Urethral Stricture Disease in Children

0022-5347/81/1265-0650$02.00/0

Vol. 126, November

THE JOURNAL OF UROLOGY

Printed in U.S.A.

Copyright© 1981 by The Williams & Wilkins Co.

URETHRAL STRICTURE DISEASE IN CHILDREN MARK W. HARSHMAN,* WILLIAM J. CROMIE, ALAN J. WEIN

AND

JOHN W. DUCKETTt

From the Division of Urology, Hospital of the University of Pennsylvania and Children's Hospital of Philadelphia, Philadelphia, Pennsylvania

ABSTRACT

We review our experience with urethral stricture disease in 25 children who were seen between 1970 and 1979. Trauma was the most common etiology (48 per cent). Excellent results were obtained by 1-stage repair and multistage skin inlay urethroplasty. An average of 3.8 procedures per patient was required for multistage repairs versus 1.1 procedures per patient for 1-stage repair. Dilation alone averaged 1.7 procedures per patient and was successful in only 20 per cent. Dilation is unacceptable for management of most strictures in children and urethroplasty should be considered early in the treatment plan. One-stage urethroplasty, when applicable, is preferable to multistage repair. Seven children had a fractured pelvis with prostatomembranous urethral disruption, which was complete in 6 cases and partial in 1. Successful anatomical results were achieved ultimately in all of these cases. No patient who had erections preoperatively was impotent afterward. One child has been totally incontinent since the accident and 3 others have stress incontinence, only 1 of whom requires treatment. There have been significant advances in the management of adult urethral stricture disease during the last 2 decades. Multistage repair, once the preferred treatment for most strictures, is now reserved for specific indications and single stage repairs have become increasingly popular. The same trend is true for the pediatric population as well. However, urethral stricture disease in children has been reviewed infrequently and most of the articles deal primarily with posterior urethral strictures. 1- 10 Herein we review our experience with urethral strictures at the Children's Hospital of Philadelphia from 1970 to 1979. Strictures secondary to hypospadias and meatal stenosis have been excluded.

TREATMENT

A summary of the treatment used is listed in tables 2 and 3. Dilation was the initial procedure performed in 10 children and stricture recurred in 8. In 7 patients excision of the stricture and end-to-end anastomosis were performed (fig. 2). 11 These strictures were located in the anterior urethra (1), bulbous urethra (3) and membranous urethra (3). Two of the membranous strictures treated in this fashion were approached from a combined abdominal-transperineal approach. In 6 patients with bulbous urethral strictures treatment consisted offull thickness skin grafts (fig. 3). 12 Penile skin was used in all cases and the graft sizes ranged from 2.5 X 0.5 cm. to 8.5 X 1.5 cm. The last dilation was 3 to 5 months before repair in all cases. Of these patients 3 had multiple strictures, including 1 who had 3 strictures that spanned a total distance of 7 cm. In 7 patients a multistage skin inlay procedure was used for strictures located in the membranous (4), bulbous (1) and anterior urethra (1) (fig. 4). 13- 15 One child had strictures in all 3 locations. The interval between the first and second stages ranged from 4 to 42 months, averaging 15 months. Two patients had a transpubic membranous urethroplasty for complete prostatomembranous urethral disruption secondary to severe pelvic fractures (fig. 5). 16 A 7-year-old boy who had undergone a suprapubic tube and primary catheter realignment elsewhere was sent to us the next day. Subsequently, we applied 400 gm. of traction to the catheter at 45 degrees for 6 days. 4 Nevertheless, a long membranous urethral stricture developed, which was managed successfully by a transpubic repair 3 months later. An omental pedicle graft was used in 2 patients with gratifying results. In 1 case a transpubic membranous urethroplasty was done, while in the other boy a combined abdominaltransperineal approach was used with excision of a membranous stricture and reanastomosis.

MATERIALS AND METHODS

Our study consists of 25 male patients, ranging in age at the time of diagnosis from 2 to 20 years (fig. 1). The presenting signs and symptoms are listed in table 1. The preoperative evaluation consisted of a voiding cystourethrogram and/or cystoscopy. These studies were abnormal in all children studied. An excretory urogram was obtained as part of the evaluation in 19 patients and was abnormal in 9. Dilated upper tracts were seen in 7 children, 4 of whom had dilated systems prior to the onset of the strictures following urethral valve surgery. A unilateral poorly functioning kidney was seen in 2 trauma cases, which, subsequently, required nephrectomy. There were 12 cases of traumatic strictures, including 6 from straddle injuries, 5 from vehicle injuries and 1 from a 5-story fall. Of 11 iatrogenic cases 5 occurred after cystoscopy, 5 after transurethral surgery and 1 after repair of a high imperforate anus. There were 2 cases of inflammatory strictures, including a 16-year-old boy with a history of urethritis and urethral discharge 2 years before presentation and an 11-year-old boy with a 1½-year history of hematuria and inflammatory polyps. Twenty patients had a single stricture, while 5 had multiple strictures. The strictures were located in the anterior urethra in 4 patients, bulbous urethra in 13 and membranous urethra in 10. One patient had strictures in all 3 locations.

RESULTS

Accepted for publication December 19, 1980. Read at annual meeting of Mid-Atlantic Section, American Urological Association, Hot Springs, Virginia, October 1-5, 1980. • Current address: 335 Washington Ave., Downingtown, Pennsylvania 19335. t Requests for reprints: Division of Urology, Children's Hospital of Philadelphia, 34th St. and Civic Center Blvd., Philadelphia, Pennsylvania 19104.

Followup was available in all but 1 patient and varied from 3 to 96 months, averaging 28 months. The results are listed in tables 2 to 4. A successful final result implied normal x-ray studies (done in 21 patients) and/or endoscopy (done in 4 patients), and lack of symptoms. No child currently requires dilation. Seven patients had posterior urethral strictures following pelvic fractures with prostatomembranous urethral disruption,

650

651

URETHRAL STRICTURE DISEASE IN CHILDREN

whi.ch were partial in I case and complete in 6. Patient 6 was incontinent after the original accident and remained so after an otherwise successful urethroplasty. Vesicostomy was the initial µv,c,c-,n .. ,c;w,ca.,c procedure in this case. Patients 2, 4 and

8

9 10 11 12 AGE (YrnRS)

13

14

15

16

17

18

19

COMPLICATIONS

Fm. 1. Age of children at diagnosis TABLE

TABLE

The only complication was recurrent stricture, which involved 4 patients. Additional procedures were necessary in each case, including l patient who had undergone a transpubic as described previously. Of the 7 children who underwent multistage inlay procedures 2 strictures developed after the first stage repair_ One case was treated successfully with dilation alone and l required formal revision. Two failures occurred after the second stage procedure, of which 1 was treated successfully dilation but the other required formal repair. Extravasation of contrast material noted on a tourethrogram 7 to 10 days postoperatively was not cuJ,1,:,i,ws1 a complication unless operative intervention became necessary_ This finding was seen in 3 patients and was treated remser

1. Signs and symptoms

Acute urinary retention Dysuria Hesitancy Hematuria Weak stream Flank pain Enuresis Blood in shorts Fever of unlmovvn origin Frequency Daytime and nighttime incontinence Recurrent urinary tract infection Periurethral abscess Evaluation for undiversion

20

7 have postoperative stress incontinence, which is moderate l (controlled by imipramine) and mild in 2 (no treatment). The 3 other patients in our series with membranous strictures have no incontinence following surgical repair. Of these 7 patients 5 have had definite post-repair erections, 1 is indeterminant and a 9-year-old boy has not had an erection since the accident 18 months previously. This child had sustained multiple severe injuries. Analysis of the various procedures indicates excellent results with excision and reanastomosis, full thickness skin and multistage skin inlay procedures (table 3)_ Transpubic membrnnous urethral repair was successful in 1 of 2 children. Subse .. quently, the transpubic failure was repaired successfuHy with a multistage skin inlay procedure. The number of separate cedures necessary to achieve these results, however, was ent for each method of repair (table 3). ru,cuL>ui,:u the final success rate for multistage repair was 100 per cent an average of 3.8 procedures per patient was required. Similar have been reported in adults. 13

10 6 6 6 6 4 4

4 3

2 2

2 1 3

TABLE

2. Summary of treatment No. Cases

I-Stage urethroplasty: Excision and :reanastomosis 11 FuU thickness patch graft 12 Transpubic repair 16 Totals Multistage skin inlay procedure: 1'- 15 First stage Second stage ' Responded to catheter drainage.

7 6

2 15

7 7

Restenosis

Fistula''

Technique

0

0

0 1

1 1

1

2

2 2

0 1

Excision and reanastomosis Full thickness patch graft Transpubic repair Multistage skin inlay procedure Dilation

3. Procedures performed

No. Pts.

No. Procedures

Final Success No.(%)

No. Procedures per Pt.

7

7

7 (100)

6

6

6 (JOO)

2 7

4 27

1 (100) 7 (100)

2 3.8

10

17

2 (20)

L'I

retrograde and anterograde urethrogram shows traumatic membranous urethral stricture. B, :result following exci.sion and

652

HARSHMAN AND ASSOCIATES

Fm. 3. A, retrograde urethrogram shows preoperative bulbous urethral stricture. B, result following full thickness patch graft urethroplasty

Fm. 4. A, retrograde urethrogram shows traumatic membranous urethral stricture. B, result following multistage skin inlay procedure

tion of the catheter for 1 week. A good result was obtained in all of these patients without further surgery. Although extravasated urine has been associated with recurrent stricture 17 none of our patients had this problem. DISCUSSION

The spectrum of stricture disease in our series of 25 children basically is similar to that in adults. The most common etiology was trauma (48 per cent) as is the case in most adult 18 and previously reported pediatric series. 1' 2 Unfortunately, iatrogenic injury continues to be a common cause of childhood stricture disease. Our experience indicates that strictures following instrumentation are often due to inappropriate dilation or cystoscopy with too large of an instrument. Three children in our series had been investigated for hematuria, which was gross in only 1 patient. Cystoscopy for hematuria in children should be reserved for clear indications 19 and extreme care should be exercised in the management of inflammatory polypoid lesions that occasionally are seen in these cases. In 1 of our patients a stricture developed after transurethral fulguration of such a lesion. There were no congenital strictures in our series. These congenital membranes, as described by Stephens, 20 are a rare cause of urethral obstruction in boys and are grossly overdiagnosed. Often a congenital stricture is diagnosed in boys with ill-defined voiding symptoms. We recommend avoidance of the term congenital urethral stricture altogether. Perhaps in this way frequently needless urethral dilation could be avoided. There are numerous options for managing urethral stricture disease, including dilation, primary catheter realignment,4 excision and reanastomosis, 11 internal urethrotomy, 21 multistage repairs, 13- 15 full thickness patch graft, 12 full thickness tube graft, 22 island patch graft, 23 and transpubic urethroplasty. 16 The

type of repair selected is dependent on a number of factors, such as the length, location, number and character of the stricture(s) as well as the presence of infection, associated injuries, condition of the patient, experience of the surgeon and type of previous repair, if any. The goal should be to select a repair that gives the best results with the least morbidity and requires the fewest surgical procedures. Dilation often is the initial treatment for urethral strictures but it is rarely permanently successful (only 2 of 10 in our series). Other series of children reveal similarly poor results. 1- 3 Long-term results of dilation were reviewed by Devereux and Burfield, who noted that routine dilation in younger patients placed them at an increased risk of having lower urinary tract complications. 24 Furthermore, Blandy and associates reported that among patients whom they considered to be doing well with routine dilation 43 per cent had some complication. 25 Optical internal urethrotomy for management of short urethral strictures is gaining popularity, and Glassberg and associates recommend its usage in pediatric patients. 10 However, Turner-Warwick cautions that failure with this technique results in local scarring and may make subsequent reconstruction more difficult. 28 Stricture excision and oblique reanastomosis 11 often are possible with short post-traumatic strictures. Those procedures were done in 7 of our 15 patients who had a 1-stage repair. All 7 repairs were successful. It is important to identify and reanastomose the normal urethra proximally and distally without tension. The extent of intrinsic urethral scarring must be verified at the time of operation, since radiologic estimation of this often is misleading. For strictures in the membranous urethra transpubic exposure often greatly facilitates the urethroplasty. We had 2 such cases in our series. In 1 case stricture recurred and at surgery the urethra was found to be encased in a fibro-

URETHRAL STRICTURE DISEASE IN CHILDREN

Fm. 5. A, retrogTade and imte:rograde urethrogram demonstrates preoperative membranous urethral stricture. B, result following transpubic repair. TABLE 4

Age

Case No.

2 3

at

Etiology

Location (No.)

9

Traumatic

Membranous

16 6

Traumatic TTaurn.atic

]i/lembranous

Traumatic

Membranous

Traumatic Traumatic

Membranous Membranous

Membranous

5 6

20

7

8

T:raumatic

Membranous

8

5 16

Traumatic

Bulbous (3) Bulbous (3) Bulbous

10

9

Traumatic Traumatic

Type of Repair Primary catheter Yealignment, * dilation,* suprapubic tube, skin inlay repair of stricture and reanastomosis transpubic repair, dilation, multistage skin Primary followed by stricture, t:ranspubic repair omentum Excision of stricture and. reanastomosis multistage skin inlay repair, Suprapubic reanastomosis with omentum Dilation patch graft Dilation, graft tube,* dilation, excision of stricture and

Length of Followup Voiding (mos.)

Complications

68

Good

None

14 41

Good Good

None (stress incontinence) Restricturn

9

Good

8

56

Good Good

(No erections, stress incontinence) None None (incontinence)

3

Good

None (stress incontinence)

5 6 9

Good Good Good

None None None

65

Good

None

56 26

Poor Good

None Restricture

96

Good Good

Restiricture

Good Good Good

None None Restricture

Good Good Good Good Good Good

None None None None None None

reanaston1.osis

11

13

Traumatic

Bulbous

Suprapubic tube,* excision of stricture and reanastomosis

12

15

Bulbous Membranous

8

T:raumatic

2

Iatrogenic

2

Iatrogenic Iatrogenic

Membranous Ante,~or, bulbous and

Iatrogenic Iatrogenic Iatrogenic

Buibous (2) Bulbous

J.l

Iatrogenic Iatrogenic Iatrogenic Iatrogenic Iatrogenic Inflammatory

Bulbous Bulbous Anterior Bulbous (2) Bulbous Bulbous

J.6

Inflammatoxy

Bulbous

9

membranous

16 5 18

2

19 20

10

21 22 23 24 25

7 16 16 12

Anterior

Dilation 3 Vesicostomy, multistage skin inlay repair, dilation 3, 2nd stage soft dilation 6 wks., dilation Multistage :repaiT, dilation 1 :revision, 2nd stage, dilation, :repaiT, transurethral resection of

8

None

stricture

Dilation, graft Dilation Dilation 5 1 '~ skin inlay repair, transurethral of stricture, dilation Dilation, graft Excision strictu.rre and reanastomosis Excision of stricture and reanastomosis Multistage skin inlay repair Dilation 3, patch graft Dilation 2, * fulguration urethral polyps 2, dilation, patch graft Suprapubic tube, dilation

5

42 36

18 26

22 9

20 28

Lost

~, Procedure done elsewhere.

osseous tunnel. An omental graft might have nn,,nmt.rc>rl Good results with cu,u;:, µui"'" urethro5 AddiWaterhouse in 6 of 7 .,,,,,,,..,,..t,,rl no ol1;ho,oectJ.c deformity in > 100 adult and µct,cn,1,u1> he has managed in this fashion,8 which has expenEm(:e in our smaller series as well. 29 1

A full thickness graft is an alternative 1-stage technique. All 6 of our patients whose bulbar strictures were treated by this means had a good result. Other series report good results in children using this technique. 3 ' 7 Multistage skin inlay procedures, using perinea! or scrotal skin, were done in 7 patients. There was l fistula and no

654

HARSHMAN AND ASSOCIATES

diverticula. Management of acute posterior urethral trauma remains controversial. Immediate repair versus initial suprapubic urinary diversion with delayed repair is the issue. Immediate intervention with realignment over a catheter is advocated by Myers and DeWeerd who reported good results in 20 of 22 patients. 30 Of their patients 7 were <12 years old. However, Waterhouse and Gross reported that all of their 9 patients, 2 of whom were children, treated by primary catheter realignment with traction subsequently had strictures. 31 Morehouse and MacKinnon have been the prime advocates of initial suprapubic cystostomy for significant urethral injury followed by delayed repair if a stricture develops. 32 Of 5 children treated by us in this fashion 4 had a good result and we recommend this approach. Care should be taken when passing a retrograde catheter, since injudicious passage may convert a partial tear into a complete tear. 10 In 2 to 4 weeks a voiding study should be obtained and, if normal, the suprapubic tube may be removed. If disruption exists repair should be performed after the tissue reaction has subsided. We would perform the repair earlier than the usual 6-month delay. 33 The rate of impotence after posterior urethral injuries ranges from Oto 78 per cent of patients. 32 ' 34- 36 Chambers and Balfour reported that impotence was temporary in 7 of 31 adult patients, lasting from 4 months to 4 years, with an average of 19 months. 35 Turner-Warwick warns that excessive mobilization of the prostate in cases of posterior urethral disruption increases the chance for impotence. 37 Reported experience in children in this regard is scarce but Malek4 and Blandy38 and their associates noted no impotence in 7 and 36 children, respectively, secondary to reconstructive surgery. Waterhouse and Gross reported no cases among 32 children. 31 Inability to achieve an erection has persisted for 18 months following an accident in a 9-year-old boy in our series. Urinary continence is dependent on an intact bladder neck sphincter or an intact distal urethral mechanism, consisting of intrinsic urethral resistance and an intact external sphincter. 37 Incontinence following severe injuries to the posterior urethra is reported to range from Oto 33 per cent. 4 • 5• 32 ' 38• 39 Four of our 7 patients who had severe pelvic fractures with posterior urethral disruption have some degree of incontinence. The 3 boys with minor incontinence may experience some improvement at puberty when prostatic growth will contribute to intrinsic urethral resistance. REFERENCES 1. Leadbetter, G. W., Jr. and Leadbetter, W. F.: Urethral strictures in male children. J. Urol., 87: 409, 1962. 2. Devereux, M. H. and Williams, D. I.: The treatment of urethral stricture in boys. J. Urol., 108: 489, 1972. 3. Gibbons, M. D., Koontz, W.W., Jr. and Smith, M. J. V.: Urethral strictures in boys. J. Urol., 121: 217, 1979. 4. Malek, R. S., O'Dea, M. J. and Kelalis, P. P.: Management of ruptured posterior urethra in childhood. J. Urol., 117: 105, 1977. 5. Waterhouse, K.: The surgical repair of membranous urethral strictures in children. J. Urol., 116: 363, 1976. 6. Waterhouse, K.: Injuries to the urinary tract in children. In: Reviews in Paediatric Urology. Edited by J. H. Johnston and W. E. Goodwin. Amsterdam: Excerpta Medica, chapt. 10, pp. 241-267, 1974. 7. Fuqua, F.: Utilization of full thickness skin grafts for the management of urethral strictures in the male child. In: Urinary System Malformations in Children. Edited by D. Bergsma and J. W. Duckett. New York: Alan R. Liss, Inc., sect. V, pp. 223-225, 1977. 8. Waterhouse, K.: The surgical repair of membranous urethral strictures in children. In: Urinary System Malformations in Children. Edited by D. Bergsma and J. W. Duckett. New York: Alan R. Liss, Inc., sect. V, pp. 227-234, 1977. 9. Glassberg, K. I., Kassner, E. G., Haller, J. 0. and Waterhouse, K.: The radiographic approach to injuries of the prostatomembranous urethra in children. J. Urol., 122: 678, 1979. 10. Glassberg, K. I., Tolete-Velcek, F., Ashley, R. and Waterhouse, K.: Partial tears of the prostatomembranous urethra in children.

Urology, 13: 500, 1979. 11. Turner-Warwick, R. T.: Three approaches to management of acute disruption of the membranous urethra. In: Current Controversies in Urologic Management. Edited by R. Scott, Jr. Philadelphia: W. B. Saunders Co., chapt. 8, essay 3, pp. 144-150, 1972. 12. Devine, P. C., Fallon, B. and Devine, C. J., Jr.: Free full thickness skin graft urethroplasty. J. Urol., 116: 444, 1976. 13. Wein, A. J., Leoni, J. V., Sansone, T. C., Mulholland, S. G. and Bogash, M.: Two-stage urethroplasty for urethral stricture disease. J. Urol., 118: 392, 1977. 14. Turner-Warwick, R. T.: The repair of urethral strictures in the region of the membranous urethra. J. Urol., 100: 303, 1968. 15. Johanson, B.: Reconstruction of the male urethra in stricture. Acta Chir. Scand., suppl., 176: 1, 1953. 16. Waterhouse, K., Abrahams, J., Gruber, H., Hackett, R. E., Patil, U. B. and Peng, B. K.: The transpubic approach to the lower urinary tract. J. Urol., 109: 486, 1973. 17. Singh, M. and Blandy, J.P.: The pathology of urethral stricture. J. Urol., 115: 673, 1976. 18. Devine, C. J., Jr.: Surgery of the penis and urethra. In: Campbell's Urology, 4th ed. Edited by J. H. Harrison, R. F. Gittes, A. D. Perlmutter, T. A. Stamey and P. C. Walsh. Philadelphia: W. B. Saunders Co., vol. 3, chapt. 81, pp. 2390-2437, 1979. 19. Walther, P. C. and Kaplan, G. W.: Cystoscopy in children: indications for its use in common urologic problems. J. Urol., 122: 717, 1979. 20. Stephens, F. D.: Urethral obstruction in childhood. In: Congenital Malformations of the Rectum, Anus, and Genitourinary Tracts. Edited by R. Webster. London: E. & S. Livingstone, Ltd., pp. 225-227, 1963. 21. Sacknoff, E. J. and Kerr, W. S., Jr.: Direct vision cold knife urethrotomy. J. Urol., 123: 492, 1980. 22. Devine, P. C., Wendelken, J. R. and Devine, C. J., Jr.: Free full thickness skin graft urethroplasty: current technique. J. Urol., 121: 282, 1979. 23. Orandi, A.: One-stage urethroplasty. Brit. J. Urol., 40: 717, 1968. 24. Devereux, M. H. and Burfield, G. D.: Prolonged follow-up of urethral strictures treated by intermittent dilation. Brit. J. Urol., 42: 321, 1970. 25. Blandy, J. P., Wadhwa, S., Singh, M. and Tresidder, G. C.: Urethroplasty in context. Brit. J. Urol., 48: 697, 1976. 26. Waterhouse, K.: Symposium-surgical management of urethral strictures. Contemp. Surg., 16: 52, 1980. 27. Wein, A. J., Malloy, T. R., Greenberg, S. H., Carpiniello, V. L. and Murphy, J. J.: Omental transposition as an aid in genitourinary reconstruction procedures. J. Trauma, 20: 473, 1980. 28. Turner-Warwick, R.: The use of the omental pedicle graft in urinary tract reconstruction. J. Urol., 116: 341, 1976. 29. Malloy, T. R., Wein, A. J. and Carpiniello, V. L.: Transpubic urethroplasty for prostatomembranous urethral disruption. J. Urol., 124: 359, 1980. 30. Myers, R. P. and DeWeerd, J. H.: Incidence of stricture following primary realignment of the disrupted proximal urethra. J. Urol., 107: 265, 1972. 31. Waterhouse, K. and Gross, M.: Trauma to the genitourinary tract: a 5-year experience with 251 cases. J. Urol., 101: 241, 1969. 32. Morehouse, D. D. and MacKinnon, K. J.: Management of prostatomembranous urethral disruption: 13-year experience. J. Urol., 123: 173, 1980. 33. Waterhouse, K., Laungani, G. and Patil, U.: The surgical repair of membranous urethral strictures: experience with 105 consecutive cases. J. Urol., 123: 500, 1980. 34. Gibson, G. R.: Urological management and complications of fractured pelvis and ruptured urethra. J. Urol., 111: 353, 1974. 35. Chambers, H. L. and Balfour, J.: The incidence of impotence following pelvic fracture with associated urinary tract injury. J. Urol., 89: 702, 1963. 36. Jackson, D. H. and Williams, J. L.: Urethral injury: a retrospective study. Brit. J. Urol., 46: 665, 1974. 37. Turner-Warwick, R.: Complications of urethral surgery in the male. In: Complications of Urologic Surgery. Prevention and Management. Edited by R. B. Smith and D. G. Skinner. Philadelphia: W. B. Saunders Co., chapt. 17, pp. 336-372, 1976. 38. Blandy, J. P., Singh, M., Notley, R. G. and Tresidder, G. C.: The results and complications of scrotal-flap urethroplasty for stricture. Brit. J. Urol., 43: 52, 1971. 39. Allen, T. D.: The transpubic approach for stricture of the membranous urethra. J. Urol., 114: 63, 1975.