Spina bifida is a neural tube defect, a congenital abnormality causing disability, whereby the spinal cord and vertebrae do not form completely and the neural tube fails to develop normally. The worldwide incidence of spina bifida varies between 0.17 and 6.39 per 1000 live births (Bowman, Boshnjaku & Mclone 2009; Kinasha & Manji 2002; Msamati, Igbigbi & Chisi 2000; Shaer et al. 2007). In Uganda, incidence data are not available. Warf et al. estimated a birth incidence of 1 in 1000, translating into 1400 children born with spina bifida in the country annually (Warf et al. 2011).

Most children with spina bifida have some degree of paralysis, which affects mobility as well as bowel and bladder control (Northrup & Volcik 2000; Verpoorten & Buyse 2008). This affects participation in daily activities (Danielsson et al. 2008; Jansen et al. 2009). Sixty-six per cent of the children with spina bifida develop hydrocephalus (Warf & Campbell 2008).

Surgery and rehabilitative care are expensive and inaccessible for many born with the disability in Africa. Families need to find resources for children with disabilities whilst already constrained. In Uganda, the initial surgery (closure of the spine) at the time of this study was only available in Mulago National Referral Hospital in Kampala, and CURE Children’s Hospital in Mbale, eastern Uganda. Recently, Mbarara Regional Referral Hospital started offering the same. In north, west and central Uganda, three rehabilitation centres offer specific occupational therapy, physiotherapy, continence management and social support services for children with spina bifida and their families in Uganda (Supplement Figure 1: map). Policies such as the United Nations Convention on the Rights of Persons with Disabilities and Disability Act, amongst others, are in place and efforts are made by the Ugandan government and not-for-profit organisations and hospitals to provide basic services for children with spina bifida and other disabilities (Mertens & Bannink 2012; Oyaro 2014). Despite these efforts, poverty continues to affect access to services for the majority of the population and more so for the families of children with disabilities (Bannink et al. 2015; Lwanga-Ntale 2003; Miles 2002).

Parental functioning is of great importance for children with severe disabilities who depend for a large part of their functioning on the parent or caregiver. Research on family functioning and psychosocial adjustment of families of children with spina bifida in high-income countries support a resilience–disruption view of family functioning, whereby the presence of a child with spina bifida disrupts normative family functioning at first, but after a period of time families adapt and exhibit considerable resilience (Holmbeck et al. 1997; Vermaes et al. 2007). The level of impairment has not been related to the general level of family functioning in Europe and the United States (Ulus et al. 2012; Wiegner & Donders 2000). Nevertheless, spina bifida has been found to have negative medium–large effects on parents’ psychological adjustment and functioning (Holmbeck & Devine 2010; Vermaes et al. 2005, 2008).

In the review by Vermaes et al. (2005), child, parent, family and environmental factors were found to be associated with variations in parents’ psychological adjustment. Mothers are often at higher risk for parenting stress than fathers because of role differences in care and work (Vermaes et al. 2008). Children with hydrocephalus (literally ‘excess water in the brain’) are often in need of neurosurgical treatment, which may involve insertion of a shunt or creation of a bypass within the brain to allow the cerebrospinal fluid drain. Shunts are prone to infections, which can be life threatening. For children with both spina bifida and hydrocephalus, such shunt dependency has been associated with higher levels of parental anxiety and depression (Malm-Buatsi et al. 2015).

Cultural differences have been described in a study of Hispanic parents in the United States, in which mothers of children with spina bifida were found to be at risk for lower feelings of satisfaction and competence as parents compared to non-Hispanic parents (Devine et al. 2012). In Malaysia, mothers of children with spina bifida had significantly higher stress scores compared to mothers with children without a disability (Ong et al. 2011). In the same country, clean intermittent catheterisation was the only medical factor associated with such stress. This is a technique used to empty urine from the bladder using a catheter. Most children with myelomeningocele, the severe type of spina bifida in children involved in this study, are incontinent of urine and benefit from practicing intermittent bladder catheterisation as this helps them to participate in daily activities without losing urine (IFSBH 2014). Parental stress in Malaysia was also mediated by single parenthood, caregiver status and the child’s adaptive skills (Kanaheswari et al. 2011).

African studies on parental stress in parents of children with spina bifida are limited. In South Africa, Greeff and Nolting showed that families of children with developmental disabilities adapt better when they are more accepting of the situation, have more investment in the family unit and have positive patterns of communication and attitudes towards challenges (Greeff & Nolting 2013). In Nigeria and Tunisia, parents of children with neurodevelopmental disabilities and learning disabilities had higher rates of depression and anxiety compared to parents of children without a disability (Abasiubong, Obembe & Ekpo 2006; Ben Thabet et al. 2013). In Malawi, parents of children with neurodisability following brain injury experienced barriers to care and support (Paget et al. 2016). Similar findings were seen in South Africa in families of children with mental health disabilities (Coomer 2013). In Kenya, mothers of children with spina bifida struggled with the financial implications of the child’s disability; most of them received some support from other parents and religious communities (Vant Veer et al. 2008).

In the study, we aim to explore perceived stress and support of parents of children with spina bifida living in Uganda and examine the factors that influence it. By understanding the stress factors, we hope to inform health and community-based rehabilitation services for children and parents with spina bifida in the country. We believe parental stress and support is highly dependent on the cultural and socio-economic context where parents live. Contextual factors such as the differences in rural and urban setting, and the 22-year conflict between the Lord’s Resistance Army and the Government of Uganda, which displaced over 2 million people in the north of the country (Muyinda & Whyte 2011), will be taken into account.

Ethical approval and research clearance for the study were obtained from Ghent University, Belgium, the Uganda Virus Research Institute and the Uganda National Council for Science and Technology. Informed consent was obtained from all parents.

Selection of participants was performed in Mbarara, Kampala and Mbale, where CURE holds bi-monthly clinics. CURE hospital and the partnering rehabilitation centres in Kampala and Mbarara were requested to list all children registered in their follow-up programmes and inform them during home visits and through telephone calls to attend the clinic. In Gulu and Lira where no follow-up system or registry of the children was in place at the time, radio announcements were aired to inform parents about the upcoming review clinics in the area, and specifically invited parents with children with spina bifida between the age of 4 and 14 to attend.

Data collection

Explorative mixed-study methods were employed to explore factors affecting parental stress and support in the Ugandan setting (Creswell & Clark 2007) (Singh 2007). Both quantitative and qualitative data were collected concurrently from 134 parents between June 2011 and December 2014.

Quantitative instruments were used to explore daily functioning and stress levels. A selection of 10 items of The Vineland Adaptive Behaviour Scales (VABS) Daily Living Skills subscale relevant to the Ugandan setting were used. Items included measures of daily functioning tasks such as removing a jumper, drinking from a cup, washing ones’ face, fetching water and dressing independently. Items were scored 2 (behaviour is usually or habitually performed), 1 (sometimes or partly performed) or 0 (never performed) (Sparrow et al. 1984).

The Parental Stress Index Short Form (PSI/SF) consists of 36 items scored on a 5-point Likert scale. The items are divided over three subscales: parental distress (PD), parent–child dysfunctional interaction (P-CDI) and difficult child (DC). A total stress score is computed from the three subscales and indicates the overall level of parenting stress in the areas of personal PD, stresses derived from the parent’s interaction with the child and stresses that result from the child’s behavioural characteristics. Parents who obtain a total stress score above 90 are experiencing clinically significant levels of stress (Abidin 1995).

The presence of a (house) helper or other adults at home involved in the care of the child alongside the primary caregiver was registered as a measure of perceived support. Membership of a parent support group was documented to understand wider support networks.

Qualitative methods were used to explore the day-to-day reality of parents of children with spina bifida in the Ugandan setting. Semi-structured interviews were held with all 134 parents. In addition, focus group discussions were held with four parents’ support groups, in Gulu, Kampala, Mbale and Mbarara, respectively. Questions for the interviews and focus group discussions were formulated to develop a more in-depth understanding of perceived parental stress, coping strategies and the role of support from others. Specific focus group discussions with parent support groups were held to further understand their activities and possible contributions in reducing parental stress and increasing perceived support. In total 5 of the 134 parents took part in the focus group discussions, the other members of the focus groups (26) were not interviewed individually.

The interviews with the parents were held in the local language of the area, and a translator was hired and trained for each area to assist in conducting the interviews and focus group discussions. Some of the interviews were conducted in English, if parents were fluent and requested for this.

The semi-structured interviews and focus group discussions were transcribed, translated, coded and analysed using thematic analysis in NVivo version 10. Basic demographic data, details on the child’s impairment, social support and scores for the VABS Daily Functioning Subscale and PSI/SF were entered and analysed using SPSS16. The sub-total scores for each subscale was calculated and compared with > 90% percentile scores from the PSI manual, which indicates high levels of stress. To understand relationships between demographic and impairment variables and parental stress levels, Spearman’s rho correlations were computed. To compare regional differences, a multivariate analysis of variance (MANOVA) between PSI scores between the various regions was carried out. Linear regression analysis was conducted to understand the factors that mostly contributed to parental stress.

Table 1 describes the demographics of the study population. Parents’ ages ranged from 24 to 46 years with an average age of 32.9 (SD 5.2) years. The majority of the parental respondents were the mother to the child, followed by fathers and grandmothers. Over 75% was married and Christian. Almost half of them were farmers.

The mean age of the 80 (59.7%) male and 54 (40.3%) female children with spina bifida (myelomeningocele type) was 6.1 (SD = 2.0) years, ranging from 4.0 to 14.0 years of age. Only 76 (57.3%) of them were going to school: 49 (37.4%) were in nursery school, 22 (16.8%) in primary school and 4 (3.1%) in secondary school. The household size ranged from 2 to 13 with an average of 6.5 persons per household; an average 4.3 children (SD 2.2) and 2.3 adults (SD 1.0) per household. The average monthly household income is 82 euros (range, 12–604 euros). The majority of the children (128, 95.5%) had undergone surgery to close their spine (myelomeningocele closure) earlier in life. Of the 61 children who had both spina bifida and hydrocephalus, 22 (35.9%) had undergone endoscopic third ventriculostomy whilst 1 (18.6%) had ventriculo-peritoneal shunts placed. Only 2 (3.6%) of the children of parents in the study never had surgery. Most parents took their children (118, 90.8%) for rehabilitation services such as physiotherapy and occupational therapy.

The parent stress index questionnaire was administered to all parents. Scores were normally distributed for all items including the total subscales. We excluded three cases from the analysis as scores showed defensive responding (score < 10). Table 2 shows the subscales scores. More than half of the parents (52.7%) scored above the 90% percentile on total stress. Higher scores were seen on the P-CDI domain with 48.1% (63) scoring above the 90% percentile, followed by the DC Domain with 32.8% (43) scoring above the 90% percentile and the PD Score in which 14.5% (19) of the parents scored above the 90% percentile.

Spearman’s rho correlations were calculated for demographic variables, impairment-related variables, for example incontinence and mobility, daily functioning skills and perceived support from another adult and parent support groups. Table 3 shows the variables that had significant Spearman’s rho correlations. Variables with significant correlations with PSI outcomes were the child’s ability to walk, continence and use of clean intermittent catheterisation and bowel management, receiving rehabilitative care, household income, location (region where the child and family lives) and having support from another parent in taking care of the child. Household income correlated positively with school going, region, parental education, the use of assistive devices, receiving rehabilitation services, using catheterisation and having support from another adult.

Based on the significant correlations found, linear regression analysis was performed to investigate factors that were determinants for high scores in the PD, DC and P-CDI subdomains of the PSI/SF.

All variables with significant correlations were entered in a stepwise regression model. The best predictive models are displayed in Table 4. Results are expressed as beta coefficients, R² values and F values in Table 4. The ability to walk, having support of another adult and the location where the child and family are residing were the significant contributing predictors.

MANOVA results showed that parents in northern Uganda had significantly higher > 90 percentile scores on the PSI subscales compared to parents in other regions: PD F = 5.467*, P-CDI F = 8.815** and DC F = 10.489**. No significant differences were found between the other regions.

The inability to walk was the largest contributor to parental stress and perceived parental difficulty in managing and caring for the child. Parents of children with both spina bifida and hydrocephalus had higher scores on dysfunctional interaction between parent and child compared to those with only spina bifida only.

The majority of the children with spina bifida whose parents were interviewed were able to sit without assistance and could speak. Half of them were able to walk without assistive devices. Of the ones who were in need of assistive devices, a third used crutches, another third a wheelchair, whilst the others had no access to assistive devices and crawled. In our study population, 88.6% of the children were incontinent and 81.1% of them used clean intermittent catheterisation to manage the incontinence. Table 5 shows the various percentages for the impairment-related variables.

In the interviews, parents indicated mobility was a major challenge and stressor. Many parents narrated how they carried their children on their back to the land they cultivate (most of them are subsistence farmers), to public transport and sometimes to school when the children were young. Some still continue to do so as assistive devices are not always available or applicable for use in their setting.

Although incontinence did not explain parental stress for a large part on the PSI outcomes, apart from practising bowel management on the PD subscale, in the focus group discussions and interviews the majority of parents said incontinence was the most complex factor in managing their child’s health, more than the (partial) paralysis. Whilst the majority did practice catheterisation, parents complained practicing catheterisation and bowel management outside home and the rehabilitation posed a big challenge. Managing their children’s catheterisation meant that parents had to be close by and available to help the child practice catheterisation, for example at school every 6 hours. Accidents or ill-managed catheterisation also caused stress as the child would be wet and smelly, which would result in others commenting or pitying the parents.

Almost half of the parents (63, 47.7%) mentioned they have support of another adult in the household to care for their child with spina bifida. Having another adult to support in providing care significantly contributed to having less dysfunctional parent–child interaction on the PSI outcomes. This other adult would be a spouse, relative or house help. In the focus group discussion and interviews, parents who did not have support from another adult explained they found it very stressful to be constantly responsible for their child and often felt isolated, as they could not attend family and community functions because of their care obligations. Whilst some could leave their other children with neighbours or friends, they said it was hard to find someone who would be ready to take care of their child with spina bifida. Parents in the north and east had less support from other adults compared to those in the central and western regions.

In total, 55 (41%) parents were members of a parent support group. No interaction was found between PSI outcomes and membership of a parent support group. Table 6 provides details of the reasons of (not) participating in a parent support group. From the interview, those who were members of a parent support group indicated it mostly helped them to learn more about taking care of their child (50.9%), followed by feeling encouraged by other parents (14.5%), enjoying sharing experiences and learning from each other (14.5%) and income-generating opportunities (12.7%).

The majority of parents who were not members of a parent support group said they were not aware parent support groups existed (55.1%), and some mentioned they had heard about parent support groups but lived too far away from the rehabilitation centre to attend meetings regularly.

To explore the support given by the PSG further, focus group discussions were held with 26 mothers and 6 fathers active in parents support groups in Gulu (9: 8 mothers, 1 father), Kampala (6: 5 mothers, 1 father), Mbale (8: 6 mothers, 2 fathers) and Mbarara (9: 7 mothers, 2 fathers).

Parents explained the groups started with help of the rehabilitation centres in three sites; in Gulu, the group started based on a community-based rehabilitation initiative which was started up during the conflict in the area. The main objectives of the groups were to motivate and support each other in caring for their children, provide information and training on care and rehabilitation and create awareness in their communities about the disability. Parents said they themselves enjoyed participating in the PSG as they would learn more about taking care of their child and were able to advise and encourage others. The majority indicated that the feeling of ‘not being alone’ in this situation and being able to share specific challenges with others with similar experiences helped them to continue caring for and loving their child more.

Some parents mentioned the group had given them new friends and support networks. For example, in case of advocating for children, parents in Kampala and Mbarara gave examples on how other parents would join them in visiting schools trying to get a space for their child. Many schools would refuse children with physical impairments, but if going as a group or pair, head teachers were said to take their call more seriously and were more likely to give them a space. Similar advocacy took place on inclusion and reducing stigma in communities, where parents would move around with their children explaining the impairment to community members and the need for their children and families to be included. In Mbale, most members felt the group gave them spiritual support too.

In northern Uganda, most parents felt the group gave them an opportunity to start income-generating activities together. They had started a rotating loan scheme, which was helping members to set up small businesses. They especially felt stigma was high in their area, and support for them was difficult as some people believed the impairment was contagious. Similar activities were mentioned in Mbarara and Kampala. Stigma was mentioned in these areas too, but less directly connected to physical contact as in the northern region.