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Type IA Urethral Duplication CASE REPORT Bali Medical Journal (Bali Med J) 2019, Volume 8, Number 3: 587-591 P-ISSN.2089-1180, E-ISSN.2302-2914 Type IA urethral duplication: A case report Case Report Miftah Adityagama,1 Yonas Immanuel Hutasoit2* Published by DiscoverSys CrossMark Doi: http://dx.doi.org/10.15562/bmj.v8i3.1602 ABSTRACT Background: Urethral duplication is a rare congenital malformation stopped filling 2 cm from the meatus in the dorsal penoscrotal Volume No.: 8 mainly affecting men and boys. Although a number of theories have with a total dead-end impression as an accessory urethra and been proposed to describe this condition, the actual mechanism of there was no abnormalities in the principal urethra looked from this disorder is still not clear. The most frequent anomaly occurs in the the contrast filled the penile urethra, bulbous urethra, prostatic Issue: 3 sagittal plane, in which the duplicated urethra is in either dorsal or urethra, and bladder. That was the form of Effmann Type IA urethral ventral position in relation to the orthotopic urethra. The therapeutic duplication. The dorsal accessory urethra was excised entirely management of these conditions is complicated and depends on the without complication and he remains symptom-free eight months presence of symptoms as well as the type of anomaly. after surgery. First page No.: 587 Case Description: We present a case of urethral duplication in a Conclusion: In summary, urethral duplication is a rare clinical 2 years old male child. The malformation was characterised by the condition. It has many variants that were classified by Effman. presence of the meatus in the dorsal penoscrotal and accompanied Management depends on the anatomy of the duplication involved and P-ISSN.2089-1180 by the sign of infection in it. Urethrography showed contrast the patient’s clinical symptoms. Keywords: Double urethra, urethral duplication, Effman’s classification, reconstructive surgery. E-ISSN.2302-2914 Cite This Article: Adityagama, M., Hutasoit, Y.I. 2019. Type IA urethral duplication: A case report. Bali Medical Journal 8(3): 587-591. DOI:10.15562/bmj.v8i3.1602 1Department of Urology, Faculty INTRODUCTION patients. The treatment aims to preserve renal of Medicine Universitas Indonesia, function, achieve continence, and achieve the Cipto The urethral duplication, also known as double 2Mangunkusumo National General best possible cosmetic outcome. The ventral part urethra, is a rare anomaly occurring in pediatric of the urethra is always the functioning urethra, Hospital, Jakarta, Indonesia cases, this deformity is most often found in male 2Department of Urology, complete with sphincters and the urinary bladder. Fatmawati General Hospital, children while for female children it is very rare, Several methods may be used in obliterating the Jakarta, Indonesia with a previous literature recording no more than accessory channels, such as injection of sclerosing 40 cases of double urethra found in female chil- 1,2 agent or other ablative interventions. The tech- dren. The clinical entity comprised of a wide niques, however, are slowly being abandoned due spectrum with various anatomical variations. Thus, to the risk of corporeal thrombosis and fibrosis. there are several classifications used in defining Reconstruction by combining the ventral and each case of double urethra or urethral duplication, 3 accessory urethra parts may be utilized, although such as the Effmann classification system. It can it carries a potential risk for meatal stenosis and be complete or incomplete, the majority of cases obliteration of dorsal meatus. The current golden occur in the sagittal plane, further divided into standard is surgical excision of dorsal accessory dorsal and ventral duplication according to the urethra. The procedure itself needs to be done position of the ectopic urethra. Collateral duplica- delicately in order to avoid damage to the external tion without bladder duplication may also be found sphincter and neurovascular bundle.1 without accompanying bladder duplication. The relative rarity of the case combined with the wide spectrum of manifestation creates some difficulties CASE REPORT in pinpointing the exact pathophysiological mech- *Correspondence to: dr. Yonas Immanuel Hutasoit; anism. Several literatures had suggested that there A 2-years-old male patient was referred to urology Department of Urology, Fatmawati were multiple mechanisms that may cause urethral polyclinic at Fatmawati General Hospital with the General Hospital, Jakarta, Indonesia; duplication, each possibly related with different chief complaint there was whitish discharge from [email protected] types of manifestation.1 a hole in the dorsal penoscrotal. That condition Individually tailored management is required has been realised by the parents since the last Received: 2019-09-11 due to the spectrum above of clinical manifesta- one month, but they have noticed the small hole 1,4 Accepted: 2019-10-10 tions in cases of urethral duplication. The general in the dorsal penoscrotal since the patient was Published: 2019-12-01 consensus is, presently, to treat only symptomatic 3-months-old. Previously they took the patients Open access: www.balimedicaljournal.org and ojs.unud.ac.id/index.php/bmj 587 CASE REPORT to a paediatrician because the patients were fussy when urinating and looked reddish around the hole then stated there was inflammation. There was no difficulty in urinating. Good emission of urine, not branched, and there was no urine comes out of a hole in the dorsal penoscrotal. There were no abnormalities in growth and development or other congenital abnormalities. From the physical examination we found a normal external urethral meatus on the tip of the glans penis (Figure 1a) and the second meatus with a diameter of 2 mm in the Figure 1 The normal external urethral meatus dorsal penoscrotal (Figure 1b). The two testicles (a) and the second meatus in the dorsal were found normal. penoscrotal (b) During urethrography and urethroscopy exam- ination, it was obtained a meatus from the dorsal penoscrotal as long as 2 cm, a pinpoint hole and debris appeared, and the wire could not be inserted. Contrast stopped filling 2 cm from the meatus in the skin, with a total dead-end impression (Figure 2a). 7 Fr ureteroscopy sheath was inserted through the external urethral meatus on the tip of the glans penis; urethra, verumontanum, bladder neck and bladder were found normal. There were no holes along the urethra; the right and left uret- Figure 2 Urethrography showed contrast stopped eral opening were identified. Contrast filled the filling 2 cm from the meatus in the dor- penile urethra, bulbous urethra, prostatic urethra, sal penoscrotal, with a total dead-end and bladder. There was no stenosis and there were impression (a), and contrast filled the no tracts to another place (Figure 2b). penile urethra, bulbous urethra, pros- Patients had no other complaint besides a whit- tatic urethra, and bladder (b) ish discharge from a meatus in the dorsal peno- scrotal which is then followed by the presence of pain and redness around it. The patient was then planned for exploration and excision of the tract from the meatus in the dorsal penoscrotal. Surgical Technique Patient was positioned under general anaesthe- sia. The aseptic and antiseptic procedure was conducted at surgical site and the surrounding area. Evaluation of genitalia external was carried out; the Figure 3 Penile degloving and identification the external urethral meatus was located at the tip of dorsal tract (a), the dorsal tract appears glans penis, two testicles were found normal, and to the symphysis (b) there was a second meatus at the dorsal penoscrotal. Mattress stitch on glans penis using suturing thread prolene 4-0. Foley catheter 8 Fr was inserted at the opening of external urethral meatus at the tip of the glans penis. Circumglandular incision was made following marker and penile deglov- ing was conducted, identification of the dorsal tract (Figure 3a). The dorsal tract appears to the symphysis (Figure 3b). Excision of the dorsal tract Figure 4 Excision of the dorsal tract and released and released up to the symphysis, obliteration of the up to the symphysis (a), excised tissue tissue appeared (Figure 4). The tissue was sent to be of the dorsal tract as long as 2 cm (b) 588 Published by DiscoverSys | Bali Med J 2019; 8(3): 587-591 | doi: 10.15562/bmj.v8i3.1602 CASE REPORT examined into the anatomical pathology laborato- bones.5 Arena et al.,6 found that in 60% of cases rium. Raw surface was the covered using the byars is accompanied by genitourinary malformation flap technique with PDS 5-0 interrupted (Figure 5). such as ureteropelvic junction obstruction, extra rotation of the penis, vesicoureteral reflux, renal RESULT ectopia, renal agenesis, or posterior urethral valves. There was no additional abnormality in our patient. The results of examination from anatomical pathol- Various theories have been proposed to explain ogy of the tissue shown a picture of the urethra the embryological development of urethral dupli- with a chronic inflammatory reaction and no signs cation, including disorders of mesoderm develop- of malignancy appeared. Ten days after surgery ment, abnormal Mullerian ducts, ischemia during (Figure 6), patient came back to our urology poly- embryogenesis, and defects in the development of clinic. Physical examination showed good surgical the urogenital sinus.7–10 Casselman and Williams10 wound healing, there was no discharge from the suggested that a partial failure or an irregularity surgical wound in the dorsal penoscrotal was found of the ingrowth of the lateral mesoderm between as before and there were no problems with voiding. the ectodermal and endodermal layers of the cloacal membrane in the midline accounts for the DISCUSSION forms with a dorsal epispadias channel. Das and Brosman8 stated that abnormal termination of the Double urethra or urethral duplication is a congen- Mullerian duct was responsible for the development ital disorder which rarely happens. Approximately of urethral duplication.
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