ORIGINAL ARTICLE
Ahead of print publication  

A 30-year retrospective study of rare ectopic seminal tract opening cases


1 Department of Urology, Nanjing BenQ Hospital Affiliated to Nanjing Medical University, Nanjing 210019, China
2 Department of Urology, Nanjing First Hospital, Nanjing Medical University, Nanjing 210006, China
3 Department of Urology, Gaogang Hospital of Traditional Chinese Medicine, Taizhou 225321, China
4 Fourth Clinical Institute, Nanjing Medical University, Nanjing 211166, China
5 Department of Urology, Jiangsu Cancer Hospital and Jiangsu Institute of Cancer Research and Nanjing Medical University Affiliated Cancer Hospital, Nanjing 210019, China

Date of Submission07-Nov-2018
Date of Acceptance31-Mar-2019
Date of Web Publication02-Jul-2019

Correspondence Address:
Hong-Fei Wu,
Department of Urology, Nanjing BenQ Hospital Affiliated to Nanjing Medical University, Nanjing 210019, China
China
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/aja.aja_63_19

PMID: 31274477

  Abstract 


Ectopic seminal tract opening is a rare congenital malformation. Until recently, there has been a lack of comprehensive reporting on the condition. The purpose of this retrospective study is to summarize the experience of diagnosis and treatment of this condition based on 28 clinical practice cases throughout the past 30 years. We conducted auxiliary examinations on such patients including routine tests, imaging examinations, and endoscopy. Among these 28 cases, there were ectopic opening of vas deferens into enlarged prostatic utricles (6 cases); ejaculatory ducts into enlarged prostatic utricles, Müllerian ducts cysts, and urethras (18 cases, 2 cases, and 1 case, respectively); and ectopic opening of the unilateral vas deferens and the contralateral ejaculatory duct into enlarged prostatic utricle (1 case). The size of the enlarged prostatic utricle, the type of ectopic seminal tract opening, and the opening's location effectively assisted in the selection of clinical treatment methods, including transurethral fenestration of the utricle, transurethral cold-knife incision, open operation, laparoscopic operation, and conservative treatment. Satisfactory effect was achieved during follow-up. In conclusion, a definite diagnosis and personalized treatment are especially important for patients with ectopic seminal tract opening.

Keywords: congenital malformation; ectopic opening; ejaculatory duct; enlarged prostatic utricle; vas deferens


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How to cite this URL:
Wu HF, Zhu JG, Lin JZ, Shi GD, Yu JQ, Xu WZ, Yu HB. A 30-year retrospective study of rare ectopic seminal tract opening cases. Asian J Androl [Epub ahead of print] [cited 2019 Sep 17]. Available from: http://www.ajandrology.com/preprintarticle.asp?id=261960

Hong-Fei Wu, Jia-Geng Zhu
These authors contributed equally to this work.





  Introduction Top


Ectopic seminal tract opening (ESTO) is a rare congenital malformation, which is mainly divided into two types, ectopic vas deferens opening (EVDO) and ectopic ejaculatory duct opening (EEDO). Since EVDO was first reported in 1895, only about 66 cases have been reported. Among the cases of EVDO, an ectopic opening into the ureter is relatively common,[1],[2],[3],[4] and ectopic opening into the bladder,[5],[6] the Müllerian duct cyst (MDC),[7],[8] the enlarged prostatic utricle (EPU),[9],[10] and renal collective system [11] could also be found in some rare cases. EEDO, first documented in 1939, is even rarer than EVDO, with only about 11 cases having been recorded until now.[12],[13],[14]

Various malformations often coexist with ESTO, including congenital imperforate anus,[15] rectourethral fistula,[10] ipsilateral renal dysplasia,[14],[16],[17] renal agenesis and dysmorphosis,[1],[8],[18],[19] hypospadias and cryptorchidism,[11],[15],[20],[21] and vesicoureteral reflux.[3],[22] Hence, associated symptoms due to these diseases are often present in patients with ESTO. In addition, other factors such as the type of ectopic opening, the grade and position of the EPU, and the unilateral or bilateral opening also affect clinical symptoms. Besides clinical manifestations, physical examination, urine tests, semen tests, and routine imaging examination are often conducted for diagnosis. Some special imaging examinations, such as percutaneous vasopuncture vesiculography and retrograde contrast radiography through the opening of the EPU under urethroscope, play a crucial role for a definite diagnosis.[23] However, the rare incidence and atypical symptoms of these conditions make clinical diagnosis and treatment difficult. In this study, we retrospectively analyzed 28 such cases, proposed a new classification of ESTO, and applied it in clinical practice to provide valuable references for diagnosis and treatment of the condition.


  Patients and Methods Top


Study population and clinical manifestations

From August 1985 to August 2015, 28 cases of ESTO in patients aged 2–48 years old were diagnosed and treated in the First Affiliated Hospital of Nanjing Medical University and Nanjing BenQ Hospital (Nanjing, China). The study protocol was approved by the ethics committees of the First Affiliated Hospital of Nanjing Medical University and Nanjing BenQ Hospital. Patients themselves or their parents when necessary provided written informed consent before enrollment.

The results of the physical examination, laboratory tests, and imaging are described in detail in [Supplementary Table 1 [Additional file 1]]. Briefly, the scrotum and perineum were carefully examined. Semen analyses were conducted after liquefaction at the clinics using the WLJY-9000 CASA system (Beijing Weili New Century Science & Tech. Deve. Co. Ltd., Beijing, China). B ultrasound and computed tomography (CT) on the urinary system were performed routinely. Enhanced CT or magnetic resonance imaging (MRI; 1.5T, Siemens Magnetom TrioTim, Munich, Germany) were performed in some cases.

Percutaneous vasopuncture vesiculography

Percutaneous vasopuncture vesiculography was carried out in 26 patients, all except 1 infant aged 2 years old and 1 patient with bilateral cryptorchidism. Briefly, the vas deferens on superficial anterior wall of scrotum was fixed and then the anterior wall was punctured with the 8th sharp needle at the most prominent and central part of the vas deferens. The 8th sharp needle was pulled out and inserted into the 6th blunt needle through the opening into the vas deferens immediately. If successful, 50% meglumine diatrizoate (XuDong HaiPu Pharmaceutical Co., Ltd., Shanghai, China) of approximately 2.5 ml was slowly injected into the unilateral vas deferens. Photographs were taken when patients felt urgency to urinate [Figure 1]a. The contralateral vas deferens was processed in the same way. Photography was delayed if necessary.
Figure 1: EVDO into the EPU. (a) Percutaneous vasopuncture vesiculography displayed bilateral EVDO into the body of EPU, absence of bilateral seminal vesicles, and no reflux of contrast medium to the bladder; (b) the EPU was verified by retrograde contrast urethrography through the central outlet of seminal caruncle. No reflux of contrast medium to bilateral vas deferens was observed; (c) percutaneous vasopuncture vesiculography showed that the left vas deferens opened ectopically into the bottom of the EPU and the left seminal vesicle and the distal segment of the right vas deferens was absent; (d) right vas deferens angiography during open surgery proved right EVDO into the bottom of EPU. (e) The bilateral vas deferens during open surgery was found to open ectopically into the bottom of the EPU (Grade II), most of the wall was removed, the left vas deferens was ligated, and the wall of EPU was partially preserved to ensure the patency of the right seminal duct. VD: vas deferens; EVDO: ectopic vas deferens opening; EPU: enlarged prostatic utricle.

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Staining urethrocystoscopy and contrast radiography through intubating opening of the EPU or the MDC

Such methods were employed when the ectopic opening into the MDC or EPU could not be precisely diagnosed through percutaneous vasopuncture vesiculography. Briefly, the central opening of the seminal caruncle and bilateral opening of the ejaculatory duct was found via urethrocystoscope and then diluted methylene liquid was injected slowly into the vas deferens. The case was identified as ESTO into the EPU when the liquid flowed from the central opening [Figure 1]b. The case was determined as having a normal opening when the liquid flowed from a bilateral opening. If the liquid did not flow out but needed to be pumped out, the case was diagnosed as ESTO into the MDC.

If urethrocystoscopy staining was carried out alone, the F5 ureteral catheter (Shangyi Kangge Medical Instruments Co., Ltd., Shanghai, China) was inserted into the EPU through the central opening of the seminal caruncle. Subsequently, 50% meglumine diatrizoate was injected into the ureter through the ureteral catheter, and photographs were taken with dynamic observation. If there was no opening in the middle of the seminal caruncle observed by naked eye, which was then confirmed by the F5 ureteral catheter, we punctured the center of the seminal caruncle with the needle under the cystoscope. The final step was to withdraw the fluid, followed by injecting the contrast medium to observe dynamically.


  Results Top


Test results

In our group, routine urine tests indicated that leukocytes and erythrocytes were found to be higher than normal in 23 samples. In addition, urine culture tests indicated that 15 samples were positive for Escherichia coli. Semen analysis of 26 cases revealed 11 cases of normal sperm, 10 cases of azoospermia, 4 cases of oligospermia and asthenospermia, and 1 case of necrospermia.

Diagnosis

Six patients were diagnosed with EVDO in the EPU [Figure 1]. Eighteen patients were proven to suffer from EEDO into the EPU [Figure 2], of which seven patients experienced complications of intracapsular calculus [Figure 2]a and [Figure 2]b. One case turned out to have unilateral EVDO and contralateral EEDO into the EPU [Figure 3]a. Two cases were verified as having EEDO into the MDC [Figure 4]a and [Figure 4]c. One patient was diagnosed with EEDO in the urethra [Figure 5]. Relevant diagnostic auxiliary examination results are listed in [Supplementary Table 1].
Figure 2: EEDO into the EPU. (a) Percutaneous vasopuncture vesiculography showed that ectopic opening of the EPS of the bilateral ejaculatory duct into the bottom of EPU (Grade I) with intracapsular stones; (b) contrast radiography exhibited left EEDO into the bottom of the EPU (Grade II). The left seminal vesicle was dilated. The right vas deferens failed to be punctured; (c) percutaneous vasopuncture vesiculography displayed that the middle segment of the left ejaculatory duct opened ectopically into the body of the EPU; (d) computed tomography showed left EEDO into the body of the EPU, accompanying by hypoplasia of the right seminal tract; (e) percutaneous vasopuncture vesiculography failed, but retrograde contrast radiography under the cystoscope proved right EEDO into the neck of the EPU (Grade II); (f) delayed contrast radiography after retrograde intubation displayed bilateral EEDO opened ectopically into the neck of EPU (Grade II); (g) percutaneous vasopuncture vesiculography revealed that right EEDO opened ectopically into bottom of the EPU and normal left ejaculatory duct. ED: ejaculatory duct; EEDO: ectopic ejaculatory duct opening; EPU: enlarged prostatic utricle; EPS: external-prostatic-segment.

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Figure 3: Unilateral EVDO and contralateral EEDO into the EPU. (a) Percutaneous vasopuncture vesiculography showed right EVDO and left EEDO into the bottom of EPU (Grade II); (b) retrograde contrast radiography indicated that the EPU and the contrast medium flowed into the bilateral seminal tract. EPS: external-prostatic-segment; EVDO: ectopic vas deferens opening; VD: vas deferens; ED: ejaculatory duct; EEDO: ectopic ejaculatory duct opening; EPU: enlarged prostatic utricle.

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Figure 4: EEDO into the MDC. (a) Percutaneous vasopuncture vesiculography displayed that the distal segment of the bilateral ejaculatory duct opened ectopically into the MDC (Grade II). No contrast medium flowed into the bladder through the urethra; (b) MDC was developed through the cystic puncture and contrast medium injection under cystoscope; (c) transurethral fenestration of the cyst was performed. ED: ejaculatory duct; EEDO: ectopic ejaculatory duct opening; MDC: Müllerian duct cyst.

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Figure 5: EEDO in the urethra. Percutaneous vasopuncture vesiculography showed right EEDO into the urethra accompanied by dysplasia of the seminal vesicle and the left vas deferens. Urethroscopy proved right ESTO into the urethra. ESTO: ectopic seminal tract opening; VD: vas deferens; ED: ejaculatory duct; EEDO: ectopic ejaculatory duct opening.

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Treatment

Two cases of EEDO in the MDC underwent transurethral fenestration of the cyst [Figure 4]c, and the surgical margin was between the central opening of the caruncle and the neck of the bladder. Among 24 adults, 2 patients whose EPUs were relatively smaller underwent transurethral cold-knife incision on the opening and anterior wall of the EPU [Figure 2]a, and transurethral fenestration of the EPU was performed in 19 patients whose EPUs were relatively larger. In addition, in some patients, intracapsular calculus measuring 3–7 mm were cleaned [Figure 6]. Open or laparoscopic resection of the cyst was performed in three cases of ESTO in the neck of the EPU when conservative treatment was insufficiently effective [Figure 2]e and [Figure 2]f. A 2-year-old infant diagnosed with bilateral EVDO in the EPU during operation underwent vasoligation on the left-epididymitis side and resection of most of the EPU [Figure 1]e with some cystic wall preserved for tubular reconstruction to ensure smooth flow of the contralateral seminal tract. Conservative therapy practiced in one patient proved to indicate right EEDO in the urethra [Figure 5].
Figure 6: Transurethral fenestration of the EPU. The operation was performed; multiple intracapsular stones were visible, varying 3–7 mm in size. EPU: enlarged prostatic utricle.

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Follow-up

Twenty-seven patients were followed up for up to 43 months. Preoperative symptoms of discomfort were remarkably relieved after surgery and anti-inflammatory treatment. Although semen quality improved in those who complained of infertility after surgery, no natural pregnancy was achieved. Fertility was obtained in two patients through intracytoplasmic sperm injection.


  Discussion Top


In the present clinical practice, ESTO is diagnosed mainly through intravenous urography, MRI, and voiding cystic urethrography.[23] Hence, cases of ESTO in the urinary tract can be identified effectively. On the contrary, we carried out percutaneous vasopuncture vesiculography in these cases, for which more cases of ESTO in the EPU and MDC can be discovered. Among these cases, complaints of hemospermia, hematuria, infertility, dysuria, or distending pain in the perineal emerged frequently, indicating that such patients often display these symptoms. Therefore, we need to consider the possibility of ESTO when diagnosing patients with such manifestations. Notably, percutaneous vasopuncture vesiculography should be considered when necessary.

Based on previous reports and our experience within the past 30 years, we proposed the following classification, which proved to be helpful to diagnose and treat these conditions in our clinical practice.

Overall classification

In general, they can be divided into three types according to the location of ESTO: (1) ESTO into the urinary tract, which can be located in the renal pelvis and calyx, ureter, bladder, and urethra; (2) ESTO into the Müllerian duct residual cyst, which can emerge in the MDC and the EPU; and (3) ESTO into the rectum, which was very rare.

Mainly, they can also be classified into EVDO and EEDO according to different parts of the seminal duct. Moreover, other types such as unilateral EVDO with contralateral EEDO and unilateral ESTO with normal or agenetic contralateral seminal tract have been found.

Types of ESTO into the EPU in our group

Five classification methods have been applied to these cases. First, according to the segment of the seminal tract for the ectopic opening, they can be divided into the following three types: (1) EVDO in the EPU is referred as Type I: 6 cases; (2) EEDO in the EPU is identified as Type II: 18 cases; and (3) unilateral EVDO with contralateral EEDO in the EPU is defined as Type III (also named mixed type): 1 case [Figure 3].

Second, in accordance with the segment of the ejaculatory duct for the ectopic opening, they can be divided into the following types: (1) external-prostatic-segment EEDO: 4 cases [Figure 2]a and [Figure 2]b; (2) middle-segment EEDO: 3 cases [Figure 2]c; and (3) distal-segment EEDO: 11 cases [Figure 2]e and [Figure 2]f.

Third, in accordance with the location of ESTO into the EPU, they can be classified into the following types: (1) bottom type: 10 cases ([Figure 1]c, [Figure 1]d, [Figure 1]e, [Figure 2]a and [Figure 2]b; (2) body type: 12 cases [Figure 1]a and [Figure 2]c; and (3) neck type: 3 cases [Figure 2]e and [Figure 2]f.

Fourth, based on the opening of the bilateral vas deferens and development of the contralateral seminal tract, they can be classified into the following types: (1) unilateral EVDO in the EPU with normal contralateral seminal tract is referred as Type Ia: none; (2) bilateral EVDO into the EPU is identified as Type Ib: 4 cases [Figure 1]a; and (3) unilateral EVDO into the EPU with agenetic contralateral seminal tract is defined as Type Ic: 2 cases [Figure 1]c.

Fifth, based on the opening of the bilateral ejaculatory duct and development of the contralateral seminal tract, they can be divided into the following three types: (1) unilateral EEDO into the EPU with normal contralateral seminal tract is referred as Type IIa: 10 cases [Figure 2]g; (2) bilateral EEDO into the EPU is identified as Type IIb: 5 cases [Figure 2]a and [Figure 2]f; and (3) unilateral EEDO into the EPU with agenetic contralateral seminal tract is defined as Type IIc: 3 cases [Figure 2]c and [Figure 2]d.

A simple diagram of the above classification is shown in [Supplementary Figure 1 [Additional file 2]], [Supplementary Figure 2 [Additional file 3]], [Supplementary Figure 3 [Additional file 4]], [Supplementary Figure 4 [Additional file 5]]. In practical clinical work, the types of ESTO into EPU are mainly based on different anatomical positions of the vas deferens and the ejaculatory duct opening's location in the EPU and development of the contralateral seminal tract. ESTO can be a single type or be different combinations of the above types.

Clinical application

This classification is important because the type of ESTO and location of the opening in the EPU are tightly connected with clinical manifestations and the selection of therapeutic methods.

Based on our clinical experience, patients with EVDO in the EPU exhibited more severe symptoms than those with EEDO in the EPU owing to a higher grade and larger size of the cyst. In our group, there were six cases of EVDO in the EPU and their cysts were in Grade II (IKOMA grade),[21] of which the largest one was 7 cm × 12 cm in size. These cases were all complicated with agenesis of the ipsilateral seminal vesicle [Figure 1]. In addition, patients diagnosed with external-prostatic-segment EEDO in the EPU displayed more serious manifestations than those with distal-segment EEDO in the EPU. Symptoms including infection, oligospermatism, asthenospermia, infertility, and perineal discomfort are also positively related with the grade of EPU.

For patients with bilateral ESTO in the bottom or body of the EPU, therapeutic methods should be selected according to the EPU grade. Specifically, patients with relatively smaller EPU should undergo transurethral cold-knife incision on the opening and anterior wall of the cyst, while transurethral fenestration of the EPU should be conducted on patients with a relatively larger one. In addition, excision of the EPU is not recommended because it may contribute to several unexpected conditions. For instance, the seminal tract located at the opening of the EPU may be removed; therefore, semen would not be excreted smoothly. However, if nonoperative treatments of severe symptoms have achieved little efficacy, surgery can also be considered. For patients with ESTO into the neck of the EPU [Figure 2]e and [Figure 2]f, open resection or under-laparoscopic resection of the EPU can be considered. In the surgery, we should protect the opening of the seminal tract at the neck of the EPU and carve the opening of the EPU into the urethra simultaneously to ensure smooth semen flow.

For patients with unilateral ESTO into the EPU with a normal opening of the contralateral seminal tract [Figure 2]g, therapeutic methods are mostly identical to those for patients with bilateral ESTO into the EPU, and normal opening should be protected during the operation. Patients with unilateral ESTO in the EPU with dysplasia or agenesis of the contralateral seminal tract [Figure 2]c and [Figure 2]d can be treated with the same approaches as those for patients with bilateral ESTO into the EPU.

Finally, for patients with ESTO in the EPU whose EPU is relatively smaller and related symptoms are mild, symptomatic treatment should be considered first and should be followed up thereafter.


  Conclusion Top


ESTO is rare and often associated with multiple malformations. Commonly, it is divided into three types named EVDO, EEDO, and mixed type (unilateral EVDO with contralateral EEDO). Locations of the ectopic opening include the urinary tract, the residual MDC, and the rectum. Moreover, it can be classified into unilateral and bilateral ectopic openings as well. Specifically, patients with unilateral ESTO can have normal or incompletely developed or absent contralateral seminal tract. In addition, EVDO is also associated with agenesis of the ipsilateral seminal vesicle. EEDO is divided into three types: external-prostatic-segment EEDO, middle-segment EEDO, and distal-segment EEDO. In addition, ESTO into the EPU can be classified into three groups consisting of the bottom type, body type, and neck type. In general, symptom data, physical examination, laboratory tests, imaging tests, and other methods are indispensable for diagnosis, among which percutaneous vasopuncture vesiculography is the most reliable. In addition, retrograde contrast radiography through opening of the EPU under urethrocystoscope can help us determine an exact diagnosis. Finally, treatment should adhere to the principle of individualization, aiming at eliminating symptoms, correcting malformations, protecting renal function, and restoring the semen outflow tract patency. Based on our experience and previous reports, a classification of ESTO has been proposed, which exerts a great impact on surgical method selection. Our study has some limitations. For example, we paid more attention to adults with hemospermia and infertility than to other patients and investigated more cases of ectopic opening into the EPU than those of ectopic opening into other structures.


  Author Contributions Top


HFW and JGZ conceived of the study, designed and carried out the surgeries, made contributions in acquisition and analysis of the data, and drafted the manuscript. JZL, JQY, and WZX helped draft the manuscript and assisted in the analysis of the data. GDS and HBY helped acquire and collect the data. All authors read and approved the final manuscript.


  Competing Interests Top


The authors declared no competing interests.

Supplementary Information is linked to the online version of the paper on the Asian Journal of Andrology website.

 
  References Top

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