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

Abstract

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 Study population and clinical manifestationsFrom 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. 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. INTRODUCTIONEctopic 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, 1-4 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]

A rare abnormality of ejaculatory duct opening in the bladder trigone in a 33‐year‐old male associated with primary infertility: Case report and literature review

“…In addition, we provide the findings of our literature review of published cases of ectopic openings of EDs (Table 1). We identified 24 such published cases (Khunovich et al, 2020; Lin et al, 2012; Reisman, 1977; Wang et al, 2009; Wu et al, 2020); of which only one reported case was of bilateral ectopic EDs openings in the bladder trigone observed in a 3‐year‐old child (Khunovich et al, 2020). Hence, to our knowledge, our case is the second ever reported of bilateral ectopia of EDs openings in the bladder trigone; and the first observed in an adult associated with primary infertility.…”

“…In terms of the anomaly, the literature review we undertook (Table 1) depicts that ectopic EDs most commonly open in the midline cyst, raising suspicion of an enlarged prostatic utricle (Lin et al, 2012; Wu et al, 2020). In a few rare cases, the ectopic ED invades the bladder trigone, bladder neck and urethra (Reisman, 1977; Wang et al, 2009; Wu et al, 2020). One patient had the ED ending in a large cystic dead‐end, compressing the bladder and the contralateral ED (Wang et al, 2009).…”

“…However, bilateral EDs opening into the bladder trigone was only reported once (Khunovich et al, 2020). Some ESTO classifications have been suggested, premised on the seminal tract segment involved, the ED segment opening ectopically, the ESTO location in the enlarged prostatic utricle (EPU), and the relationship between the ED or vas opening and the contralateral seminal tract (Wu et al, 2020). Interestingly, all the ESTO classification were restricted to ectopic openings of either VD or ED into the EPU without approaching ureteric or vesical ectopia (Wu et al, 2020).…”

Multiple vas deferens with polyorchidism and many congenital malformations in a symptomatic 11-year-old male patient: a rare case report

“…The congenital anomalies of the ductus deferens, which are rare, affect approximately 0.05% of the general population. This phenomenon has two main types, ectopic ejaculatory duct opening, and ectopic vas deferens opening [ 1 ]. In general, Ductus deferens' precise etiology is still unknown.…”

Invited Speakers

“…The function of testicles is usually impaired and their structure is abnormal, while external genitalia are often ambiguous. Until now only a single causative mutation responsible for Persistent Müllerian Duct Syndrome (PMDS) was identified in dogs(26). The mutation of Müllerian inhibiting substance type II receptor (MISRII) gene occurred in Miniature Schnauzers and was reported in different populations of this breed(12).…”