gms | German Medical Science

GMS German Medical Science — an Interdisciplinary Journal

Association of the Scientific Medical Societies in Germany (AWMF)

ISSN 1612-3174

Transverse testicular ectopia, a case report and review of literature

Transverse Hodenektopie, Fallbericht und Literaturübersicht

Case Report

  • corresponding author Mohammad Kazem Moslemi - Department of Urology, Kamkar Hospital, School of Medicine, Qom University of Medical Sciences, Qom, Iran
  • author Mohammad Reza Ebadzadeh - Department of Urology, School of Medicine, Kerman University of Medical Sciences, Kerman, Iran
  • author Shabir Al-Mousawi - Division of Urology, Department of Surgery, Al-Amiri Hospital, Kuwait City, Kuwait

GMS Ger Med Sci 2011;9:Doc15

doi: 10.3205/000138, urn:nbn:de:0183-0001386

Received: March 2, 2011
Revised: June 6, 2011
Published: July 7, 2011

© 2011 Moslemi et al.
This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by-nc-nd/3.0/deed.en). You are free: to Share – to copy, distribute and transmit the work, provided the original author and source are credited.


Abstract

Crossed testicular ectopia (CTE)/transverse testicular ectopia (TTE) is a rare but well known congenital anomaly, in which both gonads migrate toward the same hemiscrotum. It is usually associated with other abnormalities such as persistent Mullerian duct syndrome, true hermaphroditism, inguinal hernia, hypospadias, pseudohermaphroditism, and scrotal anomalies. About 100 cases of transverse testicular ectopia have been reported in published studies. We report a case of transverse testicular ectopia in an 8-month-old boy who presented with right inguinal hernia and nonpalpable left testis. On exploration, both testes were present in the right inguinal region. Bilateral orchiopexy was performed by crossing the left testis in the extra-peritoneal space and ipsilateral scrotal orchiopexy. The diagnosis could not be made preoperatively in most of reported cases.

Keywords: testis, undescended testis, cryptorchidism, testicular ectopia

Zusammenfassung

Gekreuzte Hodenektopie oder transverse Hodenektopie ist eine seltene, aber bekannte angeborene Anomalie, bei der beide Hoden in denselben Teil des Hodensackes (Hemiskrotum) wandern. Die Anomalie ist gewöhnlich mit anderen Fehlbildungen assoziiert wie z.B. persistierender Müller-Gang, echter Hermaphroditismus, Leistenhernie, Hypospadien, Pseudohermaphroditismus und Fehlbildungen am Hodensack. Etwa 100 Fälle mit gekreuzter Hodenektopie sind als Fallberichte publiziert worden.

Wir berichten über einen Fall von transverser Hodenektopie bei einem 8 Monate alten Jungen, der mit einer Leistenhernie rechts und einem nicht tastbaren linken Hoden vorgestellt wurde. Die Untersuchung ergab, dass beide Hoden in der rechten Leistenregion vorhanden waren. Eine bilaterale Orchiopexie mit Überkreuzung des linken Hodens im extraperitonalen Bereich und Fixation im seitengleichen Skrotum wurde durchgeführt. In den meisten berichteten Fällen konnte die genaue Diagnose präoperativ nicht gestellt werden.

Schlüsselwörter: Nicht abgestiegene Hoden, Kryptorchismus, Hodenektopie


Introduction

Transverse testicular ectopia (TTE) also named testicular pseudoduplication, unilateral double testis, and transverse aberrant testicular mal-descent, is a rare anomaly in which both testes descend or migrate through a single inguinal canal or hemiscrotum [1]. Often, the diagnosis is made during surgical exploration. In the literature more than 100 cases of TTE have been reported. We report a case of TTE discovered accidentally during surgery for right inguinal hernia and bilateral undescended testes.


Case presentation

The patient was a 7 months old infant, who admitted for the left nonpalpable testis, right undescended testis and right inguinal hernia. The patient was born with normal vaginal delivery, with a normal Apgar score. There was no history of illnesses or poor feeding or failure to thrive (FTT). General physical examination was unremarkable. Hematological examination and biochemistry lab data was normal. In external genitalia examination, the right testis was palpable inguinally with an evident hernia and the left hemiscrotum was empty. The patient scheduled for synchronous bilateral orchiopexy and right inguinal herniotomy. Firstly, left inguinal incision was done, and no testicular tissue or even spermatic cord was found. Then, right inguinal incision was made. The right testis with its overlying tunica vaginalis was found at the deep inguinal ring. After opening of the tunica, the fluid inside of it drained and testis was found. At the proximal part of the cord another testis was found (Figure 1 [Fig. 1]). The suspicion of left testis anorchia was changed to right side transverse testicular ectopia. Then, the plan changed to releasing of cords and bilateral orchiopexy. Two cords had common origin for 3-4 cm. The left cord was released at the site of its bifurcation, to the most proximal site, that distal to it releasing dissection was completed. Finally, the left testis was transferred with its cord to the left hemiscrotum easily and extra-peritoneally. Both testes were fixed in the sub-dartos pouch.


Discussion

TTE is a rare form of testicular ectopia. It was first reported by Von Lenhossek in 1886 [2]. More than 100 cases have been reported in the literature [3]. Several theories have been reported to explain the genesis of TTE. Berg [4] proposed the possibility of the development of both testes from the same genital ridge. Kimura [5] concluded that if both vasa deferentia arose from one side, there had been unilateral origin but if there was bilateral origin, one testis had crossed over. Gupta and Das [6] postulated that adherence and fusion of the developing Wolffian ducts took place early, and that descent of one testis caused the second one to follow.

An inguinal hernia is invariably present on the side to which the ectopic testis has migrated. On the basis of the presence of various associated anomalies, TTE has been classified into 3 types: Type 1, accompanied only by hernia (40% to 50%); type 2, accompanied by persistent or rudimentary Mullerian duct structures (30%); and type 3, associated with disorders other than persistent Mullerian remnants (inguinal hernia, hypospadias, pseudohermaphroditism, and scrotal abnormalities) (20%). According to that classification, our case was type 1 TTE. TTE associated with fused vas deferens is extremely rare. This condition may hinder the testis from being placed into the scrotum during orchiopexy [7].

The mean age at presentation is 4 years. The clinical presentation generally includes an inguinal hernia on one side and a contralateral or sometimes a bilateral cryptorchidism [8], [9]. Usually, the correct diagnosis is not made before surgical exploration, like our case, and it is revealed during herniotomy [9]. The diagnosis of TTE can be made preoperatively by using ultrasonography [10] by an experienced sonologist. Patients with TTE are at increased risk of malignant transformation. In fact, the overall incidence of malignant transformation of gonads is 18% [11]. There have been reports of embryonal carcinoma [12], seminoma, yolk sac tumor [13], and teratoma [11]. Walsh et al. [14] in their study concluded that testicular cancer was nearly 6 times more likely to develop in cryptorchid cases whose operations were delayed until after age 10 to 11 years. Wood et al. [15] in their study showed that risk of malignancy in undescended testicles decreased if their orchiopexy performed before ages 10 to 12 years. In 2% to 97% of patients with crossed testicular ectopia, disorders of the upper and lower urinary tract system have been reported [16]. Once diagnosis of TTE is made, a conservative surgical approach in the form of orchiopexy is recommended for the preservation of fertility [9]. Laparoscopy is useful for both diagnosis and treatment of TTE and associated anomalies [17]. Management for testicular ectopia is either trans-septal or extra-peritoneal transposition orchiopexy [18], [19], a search for Mullerian remnants and other anomalies, and long-term postoperative follow-up. There were two options for left orchiopexy in our case: extra-peritoneal orchiopexy and trans-septal orchiopexy. In the extra-peritoneal technique the testis is brought to the contra-lateral hemiscrotum after its passing near the root of penis. In the trans-septal technique the testis should traverse the scrotal mediastinum to be fixed in it. In the case of fused vas deferens, unlike our case, a trans-septal orchiopexy is recommended. It may be misdiagnosed as an inguinal hernia and intersex [17] or present as an irreducible hernia, requiring urgent surgery [20].


Conclusion

TTE is a rare anomaly of which the pathogenesis remains unclear, although experimental evidence suggests that the gubernaculums may play an important role. The ectopic testis can lie in the hemiscrotum, in the inguinal canal, or at the deep inguinal ring. The diagnosis should be considered when unilateral hernia and concurrent cryptorchidism of the contralateral side are present. In suspected cases, laparoscopy and ultrasonographic evaluation may be helpful in diagnosing of this condition before surgery. Transseptal orchiopexy is highly recommended to manage TTE. Laparoscopy, at present, is useful for both diagnosis and management of TTE and associated anomalies.


Notes

Competing interests

The authors declare that they have no competing interests.


References

1.
Barrack S. Crossed testicular ectopia with fused bilateral duplication of the vasa deferential: an unusual finding in cryptochidism. East Afr Med J. 1994;71(6):398-400.
2.
Von Lenhossek MN. Ectopia testis transversa. Anat Anz. 1886;1:376-81.
3.
Fourcroy JL, Belman AB. Transverse testicular ectopia with persistent Müllerian duct. Urology. 1982; 19(5):536-8. DOI: 10.1016/0090-4295(82)90614-8 External link
4.
Berg AA. Transverse ectopy of the testis. Ann Surg. 1904;40:223-4.
5.
Kimura T. Transverse ectopy of the testis with masculine uterus. Ann Surg. 1918;68(4):420-5. DOI: 10.1097/00000658-191810000-00009 External link
6.
Gupta RL, Das P. Ectopia testis transversa. J Indian Med Assoc. 1960;16:35:547-9.
7.
Chacko JK, Furness PD 3rd, Mingin GC. Presentation of fused vas deferens. Urology. 2006; 67(5):1085.e17-8. DOI: 10.1016/j.urology.2005.11.056 External link
8.
Feizzadeh Kerigh B, Mohamadzadeh Rezaei M. Crossed testicular ectopia: a case report. Urol J. 2005;2(4):222-3. Available from: http://www.urologyjournal.org/index.php/uj/article/view/229/226 External link
9.
Acikalin MF, Pasaoglu O, Tokar B, Ilgici D, Ilhan H. Persistent Mullerian duct syndrome with transverse testicular ectopia: a case report with literature review. Turk J Med Sci. 2004;34:333-6.
10.
Nam YS, Baik HK, Kim SJ, Lee HK, Park HK. Transverse testicular ectopia found by preoperative ultrasonography. J Korean Med Sci. 1998; 13(3):328-30.
11.
Berkmen F. Persistent müllerian duct syndrome with or without transverse testicular ectopia and testis tumours. Br J Urol. 1997; 79(1):122-6.
12.
Melman A, Leiter E, Perez JM, Driscoll D, Palmer C. The influence of neonatal orchiopexy upon the testis in persistent Müllerian duct syndrome. J Urol. 1981;125(6):856-8.
13.
Eastham JA, McEvoy K, Sullivan R, Chandrasoma P. A case of simultaneous bilateral nonseminomatous testicular tumors in persistent müllerian duct syndrome. J Urol. 1992;148(2 Pt 1):407-8.
14.
Walsh TJ, Dall'Era MA, Croughan MS, Carroll PR, Turek PJ. Prepubertal orchiopexy for cryptorchidism may be associated with lower risk of testicular cancer. J Urol. 2007; 178(4 Pt 1):1440-6. DOI: 10.1016/j.juro.2007.05.166 External link
15.
Wood HM, Elder JS. Cryptorchidism and testicular cancer: separating fact from fiction. J Urol. 2009; 181(2):452-61. DOI: 10.1016/j.juro.2008.10.074 External link
16.
Tolete-Velcek F, Bernstein MO, Hansbrough F. Crossed testicular ectopia with bilateral duplication of the vasa deferentia: an unusual finding in cryptorchism. J Pediatr Surg. 1988; 23(7):641-3. DOI: 10.1016/S0022-3468(88)80636-5 External link
17.
Gornall PG, Pender DJ. Crossed testicular ectopia detected by laparoscopy. Br J Urol. 1987; 59(3):283. DOI: 10.1111/j.1464-410X.1987.tb04627.x External link
18.
Esteves E, Pinus J, Maranhão RF, Abib Sde C, Pinus J. Crossed testicular ectopia. Sao Paulo Med J. 1995; 113(4):935-40.
19.
Pandey A, Gupta DK, Gangopadhyay AN, Sharma SP. Misdiagnosed transverse testicular ectopia: a rare entity. Hernia. 2009; 13(3):305-7. DOI: 10.1007/s10029-008-0434-5 External link
20.
Vaos G, Zavras N. Irreducible inguinal hernia due to crossed testicular ectopia in an infant. Hernia. 2004; 8(4):397-8. DOI: 10.1007/s10029-004-0232-7 External link