Child, Parent, and Family Adjustment for Patients Followed in a Multidisciplinary Spina Bifida Clinic

Abstract

Objectives:

To characterize child, parent, and family adjustment for patients followed in a multidisciplinary spina bifida (SB) clinic.

Methods:

Participants were drawn from clinical cases seen through a multidisciplinary outpatient SB clinic at a children’s hospital between 2017 and 2019. Participants included 209 youth under 19 years old who were diagnosed with SB and their parents. Self-reported internalizing symptoms were measured in youth in grade 3 through 12 using the 25-item Revised Children’s Anxiety and Depression Scale-25 (RCADS-25). Self- and parent-reported quality of life and family functioning were obtained using the Pediatric Quality of Life Inventory (PedsQL) 4.0 Generic Core Scales and Family Impact Modules.

Results:

A total of 45.7% of children and adolescents reported at-risk psychosocial functioning on the PedsQL. In contrast, only 5% of patients reported clinically elevated internalizing symptoms on the RCADS. Parents’ quality of life and family functioning in the study were higher than in most studies of parents of children with other chronic health conditions, children with attention deficit-hyperactivity disorder, and healthy control samples.

Conclusion:

Our findings indicate that children and adolescents with SB are at risk for poor health-related quality of life (HRQOL); however, poorer HRQOL may not necessarily be associated with more severe psychiatric symptoms in this population. Examining resilience factors that may help to buffer against challenges to HRQOL will be important in informing future interventions.

Introduction

Spina bifida (SB) is a chronic condition that has a multisystemic impact on children and adolescents.^1,2 Youth with SB are at risk for physical, neurocognitive, psychosocial, and family challenges, but there is also substantial heterogeneity within the SB population. Previous studies of psychosocial functioning in youth with SB and their families have produced mixed results. Some previous research has indicated that youth with SB and their parents are more likely to experience high levels of stress and emotional difficulties, but other research has highlighted resilience in this population.^2–7 We previously investigated parent-reported stress and adolescent internalizing symptoms for youth with SB followed in a multidisciplinary SB clinic⁸ and found low levels of parent and adolescent distress. To further our understanding of child, parent, and family adjustment for children seen in the clinic, this study built on our previous study by capturing a broader age range of the youth sample and by adding measures of child and adolescent health-related quality of life (HRQOL) and parent-reported family functioning. In addition, we investigated associations among child health-related variables, child-reported internalizing symptoms and HRQOL, and parent quality of life (QOL) and parent-reported family functioning in the SB sample to gain further insight into factors that may influence adjustment within this population.

Child Adjustment

Quality of life

Children with SB are at significant risk for lower HRQOL. Children and adolescents with SB not only report greater impairment in HRQOL than healthy controls but also report more impaired HRQOL than youth with other chronic health conditions (e.g., asthma, diabetes).^9–14 Youth with SB face many medical and neurocognitive challenges, which have important implications for physical health and psychosocial development. Several medical factors have been found to be related to lower HRQOL in youth with SB, including presence of a shunt,^13,15 impaired bowel and bladder continence,^15,16 history of Chiari malformation type II decompression,¹⁵ low mobility independence,^11,15,17 pain,¹¹ and SB diagnosis of myelomeningocele.¹⁵ Age has also been found to be related to HRQOL in youth with SB, with younger children reporting better HRQOL than older children.¹⁵ Cognitive and psychosocial factors such as reduced executive functioning skills; higher life stress and depressive symptoms; and lower social competence, peer social engagement, and family functioning have also been linked to lower HRQOL.^1,11,18 Many youth with SB are less socially engaged and socially mature, are more passive, report fewer romantic relationships, and have fewer positive peer and social experiences compared to typically developing children.^3,19–21

Child anxiety and depression

Research findings are varied regarding whether youth with SB are at increased risk for elevated internalizing symptoms. In several studies, adolescents and young adults with SB have reported high rates of anxiety and depressive symptoms, low self-worth, and elevated suicidal ideation.^22–24 In contrast, our previous study found low levels of adolescent internalizing symptoms compared to both the general population and populations with other health conditions.⁸ Even in studies where adolescents with SB exhibit similar median or mean levels of internalizing symptoms as typically developing teens, there remained a large subset of SB adolescents with clinically elevated symptoms of anxiety/depression (e.g., 23.5% of SB adolescents with elevated scores vs. 7.4% in the control group).^3,25 Internalizing symptoms in youth with SB are often related to family functioning, perceived parental support, school experiences, youth attitudes toward SB, and self-reported pain.^{11,19,22–24,26} In a longitudinal study, Lennon et al.⁴ found that in youth with SB, better neurocognitive functioning predicted better social competence, which was associated with lower depressive symptoms 2 years later.

Parent Adjustment

Parenting a child with a chronic illness such as SB often involves acute, stressful life events (e.g., diagnosis, surgery) and daily chronic stressors (e.g., bladder/bowel management, mobility difficulties).⁷ The presence of SB in families has been shown to predict higher levels of parent psychological distress, with particular stress reported around medical, psychosocial, financial, and time responsibilities associated with caring for a child with SB.^6,7,27 One study of SB caregivers found a large percentage of parents had elevated anxiety and depressive symptom scores (e.g., 49% reported elevated anxiety and 21% reported elevated depression symptoms ).²⁵ Caregivers of children with SB report fewer hours of sleep each night and less engagement in leisure and social activities as compared to parents of typically developing children.²⁸ Parents of youth with SB who have a shunt report more anxiety and depressive symptoms and perceive their child as being more vulnerable than children with SB without a shunt.²⁹ In addition to shunt status, a number of other factors have been shown to be associated with adjustment among parents of children with SB including child factors (e.g., age, internalizing symptoms, independence, bowel/bladder management, ambulation status), parent factors (e.g., extraversion, emotional stability, parenting competence, hope), family factors (e.g., income, partner relationship), and psychosocial/contextual factors (e.g., social support, life stressors).^6,7,30

Family Adjustment

Challenges faced by youth with SB and their families, such as bladder and bowel management, delayed autonomy development, and executive functioning and social skills difficulties, can place significant strain on family relationships.³¹ SB families may have particular difficulties with establishing and maintaining defined roles and responsibilities within the family.²² Observational assessments of family behavior suggested that SB families are less cohesive and youth are more passive compared to control families; this relationship was partially mediated by lower child verbal IQ.³² Additionally, low family socioeconomic status was an added risk factor for heightened conflict and less family cohesion among SB families.³² Difficulties with bladder and catheterization management may also contribute to strain in SB family relationships.³³ Despite these challenges, rates of dysfunction in SB families are lower than that found in some other pediatric populations with chronic health conditions.³⁰

Although demands associated with SB can disrupt normative family functioning in some areas, many families display significant adaptation and resilience.^2,8 Notably, cultural factors can play a protective role in family functioning. Latino/a SB families demonstrated less family conflict during interactions compared to non-Latino/a, Caucasian SB families.³⁴ Family conflict was further associated with friendship quality among Latino/a SB youth.³⁴ SB families may present with unique strengths and trajectories that are simply different from that of typically developing youth. For example, SB families experience less parent–child conflict during adolescence compared to control families, perhaps due to differences in autonomy development.^35,36

Multidisciplinary Care

The challenges of SB can be managed, but they require daily attention and oversight that can be difficult for many youth and families. Suboptimal management can have lasting (and even in some extreme cases, fatal) effects. Child, family, and parent adjustment is increasingly being understood as a primary driver in optimal outcomes for children with SB, particularly as it relates to adherence and overall adjustment. For example, Holmbeck and Devine² propose a bioneuropsychosocial model of adjustment, specific for youth with SB, which states that adjustment and adherence to medical regimens is determined by biological, neuropsychological, and social influences. This highlights the importance of the individual, parent, and family systems as they relate to both physical and psychosocial outcomes.

Multidisciplinary teams have become common in SB clinics,³⁷ and yet they often lack a psychologist who is regularly integrated into clinic visits, care teams, and treatment plans.^38,39 This is surprising, given that systems of care, psychosocial context for self-management, and neuropsychology are all explicitly included as main areas of focus within the 2018 Guidelines for the Care of People with Spina Bifida, designed to highlight necessary elements of care delivery for clinics serving people with SB.^40,41 Pediatric psychologists are specifically trained to work with a multidisciplinary team to address the complex physical and psychosocial needs of families and youth with chronic health conditions. Beginning in 2020, SB clinics that successfully adopt the newly updated guidelines can be designated as a Spina Bifida Association (SBA) Clinic Partner and become associated with the only agency in the United States whose focus is on improving the lives of people with SB³⁹ through consolidated efforts for research, education, delivery of care, and other services essential for optimal outcomes.

Current Study

Building upon our previous study that characterized parent-reported stress for parents of children with SB,⁸ this study utilized standardized measures that capture a wider age range of participants and additional adjustment domains including child HRQOL and parent-reported family functioning. Prior research has assessed SB family functioning in a variety of ways, including the use of the Family Assessment Device, Family Environment Scale, and observations of family interactions.³¹ However, to our knowledge, no prior studies of SB have assessed family functioning using the Pediatric Quality of Life Inventory (PedsQL) Family Impact Module,⁴² despite its use with many other pediatric health populations.^42–46 The PedsQL Family Impact Module is uniquely designed to assess family functioning within the context of caring for a child with chronic health conditions.

The current study did not include a comparison group, but we made statistical comparison to previously reported results for community samples and samples of children with psychiatric or other chronic health conditions. We hypothesized that our sample of children with SB and their parents would report poorer HRQOL, higher rates of internalizing symptoms, and more impaired family functioning compared to community and healthy control samples. We additionally investigated associations among child health-related variables, child-reported internalizing symptoms and HRQOL, and parent QOL and parent-reported family functioning in the SB sample to gain further insight into factors that may influence adjustment within this population. We hypothesized that higher child internalizing symptoms and markers of more severe medical problems (e.g., higher lesion level, presence of shunt, more frequent bladder/bowel accidents, reduced mobility) would be associated with poorer self-reported HRQOL, parent QOL, and family functioning.

Methods

Participants

The project was a retrospective chart review of clinical data collected as part of the standard of care in a multidisciplinary outpatient SB clinic and was approved by the university-affiliated institutional review board. Participants were drawn from clinical cases seen through a SB clinic at a children’s hospital between 2017 and 2019. Patients were considered eligible if they were under 19 years old at the time of their clinic appointment and had a diagnosis of SB. Of the 218 eligible patients, 209 completed at least one questionnaire and were included in the study. There were no demographic differences for the nine participants who were excluded from the current study due to missing data, with the exception of age at encounter. Excluded patients were on average younger (M = 6.33 years, SD = 3.04) than patients included in the study (M = 8.78, SD = 4.14) (t = −2.27, p = .046). Self-reported internalizing symptoms (n = 122) and psychosocial HRQOL (n = 116) were measured in youth in grade 3 through 12 using the 25-item Revised Children’s Anxiety and Depression Scale-25 (RCADS-25) and the PedsQL.⁴⁷ Caregivers were asked to complete measures of family functioning (n = 184) and parent QOL (n = 172) using the PedsQL Family Impact Module. Caregiver respondents included 158 mothers, 18 fathers, 5 guardians, and 6 identified as “other” or “N/A.” Independent samples t tests revealed no significant differences between parent and child variables when comparing mothers and fathers. Demographic and health-related information for participants is provided in Table 1.

Table 1.

Participant demographics

Sample demographics (N = 209)	n (%)	M (SD)
Sex (male)	107 (51.2)
Latinoethnicity	80 (38.3)
Race
White	151 (72.2)
Asian	14 (6.7)
Black/African American	4 (1.9)
American Indian/Alaska Native	1 (0.5)
Multiracial	6 (2.9)
Other	29 (13.9)
Unknown/Not reported	4 (1.9)
Age, years		8.78 (5.14)
Diagnosis
Myelomeningocele	173 (82.8)
Lipomyelomeningocele	28 (13.4)
Meningocele	8 (3.8)
Lesion level*
Sacral	62 (29.7)
Low lumbar	27 (12.9)
Mid lumbar	95 (45.5)
High lumbar	10 (4.8)
Thoracic	14 (6.7)
Shunt	130 (62.2)
Ambulation status**	53 (25.4)
Nonambulator	25 (12.0)
Therapeutic and/or household ambulator	25 (12.0)
Community ambulator	111 (53.1)
Bladder continence***
Never accidents	74 (35.4)
Less than monthly (less than one accident per month)	18 (8.6)
Monthly (accidents once a month or more)	24 (11.5)
Weekly (accidents once a week or more)	25 (12.0)
Daily (accidents once a day or more)	61 (29.2)
Insurance type
Private	86 (41.1)
Public	123 (58.9)

^*Missing data for n = 1.

^**Missing data for n = 20.

^***Missing data for n = 7.

Procedure

Parents of patients aged 19 and younger were asked to complete the PedsQL. All patients in grade 3 through 12 were asked to complete the PedsQL 4.0 Generic Core Scales ⁴⁸ and RCADS-25.⁴⁷ Background information was collected from interviews and the electronic medical record. Lesion level was determined based on the following functional indicators during physical examination: thoracic lesion level displayed flaccid lower extremities, high lumbar lesion level displayed the presence of hip flexion, mid lumbar lesion level displayed the presence of knee extension, low lumbar lesion level displayed the presence of foot dorsiflexion, and sacral lesion level displayed the presence of foot plantarflexion. If functional status was different on the right versus left side, lesion level was determined based on the side of the body with more severe functional impairment.

The multidisciplinary outpatient SB clinic runs 1 day per week from 8 a.m. to 12 p.m. Up to eight patients are typically scheduled. Providers from the following disciplines are scheduled to see most patients: rehabilitation medicine, neurosurgery, orthopedics, urology, genetics, physical therapy, nutrition, rehabilitation psychology, and social work. Many patients are also seen by the hospital’s adaptive sports program coordinator, who provides information regarding local adaptive sport opportunities and assists with registration. The standard questionnaires gathered during clinic are handed out by a medical assistant to parents and patients upon check-in. Parents and patients are provided a brief description of the measures and instruction on how to complete them. The psychologist utilizes the questionnaire data to assist with evaluation of parent and child psychosocial functioning during the clinic visit. If concerns with patient or parent psychosocial functioning are identified, brief intervention, in addition to internal or community referrals for follow-up behavioral health treatment when indicated, is provided by the psychologist during the clinic appointment.

Measures

Health-related quality of life

Child and adolescent HRQOL was assessed with the age-appropriate version of the PedsQL 4.0. The PedsQL consists of 23 items in physical, emotional, social, and school functioning domains. The 15 items assessing emotional, social, and school functioning are combined to create a single psychosocial health summary score. The eight physical functioning items were modified to be more inclusive of youth with varying levels of mobility (e.g., “It is hard for me to walk or push my wheelchair more than one block”). Despite these modifications, the questions focus on physical functioning rather than perceptions of how SB impacts engagement in various physical activities, which increased the risk for negatively skewed results for participants in this study. Therefore, we chose to only include the psychosocial subscale in data analyses. In addition, the physical functioning subscale is no longer included as part of the standard questionnaires used in our SB clinic. Responses are reverse scored and then converted to a scale from 0 (poor HRQOL) to 100 (excellent HRQOL). A self-report PedsQL psychosocial health summary score below 66.03 indicates at-risk status for impaired psychosocial HRQOL.⁴⁹ Within the current sample, the PedsQL was found to have very good internal consistency (α = .91).

Parent quality of life and family functioning

Parent report of their QOL and family functioning within the context of having a child with a chronic health condition was assessed using the 36-item PedsQL Family Impact Module. The PedsQL Family Impact Module was designed to measure the impact of pediatric chronic health conditions on parents and the family. Specifically, parents are asked to rate on a scale of 0 (never) to 4 (almost always) how much of a problem they have had and the family has had over the past month in the following domains as a result of their child’s health: 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). A Parent QOL summary score is computed by averaging the 20 items comprising the Physical, Emotional, Social, and Cognitive Functioning scales. The family functioning summary score is computed by averaging the eight items comprising the Daily Activities (e.g., “family activities taking more time and effort,” “difficulty finding time to finish household tasks”) and Family Relationships (e.g., “conflicts between family members,” “difficulty solving problems together,” “stress or tension between family members”) scales. Within the current sample, the PedsQL Family Impact Module was found to have excellent internal consistency (α = .97).

Child and adolescent internalizing symptoms

To evaluate child and adolescent internalizing symptoms, the RCADS-25⁴⁷ was completed by youth in grades 3 through 12 (n = 122). The RCADS-25 asks participants to rate on a scale of 0 (never) to 3 (always) how often they have experienced 15 different anxiety symptoms and 10 different depressive symptoms. Scores are summed and yield two subscales (total anxiety and total depression) and one overall score (total anxiety and depression). In this study, we used the total anxiety and depression score as a measure of internalizing symptoms. Both the anxiety and depression subscales have a strong relation to diagnostic groups. The subscales of the RCADS-25 have been found to be reliable and valid in a variety of clinic- and school-based samples.⁴⁷ Within the current sample, the RCADS-25 was found to have good internal consistency (α = .85). The developer uses a t score of 70 as the clinical threshold and t scores of 65 to 69 to describe the borderline clinical range.

Data Preparation

Patients and/or parents who completed at least one questionnaire were included in the current study. We examined all variables for departures from normality and extreme outliers. All variables had acceptable skew and kurtosis (between −2 and +2). We created psychosocial HRQOL, parent QOL, family functioning, and internalizing symptoms summary scores (described previously) as recommended by the publishers.^42,48

To characterize parent and child psychosocial adjustment in the present sample, primary analyses first examined descriptive statistics for child HRQOL, parent QOL, parent-reported family functioning, and child internalizing symptoms. We used independent sample t tests to compare our sample’s functioning on these four constructs to other previously published samples. Previously published samples were identified via literature search using the PubMed database and included studies utilizing an identical measure within a child or adolescent population. Comparison samples were organized into categories based on sample characteristics: participants with SB, other chronic health conditions, and psychiatric disorders; community samples; and healthy controls. We identified no comparison samples that reported t scores from the RCADS-25 but found five comparison samples that published t scores from the long version of the instrument (i.e., RCADS-47). We compared the short and long versions of the RCADS and found that although the items assessing anxiety are different across the two instruments, the items in the depression subscale are the same on both measures. Therefore, for the sample comparisons, we looked only at the depression subscale.

Second, we examined associations among parent QOL, parent-reported family functioning, child and adolescent HRQOL, child and adolescent internalizing symptoms, demographic variables, and child health-related variables using Pearson correlations in this sample. For these analyses, we used the Total Anxiety and Depression Scale from the RCADS rather than just the depression subscale to maximize reliability.

Results

Psychosocial questionnaires

Descriptive statistics for the psychosocial screeners are presented in Tables 2–6. A total of 45.7% of children and adolescents reported at-risk psychosocial functioning on the PedsQL. In contrast, only 5% of patients reported clinically elevated internalizing symptoms on the RCADS (based on the total anxiety and depression score on the RCADS-25). We did not identify a previously published study documenting a recommended clinical cutoff or rates of impairment for the PedsQL Family Impact Module.

Table 2.

Pediatric Quality of Life Inventory self-report psychosocial score comparisons

Study	Sample	n	M	SD	Cohen’s d	t	df	p
Current study	Children aged 8–19 with SB	116	67.47	17.46
Spina bifida
Ramachandra et al., 201313	Children aged 5–18 with SB	33	70.80	17.10	0.19	−0.98	52	.3304
Murray et al., 201510	Children aged 8–15 with SBa	134	65.19	16.45	−0.13	1.06	238	.2912
Abresch et al., 20079	Children aged 11–21 with SBb	42	65.07	20.18	−0.13	0.68	64	.496
Chronic health conditions
Klinnert et al., 201451	Children aged 5–18 with eosinophilic esophagitis	59	74.90	17.10	4.75	−2.70	118	.008
Varni et al., 200754	Children aged 5–18 with diabetes	300	77.34	14.62	0.61	−5.40	180	<.00001
Varni et al., 200148	Children aged 5–18 with chronic health conditions	367	77.10	15.84	0.58	−5.29	178	<.00001
Varni et al., 200754	Children aged 5–18 with gastrointestinal conditions	280	76.18	14.63	0.54	−4.73	185	<.00001
Abresch et al., 20079	Children aged 11–21 who were obese	21	74.4	11.09	0.48	−2.40	40	.0212
Varni et al., 200754	Children aged 5–18 with cardiac conditions	250	74.88	16.10	0.44	−3.87	208	.0002
Varni et al., 200255	Children aged 5–18 with rheumatic conditions	231	74.23	16.46	0.40	−3.47	218	.0006
Varni et al., 200754	Children aged 5–18 with end stage renal disease	85	73.54	14.80	0.38	−2.66	194	.0084
Varni et al., 200754	Children aged 7–12 with asthma	162	73.95	18.35	0.36	−2.99	254	.003
Graves et al., 201650	Children aged 10–17 with sickle cell disease	75	73.30	15.90	0.35	−2.38	168	.0184
Varni et al., 200754	Children aged 5–18 with rheumatologic conditions	336	72.67	17.07	0.30	−2.78	196	.006
Varni et al., 200253	Children aged 5–18 with newly diagnosed cancer on treatment	219	72.62	16.41	0.30	−2.62	222	.0094

Table 2.

Pediatric Quality of Life Inventory self-report psychosocial score comparisons (cont.)

Study	Sample	n	M	SD	Cohen’s d	t	df	p
Varni et al., 200754	Children aged 5–18 with cancer	393	72.10	16.31	0.27	−2.55	178	.0118
Varni et al.49	Children aged 5–16 with chronic health and psychiatric conditions (e.g., asthma, ADHD, depression, diabetes)	573	71.32	17.13	0.22	−2.17	162	.0312
Varni et al., 200754	Children aged 5–18 with obesity	63	72.10	14.10	0.29	−1.93	151	.056
Mah et al. 200652	Adolescents presenting to a pediatric neurology clinic	104	69.70	15.90	0.13	−0.99	217	.3226
Varni et al., 200754	Children aged 5–18 with cerebral palsy	77	68.11	16.52	0.04	−0.26	169	.797
Abresch et al., 20079	Children aged 11–21 with spinal cord injuryb	61	67.76	15.38	0.02	−0.11	136	.9098
Psychiatric disorders
Limbers et al., 201156	Children aged 5–18 with a physician diagnosis of ADHD and a comorbid psychiatric disorder seen in an outpatient psychiatric clinic	179	61.89	17.57	−0.32	2.67	246	.008
Limbers et al., 201156	Children aged 5–18 with a physician diagnosis of ADHD seen in a general pediatric clinic	17	70.69	18.35	0.18	−0.68	20	.5044
Varni et al., 200754	Children aged 5–18 with psychiatric disorders	296	67.40	14.70	0.00	0.04	182	.9696
Community samples
Varni et al., 200349	Children aged 5–16 recruited from a community sample, including healthy children and children with chronic health conditions	5963	80.73	14.70	0.82	−8.12	118	<.00001
Varni et al., 200148	Children aged 5–18 who were acutely ill and presenting to a specialty clinic, with no chronic health conditions	148	78.68	14.66	0.70	−5.55	223	<.00001

Table 2.

Pediatric Quality of Life Inventory self-report psychosocial score comparisons (cont.)

Study	Sample	n	M	SD	Cohen’s d	t	df	p
Mansour et al., 200357	Children aged 6–13 living in an urban and predominately low-income setting	525	64.60	17.20	−0.17	1.61	167	.11
Healthy controls
Abresch et al., 20079	Healthy children aged 11–21	39	87.71	8.77	1.47	−9.44	130	<.00001
Varni et al., 200349	Healthy children aged 5–16	5070	81.83	13.97	0.91	−8.79	118	<.00001
Varni et al., 200754	Healthy children aged 5–18	5480	81.87	14.09	0.91	−8.82	118	<.00001
Varni et al., 200148	Healthy children aged 5–18	399	82.38	15.51	0.90	−8.30	171	<.00001

Note: Light grey shading indicates that the current study scored significantly lower than the comparison sample. No shading indicates no significant differences. ADHD = attention deficit-hyperactivity disorder; SB = spina bifida; SCI = spinal cord injury.

^aWeighted mean of two time points.

^bWeighted mean of youth who were and were not obese.

Table 6.

Pearson correlations among patient- and parent-reported psychosocial variables

		1	2	3	4
1.	Child psychosocial HRQOL
2.	Child internalizing symptomsa	−.55
3.	Parent HRQOL	.37	−.33
4.	Family functioning	.33	−.28	.76

Note: HRQOL = health-related quality of life.

p < .05.

^aA composite of anxiety and depressive symptoms.

Sample comparisons

Health-related quality of life

Child and adolescent self-report of psychosocial HRQOL in this study was similar to other studies of pediatric SB populations (see Table 2).^{9,10,13,48–57} Further, patients had reduced psychosocial HRQOL when compared to youth with other chronic health conditions and community and healthy control samples in other studies (Table 2).

Parent quality of life and family functioning

Parents QOL and family functioning in this study were higher than in most studies of parents of children with other chronic health conditions, children with attention deficit-hyperactivity disorder (ADHD), and healthy control samples (Tables 3 and 4).^{42,46,51,56,58–60}

Table 3.

Parent health-related quality of life comparisons

Study	Sample	n	M	SD	Cohen’s d	t	df	p
Current study	Children aged 0–19 with spina bifida	172	78.86	19.475
Chronic health condition
Varni et al., 200442	Children with complex health conditions living at home (e.g., severe CP, birth defects)	11	62.94	19.83	−0.81	2.58	11	.0254
Scarpelli et al., 200846	Children aged 2–18 undergoing cancer treatmenta	95	65.24	16.09	−0.76	6.13	226	<.00001
Medrano et al, 201358	Children aged 2–7 with chronic health conditions	383	66.9	16.1	−0.67	7.05	280	<.00001
Klinnert et al., 201451	Children aged 2–19 with eosinophilic esophagitis	97	68.9	19.9	−0.51	3.97	195	.0002
Isa et al., 201359	Children aged 2–18 with disabilities	425	75	18.74	−0.20	2.22	305	.0274
Kunz et al., 201160	Children aged 11–18 with inflammatory bowel diseaseb	129	81.69	16.57	0.16	−1.36	294	.175
ADHD
Limbers et al., 201156	Children aged 5–18 with a physician diagnosis of ADHD and a comorbid psychiatric disorder seen in an outpatient psychiatric clinic	177	58.94	20.27	−1.00	9.36	346	<.00001
Limbers et al., 201156	Children aged 5–18 with a physician diagnosis of ADHD seen in a general pediatric clinic	17	76.01	19.92	−0.14	2.58	11	.0254
Healthy controls
Medrano et al, 201358	Children aged 2–17 without chronic health conditions	546	71.2	14.9	−0.4	4.74	237	<.00001

Note: Dark grey shading indicates that the current study scored significantly higher than the comparison sample. No shading indicates no significant differences. ADHD = attention deficit-hyperactivity disorder; CP = cerebral palsy.

^aWeighted mean of inpatient and outpatient sample.

^bWeighted mean of maternal and paternal report.

Table 4.

Family functioning comparisons

Study	Sample	n	M	SD	Cohen’s d	t	df	p
Current study	Children aged 0–19 with spina bifida	184	77.38	21.038
Chronic health condition
Scarpelli et al., 200846	Children aged 2–18 undergoing cancer treatmenta	95	59.67	22.17	−0.82	6.43	181	<.00001
Medrano et al., 201358	Children aged 2–17 with chronic health conditions	383	62.50	18.30	−0.75	8.22	319	<.00001
Varni et al., 200442	Children with complex health conditions living at home (e.g., severe CP, birth defects)	11	68.81	24.11	−0.38	1.15	10	.2758
Klinnert et al., 201451	Children aged 2–18 with eosinophilic esophagitis	97	69.40	23.90	−0.35	2.77	175	.0062
Kunz et al., 201160	Children aged 11–18 with inflammatory bowel diseaseb	129	81.40	17.77	0.21	−1.83	300	.0688
ADHD
Limbers et al., 201156	Children aged 5–18 with a physician diagnosis of ADHD and a comorbid psychiatric disorder seen in an outpatient psychiatric clinic	177	47.71	23.84	−1.32	12.52	349	<.00001
Limbers et al., 201156	Children aged 5–18 with a physician diagnosis of ADHD seen in a general pediatric clinic	17	61.91	22.16	−0.72	2.77	18	.0128
Healthy controls
Medrano et al., 201358	Children aged 2–17 without chronic health conditions	546	67.60	18.40	−0.49	5.62	283	<.00001

Note: Dark grey shading indicates that the current study scored significantly higher than the comparison sample. No shading indicates no significant differences. ADHD = attention deficit-hyperactivity disorder; CP = cerebral palsy.

^aWeighted mean of inpatient and outpatient sample.

^bWeighted mean of maternal and paternal report.

Child and adolescent depressive symptoms

Depression T scores in this study were similar to children with inflammatory bowel disease and children seen in an urban primary care clinic. Depression scores in this study were lower compared to children with dengue fever, ADHD, and healthy control samples (Table 5).^61–64

Table 5.

Revised Children’s Anxiety and Depression Scale, Depression T score comparisons

Study	Sample	n	M	SD	Cohen’s d	t	df	p
Current study	Children aged 8–19 with spina bifida	119	43.73	9.423
Pediatric health conditions
Herbuela et al., 202061	Children aged 8–17 with dengue fever	225	52.3	9.87	0.89	−7.89	250	<.00001
Walter et al., 201662	Children aged 11–18 with inflammatory bowel disease	161	44.22	12.27	0.04	−0.38	277	.7058
ADHD
Becker et al., 201963	Children aged 8–12 evaluated for suspected ADHD at an ADHD specialty clinic (83% met criteria for ADHD)	117	46.88	11.17	0.30	−2.34	226	.0202
Community samples
Hourigan et al., 201564	Children aged 8–17 in an urban pediatric primary care clinic	197	45.34	10.19	0.16	−1.43	264	.1548
Healthy controls
Herbuela et al., 202061	Healthy children aged 8–17	260	49.30	8.64	0.62	−5.48	211	<.00001

Note: Light grey shading indicates that the current study scored significantly lower than the comparison sample. No shading indicates no significant differences. ADHD = attention deficit-hyperactivity disorder.

Associations with parent and child psychosocial functioning

Self-reported HRQOL, self-reported total internalizing symptoms, parent-reported QOL, and parent-reported family functioning were all unrelated to demographic variables (sex, race/ethnicity, age, and insurance type). HRQOL, internalizing symptoms, and parent QOL were also unrelated to ambulation status, bladder continence, diagnosis, and lesion level. Family functioning was lower on average for patients with a shunt (M = 74.50, SD = 20.79) compared to those without (M = 81.86, SD = 20.77) [t(182) = 2.34, p = .02]. It is important to note, however, that parent-reported family functioning in the subset of patients with a shunt was still higher than in most studies of parents of children with other chronic health conditions, children with ADHD, and healthy control samples (Table 4).

The associations among patient- and parent-reported psychosocial variables are presented in Table 6. Although rates of clinically elevated self-reported internalizing symptoms were low, internalizing symptoms showed moderate associations with self-reported HRQOL. In addition, significant associations were also found between child psychosocial HRQOL, parent QOL, and family functioning.

Discussion

Building upon our previous study that characterized parent-reported stress for parents of children with SB,⁸ this study utilized standardized measures that capture a wider age range of participants and additional adjustment domains including child HRQOL and parent-reported family functioning. We hypothesized that higher child internalizing symptoms and markers of more severe medical problems (e.g., higher lesion level, presence of shunt, more frequent bladder/bowel accidents, reduced mobility) would be associated with poorer self-reported HRQOL, parent QOL, and family functioning.

Previous research investigating the impact of SB on child, parent, and family adjustment has shown that, on average, children with SB and their parents report poorer QOL,^9–14 higher rates of anxiety and depressive symptoms,^3,22–25 and more impaired family functioning³² compared to community and healthy control samples.

Consistent with prior literature, slightly less than half of the children and adolescents in our sample reported impaired psychosocial HRQOL. When compared to numerous other studies of youth with chronic health conditions and community and healthy control samples, children and adolescents in this study exhibited reduced psychosocial HRQOL.^9,10,48–55 This result appears to highlight the multisystemic impact SB has on the adjustment and functioning of children and adolescents across various stages of development.

Similar to our previous study,⁸ parent QOL in our current sample population was better than most studies of parents of children with a chronic health condition, children with ADHD, and healthy control samples. These data speak to resilience in this population and converge with previous studies^8,32,35 in suggesting that parents of children with SB can be well adjusted in many ways, despite the numerous acute and chronic stressors associated with caring for a child with SB.

Although rates of clinically elevated self-reported internalizing symptoms were low (only 5% in the present sample), internalizing symptoms showed moderate associations with self-reported HRQOL and parent-reported QOL. Contrary to our prediction, both self- and parent-reported psychosocial functioning were unrelated to demographic variables (sex, race/ethnicity, and age) and health-related variables (SB diagnosis, lesion level, ambulation status, bladder continence). However, we did find that parent-reported family functioning was lower for youth with SB who had a shunt than those who did not. This result is consistent with prior research showing health-related variables to be negatively associated with both self-reported HRQOL and parent QOL in youth and young adults with SB.^6,7,15,16,30 The reason for the association between shunted hydrocephalus and family functioning is not known. Hydrocephalus conveys increased risk for attentional weaknesses and other neurobehavioral challenges, which could in turn negatively impact the ability of some participants to participate in daily household tasks and family activities. Parents of children with a shunt may also experience heightened stress due to the need to monitor for potential shunt malfunctions and participate in additional medical appointments, which could in turn impact family functioning.

It is important to note the striking differences we found in psychosocial adjustment across measures. We found clear evidence of HRQOL concerns in the present sample, but little evidence of child and adolescent internalizing symptoms. It appears that the concept of HRQOL better captures the type of adjustment difficulties that this population faces as compared to a focus on symptoms of depression and anxiety. Results also converge with our previously published study⁸ in demonstrating resilience in this population. Despite poorer HRQOL than children with many other chronic health conditions, children with SB in our sample had lower rates of depressive symptoms than a healthy control sample. Their parents also reported better family functioning than families of children with several other chronic health conditions and even better than families of healthy controls. Various protective factors in the current sample may act as a buffer to the challenges faced in social, emotional, and school functioning and defend against the development of more severe internalizing symptoms. Prior research has linked specific individual and family resiliency factors, such as better attitudes about having SB and higher levels of parental responsiveness, to more adaptive outcomes in individuals with SB.^24,65 Additional resiliency factors related to individual strengths and supports (e.g., problem-solving skills and close friends) and factors related to the youth’s context (e.g., effective schools and religious or spiritual beliefs) may also assist in protecting individuals with SB against the risks they face as they manage the multisytemic impact of their condition across the course of development.⁶⁶

The current findings must be interpreted in the context of several limitations. First, we utilized a clinically referred sample of convenience. Second, we did not include a control group of healthy children or children with another chronic health condition, and comparisons were thus made statistically with previously published results. This means that some of the obtained differences in our results could be due to differences in the demographic characteristics between our sample and the other samples, rather than an effect of SB per se. Third, the striking differences we found in psychosocial adjustment across measures could be due to response biases. Questions regarding mental health challenges can be viewed as a sensitive topic for many, and it is possible that the multidisciplinary clinic setting in which questionnaire results were collected did not offer the level of privacy and comfort needed for authentic responding. In addition, having a parent or guardian present while the child or adolescent was completing the measure may have evoked socially desirable responding to avoid the possibility of upsetting the parent.

Despite these limitations, the current study makes a valuable contribution to the growing body of research investigating parent, child, and family adjustment in youth with SB. The results of this study highlight the importance of careful measure selection when screening for adjustment challenges in children and adolescents with chronic health conditions in clinical care settings. Utilizing a screening measure that captures a wider range of social, emotional, and educational adjustment (e.g., HRQOL measure) may be important to ensure adequate assessment of psychosocial concerns and early intervention, if indicated. Future research should explore optimal measures for assessing HRQOL among youth with physical disabilities and the population with SB specifically. For example, the PedsQL has several different disease-specific modules (e.g., asthma, diabetes, cancer) but does not currently have a SB-specific module. Additional studies that include measures designed for children with physical disabilities are needed to corroborate the current results. In addition, future research should explore the use of resilience screening tools in clinical care settings to assist with identifying specific promotive and protective factors that help to buffer against physical, social, emotional, and academic challenges endorsed by this population. Further research is also needed to better understand families’ experiences in managing children in this population who have a shunt, as findings suggest this health variable may represent a target for family intervention. Lastly, it will be important to further explore how multidisciplinary clinics impact patient and family adjustment and well-being within the SB population. The comprehensive services offered to families in the sample may explain aspects of their resilience and is worth further investigation to encourage dissemination of similar treatment models if shown to be effective.

Funding Statement

Financial Support Internal funds were provided from the University of Colorado School of Medicine, Department of Rehabilitation Medicine to assist with research assistant support for data entry and data cleaning and for travel/conference attendance support.