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American Journal of Public Health Jan 2016Birth defects remain a significant source of worldwide morbidity and mortality. Strong scientific evidence shows that folic acid fortification of a region's food supply... (Meta-Analysis)
Meta-Analysis Review
BACKGROUND
Birth defects remain a significant source of worldwide morbidity and mortality. Strong scientific evidence shows that folic acid fortification of a region's food supply leads to a decrease in spina bifida (a birth defect of the spine). Still, many countries around the world have yet to approve mandatory fortification through government legislation.
OBJECTIVES
We sought to perform a systematic review and meta-analysis of period prevalence of spina bifida by folic acid fortification status, geographic region, and study population.
SEARCH METHODS
An expert research librarian used terms related to neural tube defects and epidemiology from primary research from 1985 to 2010 to search in EMBASE and MEDLINE. We searched the reference lists of included articles and key review articles identified by experts.
SELECTION CRITERIA
Inclusion criteria included studies in English or French reporting on prevalence published between January 1985 and December 2010 that (1) were primary research, (2) were population-based, and (3) reported a point or period prevalence estimate of spina bifida (i.e., prevalence estimate with confidence intervals or case numerator and population denominator). Two independent reviewers screened titles and abstracts for eligible articles, then 2 authors screened full texts in duplicate for final inclusion. Disagreements were resolved through consensus or a third party.
DATA COLLECTION AND ANALYSIS
We followed Preferred Reporting Items for Systematic Reviews and Meta-Analyses, or PRISMA, abstracting data related to case ascertainment, study population, folic acid fortification status, geographic region, and prevalence estimate independently and in duplicate. We extracted overall data and any subgroups reported by age, gender, time period, or type of spina bifida. We classified each period prevalence estimate as "mandatory" or "voluntary" folic acid fortification according to each country's folic acid fortification status at the time data were collected (as determined by a well-recognized fortification monitoring body, Food Fortification Initiative). We determined study quality on the basis of sample representativeness, standardization of data collection and birth defect assessment, and statistical analyses. We analyzed study-level period prevalence estimates by using a random effects model (α level of < 0.05) for all meta-analyses. We stratified pooled period prevalence estimates by birth population, fortification status, and continent.
RESULTS
Of 4078 studies identified, we included 179 studies in the systematic review and 123 in a meta-analysis. In studies of live births (LBs) alone, period prevalences of spina bifida were (1) lower in geographical regions with mandatory (33.86 per 100,000 LBs) versus voluntary (48.35 per 100,000 LBs) folic acid fortification, and (2) lower in studies of LBs, stillbirths, and terminations of pregnancy in regions with mandatory (35.22 per 100,000 LBs) versus voluntary (52.29 per 100,000 LBs) fortification. In LBs, stillbirths, and terminations of pregnancy studies, the lowest pooled prevalence estimate was in North America (38.70 per 100,000). Case ascertainment, surveillance methods, and reporting varied across these population-based studies.
CONCLUSIONS
Mandatory legislation enforcing folic acid fortification of the food supply lags behind the evidence, particularly in Asian and European countries. This extensive literature review shows that spina bifida is significantly more common in world regions without government legislation regulating full-coverage folic acid fortification of the food supply (i.e., Asia, Europe) and that mandatory folic acid fortification resulted in a lower prevalence of spina bifida regardless of the type of birth cohort. African data were scarce, but needed, as many African nations are beginning to adopt folic acid legislation.
Topics: Female; Folic Acid; Food, Fortified; Global Health; Humans; Pregnancy; Prevalence; Spinal Dysraphism; Vitamin B Complex
PubMed: 26562127
DOI: 10.2105/AJPH.2015.302902 -
The Cochrane Database of Systematic... Apr 2016There is evidence that the process of transition from paediatric (child) to adult health services is often associated with deterioration in the health of adolescents... (Review)
Review
BACKGROUND
There is evidence that the process of transition from paediatric (child) to adult health services is often associated with deterioration in the health of adolescents with chronic conditions.Transitional care is the term used to describe services that seek to bridge this care gap. It has been defined as 'the purposeful, planned movement of adolescents and young adults with chronic physical and medical conditions from child-centred to adult-oriented health care systems'. In order to develop appropriate services for adolescents, evidence of what works and what factors act as barriers and facilitators of effective interventions is needed.
OBJECTIVES
To evaluate the effectiveness of interventions designed to improve the transition of care for adolescents from paediatric to adult health services.
SEARCH METHODS
We searched The Cochrane Central Register of Controlled Trials 2015, Issue 1, (including the Cochrane Effective Practice and Organisation of Care Group Specialised Register), MEDLINE, EMBASE, PsycINFO, and Web of Knowledge to 19 June 2015. We also searched reference lists of included studies and relevant reviews, and contacted experts and study authors for additional studies.
SELECTION CRITERIA
We considered randomised controlled trials (RCTs), controlled before- and after-studies (CBAs), and interrupted time-series studies (ITSs) that evaluated the effectiveness of any intervention (care model or clinical pathway), that aimed to improve the transition of care for adolescents from paediatric to adult health services. We considered adolescents with any chronic condition that required ongoing clinical care, who were leaving paediatric services and going on to receive services in adult healthcare units, and their families. Participating providers included all health professionals responsible for the care of young people.
DATA COLLECTION AND ANALYSIS
Two review authors independently extracted data from included papers, assessed the risk of bias of each study, and assessed the certainty of the evidence for the main comparisons using GRADE. Discrepancies were resolved by discussion. Authors were contacted for missing data. We reported the findings of the studies as pre- and post-intervention means and calculated the unadjusted absolute change from baseline with 95% confidence intervals (CI).
MAIN RESULTS
We included four RCTs (N = 238 participants) that explored: a two-day workshop-based transition preparation training for adolescents with spina bifida; a nurse-led, one-on-one, teaching session with the additional support of a 'health passport' for adolescents with heart disease; a web- and SMS-based educational intervention for adolescents with a range of different conditions; and a structured comprehensive transition programme with a transition co-ordinator for adolescents with type 1 diabetes.One study evaluating a one-on-one nurse-led intervention, and one evaluating a technology-based intervention suggested that these interventions may lead to slight improvements in transitional readiness and chronic disease self-management measured at six- to eight-month follow-ups (low certainty evidence). Results with the TRAQ self-management tool were: MD 0.20; 95% CI -0.16 to 0.56 and MD 0.43; 95% CI; -0.09 to 0.95; with the TRAQ self-advocacy tool: MD 0.37; 95% CI -0.06 to 0.80; and with the PAM tool were: MD 10; 95% CI 2.96 to 17.04. In contrast, transition-preparation training delivered via a two-day workshop for patients with spina bifida may lead to little or no difference in measures of self-care practice and general health behaviours when measured using the DSCPI-90©.Two studies evaluated the use of health services. One study evaluated a technology-based intervention and another a comprehensive transition programme; these interventions may lead to slightly more young people taking positive steps to initiate contact with health professionals themselves (Relative risk (RR): 4.87; 95% CI 0.24 to 98.12 and RR 1.50; 95% CI 0.32 to 6.94, respectively; low certainty evidence.Young people's knowledge of their disease may slightly improve with a nurse-led, one-on-one intervention to prepare young people for transition to an adult congenital heart programme (MD 14; 95% CI 2.67 to 25.33; one study; low certainty evidence).Disease-specific outcome measures were reported in two studies, both of which led to little or no difference in outcomes (low certainty evidence). One study found little or no difference between intervention and control groups. A second study found that follow-up HbA1c in young people with type 1 diabetes mellitus increased by 1.2% for each percentage increase in baseline HbA1c, independent of treatment group (1.2%; 95% CI 0.4 to 1.9; P = 0.01).Transition interventions may lead to little or no difference in well-being or quality of life as measured with the PARS III or PedsQ (two studies; low certainty evidence). Both the technology-based intervention and the two-day workshop for young people with spina bifida found little or no difference between intervention and control groups (MD 1.29; 95% CI -4.49 to 7.07). One study did not report the data.Four telephone support calls from a transition co-ordinator may lead to little or no difference in rates of transfer from paediatric to adult diabetes services (one study; low certainty evidence). At 12-month follow-up, there was little or no difference between groups of young people receiving usual care or a telephone support (RR 0.80; 95% CI 0.59 to 1.08)). They may slightly reduce the risk of disease-related hospital admissions at 12-month follow-up (RR 0.29; 95% CI 0.03 to 2.40).
AUTHORS' CONCLUSIONS
The available evidence (four small studies; N = 238), covers a limited range of interventions developed to facilitate transition in a limited number of clinical conditions, with only four to 12 months follow-up. These follow-up periods may not be long enough for any changes to become apparent as transition is a lengthy process. There was evidence of improvement in patients' knowledge of their condition in one study, and improvements in self-efficacy and confidence in another, but since few studies were eligible for this review, and the overall certainty of the body of this evidence is low, no firm conclusions can be drawn about the effectiveness of the evaluated interventions. Further research is very likely to have an important impact on our confidence in the intervention effect and likely could change our conclusions. There is considerable scope for the rigorous evaluation of other models of transitional care, reporting on clinical outcomes with longer term follow-up.
Topics: Adolescent; Adolescent Health Services; Chronic Disease; Diabetes Mellitus, Type 1; Heart Diseases; Humans; Practice Patterns, Nurses'; Randomized Controlled Trials as Topic; Self Care; Spinal Dysraphism; Transition to Adult Care
PubMed: 27128768
DOI: 10.1002/14651858.CD009794.pub2 -
Scientifica 2020Spina bifida is an abnormal closure of the neural tube during the fourth week of development. It is the major cause of fetal loss and considerable disabilities in... (Review)
Review
Spina bifida is an abnormal closure of the neural tube during the fourth week of development. It is the major cause of fetal loss and considerable disabilities in newborns. The aim of this review is to determine the pooled prevalence of spina bifida among newborns in Africa. PubMed/Medline, Google Scholar, Science Direct, Joanna Briggs Institute (JBI) Library, Cochrane Library, Web of Science, African Journals Online, and Embase databases were systematically searched. Cochran Q test and test statistics were applied to assess heterogeneity across studies. A random-effect model was applied to calculate the pooled prevalence of spina bifida. Forest plot and Galbraith's plot were used to visualize heterogeneity. Subgroup, sensitivity, meta-regression, and meta-cumulative analyses were performed. All essential data were extracted using a standardized data extraction format, and the JBI quality appraisal checklist was used to assess the quality of studies. Egger's test and Begg's test were used in order to detect the publication bias. In the present systematic review and meta-analysis, 6,587,298 births in twenty-seven studies were included. The pooled birth prevalence of spina bifida in Africa was 0.13% with a range between 0.12% and 0.14%. In Africa, the highest burden of spina bifida was detected in Algeria (0.43%), Ethiopia (0.32%), Tanzania (0.26%), Cameron (0.12%), Egypt (0.10%), and South Africa (0.10%). The lowest burden of spina bifida was detected in Libya (0.006%) and Tunisia (0.009%). The high birth prevalence of spina bifida was detected in Africa. There was a significant variation in the prevalence of spina bifida among study countries in Africa. The authors recommend that special awareness creation with the help of health education intervention should be provided for mothers to focus on prevention in order to reduce the burden of spina bifida.
PubMed: 33083093
DOI: 10.1155/2020/4273510 -
PloS One 2021A systematic review was conducted in high-income country settings to analyse: (i) spina bifida neonatal and IMRs over time, and (ii) clinical and socio-demographic... (Meta-Analysis)
Meta-Analysis
OBJECTIVES
A systematic review was conducted in high-income country settings to analyse: (i) spina bifida neonatal and IMRs over time, and (ii) clinical and socio-demographic factors associated with mortality in the first year after birth in infants affected by spina bifida.
DATA SOURCES
PubMed, Embase, Ovid, Web of Science, CINAHL, Scopus and the Cochrane Library were searched from 1st January, 1990 to 31st August, 2020 to review evidence.
STUDY SELECTION
Population-based studies that provided data for spina bifida infant mortality and case fatality according to clinical and socio-demographical characteristics were included. Studies were excluded if they were conducted solely in tertiary centres. Spina bifida occulta or syndromal spina bifida were excluded where possible.
DATA EXTRACTION AND SYNTHESIS
Independent reviewers extracted data and assessed their quality using MOOSE guideline. Pooled mortality estimates were calculated using random-effects (+/- fixed effects) models meta-analyses. Heterogeneity between studies was assessed using the Cochrane Q test and I2 statistics. Meta-regression was performed to examine the impact of year of birth cohort on spina bifida infant mortality.
RESULTS
Twenty studies met the full inclusion criteria with a total study population of over 30 million liveborn infants and approximately 12,000 spina bifida-affected infants. Significant declines in spina bifida associated infant and neonatal mortality rates (e.g. 4.76% decrease in IMR per 100, 000 live births per year) and case fatality (e.g. 2.70% decrease in infant case fatality per year) were consistently observed over time. Preterm birth (RR 4.45; 2.30-8.60) and low birthweight (RR 4.77; 2.67-8.55) are the strongest risk factors associated with increased spina bifida infant case fatality.
SIGNIFICANCE
Significant declines in spina bifida associated infant/neonatal mortality and case fatality were consistently observed, advances in treatment and mandatory folic acid food fortification both likely play an important role. Particular attention is warranted from clinicians caring for preterm and low birthweight babies affected by spina bifida.
Topics: Female; Humans; Infant; Infant Mortality; Infant, Newborn; Pregnancy; Premature Birth; Spinal Dysraphism
PubMed: 33979363
DOI: 10.1371/journal.pone.0250098 -
Neuroradiology Feb 2022Open spina bifida (OSB) encompasses a wide spectrum of intracranial abnormalities. With foetal surgery as a new treatment option, robust intracranial imaging is... (Meta-Analysis)
Meta-Analysis Review
PURPOSE
Open spina bifida (OSB) encompasses a wide spectrum of intracranial abnormalities. With foetal surgery as a new treatment option, robust intracranial imaging is important for comprehensive preoperative evaluation and prognostication. We aimed to determine the incidence of infratentorial and supratentorial findings detected by magnetic resonance imaging (MRI) alone and MRI compared to ultrasound.
METHODS
Two systematic reviews comparing MRI to ultrasound and MRI alone were conducted on MEDLINE, EMBASE, and Cochrane databases identifying studies of foetal OSB from 2000 to 2020. Intracranial imaging findings were analysed at ≤ 26 or > 26 weeks gestation and neonates (≤ 28 days). Data was independently extracted by two reviewers and meta-analysis was performed where possible.
RESULTS
Thirty-six studies reported brain abnormalities detected by MRI alone in patients who previously had an ultrasound. Callosal dysgenesis was identified in 4/29 cases (2 foetuses ≤ 26 weeks, 1 foetus under any gestation, and 1 neonate ≤ 28 days) (15.1%, CI:5.7-34.3%). Heterotopia was identified in 7/40 foetuses ≤ 26 weeks (19.8%, CI:7.7-42.2%), 9/36 foetuses > 26 weeks (25.3%, CI:13.7-41.9%), and 64/250 neonates ≤ 28 days (26.9%, CI:15.3-42.8%). Additional abnormalities included aberrant cortical folding and other Chiari II malformation findings such as lower cervicomedullary kink level, tectal beaking, and hypoplastic tentorium. Eight studies compared MRI directly to ultrasound, but due to reporting inconsistencies, it was not possible to meta-analyse.
CONCLUSION
MRI is able to detect anomalies hitherto underestimated in foetal OSB which may be important for case selection. In view of increasing prenatal OSB surgery, further studies are required to assess developmental consequences of these findings.
Topics: Brain; Female; Fetus; Humans; Infant, Newborn; Magnetic Resonance Imaging; Pregnancy; Spinal Dysraphism; Ultrasonography, Prenatal
PubMed: 34792623
DOI: 10.1007/s00234-021-02853-1 -
Translational Andrology and Urology May 2018Urinary tract infection (UTI) is a source of morbidity and healthcare costs in adults with spina bifida (ASB). UTI prevention strategies are often recommended, but the... (Review)
Review
Urinary tract infection (UTI) is a source of morbidity and healthcare costs in adults with spina bifida (ASB). UTI prevention strategies are often recommended, but the evidence of various approaches remains unclear. We performed a systematic review to inform a best practice policy statement for UTI prevention in ASB. On behalf of the Neurogenic Bladder Research Group (NBRG.org), we developed an a priori protocol and searched the published English literature for 30 outcomes questions addressing UTI prevention in ASB. The questions spanned the categories of antibiotics, oral supplements, bladder management factors and social support. Where there was little literature in ASB, we included literature from similar populations with neurogenic bladder (NB). Data was abstracted and then reviewed with recommendations made by consensus of all authors. Level of Evidence (LoE) and Grade of Recommendation (GoR) were according to the Oxford grading system. Of 6,433 articles identified by our search, we included 99 publications. There was sufficient evidence to support use of the following: saline bladder irrigation (LoE 1, GoR B), gentamicin bladder instillation (LoE 3, GoR C), single-use intermittent catheterization (IC) (LoE 2, GoR B), hydrophilic catheters for IC (LoE 2, GoR C), intradetrusor onabotulinumtoxinA injection (LoE 3, GoR C), hyaluronic acid (HA) instillation (LoE 1, GoR B), and care coordination (LoE 3, GoR C). There was sufficient evidence to recommend against use of the following: sterile IC (LoE 1, GoR B), oral antibiotic prophylaxis (LoE 2, GoR B), treatment of asymptomatic bacteriuria (LoE 2, GoR B), cranberry (LoE 2, GoR B), methenamine salts (LoE 1, GoR B), and ascorbic acid (LoE1, GoR B). There was insufficient evidence to make a recommendation for other outcomes. Overall, there are few studies in UTI prevention in the specific population of ASB. Research in populations similar to ASB helps to guide recommendations for UTI prevention in the challenging patient group of ASB. Future studies in UTI prevention specific to ASB are needed and should focus on areas shown to be of benefit in similar populations.
PubMed: 29928619
DOI: 10.21037/tau.2018.04.21 -
The Journal of Urology Jun 2022We systematically reviewed the variability in definitions of kidney abnormality (KA) outcomes in individuals with spina bifida (SB). (Review)
Review
PURPOSE
We systematically reviewed the variability in definitions of kidney abnormality (KA) outcomes in individuals with spina bifida (SB).
MATERIALS AND METHODS
A systematic scoping review was conducted using MEDLINE, Embase™, Cochrane Library, CINAHL, PsycInfo®, Web of Science™ and ClinicalTrials.gov for articles from time of database inception to September 2020. No language or patient age restrictions were applied. Primary research articles involving individuals with SB where KA was assessed as an outcome were included. Means of assessing KA and defining KA severity were abstracted.
RESULTS
Of 2,034 articles found, 274 were included in the review. Most articles were published after 1990 (63.5%) and included pediatric-only populations (0-18 years; 60.5%). KA outcomes were identified by imaging-based anatomical outcomes (84.7%), serum-based outcomes (44.9%), imaging-based functional outcomes (5.5%), urine-based outcomes (3.3%) and diagnoses of end-stage kidney disease (2.6%) or chronic kidney disease otherwise unspecified (1.8%). Hydronephrosis was the most commonly used specific outcome (64.6%, 177/274) with 19.8% (35/177) of articles defining hydronephrosis severity. Hydronephrosis was used more frequently in articles with pediatric-only populations. Creatinine and cystatin-C were used in 82.1% (101/123) and 17.9% (22/123) of articles reporting serum-based outcomes, respectively, with 32.7% and 50.0% of articles defining estimated glomerular filtration rate (GFR) severity. Serum-based outcomes were more common in articles including adults >18 years. Measured GFR was assessed in 9.9% (27/274) of articles, with 44.4% (12/27) of articles defining GFR severity.
CONCLUSIONS
Significant variability exists in how authors define KA with few specifically defining KA severity. Consensus and consistency in defining KA outcomes are needed.
Topics: Adult; Child; Female; Glomerular Filtration Rate; Humans; Hydronephrosis; Kidney; Male; Renal Insufficiency, Chronic; Spinal Dysraphism
PubMed: 35050700
DOI: 10.1097/JU.0000000000002432 -
Ultrasound in Obstetrics & Gynecology :... Mar 2019Linked Comment: Ultrasound Obstet Gynecol 2018; 53: 293-301 Linked Comment: Ultrasound Obstet Gynecol 2018; 53: 302-308 Linked Comment: Ultrasound Obstet Gynecol 2018;...
Linked Comment: Ultrasound Obstet Gynecol 2018; 53: 293-301 Linked Comment: Ultrasound Obstet Gynecol 2018; 53: 302-308 Linked Comment: Ultrasound Obstet Gynecol 2018; 53: 309-313.
Topics: Female; Fetal Therapies; Humans; Neural Tube Defects; Outcome Assessment, Health Care; Pregnancy; Prenatal Diagnosis; Spinal Dysraphism
PubMed: 30835370
DOI: 10.1002/uog.20226 -
Ultrasound in Obstetrics & Gynecology :... Aug 2016To assess and compare the rate of procedure-related complications after intrauterine treatment of spina bifida by endoscopic surgery and by open fetal surgery. (Comparative Study)
Comparative Study Meta-Analysis Review
Procedure-related complications of open vs endoscopic fetal surgery for treatment of spina bifida in an era of intrauterine myelomeningocele repair: systematic review and meta-analysis.
OBJECTIVE
To assess and compare the rate of procedure-related complications after intrauterine treatment of spina bifida by endoscopic surgery and by open fetal surgery.
METHODS
Systematic literature searches in PubMed and SCOPUS databases were performed on 20 September 2015 to identify randomized controlled trials and observational studies on treatment of human spina bifida by endoscopic or open fetal surgery techniques. Only studies with ≥ 10 cases that were published in or after 2000 were included in the meta-analysis in order to reduce the risk of bias. Primary outcomes (complete dehiscence, focal dehiscence and/or markedly thin hysterotomy scar; preterm delivery < 34 weeks; mean gestational age at delivery) and secondary outcomes (oligohydramnios, prelabor rupture of membranes, placental abruption, chorioamnionitis and perinatal death) were assessed for both techniques. Precision of the estimated proportions was evaluated with 95% CIs. Inconsistency was assessed using the I(2) statistic.
RESULTS
The search identified 1080 records that were examined based on title and abstract, of which 28 full-text articles were examined completely for eligibility. Nine records were excluded because cases were also described in other studies, leaving 19 records for analysis. When comparing endoscopic vs open fetal surgery, the rate of complete dehiscence, focal dehiscence and/or markedly thin hysterotomy scar was, respectively, 1% (95% CI, 0-4%) vs 26% (95% CI, 12-42%); preterm delivery < 34 weeks was 80% (95% CI, 41-100%) vs 45% (95% CI, 38-53%); oligohydramnios was 39% (95% CI, 9-75%) vs 14% (95% CI, 7-24%); prelabor rupture of membranes was 67% (95% CI, 12-100%) vs 38% (95% CI, 26-50%); and perinatal death was 14% (95% CI, 1-38%) vs 5% (95% CI, 3-8%).
CONCLUSION
Open fetal surgery for spina bifida seems to show lower rates of procedure-related complications than does endoscopic surgery, but the rate of hysterotomy scar complications is high after open surgery. Because of the low quality of evidence, the conclusions should be interpreted with caution. Copyright © 2015 ISUOG. Published by John Wiley & Sons Ltd.
Topics: Female; Fetoscopy; Humans; Infant, Newborn; Meningomyelocele; Observational Studies as Topic; Postoperative Complications; Pregnancy; Randomized Controlled Trials as Topic; Spinal Dysraphism; Treatment Outcome
PubMed: 26612040
DOI: 10.1002/uog.15830 -
Ultrasound in Obstetrics & Gynecology :... Jun 2020The Management of Myelomeningocele Study (MOMS) trial demonstrated the safety and efficacy of open fetal surgery for spina bifida aperta (SBA). Recently developed... (Meta-Analysis)
Meta-Analysis
OBJECTIVE
The Management of Myelomeningocele Study (MOMS) trial demonstrated the safety and efficacy of open fetal surgery for spina bifida aperta (SBA). Recently developed alternative techniques may reduce maternal risks without compromising the fetal neuroprotective effects. The aim of this systematic review was to assess the learning curve (LC) of different fetal SBA closure techniques.
METHODS
MEDLINE, Web of Science, EMBASE, Scopus and Cochrane databases and the gray literature were searched to identify relevant articles on fetal surgery for SBA, without language restriction, published between January 1980 and October 2018. Identified studies were reviewed systematically and those reporting all consecutive procedures and with postnatal follow-up ≥ 12 months were selected. Studies were included only if they reported outcome variables necessary to measure the LC, as defined by fetal safety and efficacy. Two authors independently retrieved data, assessed the quality of the studies and categorized observations into blocks of 30 patients. For meta-analysis, data were pooled using a random-effects model when heterogeneous. To measure the LC, we used two complementary methods. In the group-splitting method, competency was defined when the procedure provided results comparable to those in the MOMS trial for 12 outcome variables representing the immediate surgical outcome, short-term neonatal neuroprotection and long-term neuroprotection at ≥ 12 months of age. Then, when raw patient data were available, we performed cumulative sum analysis based on a composite binary outcome defining successful surgery. The composite outcome combined four clinically relevant variables for safety (absence of extreme preterm delivery < 30 weeks, absence of fetal death ≤ 7 days after surgery) and efficacy (reversal of hindbrain herniation and absence of any neonatal treatment of dehiscence or cerebrospinal fluid leakage at the closure site).
RESULTS
Of 6024 search results, 17 (0.3%) studies were included, all of which had low, moderate or unclear risk of bias. Fetal SBA closure was performed using standard hysterotomy (11 studies), mini-hysterotomy (one study) or fetoscopy by either exteriorized-uterus single-layer closure (one study), percutaneous single-layer closure (three studies) or percutaneous two-layer closure (one study). Only outcomes for standard hysterotomy could be meta-analyzed. Overall, outcomes improved significantly with experience. Competency was reached after 35 consecutive cases for standard hysterotomy and was predicted to be achieved after ≥ 57 cases for mini-hysterotomy and ≥ 56 for percutaneous two-layer fetoscopy. For percutaneous and exteriorized-uterus single-layer fetoscopy, competency was not reached in the 81 and 28 cases available for analysis, respectively, and LC prediction analysis could not be performed.
CONCLUSIONS
The number of cases operated is correlated with the outcome of fetal SBA closure, and the number of operated cases required to reach competency ranges from 35 for standard hysterotomy to ≥ 56-57 for minimally invasive modifications. Our observations provide important information for institutions looking to establish a new fetal center, develop a new fetal surgery technique or train their team, and inform referring clinicians, potential patients and third parties. Copyright © 2019 ISUOG. Published by John Wiley & Sons Ltd.
Topics: Adult; Female; Fetoscopy; Fetus; Humans; Hysterotomy; Learning Curve; Pregnancy; Spina Bifida Cystica
PubMed: 31273862
DOI: 10.1002/uog.20389