Urologic self-management through intermittent self-catheterization among individuals with spina bifida: A journey to self-efficacy and autonomy

In the United States, spina bifida (SB), with a birth prevalence of approximately 30 per 100,000, is the most common non-chromosomal birth defect resulting in a complex genitourinary anomaly [1]. Given recent medical advances, individuals seeking care across our nation for SB related conditions are growing older and are increasingly likely to be adults [2]. Many of these adults have a difficult time reaching full participation in society. Although the reasons for this are multifactorial, it is known that urinary incontinence is associated with a lower health-related quality of life and potentially hinders full participation [3]. Hence, it is clear that preventive steps must be taken to assure that the growing number of individuals living with SB enjoy the best quality of life possible and transition well into an independent adulthood. Early instruction, therefore, must foster urologic self-management skills.

Clean intermittent catheterization (CIC) has been shown to be of great benefit in the care of SB-related neurogenic bladder and leads to improved long-term outcomes [4, 5]. Along with improving health-related quality of life, it reduces the frequency of urinary tract infections and preserves renal function [6, 7]. Thus, there has been growing interest in promoting timely intermittent self-catheterization (ISC) among patients with neurogenic bladder and on methods to predict patients’ ability to practice and master required skills [8, 9]. From improving self-care and independence to subsequent intimacy, this interest in ISC is not unwarranted, as there is a growing body of evidence that ISC has the potential to promote a healthy sense of autonomy and wellbeing [10, 11, 12].

In spite of the promising benefits to early implementation of ISC, to date little is known about when children can be expected to be involved in ISC. A few early studies of small cohorts indicated that self-catheterization can be achieved by five years of age in patients with myelodysplasia [13, 14]. However, in recent decades, there has been a lack of educational initiatives targeting promotion of self-catheterization at young ages. Moreover, there seems to be a hesitancy in training young children, who may be perceived to have fine-motor and/or cognitive impairments, to be shunted, or to lack an accessible channel, to perform ISC as compared to an earlier era [13]. Therefore, the purpose of this study was to describe the age of independence in ISC in a diverse patient population and to identify factors associated with the use of intermittent self-catheterization in children with spina bifida in an effort to promote early self-management.

The study participants were recruited from the Texas Children’s Hospital/Baylor College of Medicine multi-disciplinary pediatric Spina Bifida Clinic. Beginning in July 2015 through January 2017, 200 patients who were ⩾ 3 years of age were enrolled. Only patients that utilized catheterization for bladder management and had a diagnosis of myelomeningocele or lipomyelomeningocele were included. At this institution, patients were not routinely started on CIC at birth; the decision to initiate CIC was made in collaboration with the Urology service based on medical need (clinical or radiologic indicators of a hostile bladder) or patient desire for continence. A standard SB Clinic Questionnaire form was completed by the patient and/or caregiver upon arrival to the multi-disciplinary SB clinic, which provided data regarding independence in self-catheterization, dressing, bathing, and skin checks (Fig. 1). This information was then discussed by a Developmental Pediatrician with the patient and caregiver to ascertain the age at which ISC was performed mostly by the patient. Independence in dressing, bathing, and skin checks was also elucidated during this appointment to specify that independence meant that no caregiver was needed to complete these activities. The functional level of the spinal lesion was determined by direct neurological examination at each clinic visit. Cognitive ability was assessed based on available results of neuropsychological testing, through review of school-based individualized educational program (IEP) classifications, or from clinical neurodevelopmental assessments performed by one of the SB clinic Developmental Pediatricians. Cognitive ability and independence in dressing, bathing, and skin checks were only assessed on patients ⩾ 5 years of age at the time of the visit. Fine-motor ability was evaluated using the Beery-Buktenica Developmental Test of Visual-Motor Integration-Sixth Edition (Beery VMI) performed as part of a neuropsychology evaluation or a neurodevelopmental assessment performed by one of the Developmental Pediatricians. Patients who learned ISC prior to the creation of a surgically created channel (e.g. Mitrofanoff, Monti) were classified in the urethral ISC group for analysis of age of independence. Written informed consent were obtained from all participants and/or caregivers. The study was approved by the Institutional Review Board of Baylor College of Medicine.

Of the 200 individuals in the study, 92.5% had a diagnosis of myelomeningocele and 7.5% had a diagnosis of lipomyelomeningocele. The age range at the time of the study visit was 3 to 19 years. One hundred and eleven patients (55.5%) reported that they performed ISC for bladder management.

4. Discussion

These findings suggest that a higher level of lesion and female gender are associated with a lower rate of ISC in children/adolescents with SB. In the absence of a surgically created channel, it would be understood that the ease of access to catheterizing the urethra would explain the observed difference in gender. Moreover, though few studies have looked at the relationship between level of lesion or shunted hydrocephalus and ISC [15], this study lends credence to the notion that patients with lower level lesions are more likely to utilize ISC than patients with thoracic lesions. However, these results do not support the assertion that patients with shunted hydrocephalus are less likely to be able to perform ISC than those without a shunt [16], as no significant statistical difference in the rate of shunted hydrocephalus was found between patients who did and did not perform ISC.

Among those who catheterized via a surgically created channel, female gender and Non-Hispanic ethnicity are associated with increased self-management. This finding is consistent with Faleiros et al. who reported that among a cohort of 92 Brazilian and German patients, cultural factors influenced the performance of ISC [16, 17]. In this US cohort, Hispanic ethnicity was associated with a lower incidence of surgically created channel use, which may reflect a cultural preference for a non-invasive approach or a disparity in access to surgical care. As Hispanics are the greatest population in need of myelomeningocele surgical repair in the US and concurrently have a higher urinary incontinence rate and lower satisfaction with bladder management [18, 19], these findings are relevant to the management of this growing population within our nation. Overall, the average age of ISC emerges as a target for culturally-appropriate educational interventions to stimulate earlier self-management. To our knowledge, this is the largest US cohort of children in whom factors associated with ISC have ever been studied.

Although methods of teaching ISC have been described [8, 13, 20, 21], no large studies have examined the appropriate age of commencing such education. Campbell et al. reported that 45 Canadian children learned to perform ISC at a mean age of 8 years (range 4.7–15.3 years) [22]. In this US cohort, the mean age to ISC was 9.45 years (SD = 2.97 years; range 2 to 17 years) and in those individuals with known intellectual disability, it was 12.2 years (SD = 2.96 years). This illustrates that while individuals with intellectual disability may lag in initiating ISC, their cognitive ability alone should not preclude them from being taught these skills. Though controversy exists concerning the indications for commencement of clean intermittent catheterization and treatment with anticholinergic medications [23], once underway a case can be made for a proactive approach to ISC. Given the improved longevity of individuals with SB, transitional care issues (such as health-related independence) are becoming progressively more vital. Thus, given the wide age range for initiating ISC, it can be advocated that it is never too early or late to initiate participation in urological self-care skills, and that ISC should commence at the earliest possible age [24].

Determinants of successful ISC teaching programs for children have long been of interest [25]. However, in an era of limited access to comprehensive evaluations and of diminishing reimbursement for such evaluations, there is a need to be able to easily assess a child’s readiness to initiate ISC. In this study, while both compared groups had expected below average VMI standard scores, there was no difference between those who used ISC and those who did not, suggesting that fine motor abilities are not the major determinant of urologic self-management. Furthermore, cognitive impairment did not preclude ISC, since in the ISC group 15% had mild to moderate intellectual disability. Most importantly, the findings demonstrate that existent self-efficacy regarding activities of daily living (i.e. dressing, bathing, skin care) are more likely associated with ISC.

Peny-Dahlstrand et al. documented that many children living with SB had difficulties executing activities of daily living in an effortless, efficient and independent way, which at the time was understood to be modulated by both motor and process abilities [26]. Subsequently, the neurostructural association between fine motor dexterity and executive function has been demonstrated [27]. Thus, the assertion that to reach independence in daily life, children with SB need guidance to overcome process deficits, to acquire not only ‘how to do things’ but also ‘how to get things done’ is becoming evident [26]. Consequently, as with other activities of daily living, increasing the use of ISC will necessitate intentional parental support and encouragement early in life for children to attain the set of skills required for later autonomy.

The role of an ‘independent spirit’ within the training and rehabilitation process has long been believed to be important [28]. In this study, a sense of independence was seemingly illustrated in the relationship observed among ISC and independence in dressing, bathing and skin care. Although the impact of parental behaviors on ISC was not addressed in this study, the potential role it may play in the initiation and adherence to ISC cannot be ignored [29]. This observation would be in direct agreement with current literature linking parenting behaviors with medical adherence and autonomy in the SB population, as it has been suggested that higher levels of adaptive parent behaviors (i.e., enforcing developmentally appropriate limits, termed behavioral control) buffers against the negative effect of expected lower levels of executive functioning in children with SB [30, 31]. Concurrently, it has been suggested that the level of maladaptive parent behaviors (i.e., intrusive actions that inhibit self-sufficiency development, termed psychological control) is inversely related to levels of medical autonomy and adherence [30, 31, 32]. Thus, it has been described that parental behavioral control and psychological control may moderate the relationship between executive functions and medical adherence [31]. As a whole, these studies intrinsically conjoin the role parents have in supporting the early initiation of and constancy with self-management practices.

Lastly, similar to the role of parents, the responsibility of medical providers cannot be ignored. Historically, ISC has been an area of nursing expertise, and physicians have often readily delegated this task. However, in this new era, an inter-professional approach within a multidisciplinary clinic is an ideal environment to support early ISC training through multiple providers. Thus at an early age, providers can discuss parental attitudes and expectations regarding self-catheterization in order to address barriers to adopting this self-management skill.

5. Conclusions

These findings suggest that a higher level of lesion and female gender are associated with a lower rate of ISC in children/adolescents with SB. In this study, the mean age to ISC was 9.45 years (SD = 2.97 years), and thus, the average age of ISC emerges as a target for culturally-appropriate educational interventions to encourage early independence. While the mastery of activities of daily living was not conclusively proven to be a predictor for readiness to participate in ISC, our study does set the stage for future research on factors that may foster an ‘independent spirit’ early in childhood. Encouraging self-care is an opportunity to provide early guidance in the journey to self-management and transition to adulthood.