Comparative Analysis of the Quality of Life in Families with Children or Adolescents Having Congenital versus Acquired Neuropathology

Aim: This research aims to determine whether the time of injury (congenital or acquired) affects the quality of life (QOL) in families having a child or adolescent with neurological impairment. Design: Comparative, cross-sectional study. Material and methods: To find out if there are differences in the quality of life domains between these two groups, 66 subjects (31 mothers of patients with congenital disorders and 35 mothers of patients with acquired disorders) completed the PedsQL-Family Impact Module (PedsQL-FIM), the World Health Organization Quality of Life Instrument—Short Form (WHOQOL-BRIEF), and the Cognitive Emotion Regulation Questionnaire (CERQ). Results: Analyzing the PedsQL-FIM dimensions, we found significant differences between groups in terms of emotional functioning, communication, and worry, which favor the congenital group. There are no statistically significant differences between social functioning, cognitive functioning, and daily activities groups. No significant differences between groups when evaluating the WHOQOL-BRIEF’s domains (physical health, psychological health, social relationships, environment) have been found. According to CERQ results, adaptive strategies had higher mean scores in the congenital than in the acquired group. The mean score for maladaptive strategies in the congenital group is higher than that in the acquired one, except for catastrophizing, which is higher for acquired. Conclusion: Our findings show that the mothers of patients with acquired neuropathology have a lower quality of life in the emotional functioning, communication, and worry domains.


Introduction
Given the complexity of the concept of disability, there have been numerous attempts to define it. Thus, disability is a "shorthand expression" [1] or, according to the World Health Organization (WHO)'s International Classification of Functioning, Disability and Health (ICF), an "umbrella term for impairments, activity limitations or participation restrictions" [2,3] that are present from birth or develop over time. The impairments, mainly motor, sensory or cognitive, limit a child's or an adolescent's ability to perform necessary/required activities and interact with the surrounding environment [4].
For example, cerebral palsy is a "congenital" condition when the timing of the lesion is unknown (develops before birth, at birth, or shortly after delivery) [5,6]. It is usually PedsQL-Family Impact Module (PedsQL-FIM) [32] and World Health Organization Quality of Life Instrument, Short Form (WHOQL-BRIEF) [33], Romanian version. We also used the Cognitive Emotion Regulation Questionnaire (CERQ) [34] to determine the cognitiveemotional strategies our participants adopt when confronted with the challenge of the children's illness. All items in each questionnaire were surveyed.
The design of our study did not include a specific questionnaire to address the relationship between the family and the child with disabilities.
Subjects were invited to participate in the study voluntarily, enrollment taking place after they signed an informed consent form outlining specific aspects of the study (research description, confidentiality of collected data, their right to withdraw with researchers' notification, and data exclusion).
Subjects were asked to complete the Romanian paper version of the PedsQL-FIM, WHOQL-BRIEF, and CERQ questionnaires, which assessed their functionality in various dimensions/domains and conscious cognitive emotion regulation strategies. Basic demographic data were also gathered, including education, marital status, residency, age of their children, the time between the moment of the accident and data collection (for acquired neuropathology), and impairment severity. Medical records from the hospital were used to obtain information about the impairment severity. The classification by severity level was used to differentiate children with cerebral palsy into mild, moderate, and severe categories.
To determine the severity of traumatic brain injury (TBI), the Glasgow Coma Scale was used (mild, moderate, or severe).
All the data were collected between December 2019 and January 2022. We divided the enrolled subjects into two groups: Group 1 consisted of 31 mothers of patients with the congenital disorder (cerebral palsy of prenatal and perinatal origin), and Group 2 of 35 mothers of patients with the acquired disorder (traumatic brain injury after road/car accidents).

Research Tools
The first author received permission to use the Romanian version of the scales for this research, PedsQL-FIM from MAPI Research Trust, and WHOQOL-BREF from the WHO. For CERQ, permission to use the questionnaire was obtained from Cognitrom, a Romanian research organization authorized to commercialize its version in the Romanian language.

PedsQL-Family Impact Module (PedsQL-FIM)
The PedsQL-FIM is a tool created to assess the impact of having a child with health issues in the family [35]. According to the authors who developed it, its internal consistency reliabilities passed the acceptable minimal alpha coefficient standard of 0.70 for group comparisons [22]. This instrument contains 36 items divided into 8 dimensions, which are as follows: physical functioning (6 items), emotional functioning (5 items), social functioning (4 items), cognitive functioning (5 items), communication (3 items), worry (5 items), daily activities (3 items), and family relationships (5 items) [36]. We surveyed all the 36 items of the questionnaire. The mothers filled out the questionnaire based on how many problems they had in the previous month because of their child's illness.
For each evaluated item, a 5-point Likert scale ranging from 0 (never a problem) to 4 (almost always a problem) is used [37].
As higher the result of each component, as higher the quality of life is (the scores are obtained after the transformation of the variables).

World Health Organization Quality of Life Instrument, Short Form (WHOQL-BRIEF)
WHOQOL-BREF is a shorter version of the WHOQOL−100 that is used to assess QOL in healthy people and groups of patients with various pathologies [38][39][40].
Used in clinical studies or research, WHOQOL-BREF is regarded as a relevant and trustworthy measure for assessing the quality of life in the medical field [41]. Cronbach alpha values for each of the four domain scores ranged from 0.66 to 0.84 [42]. It has 26 questions, with Q1 and Q2 being two global items that inquire about "individual's overall perception of quality of life" (Q1) and "individual's overall perception of their health" (Q2) [33]. We surveyed all 26 items of the questionnaire. The WHOQOL-BREF has four domains: physical health (7 items), psychological health (6 items), social relationships (3items), and environment (8 items) [33]. Each item is graded on a 5-point Likert scale ranging from 1 to 5. Raw WHOQOL domain scores are converted to a 4-20 score based on standards. Domain scores are scaled in a synergist, positive direction; lower scores indicate lower QOL. The domain score is calculated using the mean score of items within each domain, then translated linearly to a 0-100 scale [33]. This questionnaire is usually ordered in the same manner as the quality of life, i.e., the higher values correspond to higher QOL.

Statistical Analysis
To process statistical data analysis, we used Excel 2019 (Microsoft Corporation, Redmond, WA, USA) and SPSS version 27 (IBM, Armonk, New York, NY, USA).
In case the Kolmogorov-Smirnov test on both groups accepted the normality of data. Hence, the acceptance of use of the t-test for Equality of Means use of the same independent groups was used to determine if the QOL is lower because of the injury (congenital or acquired). If not, the Mann-Whitney U test (for Equality of Medians) was used [46].
Computed p-values < 0.05 were considered to be statistically significant.

Socio-Demographic Features of the Groups
Sixty-six mothers of children or adolescents with congenital versus acquired neuropathology (Groups 1 and 2) completed PedsQL-FIM, WHOQOL-BREF, and CERQ questionnaires.
Children and adolescents in Group 1 ranged in age from 72 to 204 months (M = 101.77, SD = 28.63), while those in Group 2 ranged in age from 72 to 216 months (M = 154.57, SD = 45.56).
The time between injury and data collection for mothers of children with acquired neuropathology was between 12 and 24 months.
In terms of severity, 7 (22.58%) of children or adolescents in Group 1 had a severe impairment, 13 (41.94%) had a moderate impairment, and 11 (35.48%) had mild impairment. In Group 2, 12 (34.29%) children or adolescents had severe, 9 (25.71%) moderate, and 14 (40%) mild impairment. Table 1 summarizes the characteristics of our research groups in terms of education level, marital status, and residence. Using the Mann-Whitney U test, we found that the mean rank for Group 1 is 34.18 and for Group 2 is 32.90; p = 0.686, which is higher than 0.05, indicating that our groups are not statistically significantly different in education level. In terms of marital status, Group 1 has a mean rank of 30.35, while Group 2 has a mean rank of 36.29; p = 0.084. Our groups are not statistically significantly different neither on this feature. Group 1 has a mean rank of 33.53, whereas Group 2 has a mean rank of 33.47; p = 0.988. There are no statistically significant differences between groups based on the criterion of the place of residence.

Descriptive Statistics
We calculated the mean, median, and standard deviation for each group and each questionnaire. The results are presented in Table 2.

Data Analysis for PedsQL-FIM Dimensions
To determine whether there is a difference in the quality of life in a family having children with congenital versus acquired neuropathology, we examined the data for PedsQL-FIM dimensions. We used Kolmogorov-Smirnov test to verify the normality of data for both groups. We also found a possibly normal distribution of data for some variables in Group 2-physical functioning (p = 0.160), emotional functioning (p = 0.076), communication (p > 0.200), and worry (p > 0.200).
The Kolmogorov-Smirnov test rejected the normality of data for the following variables in Group 2: social functioning (p = 0.034), cognitive functioning (p = 0.040), daily activities (p = 0.002), and family relationships (p = 0.011).
We used the t-test to determine whether there were any statistically significant differences between groups for the variables with a normal data distribution (physical functioning, emotional functioning, communication, and worry).
p-values less than 0.05 were achieved for emotional functioning (p = 0.034), communication (p = 0.042), and worry (p = 0.036) indicating that there are significant differences between the two groups, Group 2 obtaining lower values (Table 3).
For physical functioning, we cannot assert the same; we can only say that the number of cases treated is too small to draw (statistically) a conclusion ( Table 3).
The Mann-Whitney U test (for Equality of Medians) was used to determine whether there are significant differences in social functioning, cognitive functioning, daily activities, and family relationships between groups (where the data were not normally distributed).
Social functioning (p = 0.489), cognitive functioning (p = 0.199), and daily activities (p = 0.103) all have p-values higher than 0.05, indicating that there are no statistically significant differences between the enrolled groups for these dimensions.
The result is not statistically significant for the family relationships (p = 0.057).

Data Analysis for WHOQOL-BREF Domain
We have also analyzed the data for each WHOQOL-BREF domain. Kolmogorov-Smirnov test rejected the normality of the related data, except for the psychological health variable (in Group 1, p = 0.069 and Group 2, p > 0.200), where this should be used with precaution.
To determine if there are statistically significant differences between groups for the variable psychological health, we used a t-test. Notice both means are apparently "close" one to the other mean for Group 1 is 14.96 and for Group 2 is 14.40 (Table 2).
Thus, the following result (p = 0.191) is not surprising and suggests no difference between groups regarding psychological health (Table 4). We used the Mann-Whitney U test (for Equality of Medians) for the variables: physical health, social relationships, and environment to see if there are statistically significant differences between groups on these variables.
The variables, physical health (p = 0.370) and environment (p = 0.479) have p-values greater than 0.05, indicating no statistically significant differences between the enrolled groups in these domains.
The difference is not statistically significant for social relationships (p = 0.058).

Data Analysis for CERQ Questionnaire
We calculated the mean score for each subscale of the cognitive strategies for regulating emotions that our groups used when faced with the challenge represented by their children's illness (Table 2).
Furthermore, we calculated the frequency of the subjects with the highest scores for each strategy for each group (Table 5). The Kolmogorov-Smirnov test revealed a normal distribution of data with p > 0.05 for: acceptance, rumination, and refocus on planning in both groups. The t-test indicated for: acceptance (p = 0.804), rumination (p = 0.155), refocus on planning (p = 0.332), no statistically significant differences between groups.
We used Mann-Whitney U test (for Equality of Medians) to see any significant differences between groups for the following variables that did not have a normal distribution of data: self-blame, positive refocusing, positive reappraisal, putting into perspective, catastrophizing, and other blame.

Discussion
To our knowledge, only studies comparing the quality of life of families with disabled children to those with normal development [47][48][49] or studies separately analyzing the quality of life of parents of children with congenital (cerebral palsy) [50] or acquired brain injuries (traumatic brain injury) [51,52] were available in the literature.
We did not find any comparative research on the quality of life in families with children with congenital versus acquired neurological disorders.
We believe that the importance of this study comes from the fact that no one has done anything similar before; therefore, we set out to perform it.
We found that there were no major differences between the groups. However, we supposed that the occurrence of a disabling event at a certain point in life could lead to a gap in previous normal life. As a result, coping strategies and quality of life differ from mothers with children with congenital impairment. We appreciate that for the family, regardless of when the injury occurs, at birth or after a period of normal development, it is a shock and a permanent challenge in the struggle with child disabling.
We found no statistically significant differences between our groups in terms of education, marital status, and place of residence. However, we cannot appreciate how the socio-demographic characteristics influence the quality of life in our studied groups.
We believe that differences in quality of life between families having a child with chronic disease due to economic status should be considered. A higher economic status influences the family, which has easier access to higher quality and innovative treatment in more developed countries and more performant medical assistive devices that are rarely compensated by standard health social programs.
The US literature showed that socio-demographic factors were associated with reduced QoL in families with children with chronic disease [53,54]. Some European countries with advanced economies and public health care identified them as having a risk for lower HRQOL in childhood. However, there are also European countries with public health care in which some studies report a minimal link between socioeconomic factors and QOL [53].
More studies agree that higher parental education can improve caregivers' knowledge by supporting better coping strategies for managing children's health and ensuring a better quality of life for themselves and their children [54,55]. Marital status is also important because tasks related to a disabled child's care and education are usually shared in married families, with the burden of care not falling solely on a single parent [55].
Because no comparative studies were found in the literature, based on our statistical analysis, we can estimate that regardless of the type of impairment, congenital or acquired, the domains of the quality of life, social functioning, cognitive, and daily activity were equally valued in both groups.
Given the significant parental burden caused by the physical and psychosocial difficulties of children with a chronic or acquired disease, we can appreciate that the family's structure, social, emotional, and relational functioning are disrupted.
In families with a disabled child, research has shown that illness severity and functional disability have a higher impact and may raise the chance of psychosocial issues [18,56].
Because the result for the family relationships has no statistical significance, we ca not clearly assess which group may have a higher or lower QOL in this domain.
We cannot say which group may have a higher or lower QOL on social relationships because the result for this domain has no statistical significance.
Considering the length of time of care and severity of disease could explain why most QoL parameters are nearly comparable in the two studied groups.
In families with children who have suffered a severe traumatic brain injury, long-term studies show that family relationships and functioning worsen between 3 and 36 months after the injury [57]. Long-term dependence harms family functioning in families with children with cerebral palsy [58].
Additionally, when completing the three questionnaires, the results enabled us to draw some conclusions regarding coping strategies.
Analyzing the mean score of CERQ, we found that the mean score for all adaptive strategies in Group 1 is higher than the mean score for all adaptive strategies in Group 2 ( Table 2). We also found that the mean score for maladaptive strategies in Group1 is higher than the mean score for maladaptive in Group 2, except for catastrophizing, where the mean score is higher for Group 2 ( Table 2).
Putting into perspective (45.1%) for Group 1 and catastrophizing (48.5%) for Group 2 are the strategies with the highest score frequency on the CERQ questionnaire.
Given the positive refocusing and putting into perspective strategies found to be better in Group 1, we can appreciate that parents with children with a chronic illness changed their perspective on life. Our study has the following limitations. (1) Self-administration of questionnaires could be considered a limitation due to misunderstanding questions. As a result, causing inaccurate responses despite the constant availability of investigators to clarify any question. (2) Considering that the evaluation tools gather data from a specific period (last month for PedsQL, last 2 weeks for WHQOL-BREF), mothers' responses are limited at this period. (3) Our study analyzed information collected only from caregivers admitted to the hospital. This could be an objective limitation of the study in the biparental families where only one parent completed the questionnaires.
Further research is needed with an increased number of participants and, consequently, higher statistical power for a deeper analysis.

Conclusions
According to our findings from the questionnaire analysis, the QOL of the mothers of patients with traumatic brain injury (acquired neuropathology) is lower than that of patients with cerebral palsy (congenital neuropathology) for emotional functioning and communication and worry domains.
At the same time, both groups chose adaptive strategies that psychologically support them in struggling with their children's illness and continuing the rehabilitation treatment. Informed Consent Statement: Informed consent was obtained from all subjects involved in the study. The participation of mothers of children or adolescents with congenital or acquired neuropathology in the study was voluntary. The enrollment was done after the parent/legal representative of the patient signed the informed consent where specific aspects of the study were explained.

Data Availability Statement:
The data sets used and analyzed during the current study are available from the corresponding author upon reasonable request.