Hydrocephalus-related quality of life as assessed by children and their caregivers

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  • 1 Department of Neurosurgery, Division of Pediatric Neurosurgery,
  • | 3 Department of Ophthalmology, and
  • | 4 Department of Biostatistics, University of Alabama at Birmingham, Alabama; and
  • | 2 School of Medicine, University of Mississippi Medical Center, Jackson, Mississippi
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OBJECTIVE

Hydrocephalus is a chronic medical condition that has a significant impact on children and their caregivers. The objective of this study was to measure the quality of life (QOL) of children with hydrocephalus, as assessed by both caregivers and patients.

METHODS

Pediatric patients with hydrocephalus and their caregivers were enrolled during routine neurosurgery clinic visits. The Hydrocephalus Outcomes Questionnaire (HOQ), a report of hydrocephalus-related QOL, was administered to both children with hydrocephalus (self-report) and their caregivers (proxy report about the child). Patients with hydrocephalus also completed measures of anxiety, depression, fatigue, traumatic stress, and headache. Caregivers completed a proxy report of child traumatic stress and a measure of caregiver burden. Demographic information was collected from administration of the Psychosocial Assessment Tool (version 2.0) and from the medical record. Child and caregiver HOQ scores were analyzed and correlated with clinical, demographic, and psychological variables.

RESULTS

The mean overall HOQ score (parent assessment of child QOL) was 0.68. HOQ Physical Health, Social-Emotional Health, and Cognitive Health subscore averages were 0.69, 0.73, and 0.54, respectively. The mean overall child self-assessment (cHOQ) score was 0.77, with cHOQ Physical Health, Social-Emotional Health, and Cognitive Health subscore means of 0.84, 0.79, and 0.66, respectively. Thirty-nine dyads were analyzed, in which both a child with hydrocephalus and his or her caregiver completed the cHOQ and HOQ. There was a positive correlation between parent and child scores (p < 0.004 for all subscores). Child scores were consistently higher than parent scores. Variables that showed association with caregiver-assessed QOL in at least one domain included child age, etiology of hydrocephalus, and history of endoscopic third ventriculostomy. There was a significant negative relationship (rho −0.48 to −0.60) between child-reported cHOQ score and child-reported measures of posttraumatic stress, anxiety, depression, and fatigue. There was a similar significant relationship between caregiver report of child’s QOL (HOQ) and caregiver assessment of the child’s posttraumatic stress symptoms as well as their assessment of burden of care (rho = −0.59 and rho = −0.51, respectively). No relationship between parent-reported HOQ and child-reported psychosocial factors was significant. No clinical or demographic variables were associated with child self-assessed cHOQ.

CONCLUSIONS

Pediatric patients with hydrocephalus consistently rate their own QOL higher than their caregivers do. Psychological factors such as anxiety and posttraumatic stress may be associated with lower QOL. These findings warrant further exploration.

ABBREVIATIONS

ASC-Kids = Acute Stress Checklist for Kids; cHOQ = children’s HOQ; CSDC = Child Stress Disorders Checklist; ETV = endoscopic third ventriculostomy; EVD = external ventricular drain; HCRN = Hydrocephalus Clinical Research Network; HOQ = Hydrocephalus Outcomes Questionnaire; PAT = Psychosocial Assessment Tool; PedMIDAS = Pediatric Migraine Disability Assessment; PROMIS = Patient-Reported Outcomes Measurement Information System; PTSS = posttraumatic stress symptoms; QOL = quality of life; VP = ventriculoperitoneal.

OBJECTIVE

Hydrocephalus is a chronic medical condition that has a significant impact on children and their caregivers. The objective of this study was to measure the quality of life (QOL) of children with hydrocephalus, as assessed by both caregivers and patients.

METHODS

Pediatric patients with hydrocephalus and their caregivers were enrolled during routine neurosurgery clinic visits. The Hydrocephalus Outcomes Questionnaire (HOQ), a report of hydrocephalus-related QOL, was administered to both children with hydrocephalus (self-report) and their caregivers (proxy report about the child). Patients with hydrocephalus also completed measures of anxiety, depression, fatigue, traumatic stress, and headache. Caregivers completed a proxy report of child traumatic stress and a measure of caregiver burden. Demographic information was collected from administration of the Psychosocial Assessment Tool (version 2.0) and from the medical record. Child and caregiver HOQ scores were analyzed and correlated with clinical, demographic, and psychological variables.

RESULTS

The mean overall HOQ score (parent assessment of child QOL) was 0.68. HOQ Physical Health, Social-Emotional Health, and Cognitive Health subscore averages were 0.69, 0.73, and 0.54, respectively. The mean overall child self-assessment (cHOQ) score was 0.77, with cHOQ Physical Health, Social-Emotional Health, and Cognitive Health subscore means of 0.84, 0.79, and 0.66, respectively. Thirty-nine dyads were analyzed, in which both a child with hydrocephalus and his or her caregiver completed the cHOQ and HOQ. There was a positive correlation between parent and child scores (p < 0.004 for all subscores). Child scores were consistently higher than parent scores. Variables that showed association with caregiver-assessed QOL in at least one domain included child age, etiology of hydrocephalus, and history of endoscopic third ventriculostomy. There was a significant negative relationship (rho −0.48 to −0.60) between child-reported cHOQ score and child-reported measures of posttraumatic stress, anxiety, depression, and fatigue. There was a similar significant relationship between caregiver report of child’s QOL (HOQ) and caregiver assessment of the child’s posttraumatic stress symptoms as well as their assessment of burden of care (rho = −0.59 and rho = −0.51, respectively). No relationship between parent-reported HOQ and child-reported psychosocial factors was significant. No clinical or demographic variables were associated with child self-assessed cHOQ.

CONCLUSIONS

Pediatric patients with hydrocephalus consistently rate their own QOL higher than their caregivers do. Psychological factors such as anxiety and posttraumatic stress may be associated with lower QOL. These findings warrant further exploration.

ABBREVIATIONS

ASC-Kids = Acute Stress Checklist for Kids; cHOQ = children’s HOQ; CSDC = Child Stress Disorders Checklist; ETV = endoscopic third ventriculostomy; EVD = external ventricular drain; HCRN = Hydrocephalus Clinical Research Network; HOQ = Hydrocephalus Outcomes Questionnaire; PAT = Psychosocial Assessment Tool; PedMIDAS = Pediatric Migraine Disability Assessment; PROMIS = Patient-Reported Outcomes Measurement Information System; PTSS = posttraumatic stress symptoms; QOL = quality of life; VP = ventriculoperitoneal.

In Brief

The objective of this study was to measure quality of life (QOL) of children with hydrocephalus, as assessed by caregivers and patients themselves. Using a convenience cross-sectional sample of children with hydrocephalus and their parents, the authors evaluated QOL using standardized survey instruments. This study is important because in order to improve the QOL of children with hydrocephalus, risk factors for lower QOL must be identified.

Hydrocephalus is a life-altering, chronic condition that has a significant impact on children and their caregivers. It is recognized that quality of life (QOL) can be negatively affected in pediatric patients with hydrocephalus and their families. Previous studies have demonstrated that children with hydrocephalus have lower QOL scores than children without hydrocephalus, as assessed by their caregivers.1 Lower QOL has been associated with socioeconomic factors such as family income and parental education, as well as disease-related factors such as the presence of epilepsy or the etiology of hydrocephalus.2

Almost all studies of QOL in children with hydrocephalus have used surveys completed by the parent or caregiver. To our knowledge, only 1 study has assessed child self-reported QOL;3 in that study, Kulkarni et al. reported a correlation between child and parent report of QOL.

The primary objective of this study was to measure the health-related QOL of children with hydrocephalus, as assessed by both caregivers and pediatric patients themselves. The secondary objective was to directly compare QOL assessment between caregiver-child dyad pairs. In addition, we sought to determine what factors were associated with lower QOL for these children and their caregivers, assessing factors previously established in the existing literature, as well as conducting the first study of psychological comorbidities and their association with QOL. We hypothesize that factors such as anxiety, depression, fatigue, severity of headache, and presence of posttraumatic stress symptoms (PTSS) will negatively correlate with QOL.

Methods

Setting

Study enrollment took place over a 7-month period in 2017–2018. Participants were recruited during their annual neurosurgical clinic visit at Children’s of Alabama, a tertiary referral center for pediatric neurosurgery in the southeastern US.

Study Population

Caregivers of pediatric patients with hydrocephalus and pediatric patients themselves were eligible for enrollment. Patients were eligible for enrollment if they were between 5 and 21 years of age and were capable of independently completing the survey as assessed by caregiver and research staff. Recognizing that some children with hydrocephalus have cognitive difficulties, research staff along with the caregiver determined if a child was capable of independently completing the questionnaire. This determination involved an assessment of level of consciousness, ability to read or to understand questions read aloud, ability to respond to the questions either verbally or in writing, and maturity level. Patients were eligible for enrollment regardless of whether their caregiver chose to enroll in the study.

Caregivers were eligible for enrollment if they were the legal guardian of a child with hydrocephalus between 5 and 21 years of age. In the event that the caregiver’s child was not eligible for enrollment due to inability to independently complete the research survey or refusal to participate, the caregiver was still eligible for enrollment. If more than one caregiver was present at their child’s clinic visit, both caregivers were eligible for enrollment; however, during the course of the study, in every case, only one caregiver was present or willing to participate. Therefore, study results represent the responses of the caregiver who was the primary caregiver in the context of the child’s neurosurgical clinic visit.

Because caregiver and patient enrollment were not mutually exclusive, there were 3 different study populations: 1) caregivers of children with hydrocephalus, 2) children with hydrocephalus, and 3) caregiver-child pairs (“dyads”) who both enrolled in the study. Only English-speaking participants were eligible, given the lack of availability of the study instruments in other languages. Study data were collected and managed using Research Electronic Data Capture tools hosted at the University of Alabama at Birmingham.4

Data Collection

Prior to enrollment, ethical approval for the study was obtained from the IRB of the University of Alabama at Birmingham. A research assistant, trained in both informed consent and research ethics, obtained consent from study participants during their routine pediatric neurosurgery clinic visit. Both parent/caregiver and child study participants completed the Hydrocephalus Outcome Questionnaire (HOQ), a 51-question instrument, to measure the health status in children living with hydrocephalus.5 The HOQ is a validated instrument for measuring QOL in children with hydrocephalus. The HOQ measures overall QOL and provides subscores to assess 3 domains: Social-Emotional Health, Cognitive Health, and Physical Health. The HOQ is primarily used as a parental proxy evaluation, with the parent or caregiver performing the assessment of their child. For children with hydrocephalus, the children’s HOQ (cHOQ) is a self-report version of the HOQ, including the same 51 items, scored in the same way.3 The HOQ is scored on a scale of 0–1, with higher scores indicating higher QOL. The HOQ has previously been used in caregivers of patients 5 years old and older, and the cHOQ has been used in pediatric patients (ages 6–19 years) with hydrocephalus.3,5

Demographic social variables were collected from the Psychosocial Assessment Tool (PAT 2.0), a validated parent report screen of psychosocial risk in pediatric health, and are summarized in Table 1.6 Distance of the child’s residence to Children’s of Alabama hospital was calculated from the patient’s most recent zip code, as obtained from the patient’s electronic medical record. Clinical variables such as etiology of hydrocephalus, number of hydrocephalus-related procedures, and history of shunt infection were collected from institutional records. All of these data points are collected prospectively as part of institutional participation in the Hydrocephalus Clinical Research Network (HCRN) registry. Additionally, the electronic medical record was utilized to collect information such as child’s gestational age at birth, total number of nontemporary hydrocephalus procedures, and total number of external ventricular drain (EVD) placements. Recognizing that shunts can fail repeatedly, and that this may increase the risk of PTSS, we defined a new variable to capture those patients who had a history of a cluster of shunt revisions. The variable, shunt cluster, was defined as having 3 or more hydrocephalus operations within a 2-month period.

TABLE 1.

Demographic and clinical variables

VariableCaregiver (n = 91)Child (n = 39)
Mean age ± SD, yrs11.41 ± 5.02 (n = 83)14.03 ± 4.50 (n = 39)
Sex, n (%)
 Female37 (41)19 (49)
 Male46 (51)20 (51)
Race, n (%)
 White55 (60)27 (69)
 Non-white28 (31)12 (31)
Etiology of hydrocephalus, n (%)
 Aqueductal stenosis4 (4)0 (0)
 Intraventricular hemorrhage22 (24)7 (18)
 Myelomeningocele17 (19)6 (15)
 Postinfectious7 (8)3 (8)
 Spontaneous hemorrhage1 (1)1 (3)
 Midbrain tumor or lesion3 (3)3 (8)
 Post-head injury4 (4)2 (5)
 Posterior fossa cyst1 (1)2 (5)
 Other intracranial cyst1 (1)1 (3)
 Communicating congenital12 (13)8 (21)
 Other congenital1 (1)0 (0)
 Unknown/missing2 (2)2 (5)
ETV alone (no shunt in lifetime), n (%)
 No70 (77)30 (77)
 Yes7 (8)4 (10)
History of shunt infection, n (%)
 No57 (63)27 (69)
 Yes21 (23)8 (21)
History of clustered shunt failure, n (%)
 No clusters64 (70)26 (67)
 Clusters12 (13)7 (18)
Total no. of EVDs, n (%)
 None48 (53)22 (56)
 ≥129 (32)12 (31)
Mean gestational age at birth ± SD, wks34.18 ± 5.98 (n = 74)35.19 ± 5.68 (n = 31)
Mean no. of hydrocephalus procedures ± SD4.28 ± 5.06 (n = 78)4.54 ± 6.54 (n = 35)
Type of health insurance, n (%)
 Other43 (47)19 (49)
 Private36 (40)17 (44)
Age of caregiver, n (%)
 ≥21 yrs73 (80)32 (82)
 Other8 (9)5 (13)
Caregiver education, n (%)
 College/graduate education42 (46)18 (46)
 Other39 (43)19 (49)
Caregiver marital status, n (%)
 Married/partnered57 (63)22 (56)
 Other24 (26)15 (38)
Mean distance from hospital ± SD, miles87.76 ± 57.55 (n = 77)91.44 ± 49.31 (n = 34)
Mean time from last surgery ± SD, mos67.66 ± 58.78 (n = 82)76.16 ± 71.58 (n = 39)
Child w/ developmental problems, n (%)
 Yes, or getting help22 (24)9 (23)
 Sometimes19 (21)9 (23)
 No28 (31)16 (41)

Average survey completion time was 30 minutes. No specific precautions were taken to avoid survey fatigue. Participants were not compensated for study participation. If subjects did not complete all survey measures, only completed surveys were included in data analysis.

Psychological Variables

To measure psychological variables, we used several validated survey instruments. To assess PTSS, we administered the Child Stress Disorders Checklist (CSDC), a parent report of child posttraumatic stress in children ages 2–18 years.7 In addition to this parent report of PTSS, we administered the Acute Stress Checklist for Kids (ASC-Kids), a child self-report measure for children ages 8–17 years.8 Pediatric patients completed three PROMIS (Patient-Reported Outcomes Measurement Information System) measures of psychological comorbidities in children ages 8–17 years: PROMIS Anxiety, PROMIS Depression, and PROMIS Fatigue scales.9 To measure the burden of headache, pediatric patients completed the Pediatric Migraine Disability Assessment (PedMIDAS), an assessment of migraine disability in patients ages 4–18 years.10 Finally, caregivers completed the Zarit Burden Interview, an assessment of caregiver perception of the burden of providing care for a loved one with a chronic illness and its impact on health, personal, and social well-being.11

Statistical Analysis

The 3 study group were analyzed. Demographic characteristics that are categorical were summarized using counts and percentages, while continuous demographic and outcome variables were summarized using means and standard deviations. The Kruskal-Wallis test was used to compare the continuous QOL and psychological scores for different categories of categorical variables that had small numbers in some categories (for example, age of caregivers, etiology of hydrocephalus, and endoscopic third ventriculostomy (ETV)–alone hydrocephalus treatment). In these cases, we prefer to use nonparametric tests, because tests for normality may not perform as well as needed with small numbers. For uniformity, we used the Kruskal-Wallis test for all other categorical variables as well. Scatterplots were examined, and Pearson correlation was estimated and tested (null hypothesis: correlation = 0) to examine the association among pairs of continuous variables. Parametric tests were used for these comparisons because the issue of small numbers noted above was not a concern. For the dyads, we calculated the differences in responses between the matched parent and child. We presented the mean and standard deviation of these differences. To determine if the parent responses differ significantly from the child, we used the Wilcoxon signed-rank test. Due to multiple tests performed, we used the Bonferroni method by dividing 0.05 by the total number of tests. Because this method is very conservative and this study is exploratory in nature, we made adjustments separately for parent and child analyses. In investigating which demographic and clinical characteristics are significantly associated with HOQ scores, the total number of variables used was 17 (listed in Table 1), hence a cutoff for determining significance of 0.0029. For comparing HOQ with 7 psychological variables, we use the cutoff of 0.0071 (i.e., 0.05/7). In all the tables, raw unadjusted p values were presented. All analyses were performed using the SAS statistical program (version 9.4, SAS Institute Inc.).

Missing Data

Missing clinical and demographic data were due to the following: caregiver did not complete PAT, pediatric patient not enrolled in the HCRN registry, and information not available in the electronic medical record. Only those with available data were included in a given analyses.

Results

Ninety-one caregivers and 39 children with hydrocephalus were enrolled in this study (Fig. 1). Among these cohorts, there were 39 dyad-pairs, in which both a child with hydrocephalus and their caregiver completed the HOQ and cHOQ. Detailed information on the sample of both caregivers and children can be found in Table 1.

FIG. 1.
FIG. 1.

Enrollment diagram.

The mean overall HOQ score (parent assessment of child QOL) was 0.68. HOQ Physical Health, Social-Emotional Health, and Cognitive Health subscore averages were 0.69, 0.73, and 0.54, respectively. Mean overall child self-assessment (cHOQ) score was 0.77, with cHOQ Physical Health, Social-Emotional Health, and Cognitive Health subscore means of 0.84, 0.79, and 0.66, respectively. We analyzed 39 dyads, in which both a child with hydrocephalus and his or her caregiver completed the cHOQ and HOQ, respectively. As expected, linear regression shows a positive correlation between parent and child scores (p < 0.004 for all subscores). Physical health showed the strongest correlation between parent and child scores (rho = 0.77). However, child scores were consistently higher than parent scores. This difference was statistically significant for physical QOL (Table 2).

TABLE 2.

Caregiver and child HOQ and dyad scores

Mean Caregiver Assessment ± SDDyad DifferenceDyad Pearson Correlation
HOQ Scoren = 91*n = 39Mean Child Self-Report ± SD (n = 39)Mean ± SDp ValueCorrelationp Value
Overall health0.68 ± 0.170.74 ± 0.140.77 ± 0.130.04 ± 0.130.0960.520.0007
Physical health0.69 ± 0.210.80 ± 0.140.84 ± 0.140.05 ± 0.090.00350.77<0.0001
Social-emotional health0.73 ± 0.170.76 ± 0.160.79 ± 0.140.03 ± 0.160.250.440.005
Cognitive health0.54 ± 0.280.62 ± 0.270.66 ± 0.230.04 ± 0.240.320.520.0006

Boldface type indicates statistical significance.

Parents who completed the survey instruments.

Parents who were part of a dyad.

Dyad difference = child score − caregiver score.

Variables that showed association with caregiver assessment of at least one domain of QOL prior to applying the Bonferroni correction (p < 0.05) were child age, sex, etiology of hydrocephalus, number of procedures, history of ETV alone (no ventriculoperitoneal [VP] shunt), and history of shunt infection (Table 3). After applying the Bonferroni correction, only child age (older age associated with higher physical QOL score), etiology of hydrocephalus (aqueductal stenosis associated with higher overall and physical QOL than other etiologies), and history of ETV (ETV without VP shunt associated with higher overall QOL) maintained significance (Table 3).

TABLE 3.

Caregiver-report HOQ correlation with clinical and demographic factors

Overall HealthCognitive HealthPhysical HealthSocial-Emotional Health
VariableMean (SD) or rhop ValueMean (SD) or rhop ValueMean (SD) or rhop ValueMean (SD) or rhop Value
AgeRho = 0.240.02rho = 0.320.0020.0950.38
Sex0.0080.020.020.05
 Male0.72 (0.16)0.60 (0.26)0.75 (0.18)0.76 (0.17)
 Female0.62 (0.17)0.47 (0.29)0.63 (0.23)0.69 (0.17)
Etiology of hydrocephalus0.020.030.00020.67
 Myelomeningocele0.62 (0.18)0.51 (0.25)0.57 (0.18)0.72 (0.20)
 Hemorrhage0.60 (0.20)0.40 (0.28)0.60 (0.25)0.71 (0.19)
 Aqueductal stenosis0.78 (0.16)0.68 (0.30)0.80 (0.15)0.83 (0.13)
 Other0.72 (0.14)0.62 (0.27)0.78 (0.18)0.74 (0.17)
No. of proceduresrho = −0.300.021rho = −0.230.04rho = −0.170.12rho = −0.300.007
ETV alone
 Yes0.85 (0.07)0.0020.82 (0.16)0.020.86 (0.10)0.020.86 (0.08)0.044
 No0.66 (0.17)0.52 (0.28)0.68 (0.22)0.72 (0.18)
Shunt infection
 Yes0.63 (0.16)0.130.57 (0.29)0.210.60 (0.24)0.0210.72 (0.17)0.57
 No0.70 (0.18)0.48 (0.27)0.73 (0.20)0.74 (0.18)
Age of main caregiver
 ≥21 yrs0.67 (0.17)0.300.54 (0.28)0.440.69 (0.21)0.580.73 (0.18)0.86
 Other0.73 (0.18)0.62 (0.27)0.79 (0.25)0.75 (0.15)
Child race
 White0.680.180.56 (0.29)0.330.69 (0.23)0.780.73 (0.18)0.86
 Non-white0.680.150.51 (0.26)0.72 (0.19)0.74 (0.16)
Caregiver education
 College/grad0.69 (0.16)0.900.55 (0.26)0.930.69 (0.22)0.710.75 (0.16)0.42
 Other0.68 (0.18)0.55 (0.29)0.71 (0.21)0.72 (0.19)
Marital status
 Married/partnered0.67 (0.17)0.230.54 (0.28)0.740.68 (0.21)0.130.72 (0.18)0.32
 Other0.71 (0.17)0.56 (0.28)0.74 (0.20)0.76 (0.16)
Type of health insurance
 Other0.67 (0.16)0.610.52 (0.26)0.150.71 (0.19)0.940.73 (0.18)0.82
 Private0.70 (0.18)0.60 (0.29)0.70 (0.22)0.74 (0.17)
No. of clusters
 None0.68 (0.18)0.450.54 (0.28)0.660.71 (0.22)0.140.74 (0.17)0.21
 ≥10.64 (0.17)0.58 (0.29)0.64 (0.20)0.67 (0.21)
Total no. of EVDs
 None0.70 (0.17)0.180.57 (0.28)0.310.73 (0.20)0.030.74 (0.17)0.58
 ≥10.64 (0.18)0.50 (0.28)0.63 (0.23)0.72 (0.18)
Gestational age at birthrho = 0.130.23rho = 0.200.08rho = 0.210.14rho = −0.0030.98
Distance from hospitalrho = 0.050.68rho = 0.010.94rho = 0.830.02rho = 0.070.54

Boldface type indicates statistical significance after correction for multiple measures (p < 0.0029).

As shown in Table 4, when considering child self-reported QOL, fewer variables showed significant association prior to application of correction for multiple measures (p < 0.05). Increasing number of hydrocephalus procedures was associated with lower overall (rho = −0.34, p = 0.03) and cognitive QOL scores (rho = −0.30, p = 0.008). Children whose parents had college or graduate school education had lower cognitive QOL than children of parents with lower levels of education (0.60 vs 0.75, p = 0.041). However, none of these associations maintained significance (p < 0.0029) after Bonferroni correction.

TABLE 4.

Child-report HOQ correlation with clinical and demographic factors

Overall HealthCognitive HealthPhysical HealthSocial-Emotional Health
VariableMean (SD) or Regression Coefficientp ValueMean (SD) or Regression Coefficientp ValueMean (SD) or Regression Coefficientp ValueMean (SD) or Regression Coefficientp Value
No. of hydrocephalus proceduresrho = −0.340.03rho = −0.300.008rho = −0.220.18rho = −0.260.10
Caregiver education
 College/grad0.76 (0.12)0.60 (0.23)0.0410.84 (0.12)0.360.78 (0.12)0.43
 Other0.81 (0.12)0.75 (0.19)0.87 (0.14)0.80 (0.15)
Age of main caregiver
 ≥21 yrs0.78 (0.13)0.840.68 (0.22)0.840.85 (0.13)0.210.79 (0.14)0.96
 Other0.80 (0.12)0.67 (0.23)0.90 (0.12)0.81 (0.12)
Sex
 Male0.83 (0.12)0.050.72 (0.21)0.180.88 (0.12)0.180.84 (0.11)0.06
 Female0.75 (0.12)0.63 (0.24)0.84 (0.14)0.75 (0.14)
Race
 White0.80 (0.14)0.330.70 (0.24)0.290.87 (0.13)0.360.81 (0.14)0.43
 Non-white0.77 (0.10)0.64 (0.22)0.84 (0.13)0.78 (0.13)
Marital status
 Married/partnered0.77 (0.13)0.610.67 (0.23)0.920.85 (0.14)0.770.78 (0.13)0.37
 Other0.80 (0.12)0.69 (0.22)0.86 (0.12)0.81 (0.14)
Type of health insurance
 Other0.77 (0.13)0.320.66 (0.22)0.440.82 (0.14)0.050.78 (0.14)0.91
 Private0.81 (0.12)0.71 (0.24)0.90 (0.09)0.80 (0.12)
ETV alone
 No0.80 (0.13)0.320.69 (0.24)0.750.88 (0.12)0.110.80 (0.14)0.56
 Yes0.73 (0.15)0.63 (0.22)0.74 (0.17)0.78 (0.14)
Etiology of hydrocephalus
 Myelomeningocele0.73 (0.10)0.120.61 (0.19)0.250.82 (0.09)0.090.72 (0.14)0.11
 Hemorrhage0.78 (0.17)0.70 (0.25)0.83 (0.19)0.78 (0.18)
 Aqueductal stenosis0.990.961.001.00
 Other0.81 (0.12)0.69 (0.25)0.89 (0.11)0.93 (0.10)
No. of clusters
 None0.81 (0.12)0.200.71 (0.23)0.230.89 (0.11)0.080.81 (0.13)0.42
 ≥10.73 (0.15)0.61 (0.23)0.79 (0.18)0.75 (0.16)
History of shunt infection
 Yes0.80 (0.13)0.180.59 (0.25)0.230.87 (0.14)0.270.75 (0.15)0.37
 No0.74 (0.10)0.70 (0.23)0.85 (0.10)0.81 (0.13)
Total no. of EVDs
 None0.79 (0.13)0.810.68 (0.23)0.930.87 (0.14)0.850.80 (0.13)0.78
 ≥10.79 (0.14)0.67 (0.26)0.86 (0.12)0.80 (0.16)
Gestational age at birthrho = −0.110.51rho = 0.200.89rho = −0.070.68rho = −0.200.25
Child agerho = 0.170.27rho = 0.250.10rho = 0.150.32rho = 0.030.83
Distance from hospital (miles)rho = −0.060.72rho = 0.030.83rho = −0.280.08rho = 0.0120.91

Boldface type indicates statistical significance.

Association of QOL With Psychological Comorbidities

When comparing overall QOL with components of the psychological assessment, several statistically significant associations were apparent. There was a negative relationship (rho −0.48 to −0.60) between cHOQ score and most other child-report measures (ASC-Kids, and the PROMIS Anxiety, Depression, and Fatigue scores). The exception was headache-related disability (PedMIDAS), which only approached statistical significance. Children who rated themselves higher on these scales also rated their overall QOL lower. However, there was not a significant association between parent-reported psychosocial assessments and child-reported cHOQ (Table 5). Similarly, we observed a significant relationship between the parent’s report of the child’s QOL (HOQ) and parent assessment of the child’s PTSS (CSDC) as well as their assessment of burden of care (rho = −0.59 and rho = −0.51, respectively). Here again, higher perceived burden of care and more severe PTSS were associated with lower overall QOL. No significant relationship between parent-reported QOL and child-reported psychosocial factors was seen. Correlations can be seen in Figs. 24.

TABLE 5.

HOQ correlation with psychological factors

Child-Reported QOLParent-Reported QOL
SurveyLRC (rho)p ValueLRC (rho)p Value
Child-reported measures
 ASC-Kids−0.490.0090.040.85
 PedMIDAS Headache Tool−0.500.0061−0.370.053
 PROMIS Anxiety−0.550.001−0.120.52
 PROMIS Depression−0.480.00550.060.76
 PROMIS Fatigue−0.60<0.001−0.250.18
Parent-reported measures
 Zarit Burden Interview−0.390.018−0.51<0.0001
 CSDC−0.1600.36−0.59<0.0001

LRC = linear regression coefficient.

Boldface type indicates a statistically significant association after correction for multiple measures (p < 0.007).

FIG. 2.
FIG. 2.

Graphs showing the correlation of child (left) and adult (right) HOQ with the PedMIDAS.

FIG. 3.
FIG. 3.

Graphs showing the correlation of child (left) and adult (right) HOQ with ASC-Kids.

FIG. 4.
FIG. 4.

Correlation of child (left column) and adult (right column) HOQ with PROMIS measures: Anxiety (A and B), Depression (C and D), and Fatigue (E and F).

Discussion

The purpose of this study was to evaluate QOL in pediatric patients with hydrocephalus, as assessed both by the children themselves and their caregiver. In addition, we sought to verify the relationship of QOL with clinical variables from previously published studies and conduct the first examination of psychological variables with QOL. Several previous studies have shown correlations between clinical variables and QOL.2 For example, children with myelomeningocele or intraventricular hemorrhage as the etiology of their hydrocephalus have been reported to have lower HOQ scores.12–14 We have found a similar relationship here, although only the physical component of QOL had a statistically significant association with hydrocephalus etiology. This may indicate that other components of these conditions (myelomeningocele and intraventricular hemorrhage of prematurity) that result in physical disability are a driving factor in lower QOL in these children.

Previous studies have suggested that children with more shunt-related complications have lower QOL scores.5,13 We have assessed this by testing the total number of lifetime shunt operations, history of shunt infection, total number of EVDs, and history of a cluster of shunt malfunctions (defined as 3 or more operations within 2 months). None of these variables showed significant association with QOL in our analysis. The total number of shunt operations approached statistical significance for overall, cognitive, and social-emotional QOL, but does not meet the significance threshold when controlling for multiple measures. In addition, correlation coefficients for these associations are low (rho = −0.23 to −0.3), indicating weak correlation.

Social and economic factors that have previously been shown to correlate with QOL scores include family income, family functioning, and parental education.13 We assessed these factors using components of the PAT. We found no significant association with child race, type of health insurance (private or public), age of caregiver (over age 21 years or under), caregiver marital status (married/partnered or single), or distance from the hospital. Caregiver education showed no relationship with parent-rated QOL but did show a possible relationship with child-related cHOQ score. This relationship appears paradoxical, with children whose parents had lower levels of education reporting higher cognitive QOL scores. However, this relationship did not maintain significance when corrected for multiple measures. Therefore, we would caution against placing much importance on this observation.

There were 39 parent-child dyads in this study, in which both caregiver and child completed the HOQ and cHOQ, respectively. It is important to note that these both measure the QOL of the child, and so a high level of correlation would be expected. Indeed, that is what we observed. Importantly, however, children rated their QOL higher than their parents, although that difference was only statistically significant for physical health scores. This may indicate that children with hydrocephalus perceive their own physical abilities differently than their parents. This is important to consider when designing studies or interventions focused on QOL. While parent QOL has been shown to be a reliable proxy, there may be enough difference to warrant specific attention on child self-report when possible.3

This is the first study to examine the relationship between QOL and psychological comorbidities such as anxiety, depression, fatigue, and PTSS. In contrast to clinical and demographic variables, where we observed few significant relationships, all psychological factors showed a correlation with QOL. Children who reported higher levels of PTSS, anxiety, depression, or fatigue all reported significantly lower overall QOL. However, none of these self-reported psychological factors showed significant association with caregiver-reported overall QOL. On the other hand, parental assessment of their child’s PTSS and of the level of burden involved in caring for the child both correlated with parent report of the child’s QOL. But these two parent-report measures had no association with child-reported QOL. It would appear that when assessing psychological comorbidities in hydrocephalus, it is critical to consider who is making the report.

This study has several limitations. Approximately 50% of the eligible patients seen in the clinic during the enrollment period were not screened for enrollment due to unavailability of the research assistant. A relatively smaller proportion declined participation in the study. Enrollment of less than 50% of eligible subjects could lead to a biased sample. However, given that the majority of nonparticipation was due to lack of availability of the research assistant, it is unlikely that this introduces significant bias. Additionally, caregivers and research staff made assessments of whether a child was capable of completing the child-report questionnaires. Because these judgements were somewhat subjective, there is a possibility of selection bias in the child self-report cohort, as only children able to complete the survey independently were able to enroll. Children with significant impairments, developmental delay, cognitive disability, and low maturity level were not eligible for enrollment, so QOL and psychosocial comorbidities were unable to be assessed in these populations. It is also possible that children with more complicated hydrocephalus or more frequent shunt problems were in clinic more often, and thus more likely to be approached for participation in this study. This would also lead to selection bias, potentially enriching our sample for more severely affected children. The HOQ itself contains questions that have some overlap with PROMIS measures. This could artificially increase the degree of correlation observed between these measures. Nevertheless, we suggest that anxiety, depression, and fatigue are important comorbidities for which screening and treatment may improve QOL in these patients.

Conclusions

Psychological factors such as anxiety, depression, fatigue, and traumatic stress may have a significant association with QOL for pediatric patients with hydrocephalus, although these factors have not been well-studied in this population. Child-reported QOL was significantly associated with child-reported symptoms, but not caregiver-reported psychological symptoms, so future studies should focus on child-reported symptoms whenever feasible. In contrast to many of the clinical and demographic variables that have been the focus of previous QOL studies, psychological factors may be modifiable. Further study of traumatic stress, anxiety, depression, and fatigue is warranted. Routine assessment of these comorbidities in the clinical setting could lead to recognition of problems and referral for appropriate treatment. If QOL is to be improved in this population, further research must include these factors when assessing the overall health and well-being of pediatric patients with hydrocephalus.

Acknowledgments

This work was supported by NIH grant no. TL1 5TL1TR001418-03 to K.Z.

Disclosures

The authors report no conflict of interest concerning the materials or methods used in this study or the findings specified in this paper.

Author Contributions

Conception and design: Rocque, Zimmerman, Arynchyna, Dreer. Acquisition of data: Rocque, Zimmerman, May, Barnes, Arata Wessinger. Analysis and interpretation of data: Rocque, Zimmerman, May, Arynchyna, Aban. Drafting the article: Rocque, Zimmerman, May, Alford, Chagoya, Arata Wessinger, Aban. Critically revising the article: all authors. Reviewed submitted version of manuscript: Rocque, Zimmerman, May, Barnes, Arynchyna, Alford, Chagoya, Arata Wessinger, Dreer, Johnston, Rozzelle, Blount. Approved the final version of the manuscript on behalf of all authors: Rocque. Statistical analysis: Rocque, Zimmerman, Aban. Administrative/technical/material support: Rocque, Zimmerman, Barnes, Arynchyna. Study supervision: Rocque, Zimmerman, Arynchyna, Johnston, Rozzelle, Blount.

Supplemental Information

Previous Presentations

Portions of this work were presented orally at the 2018 AANS/CNS Section on Pediatric Neurosurgery in Nashville, Tennessee, and the 2019 AANS Annual Meeting in San Diego, California.

References

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    Pai ALH, Patiño-Fernández AM, McSherry M, et al. The Psychosocial Assessment Tool (PAT2.0): psychometric properties of a screener for psychosocial distress in families of children newly diagnosed with cancer. J Pediatr Psychol. 2008;33(1):5062.

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    Kassam-Adams N. The Acute Stress Checklist for Children (ASC-Kids): development of a child self-report measure. J Trauma Stress. 2006;19(1):129139.

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  • 9

    DeWalt DA, Gross HE, Gipson DS, et al. PROMIS® pediatric self-report scales distinguish subgroups of children within and across six common pediatric chronic health conditions. Qual Life Res. 2015;24(9):21952208.

    • Search Google Scholar
    • Export Citation
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    Hershey AD, Powers SW, Vockell AL, et al. PedMIDAS: development of a questionnaire to assess disability of migraines in children. Neurology. 2001;57(11):20342039.

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    Bédard M, Molloy DW, Squire L, et al. The Zarit Burden Interview: a new short version and screening version. Gerontologist. 2001;41(5):652657.

    • Search Google Scholar
    • Export Citation
  • 12

    Karmur BS, Kulkarni AV. Medical and socioeconomic predictors of quality of life in myelomeningocele patients with shunted hydrocephalus. Childs Nerv Syst. 2018;34(4):741747.

    • Search Google Scholar
    • Export Citation
  • 13

    Kulkarni AV, Cochrane DD, McNeely PD, Shams I. Medical, social, and economic factors associated with health-related quality of life in Canadian children with hydrocephalus. J Pediatr. 2008;153(5):689695.

    • Search Google Scholar
    • Export Citation
  • 14

    Kulkarni AV, Shams I. Quality of life in children with hydrocephalus: results from the Hospital for Sick Children, Toronto. J Neurosurg. 2007;107(5)(suppl):358364.

    • Search Google Scholar
    • Export Citation

Illustration from Guida et al. (pp 346–352). Copyright Lelio Guida. Published with permission.

Contributor Notes

Correspondence Brandon G. Rocque: University of Alabama at Birmingham, AL. brandon.rocque@childrensal.org.

INCLUDE WHEN CITING Published online July 10, 2020; DOI: 10.3171/2020.4.PEDS19660.

Disclosures The authors report no conflict of interest concerning the materials or methods used in this study or the findings specified in this paper.

  • View in gallery

    Enrollment diagram.

  • View in gallery

    Graphs showing the correlation of child (left) and adult (right) HOQ with the PedMIDAS.

  • View in gallery

    Graphs showing the correlation of child (left) and adult (right) HOQ with ASC-Kids.

  • View in gallery

    Correlation of child (left column) and adult (right column) HOQ with PROMIS measures: Anxiety (A and B), Depression (C and D), and Fatigue (E and F).

  • 1

    Khan SA, Khan MF, Bakhshi SK, et al. Quality of life in individuals surgically treated for congenital hydrocephalus during infancy: a single-institution experience. World Neurosurg. 2017;101:247253.

    • Search Google Scholar
    • Export Citation
  • 2

    Kulkarni AV. Quality of life in childhood hydrocephalus: a review. Childs Nerv Syst. 2010;26(6):737743.

  • 3

    Kulkarni AV, Cochrane DD, McNeely PD, Shams I. Comparing children’s and parents’ perspectives of health outcome in paediatric hydrocephalus. Dev Med Child Neurol. 2008;50(8):587592.

    • Search Google Scholar
    • Export Citation
  • 4

    Harris PA, Taylor R, Thielke R, et al. Research electronic data capture (REDCap)—a metadata-driven methodology and workflow process for providing translational research informatics support. J Biomed Inform. 2009;42(2):377381.

    • Search Google Scholar
    • Export Citation
  • 5

    Kulkarni AV, Rabin D, Drake JM. An instrument to measure the health status in children with hydrocephalus: the Hydrocephalus Outcome Questionnaire. J Neurosurg. 2004;101(2)(suppl):134140.

    • Search Google Scholar
    • Export Citation
  • 6

    Pai ALH, Patiño-Fernández AM, McSherry M, et al. The Psychosocial Assessment Tool (PAT2.0): psychometric properties of a screener for psychosocial distress in families of children newly diagnosed with cancer. J Pediatr Psychol. 2008;33(1):5062.

    • Search Google Scholar
    • Export Citation
  • 7

    Saxe G, Chawla N, Stoddard F, et al. Child Stress Disorders Checklist: a measure of ASD and PTSD in children. J Am Acad Child Adolesc Psychiatry. 2003;42(8):972978.

    • Search Google Scholar
    • Export Citation
  • 8

    Kassam-Adams N. The Acute Stress Checklist for Children (ASC-Kids): development of a child self-report measure. J Trauma Stress. 2006;19(1):129139.

    • Search Google Scholar
    • Export Citation
  • 9

    DeWalt DA, Gross HE, Gipson DS, et al. PROMIS® pediatric self-report scales distinguish subgroups of children within and across six common pediatric chronic health conditions. Qual Life Res. 2015;24(9):21952208.

    • Search Google Scholar
    • Export Citation
  • 10

    Hershey AD, Powers SW, Vockell AL, et al. PedMIDAS: development of a questionnaire to assess disability of migraines in children. Neurology. 2001;57(11):20342039.

    • Search Google Scholar
    • Export Citation
  • 11

    Bédard M, Molloy DW, Squire L, et al. The Zarit Burden Interview: a new short version and screening version. Gerontologist. 2001;41(5):652657.

    • Search Google Scholar
    • Export Citation
  • 12

    Karmur BS, Kulkarni AV. Medical and socioeconomic predictors of quality of life in myelomeningocele patients with shunted hydrocephalus. Childs Nerv Syst. 2018;34(4):741747.

    • Search Google Scholar
    • Export Citation
  • 13

    Kulkarni AV, Cochrane DD, McNeely PD, Shams I. Medical, social, and economic factors associated with health-related quality of life in Canadian children with hydrocephalus. J Pediatr. 2008;153(5):689695.

    • Search Google Scholar
    • Export Citation
  • 14

    Kulkarni AV, Shams I. Quality of life in children with hydrocephalus: results from the Hospital for Sick Children, Toronto. J Neurosurg. 2007;107(5)(suppl):358364.

    • Search Google Scholar
    • Export Citation

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