Surgical Reconstruction of Cloacal Malformation Can Alter Bladder Function: A Comparative Study With Anorectal Anomalies

Warne, S.A.; Godley, Margaret L.; Wilcox, Duncan T.

doi:10.1097/01.ju.0000145201.94571.67

Cited by 54 publications

(46 citation statements)

References 14 publications

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“…The neuroanatomy of males with rectourethral fistula due to ARM has demonstrated that both pelvic splanchnic nerves come into close proximity at the level of the distal rectum and are only separated by the thin fistula itself (17). Other authors have also observed a change in urodynamics after extensive dissection needed for correction of ARM (18,19). Warne et al demonstrated 60% incidence of bladder dysfunction at presentation in 20 patients with ARM, however a change in bladder function following reconstructive surgery was noted in only one of the patients who had a recto-bladder neck fistula repaired with a combined posterior sagittal and abdominal approach (18).…”

Section: Discussionmentioning

confidence: 95%

Epididymitis in Patients with Anorectal Malformations: A Cause for Urologic Concern

et al. 2014

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ARTICLE INfO ______________________________________________________________ ______________________Introduction: Epididymitis in patients with anorectal malformation (ARM) represents a unique problem because unlike the general population, an underlying urinary tract problem is frequently identified. We review our experience with epididymitis in ARM population with an emphasis on examining urologic outcomes. Materials and Methods:We performed a retrospective review of male patients with ARM cared for from 1980 to 2010. Clinical and pathologic variables recorded included age at presentation, recurrence, associated urologic anomalies, incidence of ureteral fusion with mesonephric ductal structures, glomerular filtration rate and urodynamic parameters.Results: Twenty-six patients were identified with documented episodes of epididymitis.Renal injury was noted in five patients (19%), all of whom were diagnosed with neurogenic bladder (NGB) several years after anorectoplasty. NGB was found in ten patients (38%) in our series. Ectopic insertion of ureter into a mesonephric ductal structure was discovered in five patients (19%). Twelve patients (46%) had recurrent episodes of epididymitis, with seven of these patients (58%) being diagnosed with NGB. Two patients in the pubertal group presented with a history of epididymitis and complained of ejaculatory pain. Conclusion: Epididymitis in a patient with ARM warrants a comprehensive urologic investigation, particularly in recurrent episodes. Attempts at surgical intervention (e.g. vasectomy) should be avoided until functional assessment of the urinary tract has occurred. Failure to recognize this association may lead to potentially avoidable complications and morbidity. Long term urological follow up of these patients is warranted to identify at risk patients and minimize renal deterioration

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Section: Discussionmentioning

confidence: 95%

Epididymitis in Patients with Anorectal Malformations: A Cause for Urologic Concern

et al. 2014

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“…The indicators of neuropathy in the context of ARA are unclear, but we consider that neuropathy affecting the bladder is one of the best indicators because of similar embryological origin of hindgut and bladder [20,21]. Morbidity related to neuropathy may be associated with the original ARA or acquired during reconstruction of anorectal and cloacal malformations [18,19,21]. Thus, urodynamic assessment may be necessary to detect bladder dysfunction in these patients.…”

Section: Introductionmentioning

confidence: 95%

“…Neuropathy and megarectum are likely to impact adversely on the functional outcome even in the presence of a good sphincter [2,[17][18][19][20]. The indicators of neuropathy in the context of ARA are unclear, but we consider that neuropathy affecting the bladder is one of the best indicators because of similar embryological origin of hindgut and bladder [20,21].…”

Section: Introductionmentioning

confidence: 97%

Evaluation of outcome of anorectal anomaly in childhood: the role of anorectal manometry and endosonography

et al. 2008

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The aim of this study was to evaluate role of anorectal manometry (ARM) and anal endosonography (ES) in assessment of the internal anal sphincter (IAS) quality on continence outcome following repair of anorectal anomalies (ARA). We devised a scoring system to evaluate the quality of the IAS based on ARM and ES and correlated the scores with clinical outcome, using a modified Wingfield score (MWS) for faecal continence. We also assessed the implication of megarectum and neuropathy on faecal continence. Of 54 children studied, 34 had high ARA and 20 had low ARA. Children with high ARA had poor sphincters on ES and ARM, and also poor faecal continence compared to those with low ARA. The presence of megarectum and neuropathy was associated with uniformly poor outcome irrespective of the IAS quality. The correlations between MWS on one hand, and ES and ARM scores for IAS on the other hand were weak in the whole study group, ES r = 0.27, P < 0.04, and ARM r = 0.39, P < 0.004. However, the correlations were strong in those who had isolated ARA without megarectum or neuropathy, ES r = 0.51, P < 0.02 and ARM r = 0.55, P < 0.01, respectively. In conclusion, the ARM and ES are valuable in evaluation of continence outcome in children after surgery for ARA and those with good quality IAS had better faecal continence. The IAS is a vital component in functional outcome in absence of neuropathy and megarectum.

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“…About 1:50 000 human births have a 'persistent cloaca' (2). Persistent cloaca is a potentially devastating disease, generally requiring multiple rounds of corrective surgery and, even with the best current treatments, there can be significant urological and gynaecological sequelae, including incontinence and infertility (3)(4)(5). Upper renal tract malformations are common associations.…”

Section: Introductionmentioning

confidence: 99%

Mutational analyses of UPIIIA, SHH, EFNB2, and HNF1β in persistent cloaca and associated kidney malformations

Jenkins

Bitner‐Glindzicz

Thomasson

et al. 2007

Journal of Pediatric Urology

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Objectives-'Persistent cloaca' is a severe malformation affecting females in which the urinary, genital and alimentary tracts share a single conduit. Previously, a Uroplakin IIIA (UPIIIA) mutation was reported in one individual with persistent cloaca, and UPIIIA, Sonic Hedgehog (SHH), Ephrin B2 (EFNB2) and Hepatocyte Nuclear Factor 1β (HNF1β) are expressed during the normal development of organs that are affected in this condition. HNF1β mutations have been associated with uterine malformations in humans, and mutations of genes homologous to human SHH or EFNB2 cause persistent cloaca in mice.Patients and Methods-We sought mutations of coding regions of UPIIIA, SHH, EFNB2 and HNF1β genes by direct sequencing in a group of 20 patients with persistent cloaca. Most had associated malformations of the upper renal tract and over half had impaired renal excretory function. The majority of patients had congenital anomalies outside the renal/genital tracts and two had the VACTERL association.Results-Apart from a previously described index case, we failed to find UPIIIA mutations, and no patient had a SHH, EFNB2 or HNF1β mutation.Conclusion-Persistent cloaca is only rarely associated with UPIIIA mutation. Despite the fact that SHH and EFNB2 are appealing candidate genes, based on their expression patterns and mutant mice phenotypes, they were not mutated in these humans with persistent cloaca. Although HNF1β mutations can perturb paramesonephric duct fusion in humans, HNF1β was not mutated in persistent cloaca.

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Surgical Reconstruction of Cloacal Malformation Can Alter Bladder Function: A Comparative Study With Anorectal Anomalies

Cited by 54 publications

References 14 publications

Epididymitis in Patients with Anorectal Malformations: A Cause for Urologic Concern

Epididymitis in Patients with Anorectal Malformations: A Cause for Urologic Concern

Evaluation of outcome of anorectal anomaly in childhood: the role of anorectal manometry and endosonography

Mutational analyses of UPIIIA, SHH, EFNB2, and HNF1β in persistent cloaca and associated kidney malformations

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