0022-5347 /81/1254-0534$02.00/0 Vol. 125, April
THE JOURNAL OF UROLOGY
Printed in U.S.A.
Copyright© 1981 by The Williams & Wilkins Co.
URODYNAMICS IN CHILDREN. PART IL THE PSEUDONEUROGENIC BLADDER MONEER K. HANNA,* WILLIAM D1 SCIPIO, KYOUNG K. SUH, STANLEY J. KOGAN, SELWYN B. LEVITT AND KENNETH DONNER From the Division of Pediatric Urology, Children's Hospital of New Jersey, Newark, New Jersey, and Division of Pediatric Urology, Departments of Anesthesia and Psychiatry, Hospital of the Albert Einstein College of Medicine and Montefiore Hospital and Medical Center, Bronx, New York
ABSTRACT
We evaluated 83 children for recurrent urinary infections, day and night urinary incontinence or both for more than 1 year in the urodynamic laboratory. Bladder instability and/ or bladder sphincter dyssynergia was encountered in 74 children. Pharmacologic manipulation of detrusor and sphincter function and biofeedback therapy were highly successful. Routine and random urethral dilation, urethrotomy and fulguration, and/ or resection of ill-defined urethral obstructions is unwarranted. Rational treatment requires individualization based upon an objective evaluation by thorough and careful urodynamic testing. In 1973 Hinman and Baumann reported on 14 boys with incoordination of micturition, enuresis and recurrent urinary tract infections in whom no neurologic or obstructive disease could be detected. 1 Allen supported their thesis by reporting on 21 children with "non-neurogenic neurogenic" bladders. 2 Recently, this syndrome has received greater recognition and a number of surgical failures of urinary tract reconstruction procedures proved to be owing to voiding dysfunction. Various names have been given and various treatments applied. The group of 14 boys described by Hinman and Baumann provide a sound basis for understanding this disorder. However, a much larger series of cases will present additional clinical features and, indeed, additional difficulties in management. Herein we report on 83 children with voiding dysfunction and pseudoneurogenic bladders. We wish to make a distinction between "occult" and "pseudo" neurogenic bladder. The first implies the presence of a subtle spinal canal disease, resulting in bladder neuropathy associated with minor or no skeletal abnormalities. We have encountered such a condition on 4 occasions and this group will be reported on elsewhere. Myelographic study and neurosurgical procedures are indicated for occult neuropathy. The pseudoneurogenic bladder probably represents a self-sustained maladaptive voiding habit resulting in bladder changes, namely trabeculation and sacculation, that mimic obstructed and neuropathic disorders. CLINICAL MATERIAL AND METHODS
recordings of the subtracted bladder pressure, urine flow rate, urethral sphincter electromyography and fluoroscopic cystourethrography were obtained. RESULTS
Clinical and urodynamic features. The clinical presentation is outlined in table 1. Most of the children had a prolonged history of urinary tract infections and incontinence. All patients were "hard core" cases, inasmuch as they had symptoms and infections while taking continuous antibacterial medications. Review of the patient's clinical history revealed no common denominator. Constipation and fecal soiling were common. Two children had renal impairment at presentation. Neurological examination was unremarkable in all. Lumbosacral spina bifida occulta was observed on the IVP in 8.4 per cent. Ureterai fullness and pyelonephritic scarring were present in 12 per cent (table 2). Voiding cystourethrography revealed vesicoureteral reflux in 17 children, significant trabeculation in 32 and a large capacity bladder in 12 (table 3). Urodynamic testing indicated that detrusor-sphincter incoordination is the most common underlying abnormality (table 4). Nine children proved to be normal urodynamically. At cystourethroscopy prominent mucosa! folds or "mini valves" were noted in 12 boys. All proved to have detrusor-sphincter incoordination and 3 also had mild bladder instability. Although 2 boys underwent transurethral fulguration of the valve leaflets the symptoms persisted. Subsequently, both patients responded to biofeedback therapy (fig. 1).
The study consisted of 37 boys and 46 girls, between 2½ and 16 years old, who were referred for urodynamic evaluation because of persistent bladder symptoms despite long-term antibacterial therapy and/ or various urethral manipulations. Many had been taking continuous antibacterial medication for years and the majority of girls had been subjected to urethral dilations at some stage. Operations were done in 3 children: 2 boys underwent resection of valves and 1 girl underwent internal urethrotomy and, subsequently, Y-V plasty of the bladder neck. Excretory urograms (IVPs) and voiding cystourethrograms were performed in all children. Urodynamic testing was done by the method reported previously. 3 Urine flow rate, urethral pressure profilometry and provocative cystometry were performed before the voiding study when simultaneous
Treatment. There were 60 children who had completed the treatment and were available for evaluation. The treatment is TABLE
1. Pseudoneurogenic bladder: clinical presentation No. Pts.
Recurrent urinary tract infection Day and night wetting Both Total
TABLE
2. !VP in pseudoneurogenic bladder No. Pts. (%)
Spina bifida occulta Pyelonephritic scarring and/ or ureteral fullness Normal Total
Accepted for publication July 14, 1980. * Requests for reprints: Children's Hospital of New Jersey, 15 South Ninth St., Newark, New Jersey 07107. 534
28 16 39 83
7
(8.4)
10 (12.1) 66 (79.5) 83 (100)
535
l7RODYNA11llCS LN C~HILDREN
divided into 3 (table 5). Large doses of anticholinergic medications, either alone or in various combinations, were prescribed for bladder instability (1 to 2 mg./kg. propantheline bromide and imiprarnine chloride in 3 divided doses daily, respectively). Sphincter dyssynergia was treated with 0.3 mg./ kg. phenoxybenzamine daily or l to 3 mg. per day diazepam, either alone or in combination, for 3 to 6 months. Transurethral fulguration or resection of the valves and Y-V plasty of the bladder neck were performed in 3 children. More recently, we have added contingency and biofeedback therapy to pharmacologic manipulation (figs. 2 to 4). The results of the various treatments are shown in table 5. Children who failed to respond to pharmacologic manipulation and surgery were treated by behavioral modification. This modality, used in conjunction with pharmacologic manipulation, yielded successful results in some children when the other modalities, either alone or in combination, had failed. Of 17 children 9 ceased to have reflux following conservative treatment within a 6-month period. DISCUSSION
Functional bladder outflow obstruction is characterized urodynamically by a high voiding pressure and low flow rate, often with intermittency. Finkbeiner and Lapides have demonstrated TABLE 3.
0
Voiding cystourethrography and endoscopy in pseudoneurogenic bladder No. Pts.
Bladder trabeculation (reflux in 13) Large capacity bladder (reflux in 4) Prominent posterior urethral folds ("mini-valves") Normal Total
TABLE 4.
experimentally that overdistension of the dog bladder results in reduced blood flow, th, YPhv interfering with local resistance to infection. 4 Bladder ischemia during involuntary detrusor contractions and/or the high voiding pressure might conceivably result in a decreased blood flow to the bladder and, thereby, increase its susceptibility to bacterial invasion. Alternatively, turbulent urine flow secondary to low flow rates and intermittency might be incriminated. Significant residual urine would be a further factor that w-ould be incriminated. It is not clear whether the detrusor instability precedes or follows sphincter spasticity. One may postulate that some children voluntarily contract the striated muscle of the external sphincter in response to involuntary bladder contraction to prevent urinary incontinence. The child learns to contract the external sphincter at a time of increased bladder pressure and this maladaptive habit may become a self-sustained behavioral pattern. It also is possible that the sphincter spasm may result in detrusor hypertrophy and instability, as well as altered neuromuscular transmission. Biofeedback has emerged as a new and exciting modality. It is based upon established principles of learning and motivation. It is a method aimed at the acquisition and maintenance of new learning. Instrumentation is applied to detect, code and amplify signals so that continuous and immediate feedback is provided. Externalization of bladder and urethral function can be accomplished readily by application of surface electrodes to the per-
5. Methods and results of treatment in 60 children with bladder instability and detrusor-sphincter incoordination
TABLE
32 12 12 27
Result Method
83
Improvedt
Failed:j:
18
6
24
7
10§ 2§ 4
Pharmacologic (34 pts.) Surgical (3 pts.) Pharmacologic + psychologic (35 pts.)
Pseudoneurogenic bladder: urodynamic abnormality
Bladder instability Sphincter dyssynergia Both Normal Total
Satisfactory*
Free of symptoms for >3 months and improved radiologically or significant urodynamic improvement (flow rate and electromyography). t Occasionally symptomatic and stable radiologically or moderate urodynamic improvement. :j: Unchanged symptoms and absent objective improvement. § Failures were treated by pharmacologic plus psychologic methods.
18 23 33 9 83
B
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FLOW RATE
25[ ml/sec VOIDED 250
FLOW RATE
MAX. FLOW AATE 14cc/aoc. MEAN FLOW RATE 14cc/HC.
0
--
ml/aiec
VOIDED 300
MAX. FLOW RATE 21cc/10c
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MEAN FLOW RATE 2:c/,ac /
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Frc. L J. S., 14-year-old boy, presented with diurnal frequency and nocturnal enuresis. Clinical examination was unremarkable. A, urodynamic evaluation reveals bladder sphincter incoordination. B, voiding study after biofeedback therapy shows sphincter relaxation and improved urine flow. Child remains asymptomatic.
536
HANNA AND ASSOCIATES
FIG. 2. Five-year-old boy presented with day and night wetting. Clinical examination was unremarkable. A, voiding cystourethrogram shows fullness of posterior urethra and reflux into upper moiety of left duplex kidney. B, IVP reveals bilateral hydronephrosis and complete duplication. Diagnosis was grade I posterior urethral valves and transurethral resection was performed elsewhere. Symptoms persisted. C, followup voiding cystourethrogram 6 months later shows increased bladder trabeculation and persistent left reflux.
ELECTROMYOGRAPHY
RECTAL PRESSURE
---~-"-'--~----
80[ 0
SUBTRACTED DETRUSOR PRESSURE (TOTAL-RECTAL)
__ _.r.N,1\;"-----.------~
I '"'"mr_TE____
.\.c.,, ______ -- ____ ,~.-._,~
A
---~·1:
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FIG. 3. Urodynamic evaluation of patient described in figure 2 reveals consistent bladder sphincter incoordination and intermittency of urine flow.
ianal sphincter or by observing the flow rate chart. Contingency management can be applied by means of token reinforcements for correct responses, for example aimed at improving flow rate as the child monitors his own urine flow rate during voiding. Complete electrical silence of the pelvic floor musculature as indicated on an oscilloscope or electromyograph tracing can be achieved by teaching the child to relax this muscle with biofeedback from a surface electromyographic electrode. The application of psychotherapy to children with "pseudoneurogenic" voiding dysfunctions has added a new dimension to our treatment armamentarium and has salvaged about 60 per cent of our treatment failures from pharmacologic manipulation and/or surgical treatment, either alone or in combination. These data lend support to the concept of maladaptive voiding habits as being at least 1 part of voiding dysfunction in children. Endoscopic observation of the urethra defines static anatomy and is no substitute for a dynamic evaluation. The mere presence of prominent urethral folds does not imply functional significance. The 12 children who were thought to have prom-
FIG. 4. IVP in patient described in figure 2 after bladder training (biofeedback therapy) shows significant improvement of upper urinary tract. Wetting has improved.
UR.0DYNA1VIICS
inent urethral mucosal folds radiologically and endoscopically to have sphincter dyssynergia. Because of the 2 children who were treated initially by transurethral fulguration of the folds without alleviation of the symptoms and who, subsequently, improved dramatically on pharmacotherapy and/ or psychotherapy, one seriously questions the functional significance of the frequently observed prominent mucosal folds that have been regarded by some recently as obstructive type I posterior urethral folds. 5 Persistent abnormal voiding symptoms in children, as well as frequently recurring urinary tract infections despite adequate antibiotic therapy, require thorough and careful urologic evaluation. Appropriate, properly performed urodynamic tests help to pinpoint the major contributing factors and allow more rational and objective therapy to be prescribed. This should include pharmacologic manipulation with anticholinergics, aadrenergic stimulators and inhibitors, biofeedback psychother-
11
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537
apy or surgery, either alone or in combination. Random routine empiric fulguration or urethral dilation and/or urethrotomy in girls is no longer justified. REFERENCES 1. Hinman, F. and Baumann, F. W.: Vesical and ureteral damage from
2. 3.
4. 5.
voiding dysfunction in boys without neurologic or obstructive disease. J. Urol., 109: 727, 1973. Allen, T. D.: The non-neurogenic neurogenic bladder. J. Urol., 117: 232, 1977. Hanna, M. K., Di Scipio, W., Suh, K. K., Kogan, S. J., Levitt, S. B. and Donner, K.: Urodynamics in children. Part I. Methodology. J. U:rol., 125: 530, 1981. Finkbeiner, A. and Lapides, J.: Effect of distension on blood flow in dog's urinary bladder. Invest. Urol., 12: 210, 1974. Hendren, W. H.: Evaluation of the child who wets. Paediatrician, 3: 251, 1974.