Health-related Quality of Life in Children with Congenital Hydrocephalus and the Parental Concern: An Analysis in a Developing Nation.

PURPOSE: To analyze quality of life of children operated for congenital hydrocephalus and the concern of parents in taking care of these children.
METHODS: Thirty patients who underwent ventriculo-peritoneal shunt were randomly selected with minimum gap of 1 year between surgery and study. Canadian validated questionnaire was used. Overall health score (OHS) and parental concern score (PCS) were correlated with gender, family type and number of surgeries.
RESULTS: Mean OHS was 159.43 which was summation of physical health (mean 45.76), social-emotional (mean 80.03) and cognitive health scores (mean 33.66). Mean OHS was 151.57 for males and 177.77 for females (p-value 0.233). Nuclear and joint families had mean OHS of 160.36 and 158.89 respectively (p-value 0.944). Those who underwent one surgery had mean OHS of 167.48 and PCS of 23.10 whereas mean OHS was 140.66 and PCS was 27.78 for those with multiple procedures. Mean PCS for males was 26.71 and for females was 19.33 (p-value 0.036 statistically significant). This was not statistically significant between nuclear (24.73) and joint families (24.26).
CONCLUSIONS: Quality of life of survivors of hydrocephalus is reasonably good even in developing countries due to keen parental involvement irrespective of gender, family type and number of surgeries.

INTRODUCTION

Congenital hydrocephalus (CHC) is a common neurosurgical condition which if untreated, leads to the progressive cognitive deficit and early death. Cerebrospinal fluid diversion by a shunt is the procedure of choice, which is however associated with complications, mainly blockage and infection.[1] Even in the absence of these complications, a significant number of survivors have cognitive, social, emotional and physical deficits which impact their health-related quality of life (HRQOL). Studies related to long-term outcome have been done anecdotally in the developed world, mainly in their adolescence or adulthood.[23] Hence, we made an attempt to assess the HRQOL of hydrocephalus survivors in pediatric age group in resource constrained setting of a developing country. We analyzed 30 children without associated spina bifida in terms of HRQOL and the parental concern using a Canadian validated questionnaire.[45]

METHODS

Study group

A total of 30 consecutive patients of isolated CHC in the review outpatient department, who underwent a ventriculoperitoneal shunt in infancy, and had a minimum postoperative follow-up of 1 year were selected and analyzed for HRQOL. Those who did not fulfil the inclusion criteria (such as cases of CHC with spina bifida, cases who did not complete 1 year after surgery) were excluded from the study. There were 21 males and 9 females with their age at the time of study ranging from 4 to 17 years (median 8 years).

Instrument

Hydrocephalus outcome questionnaire given by Kulkarni et al. in 2004, comprising of 51 questions, was completed by the parents after informed consent, assuring the privacy of the interview.[4] The items in the questionnaire were categorized into three domains with 15 for physical, 24 for socialemotional, and 12 for cognitive health. The subscore was calculated with respect to each of these 3 domains for every patient, and their summated value was taken as overall health score (OHS). The OHS was analyzed in terms of gender, family type, and the number of surgeries. We also attempted to measure The Parental Concern Score (PCS) by using the questionnaire designed by Kulkarni et al. in 2006, which has items in an understandable and answerable pattern with ease.[5] The PCS was also analyzed with respect to gender, family type, and the number of surgeries.

Both mothers and fathers in the study showed equal interest in answering the questionnaire, alleviating the need for comparison of the reliability of answering between them. Despite the attempt to take socioeconomic status as a part of our study, we could not rely on the answers given regarding the exact per capita income to categorize them into low, middle, or high socioeconomic status, since almost all parents were reluctant in giving this detail.

Statistics

Unpaired t-test on Graph Pad software (GraphPad Software, Inc, California, USA) was used for statistical analysis.

OBSERVATION AND RESULTS

Epidemiological factors

On observing the occupation of parents, all fathers were working, but all mothers except two, were home makers taking care of the family. However, eight out of 30 (26.7%) mothers in the study attributed the child's illness as a cause for sacrificing the job, in spite of their adequate education. Eleven families were nuclear and 19 were joint with maternal or paternal relatives with their children staying in the same family. Approximately 87% (26/30) of children were studying in school of which only 2 (7.69%) of them attending special school for mentally challenged kids. Most of the parents attributed nonavailability of special school in their home town as a reason for sending the child to a normal school with compromise.

Surgery related factors

All 30 children had a functioning shunt at the time of the study. Twenty-one out of 30 (70%) children had undergone single surgery. Nine patients required more than one surgery for shunt related complications in the form of revision of shunt or endoscopic third ventriculostomy. Nearly 20% (6/30) of patients were on anti-epileptic medications on long-term basis.

Scores

The OHS ranged from 34 to 204 (mean 159.43). This was the amalgamation of 3 separate domains, namely physical health score, socialemotional health score and cognitive health score (CHS) as shown in Table 1.

Table 1

Mean and range of the health scores in the study population

The mean OHS was 151.57 for males and 177.77 for females, which was not statistically significant (P = 0.233). Nuclear and joint families had the mean OHS of 160.36 and 158.89, respectively (P = 0.944). Those who underwent one surgery had mean OHS of 167.48, and it was 140.66 for those who underwent more than one surgical procedure (P = 0.226). The PCS ranged from 4 to 36 with the mean of 24.5. The mean PCS for males was 26.71, whereas it was 19.33 for females with statistical significance (P = 0.036). The mean PCS was not having the statistically significant difference between nuclear and joint families, who had the score of 24.73 and 24.26, respectively (P = 0.894). Parents of the children who had undergone single surgery had a mean PCS of 23.10 and of those with more than one surgery had the score of 27.78 (P = 0.194) [Figure 1]. Almost all 30 parents had a concern about the block or infection of the shunt.

Figure 1

Overall health score and parental concern score for the variables analyzed

On categorizing the children in three age groups (4–7, 8–12, and 13–17 years), we did not observe any significant difference in the mean OHS or PCS with increasing age [Table 2].

Table 2

Overall health score and parental concern score in relation to different age groups

Additional observations

Two out of 30 children were left handers and 3 out of 30 children had precocious puberty for which the parents did not show any special attention or concern. One child with complete loss of vision was found to have OHS 146 out of 204 and CHS 40 out of 48.

DISCUSSION

Despite CHC being a common pediatric neurosurgical disorder, most of the available literature in English has concentrated on its pathogenesis, investigations, treatment modalities, and complications without much discussion on the HRQOL.[67] Apart from that, there is a Brazilian study emphasizing on the stress of caregivers of children with hydrocephalus, in which almost all the caregivers were in some or the other stage of stress.[8]

Because of its subjective nature, there is no definite assessment tool for evaluation of the quality of life; although, there are long-term studies which have assessed one or more of the parameters affecting the HRQOL in patients with CHC.[910] Studies which reported 10-year outcomes of hydrocephalus demonstrated a higher rate of mortality and shunt revision within the first decade of shunt placement without analyzing the HRQOL.[1112] Paulsen et al. in their 20-year follow-up study have reported a similar outcome in terms of shunt revision with HRQOL comparable to other young adults. However, the limitation of their study was the inability to apply Short Form-36 (SF-36) questionnaire in all these patients.[1] Further studies have used other assessment tools such as 15D overcoming the deficit of SF-36 by the inclusion of questions on memory functions.[2] Their observation was concordant with Paulsen et al. that, the associated motor impairment or epilepsy had some negative role in HRQOL. However, Lindquest et al. observed a better overall outcome than the previous studies using 15D.[11112] Associated spina bifida in these group of patients affects the HRQOL due to the motor deficit which was not a confounding factor in our study. Andersson et al. demonstrated the difference in the patterns of perpetual and cognitive visual dysfunction in operated patients of hydrocephalus and insisted on the care of vision to minimize the educational and social disadvantage.[13]

Parenting characteristics, anxiety, and depression of parents and caregivers of children with spina bifida have been analyzed and the impact of caregiver's age, child's age, shunt status, employment status, and income has also been demonstrated.[14] However, this type of parental assessment for children with CHC without spina bifida has not been done with a proper tool.

When we reviewed the literature with an attempt to validate HRQOL and parental assessment of CHC patients, we found the Canadian validated questionnaire designed by Kulkarni et al. as a better assessment tool and used in our population for analyzing those parameters. To the best of our knowledge, this is the first study analyzing the quality of life of hydrocephalus children in the setting of a developing nation. On analyzing the data, the HRQOL in our patients seems to be at a better level against anticipation. HRQOL and PCS were not significantly different with respect to family type and the number of surgeries. HRQOL was comparable between males and females. The significant difference of PCS between the parents of boys and girls depict the social stigma in developing countries with preferential care given to male children by parents. We believe that this type of HRQOL assessment studies can evoke enthusiasm for further similar studies in the developing countries, which in turn can elucidate the factors affecting the HRQOL in these children. Ubiquitous provision of special schooling can be one such modification which will help in improvising the quality of life of these children.

The pitfall in our study was the lack of control group due to which the real implication of OHS and PCS could not be elucidated with evidence. Unfortunately, we did not come across any previous study which used the same assessment tool, to help us in comparing the real difference of these scores between developed and developing countries.

CONCLUSIONS

The quality of life of survivors of CHC is reasonably good even in developing countries irrespective of gender, family type and a number of surgeries, although the parental concern was significant higher for male children. Keen parental involvement and concern seems to be the reason for the better than anticipated quality of life of these children even in resource constrained nations.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.