Trends in incidence and long-term outcomes of myelomeningocele in British Columbia

North, Taylor; Cheong, Alexander; Steinbok, Paul; Radic, Julia

doi:10.1007/s00381-017-3685-6

Cited by 30 publications

(41 citation statements)

References 16 publications

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“…In this large population‐based study examining children born with spina bifida between years 1983 and 2006, the probability of survival was 93% up to age 7 days, 92% up to 1 month, 88% up to 1 year, 86% up to 5 years, and 82% up to 25 years . Time trends in spina bifida survival are also not well examined globally; two studies from the United States and Canada showed an improvement in survival among cohorts born in later years compared with those born during the late 1970s and the early 1980s …”

Section: Introductionmentioning

confidence: 99%

Analysis of Mortality among Neonates and Children with Spina Bifida: An International Registry‐Based Study, 2001‐2012

Bakker

Kancherla

Canfield

et al. 2019

Paediatric Perinatal Epid

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BackgroundMedical advancements have resulted in better survival and life expectancy among those with spina bifida, but a significantly increased risk of perinatal and postnatal mortality for individuals with spina bifida remains.ObjectivesTo examine stillbirth and infant and child mortality among those affected by spina bifida using data from multiple countries.MethodsWe conducted an observational study, using data from 24 population‐ and hospital‐based surveillance registries in 18 countries contributing as members of the International Clearinghouse for Birth Defects Surveillance and Research (ICBDSR). Cases of spina bifida that resulted in livebirths or stillbirths from 20 weeks' gestation or elective termination of pregnancy for fetal anomaly (ETOPFA) were included. Among liveborn spina bifida cases, we calculated mortality at different ages as number of deaths among liveborn cases divided by total number of liveborn cases with spina bifida. As a secondary outcome measure, we estimated the prevalence of spina bifida per 10 000 total births. The 95% confidence interval for the prevalence estimate was estimated using the Poisson approximation of binomial distribution.ResultsBetween years 2001 and 2012, the overall first‐week mortality proportion was 6.9% (95% CI 6.3, 7.7) and was lower in programmes operating in countries with policies that allowed ETOPFA compared with their counterparts (5.9% vs. 8.4%). The majority of first‐week mortality occurred on the first day of life. In programmes where information on long‐term mortality was available through linkage to administrative databases, survival at 5 years of age was 90%‐96% in Europe, and 86%‐96% in North America.ConclusionsOur multi‐country study showed a high proportion of stillbirth and infant and child deaths among those with spina bifida. Effective folic acid interventions could prevent many cases of spina bifida, thereby preventing associated childhood morbidity and mortality.

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Section: Introductionmentioning

confidence: 99%

Analysis of Mortality among Neonates and Children with Spina Bifida: An International Registry‐Based Study, 2001‐2012

Bakker

Kancherla

Canfield

et al. 2019

Paediatric Perinatal Epid

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“…11 Delaying the repair (3 or more days after birth) increases the risk of meningitis and other complications. 12 The proportion of cases repaired soon after birth has significantly improved in the past few decades, 4 and the technology and practice of fetal surgeries for myelomeningocele in selected cases are gaining prominence. 13 Timely myelomeningocele repair is not ubiquitous in the United States.…”

mentioning

confidence: 99%

“…16 Recently, a hospital-based study from British Columbia reported that 98% of myelomeningocele cases born between 1996 and 2006 had a repair surgery within 48 hours after birth. 4 The objective of our study was to examine the prevalence of timely surgical repair (0-2 days after birth) and associated clinical, demographic, and delivery hospital characteristics among infants with myelomeningocele born in California from 2006 to 2011. We used population-based data from the California Perinatal Quality Care Collaborative (CPQCC) to identify infants with myelomeningocele, in conjunction with additional information from hospital discharge and vital records data.…”

mentioning

confidence: 99%

Factors Associated with Timeliness of Surgical Repair among Infants with Myelomeningocele: California Perinatal Quality Care Collaborative, 2006 to 2011

Kancherla

Grant

et al. 2019

Am J Perinatol

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Objective To examine factors associated with timely (0–2 days after birth) myelomeningocele surgical repair. Study Design We examined 2006 to 2011 births from the California Perinatal Quality Care Collaborative, linking to hospital discharge and vital records. Selected maternal, infant, and delivery hospital characteristics were evaluated to understand disparities in timely repair. Poisson regression was used to estimate adjusted risk ratios (aRRs) and 95% confidence intervals (CIs). Results Overall, 399 of the 450 (89%) infants had a timely repair and approximately 80% of them were delivered in level III/IV hospitals. Infants with hydrocephalus were significantly less likely to have a delayed myelomeningocele repair compared with those without (aRR = 0.22; 95% CI = 0.13, 0.39); infants whose medical care was paid by Medi-Cal or other nonprivate insurance were 2.2 times more likely to have a delayed repair compared with those covered by a private insurance (aRR = 2.23; 95% CI = 1.17, 4.27). Low birth weight was a significant predictor for delayed repair (aRR = 2.06; 95% CI = 1.10, 3.83). Conclusion There was a significant disparity in myelomenigocele repair based on medical care payer. Families and hospitals should work together for timely repair in hospitals having specialized multidisciplinary teams. Findings from the study can be used to follow best clinical practices for myelomeningocele repair.

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“…Clinically, these present as long-term disabilities affecting ambulatory status, bowel and bladder continence and sexual function, as well as decreased cognitive function in ~ 15% of cases mainly due to complications of cerebrospinal fluid shunting. (3,6) Typically, the lesion is closed surgical shortly after birth (postnatal repair), but prenatal repair is believed to prevent damage to the spinal cord and improve neonatal and long-term functional outcomes. (7) (8)(9)(10)(11) In 2011, The Management of Myelomeningocele Study (MOMS), a randomised controlled trial comparing prenatal to postnatal repair indicated a decrease in the need for ventriculoperitoneal shunting, reversal of hindbrain herniation, and better neurologic function.…”

Section: Introductionmentioning

confidence: 99%

Development of a Core Outcome Set for Fetal MyelomenIngoCele (COSMiC): study protocol

Altoukhi

Whitehead

Ryan

et al. 2020

Preprint

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Background: Open spina bifida (OSB) is one of the most common congenital central nervous system defects and leads to long-term physical and cognitive disabilities. Open fetal surgery for OSB improves neurological outcomes and reduces the need for ventriculoperitoneal shunting, compared to postnatal surgery, at the expense of increased prematurity. It however confers significant morbidity to the mother in the index pregnancy and potential risks to future pregnancies. Fetoscopic surgery may prevent maternal morbidity yet the question remains whether the procedure is neuroprotective and reduces prematurity. Comparison of outcomes between different treatment options is challenging due to inconsistent outcome reporting. We aim to develop and disseminate a core outcome set (COS) for fetal OSB, to ensure that outcomes relevant to all stakeholders are collected and reported in a standardised fashion in future studies. Methods: The COS will be developed using a validated Delphi methodology. A systematic literature review will be performed to identify previously reported outcomes focused on prenatally diagnosed OSB. Outcomes assessed will include maternal (primary and subsequent pregnancies), fetal, neonatal and childhood to adolescence. In a second phase, semi-structured interviews with stakeholders will be performed to ensure representation of additional relevant outcomes that may not have been reported in the literature. We will include patients and parents, as well as health professionals involved in the care of these pregnancies and children (fetal medicine specialists, fetal surgeons, neonatologists/paediatricians, and allied health). Subsequently, an international group of key stakeholders will rate the importance of the identified outcomes using three sequential, online, rounds of a modified Delphi Survey. Final agreement on outcomes to be included in the COS, their definition and measurement will be achieved through a final face-to-face consensus meeting with all stakeholder groups represented. Dissemination of the final COS will be ensured through different media and relevant societies. Discussion: Development and implementation of a COS for fetal OSB will ensure consistent outcome reporting in future clinical trials, systematic reviews, and clinical practice guidelines. This will lead to higher quality research, better evidence-based clinical practice and ultimately improved maternal, fetal and long term childhood outcomes. Trial registration: Core Outcome Measures in Effectiveness Trials (COMET): 1187. International Prospective Register of Systematic Reviews (PROSPERO): CRD42018104880.

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Trends in incidence and long-term outcomes of myelomeningocele in British Columbia

Cited by 30 publications

References 16 publications

Analysis of Mortality among Neonates and Children with Spina Bifida: An International Registry‐Based Study, 2001‐2012

Analysis of Mortality among Neonates and Children with Spina Bifida: An International Registry‐Based Study, 2001‐2012

Factors Associated with Timeliness of Surgical Repair among Infants with Myelomeningocele: California Perinatal Quality Care Collaborative, 2006 to 2011

Development of a Core Outcome Set for Fetal MyelomenIngoCele (COSMiC): study protocol

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