<i>In utero</i> myelomeningocoele repair and urological outcomes: the first 100 cases of a prospective analysis. Is there an improvement in bladder function?

Macedo, António; Ottoni, Sérgio Leite; Garrone, Gilmar; Liguori, Riberto; Cavalheiro, Sérgio; Moron, Antônio Fernandes; Cruz, Marcela Leal da

doi:10.1111/bju.14639

Cited by 22 publications

(17 citation statements)

References 13 publications

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“…While the MOMS trial demonstrated [3] (and our center delivered confirmative data [1]) that fetal OSB repair indeed is able to preserve neurologic lower extremity function and dramatically decreases rates of shunt-dependent hydrocephalus, the effect on the urological outcome remains unclear. Several studies on the urological outcome after in utero OSB repair were published [4][5][6][7][8][9][10][11][12][13][14][15][16][17], however reporting controversial results. The aim of this study was to further increment urological outcome data by analyzing our own cohort of patients who underwent in utero OSB repair, and to demonstrate that our earlier published positive preliminary results [7] are permanent.…”

Section: Introductionmentioning

confidence: 99%

Urological Outcome after Fetal Spina Bifida Repair: Data from the Zurich Cohort

et al. 2020

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Introduction: Neurogenic lower urinary tract dysfunction (NLUTD) represents a severe burden for patients with open spina bifida (OSB). The effect of fetal OSB repair on the urological outcome remains unclear, as controversial data exist. The aim of this study was to further increment existing outcome data and to demonstrate that our earlier published positive preliminary results are not erratic. Methods: Data from standardized urological follow-up appointments of patients with fetal OSB repair operated at our center were analyzed. Data were obtained from urodynamic studies (UDSs) and radiologic exams performed in the newborn (gestational age 37-39 weeks), at ages of 6, 12, 18, and 24 months, and then at yearly intervals. Results: Of 82 patients (mean age 2.6 years, range 6 months to 7 years), 26 (32%) had a normal bladder function as demonstrated by UDSs. Of the 56 (68%) patients with NLUTD, 29 (51%) patients showed initially a normal UDS, but developed NLUTD in the follow-up, 19 (66%) of them spontaneously and another 10 (34%) in association with growth and development, or surgery of inclusion cysts. Radiologic abnormalities (upper tract dilatation and vesico-uretero-renal reflux) were seen in 15%, mainly patients with NLUTD. Conclusion: Our results add an important set of information to the existing body of evidence. The data reconfirm our earlier published favorable preliminary results and support other studies that show a possible benefit of prenatal OSB repair on the urological outcome, but they also demonstrate that the positive effect remains limited.

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Section: Introductionmentioning

confidence: 99%

Urological Outcome after Fetal Spina Bifida Repair: Data from the Zurich Cohort

et al. 2020

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“…Since first MMC closure 1998 (44), nine reports of urological outcomes on 291 cases have been published worldwide (45–52). Most reports that compared early bladder function of MMC patients treated pre- or post-natally failed to show nay advantages.…”

Section: Resultsmentioning

confidence: 99%

Effectiveness of Prenatal Intervention on the Outcome of Diseases That Have a Postnatal Urological Impact

et al. 2019

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We reviewed the literature addressing outcomes of fetal intervention of conditions that require post-natal urological management including lower urinary tract obstruction, hydrometrocolpos, congenital adrenal hyperplasia, and myelomeningocele. Despite several decades of fetal intervention for these conditions, benefits remain elusive in part because of the enormous difficulty of conducting prospective randomized studies. In this review, we reached the following conclusions: Prenatal intervention in lower urinary tract obstruction (LUTO) improves survival in the worst cases by improving pulmonary function and it may be advantageous for renal function, particularly in patients with adverse urine parameters. Prenatal treatment of female fetuses at risk of having congenital adrenal hyperplasia (CAH) reduces virilization. Non-invasive fetal DNA analysis allows earlier diagnosis, reducing the risk of treating with dexamethasone males and non-affected fetuses. Fetal treatment of myelomeningocele (MMC) is gaining momentum. Prospective studies including standardized urologic evaluation will determine if the encouraging results reported by some hold on the long term follow-up.

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“…The initial evaluation, with an average age of 5.8 months, showed high risk in 52.6%, incontinent in 27.4%, with underactive bladder in 4.2% and only 14.7% had a normal bladder profile. CIC was initiated in 57.3% of the patients and anticholinergics in 52.6% 10 . In another comparative study involving patients from the same database, we evaluated 88 patients in group 1 (prenatal surgery), 86 in group 2 (postnatal surgery), and a third group, subgroup 2, with 38 patients who started urological evaluation at <12 months of age.…”

Section: Discussionmentioning

confidence: 99%

In utero myelomeningocele repair: The natural history of patients with incontinent pattern (sphincteric deficiency: leakage below 40 CMH20)

Macedo

Migueli²,

Cavalheiro

et al. 2020

Neurourology and Urodynamics

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Introduction: Since 2011 we have been following prospectively myelomeningocele patients treated in utero with particular interest to patients with sphincter weakness/deficiency. We investigated the changes of bladder pattern and upper urinary tract with time in children who underwent in utero repair and had low-pressure incontinence based on urodynamic evaluation (UE). Materials and Methods: From the 120 patients in our database, 117 had at least one UE. Of these, 30 were classified as incontinent when leaking at low pressure (<40 cmH20). We reviewed clinical evaluation, urinary tract ultrasound, voiding cystourethrography (VCUG), and other UE parameters at first and last evaluation. Results: We found 30 cases (25.64%). Mean age at initial evaluation was 4.97 months followed by UE done initially at mean age of 5.73 months. Follow-up was 28.4 months. Febrile urinary tract infection has been found in four patients (13.3%), hydronephrosis in four patients, and bladder neck thickening in three (10%). The VCUG showed vesicoureteral reflux in three cases (3/27, 11.1%). A total of 90% of patients had detrusor overactivity with mean maximum detrusor pressure (33.37 cmH20). Only 16.67% of patients showed normal bladder capacity. From the 30 patients, 23 had at least two UE. We noticed a change of bladder pattern as follows: six patients became of high-risk pattern, five normal, and two with underactive bladder pattern. The average interval between the first and last UE was 25.5 months (median: 15 months). Conclusion: We concluded that 43.47% of patients with low DLPP have kept the incontinent pattern. If the initial LPP was below 30 cmH20, 70% remained with the incontinet pattern.

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In utero myelomeningocoele repair and urological outcomes: the first 100 cases of a prospective analysis. Is there an improvement in bladder function?

Cited by 22 publications

References 13 publications

Urological Outcome after Fetal Spina Bifida Repair: Data from the Zurich Cohort

Urological Outcome after Fetal Spina Bifida Repair: Data from the Zurich Cohort

Effectiveness of Prenatal Intervention on the Outcome of Diseases That Have a Postnatal Urological Impact

In utero myelomeningocele repair: The natural history of patients with incontinent pattern (sphincteric deficiency: leakage below 40 CMH20)

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