Reviewing definitions of urinary continence in the contemporary spina bifida literature: A call for clarity

Journal of Pediatric Urology (2013) 9, 567e574

Reviewing definitions of urinary continence in the contemporary spina bifida literature: A call for clarity Jessica C. Lloyd*, Unwanaobong Nseyo, Ramiro J. Madden-Fuentes, Sherry S. Ross, John S. Wiener, Jonathan C. Routh Division of Urologic Surgery, Duke University Medical Center, DUMC 3831, Durham, NC 27710, USA Received 4 December 2012; accepted 19 February 2013 Available online 16 March 2013

KEYWORDS Neural tube defect; Urinary incontinence; Urology; Pediatrics

Abstract Background: Urinary continence is a common goal for children with spina bifida and their physicians. However, definitions of urinary continence vary widely across published studies. We systematically assessed the utilization of common definitions of urinary continence in the spina bifida literature. Methods: We searched library databases for reports (2000e2012) describing urinary continence outcomes in children with spina bifida. We assessed various patient-level factors such as age, lesion level, surgical history, and use of additional therapies, as well as study-level factors such as study design, country of origin, continence definition(s), and method of data collection. Results: Of 473 identified articles, 105 met inclusion criteria, comprising a total of 3209 patients. Of these, 1791 patients (56%) were deemed continent by the study authors. Only 60 studies (57%) clearly defined what they considered to be “continent”. The most common definition, used in 24% of all reports, was “always dry”. There was no association between journal of publication (p Z 0.13), publication year (p Z 0.86), study size (p Z 0.26), or study country (p Z 0.43) and likelihood of a continence definition being included in the manuscript. Conclusions: The most frequent definition of urinary continence in the spina bifida literature is “always dry”. However, definitions were highly variable, and many authors did not define continence at all. Clinicians and researchers alike would be better able to apply research findings toward improving patient care if continence definitions were more explicitly reported and less variable. ª 2013 Journal of Pediatric Urology Company. Published by Elsevier Ltd. All rights reserved.

Abbreviations: SB, spina bifida; UI, urinary incontinence. * Corresponding author. Tel.: þ1 919 684 6994; fax: þ1 919 681 5507. E-mail address: [email protected] (J.C. Lloyd). 1477-5131/$36 ª 2013 Journal of Pediatric Urology Company. Published by Elsevier Ltd. All rights reserved. http://dx.doi.org/10.1016/j.jpurol.2013.02.006

568

Introduction Urinary incontinence (UI) is a frequently-encountered, clinically-significant problem in children with spina bifida (SB) [1]. Urinary incontinence reduces children’s selfesteem, quality of life and independence [2], in addition to causing physical problems such as skin irritation and wound complications [3]. However, procedures to promote continence are not without risks, including need for repeated operations, frequent urinary tract infections, and decline in renal function [4e7]. Given its clinical significance, a substantial body of medical literature is devoted to the attainment of urinary continence in children with SB; however, definitions of urinary continence are surprisingly variable across studies. In addition, there is no general consensus on which methods researchers should use to assess continence. With such a wide range of definitions and often unclear methodology, it is no wonder that researchers and clinicians alike struggle to interpret study results. Only a uniform, standard definition of urinary continence will allow accurate comparisons between studies of continence outcomes in children with SB. Indeed, there has been significant effort to standardize definitions in other fields, and numerous working groups have created reporting guidelines for the explicit purpose of making scientific literature more complete, clear, and transparent [8e10]. Because the population of children with SB is relatively small, being able to pool and compare data across studies is particularly critical; however, deficiencies in reporting, such as incomplete or selective reporting of outcomes, poor methodology descriptions, or failing to clearly define outcomes (in this case, continence), make this task even more challenging [11]. A single, widely accepted definition of urinary continence will increase the clinical utility and applicability of the SB literature. Given the current lack of consensus, we sought to investigate the degree of standardizationdor lack thereofdof urinary continence definitions in children with SB. We therefore reviewed definitions of urinary continence that were reported in the contemporary spina bifida literature. Our objective was to assess the most common definitions used to describe continence and the most common methods by which authors determine continence rates.

Patients & methods Search strategy We searched MEDLINE, EMBASE, CINAHL, the Cochrane Library, the Web of Science and the Conference Papers Index for studies published between January 2000 and August 2012 based upon AMSTAR [12] and PRISMA [13] guidelines. This date range was chosen to provide a contemporary selection of studies. We used the MeSH search term “urinary incontinence” or the text terms “urinary” AND “incontinence”. Using Boolean operators, we then restricted the results to articles retrieved under a second search for the MeSH terms “spinal dysraphism”, “spina bifida”, “meningocele” or “neural tube defect”. In our second search, the

J.C. Lloyd et al. text terms “myelomeningocele”, “lipomyelomeningocele”, or “lipomeningocele” were also included.

Selection criteria We reviewed all retrieved manuscripts that assessed continence status in a predominantly pediatric cohort with any of the neural tube defects listed above. We defined a pediatric cohort as one with an average age of <18 years. No manuscript was excluded based on study design, method of analysis, definition of success, or perceived quality. We included non-English-language reports; these were translated by study authors fluent in those languages. We required that the number of patients treated and the fraction for which the treatment was successful be reported. For studies reporting both fecal and urinary continence rates, we required that urinary continence be clearly delineated separately from fecal continence. Reviews and metaanalyses that did not include a unique study population but instead aggregated data from other sources were excluded.

Data abstraction Two reviewers (JCL and UN) independently reviewed study abstracts in parallel with questions and disagreements resolved by consensus between the two reviewers. The full text of articles that met selection criteria were reviewed and study data abstracted. Abstracted data included: the site and country in which the study was performed, study period, study design, total number of patients, patient gender, patient age, underlying disorder, operations performed, duration of follow-up, definition of “continence”, method for determining continence, use of clean intermittent catheterization via the native urethra or continent catheterizable channel, and use of anticholinergic medications. In cases where authors did not provide sufficient information in their articles to complete our dataset, we did include the study but noted the missing data. Our primary outcome of interest was the definition of continence employed in each study, or in other words what clinical benchmark was required for a patient to be deemed “continent” (e.g. no urinary leakage at any time, no leakage for 2 h intervals, use of security pads only, etc.).

Statistical analysis We performed bivariate tests of association between predictor variables and study outcomes using Fisher’s exact test and chi-squared test for categorical variables, and the KruskaleWallis test for continuous variables. Outcomes assessed included continence definitions and methods of continence ascertainment. Statistical analyses were performed using Stata 11.2 (College Station, TX, USA).

Results Systematic literature review Our search strategy identified 473 abstracts. After our initial screen of abstracts and titles, 176 studies were

Definitions of continence in spina bifida patients reviewed in full, and 105 studies met our inclusion criteria (Fig. 1). In total, 26 countries were represented, with the USA (31%), Canada (12%), Germany (8%) and Iran (8%) being the most frequent (Table 1). Studies were almost universally case series (92%), with a small number of caseecontrol (2%), prospective cohort (1%), retrospective cohort (2%), and randomized controlled trials (3%). Median follow-up among studies was 3.8 years (range 0.08e16.3 years). The median study size was 20 patients (range 2e244). Twentysix journals were represented, with the most frequent being the Journal of Urology (47%), followed by the British Journal of Urology International (10%), and Urology (6%). The vast majority of manuscripts were published in English (103, 98%), though one French-language and one Germanlanguage study met inclusion criteria. A single article each in Czech and Korean were reviewed, but did not meet inclusion criteria.

Patient characteristics Our systematic review included 3209 patients. Of these, 2460 patients (82%) were diagnosed with spina bifida or other neural tube defect, though 5 authors did not report the proportions of patients with each underlying condition in the study. Amongst studies that listed subject gender, there were 1247 males (51%), though 20 studies did not differentiate patients by gender (Table 2). The mean age at the time of enrollment was 10 years (SD 4.7 years). Across all studies, the average youngest age at enrollment was 4.4 years (SD 3.5 years) and the average oldest age at enrollment was 17.5 years (SD 8.5). Among the 13 studies (12%) that commented on lesion level, lumbar lesions were most common (189/494, 38%).

Reporting of continence outcomes Of 3209 included children, 1791 (56%) were defined as continent by the study authors. However, only 60 studies (57%) clearly defined what they considered to be “continent”. Reported definitions ranged from ‘always dry’ (n Z 25, 25%), ‘dry with voiding/CIC every 2 h’ (n Z 2, 2%), ‘dry with voiding/CIC every 3 h’ (n Z 8, 8%), ‘dry with voiding/CIC every 4 h’ (n Z 13, 13%), ‘no pad use’ (n Z 9, 9%), ‘security pads only’ (n Z 1, 1%) and ‘no leakage observed on urodynamics’ (n Z 2, 2%) (Table 3). The most commonly reported definition, used in 25 studies (42% of all

473 Articles Identified Abstracts reviewed

176 Studies Reviewed in Full Papers reviewed

105 Studies Included

Figure 1

297 Articles Excluded -No continence outcome (81) -Review (64) -Editorial comment (21) -Duplicate study (29) -Adult patient population (41) -Single patient case study (31) -No unique patients (11) -Not spina bifida cohort (19)

71 Studies Excluded -Insufficient discussion of continence (29) -Technical review (4) -Duplicate study (1) -Cohort average age >18 (17) -Single patient case study (3) -No unique patients (1) -Not spina bifida cohort (16)

Flow diagram of literature search and study review.

569 studies that report a definition), was “always dry” throughout the day and night. Though all studies referenced continence outcomes, 45 studies (43%) did not include a definition of “continence”. There was no association between journal of publication (p Z 0.13), publication year (p Z 0.86), study size (p Z 0.26), or study country (p Z 0.43) and likelihood of continence being defined in the manuscript. The methods by which continence data were determined were less clear. Of the 105 studies, 66 (63%) did not describe how continence status was ascertained (i.e., whether by patient report, parental report, voiding diary, etc). In studies that did characterize this aspect of data acquisition, authors reported using patient report to study staff (n Z 9, 23%), patient or parent report to study staff (n Z 6, 15%), voiding diary (n Z 8, 21%), pad weight (n Z 2, 5%), patient or parent paper surveys (n Z 5, 13%), chart review (n Z 6, 15%), and physician observation at urodynamics testing (n Z 2, 5%). Although a greater percentage of recent studies explicitly described the methods they used to ascertain continence (Table 4), this trend did not reach statistical significance (p Z 0.12).

Reporting variation across multiple studies by a single author Several centers published multiple studies included in our analysis. In total, 11 authors published 29 of the reports included in this study, encompassing 941 patients (29%). Notably, repeat authors were more likely to include a continence definition in their manuscript (72% of studies written by repeat authors included a continence definition, vs 49% of single-study authors, p Z 0.077). However, of the 11 repeat authors, only 5 (45%) were consistent across studies in their continence definitions. Of the 6 authors whose definitions varied, 3 failed to include a definition in early manuscripts and then did define continence in later studies, whereas 3 simply changed definitions between studies. Notably, no author defined continence in early studies and then failed to do so in later studies. Repeat authors also appeared more likely to delineate the methods by which they ascertained continence (41% vs 36%); however this failed to reach statistical significance (p Z 0.65). In total, 8 repeat authors (73%) were consistent in their methodological descriptions (or lack thereof). Of the 3 repeat authors whose continence ascertainment methods varied, we noted a pattern of evolving awareness of including methodology in their manuscripts, with all 3 demonstrating a change from ‘no method specified’ to inclusion of a methodological description.

Discussion Children with SB often struggle to achieve urinary continence. As such, research related to SB frequently addresses urinary continence outcomes. In this study, we found that only 57% of authors clearly defined the parameters used to define “continence”, despite the fact that all reported continence outcomes. The most common definition we encountered was “always dry”, but even this was only used

570 Table 1

J.C. Lloyd et al. Characteristics of included studies.

First author

Publication date

Country

Study design

Ahmadi Albouy Almodhen Armando Bagrodia Barbosa Bar-Yosef Battaglino Bersch Bissada

2010 2007 2007 2012 2011 2009 2011 2005 2009 2007

Iran France Canada Italy USA Brazil USA Italy Switzerland USA

Case Case Case Case Case Case Case Case Case Case

Block Bugg Canales Castera Cole Colvert Daher Dave Dean Dean DeFoor

2003 2003 2006 2001 2003 2002 2001 2008 2007 2009 2012

USA USA USA Argentina USA USA France Canada USA USA USA

Deshpande

2010

Australia

Diamond Diamond Dik Dik Dik Ghanem Godbole Godbole Guerra Gundeti Guys Guys Guys

2000 2004 2010 2003 2003 2012 2003 2003 2006 2010 2006 2001 2004

USA USA Canada Holland Holland Holland UK UK Canada USA France France France

Case series Case series Case series Case series Case series Case series Case series Case series Case series Case series Retrospective cohort Prospective cohort Case series Case series Case series Case series Case series Case series Case series Case series Case series Case series Case series Case series RCT

11 109 13 35 24 24 15 19 24 6 49 44 42

Haferkamp Halachmi Harris Hayashi Herdon Hoebeke Hurley Idris Jorgensen Kajbafzadeh Kajbafzadeh Kajbafzadeh Kajbafzadeh Kajbafzadeh Kajbafzadeh Kassouf Kaye Kessler

2000 2004 2000 2007 2004 2000 2000 2010 2010 2008 2011 2006 2010 2008 2010 2001 2010 2006

Germany Canada USA Japan USA Belgium Canada Malaysia Denmark Iran Iran Iran Iran Iran Iran Canada USA Austria

Case Case Case Case Case Case Case Case Case Case Case Case Case RCT Case Case Case Case

32 33 50 4 15 17 19 42 60 20 15 26 44 30 12 20 10 123

series series series series series series series series series series

series series series series series series series series series series series series control series series series series

# Patients 15 14 37 12 4 23 21 7 51 17 19 15 9 49 43 20 11 15 34 6 5 7

Age (y)

Definition of dry

Ascertainment method

Patients dry (%)

7.80 14.00

None given Q3 h interval Q4 h interval Urodynamics Always dry None given None given None given None given Q3 h interval

53 93 59 67 100 91 81 86 90 94

9.3

None given Q4 h interval Q3 h interval None given Q4 h interval None given None given Q4 h interval None given Q3 h interval None given

Not specified Chart review Not specified Not specified Not specified Chart review Chart review Not specified Patient report Patient or f amily report Patient report Not specified Voiding diary Not specified Not specified Not specified Not specified Not specified Not specified Not specified Not specified

16

None given

Survey

14.6 9.7

None given None given None given None given None given Always dry None given Always dry None given Q4 h interval Q4 h interval Q4 h interval Always dry None given Always dry Q4 h interval None given Q3 h interval None given Always dry None given None given None given Always dry Always dry Always dry Always dry Always dry Always dry Always dry Q4 h interval Always dry

Not specified Not specified Not specified Not specified Not specified Not specified Not specified Not specified Not specified Patient report Not specified Not specified Physician observation Not specified Not specified Not specified Voiding diary Not Specified Not specified Not specified Not specified Not specified Voiding diary Voiding diary Voiding diary Not specified Voiding diary Voiding diary Patient report Patient report Not specified

a a

9.75 3.64 10.4 13.00 38.7 10.2 11.7 a a

14 10.1 8.7 a

11.8 11.7 a

a

9.9 9 a

9.92 a

6.00 9.75 14.00 13.00 11.90 12.00 12.40 13.10 1.75 11.00 13.50 a a

0.00 8.70 7.80 6.90 9.20 5.60 5.04 13.30 9.4 17

0 60 89 100 74% 70 100 13 71 83 20 0 18 86 85 54 92 100 20 21 54 67 33 34 2 66 9 98 75 100 100 47 17 60 85 53 73 27 37 17 5 70 74

Definitions of continence in spina bifida patients

571

Table 1 (continued ) First author

Publication date

Country

Study design

Khoury Kumar Landau Lehnert Liard Lopez-Pereira MacNeily MacNeily

2008 2010 2009 2012 2001 2005 2003 2005

Canada Canada Israel Germany France Spain Canada Canada

Marte Mazzone Medel

2002 2006 2002

Italy Switzerland Argentina

Case series Case series Case series Case series Case series Case series Case series Retrospective cohort Case series Case series Case series

Metcalfe Mitsui Moore Mosiello Neel

2011 2008 2004 2011 2010

Neel

2007

Nguyen Nguyen Nomura Obermayr

2003 2005 2002 2011

Canada Japan USA Italy Saudi Arabia Saudi Arabia USA USA Japan Germany

Case Case Case Case

Ong

2002

Palmer Pascali Perez Peters Potter

2012 2011 2000 2010 2002

Ramsak Rawashdeh Sanders

2004 2004 2002

Schulte-Baukloh

# Patients 48 9 12 21 23 17 17 36

Age (y)

Definition of dry

Ascertainment method

Patients dry (%)

a

Q4 h interval Q4 h interval None given None given Always dry Always dry No pad use Q3 h interval

60 100 100 62 65 94 24 69

11 2 26

12.80 9.50 12.00

No pad use None given No pad use

155 22 50 5 13

10.00 14.40 4.50 5.30

Always dry None given No pad use Q4 h interval Always dry

Not specified Not specified Chart review Not specified Not specified Not specified Not specified Patient or f amily report Not specified Chart review Physician observation Patient report Survey Survey Not specified Not specified

23

5.60

Always dry

Not specified

39

series series series series

20 11 10 25

16.20 7.50 16.70 7.20

None given Always dry None given None given

30 55 100 76

Malaysia

Case series

66

6.00

Case Case Case Case Case

6 75 11 9 9

a

0 8.00 9.3

Case series Case series Case series

41 14 111

9.80 6.7 14

None given None given None given

Chart review Not specified Not specified

20 57 74

2012

USA Italy USA USA United Kingdom Slovenia Denmark United Kingdom Germany

Security pads only None given None given None given None given None given

Not specified Chart review Not specified Patient or family report Patient or family report Not specified Pad weight Voiding diary Not specified Not specified

Case series

17

13.00

Always dry

53

Shadpour Singh Skobejko Snodgrass

2005 2010 2001 2009

Iran India Poland USA

Case Case Case Case

series series series series

6 68 14 41

11.17

Q2 h interval Always dry Q3 h interval No pad use

Patient or family report Not specified Not specified

Spiess Stehr Stein Stein Stein Storm Taskinen Thorup Torre VanDerBrink VanSavage Walker

2002 2000 2000 2005 2005 2008 2007 2010 2008 2011 2001 2001

Canada Germany Germany Germany Germany USA Finland Denmark Italy USA USA USA

Case Case Case Case Case Case Case Case Case Case Case Case

series series series series series series series series series series series series

30 9 129 70 24 2 13 52 244 43 5 35

Case Case Case Case Case

series series series series series

RCT

control series series series series

a

7 7.6 8.00 14.40 10.20 a

a

a

a

11.00 8.00 12.60 9.00 12.10 15.30 10.80 9.50 15.50 6.00 13.50 9.00 15.00 a

Always dry Always dry No pad use No pad use No pad use None given No pad use Always dry Q4 h interval None given None given Q3 h interval

64 0 42 35 82 20 100 54

9 67 71 64 0 22

100 9 86 83

Patient or family report Not specified 63 Not specified 67 Patient report 32 Patient report 90 Patient report 83 Not specified 100 Pad weight 0 Not specified 38 Not specified 45 Chart review 95 Not specified 100 Not specified 49 (continued on next page)

572

J.C. Lloyd et al.

Table 1 (continued) First author

Publication date

Walker Wille Xiao Youdim Zerhau

2000 2011 2005 2002 2011

a

Country

Study design

USA USA USA USA Czech Republic

Case Case Case Case Case

series series series series series

# Patients 15 11 20 25 17

Age (y)

Definition of dry

Ascertainment method

Patients dry (%)

12.90 10.40 11.00 9.50 9.50

Always dry None given Q2 h interval None given None given

Survey Not specified Not specified Survey Not specified

33 100 85 72 12

Data not provided by authors.

by a pluralitydnot a majoritydof studies. Only 24% of all authors, or 42% of authors who defined continence, used this definition. Only 45% of authors who published more than one manuscript were consistent in their definitions between studies. The methods used to ascertain continence were rarely reported; when reported, these were also highly variable. Only 37% of authors clearly described the methods they employed to determine patients’ continence status. Undefined outcomes are unfortunately frequent in published clinical research. In the exstrophyeepispadias literature, we previously found that 32% of authors did not define “continence” in studies of urinary continence rates [14]. Similarly, a recent study on “stone-free” status after intervention for kidney stones found that nearly one-third of authors failed to define this outcome measure [15]. However, since success rates at least in part reflect how an author defines “success”, it is essential that authors make clear the exact parameters that they use to define a successful outcome. Our review of the contemporary SB literature underscores the lack of consensus regarding the meaning of urinary continence, a fundamental concept in the urologic care of children. We identified 7 different definitions of urinary continence in the 105 studies we analyzed. As previous authors have noted, study results varyd sometimes dramaticallyddepending on how a positive outcome is defined, in outcomes as diverse as childhood obesity [16], anemia in adolescents [17], and bacterial

Table 2

colonization of children with cystic fibrosis [18]. Only a uniform, transparent definition will allow for accurate comparisons between studies. The call for a comprehensive and generalizable definition of urinary continence is not novel [14]. Numerous authors have called for consensus definitions, across a broad range of clinical areas of study, including chronic constipation [19], childhood obesity [20], childhood asthma [21], and cystic fibrosis pulmonary exacerbations [22]. We, likewise, believe that authors reporting continence outcomes in children with SB should use definitions which are consistent with other literature and methodologically transparent. Moreover, authors should precisely report the methods by which they ascertained their subjects’ urinary continence status. Given that high quality clinical research drives both bedside patient care and future research investigations, communicating research outcomes is clearly critical. Only then will clinicians and researchers, as well as patients and their families, be able to accurately compare studies and make the most informed decisions with regard to the care and study of SB patients. In our analysis, the most common definition of urinary continence was “always dry” throughout the day and night; continence status was most commonly ascertained by patient report. There are certainly advantages to this definition and methodology. First, it is a true gold-standard of urinary continence; there is no continence status superior to “always dry”. Second, “always dry by patient report” is a profoundly patient-centered outcome. While

Clinical characteristics of included patients. Number of studies reportinga

Female Male (n reported Z 2455) Mean age, years (range, SD) Mean follow-up, years (range, SD) Lesion level Thoracic Lumbar Lumbosacral Sacral (n reported Z 494) Use of CIC (n reported Z 2061) Use of anticholinergic medications (n reported Z 1247) History of bladder augmentation (n reported Z 1922) a

Out of 105 studies included in this analysis.

1208 1247 10.0 3.8

(49%) (51%) (4.4e17.5, 4.7) (0.08e16.3, 3.5)

85

(10%) (38%) (28%) (24%)

13

1475 (72%) 717 (57%) 954 (50%)

75 39 68

49 189 137 119

86 71

Definitions of continence in spina bifida patients Table 3

573

Reported definitions of continence.

Definition

Number of papers using definition (percentage of all papers, n Z 105)

Percentage of papers that define continence using definition (n Z 60)

Always dry Dry with Q2 h voidinga Dry with Q3 h voidinga Dry with Q4 h voidinga No pad use Use of security pads only No leakage on UDS No definition given

25 (24%) 2 (2%) 8 (8%) 13 (12%) 9 (9%) 1 (1%) 2 (2%) 45 (43%)

42% 3% 13% 22% 15% 2% 3% NA

a

Or catheterization.

encourage the use of the popular definition “always dry by patient report”, we also believe that authors must inquire how frequently SB patients are voiding/catheterizing and should report this information in their manuscripts. Journal editors and peer reviewers can also improve reporting by requiring that manuscripts include an explicit definition of continence and measurement parameters prior to publication. Our review should be interpreted in light of its limitations. As with any systematic review, our analysis was limited by the data reported by study authors. Published studies regarding urinary continence in SB patients are predominantly case series and rarely report continence outcomes in a manner that makes them readily amenable to comparison. In our series, 43% of authors failed to define urinary continence, despite the fact that it was a reported study outcome. While some reviewers may have excluded these studies, we believe drawing attention to these shortcomings in the literature is important to our ultimate goal of enhancing standardization and improving clinical usefulness of the SB literature. Additionally, we must note that 18% of patients in this study were diagnosed with conditions other than SB (such as spinal cord injury, caudal regression syndrome, or exstrophy/epispadias complex). Given that SB is a rare condition, we chose not to exclude studies with mixed populations of SB and non-SB patients. Had we excluded these studies, valuable continence outcome data would have been lost. Moreover, our review attempts to capture data as it has been presented by SB researchers; if they find utility in grouping SB patients with other pediatric voiding dysfunction patients, then we will follow this convention as well.

less quantifiable than pad weight or leakage at the time of urodynamics, “always dry” perfectly pinpoints a patient’s perception of continence. Indeed, surgeons in a variety of fields have begun to place a greater emphasis on patientrelated endpoints, rather than endpoints relating directly to the intraoperative surgical technique. Examples of this include the return to normal activity level after anterior cruciate ligament reconstruction [23], patient perception of vaginal bulge after pelvic organ prolapse surgery [24], and patient satisfaction with the appearance of breast reconstruction after mastectomy [25]. Such efforts are commensurate with the ongoing implementation of patient-reported outcomes as measures of quality healthcare. As the patient-centered model of care becomes more prevalent, we must work to make SB care truly focused on goals that are important to our patients, and not just physicians. Without knowing which continence goals are important to patients, efforts made in the area of continence achievement and improved quality of life may be misdirected. Acknowledging patient- and caregiver-driven continence goals is one way to include these stakeholders in our evolving care model. However, “always dry by patient report” does have shortcomings. In particular, it does not convey the lengths to which patients must go to achieve their always dry state. If a patient can only achieve continence with burdensome catheterization every hour or two or high doses of anticholinergic medications that provoke bothersome sideeffects, are they truly achieving the “gold-standard”? Moreover, the incentive structure of the patientephysician relationship may bias patients toward reporting overlyfavorable outcomes to their study doctors, thus skewing the data toward more positive results. As such, while we

Table 4

Studies reporting method of ascertaining continence by year (p Z 0.12).

Years

Not Patient Patient or Voiding Pad Survey Chart MD Total giving % Giving specified report family report diary weight review observation specific method specific method

2000e2004 2005e2008 2009e2012 Total

31 14 20 65

3 2 4 9

1 2 3 6

1 5 2 8

0 1 1 2

3 1 1 5

1 3 4 8

2 0 0 2

11 14 15 40

26% 50% 43% 38%

574

Conclusions In the SB literature, the most frequently used definition of urinary continence is “always dry”, as reported by the patient. However, reported definitions varied widely, and 43% of authors failed to define continence. Given the importance of urinary continence to children in general and to SB patients in particular, there is a clear need for greater clarity, transparency, and standardization of definition of urinary continence in the contemporary spina bifida literature. In this vein, we suggest “always dry by patient report” as a new standard for defining continence in the SB literature.

Ethical approval In lieu of a formal ethics committee, the principles of the Helsinki Declaration were followed.

Funding source No external funding was secured for this study.

Conflict of interest JSW currently serves on the pediatric urology working group for RTI International Best Hospitals Project which is a consultant to US News and World Report magazine regarding their ranking of pediatric urology hospitals. JSW also has served on an advisory panel for Glaxo Smith Kline. None of the remaining authors have any conflict of interest, financial or otherwise, to disclose.

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