Human Reproduction vol 11 no 9 pp 1919-1922, 1996
Natural spermatoceles in irreversible obstructive
azoospermia—reservoirs of viable spermatozoa for
assisted conception
A.V.Hirsh1-4, N.L.Dean2, PJ.Mohan1, A.G.Shaker3
and J-S.Bekir1
'Hallam Medical Centre, The London Women's Clinic, 115 Harley
Street, London WIN IDG, UK
2
Present address- Royal Victoria Hospital, Women's Pavilion, 687
Pine West Avenue, Montreal QC H3A, Canada
3
Present address Ross Hall Hospital, 221 Crookston Road,
Glasgow GS2 3NQ, UK
"•To whom correspondence should be addressed
Fine needle aspiration of asymptomatic spermatoceles
detected in five men with obstructive azoospermia was
evaluated as a minimally invasive method of sperm retrieval
for assisted conception. Sperm preparations adequate for
in-vitro fertilization were initially obtained from three men,
but there was failure of fertilization in all three cycles.
However, intracytoplasmic sperm injection (ICSI) in three
couples resulted in fertilization of 58% of metaphase II
oocytes, with embryo transfers in five of six cycles. A
successful pregnancy and delivery resulted from spermato-
zoa from a man with an irreversible vasectomy, for whom
previous epididymal microaspiration and ICSI had been
unsuccessful. Spermatoceles were found in 4% of men with
irreversible obstructive azoospermia referred for assisted
conception. These patients should therefore be examined
very carefully for small cysts near the epididymis, because
fine needle aspiration, usually without anaesthesia, can
instantly locate a ready source of viable spermatozoa for
ICSL This is less invasive than the alternative sperm
retrieval procedures, which are more traumatic and require
local or general anaesthesia.
Key words: cyst of epididymis/intracytoplasmic sperm injec-
tion/male infertility/obstructive azoospermia/spermatocele
Introduction
'It is recorded that in the 14th century the Pope granted a
petition from a gentleman to marry two wives because he
possessed three testicles' (Bailey, 1962) Men with large
spermatoceles may believe they have an extra testis, but
spermatoceles are usually very small and are defined as
retention cysts of the epididymis, efferent ducts, or rete testis,
which contain spermatozoa (Bailey, 1962). Developing after
puberty, they are more common after the age of 40 years.
Rolruck (1928) found spermatoceles in 1% of 50 000 normal
men.
In surgically irremediable obstructive azoospermia, attempts
to establish reservoirs of spermatozoa for assisted conception
by creating or implanting artificial spermatoceles (Belker,
© European Society for Human Reproduction and Embryology
1991; Miura et al, 1991), have generally been abandoned in
favour of surgical sperm retrieval for intracytoplasmic sperm
injection (ICSI) by microsurgical (MESA, Silber et al, 1994;
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Tournaye et al., 1994) or percutaneous (PESA, Shinvastav
et al, 1994) epididymal sperm aspiration, testicular sperm
extraction (TESE, Devroey et al, 1994) or aspiration (TESA,
Craft and Tsirigotis, 1995).
Among 75 men with obstructive azoospermia referred for
assisted conception, three (4%) were found to have natural
spermatoceles yielding viable spermatozoa on fine needle
aspiration (Hirsh et al, 1994). We summarize here our experi-
ence of attempting assisted conception using spermatozoa
obtained from the spermatoceles of five patients via the
technique of fine needle aspiration, which was evaluated as a
minimally invasive method of sperm retrieval. This resulted
in the birth of a child to a second wife, in congruity with the
petition quoted by Bailey (1962).
Material and methods
Patients
Among 75 men with irremediable obstrucuve azoospermia referred
for MESA with standard in-vitro fertilization (TVF) between 1991 and
1992, three (4%) were found to have a small unilateral spennatocele
containing moule spermatozoa (Hirsh et al, 1994). In 1995 similar
spermatoceles were detected in a further two patients with the same
diagnosis. The five men were aged 29—48 years (mean 41.4), their
wives were 29—44 years (mean 35.2), and the duration of infertility
was between 4 and 19 years (mean 8 4). One of the wives had
bilateral hydrosalpinges, and another polycystic ovanes, with no other
adverse female fertility factors except age, since two were >40 years.
Patient UVA had left unilateral vas aplasia with a discrete 1 cm
spennatocele at the upper pole of this testis. He had a past history
of epididymo-orchiUs. His right vas deferens was obstructed, but two
vaso-vasostomies had failed. Patients YS1 and YS2 had Young's
syndrome (Young, 1970). YS1 had a history of allergic rhinitis, nasal
sinus drainage, tuberculosis, and a meningioma. Scrotal exploration
had revealed collapsed epididymides, and bilateral epididymo-
vasostomy was unsuccessful His left spermatocele was 2 cm in
diameter. YS2 had a history of sinusitis with two unsuccessful
attempts at bilateral epididymo-vasostomy for upper epididymal
obstrucuon. His 2-3 cm right spennatocele was bilocular. Patient W
had a vasectomy in 1978, failed bilateral vaso-vasostomy in 1990,
and excision of a nght epididymal cyst in 1991. At another centre in
1994, MESA and ICSI had resulted in abnormal fertilization. His left
spermatocele was 1.5 cm in diameter. Patient UCA had right congenital
anorchia, diagnosed at 18 months during bilateral herniorrhaphy and
hydrocele repairs In 1991 left testicular torsion had necessitated
surgical fixation. His 3 mm cyst on the surface of the left corpus
epididymis was not detected by ultrasound. Two previous seminal
fluid analyses had revealed occasional immoule spermatozoa.
1919
A.V.Hlrsh et aL

Table L Analysis of fluid from spermatoceles in obstructive azoospermia Values are means (range)

Patient Aspiration Volume Spermatozoa Motihty Progression % normal

(spermatocele*) samples (ml) (XlO^/ml) (*) mode (1-4) forms

UVA 2 08 185 9 0-1 70

(1 cm) (0.5-1 0) (180-189) (1-17) (0-D
YS1 3 2.5 63.7 4.7 0-1 23
(2 cm) (1 2-5.0) (40-100) (3-6) (0-2) (21-25)
YS2 5 84 1.3 8.4 1 11
(2-3 cm) (2 1-12 0) (0 1-3.0) (5-17) (0-1)
W 2 38 29 75 1 10
(1 5 cm) (1.5-6 0) (0.7-5 0) (5-30) (1-2)
UCA 2 03 53.0 175 1-2 90
(3 mm) (0.25-0.4) (6-100) (5-30) (1-2)

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Summary 14 4.2 56.0 89 1 403
(mean = 1 . 5 cm) (0 25-12) (0 1-189) (1-30) (0-2) (10-90)

•Value in parentheses in this column is the diameter of the spermatocele aspirated.

All the men had azoospermia with normal concentrations of serum
follicle stimulating hormone (FSH) and their testes were normal
in size and tone. There was no significant engorgement of their
epididymides, except the nght epididymis of patient W . Apart from
patient UCA, the spermatoceles were all discrete, associated with the
caput epididymis, and transilluminated. The four smaller spermato-
celes were tense, whereas the largest was soft (patient YS2).
Spermatocele aspiration
Percutaneous fine needle aspiration, with a 25-gauge needle and
appropriate syringe, delivered cloudy fluid from all the spermatoceles,
apart from patient YS2, where it was clear. Even without local
anaesthesia only minimal discomfort was experienced by four of {he
men, but patient UCA (3 mm cyst) required a spermatic cord block
The spermatoceles were tapped, but not completely emptied, and all
reaccumulated to their regular size and tone within a few days. There
was no obvious blood contamination of any of the aspirates.
Sperm preparation, IVF and 1CSI
Aspirates from the spermatoceles of patients UVA, YS1 and YS2
were prepared for IVF with discontinuous Percoll mini-gradients,
pentoxifylline and 2-deoxyadenosine as described by Ord et aL
(1992). For the ICSI cycles of patients YS2, VV and UCA, the fluid
was prepared with one-layer Percoll gradients only
The female partners were down-regulated with long-term gonado-
trophin-releasing hormone agonist, with subsequent ovarian stimula-
tion using gonadotrophins. Follicular aspiration was earned out under
sedation utilizing transvaginal ultrasound guidance. Following IVF
or ICSI, embryo transfer was to the uterus with luteal support from
progesterone (Hirsh et al, 1994). ICSI was carried out by the method
of Palermo et al. (1992) on metaphase II oocytes, selected after
removing the cumulus oophorus.
Results
Spermatocele aspiration
Fine needle aspiration of the spermatoceles yielded 0.25-12 ml
of fluid, and viable spermatozoa were present in all the samples,
generally with rapid progression by at least a few cells (Table
I). There were only occasional red blood cells. The sperm
concentration varied between 0.1 and 180 XI OVml (mean
56 XlOfyml), motility was 1-30% (mean 8.9%), progression
(mode) 0-1 to 2/4 (median 1/4, Macleod scale), and the
proportion of abnormal forms 10-90% (mean 40.3%).
1920
Sperm preparations
After centnfugation and separation, a few red blood cells were
evident m all the pellets. The IVF preparations of patients
UVA, YS1 and YS2 were enhanced, with a mean sperm
concentration of 13.1 XlOtyml, mean motihty 79.7%, progres-
sion 2-3/4 (mode), and 3-9% hyperactivation (Table II). The
ICSI preparations from patients YS2, W and UCA had
improved motility (mean 22.5%), but not progression (median
1/4)
IVF and ICSI
One cycle of standard IVF was carried out in couples UVA,
YS1 and YS2, the wives being aged 33, 29 and 44 years, with
17, 11 and 3 oocytes retrieved respectively (Table II). There
was no fertilization, although a 4-cell, probably parthenogenetic
embryo was transferred in couple YS1, with no evidence of
implantation. (In 1993, prior to our ICSI programme, this
couple was referred by us to another centre for ICSI using
spermatocele spermatozoa, and this resulted in a miscarriage
at 6 weeks gestation.) Couple YS2 (wife aged 44 years) also
underwent two ICSI cycles, in each of which one oocyte was
retrieved, with transfer of one embryo in the second cycle, but
no conception. Couple VV (wife aged 41 years) underwent
two ICSI cycles with transfer of two embryos from three
oocytes in the first, and three embryos from four oocytes in
the second cycle. A singleton pregnancy was established, with
a full-term normal delivery of a healthy 3.5 kg female infant.
Couple UCA (wife aged 29 years) had two ICSI cycles with
transfers of one embryo from three oocytes in the first, and
three of four embryos generated from seven oocytes in the
second cycle, but without implantation. Overall, there was
fertilization and cleavage of 11 of 19 metaphase II oocytes
injected (58%), with implantation of one of the 11 (9.1%)
embryos transferred.
Discussion
This small series demonstrates that natural spermatoceles,
identified in 4% of men with irreversible obstructive azoosper-
mia, can be ready sources of viable spermatozoa for ICSI, and
emphasizes that patients should be examined very carefully

Table n. Sperm preparations from spennatoceles: ln-vitro fertilization (TVF) and lntracytoplasmic sperm injection (ICSI) results
Patient Spermatozoa Mouhty Progression
(XlOfyml) (%) mode (1^)
UVA 87 57 2-3
YS1 30 82 2-3
YS2 05 100 -2
03 25
01 10
W 2 25 -2
40 40
UCA 10 5 ICSI
30 30 -2
*No pronuclear stage.
b
Pregnancy achieved
before considering more invasive methods of sperm retrieval.
It also supports the view that asymptomatic spermatoceles
should not be removed.
There are two previous reports of successful assisted concep-
tion from natural spermatoceles. Hanley (1957) achieved a
pregnancy following 13 cycles of intrauterine insemination
(IUI) with spermatozoa from a 1 ml subclinical cyst of the
caput epididymis discovered during scrotal exploration of a
patient with bilateral congenital vas aplasia. The spermatocele
collapsed after five percutaneous aspirations under local anaes-
thesia, and was reconstituted by surgically inserting a ball of
amnion. Wittemer et al. (1993) achieved a successful pregnancy
following subzonal insemination (SUZT) with spermatozoa
from the '10 ml' epididymal cyst of a man with epididymal
obstruction, with poor fertilization (6.4%) in preliminary IVF
cycles.
There is a clinical distinction between the unobtrusive,
small, round solitary spermatocele, near the head of the
epididymis, and the larger, multilocular, tense epididymal cyst,
above and behind the testis, which contains crystal-clear fluid,
with the typical Chinese lantern appearance on transillumina-
tion (Bailey, 1962). Wakeley (1943) found spermatozoa in 22
of 34, and Cooper et al. (1992) in five of six cysts of the
epididymis. Small cysts within or near the caput epididymis
may be seen in anatomical specimens (Hirsh, 1995) and on
scrotal ultrasound of normal (Leung et al., 1989), vasectomized
(Jarvis and Dubbins, 1989) and infertile (Nashan et al., 1990)
men, and could be a factor contributing to the success of PESA.
The potential for spermatocele aspiration in obstructive
azoospermia is obviously low, but we should endeavour to
adopt the least invasive procedure where possible (Tsirigotis
and Craft, 1995). Our patients were therefore fortunate that
their spermatoceles were discrete enough to be palpable. It is
also probable that PESA or MESA would have been difficult
due to post-surgical scarring which had resulted in contraction
of most of their caput epididymides. Some of these spermato-
celes might have been the beneficial complications of epididy-
mal surgery since they can arise following epididymectomy
(Badenoch, 1946). To minimize the risk of obliteration (Hanley,
1957), complete evacuation of the spermatoceles was avoided
by simply tapping them, and this explains the wide variations
in the fluid volume obtained. However, in view of their
Spennatoceles in irreversible obstructive azoospermia
Cycle Wife's No of No of
age (years) oocytes embryos
IVF 33 17 0
IVF 29 11 I1
rvr 44 3 0
ICSI 1 1
1CS1 1 U
ICSI 41 3 2
ICSI 4 3b
29 3 1
ICSI 7 4
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relatively small sizes, the aspirates were often quite large
(Table I), inferring communication with cavities in the rete
testis as illustrated by Griffiths (1893), and indeed tubular
ectasia of the rete seen on ultrasound is commonly associated
with epididymal cysts (Weingarten et al, 1992)
The characteristics of the spermatocele spermatozoa of our
patients were similar to those described by Cooper et al.
(1992) and Wittemer et al. (1993), with little difference from
rrucrosurgically retrieved epididymal spermatozoa (Hirsh et al.,
1994). Preparation for IVF improved the motility, but not the
function, because there was failure of fertilization in all cycles,
even though hyperactivation was observed in all three post-
preparation samples. The low fertilization rates of spermatocele
spermatozoa (6.4%) from the patient of Wittemer et al. (1993),
and from epididymal spermatozoa (10.6%) in our MESA
series (Hirsh et al, 1994), were not substantially different.
Spermatocele spermatozoa are therefore immature or dysfunc-
tional and ICSI is essential for successful assisted conception.
The reports of occasional pregnancies after IUI from natural
(Hanley, 1957) and artificial (Belker, 1991) spermatoceles
could be explained by sperm contact with the epididymal duct
due to surgical intervention, for the presence of epididymal
duct secretions was considered responsible for the fertilizing
ability of caput epididymal spermatozoa obtained by MESA
for standard IVF in cases of congenital vas aplasia (Pryor,
1987; Patnzio et al., 1994). Cooper et al. (1992) found no
evidence of biochemical secretions of the epididymal duct in
spermatocele fluid, consistent with a communication confined
to the rete testis via an efferent duct, Haller's superior aberrant
duct, or blind-ending branches of efferent ducts within the
caput epididymis (Yeung et al, 1991). Spermatoceles and
epididymal cysts are usually lined by a monolayer of flattened
or cuboidal cells, typical of these ducts, but this is occasionally
pseudostratified epithelium (Cameron and Pugh, 1976), infer-
ring a less common origin from the body or tail of the
epididymis, or Haller's inferior aberrant duct (Wakeley, 1943).
A role for the epididymis in sperm maturation (Cooper, 1993)
can be supported by the poor IVF results with spermatocele
spermatozoa, which, in most cases, have no contact with
epididymal duct secretions. Spennatoceles could thus be a
useful source of immature spermatozoa for physiological study
1921
A.V.Hirsh et aL

Weingarten, BJ , Kellman, G.M, Middleton, WD. and Gross, M l . (1992)

Acknowledgements
We wish to thank Christopher Steer, Judith Reid and Bromley
Hospitals Assisted Conception Unit for assistance with couple UCA
during their transport ICSI cycles.
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