Pediatric Urology Bladder Neck Contracture After Antegrade Fulguration of Posterior Urethral Valves—Unusual Long-term Complication Rahul Janak Sinha, Vishwajeet Singh, Divakar Dalela, and S. N. Sankhwar Bladder neck contracture after antegrade fulguration of posterior urethral valves has not been reported in English studies to date to the best of our knowledge. A couple of reports have mentioned late-onset bladder neck contracture after retrograde valve fulguration. The present case report describes an unusual complication of bladder neck contracture that occurred long after antegrade fulguration of posterior urethral valves, which was done at an early age. This case report also discusses the possible mechanisms of bladder neck contracture, its management in brief, and highlights the importance of long-term follow-up of these patients. UROLOGY 73: 791–794, 2009. © 2009 Elsevier Inc.
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ntegrade fulguration of posterior urethral valves (PUVs) is a well-established technique.1 The chief advantage of this procedure is that urethral manipulation does not occur, reducing the risk of urethral injury. A pediatric endoscope is not needed, because antegrade fulguration can easily be performed with an adult-size cystoscope. The vision is excellent with the adult endoscope; a large image is seen, resulting in ease of performance by the surgeon. We report a rare and late event, long after antegrade fulguration in a child who underwent surgery for PUVs at 2 years of age at our center, after which he was lost to follow-up and presented with bladder neck contracture at about 10 years of age.
CASE REPORT A 10-year-old boy presented with complaints of dribbling of urine, frequency of micturition, and intermittent high-grade fever. The patient had been symptomatic for 6 months despite treatment by a general practitioner and came to us when his general condition had worsened. The patient’s attendants gave history of surgery performed at 2 years of age, and the previous medical records confirmed antegrade fulguration of PUVs at that age. Antegrade fulguration had been performed with a 20F endoscope under general anesthesia. No abnormality was detected on general examination. Ultrasonography of the abdomen showed normal upper tracts with urinary bladder capacity of 250 mL and a
From the Department of Urology, Chhatrapati Shahuji Maharaj Medical University (formerly King George’s Medical University), Lucknow, Uttar Pradesh, India Reprint requests: Rahul Janak Sinha, M.S. (General Surgery), Department of Urology, Chhatrapati Shahuji Maharaj Medical University (formerly King George’s Medical University), Lucknow, Uttar Pradesh, India. E-mail:
[email protected] Submitted: May 9, 2008, accepted (with revisions): July 21, 2008
© 2009 Elsevier Inc. All Rights Reserved
dilated posterior urethra. Uroflowmetry revealed poor urinary flow rate with low voided volume. A 6F infant feeding tube was passed per urethra but could not be negotiated into the urinary bladder. The retrograde urethrogram revealed a dilated prostatic urethra, and the contrast did not enter the bladder (Fig. 1A). At the time of admission, his serum creatinine was 1.4 mg/dL. The child was given broad-spectrum antibiotics. Initially, he underwent trocar suprapubic cystostomy, and a 16F Foley catheter was inserted into the bladder. Cystoscopy using a 7F pediatric endoscope was performed after 1 week. The cystoscope could not be negotiated beyond the verumontanum. A contracted bladder neck was visualized on antegrade cystoscopy (Fig. 1B). A guidewire was passed into the bladder in retrograde manner, and the bladder neck was dilated coaxially to 14F over the guidewire. Because the contracture was very tight, we doubted whether dilation would be effective in the long term; therefore, an antegrade bladder neck incision was done using a Collings knife (Fig. 2A). A urethral Foley catheter (14F) was kept in place for 1 week and the boy continued taking prophylactic oral antibiotics. The postoperative voiding cystourethrogram 1 month later revealed a normal-capacity bladder with Grade 4 reflux on the left side (Fig. 2B). At 1 year of follow-up, the patient was voiding with good urinary flow and had no urinary complaints.
COMMENT Definitive treatment of PUVs is surgical management.2 The valves are either fulgurated or excised or ablated using various devices, such as the Bugby electrode, cold knife, or laser, and a pediatric endoscope.3,4 Antegrade fulguration is also a viable alternative.1 At our center, antegrade fulguration is done by way of suprapubic cystotomy, because we have a limited range of smaller size endoscopes owing to economic constraints; a 0090-4295/09/$34.00 doi:10.1016/j.urology.2008.07.071
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Figure 1. (A) Preoperative retrograde urethrogram. (B) Bladder neck contracture (antegrade view through cystoscope).
Figure 2. (A) Contracted bladder neck incised using Collings knife. (B) Postoperative voiding cystourethrogram at 1 month.
common problem in many developing world countries. A cystoscope (20F) is introduced transvesically, and the valve is identified. A 6F feeding tube is introduced through the external urinary meatus and stationed distal to the valve, which helps in better identification and prevents overzealous fulguration. The Bugby electrode is introduced through the endoscope, and the valve is fulgurated. Afterward, a per urethral Foley catheter and a suprapubic Foley catheter are left in situ and removed later. Bladder neck contracture after antegrade fulguration of PUVs has not been previously reported. Androulakakis et al.5 conducted a study comprised of 18 patients previously treated for PUVs who underwent multiple urodynamic and videocystoscopy studies. During videocystoscopy, the bladder neck was found to be very thick and fixed in a semiclosed position.5 They concluded that despite early valve ablation, a large proportion of boys had gradual detrusor decompensation that was probably caused by secondary bladder neck obstruction, leading to obstructive voiding and detrusor failure. Misseri et al.6 also acknowledged that some patients had detrusor decom792
pensation probably resulting from some persistent obstructive process secondary to the bladder neck dyskinesia caused by prolonged anticholinergic therapy or a thickened and fixed bladder neck. The magnitude of bladder neck obstruction was greater in our patient compared to the patients mentioned in the earlier reports, probably because our patient underwent antegrade fulguration with an adult endoscope, which might have resulted in ischemic injury to the bladder neck from the shaft of the cystoscope.5,6 Whether the bladder neck contracture/stenosis is an ongoing process in patients with PUVs or it is due to iatrogenic injury during valve fulguration remains to be answered. The etiology may be multifactorial resulting from trauma, hypertrophy, or other unidentified factors yet to be discovered.
CONCLUSIONS Bladder neck contracture after antegrade fulguration of PUVs is a rare and late phenomenon. Bladder neck UROLOGY 73 (4), 2009
incision is the recommended treatment in such patients. Long-term close follow-up is needed in these patients. References 1. Zaontz MR, Firlit CF. Percutaneous antegrade ablation of posterior urethral valves in premature or underweight term neonates: an alternative to primary vesicostomy. J Urol. 1985;134:139-141. 2. Casale AJ. Posterior urethral valves and other urethral anomalies. In: Wein AJ, Kavoussi LR, Peters CA, et al., editors. CampbellWalsh Urology, 9th ed. Philadelphia: Saunders Elsevier, 2007: 3583-3603. 3. Dinneen MD, Duffy PG. Posterior urethral valves. Br J Urol. 1996; 78:275-281. 4. Zderic SA, Canning DA. Posterior urethral valves. In Docimo SG, Canning D, Khoury A, editors. The Kelalis-King-Belman Textbook of Clinical Pediatric Urology, 5th ed. London, UK: Informa Healthcare UK Ltd.; 2007:1059-1079. 5. Androulakakis PA, Karamanolakis DK, Tsahouridis G, et al. Myogenic bladder decompensation in boys with a history of posterior urethral valves is caused by secondary bladder neck obstruction? BJU Int. 2005;96:140-143. 6. Misseri R, Combs AJ, Horowitz M, et al. Myogenic failure in posterior urethral valve disease: real or imagined? J Urol. 2002;168:1844-1848.
EDITORIAL COMMENT The authors report a case of bladder neck contracture that presumably occurred as a sequela of antegrade instrumentation of the bladder neck to address posterior urethral valves. The initial valve fulguration was performed (by a different surgeon) in an antegrade fashion with a 20F cystoscope when the boy was 2 years old, and he presented to the authors’ clinic approximately 8 years later with this pathologic finding. Fortunately, the authors were able to manage this problem successfully with antegrade incision of the scar tissue. They are to be commended for presenting a case underscoring the potential pitfalls of excessive dilation of a neonatal or pediatric bladder neck. The antegrade approach to posterior urethral valves is a reasonable one. This is especially true if pediatric cystoscopic instruments are not available or if the patient’s anatomy precludes a retrograde approach, such as with an extremely small urethra in neonates and/or in the presence of urethral atresia. However, the antegrade passage of adult instruments may be as fraught with complications as the retrograde passage of these same instruments; just as a young boy’s urethral meatus and pendulous urethra cannot accommodate a 20F cystoscope, a young boy’s bladder neck should not accommodate a 20F cystoscope. When optimal pediatric instrumentation is not available, avulsion of the valves with a Fogarty or Foley catheter balloon are both viable alternatives, the feasibility of which have been previously reported.1,2 Before avulsing the valves, the balloon can be inflated under fluoroscopic guidance in the dilated prostatic urethra, thereby avoiding altogether a traverse of the bladder neck. The optimal short- and long-term management of posterior urethral valves remains the subject of ongoing discourse. However, when treating a disorder of such complexity, our objective ought not to be to trade one site of obstruction (valves) for another (the bladder neck). Jonathan D. Kaye, M.D., Emory University School of Medicine, Children’s Healthcare of Atlanta, Atlanta, Georgia UROLOGY 73 (4), 2009
References 1. Chertin B, Cozzi D, Puri P. Long-term results of primary avulsion of posterior urethral valves using a Fogarty balloon catheter. J Urol. 2002;168(4 Pt 2):1841-1843. 2. Kalicinski Z, Kansy J, Kotarbinska B, et al. Posterior urethral valves in infants—a therapeutic approach. Eur Urol. 1978;4: 182-184.
doi:10.1016/j.urology.2008.08.505 UROLOGY 73: 793, 2009. © 2009 Elsevier Inc.
REPLY The technique of posterior urethral valve fulguration using the antegrade approach is a well-established procedure,1 now mainly practiced in developing countries where pediatric endoscopes are unavailable because of economic constraints. At our center, numerous cases of posterior urethral valves have been treated successfully during the past decade using this technique. Thus, we are not entirely convinced by the editor’s assumption that this complication might have occurred owing to overdilation of the bladder neck by the 20F endoscope, even though this assumption might be viable. The editor also suggested that this complication could have been averted if a Fogarty catheter was used under fluoroscopic guidance, as described in published reports.2 However, in many of the developing world countries, fluoroscopic units are not available at some of the centers that treat such cases; therefore, the procedure recommended by the editor could be difficult to perform. Even if we agree that overstretching of the bladder neck by the 20F endoscope led to the bladder neck contracture, the logical question that arises is why was only this child affected and not others who underwent a similar procedure? Another pertinent question is why a similar complication has not been previously reported, although numerous studies of antegrade fulguration have been published in the past 2 decades. The etiology of bladder neck contracture after antegrade fulguration remains an enigma owing to the poor follow-up and patchy medical records. We speculate that the cause was multifactorial, as mentioned in the case report. The answer perhaps lies somewhere between a faulty operative technique and/or concomitant infection, along with other unidentified factors, and secondary bladder neck obstruction3,4 resulting from developmental defects associated with the posterior urethral valves. However, we agree wholeheartedly with the editor that one problem should not be traded for another and that, wherever possible, pediatric instruments should be used for valve fulguration in pediatric patients. This intriguing complication should always remind us that antegrade procedure comes with its own set of problems. We hope that as pediatric endoscopes become more cost-effective and affordable, the procedure of antegrade fulguration and its associated set of complications will become a relic of the past. Rahul Janak Sinha, M.S. (General Surgery), M.Ch. (Urology), and Vishwajeet Singh, M.S. (General Surgery), M.Ch. (Urology), Department of Urology, Chhatrapati Shahuji Maharaj Medical University (formerly King George’s Medical University), Lucknow, Uttar Pradesh, India 793
