CASE REPORT

Bilateral abdominal cryptorchidism with large left testicular

seminoma and failed right urogenital union
I. Vakalopoulos, G. Gkotsos, P. Kessidis, A. Malioris & D. Radopoulos
Urology Department of Aristotle University of Thessaloniki, Greece and ‘‘G. Gennimatas’’ General Hospital of Thessaloniki, Thessaloniki, Greece

Keywords Summary
Cryptorchidism—orchidopexy—testicular
cancer—testis exploration—urogenital This is a case report of a 32-year-old male with bilateral intra-abdominal crypt-
nonunion orchidism. A large seminoma had developed on the left testis with paraaortic
lymph nodes metastasis. The tumour was excised easily. The right testis was
Correspondence found just inside the deep inguinal ring, without the vas deferens in the sper-
Ioannis Vakalopoulos, 81A Egnatia Str, 54635
matic cord. The patient requested orchidopexy despite the well-explained risk
Thessaloniki, Greece.
Tel.: +30 2310269222;
of cancer development. Therefore, a second right groin incision was performed.
Fax: +30 2310340233; In the right inguinal canal, there was a normal-looking vas deferens that ended
E-mail: vakalj@otenet.gr in an atrophic nubbin of fibrous tissue without an epididymis. This is the sixth
case in the literature of failed urogenital union resulting in complete separation
Accepted: August 28, 2009 of testis and vas deferens. The patient underwent orchidopexy and had four
cycles of chemotherapy, which led to complete remission of the metastasis.
This case highlights the fact that an impalpable undescended testis and finding
of blind-end vas deferentia are not enough to establish the diagnosis of
vanished testis. The decision to undergo orchiectomy in cases of bilateral
cryptorchidism after puberty is also discussed. In our opinion, the choice
should be made by the patient after a discussion of the risk for cancer develop-
ment in the salvaged testis.

normal: 8–61 U l)1), with normal liver function. No

Case report
A 32-year-old adult male was referred to our department
due to bilateral cryptorchidism and a palpable abdominal
mass. Computed tomography (CT) investigation revealed
a 118-mm abdominal tumour with calcifications in front
of the aortic bifurcation. There was also a second 77-mm
abdominal tumour adjacent to the inferior mesenteric
artery. This tumour was not infiltrating or obstructing the
artery, and was lobular, well-defined and did not invade
the proximal organs (Fig. 1). Finally, there was a small
compact mass, probably representing the right testis, in the
right deep inguinal ring. The values of testicular tumour
markers were: a-fetoprotein (aFP) 1.97 ng ml)1 (normal
values: 0.00–10.90 ng ml)1); b-Chorionic gonadotrophin
(BhCG) 16.02 mIU ml)1 (normal value <5 mIU ml)1);
lactic dehydrogenase (LDH) 1332 U l)1 (normal: 135–225
U l)1). Additional laboratory data revealed mild anaemia
(haemoglobulin: 13.3 g dl)1; haematocrit: 39.8%) and a
slight increase in G glutamyltransferase (gGT) (71 U l)1;
other metastases were found in the abdomen, and the
chest CT and bone scan were free of pulmonary, bone or
lymph node metastases. The patient’s karyotype was
normal.
The patient was told that the tumour arising from the
left intra-abdominal testis had to be removed and the
options of either excising the right intra-abdominal testis
to offset the higher cancer risk or maintaining it and
performing orchidopexy were presented. The patient
decided to preserve his right testis and requested right
orchidopexy.
Left orchiectomy through a midline abdominal incision
was straightforward because the tumour did not invade
adjacent organs (Fig. 2). A large, fixed, palpable lymph
node mass in front of the aortic bifurcation was left in
place to be treated by radiation and/or chemotherapy,
depending on the original tumour histology. Palpation of
the liver and the rest of the abdominal organs did not
reveal signs of metastasis.

ª 2010 Blackwell Verlag GmbH Æ Andrologia xx, 1–4 1

Urogenital nonunion and seminoma in cryptorchidism
Fig. 1 Secondary metastatic paraaortic lymph node mass (77 mm).
Fig. 2 Completely mobile left testicular tumour visualised through a
midline incision.
In the right peritoneal cavity, there was a small testis
supplied by the testicular vessels just inside the deep
inguinal ring. Upon exploration of the right inguinal
canal, a large patent processus vaginalis was identified.
This was accompanied by a normal-looking vas deferens
that ended in a small atrophic nubbin of fibrous tissue.
Macroscopically, no epididymis was recognised at the
edge of the vas deferens. The small abdominal testis was
2 ª 2010 Blackwell Verlag GmbH
I. Vakalopoulos et al.
Fig. 3 Final CT after chemotherapy showing complete remission of
the metastatic mass.
delivered via the deep inguinal ring, accompanied by its
testicular vessels, but no connection could be identified
between the testis and the vas deferens.
On patient’s request, after dissection of the spermatic
cord to ensure adequate vessel length, we performed
orchidopexy of the right testis in the scrotum. Prior to
orchidopexy, three biopsies of the testis were obtained to
exclude microscopic cancer. The patient recovered well
without complications.
The pathology report of the testicular tumour revealed
classic seminoma. The biopsies of the right testis were
free of microscopic cancer, and no germ cells were found
(Sertoli cell-only syndrome). This was also confirmed
after mechanical fragmentation and collagenase elabora-
tion of testicular biopsy samples.
The patient underwent four cycles of chemotherapy
(Bleomycin, Etoposide and Carboplatinum). The final CT
investigation revealed complete remission of the paraa-
ortic lymph node metastasis (Fig. 3).
Discussion
This case represents an example of failed urogenital union
in a right intra-abdominal undescended testis along with
a large metastatic seminoma of the left testis. In the ninth
week of gestation, the cranial mesonephric tubules estab-
lish contact with the cords of the rete testis. While this is
occurring, the caudal mesonephric tubules regress. Union
takes place during approximately the 12th week of gesta-
tion, and the lumina of the rete tubules and cranial
mesonephric tubules become continuous during the 24th
week, when the latter become the ductuli efferentes.
In nonunion, the testis and vas deferens develop sepa-
rately, with epididymal structures attached to one or the
Æ Andrologia xx, 1–4
I. Vakalopoulos et al.
other, or to both. Michalek & Krepp (1972) have col-
lected 62 cases of complete or partial nonunion of testis
and vas from the literature up to 1972. Complete separa-
tion of the vas and testis with a portion of epididymis
attached to each appears to be very unusual, with only
five other cases reported in the literature (Mahour &
Woolley, 1972; Michalek & Krepp, 1972; Nowak, 1972;
Bergdahl & Andersson, 1981; Foley et al., 2005). These
cases support the hypothesis that testicular descent is
guided by the tail of the epididymis.
If surgical exploration of the inguinal canal for an
impalpable undescended testis reveals that the vas defer-
ens terminates blindly in a nubbin of tissue, it might be
assumed to represent the vestigial remains of the testis.
However, it is important to remember that cryptorchidism
is very often associated with epididymal abnormalities.
This is why in cases of unilateral or bilateral cryptorchi-
dism, open inguinal exploration that finds a blind-ended
vas deferens is not sufficient to establish a diagnosis of uni-
lateral or bilateral vanished testis. This is true with or with-
out the presence of epididymal structures. This diagnosis
would be safe only if blind-ended spermatic cord vessels
were found.
Laparoscopy is a well-established initial procedure to
look for an abdominal testis (Deans et al., 1995). If testic-
ular vessels are seen entering the deep inguinal ring, the
inguinal canal should be explored. If the vessels are
absent, irrespective of the presence of a vas deferens, the
peritoneal cavity should be carefully inspected for a testis.
Cryptorchidism is a well-known risk factor for testicu-
lar neoplasia, but the proportion of testicular cancer cases
that can be attributed to cryptorchidism is only approxi-
mately 5% (Møller, 2001). In a meta-analysis of 21 case–
control studies of the epidemiology of germ cell tumours,
the odds ratios for having testicular cancer in patients
with a history of cryptorchidism ranged from 3.5 to 17.1.
The overall relative risk was 4.8 (4.0–5.7) Dieckmann &
Pichlmeier (2004).
Bilateral cryptorchidism carries a higher risk of malig-
nancy than the unilateral condition (Møller, 2001). The
risk of developing a germ cell tumour is usually greater if
the testis is located intra-abdominally. In a recent study,
40% of persistent impalpable testes in adult men had
germ cell tumours (Carmingnani et al., 2005). Despite
this fact, as well as the frequent epididymal abnormalities
in undescended testes, our case is the only one in the
literature to present the simultaneous existence of failed
urogenital union and testicular cancer. This may be due
to the fact that the previous literature cases concern
young children with cryptorchidism, and testicular
tumours generally appear later in life.
Orchidopexy does not decrease the risk of developing
testicular cancer, but it makes examination of the testes
ª 2010 Blackwell Verlag GmbH Æ Andrologia xx, 1–4
Urogenital nonunion and seminoma in cryptorchidism
easier (Strader et al., 1988). For this reason, orchiectomy
is recommended by some authors for undescended testis,
especially with a testis located in the abdomen after
puberty. However, to exclude occult cancer in bilateral
cryptorchidism, some authors recommend orchidopexy
after performing bilateral biopsies (Kulkarni & Kamat,
1991). This recommendation holds even after puberty
and helps retain androgen production. In our opinion, in
cases of bilateral cryptorchidism where occult cancer has
been excluded, the patient should decide whether he pre-
fers bilateral orchidopexy or orchiectomy with lifelong
testosterone replacement. It is important to make sure
each patient has the data necessary to make an informed
decision.
In cases of nonobstructive azoospermia without or with
unilateral testicular cancer like the present case, there are
studies describing that except xenogeneic germ cell matura-
tion in vivo (Sofkitis et al., 2003), there are methodologies,
which have been proved sufficient to culture immature
diploid germ cells in vitro for therapeutic management of
men negative for haploid cells. Sofikitis et al. (2005)
reviewed these laboratory methods used for patients with
meiotic arrest in the stage of primary spermatocyte. In vi-
tro culture of spermatogonia or primary spermatocytes
recovered from men with early maturation arrest having
the potential to overwhelm the in vivo pre-meiotic block
opens a new perspective to the management of infertility
for these men.
However, there are potential genetic and epigenetic
risks using in vitro generated male haploid germ cells.
Transmission of animal infective agents, sex chromosomal
abnormalities, transmission of chromosomal or gene
defects and epigenetic alterations leading to chromosomal
abnormalities or tumour susceptibility and/or tumorigen-
esis may appear. (Sofikitis et al., 2005)
References
Bergdahl L, Andersson A (1981) The importance of a careful
search for intra-abdominal testes in cryptorchidism. Scand
J Urol Nephrol 15:153–155.
Carmingnani L, Morabito A, Gadda F, Bozzini G, Rocco F,
Colpi GM (2005) Prognostic parameters in adult impalpable
ultrasonographic lesions of the testicle. J Urol 174:1035–
1038.
Deans GT, O’Reilly PH, Brough WA (1995) Laparoscopy for
undescended testes: embryological considerations. Br J Urol
76:806–807.
Dieckmann KP, Pichlmeier U (2004) Clinical epidemiology of
testicular germ cell tumors. World J Urol 22:2–14.
Foley PT, Sparnon AL, Lipsett J (2005) Lipsett urogenital
nonunion – the case for laparoscopy for the impalpable
testis. Pediatr Surg Int 21:655–656.
3
Urogenital nonunion and seminoma in cryptorchidism
Kulkarni J, Kamat MR (1991) Tumors in undescended testis.
J Surg Oncol 46:257–260.
Mahour GH, Woolley MM (1972) Failure of urogenital union.
J Pediatr Surg 7:442–445.
Michalek HA, Krepp J (1972) Failure of urogenital union with
secondary amputation of the epididymal tail: a case report
with complete review of the literature. J Urol 107:436–439.
Møller H (2001) Epidemiological Studies of Testicular Germ
Cell Cancer. Thames Cancer Registry, Thesis, King’s College
London, United Kingdom (UK).
Nowak K (1972) Failure of fusion of epididymis and testicle
with complete separation of vas deferens. J Pediatr Surg
7:715–716.
Sofikitis N, Pappas E, Kawatani A, Baltogiannis D, Loutradis
D, Kanakas N, Giannakis D, Dimitriadis F, Tsoukanelis K,
4 ª 2010 Blackwell Verlag GmbH
I. Vakalopoulos et al.
Georgiou I, Makrydimas G, Mio Y, Tarlatzis V, Melekos M,
Miyagawa I (2005) Efforts to create an artificial testis: cul-
ture systems of male germ cells under biochemical condi-
tions resembling the seminiferous tubular biochemical
environment. Hum Reprod Update 11:229–259.
Sofikitis N, Kaponis A, Mio Y, Makredimas D, Giannakis D,
Yamamoto Y, Kanakas N, Kawamura H, Georgiou J, Schra-
der M, Lolis E, Giannakopoulos X, Loutradis D, Tarlatzis V,
Miyagawa I. (2003) Germ cell transplantation: a review and
progress report on ICSI from spermatozoa generated in
xenogeneic testes. Hum Reprod Update 9:291–307.
Strader CH, Wciss NS, Daling JK (1988) Cryptorchism.
Orchiopexy and the risk of testicular cancer. Am J
Epideriniol 127:1013–1018.
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