Abstract
Urethral duplication is a rare disease with diverse clinical symptoms and more common in male; only fewer than 300 instances have been reported. In this case report, a 20-year-old male patient came to the urology polyclinic with two holes on the glans penis, accompanied by daily clear discharge in the upper hole. This complaint has been experienced since birth. The patient underwent an excision of the dorsal accessory urethra to correct penile anomalies.
Keywords: Urethral duplication, Congenital disease, Effman's classification, Congenital anomalies
1. Introduction
Urethral duplication is a rare condition with diverse clinical symptoms and is more common in males. To date, less than 300 cases of urethral duplication have been reported. It is typically identified in middle or early age.1 Urethral duplication can either be total or partial, and Effman's classification system is generally used to describe both types.2 Here, we described a case report of urethral duplication in a 20-year-old male patient.
2. Case report
A 20-year-old male patient presented to the urology polyclinic with two holes on the glans penis, accompanied by daily clear discharge in the upper hole. There was no urine coming out of the upper hole. Interestingly, urine was coming out of the lower hole. The patient has experienced these complaints since birth. There were no urinary symptoms such as frequent urination, urgency, dysuria, and suprapubic pain. At the physical examination, there were two holes in the glans penis, and the urethral meatus was presence on the ventral surface of the tip glans penis. Surgical options were discussed with the patient who decided to undergo surgery to correct penile anomalies. In the preoperative laboratory test, the patient had complete blood count, blood urea, and serum creatinine tests; all within normal limits. An urethrocystoscopy was performed at the start of the operation to aid in surgical decision-making. It was found that the dorsal external urethral meatus was smaller than normal. The urethra lumen also looked small, and the more proximal the lumen, the smaller it got. Additionally, the channel ended blindly with a length of approximately 7 cm. The patient underwent excision of the dorsal accessory urethra under general anesthesia (Fig. 1).
Fig. 1.
Intraoperation. Excision of the dorsal accessory urethra (A). The blind channel after excision (B).
3. Discussion
Urethral duplication is a rare defect that is frequently associated with other anomalies. It is usually identified in middle or early age and is more commonly found in males.1 There are several hypotheses on how embryogenesis process relates to urethral duplication, but none of them can explain all possible presentations. Urethral duplication may result from an aberrant Mullerian duct termination, growth stop of the urogenital sinus, or misalignment of the cloacal membrane termination with the genital tubercle.3
According to Effmann's classification, urethral duplication can be divided into three types (Fig. 2). Type 1 is an incomplete duplication of the urethra (accessory urethra). Type I A represents a duplicated urethra that opens on the penis and does not communicate with the urethra. In type I B, the duplicated urethra extends from the urethra and ends in the periurethral tissue (this is a rare type). In type II, there is a complete duplication of the urethra. In type II A-1, two urethras arise independently from the bladder and do not communicate. The second channel of type II A-2 develops from the first and flows independently into the second meatus, whereas type II B has two urethra openings in a single meatus. Type III duplication of the urethra is a component of partial or whole caudal duplication. Depending on the type of duplication, patient may be asymptomatic. The most usual type of urethral duplication is type IA, which is typically asymptomatic.2 Based on this classification system, our patient had type IA. Our patient chose to undergo surgery because he was uncomfortable with his complaints and penile anomaly.
Fig. 2.
Effmann's classification urethral duplication (taken from Tlili et al., 2021).
Voiding cystourethrography and retrograde urethrography are diagnostic methods. Urodynamic investigations are helpful for confirming the position of the functional urethra.3 In our case, only physical examination and urethrocystoscopy were performed on the dorsal accessory urethra due to limited facilities.
Patients with incomplete urethra duplication (type IA or IB) are not required to undergo surgery as the condition can be asymptomatic. However, if the patients demonstrates purulent discharges or a localized infection, the accessory urethra must be removed through surgery.4 Consequently, the anomaly experienced by our patient was addressed.
The surgical treatment varies according to the type of deformity. Gozar et al.5 reported two cases of type I urethral duplication, while Lopes et al.4 reported three cases. The duplicate (accessory) urethra was removed with positive functional and cosmetic outcomes. In our case, we successfully performed accessory urethra excision (type IA) on an adult patient.
Lopes et al. reported complications rate was 0% for type IA, 50% for type IIA1 (1/2 patients developed urethral stenosis), and 75% for type IIA2 (6/8 patients, with 6/8 developing urethral stenosis and 2/8 requiring bladder augmentation).4 Our patient had no complications 8 days after accessory urethra excision (Fig. 3).
Fig. 3.
Day 8 post-operation.
4. Conclusion
Urethral duplication is a rare disease. Despite limited resources in the developing countries, such as Indonesia, accessory urethra excision is well suited for our patient with type IA urethral duplication and provides excellent anatomical and functional results without complication after surgery.
Sources of funding
There was no funding for this case report.
Ethical approval
Due to the nature of case report, our institution's ethical approval was not necessary.
Consent for publication
Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.
Author contribution
Faisal Muhammad conceived the idea, designed the study, collected the patient data, and wrote the original draft.
Irvan Octavian and Muhammad Ilhamul Karim confirmed the diagnosis, performed the surgery, evaluated and directed the case management.
Irvan Octavian and Muhammad Ilhamul Karim reviewed and edited the final manuscript.
Irvan Octavian supervised the project.
All authors approved the final manuscript.
Registration of research studies
N/a.
Guarantor
Irvan Octavian.
Provenance and peer review
Not commissioned, externally peer-reviewed.
Declaration of competing interest
None.
References
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