Elsevier

Journal of Pediatric Urology

Volume 12, Issue 3, June 2016, Pages 152.e1-152.e7
Journal of Pediatric Urology

Worldwide prevalence of hypospadias

https://doi.org/10.1016/j.jpurol.2015.12.002Get rights and content

Summary

Introduction

Hypospadias is a common congenital malformation. Surgical repair and management of the long-term consequences require a substantial amount of socioeconomic resources. It is generally accepted that genetic and environmental factors play a major role in the etiology of hypospadias. There have been contradictory reports on rising hypospadias rates, and regional and ethnical differences. The exact prevalence of hypospadias is of major interest for healthcare providers, clinical medicine, and research.

Objective

To review the literature regarding the worldwide prevalence of hypospadias.

Study design

Pubmed, EMBASE and Google were systematically screened for: hypospadias, congenital malformation, anomaly, incidence, prevalence, and epidemiology. Exclusion criteria were surgical and risk-factor studies. To give an additional comprehensive overview, prevalence data were harvested from the Annual Report of the International Clearinghouse Centre for Birth Defects Surveillance and Research. Prevalence was reported as per 10,000 live births.

Results

Data were available from 1910 to 2013. The median study period was 9 years (range: 1–36 years). Approximately 90,255,200 births have been screened in all studies. The mean prevalence were: Europe 19.9 (range: 1–464), North America 34.2 (6–129.8), South America 5.2 (2.8–110), Asia 0.6–69, Africa 5.9 (1.9–110), and Australia 17.1–34.8. There were major geographical, regional, and ethnical differences, with an extreme heterogeneity of published studies. Numerous studies showed an increasing prevalence; on the other hand, there were a lot of contradictory data on the prevalence of hypospadias. The summary table shows contradictory data from the five largest international studies available.

Discussion

There was huge literature available on the prevalence of hypospadias. Most data derived from Europe and North America. Many methodological factors influenced the calculation of an accurate prevalence, and even more of the true changes in prevalence over time (no generally accepted and standardized definition of hypospadias, different monitoring systems, unclear efficiency of notification and data ascertainment, etc.). There was wide variation of prevalence according to countries and ethnicity, and there were conflicting data on the recent trends of prevalence. Moreover, there weren't any epidemiologic data available from many parts of the world.

Conclusion

True prevalence of hypospadias and trends were difficult to estimate. For the future, to be able to assess the true prevalence of hypospadias and changes in prevalence collaboration of national and international prospective registers is recommended.

StudyYearCountryPer 10,000 live birthsTime periodTrend
Bergman et al.201523 European regions18.62001–2010Different trends
Chul Kim et al.2011South Korea1.4–3.32000–2004Increasing
Elliott et al.2011USA421985–2006No increase
Paulozzi1999International registries10–451964–1997Partial increase
Paulozzi et al.1997USA17–501968–1993Increasing
1970–1993

Introduction

Hypospadias is the most common congenital malformation of the penis, and it is considered by some to be a mild form of 46XY disorders of sex development. The urethral opening occurs on the ventral side of the penis; moreover, there is hypoplasia of the ventral tissues in varying degrees, penile curvature, and excessive prepuce. It is generally accepted that genetic and environmental factors play a major role in the etiology of hypospadias [1]. Surgical repair of hypospadias and management of the long-term consequences of being born with a genital condition require socioeconomic resources. Therefore, the exact prevalence of hypospadias is of major interest for the community. Recently, the epidemiology of hypospadias and whether the prevalence is rising or stable has become part of a major debate over the potential effects of environmental factors [2]. This article systematically reviewed the literature regarding the worldwide prevalence of hypospadias and changes of prevalence over time.

Section snippets

Methods

Pubmed, EMBASE, and Google (to March 2015) were screened for potentially eligible literature using the following keywords: ((hypospadias) AND epidemiology) OR ((hypospadias) and incidence) OR ((hypospadias) and prevalence) (Fig. 1). The Related Article feature of PubMed was used and the search was restricted to articles in English. For countries or regions with no articles on hypospadias the search was extended with the terms congenital malformation, and anomaly. Exclusion criteria were:

Results

Fig. 2 shows the worldwide available studies on the prevalence of hypospadias, including the mean, minimum and maximum prevalence, covering time ranges from 1910 to 2013. All studies are shown in detail in supplementary Table 1 to Table 6. The included studies were very heterogeneous – there was a whole spectrum from retrospective, hospital-based, descriptive, cross-sectional studies (with a cohort of 1000) to population-based, nationwide, prospective, birth defect registers (covering

Discussion

Since the first study by Sorensen in Denmark from 1910 to 1945, the epidemiology of hypospadias has been extensively reported [5]. In the current review, worldwide epidemiological data were collected on the prevalence of hypospadias, and many heterogeneous studies showed contradictory data.

Conclusions

Hypospadias epidemiology is of fundamental interest for pediatric urology. Over the last 50 years there have been numerous large studies from many parts of the world, with contradictory results. Many methodological factors influence the calculation of an accurate prevalence and, more so, the true changes in prevalence over time. There are no epidemiologic data available from many parts of the world. Based on this study, the authors suggest more national and international collaboration in

Conflict of interest/Funding

Nil.

References (34)

  • M.O. Aho et al.

    Geographical differences in the prevalence of hypospadias in Finland

    Environ Res

    (2003)
  • Defects ICCfB. Annual Report 2012

    (2012)
  • L.J. Paulozzi et al.

    Hypospadias trends in two US surveillance systems

    Pediatrics

    (1997)
  • C. Stoll et al.

    Genetic and environmental factors in hypospadias

    J Med Genet

    (1990)
  • World Health Organization ROfSEA

    Birth defects in South-East Asia – a public health challenge

  • E. Schmiedeke et al.

    Towards the perfect ARM center: the European Union's criteria for centers of expertise and their implementation in the member states. A report from the ARM-Net

    Pediatr Surg Int

    (2015)
  • H. Dolk et al.

    Toward the effective surveillance of hypospadias

    Environ health Perspect

    (2004)
  • Cited by (0)

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