Anomalies of the external urethral meatus in girls with nonneurogenic bladder sphincter dysfunction

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1 BJU International (1999), 83, Anomalies of the external urethral meatus in girls with nonneurogenic bladder sphincter dysfunction P. HOEBEKE, E. VAN LAECKE, A. RAES*, J.D. VAN GOOL and J. VANDE WALLE* Departments of Urology and *Paediatric Nephrology, Paediatric Uro-Nephrologic Centre, University Hospital Gent, Belgium and Paediatric Renal Centre, Department of Pediatric Nephrology, University Childrens Hospital, Utrecht, The Netherlands Objective To investigate in a prospective study the clini- in the control group (34%) (P=0.003). Nocturnal cal and urodynamic characteristics associated with enuresis was more frequent in the control group (87%) the correlation previously reported between non- than in the study group (68%, P=0.006). Urge was neuropathic bladder sphincter dysfunction and minimal more frequent in the study group (80%) than in the meatal deformities in girls. control group (65%, P=0.02), as was anterior deflec- Patients and methods From January 1995 to June 1996 tion of the urinary stream (voiding over the toilet all girls referred for urodynamics to investigate non- rim), in 48% and 4.5%, respectively (P<0.001). On neuropathic bladder sphincter dysfunction were examined video-urodynamics, dysfunctional voiding (detrusor for meatal anomalies. Two types of meatal anom- sphincter dyscoordination during voiding) was more alies were recognized, i.e. hypospadias, in which the frequent in the study group (59%) than in the control urethral meatus is displaced dorsally and meatal web group (38%, P=0.009), and vaginal voiding was or covered hypospadias, in which there is a mucosal more frequent in the study group (40%) than in the web on the dorsal side of the urethral meatus that control group (11.5%, P<0.001). deflects the urinary stream anteriorly. The girls presenting Conclusion Girls presenting with meatal anomalies have with minimal meatal deformities were com- more severe dysfunction, as estimated by urodynam- pared with those presenting a normal meatal ics. Although the natural history of these anomalies configuration for their history and video-urodynamic and the incidence in the normal population is not features. known, it is thought that there is an aetiological Results During the study, 288 girls were referred for correlation between the functional voiding disorders video-urodynamics; 88 presented with meatal anomalies and the meatal anomaly. Surgical treatment to correct (24 hypospadias and 64 covered hypospadias) and the anomalies is proposed. comprised the study group; all other girls were considered Keywords Dysfunctional voiding, female urethral as the control group. Incontinence during the meatus, anomalies day was more frequent in the study group (56%) than Introduction From January 1995 to June 1996 all patients primarily consulting for enuresis/incontinence problems under- the urethral meatus is displaced dorsally so that it lies went a standardized noninvasive screening protocol on the anterior vaginal wall (Fig. 1a), and meatal web consisting of a history, clinical examination, ultra- or covered hypospadias (Fig. 1b), in which a mucosal sonography of the urinary tract, uroflowmetry and completion web covers the urethral meatus and deflects the urinary of a voiding diary. Those patients with evidence stream anteriorly (Fig. 1c,d) [1]. After this report, a of an underlying bladder dysfunction, defined as those prospective study was designed to investigate the clinical presenting with diurnal incontinence, abnormal uroflow patterns, residual urine after voiding and small bladder Accepted for publication 29 September 1998 capacity for age as estimated from the diary, underwent Although the female urethral meatus is not extensively described in most anatomical and urological books, it has often been implicated in urological problems in girls. We previously described a group of 10 girls with meatal deformities and dysfunctional voiding, and recognized two types of meatal anomalies, i.e. hypospadias, in which and urodynamic characteristics associated with these anomalies. Therapeutic options and results are presented for part of the study group. Patients and methods BJU International

ANOMALIES OF THE EXTERNAL URETHRAL MEATUS 295 a b c d Fig. 1.

b, Meatal web or covered hypospadias, in which there is a mucosal web on the dorsal side of the urethral

d, A closer view of the anterior deflection of the urinary stream in a patients with a meatal web.

Of this group of patients, all girls mal meatal deformities and those presenting a normal referred during

2 ANOMALIES OF THE EXTERNAL URETHRAL MEATUS 295 a b c d Fig. 1. a, Hypospadias, in which the urethral meatus is displaced dorsally. b, Meatal web or covered hypospadias, in which there is a mucosal web on the dorsal side of the urethral meatus that deflects the urinary stream anteriorly. c, Web lifted by the tip of a urethral catheter. d, A closer view of the anterior deflection of the urinary stream in a patients with a meatal web. further urodynamics. Of this group of patients, all girls mal meatal deformities and those presenting a normal referred during the study period were examined for meatal configuration. Methods, definitions and units meatal anomalies. The history and video-urodynamic conform to the standards recommended by the ICS [2]. data were compared between girls presenting with mini- To treat the condition, the anomaly was corrected

3 296 P. HOEBEKE et al. surgically before bladder training started. For hypospadwith Table 1 Symptoms, history and video-urodynamics features of girls ias, a partial hymenectomy, in which the anterior part and without meatal anomalies of the hymen is resected, was performed, after which the Meatal Normal vaginal voiding disappeared. In those with a meatal web, anomalies meatus P the mucosal web was incised longitudinally and closed transversely with three interrupted polyglactin 6/0 Total no sutures, which corrected the direction of the urinary Night-time 39 (44) 132 (66) stream. Daytime incontinence 28 (32) 27 (13.5) After surgery all patients were included in a bladderincontinence Night-time+daytime 21 (24) 41 (21) NS training programme consisting of an individually Urge 70 (80) 130 (65) 0.02 adapted voiding and drinking schedule, instructions on Anterior deflection 42 (48) 9 (4.5) <0.001 toilet behaviour, biofeedback uroflowmetry, ultrasonogof urinary stream raphy for residual urine, and for those presenting with Urinary tract infection 31 (35) 62 (31) NS dysfunctional voiding, additional pelvic-floor relaxation Constipation 44 (50) 60 (30) <0.001 biofeedback. Girls with urodynamically confirmed Video-urodynamics detrusor instability received anticholinergics (oxybutynin Normal filling 7 (8) 14 (8) NS 0.3 mg/kg) and those with hypersensitivity received fla- Detrusor instability 59 (67) 119 (59.5) NS Hypersensitive 13 (15) 45 (21.5) NS voxate. The girls in the control group underwent the Lazy bladder 9 (10) 22 (11) NS same training programme. The ecects of therapy in both Dysfunctional voiding 52 (59) 76 (38) <0.001 groups were evaluated and compared. Vaginal voiding 35 (40) 23 (12) <0.001 Reflux 16 (18) 41 (21) NS Results During the study period, 288 girls with suspected nonneurogenic bladder sphincter dysfunction disorders underwent video-urodynamics; 88 girls presented with Discussion meatal anomalies (24 hypospadias and 64 covered hypo- Lyon and Tanagho [3] were the first to draw attention spadias) and comprised the study group, all other girls to the concept of distal urethral stenosis in girls pre- being included in the control group. The dicerences in senting with recurrent UTIs. Although they both later symptoms and the results of video-urodynamics are doubted the obstructive nature of the distal urethral shown in Table 1. Incontinence during the day, urge stenosis and attributed the problems to sphincter spasm and anterior deflection of the urinary stream (voiding [4,5], they initiated urethral dilatation and internal over the toilet rim) were significantly more frequent in urethrotomy, which remained in use for a long time. the study group than in the control group. Isolated Although several authors described the urethral calibre night-time wetting was significantly more frequent in in normal controls as being similar to that in the the control group than in the study group; constipation pathological conditions described [6,7] urethral dila- was significantly more frequent in the study group. On tation continued to be the commonest treatment. video-urodynamics (Table 1), filling-phase dysfunctions In 1970, Harvard criticized the concept of distal were the same in both groups, but dysfunctional voiding urethral stenosis and described an anomaly of the external (detrusor sphincter dyscoordination during voiding), urethral meatus in girls with recurrent UTIs, i.e the vaginal voiding and ballooning of the urethra were inferior aspect of the infant meatus is often thick and significantly more common in the study group than in tough and appears to be a part of the hymenal ring ; the control group; VUR was equally common in both this is similar to the meatal web. The anomaly was groups. even treated in the same way, except that instead of The outcome of treatment was evaluated for both incision, Harvard performed an excision of the meatal groups after 6 months of follow-up. The therapy was web [8]. Most of the patients treated by Harvard had considered successful when the incontinence problems recurrent UTI, but also typical symptoms of dysfunctional ceased. The accompanying drug therapy was administered voiding, e.g. straining, urge incontinence, daytime wet- for the entire course of training and afterwards ting and bed-wetting. There were even eight patients only gradually tapered, commencing 3 months after with no infection and with incontinence problems alone. continence had been achieved. After 6 months, the Ten children of the group of 76 girls showed reflux treatment was successful in 73 girls (83%) of the study (seven bilateral and four unilateral), which could reflect group and in 169 (85%) of the control group (not that this was indeed a group of patients similar to those significantly dicerent). described in the present study. Kaplan et al. [9] were the

4 ANOMALIES OF THE EXTERNAL URETHRAL MEATUS 297 first to suggest that urethral dilatation and urethrotomy [18]. (iii) In girls with hypospadias, extreme vaginal had no better outcome than medication alone. At the voiding can cause further urine loss after voiding. To same time, Hendry et al. [10] introduced the concept of prevent this, the girls may contract their sphincter dysfunctional bladder syndrome, which replaced the intensely several times a day, which might be responsible concept of distal meatal stenosis, treated by urethral for sphincter hypertrophy, finally resulting in dysfunctional dilatation and urethrotomy. voiding. Vaginal filling during voiding can even Much has been written about the functional origin of stimulate the bulbocavernosus reflex. detrusor sphincter discoordination; it is supposed to be Indirect confirmation of the association between mini- learned behaviour that can be self-sustaining [11 13], mal meatal deformities and dysfunctional voiding lies in and the concept of learned behaviour has never been the ease with which the voiding dysfunction can be challenged. However, dysfunctional voiding is thought treated after the deformity is corrected. Although girls to be multifactorial in origin and there may be dicerent with meatal anomalies show worse dysfunction, as illus- types. In the present study, we propose a new concept trated by symptoms and urodynamic findings, the outcome of minimal anatomical deformity. The theoretical background of treatment after surgical correction is the same for the importance of minimal anatomical as in those girls with no meatal anomalies. A prospective deformities in children with functional voiding disorders study is currently underway to evaluate the ecect of arises from the work of Yeung et al. [14], who reported surgical treatment on outcome. interesting new insights into bladder function in infancy. In conclusion, we propose a new concept for the role They reported that voiding with incomplete coordination of meatal anomalies in the pathogenesis of nonneurogenic between detrusor contraction and urinary sphincter bladder sphincter dysfunction in girls. Such relaxation may be normal, and thus that dysfunctional meatal anomalies occur often in girls with nonneurogenic voiding could be a physiological transition phase in bladder sphincter dysfunction and cause normal bladder maturation. Theoretically, in girls with greater dysfunction, with a higher incidence of daytime anomalies of the urethral meatus the infant dysfunctional incontinence and urge. Dysfunctional voiding is more voiding pattern could persist, because they are common and might be explained by deflection of the unable to control this dysfunction, which is sustained urinary stream or vaginal voiding caused by the anatom- by the anatomical anomaly. ical anomaly. Surgical correction of these anomalies can Female hypospadias is briefly mentioned in one textbook form part of the treatment of these girls. [15] and there is one report by van Bogaert [16] in which it is described in females with urethral syndrome. This may provide a link with the present finding References of hypospadias and dysfunctional voiding; many women 1 Hoebeke P, Van Laecke E, Raes A, Vande Walle J. Hundred with the urethral syndrome have had dysfunctional consecutive cystoscopic examinations in children. voiding in childhood. Thus, there may be a causal Indications and results. Eur Urol 1996; 30: association between dysfunctional voiding and minimal 2 Abrams P, Blaivas JG, Stanton SL, Andersen JT. The meatal deformities. Several hypotheses may explain this standardization of terminology of lower urinary tract function. Scand J Urol Nephrol Suppl 1988; 114: 5 19 association: (i) The bulbocavernosus reflex that is nor- 3 Lyon RP, Smith DR. Distal urethral stenosis. J Urol 1963; mally absent during voiding can be elicited by genital 89: stimulation. In the case of the anterior deflection of the 4 Lyon RP, Tanagho EA. Distal urethral stenosis in little urinary stream, the stream passes the clitoris and can girls. J Urol 1965; 93: stimulate the bulbocavernosus reflex, causing sphincter 5 Tanagho EA, Miller ER, Lyon RP, Fisher R. Spastic striated activity during voiding. In hypospadias, there is strong external sphincter and urinary tract infection in girls. Br vaginal voiding that could stimulate the same reflex. J Urol 1971; 43: The same is described for the neurogenic bladder with 6 Graham JB, King LR, Kropp KA, Uehling DT. The detrusor sphincter dyssynergia, where normal inhibition significance of distal urethral narrowing in young girls. of the bulbocavernosus reflex during voiding is deleted J Urol 1967; 97: by the neurological lesion [17]. In girls with dysfuncnormal female children. J Urol 1967; 97: Immergut M, Culp D, Flocks RH. The urethral caliber in tional voiding this inhibition might not be obtained 8 Harvard BM. Revision of the external urinary meatus in because of the minimal anatomic deformity. (ii) In girls girls: a clinical appraisal. J Urol 1970; 103: with anterior deflection of the urinary stream, it is 9 Kaplan GW, Sammons TA, King LR. A blind comparison possible that the only defence against voiding over the of dilatation, urethrotomy and medication alone in the rim of the toilet is a bent posture that precludes good treatment of urinary tract infection in girls. J Urol 1973; relaxation of the pelvic floor muscles during voiding, 109: thus creating a functional obstruction during voiding 10 Hendry WF, Stanton SL, Williams DI. Recurrent urinary

5 298 P. HOEBEKE et al. tract infections in girls: ecects of urethral dilatation. Br 17 Sethi RK, Bauer SB, Dyro FM, Krarup C. Modulation of the J Urol 1973; 45: bulbocavernosus reflex during voiding: loss of inhibition in 11 KoC SA. Bladder-sphincter dysfunction in childhood. upper motor neuron lesions. Muscle Nerve 1989; 12: Urology 1982; 19: Wennergren HM, Öberg BE, Sandstedt P. The importance 12 Van Gool JD, De Jonge GA. Urge syndrome an d urge of leg support for relaxation of the pelvic floor muscles. incontinence. Arch Dis Child 1989; 64: Scand J Urol Nephrol 1991; 25: Vande Walle J, Theunis M, Renson C, Raes A, Hoebeke P. Commercial television bladder dysfunction. Acta Urologica Belgica 1995; 63: Yeung CK, Godley ML, Ho CKW et al. Some new insights Authors into bladder function in infancy. Br J Urol 1995; 76: P. Hoebeke, MD, Paediatric Urologist E. Van Laecke, MD, Paediatric Urologist. 15 Elder JS. Congenital anomalies of the genitalia. In Campbell s A. Raes, MD, Paediatric Nephrologist. Urology. 6th ed. Philadelphia: WB Saunders Co., 1993: J.D. Van Gool, MD, Paediatric Nephrologist 1933 J. Vande Walle, PhD, Paediatric Nephrologist 16 Van Bogaert L. Surgical repair of hypospadias in women Correspondence: Dr P. Hoebeke, Department of Paediatric with symptoms of urethral syndrome. J Urol 1992; Urology, University Hospital, De Pintelaan 185, B : Gent, Belgium.

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