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ORIGINAL RESEARCHPSYCHOLOGY
Psychosexual Development of Children and Adolescents
with Hypospadias
Verena B. Schnbucher, MSc,* Markus A. Landolt, PhD, Rita Gobet, MD,* and Daniel M. Weber, MD*
*University Childrens Hospital, Pediatric Urology, Zurich, Switzerland; University Childrens Hospital, Department of
Psychosomatics and Psychiatry, Zurich, Switzerland
DOI: 10.1111/j.1743-6109.2007.00742.x
ABSTRACT
Introduction. Hypospadias is the most common malformation of the penis. Despite the common assumption that
hypospadias may affect childrens psychosexual development, only a few studies report on the patients psychosexual
adjustment during childhood and adolescence.
Aim. A comprehensive, cross-sectional investigation of the psychosexual development of boys operated on for
hypospadias in comparison to a healthy control group.
Methods. Sixty-eight children and adolescents (717 years) operated on for hypospadias were examined by means of
a standardized interview assessing penile self-perception, gender-role behavior, sexual experiences, and sexual
attitude. Scores were compared to an age-matched control group consisting of 68 boys after hernia repair. Predictive
values of medical variables as well as the patients knowledge of hypospadias were assessed.
Main Outcome Measures. The Pediatric Penile Perception Score, the Gender-Role Questionnaire by Ijntema and
Cohen-Kettenis, and a self-developed questionnaire on rst sexual experiences and sexual attitude comprised the
standardized assessment instruments.
Results. Boys with hypospadias did not signicantly differ from the control subjects with regard to penile selfperception, gender-role behavior, rst sexual experiences, and sexual attitude. Younger age and the patients
knowledge of hypospadias predicted a more positive penile self-perception, while a more pronounced masculine
gender-role behavior was best predicted by younger age at nal surgery.
Conclusions. Boys with corrected hypospadias may show a psychosexual development that is similar to healthy
children. Puberty could be a critical time for the patients, however, during which they might require regular
urological follow-ups and may benet from age-appropriate information about their penile condition. Moreover, the
later corrective surgery is completed, the more likely the patients may become insecure with regard to gender-role
behavior. Schnbucher VB, Landolt MA, Gobet R, and Weber DM. Psychosexual development of children
and adolescents with hypospadias. J Sex Med 2008;5:13651373.
Key Words. Hypospadias; Psychosexual Development; Childhood; Adolescence; Genital Self-Perception; GenderRole Behavior
Introduction
ypospadias is the most common malformation of the penis with a reported incidence
ranging up to 3 per 1,000 male births [1]. It is
manifested by an abnormal position of the urethral
opening, which lies proximal to its normal location
in the glans, on the ventral surface of the penis or
2008 International Society for Sexual Medicine
1366
penile malformation, most surgeons recommend
surgery as early as the rst 12 months to avoid
any negative impact on the patients psychosexual
development [47]. These suggestions on the
timing of surgical correction, however, are not
evidence based.
The term psychosexual development implies that
sexual development is not only a physical process
but also interacts with psychological factors. It
comprises many components such as the development of gender identity, gender-role behavior,
body image, and sexual behavior habits [8]. Several
factors related to hypospadias are assumed to
expose the patients to an increased risk of psychosexual maladjustment. First, the patients generally
experience the stressors of genital surgery [9,10].
Second, hypospadias surgery does not result in a
perfectly normal penile appearance and can entail
some functional complications (e.g., stulas,
stenoses) [2]. Ultimately, there is increasing evidence that prenatal exposure to androgens
not only induces the sexual differentiation of the
human genitalia but also inuences postnatal
sex-typed behavior patterns by engendering sexspecic brain structures. Most of the evidence
derives from studies on individuals with disorders
of sex development (DSD), who are exposed to
nonphysiological androgen levels [11]. The faulty
embryogenesis of the penile urethra in hypospadias is assumed to be related to abnormalities in
prenatal androgen exposure and/or androgen
receptor defects [12]. Sandberg et al. [13] hypothesized that children born with hypospadias might
show a more atypical gender-role behavior as well.
This hypothesis has not been conrmed to date.
However, despite the common assumption that
hypospadias may affect the childrens psychosexual
development, only few studies report on the
patients psychosexual adjustment during childhood and adolescence [14]. Previous ndings indicate that boys with hypospadias suffer more often
from negative genital appraisal and sexual inhibitions than healthy age-matched controls, but do
not differ from healthy adolescents with regard
to sexual activity, number of coitus partners, and
sexual desire [1517]. Furthermore, the studies
show that psychosexual development is partly
affected by the severity of hypospadias and the
number of hospitalizations, but not by the quality
of surgical outcome, age at nal surgery, number
of operations, and type of surgical procedure
[1319]. Results are inconsistent with regard to
gender-role behavior and age at rst sexual experiences [1316,19].
J Sex Med 2008;5:13651373
Schnbucher et al.
Aims
Procedure
The present study was approved by the local
research ethics committee. Children and parents
were informed about the study by letter and were
requested to return written informed consent.
The participating children and adolescents were
assessed by means of a standardized interview that
was usually conducted at the hospital. In addition,
hypospadias patients were offered a urological
examination, during which standardized photographs of their genitalia were taken.
Subjects
Patients
Children and adolescents operated on for hypospadias at the University Childrens Hospital
Zurich (19912005) were eligible for this study
(N = 147). Inclusion criteria for participation in
the study were (i) age between 6 and 17 years;
(ii) command of the German language; (iii)
follow-up since last surgery 1 year; (iv) no
current postoperative treatment of hypospadias;
and (v) residence in Switzerland. Children with
chronic diseases, disabilities, or hypospadias as a
concomitant phenomena in a dened DSD were
excluded.
1367
Age
Mean (SD)
Median (range)
Nationality (%)
Swiss
Foreigners
Socioeconomic status (%)
Lower
Middle
Upper
Age at first operation
Mean (SD)
Median (range)
Age at final operation
Mean (SD)
Median (range)
No. of operations
Mean (SD)
Median (range)
Days in hospital
Mean (SD)
Median (range)
Follow-up since last surgery
Mean (SD)
Median (range)
Severity of hypospadias (%)
Glanular
Coronal
Subcoronal
Penil
Penoscrotal
Patients (N = 68)
Controls (N = 68)
10.81 (3.18)
9.84 (6.6617.34)
11.05 (3.18)
10.39 (6.8018.23)
0.61
58.5
41.5
69.2
30.8
0.27
25.0
45.3
29.7
17.2
56.2
26.6
0.47**
P*
3.18 (2.45)
2.30 (0.2011.87)
2.97 (2.87)
2.02 (0.2014.18)
0.21
4.26 (3.18)
2.92 (0.7812.29)
3.09 (2.96)
2.05 (0.2014.18)
0.008
1.91 (2.18)
1.00 (1.0015.00)
1.10 (0.31)
1.00 (1.002.00)
<0.0001
11.87 (13.27)
8.00 (1.0077.00)
2.63 (1.18)
2.00 (1.007.00)
<0.0001
6.32 (3.23)
6.28 (1.0015.15)
7.96 (3.52)
7.46 (0.8016.13)
0.002
19.1
35.3
8.8
32.4
4.4
*U-tests according to MannWhitney for dichotomous variables, c2-tests for categorical variables.
**U-test according to MannWhitney based on SES scores.
Control Group
Each participant of the hypospadias group was
matched with regard to both current age and age
at rst operation with a boy who was treated for an
inguinal hernia. In order to have an equally large
control group, 116 children were contacted, of
which 48 (39.3%) refused to participate. Control
subjects did not signicantly differ from the
hypospadias group with regard to nationality,
socioeconomic status, age at assessment, and age at
rst operation (Table 1). However, the boys with
hypospadias had their nal surgery at an older age,
had more operations, were hospitalized for a
longer period, and showed a shorter follow-up
since their last surgery. All 68 control subjects
could be asked about penile self-perception, 49
about gender-role behaviour, and 25 about sexual
milestones and sexual attitude.
J Sex Med 2008;5:13651373
1368
Main Outcome Measures
Schnbucher et al.
Table 2 Patients PPPS scores, gender-role behavior,
sexual attitude, and sexual experiences: comparison with
controls
Patients
PPPS score (N = 133)
M (SD)
9.51 (2.12)
Gender-role behavior (N = 96)
M (SD)
69.64 (4.84)
Sexual attitude (N = 45)
M (SD)
6.65 (1.98)
Sexual experiences (N = 45)
Ever been in love? (f, f%)
Yes
19 (95.0)
No
1 (5.0)
Controls
P value*
9.77 (1.97)
-0.12
0.51
68.93 (3.79)
0.16
0.27
6.02 (2.53)
0.28
0.59
21 (84.0)
4 (16.0)
0.24
0.10
18 (85.7)
3 (14.3)
11.72 (2.35)
0.73
0.89
0.11
0.70
0.83
1.00
0.29
15 (60.0)
10 (40.0)
0.25
0.39
0.14
0.03
13.45 (1.57)
15 (60.0)
10 (40.0)
13.40 (0.83)
13 (52.0)
12 (48.0)
6 (24.0)
19 (76.0)
13.67 (0.82)
-0.06
0.73
-0.16
0.93
0.59
0.22
0.54
0.75
0.77
5 (20.0)
20 (80.0)
14.40 (0.55)
0.89
0.35
0.14
N/A
0.67
Sexual Attitude
By means of a 3-point scale (0 = not true, 1 = somewhat true, 2 = true), adolescents were asked to
express their level of agreement with the following
four statements: Sexuality is (i) something enjoyable; (ii) something important; (iii) something
embarrassing; and (iv) something I am not interested in. After recoding the negatively formulated
items, a sum score was computed (08) showing a
Cronbachs a of 0.58. Higher values indicate a
more positive sexual attitude.
Sociodemographic and Medical Characteristics
Sociodemographic and medical variables were
taken from the patients records. Socioeconomic
status (SES) was calculated on the basis of both
paternal occupation and maternal education on a
6-point scale. SES scores, ranging from 212
points, were classied into three social classes as
follows: 25, low social class; 68, middle social
class; and 912, upper social class. This measure
has been shown to be a reliable and valid indicator
of SES in our community [23].
Knowledge of Hypospadias
Patients were asked if they knew the reason for
having been operated on their penis. If the
reported reasons were related to hypospadias in
some way (e.g., I was not able to direct my urine
stream properly), the subjects were categorized as
having some knowledge of their hypospadias (1). If
the reported reasons were not related to hypospadias (e.g., I was circumcised) or if the subjects
were not able to name a reason, they were categorized as being not aware of their hypospadias (0).
Statistical Analyses
Data were analyzed using the statistical package
SPSS for Windows, release 14.0. (SPSS Inc.,
Chicago, IL, USA). All analyses were performed
with two-sided tests. A value of P 0.05 was considered signicant. For categorical comparisons,
c2-tests were used. In case that >20% of the cells
showed an expected frequency <5, Cramers V was
applied. For group comparisons with regard to
the PPPS and the sexual attitude score, Mann
Whitney U-tests were performed because both
scales showed nonnormal distribution. For group
comparisons regarding gender-role behavior and
age at sexual milestones, t-tests were conducted
because both variables were normally distributed
according to the Kolmogorov-Smirnov Test. In
addition, effect sizes (ES) of mean differences
1369
Psychosexual Development
No signicant group differences between hypospadias patients and the control group were found
with regard to penile self-perception, gender-role
behavior, and sexual attitude (Table 2). Accordingly, effect sizes were small. Overall, both groups
showed high satisfaction with penile appearance
and showed an overall positive sexual attitude.
Likewise, adolescents with hypospadias and the
control subjects did not differ with regard to
their sexual experience, age at sexual milestones,
and current relationship status (Table 2). However, adolescents with hypospadias reported
signicantly more often that they already had
experienced orgasm through masturbation. Furthermore, regarding age at rst petting, effect
size was large, suggesting that adolescents with
hypospadias experienced their rst petting at an
older age. Yet, due to the small number of adolescents who already had experienced petting,
effect size did not reach signicance (Cl 95%:
-0.542.08).
Determinants of Penile Self-Perception and
Gender-Role Behavior
Table 3 displays correlations between sociodemographic and medical characteristics as well as
knowledge of hypospadias with PPPS score and
gender-role behavior, respectively. The PPPS
score was signicantly negatively associated with
current age and positively related to knowledge of
J Sex Med 2008;5:13651373
1370
Schnbucher et al.
group. As opposed to both our hypothesis and
previous results [1517], boys with hypospadias
showed an overall positive penile self-perception
that was similar to those of the control subjects.
This unexpected nding might be a result of the
recently achieved improvements in treatment,
which result in better postoperative functional and
cosmetical outcomes. Yet, it can be argued that the
different age ranges of samples between the studies
have contributed to the inconsistent results since
previous studies included children and adolescents
at somewhat higher mean ages [1517].
Likewise in contradiction to our hypotheses are
the actual ndings that boys with hypospadias did
not differ from the control subjects with regard to
gender-role behavior, rst sexual experiences, and
sexual attitude. This corresponds with ndings
from previous studies by Sandberg et al. [13] and
Mureau et al. [16] and suggests that children and
adolescents with hypospadias may show a psychosexual development that is similar to healthy children. However, we did not elicit any information
from the subjects experiences of their sexual milestones, e.g., to what degree they had suffered from
sexual anxieties. The quality, rather than the quantity, of the patients sexual activity may be more
important for their well-being and should be
examined in future studies.
This study also examined medical, sociodemographic, and psychosocial correlates of penile
self-perception and gender-role behavior. Both
bivariate and multivariate analyses revealed that
older age and not being aware of ones hypospadias
were negatively associated with penile selfperception. The nding that adolescent patients
are less satised with penile appearance than
younger children is in line with previous results
[17] and might be the consequence of a change
in physical self-perception as well as an increasing
importance of sexuality during puberty. The
nding that patients who were informed about
their hypospadias showed a more positive penile
Current age
SES
Nationality
Severity
No. of operations
Age at first operation
Age at final operation
Days in hospital
Follow-up since last surgery
PPPS urologists
Knowledge of hypospadias
PPPS patient
Gender-role
behavior
-0.33*
0.10
0.02
-0.02
0.12
-0.10
0.05
0.11
-0.09
0.15
0.35**
-0.22
0.17
-0.20
0.08
-0.10
-0.28
-0.36*
-0.13
0.38*
0.03
0.01
hypospadias. Gender-role behavior was signicantly negatively correlated to the patients age
at last surgery and positively associated with
follow-up since last surgery. There was also a correlation between gender-role behavior and age at
rst surgery, but the association scarcely reached
signicance (P = 0.07).
Statistics for the multiple regression analyses
are presented in Table 4. With regard to the PPPS
score, younger age and knowledge about hypospadias signicantly contributed to the prediction of a
more positive penile self-perception. In contrast, a
more pronounced masculine gender-role behavior
was best predicted by younger age at nal surgery.
Overall, the selected variables accounted for 27%
and 26%, respectively, of the variance of the
outcome measure.
Discussion
Summary of multiple regression analyses for predictors of patients PPPS score and gender-role behavior
Equation 1: PPPS patient
Variable
SEB
SEB
PPPS urologist
Age at final operation
Current age
Knowledge of hypospadias
0.10
0.03
-0.19
1.35
0.14
0.09
0.09
0.49
0.11
0.06
-0.31*
0.37**
-0.08
-1.07
-0.22
0.98
0.40
0.36
0.58
1.66
-0.03
-0.50**
-0.07
0.10
Limitations
The strength of the present study lies in its
signicant contribution to the investigation of
the psychosexual development of children and
1371
Conclusions
1372
To the best of our knowledge, there are excellent
measures of psychosexual differentiation for both
healthy children and children with DSD [32,33].
However, validated measures of other constructs
of psychosexual development such as rst sexual
experiences, sexual behavior habits, and sexual
problems have yet to be developed.
Ultimately, our ndings also implicate some
issues for clinical management. Although our
results suggest that boys with hypospadias may
show a healthy psychosexual development, they
also indicate that puberty might be a critical time
for the patients, who therefore may benet from
regular urological follow-ups until young adulthood. Apart from urological examinations, the
patients should be asked how they feel about sexual
matters and they should be provided with ageappropriate information about their penile condition. Moreover, surgeons must be aware that the
objective surgical outcome and severity of hypospadias might be secondary for the patients psychosexual development during childhood. Yet, our
results might suggest that the later corrective
surgery is completed, the more likely the patients
may become insecure with regard to gender-role
behavior.
Corresponding Author: Verena B. Schnbucher, MSc,
Pediatric Urology, University Childrenss Hospital,
Steinwiesstrasse 75, CH-8032 Zurich, Switzerland. Tel:
+49-174-70-88882; Fax: +41-44-266-80-30; E-mail:
verena.schoenbucher@kispi.uzh.ch; vschoenb@uke.unihamburg.de
Conict of Interest: None declared.
Statement of Authorship
Category 1
(a) Conception and Design
Verena B. Schnbucher; Markus A. Landolt; Rita
Gobet; Daniel M. Weber
(b) Acquisition of Data
Verena B. Schnbucher; Markus A. Landolt; Rita
Gobet; Daniel M. Weber
(c) Analysis and Interpretation of Data
Verena B. Schnbucher; Markus A. Landolt; Rita
Gobet; Daniel M. Weber
Category 2
(a) Drafting the Article
Verena B. Schnbucher; Markus A. Landolt; Rita
Gobet; Daniel M. Weber
(b) Revising It for Intellectual Content
Verena B. Schnbucher; Markus A. Landolt; Rita
Gobet; Daniel M. Weber
J Sex Med 2008;5:13651373
Schnbucher et al.
Category 3
(a) Final Approval of the Completed Article
Verena B. Schnbucher; Markus A. Landolt; Rita
Gobet; Daniel M. Weber
References
1373