To the best of our knowledge, the PedsQL FIM module has been used for the first time in India for parents of children with the NS. Pediatric nephrologists should not be complacent with the therapeutic remission of the NS, until the parental grievances are duly addressed with adequate social support systems. 
Conflict of interest: None declared.
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Dr. Kirtisudha Mishra
Department of Pediatrics, Kalawati Saran Childrens' Hospital, Lady Hardinge Medical College, New Delhi
[Table 1], [Table 2], [Table 3], [Table 4], [Table 5], [Table 6]