Assessing quality of life of patients with hypospadias: A systematic review of validated patient-reported outcome instruments

Summary

Background

Patient-reported outcomes have the potential to provide invaluable information for evaluation of hypospadias patients, aid in decision-making, performance assessment, and improvement in quality of care. To appropriately measure patient-relevant outcomes, well-developed and validated patient-reported outcome (PRO) instruments are essential.

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 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.

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), Penile Perception Score (PPS), Genital Perception Scale (GPS) for adults, and GPS for children/adolescents. Instrument development and validation was limited. The majority of 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 patient-reported element, few have used validated PRO instruments developed in a hypospadias population. Existing instruments to measure patient-reported outcomes in hypospadias require improvement in both the breadth of content and in their development and validation methodology.

Introduction

Patient-reported outcome (PRO) instruments are questionnaires/instruments that allow for self-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 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 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 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].

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 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 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?). 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, 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], [8], [9].

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], [11], [12]. Rather, there is a need to capture the considerable long-term cognitive, behavioral, and self-esteem consequences that result from poor cosmetic or 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.

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 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.