Abstract
Objectives:
To compare differences in bowel-specific quality of life (QOL), overall qQOL, and neurogenic bowel dysfunction (NBD) severity by bowel management program in patients with spina bifida.
Methods:
We performed a retrospective cross-sectional study of patients ≥12 years old at our multidisciplinary spina bifida center who completed both a modified Peristeen NBD questionnaire (assessing bowel symptom severity and bowel-specific QOL) and the Patient-Reported Outcomes Measurement Information System Pediatric Global Health questionnaire (assessing overall QOL). Nested, multivariable models were fit for associations between outcomes and bowel management program (enemas, conservative management, and none).
Results:
A total of 173 patients, 56.1% female and 64.6% with myelomeningocele, were included in our analysis. Median age was 18.2 years old. Patients reported using enemas (n=42), conservative management (n=63), and no bowel program (n=68). When adjusting for covariates, there was no significant association between bowel-specific QOL nor overall QOL across bowel management programs. However, the use of conservative management compared to enemas was associated with worse bowel symptoms severity (adjusted beta=2.58, 95%CI=[0.09,5.06]). Additionally, greater bowel symptom severity was significantly associated with lower overall QOL (adjusted beta=−0.33, 95%CI=[−0.57,−0.10]).
Conclusions:
NBD symptom severity in spina bifida is more strongly associated with QOL than the individual bowel program being utilized. Our findings suggest that different degrees of NBD require different invasiveness of bowel programs, but it is the outcome of the bowel management program and not the specific program itself that is most associated with QOL.
Keywords: spinal dysraphism, neurogenic bowel, quality of life, enema, fecal incontinence
Introduction:
Spina bifida (SB) has an estimated prevalence in North America of 36.8 per 100,000 live births.1 The majority of patients with SB endorse symptoms of neurogenic bowel dysfunction (NBD), predominantly constipation and fecal incontinence (FI).2 More than half of adult patients with SB reporting still using some form of bowel program.2–4 Management of NBD typically follows a step-wise approach of invasiveness (i.e., oral medication to enemas), with varied efficacy by management type and high rates of program discontinuation seen across management strategies.5,6
Patients with SB and FI have reported significantly lower quality of life (QOL) compared to those with “normal” bowel function.3,7–9 Mitigation of factors that negatively affect QOL and health-related QOL is a focus of the Quality of Life Spina Bifida Guidelines,10 with targeted bowel management for those with bowel dysfunction identified as important across age groups. Prior studies have evaluated the association of individual bowel programs and QOL, either through non-comparative studies or focusing on how satisfaction with bowel program and incontinence impact QOL.11 Few studies have directly compared bowel management program type and differences in QOL.
In this study, we aim to compare differences in bowel-specific QOL, overall QOL, and NBD severity by bowel management programs in adolescents and young adults with SB. We hypothesize that enema programs confer better QOL compared to other bowel programs.
Methods:
Study Design & Patient Population
We performed a retrospective cross-sectional study at a tertiary referral multidisciplinary SB center from May 2019 to March 2020 (IRB 2020–3804). Patients ≥12 years old with SB presenting to clinic visits were administered patient-reported outcome measures (PROs) across various health-related domains as part of routine clinical care. Measures are completed independently, with the assistance of a guardian, or by the guardian as a proxy based on cognitive ability of the patient. Questionnaires are available in English and Spanish. Completed PROs/questionnaires and clinical information are collected and recorded in a secure database. Patients with a history of an anorectal malformation, history of fecal diversion, and those with incomplete NBD or QOL questionnaires were excluded from analysis.
Exposure and Outcome Measures
The primary exposure, collected by chart review, was bowel management program, categorized as: enemas, conservative management, and no bowel regimen. The groupings were defined based on perceived level of intervention, with enemas generally accepted as more invasive than medications or other measures.12 The “enema” group was composed of those patients using antegrade continence enemas, cecostomy tubes, chait tubes, and any retrograde enema (e.g., transanal irrigation, cone/balloon enema). The “conservative” management group includes use of oral medications, suppositories, digital stimulation, and manual disimpaction. The “no bowel regimen” group included timed evacuation or no program at all.
The primary outcome was bowel-specific QOL, as measured by the satisfaction score on a Peristeen Neurogenic Bowel Dysfunction (PNBD) questionnaire that was slightly modified to include non-clinical language.13 PNBD was validated in Danish patients with spinal cord injury (SCI), ages 8–88 years old.14 The NBD satisfaction score is the final question in the PNBD questionnaire but is not factored into the total score. Scoring is based on an 11-point numeric rating scale of bowel satisfaction ranging from 0–10, with 0 corresponding to total dissatisfaction and 10 as perfect satisfaction. This question is a surrogate of bowel-specific QOL.
Secondary outcomes included overall QOL and NBD severity. Overall QOL was assessed with the Patient-Reported Outcomes Measurement Information System (PROMIS) Pediatric Global Health (PGH-7) questionnaire.15 This is a non-disease specific 7-item measure with Likert scale response options. A higher score indicates a better QOL; there are no pre-defined cutoffs or groupings based on score. For research purposes, the PGH-7 summative score is normalized and converted to a T-score so the sample population may be compared to the general population. A higher T-score represents more of the concept being measured.
NBD severity was based on the PNBD questionnaire total score (10 total items). Scores range from 0–47, with higher scores indicating worse NBD severity. Severity is specified as very minor (score 0–6), minor (score 7–9), moderate (score 10–13), or severe (score 14+).
Covariates
Demographic data and clinical characteristics were obtained and recorded at time of questionnaire completion by research staff. Certain demographic and clinical characteristics were included in the analysis based on prior published associations with outcomes of interest or factors considered to be associated with NBD or QOL outcomes.8,16 Demographic variables included age, sex, race, ethnicity, highest level of education, and insurance type. Clinical characteristics included type of SB (myelomeningocele, non-myelomeningocele (fatty/thickened filum, low lying cord, lipomeningocele, terminal myocystocele, split cord malformation)), ambulatory status (ambulatory, non-ambulatory), lesion level (thoracic, lumbar, sacral), health literacy (HL) based on the Brief Health Literacy Screening Tool17 (adequate HL, marginal HL, inadequate HL), primary bladder management program (volitional void, clean intermittent catheterization, no management), presence of a bladder catheterizable channel (yes, no), bladder incontinence (yes, no/unknown), FI (yes, no), and frequency of FI (daily, 1–6 times per week, 3–4 times per month, a few times a year or less).
Statistical Analysis
Descriptive statistics by demographic and clinical characteristics were performed for proportions in the overall population and by bowel management program. Measures of association among the bowel management groups by characteristics were calculated with significance reported from ANOVA, chi-square, and Fisher’s exact tests.
Multivariable, nested linear regression models were constructed to fit the associations between bowel-specific QOL (primary outcome) and bowel management program (enema versus conservative management versus none). Models 1a-c were sequentially adjusted adding in covariates with statistically significant differences on univariable analysis or clinically meaningful associations among the bowel management groups.
For the secondary outcome of overall QOL (PGH-7 T-score), multivariable, nested linear regression models were constructed to fit the associations with bowel management program (Models 2a-c). For the secondary outcome of NBD severity, multivariable, nested linear regression models were constructed to fit the associations with bowel management program (Models 3a-b). Analysis was performed treating NBD severity as a continuous variable based on total NBD severity score given prior use as such.18,19
A post-hoc secondary analysis was performed to assess the association between overall QOL (PGH-7 T-score) and NBD severity, treating NBD score as a continuous variable. Multivariable, nested linear regression models were constructed to fit the associations with NBD severity (Models 4a-b).
Based on clinical suspicion, additional post-hoc secondary analyses were performed for subgroups of myelomeningocele versus non-myelomeningocele diagnosis and ambulatory versus non-ambulatory status to fit associations with aforementioned outcomes of bowel-specific QOL, overall QOL, and NBD severity by bowel management program using multivariable, nested linear regression models.
Statistical significance was set at an alpha of 0.05; no adjustment for multiple outcomes was performed. All analysis was performed using Stata version 17.0.20 Details of each of the Models and the covariates adjusted for are outlined in the footnotes for Tables 3–4.
Table 3:
Multivariable, nested linear regression models fit to assess the association between bowel management program and study outcomes, N=173.
| Association Between Bowel Management Program and Bowel-Specific QOL | |||
|---|---|---|---|
|
| |||
| Model [1a] | Model [1b] | Model [1c] | |
|
| |||
| Bowel management | (95% CI) | (95% CI) | (95% CI) |
|
| |||
| Enemas | reference | reference | reference |
| Conservative | −1.07 (−2.19, 0.04) | −0.78 (−2.08, 0.52) | −0.53 (−1.82 0.77) |
| None | −0.42 (−1.52, 0.68) | −0.48 (−1.81, 0.86) | −0.58 (−1.90, 0.74) |
|
| |||
| Association Between Bowel Management Program and Overall QOL | |||
|
| |||
| Model [2a] | Model [2b] | Model [2c] | |
|
| |||
| Bowel management | (95% CI) | (95% CI) | (95% CI) |
|
| |||
| Enemas | reference | reference | reference |
| Conservative | −1.50 (−4.95, 1.95) | −2.83 (−6.79, 1.13) | −2.05 (−5.99, 1.90) |
| None | 1.63 (−1.77, 5.03) | −0.66 (−4.74, 3.42) | −0.98 (−5.00, 3.05) |
|
| |||
| Association Between Bowel Management Program and NBD Severity | |||
|
| |||
| Model [3a] | Model [3b] | ||
|
| |||
| Bowel management | (95% CI) | (95% CI) | |
|
| |||
| Enemas | reference | reference | |
| Conservative | 0.81 (−1.61, 3.23) | 2.58* (0.09, 5.06) | |
| None | −4.10* (−6.49, −1.72) | −1.04 (−3.60, 1.52) | |
Model 1: Multivariable, nested linear regression models fit of the association between bowel management program and bowel-specific QOL (PNBD satisfaction score, where higher values denote better QOL).
Model [1a] is unadjusted and includes bowel management program only.
Model [1b] adds to Model [1a] and adjusts for type of spina bifida, lesion level, health literacy, bladder management program, and presence of a catheterizable bladder channel.
Model [1c] includes those adjusted for in Model [1b] and adjusts for NBD severity score.
Model 2: Multivariable, nested linear regression models of the association between bowel management program and overall QOL (PGH-7 T-score, where higher values denote better QOL).
Model [2a] is unadjusted and includes bowel management program only.
Model [2b] adds to Model [2a] and adjusts for type of spina bifida, lesion level, health literacy, bladder management program, and presence of a catheterizable bladder channel.
Model [2c] adds to Model [2b] and adjusts for NBD severity score.
Model 3: Multivariable, nested linear regression models of the association between bowel management program and NBD severity (PNBD total score, where higher values denote worse NBD symptom severity).
Model [3a] is unadjusted and includes bowel management program only.
Model [3b] adds to Model [3a] and adjusts for type of spina bifida, lesion level, health literacy, bladder management program, and presence of a catheterizable bladder channel.
Denotes statistical significance, p≤0.05
Table 4:
Secondary analysis using multivariable, nested linear regression models fit to assess the association between NBD severity and overall QOL (PGH-7 T-score).
Model 4: Multivariable, nested linear regression models of the association between NBD severity and overall QOL (PGH-7 T-score).
Model [4a] is unadjusted and includes NBD score as a continuous variable only.
Model [4b] adds to Model [4a] and adjusts for type of spina bifida, lesion level, health literacy, bladder management program, and presence of a catheterizable bladder channel.
Denotes statistical significance, p≤0.05
Results:
A total of 173/232 (74.5%) outpatients with SB who completed questionnaires met inclusion criteria for this study. Patients were excluded for incomplete questionnaires (39 incomplete PNBD questionnaires, 3 incomplete PGH-7), bowel diversion (3 colostomies), and competing diagnosis (14 anorectal malformations).
The mean age of included patients was 18.2 years old (range 12–31 years) and 56.1% identified as female. Most patients were ambulatory (80.9%) and diagnosed with myelomeningocele (64.7%) (Table 1). Patients reported using enemas (n=42, 24.3%), conservative management (n=63, 36.4%), and no bowel program (n=68, 39.3%). Of those utilizing enemas, 15 (35.7%) performed retrograde enemas and 27 (62.3%) performed antegrade enemas. In the conservative management group, 57 (90.5%) used oral therapies, 4 (6.4%) used suppositories, and 14 (22.2%) performed either digital stimulation or manual disimpaction. A few in the conservative management group utilized a multimodal conservative management approach. Univariable analysis by bowel management type is displayed in Table 1.
Table 1:
Demographic and Clinical Characteristics, study population and subgroups by bowel management program.
| Characteristics | Total population n = 173 | Enemas n = 42 | Conservative n = 63 | No Bowel Regimen n = 68 | P value |
|---|---|---|---|---|---|
|
| |||||
| Age in years, mean (std dev) | 18.2 (4.6) | 18.0 (4.8) | 18.0 (4.7) | 18.5 (4.5) | 0.79 |
|
| |||||
| Sex, n (%) | 0.22 | ||||
| Female | 97 (56.1) | 24 (57.1) | 40 (63.5) | 33 (48.5) | |
| Male | 76 (43.9) | 18 (42.9) | 23 (36.5) | 35 (51.5) | |
|
| |||||
| Race, n (%) | 0.24 | ||||
| Asian | 13 (7.5) | 3 (7.1) | 8 (12.7) | 2 (2.9) | |
| Black | 20 (11.6) | 2 (4.8) | 6 (9.5) | 12 (17.7) | |
| Multiracial | 3 (1.7) | 1 (2.4) | 1 (1.6) | 1 (1.5) | |
| Other | 13 (7.5) | 4 (9.5) | 3 (4.8) | 6 (8.8) | |
| White | 124 (71.7) | 32 (76.2) | 45 (71.4) | 47 (69.1) | |
|
| |||||
| Ethnicity, n (%) | 0.56 | ||||
| Hispanic/Latino | 50 (28.9) | 10 (23.8) | 21 (33.3) | 19 (27.9) | |
| Non-Hispanic/Latino | 123 (71.1) | 32 (76.2) | 42 (66.7) | 49 (72.1) | |
|
| |||||
| Insurance, n (%) | 0.35 | ||||
| Private | 103 (59.5) | 29 (69.1) | 36 (57.1) | 38 (55.9) | |
| Non-private | 70 (40.5) | 13 (31.0) | 27 (42.9) | 30 (44.1) | |
|
| |||||
| Type of spina bifida, n (%) | <0.01 | ||||
| Myelomeningocele | 112 (64.7) | 40 (95.2) | 36 (57.1) | 36 (52.9) | |
| Non-myelomeningocele | 61 (35.3) | 2 (4.8) | 27 (42.9) | 32 (47.1) | |
|
| |||||
| Highest level of education, n (%) | 0.73 | ||||
| Primary or secondary school | 120 (69.4) | 28 (66.7) | 46 (73.0) | 46 (67.7) | |
| Beyond secondary school | 53 (30.6) | 14 (33.3) | 17 (27.0) | 22 (32.4) | |
|
| |||||
| Who completed the questionnaires? n (%) | 0.08 | ||||
| Self-administered | 107 (61.9) | 23 (54.8) | 35 (55.6) | 49 (72.1) | |
| With assistance | 66 (38.2) | 19 (45.2) | 28 (44.4) | 19 (27.9) | |
|
| |||||
| Ambulatory status, n (%) | 0.46 | ||||
| Ambulatory | 140 (80.9) | 32 (76.2) | 50 (79.4) | 58 (85.3) | |
| Non-ambulatory | 33 (19.1) | 10 (23.8) | 13 (20.6) | 10 (14.7) | |
|
| |||||
| Lesion level, n (%) | <0.01 | ||||
| Thoracic | 33 (19.1) | 14 (33.3) | 11 (17.7) | 8 (11.8) | |
| Lumbar | 71 (41.0) | 21 (50.0) | 27 (43.6) | 23 (33.8) | |
| Sacral | 68 (39.3) | 7 (16.7) | 24 (38.7) | 37 (54.4) | |
|
| |||||
| Health Literacy, n (%) | 0.01 | ||||
| Adequate HL | 65 (38.0) | 14 (34.2) | 17 (27.0) | 34 (50.8) | |
| Marginal HL | 52 (30.4) | 9 (22.0) | 22 (34.9) | 21 (31.3) | |
| Inadequate HL | 54 (31.6) | 18 (43.9) | 24 (38.1) | 12 (17.9) | |
|
| |||||
| Primary bladder management program, n (%) | <0.01 | ||||
| Volitional void | 47 (27.2) | 1 (2.4) | 15 (23.8) | 31 (45.6) | |
| CIC | 120 (69.4) | 41 (97.6) | 46 (73.0) | 33 (48.5) | |
| No management | 6 (3.5) | 0 (0) | 2 (3.2) | 4 (5.9) | |
|
| |||||
| Does the patient have a catheterizable channel? n (%) | <0.01 | ||||
| Yes | 29 (16.8) | 20 (47.6) | 5 (7.9) | 4 (5.9) | |
| No | 144 (83.2) | 22 (52.4) | 58 (92.1) | 64 (94.1) | |
|
| |||||
| Any degree of bladder incontinence reported, n (%) | 0.33 | ||||
| Yes | 84 (48.6) | 24 (57.1) | 31 (49.2) | 29 (42.7) | |
| No or Unknown | 89 (51.5) | 18 (42.9) | 32 (50.8) | 39 (57.4) | |
|
| |||||
| Any degree of bowel incontinence reported, n (%) | 0.57 | ||||
| Yes | 45 (26.0) | 13 (31.0) | 17 (27.0) | 15 (22.1) | |
| No | 128 (74.0) | 29 (69.1) | 46 (73.0) | 53 (77.9) | |
|
| |||||
| Frequency of fecal incontinence, n (%) | 0.51 | ||||
| Daily | 1 (0.6) | 0 (0) | 1 (1.6) | 0 (0) | |
| 1–6 times/week | 14 (8.1) | 2 (4.8) | 6 (9.5) | 6 (8.8) | |
| 3–4 times/month | 30 (17.3) | 11 (26.2) | 10 (15.9) | 9 (13.2) | |
| A few times a year or less | 128 (74.0) | 29 (69.1) | 46 (73.0) | 53 (77.9) | |
The NBD severity score (range 0–47) identifies 4 subgroups based on score: very minor (score 0–6), minor (score 7–9), moderate (score 10–13), and severe (score 14+). Most patients (51.5%) scored within the “very minor” NBD severity category overall and within each bowel management group (Table 2). NBD severity score and NBD severity category, but not NBD satisfaction score nor PGH-7 T-score, varied significantly across the bowel management groups. Out of a total score of 47, the mean total NBD severity score was 8.6 for enemas, 9.4 for conservative therapies, and 4.5 for those not using a bowel management program (p<0.001).
Table 2:
Aggregated patient outcomes for NBD questionnaire and PGH-7, study population and subgroups by bowel management program.
| Total population n = 173 | Enemas n = 42 | Conservative n = 63 | No Bowel Regimen n = 68 | P value | |
|---|---|---|---|---|---|
|
| |||||
| NBD score, mean (std dev) | 7.3 (6.5) | 8.6 (4.9) | 9.4 (7.3) | 4.5 (5.7) | <0.01 |
|
| |||||
| NBD group, n (%) | <0.01 | ||||
| Severe | 29 (16.8) | 6 (14.3) | 18 (28.6) | 5 (7.4) | |
| Moderate | 26 (15.0) | 10 (23.8) | 10 (15.9) | 6 (8.8) | |
| Minor | 29 (16.8) | 9 (21.4) | 10 (15.9) | 10 (14.7) | |
| Very Minor | 89 (51.5) | 17 (40.5) | 25 (39.7) | 47 (69.1) | |
|
| |||||
| NBD Satisfaction Score, mean (std dev) | 7.1 (2.8) | 7.6 (2.3) | 6.6 (2.8) | 7.2 (3.1) | 0.15 |
|
| |||||
| Global Health QOL, PGH-7 T-score (std dev) | 47.1 (8.8) | 47.0 (9.0) | 45. 5 (8.5) | 48.7 (8.9) | 0.13 |
The overall mean NBD satisfaction score (primary outcome of bowel-specific QOL) was 7.1 (range 6.6–7.6). On univariable linear regression (Model 1a), compared to those using enemas, patients using conservative management (beta=−1.07, 95%Confidence Interval [CI]=[−2.19,0.04]) or no bowel program (beta=−0.42, 95%CI=[−1.52,0.68]) were not significantly associated with NBD satisfaction score (Table 3, Model 1). Similar null associations were found for both further-adjusted models.
For our secondary outcome of overall QOL based on PGH-7 T-score, univariable and multivariable linear regression found no significant associations with bowel management program (Table 3, Model 2).
For our secondary outcome of NBD severity, unadjusted analysis showed that those not using a bowel management program compared to those using enemas had an associated 4.1 point decrease in the NBD score (95%CI=[−6.49,−1.72], Table 3, Model 3). There was not a statistically significant difference when comparing conservative management to enemas. In multivariable analyses, there were shifts in the associations. Those using conservative management, compared to enemas, had an associated 2.58 point increase in NBD severity score (95%CI=[0.09,5.06]). There was not a statistically significant difference when comparing no bowel program to enemas.
The secondary analysis evaluating the association between overall QOL (PGH-7 T-score) and NBD severity score demonstrated a significant association (Table 4, Model 4). On both unadjusted and adjusted analysis, higher NBD severity score was associated with lower PGH-7 T-score (adjusted beta=−0.33, 95%CI=[−0.57,−0.10]).
The subgroup analyses for myelomeningocele diagnosis and ambulatory status found no significant associations between bowel management program and bowel-specific QOL, overall QOL, nor NBD severity when controlling for covariates (data not shown).
Discussion:
In our study, we sought to compare NBD outcomes across three bowel management programs commonly used in SB. We found no significant differences in bowel-specific QOL nor overall QOL in our fully-adjusted models. However, we found that use of a conservative bowel program was associated with worse NBD severity score compared to use of an enema program. Our results emphasize the need to balance the invasiveness of a bowel program with goals of care and expectations of patients. Not every patient needs an enema program to maintain good QOL if their symptoms are acceptable with less-invasive bowel programs. Shared decision-making is essential in counseling about tailored bowel programs.
Contrary to our hypotheses, we did not find any significant associations between bowel management program and QOL. For our primary outcome of bowel-specific QOL, mean NBD satisfaction score was 7.1 out of 10, but there was no significant difference between the bowel management programs for bowel-specific QOL. We similarly found no significant associations between bowel management program and overall QOL. Prior literature on this topic is conflicting. One case-control study also found that QOL did not differ by bowel management program in patients with SB.9 However, studies that assessed QOL/satisfaction before and after starting bowel programs showed improvement in both FI and QOL/satisfaction with targeted bowel therapy.21–24
We did find a significant difference when NBD severity score was treated as a continuous variable (scores ranging 0–47). Conservative management when compared to enema programs was associated with a 2.5-point worse NBD severity score on adjusted analyses. Though it is unclear whether this score difference is clinically meaningful, two randomized trials using pre-/post- designs have used NBD severity score as a continuous outcome and similarly found worse NBD scores in conservative management groups compared to enema groups.18,19 One trial noted a mean NBD severity score of 10.4 in the enema group versus 13.3 in the conservative management group.19 In these aforementioned studies, the enema groups also reported better wellbeing or improvement in QOL compared to the conservative management groups.
One key finding contextualizes the interpretation of our results. We had mostly low NBD symptom scores in our study cohort. The average NBD score for our study population overall was 7.3 out of a total possible score of 47, with most patients having “very minor” NBD severity across each bowel management group. There is also high variability in the presentation of neurogenic bowel symptoms in the spina bifida population. We see that there was a low NBD severity score (mean 4.5) in the no bowel management group and this did not negatively impact their QOL, again suggesting that therapies should be individualized. There was a larger percentage of patients in the “severe” NBD grouping using conservative therapies (28.6%) compared to enemas (14.3%), but statistical significance is likely limited by our sample size. Importantly, as this is a cross-sectional study with patients and caregivers answering the questionnaires when already maintained on their bowel therapy (versus a pre-/post- study design), the low overall NBD severity suggests patients’ bowel symptoms are already being appropriately managed. Several studies have reported improvement of bowel outcomes when utilizing a targeted bowel management program.21,25–28 Additionally, only a quarter of our study population reported any degree of FI, which is strongly weighted into the score for the NBD questionnaire.13
Taken together, our results and the prior literature suggest that properly—or improperly— managed bowels and bowel symptoms may have a greater impact on QOL than any one bowel management program in particular. The association between NBD symptoms and QOL is well-established. Published data robustly show that FI and worse bowel symptoms are associated with lower reported QOL in those with NBD.2–4,7,8,27,29 In our post-hoc secondary analysis, we confirmed this same association, with higher NBD severity score being associated with worse QOL in adjusted analyses. Our data suggest that while worse NBD symptoms are associated with worse QOL, the bowel program used to mitigate NBD symptoms can be tailored to the individual patient to accomplish the same goal of enhancing QOL. Some patients may have minimal baseline NBD symptoms that require minimal bowel programs, while others with major NBD baseline symptoms may require enema programs, but their tailored bowel programs can hopefully allow them to reach comparable post-intervention NBD symptoms and thus QOL.
Our study has limitations. First, as our study is retrospective in nature, we do not have information on duration of reported bowel programs, compliance to reported bowel programs, or pre-/post-intervention measures of NBD symptoms and QOL for intra-individual analyses. Our results are exploratory, given the lack of adjustment for multiple outcomes, the post-hoc secondary analyses, and the relatively small sample size within each bowel management group. We used the modified adult Peristeen questionnaire given that the average age was approaching adulthood. However, the questionnaire was originally validated in the SCI population and not the SB population.14 Lastly, our results may have limited generalizability. Our study cohort was cared for at a single, Midwest, urban referral center. Compared to the National Spina Bifida Patient Registry, our population had lower rates of FI in part due to the older age in our group and already being on bowel programs.30 For future research, we will aim to compare questionnaires before and after initiating a new bowel management program.
Conclusion:
There were no significant differences in bowel-specific QOL nor overall QOL between bowel management programs (enemas, conservative, and none) when controlling for covariates. There was an associated increase in NBD severity score for patients utilizing conservative management compared to enemas, suggesting there may be room for improved targeted management in this group. Consistent with previously published literature, higher NBD severity score was significantly associated with lower overall QOL. In our study cohort with overall well controlled NBD symptoms on their respective bowel management programs, it appears the individual program utilized may not be the primary factor driving differences in QOL, but rather the results of the program. Shared decision making is necessary to allow clinicians and families to focus on tailoring bowel management aimed at symptom control.
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