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Article of the week: In utero myelomeningocele repair and urological outcomes: the first 100 cases of a prospective analysis

Every week, the Editor-in-Chief selects an Article of the Week from the current issue of BJUI. The abstract is reproduced below and you can click on the button to read the full article, which is freely available to all readers for at least 30 days from the time of this post.

In addition to the article itself, there is an editorial written by a prominent member of the urological community and a podcast produced by our Resident podcasters, Giulia Lane and Kyle Johnson. The authors have also kindly produced a video describing their work.

These are intended to provoke comment and discussion and we invite you to use the comment tools at the bottom of each post to join the conversation. 

If you only have time to read one article this week, it should be this one.

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

Antonio Macedo Jr*, Sergio Leite Ottoni*, Gilmar Garrone*, Riberto Liguori*, Sergio Cavalheiro§, Antonio Moron¶§ and Marcela Leal Da Cruz*

 

*Department of Urology, CACAU-NUPEP, Department of Pediatrics, Federal University of São Paulo, Department of Neurosurgery, Federal University of São Paulo, §Santa Joana Maternity Hospital, and Department of Obstetrics-Fetal Medicine, Federal University of São Paulo, São Paulo, Brazil

 

Abstract

Objectives

To evaluate the first 100 cases of in utero myelomeningocele (MMC) repair and urological outcomes in a prospective analysis aiming to define possible improvement in bladder function.

Patients and methods

We used a protocol consisting of a detailed medical history, urinary tract ultrasonography, voiding cystourethrography, and urodynamic evaluation. Patients were categorised into four groups: normal, high risk (overactive bladder with a detrusor leak‐point pressure >40 cm H2O and high filling pressures also >40 cm H2O), incontinent, and underactivity (underactive bladder with post‐void residual urine), and patients were treated accordingly.

Fig.2. Hydronephrosis in relation to the underlying bladder pattern.

Results

We evaluated 100 patients, at a mean age of 5.8 months (median 4 months), classified as high risk in 52.6%, incontinent in 27.4%, with underactive bladder in 4.2%, and only 14.7% had a normal bladder profile. Clean intermittent catheterisation was initiated in 57.3% of the patients and anticholinergics in 52.6%. Antibiotic prophylaxis was initiated in 19.1% of the patients presenting with vesico‐ureteric reflux.

Conclusion

The high incidence of abnormal bladder patterns suggests little benefit of in utero MMC surgery concerning the urinary tract.

 

Editorial: Bladder function and fetal treatment of myelomeningocele

In utero myelomeningocele repair and urological outcome: the first 100 cases of a prospective analysis. Is there an improvement in bladder function? Comments on bladder function and fetal treatment of myelomeningocele [1].

Prenatal care with maternal screening for neural tube defects and high‐resolution maternal fetal sonography has led to the early diagnosis of fetal myelomeningocele [2]. Revolutionary fetal surgery to correct myelomeningocele in utero has reduced the need for cerebrospinal fluid shunting and improved motor outcomes in these babies, based on 30‐month follow‐up data [3]. Sponsored by the National Institute of Health, the prospective randomized ‘Management of Myelomeningocele Study’ (MOMS) trial documented the outcomes of 158 patients assessed after either fetal repair prior to 26 weeks’ gestation or standard postnatal repair of the myelomeningocele defect. The trial was stopped early when evaluation showed that the primary outcome, rate of shunt placement, in the postnatal repair group (82%) was approximately double that in the prenatal surgery group (40%). Prenatal surgery also resulted in improvement in outcomes for mental development, motor function and ambulation, also evaluated at 30 months postnatally. However, prenatal surgery was associated with an increased risk of preterm delivery and uterine dehiscence at the time of delivery.

Bladder function was also evaluated at 30 months in the MOMS trial, comparing the need for clean intermittent catheterization (CIC) in 115 patients with adequate urological follow‐up, consisting of clinical outcomes in respect to continence, sonographic appearance of the kidneys and bladder and urodynamic evaluation [4]. Prenatal surgery did not significantly reduce the need for CIC measured at 30 months of age, but was associated with less bladder trabeculation and open bladder neck. Longer follow‐up was recommended and is in progress to document further bladder outcomes.

Macedo et al. [1] report similar short‐term bladder outcomes in 100 patients undergoing in utero myelomeningocele repair. Their report documented bladder characteristics in these patients at a mean postnatal age of ~6 months. In their unique cohort, antenatal diagnosis of fetal myelomeningocele was made at ~21 weeks’ gestation, in utero surgery was performed at ~25.5 weeks’ gestation and preterm birth occurred at ~33 weeks’ gestation, parameters consistent with the patients in the MOMs trial. Short‐term evaluation showed that ~53% of the patients had high‐risk bladders with poor compliance, ~27% were incontinent with weak sphincteric activity and only ~15% had normal urodynamic profiles. CIC was initiated in ~ 57% of the Macedo et al. cohort, again similar to the MOMs trial. The long‐term outcomes after potty training and during childhood and adolescence will be especially interesting in this valuable cohort.

Some of the pitfalls that will need to be accounted for include the validation and standardization of urodynamic testing [5]. Even at the same institution with clinicians who have undergone similar training, consistent interpretation of urodynamic studies can be variable, potentially affecting therapeutic options [6].

The goal of patients, parents and providers is to avoid the urological sequelae of myelomeningocele. To date, the reality is that the majority of these children, whether or not they have undergone in utero fetal repair or postnatal surgery, will require the assistance of CIC for urological health to protect the kidneys from excess pressure, to facilitate bladder emptying and urinary continence and prevent UTI.

 

References

  1. Macedo, AOttoni, SLGarrone, G et al. In utero myelomeningocele repair and urological outcome: the first 100 cases of a prospective analysis. Is there an improvement in bladder function? BJU Int 2019123676– 81
  2. Meller, CAiello, HOtano, LSonographic detection of open spina bifida in the first trimester: review of the literature. Childs Nerv Syst 2017331101– 6
  3. Adzick, NS, Thom, EASpong, CY et al. A randomized trial of prenatal versus postnatal repair of myelomeningocele. N Engl J Med 2011364993– 1004
  4. Brock, JWCarr, MCAdzick, NS et al. Bladder Function After Fetal Surgery for Myelomeningocele. Pediatrics 2015136e906– 13
  5. Bauer, SB, Nijman, RJDrzewiecki, BASillen, UHoebeke, P, International Children’s Continence Society Standardization Subcommittee. International Children’s Continence Society standardization report on urodynamic studies of the lower urinary tract in children. Neurourol Urodyn 201534640– 7
  6. Dudley, AGCasella, DPLauderdale, CJ et al. Interrater reliability in pediatric urodynamic tracings: a pilot study. J Urol 2017197865– 70

 

Residents’ podcast: In utero myelomeningocele repair and urological outcomes

Giulia Lane M.D. is a Fellow in Neuro-urology and Pelvic Reconstruction in the Department of Urology at the University of Michigan; Kyle Johnson is a Urology Resident in the same department.

In this podcast they discuss the following BJUI Article of the Week:

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

Abstract

Objectives

To evaluate the first 100 cases of in utero myelomeningocele (MMC) repair and urological outcomes in a prospective analysis aiming to define possible improvement in bladder function.

Patients and methods

We used a protocol consisting of a detailed medical history, urinary tract ultrasonography, voiding cystourethrography, and urodynamic evaluation. Patients were categorised into four groups: normal, high risk (overactive bladder with a detrusor leak‐point pressure >40 cm H2O and high filling pressures also >40 cm H2O), incontinent, and underactivity (underactive bladder with post‐void residual urine), and patients were treated accordingly.

Results

We evaluated 100 patients, at a mean age of 5.8 months (median 4 months), classified as high risk in 52.6%, incontinent in 27.4%, with underactive bladder in 4.2%, and only 14.7% had a normal bladder profile. Clean intermittent catheterisation was initiated in 57.3% of the patients and anticholinergics in 52.6%. Antibiotic prophylaxis was initiated in 19.1% of the patients presenting with vesico‐ureteric reflux.

Conclusion

The high incidence of abnormal bladder patterns suggests little benefit of in utero MMC surgery concerning the urinary tract.

 

BJUI Podcasts now available on iTunes, subscribe here https://itunes.apple.com/gb/podcast/bju-international/id1309570262

 

 

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

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

Abstract

 

Objectives

To evaluate the first 100 cases of in utero myelomeningocele (MMC) repair and urological outcomes in a prospective analysis aiming to define possible improvement in bladder function.

Patients and methods

We used a protocol consisting of a detailed medical history, urinary tract ultrasonography, voiding cystourethrography, and urodynamic evaluation. Patients were categorised into four groups: normal, high risk (overactive bladder with a detrusor leak‐point pressure >40 cm H2O and high filling pressures also >40 cm H2O), incontinent, and underactivity (underactive bladder with post‐void residual urine), and patients were treated accordingly.

Results

We evaluated 100 patients, at a mean age of 5.8 months (median 4 months), classified as high risk in 52.6%, incontinent in 27.4%, with underactive bladder in 4.2%, and only 14.7% had a normal bladder profile. Clean intermittent catheterisation was initiated in 57.3% of the patients and anticholinergics in 52.6%. Antibiotic prophylaxis was initiated in 19.1% of the patients presenting with vesico‐ureteric reflux.

Conclusion

The high incidence of abnormal bladder patterns suggests little benefit of in utero MMC surgery concerning the urinary tract.

 

Article of the week: Testicular asymmetry in healthy adolescent boys

Every week, the Editor-in-Chief selects an Article of the Week from the current issue of BJUI. The abstract is reproduced below and you can click on the button to read the full article, which is freely available to all readers for at least 30 days from the time of this post.

In addition to the article itself, there is an accompanying editorial written by a prominent member of the urological community. This blog is intended to provoke comment and discussion and we invite you to use the comment tools at the bottom of each post to join the conversation. The authors have also supplied a video to accompany the article.

If you only have time to read one article this week, it should be this one.

Testicular asymmetry in healthy adolescent boys

Donald Vaganee*† , Frederik Daems*, William Aerts*, Rosina Dewaide*, Tinne van den Keybus*, Karen De Baets, Stefan De Wachter*† and Gunter De Win*†

*Faculty of Medicine and Health Sciences, University of Antwerp, Antwerp and Department of Urology, Antwerp University Hospital, Edegem, Belgium

Abstract

Objectives

To assess the presence of testicular asymmetry and the currently used threshold values in varicocoele management in a healthy adolescent population.

Subjects and Methods

We conducted an observational cross‐sectional study from April 2015 until December 2016 in which we recruited 539 adolescent boys aged 11–16 years. A clinical examination including testicular size measurement by ultrasonography was performed. Testicular volume (TV) was calculated using the Lambert formula (length × width × height × 0.71). The Testicular Atrophy Index (TAI) was calculated using the formula [(TV right – TV left)/largest TV] × 100. The data for all statistical analyses were stratified for Tanner stage for genital development (TSG) and pubic hair (TSP). Non‐parametric tests were used to assess the difference between right and left TV, and the prevalence of a smaller left testis for the entire population, and between each TSG and TSP. Parametric tests were used to determine the difference in mean TAI between each TSG and TSP, and to compare the mean TAI to a test value of 0.

Results

Of the 539 recruited boys, we excluded 194 due to a current or past pathology, including varicocoeles, influencing normal (testicular) growth or due to incomplete data. Most boys were in the second Tanner stage, followed by the third Tanner stage. The mean (sd) age of the entire population was 13.33 (1.25) years. Of the 345 included participants the mean (sd) left TV was 7.67 (5.63) mL and right TV was 7.97 (5.90) mL. The mean (sd) TAI was 2.85 (17.00)%. In all, 203 (58.84%) boys had a smaller left testis and 142 (41.16%) had a smaller right testis. In all, 51 boys (14.78%) had a TAI >20%, 45 (13.04%) had a TV difference (TVD) of >2 mL with a deficit in left TV, and 69 (20.00)% had a TAI >20% or a TVD of >2 mL with a deficit in left TV. Related‐samples Wilcoxon signed‐rank test showed a significant difference in mean left and right TV for the entire population, and more specifically for TSG3 (P < 0.001) and TSP3 (P = 0.004). A one‐sample t‐test showed a significant difference in the mean TAI vs the test value of 0 for the entire population (P = 0.002), and more specifically for TSG3 (P < 0.001) and TSP3 (P = 0.003).

Conclusion

Testicular asymmetry, with a smaller left testis, was seen in a considerable number of healthy adolescents. One out of five adolescents had a smaller left testis and met one of the threshold values currently used in varicocoele management. Therefore, in left‐sided unilateral inguinoscrotal pathology, a smaller ipsilateral testis in combination with a TAI of >20% and/or TVD of >2 mL requires careful interpretation and serial measurements of TV should always be performed. Furthermore, this study provides reference values for TV, TVD and TAI according to TSG and TSP for a healthy adolescent population.

Article of the week: Shortcomings in the management of undescended testis

Every week, the Editor-in-Chief selects an Article of the Week from the current issue of BJUI. The abstract is reproduced below and you can click on the button to read the full article, which is freely available to all readers for at least 30 days from the time of this post.

In addition to the article itself, there is an accompanying editorial written by a prominent member of the urological community. This blog is intended to provoke comment and discussion and we invite you to use the comment tools at the bottom of each post to join the conversation. The authors have also provided a video explanation of their work.

If you only have time to read one article this week, it should be this one.

Shortcomings in the management of undescended testis: guideline intention vs reality and the underlying causes – insights from the biggest German cohort

Philip Boehme*†, Berit Geis‡, Johannes Doerner§, Stefan Wirth* and Kai O. Hensel*

*Witten/Herdecke University, Department of Paediatrics, Centre for Clinical and Translational Research, Wuppertal University Hospital, Wuppertal, Germany; †Cardiovascular Research, Bayer Pharma AG, Wuppertal, Germany; ‡Institute of Medical Biometry and Epidemiology, Faculty of Health, Witten/Herdecke University, Witten, Germany; and §Witten/Herdecke University, Department of Surgery, Centre for General and Visceral Surgery, HELIOS University Hospital Wuppertal, Wuppertal, Germany

Abstract

Objectives

To assess the implementation of the current guideline and identify potential underlying causes for late surgery in children with undescended testis (UDT) in Germany. UDT is the most common surgical issue in paediatric urology and to avoid malignant degeneration and subfertility current guidelines recommend orchidopexy during the first year of life; however, this seems not to be implemented in practice.

Patients and Methods

In all, 5 547 patients with cryptorchidism at 16 hospitals nationwide were studied regarding age at orchidopexy between 2003 and 2016. Multivariate analysis was performed to identify factors influencing timing of surgery. Additionally, a survey on knowledge of UDT management was conducted amongst physicians treating boys and final‐year medical students.

 

Results

Between 2003 and 2008 only 4% of boys with UDT underwent surgery before the age of 1 year. After the guideline update from 2009, this figure was 5% from 2010 to 2012, and 8% from 2013 to 2016. The presence of a specialised department for paediatric surgery, as well as a high UDT case‐to‐year ratio positively influenced the timing of orchidopexy. The survey revealed discipline‐specific differences in the levels of knowledge about UDT management. One‐third of respondents did not know the guideline recommendations and 61% felt insufficiently informed. International comparisons revealed significant differences in the age at surgery of boys with UDT, with Germany and Great Britain ranging in the middle of the field.

Conclusions

Currently, only a small proportion of boys with UDT are operated upon during their first year of life. The level of knowledge in attending physicians remains in need of improvement. This should be actively addressed, i.e. by campaigns and educational programmes. Further studies are needed to investigate the underlying causes of late orchidopexy in UDT.

 

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