Pediatric Case Report Posterior Urethral Polyp With Type I Posterior Urethral Valves: A Rare Association in a Neonate Krushnakumar V. Kesan, Rahul Kumar Gupta, Paras Kothari, Abhaya Gupta, Kedar Mudkhedkar, Ravikiran Kamble, and K. Vishesh Dikshit Urethral polyp is a rare cause of bladder outlet obstruction, voiding dysfunction, and hematuria in the pediatric age group. Urethral polyps are rarely associated with other congenital urinary tract anomalies. In this study, we report a case of solitary posterior urethral polyp with type I posterior urethral valve in a 7-day-old neonate presented with urinary retention and deranged renal function. The polyp was diagnosed on cystoscopy. Transurethral resection of the polyp with posterior urethral valve fulguration was performed. Pathologic assessment revealed a fibroepithelial lesion, which was consistent with congenital posterior urethral polyp. UROLOGY -: -e-, 2014. 2014 Elsevier Inc.
C
ongenital posterior urethral polyps (PUPs) are rare benign lesions that can cause variety of symptoms in the pediatric age group.1-3 They are often described in boys. Its occurrence in girls is exceptional. These benign fibroepithelial polyps are usually diagnosed in the first decade and are not known to undergo malignant degeneration. Urethral polyps are rarely associated with other congenital urinary tract anomalies. Diagnosis is made using voiding cystourethrography (VCUG) and cystoscopy.3-5 In this study, the authors report a case of urethral polyp with type I posterior urethral valves (PUV) in a 7-day-old neonate.
CASE REPORT A 7-day-old, 2.8-kg, male neonate who was an antenatally diagnosed case of bilateral hydronephrosis and hydroureter was referred to us with urinary retention. Medical records revealed that he was born of nonconsanguineous marriage, full term, vaginally delivered, and cried immediately after birth. On examination, the baby was afebrile, normotensive, and was having palpable bladder. Hematological investigations and blood gas analysis were normal. Renal functions obtained on day 7 of life were deranged (blood urea nitrogen 36 mg/dL and serum creatinine 3.2 mg/dL). Urine routine microscopy was normal, and urine culture showed no growth. Abdominal ultrasound showed bilateral moderate hydronephrosis, hydroureter, and distended trabeculated Financial Disclosure: The authors declare that they have no relevant financial interests. From the Department of Pediatric Surgery, Lokmanya Tilak Municipal Medical College (LTMMC) and Govt. Hospital, Mumbai, Maharashtra, India Reprint requests: Krushnakumar V. Kesan, M.Ch., Department of Pediatric Surgery, LTMMC and LTMG Hospital, Sion, Mumbai, Maharashtra, India. E-mail: krishnakesan@ rediffmail.com Submitted: December 9, 2013, accepted (with revisions): February 14, 2014
ª 2014 Elsevier Inc. All Rights Reserved
Figure 1. Voiding cystourethrography showing right grade V vesicoureteric reflux, trabeculated bladder wall, and dilated posterior urethra. (Color version available online.)
bladder with thickened wall. VCUG revealed right-sided grade V vesicoureteric reflux, trabeculated bladder wall, and dilated posterior urethra (Fig. 1). The patient was catheterized and was started on injectable antibiotic. Gradually, renal functions improved (blood urea nitrogen 18 mg/dL and serum creatinine 0.9 mg/dL). Subsequently, the patient was posted for cystourethroscopy electively. Cystourethroscopy revealed normal anterior urethra, type I PUV, single pedunculated polyp between bladder neck and verumontanum, severely trabeculated bladder, and bilateral ureteric orifices could not be visualized (Fig. 2A). The base of 0090-4295/14/$36.00 http://dx.doi.org/10.1016/j.urology.2014.02.017
1
Figure 2. (A) Cystourethroscopic view showing long pedicle urethral polyp in posterior urethra. (B) Resected polyp. (Color version available online.)
polyp was coagulated with bugbee, excised and delivered perurethrally (Fig. 2B). PUV fulgurated at 12-o’clock position, and patient was catheterized (Fig. 3). Histopathology confirmed transitional epithelium over a fibrous core suggestive of fibroepithelial polyp. Postoperative course was uneventful. Perurethral catheter was removed on postoperative day 2, and the baby was discharged. Check cystoscopy and fulguration of PUV were done after 4 weeks. The patient was asymptomatic at 6 months of follow-up, and follow-up abdominal ultrasound showed bilateral decrease in hydronephrosis and hydroureter with increased parenchymal thickness.
COMMENT Urethral polyp is rare occurrence, and most of them arise from posterior urethra near the verumontanum. PUPs are mostly found in male patients and are rarely reported in female patients, to date; only 6 girls with urethral polyp have been reported in the English literature.6 PUP can occur in newborns and adults older than 70 years but is more common in adults aged 30-50 years.3,4 Gleason et al reported the findings of 16 children with urinary polyps treated during a 35-year period, with 12 (75%) located at the urethra. PUPs are considered congenital in origin because large polyps are present in healthy newborns and infants. Downs postulated PUP as a protrusion from the wall of the posterior urethra. Other etiologic factors proposed are irritative, infectious, obstructive, and traumatic.7 PUPs are a rare cause of acute urinary retention or intermittent obstruction because of prolapse into the bladder or urethra. PUPs can cause hematuria in addition to voiding dysfunction in the pediatric age group. Kearney et al8 reported obstruction (48%) to be the most common presenting symptom followed by hematuria (27%) and retention (25%; n ¼ 48). The VCUG and ultrasonography examinations aid to diagnose the PUP, but cystourethroscopy is confirmatory and also aids to excise the lesion. In our case, PUP was not detected on ultrasonography and voiding cystourethrogram, but cystourethroscopy revealed PUP with type I PUV. 2
Figure 3. Type I posterior urethral valve. (Color version available online.)
Transurethral resection of a urethral polyp using bugbee fulguration or laser excision is the treatment of a choice. Open cystotomy is rarely required only when transurethral resection is not possible.9,10 Transurethral excision using perineal urethrostomy in a 3-week-old neonate has been reported by De Wolf and Fraley. In our case, PUP was managed by transurethral resection using bugbee fulguration. PUV was fulgurated in same setting. After extensive search in literature, we could not find any report of PUV with PUP in a neonate. References 1. Tayib AM, Al-Maghrabi JA, Mosli HA. Urethral polyp verumontanum. Saudi Med J. 2004;25:1115-1116. 2. Carrion Lopez P, Domınguez Hinarejos C, Serrano Durba A, et al. Congenital fibroepithelial polyps of the urethra. Cir Pediatr. 2010; 23:7-9. 3. Jain P, Shah H, Parelkar SV, et al. Posterior urethral polyps and review of literature. Indian J Urol. 2007;23:206-207. 4. Fathi K, Azmy A, Howatson A, et al. Congenital posterior urethral polyps in childhood—a case report. Eur J Pediatr Surg. 2004;14: 215-217. 5. Noviello C, Cobellis G, Romano M, et al. Posterior urethral polyp causing haematuria in children. Pediatr Med Chir. 2011;33:134-136. 6. Klee LW, Rink RC, Gleason PE, et al. Urethral polyp presenting as interlabial mass in young girls. Urology. 1993;41:132-133.
UROLOGY
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7. Downs RA. Congenital polyps of the prostatic urethra. A review of the literature and report of two cases. Br J Urol. 1970;42: 76-85. 8. Kearney GP, Lebowitz RL, Retik AB. Obstructing polyps of the posterior urethra in boys: embryology and management. J Urol. 1979;122:802-804.
UROLOGY
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9. De Castro R, Campobasso P, Belloli G, et al. Solitary polyp of posterior urethra in children: report on seventeen cases. Eur J Pediatr Surg. 1993;3:92-96. 10. Demircan M, Ceran C, Karaman A, et al. Urethral polyps in children: a review of the literature and report of two cases. Int J Urol. 2006;13:841-843.
3
C
ongenital posterior urethral polyps (PUPs) are rare benign lesions that can cause variety of symptoms in the pediatric age group.1-3 They are often described in boys. Its occurrence in girls is exceptional. These benign fibroepithelial polyps are usually diagnosed in the first decade and are not known to undergo malignant degeneration. Urethral polyps are rarely associated with other congenital urinary tract anomalies. Diagnosis is made using voiding cystourethrography (VCUG) and cystoscopy.3-5 In this study, the authors report a case of urethral polyp with type I posterior urethral valves (PUV) in a 7-day-old neonate.
CASE REPORT A 7-day-old, 2.8-kg, male neonate who was an antenatally diagnosed case of bilateral hydronephrosis and hydroureter was referred to us with urinary retention. Medical records revealed that he was born of nonconsanguineous marriage, full term, vaginally delivered, and cried immediately after birth. On examination, the baby was afebrile, normotensive, and was having palpable bladder. Hematological investigations and blood gas analysis were normal. Renal functions obtained on day 7 of life were deranged (blood urea nitrogen 36 mg/dL and serum creatinine 3.2 mg/dL). Urine routine microscopy was normal, and urine culture showed no growth. Abdominal ultrasound showed bilateral moderate hydronephrosis, hydroureter, and distended trabeculated Financial Disclosure: The authors declare that they have no relevant financial interests. From the Department of Pediatric Surgery, Lokmanya Tilak Municipal Medical College (LTMMC) and Govt. Hospital, Mumbai, Maharashtra, India Reprint requests: Krushnakumar V. Kesan, M.Ch., Department of Pediatric Surgery, LTMMC and LTMG Hospital, Sion, Mumbai, Maharashtra, India. E-mail: krishnakesan@ rediffmail.com Submitted: December 9, 2013, accepted (with revisions): February 14, 2014
ª 2014 Elsevier Inc. All Rights Reserved
Figure 1. Voiding cystourethrography showing right grade V vesicoureteric reflux, trabeculated bladder wall, and dilated posterior urethra. (Color version available online.)
bladder with thickened wall. VCUG revealed right-sided grade V vesicoureteric reflux, trabeculated bladder wall, and dilated posterior urethra (Fig. 1). The patient was catheterized and was started on injectable antibiotic. Gradually, renal functions improved (blood urea nitrogen 18 mg/dL and serum creatinine 0.9 mg/dL). Subsequently, the patient was posted for cystourethroscopy electively. Cystourethroscopy revealed normal anterior urethra, type I PUV, single pedunculated polyp between bladder neck and verumontanum, severely trabeculated bladder, and bilateral ureteric orifices could not be visualized (Fig. 2A). The base of 0090-4295/14/$36.00 http://dx.doi.org/10.1016/j.urology.2014.02.017
1
Figure 2. (A) Cystourethroscopic view showing long pedicle urethral polyp in posterior urethra. (B) Resected polyp. (Color version available online.)
polyp was coagulated with bugbee, excised and delivered perurethrally (Fig. 2B). PUV fulgurated at 12-o’clock position, and patient was catheterized (Fig. 3). Histopathology confirmed transitional epithelium over a fibrous core suggestive of fibroepithelial polyp. Postoperative course was uneventful. Perurethral catheter was removed on postoperative day 2, and the baby was discharged. Check cystoscopy and fulguration of PUV were done after 4 weeks. The patient was asymptomatic at 6 months of follow-up, and follow-up abdominal ultrasound showed bilateral decrease in hydronephrosis and hydroureter with increased parenchymal thickness.
COMMENT Urethral polyp is rare occurrence, and most of them arise from posterior urethra near the verumontanum. PUPs are mostly found in male patients and are rarely reported in female patients, to date; only 6 girls with urethral polyp have been reported in the English literature.6 PUP can occur in newborns and adults older than 70 years but is more common in adults aged 30-50 years.3,4 Gleason et al reported the findings of 16 children with urinary polyps treated during a 35-year period, with 12 (75%) located at the urethra. PUPs are considered congenital in origin because large polyps are present in healthy newborns and infants. Downs postulated PUP as a protrusion from the wall of the posterior urethra. Other etiologic factors proposed are irritative, infectious, obstructive, and traumatic.7 PUPs are a rare cause of acute urinary retention or intermittent obstruction because of prolapse into the bladder or urethra. PUPs can cause hematuria in addition to voiding dysfunction in the pediatric age group. Kearney et al8 reported obstruction (48%) to be the most common presenting symptom followed by hematuria (27%) and retention (25%; n ¼ 48). The VCUG and ultrasonography examinations aid to diagnose the PUP, but cystourethroscopy is confirmatory and also aids to excise the lesion. In our case, PUP was not detected on ultrasonography and voiding cystourethrogram, but cystourethroscopy revealed PUP with type I PUV. 2
Figure 3. Type I posterior urethral valve. (Color version available online.)
Transurethral resection of a urethral polyp using bugbee fulguration or laser excision is the treatment of a choice. Open cystotomy is rarely required only when transurethral resection is not possible.9,10 Transurethral excision using perineal urethrostomy in a 3-week-old neonate has been reported by De Wolf and Fraley. In our case, PUP was managed by transurethral resection using bugbee fulguration. PUV was fulgurated in same setting. After extensive search in literature, we could not find any report of PUV with PUP in a neonate. References 1. Tayib AM, Al-Maghrabi JA, Mosli HA. Urethral polyp verumontanum. Saudi Med J. 2004;25:1115-1116. 2. Carrion Lopez P, Domınguez Hinarejos C, Serrano Durba A, et al. Congenital fibroepithelial polyps of the urethra. Cir Pediatr. 2010; 23:7-9. 3. Jain P, Shah H, Parelkar SV, et al. Posterior urethral polyps and review of literature. Indian J Urol. 2007;23:206-207. 4. Fathi K, Azmy A, Howatson A, et al. Congenital posterior urethral polyps in childhood—a case report. Eur J Pediatr Surg. 2004;14: 215-217. 5. Noviello C, Cobellis G, Romano M, et al. Posterior urethral polyp causing haematuria in children. Pediatr Med Chir. 2011;33:134-136. 6. Klee LW, Rink RC, Gleason PE, et al. Urethral polyp presenting as interlabial mass in young girls. Urology. 1993;41:132-133.
UROLOGY
-
(-), 2014
7. Downs RA. Congenital polyps of the prostatic urethra. A review of the literature and report of two cases. Br J Urol. 1970;42: 76-85. 8. Kearney GP, Lebowitz RL, Retik AB. Obstructing polyps of the posterior urethra in boys: embryology and management. J Urol. 1979;122:802-804.
UROLOGY
-
(-), 2014
9. De Castro R, Campobasso P, Belloli G, et al. Solitary polyp of posterior urethra in children: report on seventeen cases. Eur J Pediatr Surg. 1993;3:92-96. 10. Demircan M, Ceran C, Karaman A, et al. Urethral polyps in children: a review of the literature and report of two cases. Int J Urol. 2006;13:841-843.
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