doi:10.1016/j.jpedsurg.2008.10.111
Copyright © 2009 Elsevier Inc. All rights reserved.
Original article

Long-term physical, hormonal, and sexual outcome of males with disorders of sex
development

Yoshiyuki Kojima, a, , Kentaro Mizunoa, Akihiro Nakanea, Toshiki Katoa, Kenjiro
Kohria and Yutaro Hayashia
aDepartment of Nephro-urology, Nagoya City University Graduate School of Medical
Sciences, Nagoya 467-8601, Japan


Received 17 August 2008; revised 28 October 2008; accepted 29 October 2008.
Available online 25 July 2009.

Abstract
Purpose
We investigated the long-term physical, hormonal, and sexual outcomes of males
with disorders of sex development (DSD) and discussed the necessity of long-term
follow-up for these patients after surgery.

Patients and Method
Twelve DSD patients (average age, 21.0 ± 3.6 years old) who had been designated
as male in childhood (3 ovotesticular DSD, four 45,XO/46,XY mixed gonadal
dysgenesis, four 46,XX testicular DSD, and one 46,XY DSD; androgen insensitivity
syndrome) were enrolled. For these patients, height, penile length, and
testicular volume were evaluated in adulthood. Serum levels of luteinizing
hormone, follicle-stimulating hormone, and testosterone were also measured
during follow-up. In addition, sexual function and romantic relationships were
evaluated.

Results
Development of the penis and testes was poor. According to the hormonal study,
these patients were diagnosed with hypergonadotropic hypogonadism or
normogonadism; 90% patients had experienced penile erection and masturbation at
the time of participation, and 70% and 40% patients had experienced ejaculation
and sexual intercourse with female partners, respectively. No patients preferred
to avoid sexual contact with women.

Conclusion
Although DSD males had an undeveloped penis and testis and had hypergonadotropic
hypogonadism or normogonadism, most had male sexual potential and male sex
identity as long as testicular tissues were preserved.

Key words: Sexual function; Disorders of sex development (DSD); Ovotesticular
DSD; XX testicular DSD; Mixed gonadal dysgenesis; Testis; Penis

Article Outline
1. Materials and methods
2. Results
3. Discussion
References
Disorders of sex development (DSD) are congenital conditions with atypical
development of chromosomal, gonadal, or anatomical sex [1]. The appearance of
the external genitalia is ambiguous, often with hypospadias or clitoromegaly,
with an undeveloped or ectopic testis or inadequate vagina.

Patients reared as males with ambiguous genitalia, especially proximal
hypospadias, are commonly referred to a urologic clinic to undergo hypospadias
repair, in which DSD patients are included. Generally, these patients need not
only reconstruction surgery for ambiguous external genitalia but also surgical
exploration of gonads and internal genitalia. The aim of reconstruction surgery
in DSD is to create "functional" external genitalia consistent with the sex
assigned to the patient; therefore, one of the most important issues is whether
they can achieve sexual potential as male or female in the future; however,
information about the long-term physical, hormonal, and sexual outcome of DSD
patients cannot be easily determined until decades after surgery because many
patients are lost to follow-up [2]. As a result, there are extremely few reports
of the outcome after surgery of DSD patients, especially those raised as males
[3], [4] and [5].

In our urologic clinic, we usually attempt to observe DSD males for as long as
possible. In this study, we investigated the long-term physical, hormonal, and
sexual outcomes of DSD patients raised as males, including ovotesticular DSD,
mixed gonadal dysgenesis (MGD), 46,XX testicular DSD, and 46,XY DSD, who had
undergone hypospadias repair previously at our institute and with whom we had
discussed the necessity of long-term follow-up after surgery.

1. Materials and methods
We reviewed the medical records of DSD patients who had been designated as male
by both the medical team and family at our institution from 1975 to 1991 and
observed at least to 15 years old. Twelve patients raised as males (3
ovotesticular DSD, 4 MGD, four 46,XX testicular DSD, and one 46,XY DSD; androgen
insensitivity syndrome), who were 16 years or older at the time of the last
visit (average age, 21.0 ± 3.6 years old), were enrolled in this study. Clinical
record files of these patients detailed the initial clinical presentation,
diagnostic investigations, and therapeutic surgical intervention. The diagnostic
study between the first visit and operation had included a physical examination;
chromosomal analysis; hormonal evaluation, including human chorionic
gonadotropin (hCG) stimulation test (2400 IU/d for 3 days) and human menopausal
gonadotropin (hMG) stimulation (112.5 IU/d for 3 days); the presence of the
sex-determining region Y (SRY) sequence; magnetic resonance imaging; cystoscopy;
vaginoscopy; laparoscopy; and gonadal biopsy. After 1993, the presence of the
SRY sequence was examined by polymerase chain reaction method for most patients
during follow-up.

The clinical descriptions of 12 patients in childhood are presented in Table 1.
All patients presented with proximal hypospadias and had undergone hypospadias
repair in childhood. All patients had both Wolffian (epidydimis and/or vas
deferens) and Mullerian (fallopian tube and/or prostatic utricle) structures.
All patients had a prostatic utricle, which opened into the central area of the
verumontanum in the prostatic urethra.

Table 1.
Clinical characteristics of males with disorders of sexual development in
childhood



Diagnosis

Age at first visit

External genitalia

Internal genitalia

Gonad Gonadectomy

Chromosome

HCG Stimulation test

HMG Stimulation test

SRY



Wolffian structure Mullerian structure

1 Ovotesticular DSD 3yo hypospadias (penoscrotal type) + + Rt) ovary Rt) abdomen
Rt) ovarectomy 46,XY + + +
Lt) testis Lt) srotum
2 Ovotesticular DSD 1mo hypospadias (penoscrotal type) + + Rt) ovotestis Rt)
abdomen Bil) gonadectomy 46,XX + + -
Lt) ovotestis Lt) inguinal region
3 Ovotesticular DSD 2mo hypospadias (penoscrotal type) + + Rt) testis Rt)
scrotum Lt) ovarectomy 46,XX/46,XY/47,XX10 + - +
Lt) ovary Lt) abdomen
4 MGD 9mo hypospadias (scrotal type) + + Rt) streak gonad Rt) inguinal region
Rt) gonadectomy 45,XO/46,XY + - NA
Lt) testis Lt) scrotum
5 MGD 0mo hypospadias (scrotal type) + + Rt) streak gonad Rt) abdomen Rt)
gonadectomy 45,XO/46,XY NA NA +
Lt) testis Lt) scrotum
6 MGD 4yo hypospadias (scrotal type) + + Rt) testis Rt) scrotum Bil) gonadectomy
45,XO/46,XY NA NA +
Lt) streak gonad Lt) abdomen
7 MGD 10yo hypospadias (penoscrotal type) + + Rt) testis Lt) streak gonad Rt)
inguinal region Lt) gonadectomy 45,XO/46,XY + - +
Lt) abdomen
8 46,XX testicular DSD 2 mo hypospadias (scrotal type) + + Bil) testis Bil)
scrotum - 46,XX + - -
9 46,XX testicular DSD 8 mo hypospadias (scrotal type) + + Bil) testis Bil)
scrotum - 46,XX + - -
10 46,XX testicular DSD 9 mo hypospadias (scrotal type) + + Bil) testis Bil)
scrotum - 46,XX + - -
11 46,XX testicular DSD 5 mo hypospadias (scrotal type) + + Bil) testis Bil)
scrotum - 46,XX + - -
12 46,XY DSD 4 mo hypospadia (scrotal type) + + Bil) testis Rt) scrotum Lt)
inguinal region - 46,XY + - +


Full-size table
yo: years old.
mo: months old.
NA: not assessed.
Rt: right.
Lt: left.
Bil: bilateral.

HCG stimulation test +; response by serum testosterone after hCG stimulation, -;
response by serum testosterone after hCG stimulation.

HMG stimulation test +; response of urinary estradiol after hMG stimulation, -;
no response of urinary estradiol after hMG stimulation.


View Within Article



Three ovotesticular DSD had several complicated clinical characteristics.
Patient 1 with 46,XY had a right ovary with a fallopian tube and a uterus-like
structure in the abdomen and a left testis with an epididymis and vas deferens
in the scrotum. He had undergone right gonadectomy and resection of the
uterus-like structure. Patient 2 with 46,XX had a right ovotestis in the abdomen
and left ovotestis in the scrotum and had undergone bilateral gonadectomy.
Patient 3 with 46,XX/46,XY/47,XX10+ had a right testis with an epididymis and
vas deferens in the scrotum and a left ovary with an epididymis-like structure
in the abdomen. He had undergone left gonadectomy. The hCG response was normal
in all 3 patients, whereas the hMG response was present in 2 patients (patient
2). Patients 1 and 3 had an SRY sequence but patient 2 did not.

Four MGD patients had both a testis and streak gonad and underwent gonadectomy
for the streak gonad. Although two had undergone orchiopexy for undescended
testis in another clinic before the first visit, the other two (patients 4 and
6) had no testis in the scrotum at the first visit. All patients had a uterus
and an epididymis or fallopian tube. The hCG and hMG stimulation test was
performed for 2 patients (patients 4 and 7); the hCG response was normal,
whereas the hMG response was absent. Patients 5, 6, and 7 were confirmed as
having the SRY sequence.

Four 46,XX testicular DSD patients (patients 7-11) had not only an epididymis
and vas deferens but also a Mullerian remnant. One patient (patient 10) had
gynecomastia with age. All 46,XX testicular DSD patients had undergone open
bilateral gonad biopsy for their diagnoses, and histologic examination showed
bilateral testis. The left testis of one patient (patient 8) disappeared during
his follow-up period. The enlarged Mullerian structure (prostatic utricle) was
resected in one patient (patient 10) because of urinary tract infection. The hCG
response was normal, whereas the hMG response was absent in all patients,
showing that gonad tissues could potentially develop as testes without ovarian
tissue. All 46,XX testicular DSD patients had no SRY sequence. One patient
(patient 9) had a neurogenic bladder caused by myelomeningocele and underwent
cystectomy and neobladder replacement at the age of 25.

One 46,XY DSD patient (patient 12) had epididymis and vas deferens. He had
undergone open bilateral gonad biopsy, and histologic examination showed
bilateral testis.

For these patients, their height, penile length, and testicular volume in
adulthood were evaluated. Serum levels of luteinizing hormone (LH),
follicle-stimulating hormone (FSH), and testosterone were also measured by
radioimmunoassay (RIA) during follow-up. Radioimmunoassays for LH, FSH, and
testosterone were performed using RIA kits (LH and FSH, Japan DPC, Tokyo, Japan;
testosterone, Amersham Biosciences, Tokyo, Japan).

In addition, sexual function, such as their experiences with penile erection,
libido, orgasm, ejaculation, masturbation, and sexual intercourse, was
evaluated. Their romantic relationships and marriage were also detailed.

2. Results
Patients were observed for a median 19.8 ± 3.3 years (range, 16-28 years) after
diagnosis. Clinical features in adulthood are described in Table 2. The mean
final height of the 12 patients was 154.5 ± 3.8 cm, and most were under the 10th
percentile of the range of normal Japanese adult males. Development of the penis
and testes was poor. Average penile size was 4.1 ± 0.7 cm. No gonadal tumors
were detected during follow-up. They also had poor pubic hair.


Table 2.
Physical and sexual outcome of males with disorders of sexual development

Diagnosis Present age height Testicular volume Penile size Erection
Masturbation Ejaculation Sexual intercourse Married
1 Ovotesticular DSD 18 yo 151 cm Rt) – 4.5 cm + + + - -
Lt) 9.5 ml
2 Ovotesticular DSD 16 yo 156 cm Rt) – 3.5 cm NA NA NA NA NA
Lt) -
3 Ovotesticular DSD 19 yo 161 cm Rt) 8.0 ml 5.2 cm + + + - -
Lt) -
4 MGD 22 yo 153 cm Rt) – 3.0 cm NA NA NA NA -
Lt) 2.9 ml
5 MGD 19 yo 150 cm Rt) – 5.0 cm + + + NA -
Lt) 2.8 ml
6 MGD 21 yo 155 cm Rt) – 3.8 cm + + + - -
Lt) -
7 MGD 27 yo 150 cm Rt) 3 ml 4.0 cm + + + + -
Lt) -
8 46,XX testicular DSD 23 yo 157 cm NA 4.5 cm + + + + -
9 46,XX testicular DSD 28 yo 150 cm Rt) 5.1 ml 5.0 cm - - - - -
Lt) -
10 46,XX testicular DSD 21 yo 156 cm Rt) 3.3 ml 3.6 cm + + + + -
Lt) 3.2 ml
11 46,XX testicular DSD 20 yo 160 cm Rt) 4.2 ml 3.6 cm + + + + +
Lt) 3.8 ml
12 46,XY DSD 18 yo 155 cm Rt) 4.5 ml 4.0 cm + + - - -
Lt) 4.2 ml

Full-size table
yo: years old.
NA: not assessed.
Rt: right.
Lt: left.
Bil: bilateral.


View Within Article



Serum levels of LH, FSH, and testosterone were examined from infancy to
adulthood in 10 patients by RIA (Fig. 1). As in normal males, the concentrations
of these hormones in 10 of 12 patients, except patients 2 and 6, elevated
spontaneously during the pubertal period. Testosterone levels were low normal or
slightly decreased, and LH and FSH were elevated in these patients. These high
levels of LH and FSH began to be observed in the peripubertal stage.
Follicle-stimulating hormone was more frequently elevated than LH. According to
the concentrations of these hormones, these patients were diagnosed with
hypergonadotropic hypogonadism or normogonadism. Two patients (patients 2 and
6), who had undergone bilateral gonadectomy, received testosterone therapy
(intramuscular depot injections of testosterone esters) immediately before
puberty. One patient (patient 9), with mental retardation and neurogenic bladder
caused by myelomeningocele, also received testosterone therapy for deficiency of
sexual function at the age of 27, but he discontinued the therapy because of
adverse effects.






Full-size image (72K)

Fig. 1. Hormonal evaluation of DSD patients from childhood to adulthood after
diagnosis and operation. Serum concentrations of LH, FSH, and testosterone of
DSD patients were determined by RIA at the ages indicated.

View Within Article



Of 10 DSD males, 9 (90%) had experienced penile erection and masturbation at the
time of participation. Of 10 patients, 7 (70%) had experienced ejaculation, and
5 of these 7 patients had ejaculation problems, including dribbling, retained,
or delayed ejaculation. No patient complained of problems with libido and
orgasms. Of 10 patients, 4 (40%) had experienced sexual intercourse with female
sex partners in the past year. Only one patient (patient 11), a 46,XX testicular
DSD, was married at the time of participation, and two 46,XX testicular DSD had
a stable female partner and expressed a desire for marriage in the future. No
patients preferred to avoid sexual contact with women. There were no cases of
fertility, and the only semen analysis showed azoospermia (patient 11).

3. Discussion
There are several previous reports about the long-term physical, hormonal, and
sexual outcomes of 46,XY DSD and 46,XX DSD, such as complete androgen
insensitivity syndrome (AIS) and congenital adrenal hyperplasia, which are
usually raised as female [6] and [7]; however, there are very few reports on the
long-term outcome of DSD males. In this study, we investigated the long-term
physical, hormonal, and sexual outcomes of 12 DSD males, including ovotesticular
DSD, MGD, 46,XX testicular DSD, and 46,XY DSD.

Consistent with previous reports, 46,XX testicular DSD and MGD were shorter than
normal males [8] and [9]. Although their reduced height might be attributed to
genes on the Y chromosome that control height [10], not only 46,XX testicular
DSD but also ovotesticular DSD and 46,XY testicular DSD with a complete Y
chromosome were also short.

Recent remarkable advances in hypospadias repair have provided satisfactory
penile reconstruction, not only cosmetically but also functionally; however,
female sex assignment has been considered optimal and common in severely
undermasculinized cases because feminization with surgical and endocrine
treatment was considered more successful than masculinization [11]. The size of
the penis and its potential to develop at puberty into a sexually functional
penis are one of the important concerns. There are several reports about penile
development in sporadic hypospadias patients [12] and [13]. Mureau et al [12]
reported that penile underdevelopment was noted about 2 times more often in
hypospadias patients than among age-matched normal boys. Bracka et al [13]
reported a clear correlation between the severity of hypospadias and penile
length. On the other hand, there are few reports about penile development in DSD
males. In 46,XX testicular DSD with genital abnormalities, penile size and
testicular volume, measured directly or after surgery in the case of cryptorchid
patients, were below the normal values obtained in controls [14]. The average
penile size of Japanese men is approximately 8 cm, and the critical testicular
volume indicating normal testicular function of Japanese men is approximately 30
mL [15]. In our study, all DSD males had proximal hypospadias and had an
underdeveloped penis after puberty. This insufficient virilization of the
external genitalia may result from hypogonadism. In the previous 46,XX
testicular DSD studied, hypogonadism started in the peripubertal stage, similar
to the pattern in Klinefelter's syndrome where high FSH levels are associated
with normal or low testosterone levels [16]. Conversely, in a study of
ovotesticular DSD, peripubertal testosterone levels were lower than in 46,XX
testicular DSD [16]. In our study, 10 patients were diagnosed with
hypergonadotropic hypogonadism or normogonadism, and the concentration of
testosterone in these patients was low or low normal in young adults,
concomitant with a rise in FSH, reflecting the degeneration of spermatogenesis
and Leydig cell function. The 46,XX testicular DSD is generally characterized by
azoospermia [8]. On the other hand, phenotypic males with normal spermatogenesis
and phenotypic females with normal conception and delivery have been reported in
ovotesticular DSD [17] and [18]. Although testicular functions of DSD in
adulthood are unclear, complete testicular function may not be expected.

In our study, most patients showed potential for erection, ejaculation, and
masturbation; however, they had some problems with ejaculation, such as
dribbling, retained, or delayed ejaculation or the absence of ejaculation, which
have been reported after hypospadias repair in patients even without DSD [19].
This may be secondary to dilation of the reconstructed urethra or the presence
of a Mullerian remnant [19]. On the other hand, no patient complained of
problems with libido and orgasms. Patients with DSD had been considered to have
an increased incidence of sexual dysfunction as a result of their condition and
psychologic factors that impact on sexual function [20]. Children with DSD,
their parents, and we always face the difficulty of dealing with the diagnosis
and accepting reconstructive surgery of the genitalia to avoid psychosexual
disturbance. Sex dissatisfaction may occur more frequently in individuals with
DSD [3]. Fortunately, however, no patient had serious psychosexual problems. All
patients were interested in women and desired to have contact and to fall in
love with women, and interestingly, three 46,XX testicular DSD patients and 2
MGD patients had experienced sexual intercourse with a female sex partner, and
one 46,XX testicular DSD patient was married. Both biologic and social factors
seem responsible for the development of sex identity [3]. Because the DSD males
in our study had received prenatal androgen exposure, although insufficient, and
had been raised as male, as a result, they may have had male sex identity;
however, more systemic psychologic assessments will be needed to conclude that
these patients had no psychologic problems.

The necessity for testosterone therapy for all DSD males is controversial.
Testosterone therapy may be expected to induce puberty, growth, sexual function,
and support for psychosexual maturation [1]; however, we performed testosterone
therapy for only 3 males—2 who underwent bilateral gonadectomy and 1 who
complained of deficiency of sexual function. However, most patients with testis
underwent no hormonal therapy because they did not desire additional therapy,
and puberty and the potential for sexual function developed without hormonal
therapy. Because routine hormonal therapy for a long period may affect the
quality of life, with a greater risk of developing prostate cancer in the future
[21], careful consideration may be required.

In conclusion, although DSD males usually have an undeveloped penis and testis
and have hypergonadotropic hypogonadism or normogonadism, most may have male
sexual potential and male sex identity without hormonal therapy, if testicular
tissues are preserved. This means that testicular function in DSD males may be
insufficient for masculinization of the fetal external genitalia but sufficient
to achieve male sexual function and develop male sex identity; however, our
study showed no difference in sexual potential and sex identity between DSD
males and age-matched normal males because a limitation of this study is the
small sample size. A large multicenter study is required to obtain more detailed
information on the long-term physical, hormonal, and sexual outcomes of DSD
males.

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Corresponding author. Tel.: +81 52 853 8266; fax: +81 52 852 3179.

Journal of Pediatric Surgery Volume 44, Issue 8, August 2009, Pages 1491-1496