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Assessing quality of life of patients with hypospadias: A systematic review of validated patient-reported outcome instruments
Accepted Manuscript Assessing quality of life of patients with hypospadias: a systematic review of validated patient-reported outcome instruments K.J. Sullivan, Z. Hunter, V. Andrioli, L. Guerra, M. Leonard, A. Klassen, M. Keays PII:
S1477-5131(16)30387-4
DOI:
10.1016/j.jpurol.2016.11.010
Reference:
JPUROL 2393
To appear in:
Journal of Pediatric Urology
Received Date: 4 August 2016 Accepted Date: 8 November 2016
Please cite this article as: Sullivan KJ, Hunter Z, Andrioli V, Guerra L, Leonard M, Klassen A, Keays M, Assessing quality of life of patients with hypospadias: a systematic review of validated patient-reported outcome instruments, Journal of Pediatric Urology (2017), doi: 10.1016/j.jpurol.2016.11.010. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
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Assessing quality of life of patients with hypospadias: a systematic review of validated patient-reported outcome instruments
Department of Pediatric Surgery, Division of Urology, Children’s Hospital of Eastern Ontario,
401 Smyth Road, Ottawa, ON, Canada b
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K. J. Sullivana, Z. Hunterb, V. Andriolia, L. Guerraa,b, M. Leonarda,b, A. Klassenc, M. Keaysa,b,*
Faculty of Medicine, University of Ottawa, 451 Smyth Rd, Ottawa, ON, Canada
Departments of Pediatrics and Clinical Epidemiology & Biostatistics, McMaster University,
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1280 Main Street West, Hamilton, ON, Canada
*Corresponding author: Children’s Hospital of Eastern Ontario, 401 Smyth Road, Ottawa, ON,
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K1H 8L1, Canada. Tel.: (613) 737-7600 ext. 3864; fax: (613) 738-4271.
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E-mail address: [email protected] (Dr. Mélise Keays)
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Summary Background: Patient-reported outcomes have the potential to provide invaluable information for
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evaluation of hypospadias patients, aid in decision-making, performance assessment, and improvement in quality of care. To appropriately measure patient-relevant outcomes, welldeveloped and validated patient-reported outcome (PRO) instruments are essential.
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Objective: To identify and evaluate existing PRO instruments designed to measure quality of life and/or satisfaction of individuals with hypospadias that have been developed and validated in
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a hypospadias population.
Methods: A systematic search of MEDLINE, EMBASE, PsycINFO, CINAHL and Health and Psychosocial Instruments was conducted in April 2016. Two reviewers independently assessed studies and identified PRO instruments for inclusion. Data were extracted on study characteristics, instrument development and validation, and content domains.
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Results: A total of 32 studies were included that used or described five PRO instruments: Hypospadias Objective Scoring Evaluation (HOSE), Pediatric Penile Perception Score (PPPS),
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Penile Perception Score (PPS), Genital Perception Scale (GPS) for adults, and GPS for children/adolescents. Instrument development and validation was limited. The majority of
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identified instruments focused on postoperative cosmetic satisfaction, with only one instrument considering urinary function, and no instruments evaluating sexual function and psychosocial sequelae.
Conclusions: While many hypospadias studies have acknowledged the necessity of a patientreported element, few have used validated PRO instruments developed in a hypospadias population. Existing instruments to measure patient-reported outcomes in hypospadias require
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improvement in both the breadth of content and in their development and validation methodology.
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Keywords: Hypospadias; Patient-reported outcome (PRO); Satisfaction; Quality of life
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Introduction Patient-reported outcome (PRO) instruments are questionnaires/instruments that allow for self-
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reporting of the patient (or parent-proxy) experience, potentially including views of their
symptoms, functional status, and health-related quality of life (QoL) [1]. While originally
designed for use in research [1], PRO instruments have been adopted by healthcare professionals
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to support various clinical efforts, including quality improvement, performance assessment, and the tailoring of treatment plans to meet patient preferences and needs [2]. The latter is of
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particular importance, as healthcare professionals frequently misjudge the absolute levels of symptoms and general QoL, tending to underestimate the impact of psychological factors while emphasizing more obvious symptoms [3]. Jachuck et al. [4] observed this in their questionnaire study of hypertensive patients and their doctors, where all physicians indicated that patients had
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improved, while approximately half of the patients felt that there was no change or even deterioration. Physicians tended to ignore the factors that patients factored into their overall wellbeing, including a decline in energy, general activity, sexual inactivity, and irritability [4].
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Patient-reported outcomes are particularly useful for conditions where a large variation in care and outcomes exists, and where the impact on the patient’s QoL is currently unknown. Such
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is the case for the congenital condition of hypospadias. While the goals of hypospadias repair are generally agreed upon – including providing the patient with satisfactory urinary function, sexual function, body image or cosmesis, and quality of life – several important variations remain. First, there is variation in surgical techniques for the same condition; for example, the use of one-stage vs. two-stage repair for proximal hypospadias [5]. While advantages have been established for each technique, the current evidence base cannot definitively identify an optimal method for
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individual patients [6]. Patient input, obtained through PROs, on the relative benefits may help elucidate this (e.g. does the reduced hospital stay and anesthetic risk associated with a one-stage repair offset the increased risk of complications [5,7] compared with a two-stage repair?).
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Additional variance also exists concerning physician recommendations and parental preference for surgical correction of distal hypospadias (i.e. glanular), where some routinely
elect/recommend surgical repair and others prefer to forgo surgery in mild cases. Finally,
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variance can be observed in surgical success, where 15% of patients experience a complication (a value that at least doubles in patients with severe defects or prior complications) [7-9].
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While it remains important to measure these results using traditional surgical outcomes, such as complications and need for reoperation, these no longer sufficiently capture all important aspects from the patient’s perspective [10-12]. Rather, there is a need to capture the considerable long-term cognitive, behavioral, and self-esteem consequences that result from poor cosmetic or
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functional outcomes – such as negative genital perception [13,14], sexual avoidance [15,16], and poor school performance [17] – through the use of PRO measures. Comprehensive measurement of surgical outcomes requires a combination of objective and subjective measures.
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Clinical outcomes research in hypospadias surgery is becoming increasingly focused on the patient’s QoL and their perception of a satisfactory outcome [10]. As a result, the present
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systemic review of the published literature was conducted to identify and assess PRO instruments currently available for hypospadias patients or parent-proxies. The primary objective was to identify existing PRO instruments that have been developed and validated in a hypospadias population that assess patient satisfaction and/or quality of life. A secondary objective was to evaluate the development and validation of the instruments, and to assess the content of identified instruments.
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Methods Inclusion criteria Studies were included that described PRO instruments designed to measure quality of life and/or
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satisfaction of individuals with hypospadias or their parent-proxy that had been developed and validated in a hypospadias population. Non-English language studies were excluded, as were
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conference abstracts.
Literature search
The following databases were searched: MEDLINE including In-Process & Other Non-Indexed
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Citations (1946-April 8 2016), EMBASE (1980-week 14 2016), PsycINFO (1806-April week 1 2016), CINAHL (April 8 2016), and Health and Psychosocial Instruments (1985-January 2016). The MEDLINE search strategy was developed by a librarian experienced in systematic review searching, and peer reviewed by a second librarian using the PRESS standard [18]. The
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MEDLINE search was then adapted to allow for optimal searching of other included databases. Search strategies are presented in the Appendix. Hand-searching the reference sections of
Screening
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relevant articles identified additional publications.
At level 1 (title and abstract), studies were screened independently in duplicate using the liberal
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accelerated method [19]. Two researchers then independently assessed full-text articles (level 2 screening) and compared decisions to reach consensus for final inclusion. Disagreements were resolved by discussion or third party consultation when necessary.
Development Development and validation criteria criteria Validated instruments were identified from included studies and were evaluated for their adherence to the three-stage, rigorous, gold standard methodology for the development and validation of health outcome measures developed by the Scientific Advisory Committee (SAC) 5
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of Medical Outcomes Trust International guidelines [20] and described by Cano et al. [21]. According to this methodology, stage 1 involves generating a list of items for the PRO instruments based on patient interviews, expert opinion, and review of the literature. The number
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of items is then reduced in stage 2 according to expert opinion, item redundancy, endorsement frequency, missing data, factor analysis, and tests of scaling assumptions. This final instrument then undergoes psychometric evaluation and validation at stage 3, including determination of
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acceptability, internal consistency, reliability, and validity within scale, among others [22]. All publications that detailed instrument development and validation were reviewed to determine
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which aspects of each stage were completed to inform the final PRO instruments. Authors of each instrument were also contacted to confirm the process of development and validation.
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Finally, content domains covered by the included instruments were summarized.
Results
A search of existing literature identified 1666 articles for review, with another 14 identified
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through grey-literature and hand searching (Fig. 1). Following removal of duplications, 1219 articles were reviewed at a title/abstract level, and 245 were further examined at a full-text level.
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A total of 32 articles met the inclusion criteria and were included in the final analysis [10,17,2352]. Within these articles, five validated instruments that were developed in the hypospadias population and measured hypospadias patient-reported satisfaction and/or quality of life were identified (Table 1).
Of the included instruments, the genital perception scales (GPS) for children and adolescents (9-19 years) [28] and for adults (18-38 years) [28] were the first self-reported,
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standardized, validated instruments to be developed to assess genital perceptions of patients operated on for hypospadias. While the child/adolescent version consists of eight items investigating satisfaction with penile size, thickness, and general appearance; glans shape and
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size; meatus position; and testes/scrotum, the adult version consists of nine items (where
testes/scrotum satisfaction is replaced by penile color and position of erect penis) [28]. Both scales are intended for use by patients for self-reporting, and both showed strong internal
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consistency reliability (Cronbach’s alpha 0.89 and 0.86 for adult and child/adolescent,
description of psychometric tests).
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respectively) (Table 2) (Please see Cano et al. (2004) [21] or Voelzke et al. (2013) [53] for
The Hypospadias Objective Scoring Evaluation (HOSE) responds to the need for an impartial method to document the results of hypospadias surgery [31]. This is accomplished by individually scoring the surgical outcomes of meatal location and shape, urinary stream,
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straightness of erection, and presence of urethral fistula, where scoring the highest number indicates the most desirable outcome. Individual assessments are totaled, resulting in an overall score between 5 (minimum score) and 16 (maximum score), where ≥14 has been suggested as an
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acceptable surgical outcome [31]. The HOSE was validated for use by pediatric surgeons, nurses, and parents of patients for a pediatric population, and showed good inter-rater reliability between
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the three intended populations (mean weighted kappa 0.66) [31] (Table 2). To allow for the assessment and comparison of penile perception of patients, parents, and
surgeons following surgery, the Pediatric Penile Perception Score (PPPS) [43] and Penile Perception Score (PPS) [44] were developed. Both instruments use a 4-point Likert scale to evaluate satisfaction with penile appearance with regard to meatal positon and shape, glans shape, penile (shaft) skin, and general cosmetic appearance. The PPPS was designed for use by
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patients, parents, and urologists to evaluate a pediatric population, and it shows good inter-rater reliability (interclass correlation (ICC) 0.75-0.88) and test-retest reliability (r=0.59-0.83) [43] when used by urologists. The PPS was used by hypospadias patients >18 years of age and
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urologists, for whom the latter showed fair inter-rater reliability (ICC 0.46) and good test-retest reliability (0.78) [44]. Internal consistency reliability was satisfactory in the sample of
hypospadias patients for both instruments (Cronbach’s α=0.81 for PPPS [29] and 0.77 for PPS
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[30]) (Table 2).
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Discussion
While many hypospadias studies identified by the search strategy acknowledged the necessity of a patient-reported element, the majority used either ad hoc (instruments that are not formally developed or tested) or generic (broad based questionnaires that measure health-related QoL in a
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diverse population (e.g. International Index of Erectile Function (IIEF), Short Form-36)) instruments to collect this information. While ad hoc questionnaires might deliver useful relevant information, they are of limited use without proven reliability or validity. Without these
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psychometric analyses, one cannot be sure that measurements obtained from the instruments can be trusted. For example, it is impossible to know if the instrument is actually measuring what is
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intended without an assessment of validity, or if the instrument would produce the same results under consistent conditions without an assessment of reliability. Generic instruments are contrary to this, in that while many are psychometrically
validated, they may not capture all outcomes and domains specific to hypospadias [22]. For example, while the IIEF might be useful to assess some aspects of a hypospadias patient’s erectile function, it does not assess certain issues such as impact of persistent curvature, scarring
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or lack of sensation, which may have more of a significant impact on erectile function and sexual satisfaction in hypospadias patients. Further, they may not be responsive enough to measure
operative success in this patient population.
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changes as a result of surgery [54] and therefore are limited in their ability to determine post-
Overall, the five included instruments were similar in the domains they sought to assess (Table 3). This is not surprising, as the PPPS and PPS are the same instrument (but are intended
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for use in different populations), and the GPS for adults and for children/adolescents share seven items (representing 78% (n=7/9) and 88% (n=7/8) of total questions, respectively). Additionally,
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the focus of four of the instruments (PPPS, PPS, GPS adult, GPS child/adolescent) was to evaluate genital perception. Thus, the questions were very similar, as they all included assessment of satisfaction with position/shape of meatus, shape of glans, and general appearance of the penis. While both GPS instruments measured penile length and penile axis when erect,
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these items were removed from the PPPS, as authors felt that length is not a factor that is amendable to hypospadias repair and few pediatric patients were willing to demonstrate an erection for photographic documentation [43]. The HOSE utilized unique domains, as this was
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the only instrument to assess functional outcomes; while position/shape of meatus and penile axis when erect was evaluated, this was a functional assessment rather than an evaluation of
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satisfaction. The authors of the PPPS acknowledged that urinary flow could be added as a functional parameter, however this is suggested as a supplement to the instrument and not as a formal item [43]. While all five PRO instruments included some aspects of the three-stage system proposed by Cano et al. [21], important elements were missing from the development and validation of the instruments; for example, no instrument utilized patient interviews to generate
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items, even though this was identified by the SAC Medical Outcomes Trust as one of the best ways to generate content for newly developed self-reporting instruments [20]. Repeated interviews of patients allows for determining recurring priorities and concepts
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that are most significant and relevant to a patient’s conditions and treatment. With patient input it is possible to generate items and domains that are comprehensive with respect to patient
concerns [55], which otherwise might not have been evident to an outside observer. Further, for
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the condition of hypospadias it is essential to gather information from parents of patients, as they are often the primary decision makers concerning surgical repair (where the American Academy
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of Pediatrics suggests an optimal age of 6-12 months for genital surgery [56]). They will also be the main utilizers of hypospadias–specific PROs until the patient is able to reflect on their own condition (preferably at 12 years, although child-report is possible at 5 years [57]). Given parent’s valuable understanding of their children based on prior important experiences [57], the
compared over time.
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ideal instrument would contain both patient and parent identified items, which could be
Included instruments also showed no evidence of the use of a conceptual model to
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determine appropriate domains for the instruments. Conceptual frameworks allow for visualization of the relationships between overarching concepts, domains/subdomains, and item
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content [55], and as such are indispensable for determining the structure of the PRO instrument. Additionally, there was limited formal item reduction in included instruments, with only the GPS using factor analysis for item reduction and classification [28]. The PPPS also reported the exclusion of two items that were too difficult to collect or were not relevant for the evaluation of hypospadias repair. Item reduction is essential in finalizing instrument development, as it
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excludes items with weak psychometric properties [20,21] that would otherwise add to the overall burden of the instrument. Finally, psychometric evaluation of the instruments was sub-optimal, with acceptability,
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item total correlations, all measures of validity, and responsiveness being overlooked. Each of these measures provides important information about the instruments, including data quality (acceptability), item consistency (item total correlation), the instrument’s ability to measure what
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it is designed to measure (validity), and its ability to detect change over time (responsiveness). By overlooking these psychometric analyses and not fully evaluating the reliability and validity
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of the instrument, it is unclear is the patient, parent, or healthcare provider-reported hypospadias outcomes obtained by the instrument are trustworthy.
It is also interesting to note that the majority of included instruments were designed and validated for hypospadias patients who had undergone surgery (HOSE, PPS, and PPPS). Surgical
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treatment is not always necessary in patients with mild hypospadias, and therefore these instruments cannot be used with a high degree of confidence for the self-evaluation of the subset of patients who chose to forgo surgery. Instruments that allow for the self-evaluation both before
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and after surgery would be of use, and could be utilized to help make a decision as to whether surgery is the best course of action. Additionally, existing instruments failed to evaluate sexual
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function and psychosocial sequelae of patients with hypospadias. While studies on psychosocial functioning are limited, several comparative studies have indicated that hypospadias patients are at risk for behavioral and emotional problems when compared with the general population. For example, children with hypospadias have been shown to have a lower social competence [58] and are generally more timid and isolated compared with their healthy peers, while adults have lower self-esteem, a decreased capacity for interpersonal social and emotional relationships, and
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are generally more fearful and socially isolated [59,60]. Concerning sexual functioning, both children and adolescents with hypospadias self-report more sexual inhibitions compared with healthy age-matched controls. This included fear of rejection by sexual partners, and an
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increased disposition to hide genitals in public washrooms [61,62]. A high-quality, sensitive PRO must examine all elements that are important to a patient’s condition and treatment, and it is becoming increasingly evident that this should include an assessment of sexual functioning
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and psychosocial sequelae.
Finally, it is acknowledged that a number of well-known, validated, objective
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hypospadias-specific instruments such as HOPE (Hypospadias Objective Penile Evaluation) [63] and GMS (Glans-Urethral Meatus-Shaft) [64] were not included as, while they are valuable for physician assessment, they do not reflect the patient’s perspective. Rather GMS is utilized by physicians to described hypospadias severity [65], and is traditionally utilized pre-operatively.
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HOPE has been validated for use by pediatric urologists to assess cosmetic outcome in hypospadias patients [63]. While HOPE is currently being used in a parental questionnaire for a Dutch national registry (with an additional question concerning satisfaction with general
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cosmetic appearance), it is not yet validated for patients/parents, and has yet to produce any publications with this population. The authors of this paper acknowledge the importance of
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objective assessment of hypospadias outcomes and recommend that they continue to be used along with the subjective patient-reported outcomes.
Limitations Limitations and future directions There were several limitations to this systematic review. Only English-language studies were included, and as such it is possible that validated hypospadias-specific PRO instruments published in other languages are missing from the analysis. It is also possible that in excluding 12
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other languages, outcomes that are culturally significant (e.g. satisfaction following hypospadias surgery resulting in circumcision in cultures where circumcision is preferred) were missing. The review was also limited by the overall lack of information provided concerning instrument
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development. This necessitated contacting the authors of numerous publications to determine if the instrument met inclusion criteria, and to assess validation of included instruments. In the future, it is recommended that researchers follow the gold standard, three-stage system proposed
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by Cano et al. [21] for the development of PROs. This will ensure that all necessary elements are included in one PRO, which could then be utilized by all members of the hypospadias
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population.
Conclusions
While many hypospadias studies have acknowledged the necessity of a patient-reported element,
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few have used validated instruments developed in a hypospadias population. The five instruments that met inclusion criteria showed limited adherence to the gold standard methodology described by Cano et al. [21], based on the SAC of Medical Outcomes Trust
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international guidelines for the development and validation of health outcome measures [20]. Further, the majority of identified instruments focused on postoperative cosmetic satisfaction,
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with only one instrument considering urinary function, and no instruments evaluating sexual function and psychosocial sequelae. Future PRO instruments designed for hypospadias patients should evaluate all facets of the patient experience and should ideally undergo full development and validation, as outlined by the SAC of the Medical Outcomes Trust [20].
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Funding This research did not receive any specific grant from funding agencies in the public, commercial,
Conflict of Interest Statement
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The authors have no conflicts of interest to declare.
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or not-for-profit sectors.
Ethics
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Ethics were not required given the study design (systematic review).
Acknowledgements
We thank Margaret Sampson, MLIS, PhD, AHIP (Children’s Hospital of Eastern Ontario) for
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developing the electronic search strategies, and Linda Slater, MLIS (John W. Scott Health Sciences Library, University of Alberta) for peer review of the MEDLINE search strategy. We would also like to thank Dr. Nick Barrowman for his help with statistical evaluation of
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instruments.
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[38] Schonbucher VB, Landolt MA, Gobet R, Weber DM. Psychosexual development of children and adolescents with hypospadias. Journal of Sexual Medicine 2008;5:1365-73. [39] Dodds PR. Re: The pediatric penile perception score: an instrument for patient selfassessment and surgeon evaluation after hypospadias repair: D.M. Weber, V.B. Schonbucher, M.A. Landolt, R. Gobet. J Urol 2008; 180: 1080-4. J Urol 2009;181:1506. [40] Morel-Journel N, Courtois F, Paparel P, Ruffion A, Carrier S, Leriche A. Reconstructive surgery for major sexual congenital anomalies in adults. Sexologies 2009;18:98-104. [41] van der Toorn F. Response to Letter to the Editor RE: Introducing the HOPE (Hypospadias Objective Penile Evaluation)-score: A validation study of an objective scoring system for evaluating cosmetic appearance in hypospadias patients. J Pediatr Urol 2013;9:1017. 17
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[42] Ghavami-Adel M, Mollaeean M, Hooman N. The cosmetic results of a simple method for repairing preputial skin defects in hypospadias. Iranian Journal of Pediatrics 2014;24:406-10.
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[43] Weber DM, Schonbucher VB, Landolt MA, Gobet R. The Pediatric Penile Perception Score: an instrument for patient self-assessment and surgeon evaluation after hypospadias repair. J Urol 2008;180:1080-4. [44] Weber DM, Landolt MA, Gobet R, Kalisch M, Greeff NK. The Penile Perception Score: an instrument enabling evaluation by surgeons and patient self-assessment after hypospadias repair. J Urol 2013;189:189-93.
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[45] Deibert CM, Hensle TW. The psychosexual aspects of hypospadias repair: A review. J Urol 2011;9:279-82.
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[46] Rynja SP, de Kort L, de Jong TPVM. Urinary, sexual, and cosmetic results after puberty in hypospadias repair: current results and trends. Current Opinions in Urology 2012;22:453-6. [47] Woodhouse CRJ, Christie D. Non-surgical factors in the success of hypospadias repair. BJU International 2005;96:22-7. [48] Liu MMY, Holland AJA, Cass DT. Assessment of postoperative outcomes of hypospadias repair with validated questionnaires. Journal of Pediatric Surgery 2015;50:2071-4.
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[49] Ciancio F, Russo GL, Innocenti A, Portincasa A, Parisi D, Mondaini N. Penile length is a very important factor for cosmesis, function and psychosexual development in patients affected by hypospadias: Results from a long-term longitudinal cohort study. International Journal of Immunopathology and Pharmacology 2015;28:421-5.
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[50] Haid B, Becker T, Koen M, Berger C, Strasser C, Roesch J, et al. Penile appearance after hypospadias correction from a parent's point of view: Comparison of the hypospadias objective penile evaluation score and parents penile perception score. J Pediatr Urol 2016;12:33.e1-33.e7. [51] Örtqvist L, Fossum M, Andersson M, Nordenström A, Frisén L, Holmdahl G, et al. Long-term follow up of men born with hypospadias: Urological and cosmetic results. J Urol 2015;193:975-82. [52] Thiry S, Saussez T, Dormeus S, Tombal B, Wese FX, Feyaerts A. Long-term functional, cosmetic and sexual outcomes of hypospadias correction performed in childhood. Urologia Internationalis 2015;95:137-41. [53] Voelzke BB. Critical review of existing patient reported outcome measures after male anterior urethroplasty. J Urol 2013;189:182-8.
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[54] Velanovich V. Experience with a generic quality of life instrument in a general surgery practice. International Journal of Surgical Investigation 2000;1:447-52.
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[55] Patrick DL, Burke LB, Gwaltney CJ, Klein Leidy N, Martin ML, Molsen E, et al. Content validity - establishing and reporting the evidence in newly developed patientreported outcomes (PRO) instruments for medical product evaluation: ISPOR PRO good research practices task force report: Part 2 - Assessing respondent understanding. Value in Health 2011;14:978-88. [56] Kass E, Kogan SJ, Mainley C. Timing of the elective surgery on the genitalia of male children with particular reference to risks, benefits, psychological effects of surgery and anesthesia. Pediatrics 1996;97:590-4.
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[57] Matza LS, Patrick DL, Riley AW, Alexander JJ, Rajmil L, Pleil AM, et al. Pediatric patient-reported outcome instrument for research to support medical product labelling report of the ISPOR PRO good research practices for the assessment of children and adolescent task force. Value in Health 2013;16:461-79. [58] Sandberg DE, Meyer-Bahlburg HFL, Aranoff GS, Sconzo JM, Hensle TW. Boys with hypospadias: A survey of behavioral difficulties. Journal of Pediatric Psychology 1989;14:491-514. [59] Berg G, Berg R. Castration complex. Evidence from men operated for hypospadias. Acta Psychiatrica Scandinavica 1983;68:143-53.
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[60] Berg R, Berg G, Svensson J. Penile malformation and mental health. A controlled psychiatric study of men operated for hypospadias in childhood. Acta Psychiatrica Scandinavica 1982;66:398-416.
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[61] Mondaini N, Ponchietti R, Bonafe M, Biscioni S, Di Loro F, Agostini P, et al. Hypospadias: Incidence and effects on psychosexual development as evaluated with the Minnesota Multiphasic Personality Inventory test in a sample of 11,649 young Italian men. Urologia Internationalis 2002;68:81-5.
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[62] Mureau MA, Slijper FM, Nijman RJ, van der Meulen JC, Verhulst FC, Slob AK. Psychosexual adjustment of children and adolescents after different types of hypospadias surgery: A norm-related study. J Urol 1995;154:1902-7. [63] van der Toorn F, de Jong TPVM, de Gier RP, Callewaert PR, van der Horst EH, Steffens MG, et al. Introducing the HOPE (Hypospadias Objective Penile Evaluation) score: a validation study of an objective scoring system for evaluating cosmetic appearance in hypospadias patients. J Pediatr Urol 2013;9:1006-16. [64] Merriman LS, Arlen AM, Broecker BH, Smith EA, Kirsch AJ, Elmore JM. The GMS hypospadias score: assessment of inter-observer reliability and correlation with postoperative complications. J Pediatr Urol 2013;9:707-12.
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[65] Arlen AM, Kirsch AJ, Leong T, Broecker BH, Smith EA, Elmore JM. Further analysis of the Glans-Urethral Meatus-Shaft (GMS) hypospadias score: Correlation with postoperative complications. J Pediatr Urol 2015;11:71.e1-71.e5.
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[66] Rynja SP, de Kort L, de Jong TPVM. Urinary, sexual, and cosmetic results after puberty in hypospadias repair: current results and trends. Current Opinions in Urology 2012;22:453-6. [67] Dodds PR. Re: The pediatric penile perception score: an instrument for patient selfassessment and surgeon evaluation after hypospadias repair: D.M. Weber, V.B. Schonbucher, M.A. Landolt, R. Gobet. J Urol 2008; 180: 1080-4. J Urol 2009;181:1506.
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[68] Fasching G, Arneitz C, Gritsch-Olipp G. Foreskin reconstruction and preservation of a thin distal urethra: a challenge in tubularized incised plate urethroplasty. Pediatric Surgery International 2011;27:755-60.
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[69] Schonbucher VB, Landolt MA, Gobet R, Weber DM. Health-related quality of life and psychological adjustment of children and adolescents with hypospadias. Journal of Pediatrics 2008;152:865-72. [70] Ruppen-Greeff NK, Weber DM, Gobet R, Landolt MA. Health-related quality of life in men with corrected hypospadias: An explorative study. J Pediatr Urol 2013;9:551-8.
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[71] Örtqvist L, Fossum M, Andersson M, Nordenström A, Frisén L, Holmdahl G, et al. Long-term follow-up of men born with hypospadias: Urological and cosmetic results. J Urol 2015;193:975-82.
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Figure Legends:
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Fig. 1. PRISMA [43] flow chart.
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Table 1. Validated instruments used to measure quality of life/satisfaction of patients with hypospadias. Studies describing instrument Springer 2014 [23] Rynja et al. 2011 [27] Rynja et al. 2012 [66]
Holland et al. 2001 [31] Aulagne et al. 2010 [34] Seibold et al. 2010 [35] Liu et al. 2015 [48] Thiry et al. 2015 [52]
Pediatric Penile Perception Score (PPPS)
Springer 2014 [23] Rynja et al. 2011 [27] Kurzroc 2008 [10] Dodds 2009 [67] Morel-Journel et al. 2009 [40] van der Toorn et al. 2013 [41] Deibert et al. 2011 [45]
Penile Perception Score (PPS)
van der Toorn 2013 [41]
Fasching et al. 2011 [68] Schönbucher et al. 2008 [38] Weber et al. 2009 [33] Castagnetti et al. 2013 [37] Schönbucher et al. 2008 [69] Ghavami-Adel et al. 2014 [42] Weber et al. 2008 [43] Haid et al. 2016 [50] Thiry et al. 2015 [52]
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Hypospadias Objective Scoring Evaluation (HOSE)
Studies using instrument
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Instrument
Schlomer et al. 2014 [25] Ruppen-Greeff et al. 2013 [70] Weber et al. 2013 [44] Liu et al. 2015 [48] Ciancio et al. 2015 [49] Örtqvist et al. 2015 [71]
Mieusset et al. 2005 [32] Ringert et al. 1999 [36] Woodhouse et al. 2005 [47]
Mureau et al. 1995 [28] Mureau et al. 1997 [17]
Genital Perception Scale (GPS) for children/ Junior Genital Perception Scale
Springer 2014 [23] Rynja et al. 2011 [27] Mieusset et al. 2005 [32] Woodhouse et al. 2005 [47]
Vandendriessche et al. 2010 [24] Mureau et al. 1995 [28] Mureau et al. 1997 [17]
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Genital Perception Scale (GPS) for adults
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Table 2. Development and validation of instruments.
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Penile Perception Score (PPS)
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Patient interviews Literature Expert opinion Develop conceptual model Item reduction Expert opinion Item redundancy Endorsement frequencies Missing data Factor analysis Tests of scaling assumptions Psychometric Acceptability analyses Internal consistency reliability Item total correlations Interrater reliability Test-retest reliability Validity within scale Validity comparison with other measures Validity hypothesis testing Responsiveness
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Item generation
Pediatric Penile Perception Score (PPPS)
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Method/Evaluation
Hypospadias Objective Scoring Evaluation (HOSE)
Genital Perception Scale (GPS) for adults
Genital Perception Scale (GPS) for children and adolescents/Junior Genital Perception Scale
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Table 3. Domain analysis for patient-reported hypospadias outcomes instruments.
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Functional evaluation
Position and/or shape of meatus Shape of the glans Size of the glans Shape of penile skin Penile length (flaccid or erect) Penile axis when erect Penile thickness Penile colour General appearance of penis Meatal location Meatal shape Penile axis when erect Urinary stream Fistula
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Method/Evaluation
Genital satisfaction
Penile Perception Score (PPS)
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Hypospadias Pediatric Objective Penile Scoring Perception Evaluation Score (HOSE) (PPPS)
Genital Perception Scale (GPS) for children and adolescents/Junior Genital Perception Scale
Genital Perception Scale (GPS) for adults
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S1477-5131(16)30387-4
DOI:
10.1016/j.jpurol.2016.11.010
Reference:
JPUROL 2393
To appear in:
Journal of Pediatric Urology
Received Date: 4 August 2016 Accepted Date: 8 November 2016
Please cite this article as: Sullivan KJ, Hunter Z, Andrioli V, Guerra L, Leonard M, Klassen A, Keays M, Assessing quality of life of patients with hypospadias: a systematic review of validated patient-reported outcome instruments, Journal of Pediatric Urology (2017), doi: 10.1016/j.jpurol.2016.11.010. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
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Assessing quality of life of patients with hypospadias: a systematic review of validated patient-reported outcome instruments
Department of Pediatric Surgery, Division of Urology, Children’s Hospital of Eastern Ontario,
401 Smyth Road, Ottawa, ON, Canada b
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K. J. Sullivana, Z. Hunterb, V. Andriolia, L. Guerraa,b, M. Leonarda,b, A. Klassenc, M. Keaysa,b,*
Faculty of Medicine, University of Ottawa, 451 Smyth Rd, Ottawa, ON, Canada
Departments of Pediatrics and Clinical Epidemiology & Biostatistics, McMaster University,
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1280 Main Street West, Hamilton, ON, Canada
*Corresponding author: Children’s Hospital of Eastern Ontario, 401 Smyth Road, Ottawa, ON,
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K1H 8L1, Canada. Tel.: (613) 737-7600 ext. 3864; fax: (613) 738-4271.
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E-mail address: [email protected] (Dr. Mélise Keays)
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Summary Background: Patient-reported outcomes have the potential to provide invaluable information for
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evaluation of hypospadias patients, aid in decision-making, performance assessment, and improvement in quality of care. To appropriately measure patient-relevant outcomes, welldeveloped and validated patient-reported outcome (PRO) instruments are essential.
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Objective: To identify and evaluate existing PRO instruments designed to measure quality of life and/or satisfaction of individuals with hypospadias that have been developed and validated in
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a hypospadias population.
Methods: A systematic search of MEDLINE, EMBASE, PsycINFO, CINAHL and Health and Psychosocial Instruments was conducted in April 2016. Two reviewers independently assessed studies and identified PRO instruments for inclusion. Data were extracted on study characteristics, instrument development and validation, and content domains.
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Results: A total of 32 studies were included that used or described five PRO instruments: Hypospadias Objective Scoring Evaluation (HOSE), Pediatric Penile Perception Score (PPPS),
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Penile Perception Score (PPS), Genital Perception Scale (GPS) for adults, and GPS for children/adolescents. Instrument development and validation was limited. The majority of
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identified instruments focused on postoperative cosmetic satisfaction, with only one instrument considering urinary function, and no instruments evaluating sexual function and psychosocial sequelae.
Conclusions: While many hypospadias studies have acknowledged the necessity of a patientreported element, few have used validated PRO instruments developed in a hypospadias population. Existing instruments to measure patient-reported outcomes in hypospadias require
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improvement in both the breadth of content and in their development and validation methodology.
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Keywords: Hypospadias; Patient-reported outcome (PRO); Satisfaction; Quality of life
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Introduction Patient-reported outcome (PRO) instruments are questionnaires/instruments that allow for self-
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reporting of the patient (or parent-proxy) experience, potentially including views of their
symptoms, functional status, and health-related quality of life (QoL) [1]. While originally
designed for use in research [1], PRO instruments have been adopted by healthcare professionals
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to support various clinical efforts, including quality improvement, performance assessment, and the tailoring of treatment plans to meet patient preferences and needs [2]. The latter is of
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particular importance, as healthcare professionals frequently misjudge the absolute levels of symptoms and general QoL, tending to underestimate the impact of psychological factors while emphasizing more obvious symptoms [3]. Jachuck et al. [4] observed this in their questionnaire study of hypertensive patients and their doctors, where all physicians indicated that patients had
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improved, while approximately half of the patients felt that there was no change or even deterioration. Physicians tended to ignore the factors that patients factored into their overall wellbeing, including a decline in energy, general activity, sexual inactivity, and irritability [4].
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Patient-reported outcomes are particularly useful for conditions where a large variation in care and outcomes exists, and where the impact on the patient’s QoL is currently unknown. Such
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is the case for the congenital condition of hypospadias. While the goals of hypospadias repair are generally agreed upon – including providing the patient with satisfactory urinary function, sexual function, body image or cosmesis, and quality of life – several important variations remain. First, there is variation in surgical techniques for the same condition; for example, the use of one-stage vs. two-stage repair for proximal hypospadias [5]. While advantages have been established for each technique, the current evidence base cannot definitively identify an optimal method for
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individual patients [6]. Patient input, obtained through PROs, on the relative benefits may help elucidate this (e.g. does the reduced hospital stay and anesthetic risk associated with a one-stage repair offset the increased risk of complications [5,7] compared with a two-stage repair?).
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Additional variance also exists concerning physician recommendations and parental preference for surgical correction of distal hypospadias (i.e. glanular), where some routinely
elect/recommend surgical repair and others prefer to forgo surgery in mild cases. Finally,
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variance can be observed in surgical success, where 15% of patients experience a complication (a value that at least doubles in patients with severe defects or prior complications) [7-9].
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While it remains important to measure these results using traditional surgical outcomes, such as complications and need for reoperation, these no longer sufficiently capture all important aspects from the patient’s perspective [10-12]. Rather, there is a need to capture the considerable long-term cognitive, behavioral, and self-esteem consequences that result from poor cosmetic or
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functional outcomes – such as negative genital perception [13,14], sexual avoidance [15,16], and poor school performance [17] – through the use of PRO measures. Comprehensive measurement of surgical outcomes requires a combination of objective and subjective measures.
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Clinical outcomes research in hypospadias surgery is becoming increasingly focused on the patient’s QoL and their perception of a satisfactory outcome [10]. As a result, the present
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systemic review of the published literature was conducted to identify and assess PRO instruments currently available for hypospadias patients or parent-proxies. The primary objective was to identify existing PRO instruments that have been developed and validated in a hypospadias population that assess patient satisfaction and/or quality of life. A secondary objective was to evaluate the development and validation of the instruments, and to assess the content of identified instruments.
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Methods Inclusion criteria Studies were included that described PRO instruments designed to measure quality of life and/or
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satisfaction of individuals with hypospadias or their parent-proxy that had been developed and validated in a hypospadias population. Non-English language studies were excluded, as were
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conference abstracts.
Literature search
The following databases were searched: MEDLINE including In-Process & Other Non-Indexed
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Citations (1946-April 8 2016), EMBASE (1980-week 14 2016), PsycINFO (1806-April week 1 2016), CINAHL (April 8 2016), and Health and Psychosocial Instruments (1985-January 2016). The MEDLINE search strategy was developed by a librarian experienced in systematic review searching, and peer reviewed by a second librarian using the PRESS standard [18]. The
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MEDLINE search was then adapted to allow for optimal searching of other included databases. Search strategies are presented in the Appendix. Hand-searching the reference sections of
Screening
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relevant articles identified additional publications.
At level 1 (title and abstract), studies were screened independently in duplicate using the liberal
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accelerated method [19]. Two researchers then independently assessed full-text articles (level 2 screening) and compared decisions to reach consensus for final inclusion. Disagreements were resolved by discussion or third party consultation when necessary.
Development Development and validation criteria criteria Validated instruments were identified from included studies and were evaluated for their adherence to the three-stage, rigorous, gold standard methodology for the development and validation of health outcome measures developed by the Scientific Advisory Committee (SAC) 5
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of Medical Outcomes Trust International guidelines [20] and described by Cano et al. [21]. According to this methodology, stage 1 involves generating a list of items for the PRO instruments based on patient interviews, expert opinion, and review of the literature. The number
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of items is then reduced in stage 2 according to expert opinion, item redundancy, endorsement frequency, missing data, factor analysis, and tests of scaling assumptions. This final instrument then undergoes psychometric evaluation and validation at stage 3, including determination of
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acceptability, internal consistency, reliability, and validity within scale, among others [22]. All publications that detailed instrument development and validation were reviewed to determine
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which aspects of each stage were completed to inform the final PRO instruments. Authors of each instrument were also contacted to confirm the process of development and validation.
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Finally, content domains covered by the included instruments were summarized.
Results
A search of existing literature identified 1666 articles for review, with another 14 identified
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through grey-literature and hand searching (Fig. 1). Following removal of duplications, 1219 articles were reviewed at a title/abstract level, and 245 were further examined at a full-text level.
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A total of 32 articles met the inclusion criteria and were included in the final analysis [10,17,2352]. Within these articles, five validated instruments that were developed in the hypospadias population and measured hypospadias patient-reported satisfaction and/or quality of life were identified (Table 1).
Of the included instruments, the genital perception scales (GPS) for children and adolescents (9-19 years) [28] and for adults (18-38 years) [28] were the first self-reported,
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standardized, validated instruments to be developed to assess genital perceptions of patients operated on for hypospadias. While the child/adolescent version consists of eight items investigating satisfaction with penile size, thickness, and general appearance; glans shape and
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size; meatus position; and testes/scrotum, the adult version consists of nine items (where
testes/scrotum satisfaction is replaced by penile color and position of erect penis) [28]. Both scales are intended for use by patients for self-reporting, and both showed strong internal
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consistency reliability (Cronbach’s alpha 0.89 and 0.86 for adult and child/adolescent,
description of psychometric tests).
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respectively) (Table 2) (Please see Cano et al. (2004) [21] or Voelzke et al. (2013) [53] for
The Hypospadias Objective Scoring Evaluation (HOSE) responds to the need for an impartial method to document the results of hypospadias surgery [31]. This is accomplished by individually scoring the surgical outcomes of meatal location and shape, urinary stream,
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straightness of erection, and presence of urethral fistula, where scoring the highest number indicates the most desirable outcome. Individual assessments are totaled, resulting in an overall score between 5 (minimum score) and 16 (maximum score), where ≥14 has been suggested as an
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acceptable surgical outcome [31]. The HOSE was validated for use by pediatric surgeons, nurses, and parents of patients for a pediatric population, and showed good inter-rater reliability between
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the three intended populations (mean weighted kappa 0.66) [31] (Table 2). To allow for the assessment and comparison of penile perception of patients, parents, and
surgeons following surgery, the Pediatric Penile Perception Score (PPPS) [43] and Penile Perception Score (PPS) [44] were developed. Both instruments use a 4-point Likert scale to evaluate satisfaction with penile appearance with regard to meatal positon and shape, glans shape, penile (shaft) skin, and general cosmetic appearance. The PPPS was designed for use by
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patients, parents, and urologists to evaluate a pediatric population, and it shows good inter-rater reliability (interclass correlation (ICC) 0.75-0.88) and test-retest reliability (r=0.59-0.83) [43] when used by urologists. The PPS was used by hypospadias patients >18 years of age and
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urologists, for whom the latter showed fair inter-rater reliability (ICC 0.46) and good test-retest reliability (0.78) [44]. Internal consistency reliability was satisfactory in the sample of
hypospadias patients for both instruments (Cronbach’s α=0.81 for PPPS [29] and 0.77 for PPS
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[30]) (Table 2).
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Discussion
While many hypospadias studies identified by the search strategy acknowledged the necessity of a patient-reported element, the majority used either ad hoc (instruments that are not formally developed or tested) or generic (broad based questionnaires that measure health-related QoL in a
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diverse population (e.g. International Index of Erectile Function (IIEF), Short Form-36)) instruments to collect this information. While ad hoc questionnaires might deliver useful relevant information, they are of limited use without proven reliability or validity. Without these
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psychometric analyses, one cannot be sure that measurements obtained from the instruments can be trusted. For example, it is impossible to know if the instrument is actually measuring what is
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intended without an assessment of validity, or if the instrument would produce the same results under consistent conditions without an assessment of reliability. Generic instruments are contrary to this, in that while many are psychometrically
validated, they may not capture all outcomes and domains specific to hypospadias [22]. For example, while the IIEF might be useful to assess some aspects of a hypospadias patient’s erectile function, it does not assess certain issues such as impact of persistent curvature, scarring
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or lack of sensation, which may have more of a significant impact on erectile function and sexual satisfaction in hypospadias patients. Further, they may not be responsive enough to measure
operative success in this patient population.
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changes as a result of surgery [54] and therefore are limited in their ability to determine post-
Overall, the five included instruments were similar in the domains they sought to assess (Table 3). This is not surprising, as the PPPS and PPS are the same instrument (but are intended
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for use in different populations), and the GPS for adults and for children/adolescents share seven items (representing 78% (n=7/9) and 88% (n=7/8) of total questions, respectively). Additionally,
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the focus of four of the instruments (PPPS, PPS, GPS adult, GPS child/adolescent) was to evaluate genital perception. Thus, the questions were very similar, as they all included assessment of satisfaction with position/shape of meatus, shape of glans, and general appearance of the penis. While both GPS instruments measured penile length and penile axis when erect,
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these items were removed from the PPPS, as authors felt that length is not a factor that is amendable to hypospadias repair and few pediatric patients were willing to demonstrate an erection for photographic documentation [43]. The HOSE utilized unique domains, as this was
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the only instrument to assess functional outcomes; while position/shape of meatus and penile axis when erect was evaluated, this was a functional assessment rather than an evaluation of
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satisfaction. The authors of the PPPS acknowledged that urinary flow could be added as a functional parameter, however this is suggested as a supplement to the instrument and not as a formal item [43]. While all five PRO instruments included some aspects of the three-stage system proposed by Cano et al. [21], important elements were missing from the development and validation of the instruments; for example, no instrument utilized patient interviews to generate
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items, even though this was identified by the SAC Medical Outcomes Trust as one of the best ways to generate content for newly developed self-reporting instruments [20]. Repeated interviews of patients allows for determining recurring priorities and concepts
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that are most significant and relevant to a patient’s conditions and treatment. With patient input it is possible to generate items and domains that are comprehensive with respect to patient
concerns [55], which otherwise might not have been evident to an outside observer. Further, for
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the condition of hypospadias it is essential to gather information from parents of patients, as they are often the primary decision makers concerning surgical repair (where the American Academy
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of Pediatrics suggests an optimal age of 6-12 months for genital surgery [56]). They will also be the main utilizers of hypospadias–specific PROs until the patient is able to reflect on their own condition (preferably at 12 years, although child-report is possible at 5 years [57]). Given parent’s valuable understanding of their children based on prior important experiences [57], the
compared over time.
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ideal instrument would contain both patient and parent identified items, which could be
Included instruments also showed no evidence of the use of a conceptual model to
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determine appropriate domains for the instruments. Conceptual frameworks allow for visualization of the relationships between overarching concepts, domains/subdomains, and item
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content [55], and as such are indispensable for determining the structure of the PRO instrument. Additionally, there was limited formal item reduction in included instruments, with only the GPS using factor analysis for item reduction and classification [28]. The PPPS also reported the exclusion of two items that were too difficult to collect or were not relevant for the evaluation of hypospadias repair. Item reduction is essential in finalizing instrument development, as it
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excludes items with weak psychometric properties [20,21] that would otherwise add to the overall burden of the instrument. Finally, psychometric evaluation of the instruments was sub-optimal, with acceptability,
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item total correlations, all measures of validity, and responsiveness being overlooked. Each of these measures provides important information about the instruments, including data quality (acceptability), item consistency (item total correlation), the instrument’s ability to measure what
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it is designed to measure (validity), and its ability to detect change over time (responsiveness). By overlooking these psychometric analyses and not fully evaluating the reliability and validity
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of the instrument, it is unclear is the patient, parent, or healthcare provider-reported hypospadias outcomes obtained by the instrument are trustworthy.
It is also interesting to note that the majority of included instruments were designed and validated for hypospadias patients who had undergone surgery (HOSE, PPS, and PPPS). Surgical
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treatment is not always necessary in patients with mild hypospadias, and therefore these instruments cannot be used with a high degree of confidence for the self-evaluation of the subset of patients who chose to forgo surgery. Instruments that allow for the self-evaluation both before
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and after surgery would be of use, and could be utilized to help make a decision as to whether surgery is the best course of action. Additionally, existing instruments failed to evaluate sexual
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function and psychosocial sequelae of patients with hypospadias. While studies on psychosocial functioning are limited, several comparative studies have indicated that hypospadias patients are at risk for behavioral and emotional problems when compared with the general population. For example, children with hypospadias have been shown to have a lower social competence [58] and are generally more timid and isolated compared with their healthy peers, while adults have lower self-esteem, a decreased capacity for interpersonal social and emotional relationships, and
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are generally more fearful and socially isolated [59,60]. Concerning sexual functioning, both children and adolescents with hypospadias self-report more sexual inhibitions compared with healthy age-matched controls. This included fear of rejection by sexual partners, and an
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increased disposition to hide genitals in public washrooms [61,62]. A high-quality, sensitive PRO must examine all elements that are important to a patient’s condition and treatment, and it is becoming increasingly evident that this should include an assessment of sexual functioning
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and psychosocial sequelae.
Finally, it is acknowledged that a number of well-known, validated, objective
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hypospadias-specific instruments such as HOPE (Hypospadias Objective Penile Evaluation) [63] and GMS (Glans-Urethral Meatus-Shaft) [64] were not included as, while they are valuable for physician assessment, they do not reflect the patient’s perspective. Rather GMS is utilized by physicians to described hypospadias severity [65], and is traditionally utilized pre-operatively.
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HOPE has been validated for use by pediatric urologists to assess cosmetic outcome in hypospadias patients [63]. While HOPE is currently being used in a parental questionnaire for a Dutch national registry (with an additional question concerning satisfaction with general
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cosmetic appearance), it is not yet validated for patients/parents, and has yet to produce any publications with this population. The authors of this paper acknowledge the importance of
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objective assessment of hypospadias outcomes and recommend that they continue to be used along with the subjective patient-reported outcomes.
Limitations Limitations and future directions There were several limitations to this systematic review. Only English-language studies were included, and as such it is possible that validated hypospadias-specific PRO instruments published in other languages are missing from the analysis. It is also possible that in excluding 12
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other languages, outcomes that are culturally significant (e.g. satisfaction following hypospadias surgery resulting in circumcision in cultures where circumcision is preferred) were missing. The review was also limited by the overall lack of information provided concerning instrument
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development. This necessitated contacting the authors of numerous publications to determine if the instrument met inclusion criteria, and to assess validation of included instruments. In the future, it is recommended that researchers follow the gold standard, three-stage system proposed
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by Cano et al. [21] for the development of PROs. This will ensure that all necessary elements are included in one PRO, which could then be utilized by all members of the hypospadias
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population.
Conclusions
While many hypospadias studies have acknowledged the necessity of a patient-reported element,
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few have used validated instruments developed in a hypospadias population. The five instruments that met inclusion criteria showed limited adherence to the gold standard methodology described by Cano et al. [21], based on the SAC of Medical Outcomes Trust
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international guidelines for the development and validation of health outcome measures [20]. Further, the majority of identified instruments focused on postoperative cosmetic satisfaction,
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with only one instrument considering urinary function, and no instruments evaluating sexual function and psychosocial sequelae. Future PRO instruments designed for hypospadias patients should evaluate all facets of the patient experience and should ideally undergo full development and validation, as outlined by the SAC of the Medical Outcomes Trust [20].
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Funding This research did not receive any specific grant from funding agencies in the public, commercial,
Conflict of Interest Statement
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The authors have no conflicts of interest to declare.
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or not-for-profit sectors.
Ethics
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Ethics were not required given the study design (systematic review).
Acknowledgements
We thank Margaret Sampson, MLIS, PhD, AHIP (Children’s Hospital of Eastern Ontario) for
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developing the electronic search strategies, and Linda Slater, MLIS (John W. Scott Health Sciences Library, University of Alberta) for peer review of the MEDLINE search strategy. We would also like to thank Dr. Nick Barrowman for his help with statistical evaluation of
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instruments.
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[42] Ghavami-Adel M, Mollaeean M, Hooman N. The cosmetic results of a simple method for repairing preputial skin defects in hypospadias. Iranian Journal of Pediatrics 2014;24:406-10.
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[54] Velanovich V. Experience with a generic quality of life instrument in a general surgery practice. International Journal of Surgical Investigation 2000;1:447-52.
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[55] Patrick DL, Burke LB, Gwaltney CJ, Klein Leidy N, Martin ML, Molsen E, et al. Content validity - establishing and reporting the evidence in newly developed patientreported outcomes (PRO) instruments for medical product evaluation: ISPOR PRO good research practices task force report: Part 2 - Assessing respondent understanding. Value in Health 2011;14:978-88. [56] Kass E, Kogan SJ, Mainley C. Timing of the elective surgery on the genitalia of male children with particular reference to risks, benefits, psychological effects of surgery and anesthesia. Pediatrics 1996;97:590-4.
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[57] Matza LS, Patrick DL, Riley AW, Alexander JJ, Rajmil L, Pleil AM, et al. Pediatric patient-reported outcome instrument for research to support medical product labelling report of the ISPOR PRO good research practices for the assessment of children and adolescent task force. Value in Health 2013;16:461-79. [58] Sandberg DE, Meyer-Bahlburg HFL, Aranoff GS, Sconzo JM, Hensle TW. Boys with hypospadias: A survey of behavioral difficulties. Journal of Pediatric Psychology 1989;14:491-514. [59] Berg G, Berg R. Castration complex. Evidence from men operated for hypospadias. Acta Psychiatrica Scandinavica 1983;68:143-53.
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[61] Mondaini N, Ponchietti R, Bonafe M, Biscioni S, Di Loro F, Agostini P, et al. Hypospadias: Incidence and effects on psychosexual development as evaluated with the Minnesota Multiphasic Personality Inventory test in a sample of 11,649 young Italian men. Urologia Internationalis 2002;68:81-5.
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[62] Mureau MA, Slijper FM, Nijman RJ, van der Meulen JC, Verhulst FC, Slob AK. Psychosexual adjustment of children and adolescents after different types of hypospadias surgery: A norm-related study. J Urol 1995;154:1902-7. [63] van der Toorn F, de Jong TPVM, de Gier RP, Callewaert PR, van der Horst EH, Steffens MG, et al. Introducing the HOPE (Hypospadias Objective Penile Evaluation) score: a validation study of an objective scoring system for evaluating cosmetic appearance in hypospadias patients. J Pediatr Urol 2013;9:1006-16. [64] Merriman LS, Arlen AM, Broecker BH, Smith EA, Kirsch AJ, Elmore JM. The GMS hypospadias score: assessment of inter-observer reliability and correlation with postoperative complications. J Pediatr Urol 2013;9:707-12.
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[65] Arlen AM, Kirsch AJ, Leong T, Broecker BH, Smith EA, Elmore JM. Further analysis of the Glans-Urethral Meatus-Shaft (GMS) hypospadias score: Correlation with postoperative complications. J Pediatr Urol 2015;11:71.e1-71.e5.
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[66] Rynja SP, de Kort L, de Jong TPVM. Urinary, sexual, and cosmetic results after puberty in hypospadias repair: current results and trends. Current Opinions in Urology 2012;22:453-6. [67] Dodds PR. Re: The pediatric penile perception score: an instrument for patient selfassessment and surgeon evaluation after hypospadias repair: D.M. Weber, V.B. Schonbucher, M.A. Landolt, R. Gobet. J Urol 2008; 180: 1080-4. J Urol 2009;181:1506.
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[68] Fasching G, Arneitz C, Gritsch-Olipp G. Foreskin reconstruction and preservation of a thin distal urethra: a challenge in tubularized incised plate urethroplasty. Pediatric Surgery International 2011;27:755-60.
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[69] Schonbucher VB, Landolt MA, Gobet R, Weber DM. Health-related quality of life and psychological adjustment of children and adolescents with hypospadias. Journal of Pediatrics 2008;152:865-72. [70] Ruppen-Greeff NK, Weber DM, Gobet R, Landolt MA. Health-related quality of life in men with corrected hypospadias: An explorative study. J Pediatr Urol 2013;9:551-8.
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[71] Örtqvist L, Fossum M, Andersson M, Nordenström A, Frisén L, Holmdahl G, et al. Long-term follow-up of men born with hypospadias: Urological and cosmetic results. J Urol 2015;193:975-82.
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Figure Legends:
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Fig. 1. PRISMA [43] flow chart.
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Table 1. Validated instruments used to measure quality of life/satisfaction of patients with hypospadias. Studies describing instrument Springer 2014 [23] Rynja et al. 2011 [27] Rynja et al. 2012 [66]
Holland et al. 2001 [31] Aulagne et al. 2010 [34] Seibold et al. 2010 [35] Liu et al. 2015 [48] Thiry et al. 2015 [52]
Pediatric Penile Perception Score (PPPS)
Springer 2014 [23] Rynja et al. 2011 [27] Kurzroc 2008 [10] Dodds 2009 [67] Morel-Journel et al. 2009 [40] van der Toorn et al. 2013 [41] Deibert et al. 2011 [45]
Penile Perception Score (PPS)
van der Toorn 2013 [41]
Fasching et al. 2011 [68] Schönbucher et al. 2008 [38] Weber et al. 2009 [33] Castagnetti et al. 2013 [37] Schönbucher et al. 2008 [69] Ghavami-Adel et al. 2014 [42] Weber et al. 2008 [43] Haid et al. 2016 [50] Thiry et al. 2015 [52]
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Hypospadias Objective Scoring Evaluation (HOSE)
Studies using instrument
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Instrument
Schlomer et al. 2014 [25] Ruppen-Greeff et al. 2013 [70] Weber et al. 2013 [44] Liu et al. 2015 [48] Ciancio et al. 2015 [49] Örtqvist et al. 2015 [71]
Mieusset et al. 2005 [32] Ringert et al. 1999 [36] Woodhouse et al. 2005 [47]
Mureau et al. 1995 [28] Mureau et al. 1997 [17]
Genital Perception Scale (GPS) for children/ Junior Genital Perception Scale
Springer 2014 [23] Rynja et al. 2011 [27] Mieusset et al. 2005 [32] Woodhouse et al. 2005 [47]
Vandendriessche et al. 2010 [24] Mureau et al. 1995 [28] Mureau et al. 1997 [17]
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Genital Perception Scale (GPS) for adults
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Table 2. Development and validation of instruments.
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Penile Perception Score (PPS)
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Patient interviews Literature Expert opinion Develop conceptual model Item reduction Expert opinion Item redundancy Endorsement frequencies Missing data Factor analysis Tests of scaling assumptions Psychometric Acceptability analyses Internal consistency reliability Item total correlations Interrater reliability Test-retest reliability Validity within scale Validity comparison with other measures Validity hypothesis testing Responsiveness
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Item generation
Pediatric Penile Perception Score (PPPS)
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Method/Evaluation
Hypospadias Objective Scoring Evaluation (HOSE)
Genital Perception Scale (GPS) for adults
Genital Perception Scale (GPS) for children and adolescents/Junior Genital Perception Scale
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Table 3. Domain analysis for patient-reported hypospadias outcomes instruments.
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Functional evaluation
Position and/or shape of meatus Shape of the glans Size of the glans Shape of penile skin Penile length (flaccid or erect) Penile axis when erect Penile thickness Penile colour General appearance of penis Meatal location Meatal shape Penile axis when erect Urinary stream Fistula
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Method/Evaluation
Genital satisfaction
Penile Perception Score (PPS)
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Hypospadias Pediatric Objective Penile Scoring Perception Evaluation Score (HOSE) (PPPS)
Genital Perception Scale (GPS) for children and adolescents/Junior Genital Perception Scale
Genital Perception Scale (GPS) for adults
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