Impact of the volume of the myelomeningocele sac on imaging, prenatal neurosurgery and motor outcomes: a retrospective cohort study.
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ABSTRACT:
SUBMITTER: Corroenne R
PROVIDER: S-EPMC8222266 | biostudies-literature | 2021 Jun
REPOSITORIES: biostudies-literature
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Impact of the volume of the myelomeningocele sac on imaging, prenatal neurosurgery and motor outcomes: a retrospective cohort study.
Corroenne Romain R Mehollin-Ray Amy R AR Johnson Rebecca M RM Whitehead William E WE Espinoza Jimmy J Castillo Jonathan J Castillo Heidi H Orman Gunes G Donepudi Roopali R Huisman Thierry A G M TAGM Nassr Ahmed A AA Belfort Michael A MA Sanz Cortes Magdalena M Shamshirsaz Alireza A AA
Scientific reports 20210623 1
To investigate the association of the myelomeningocele (MMC) volume with prenatal and postnatal motor function (MF) in cases who underwent a prenatal repair. Retrospective cohort study (11/2011 to 03/2019) of 63 patients who underwent a prenatal MMC repair (37 fetoscopic, 26 open-hysterotomy). At referral, measurements of the volume of MMC was performed based on ultrasound scans. A large MMC was defined as greater than the optimal volume threshold (ROC analysis) for the prediction of intact MF a ...[more]
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Project description:Myelomeningocele, characterized by extrusion of the spinal cord through a spinal canal defect, is the most common form of spina bifida, often resulting in lifelong disability and significant orthopaedic issues. A randomized controlled trial (RCT) has shown the efficacy of prenatal repair in decreasing the need for shunting and improving motor outcomes. However, no studies have evaluated the effects of prenatal repair on orthopaedic outcomes. The purpose of this study was to determine the rates of orthopaedic conditions in patients with prenatal and postnatal repair of myelomeningocele and compare the rates of treatment required. This study analyzes the relevant outcomes from a prospective RCT (Management of Myelomeningocele Study). Eligible women were randomized to prenatal or postnatal repair, and patients were evaluated prospectively. Outcomes of interest included rates of scoliosis, kyphosis, hip abnormality, clubfoot, tibial torsion, and leg length discrepancy (LLD) at 12 and 30 months. The need for orthopaedic intervention at the same time points was also evaluated. Statistical analyses included descriptive statistics and univariate analyses. Data for the full cohort of 183 patients were analyzed (91 prenatal, 92 postnatal). There were no differences in rates of scoliosis, kyphosis, hip abnormality, clubfoot or tibial torsion between patients treated with prenatal or postnatal repair. The rate of LLD was lower in the prenatal repair group at 12 and 30 months (7 vs. 16% at 30 months, P = 0.047). The rates of patients requiring casting or bracing were significantly lower in patients treated with prenatal repair at 12 and 30 months (78 vs. 90% at 30 months, P = 0.036). Patients treated with prenatal myelomeningocele repair may develop milder forms of orthopaedic conditions and may not require extensive orthopaedic management.
| S-EPMC8099935 | biostudies-literature
Project description:The Management of Myelomeningocele Study (MOMS), a randomized trial of prenatal versus postnatal repair for myelomeningocele, found that prenatal surgery resulted in reduced hindbrain herniation and need for shunt diversion at 12 months of age and better motor function at 30 months. In this study, we compared adaptive behavior and other outcomes at school age (5.9-10.3 years) between prenatal versus postnatal surgery groups. Follow-up cohort study of 161 children enrolled in MOMS. Assessments included neuropsychological and physical evaluations. Children were evaluated at a MOMS center or at a home visit by trained blinded examiners. The Vineland composite score was not different between surgery groups (89.0 ± 9.6 in the prenatal group versus 87.5 ± 12.0 in the postnatal group; P = .35). Children in the prenatal group walked without orthotics or assistive devices more often (29% vs 11%; P = .06), had higher mean percentage scores on the Functional Rehabilitation Evaluation of Sensori-Neurologic Outcomes (92 ± 9 vs 85 ± 18; P < .001), lower rates of hindbrain herniation (60% vs 87%; P < .001), had fewer shunts placed for hydrocephalus (49% vs 85%; P < .001) and, among those with shunts, fewer shunt revisions (47% vs 70%; P = .02) than those in the postnatal group. Parents of children repaired prenatally reported higher mean quality of life z scores (0.15 ± 0.67 vs 0.11 ± 0.73; P = .008) and lower mean family impact scores (32.5 ± 7.8 vs 37.0 ± 8.9; P = .002). There was no significant difference between surgery groups in overall adaptive behavior. Long-term benefits of prenatal surgery included improved mobility and independent functioning and fewer surgeries for shunt placement and revision, with no strong evidence of improved cognitive functioning.
| S-EPMC6993457 | biostudies-literature
Project description:ObjectiveDescribe outcomes of open fetal surgery for myelomeningocele (MMC) repair in two Brazilian hospitals and the impact of surgical experience on outcome.DesignRetrospective cohort study.SettingSao Paulo, Brazil.Population237 pregnant women carrying a fetus with an open spinal defect.MethodsSurgical details, and maternal and fetal outcomes collected from all patients.Main outcome measuresAnalysis of surgical and perinatal outcome parameters.ResultsTotal surgical time was 119 ± 7.6 minutes. Preterm labour occurred in 24.2%, premature rupture of membranes in 26.7%, placental abruption in 0.8%, need for a blood transfusion at delivery in 2.1%, and dehiscence at the repair site in 2.5%. Reversal of hindbrain herniation at birth occurred in 71.4%. There were no maternal deaths or severe maternal morbidities. The failure rate with the patient anaesthetised was 0.42% and perinatal mortality was 2.1% (three intrauterine demises and two neonatal deaths). Comparing results from our study in the first 3 years with the last 3 years demonstrated improvement in the total surgical time (121.2 ± 6.4 versus 118.5 ± 8.2 minutes, P = 0.005) and an increase in reversal of hindbrain herniation at birth (64.0 versus 77.1%, P = 0.042).ConclusionOur open fetal surgical approach for MMC was effective and results were comparable to past studies. Improvements in surgical performance and perinatal outcome increased as the surgical team became more familiar with the procedure.FundingThe study was funded solely by institutional funds.Tweetable abstractBrazilian experience of in utero open surgery for myelomeningocele repair.
| S-EPMC6100098 | biostudies-literature
Project description:ObjectivesPrevious literature on the effects of marijuana exposure on neonatal outcomes has been limited by the reliance on maternal self-report. The objective of this study was to examine the relationship of prenatal marijuana exposure on neonatal outcomes in infants with marijuana exposure confirmed with meconium drug testing.DesignRetrospective cohort study.Setting and participantsMeconium drug screens obtained on infants born in a hospital system in the Pacific Northwest in the USA over a 2.5-year period. 1804 meconium drug screens were initially obtained, with 1540 drug screens included in the analysis.Primary and secondary outcome measuresNeonates with meconium drug screens positive for delta-9-tetrahydrocannabinol (THC) only were compared with neonates with negative drug screens. The following neonatal outcomes were examined: gestational age, preterm birth (<37 weeks), birth weight, low birth weight (defined as birth weight <2.5 kg), length, head circumference, Apgar scores and admission to the neonatal intensive care unit (NICU). Using multivariable logistical and linear regression, we controlled for confounding variables.Results1540 meconium drug screens were included in the analysis, with 483 positive for delta-9-THC only. Neonates exposed to delta-9-THC had significantly lower birth weight, head circumference and length (p<0.001). Neonates with THC exposure had 1.9 times the odds (95% CI 1.3 to 2.7, p=0.001) of being defined as low birth weight. Birth weight was on average 0.16 kg lower (95% CI 0.10 to 0.22, p<0.001) in those exposed to THC.ConclusionsPrenatal marijuana exposure was significantly associated with decreases in birth weight, length and head circumference, and an increased risk of being defined as low birth weight. These findings add to the previous literature demonstrating possible negative effects of prenatal marijuana use on neonatal outcomes.
| S-EPMC9528601 | biostudies-literature
Project description:BackgroundPrevious reports from the Management of Myelomeningocele Study demonstrated that prenatal repair of myelomeningocele reduces hindbrain herniation and the need for cerebrospinal fluid shunting, and improves motor function in children with myelomeningocele. The trial was stopped for efficacy after 183 patients were randomized, but 30-month outcomes were only available at the time of initial publication in 134 mother-child dyads. Data from the complete cohort for the 30-month outcomes are presented here. Maternal and 12-month neurodevelopmental outcomes for the full cohort were reported previously.ObjectiveThe purpose of this study is to report the 30-month outcomes for the full cohort of patients randomized to either prenatal or postnatal repair of myelomeningocele in the original Management of Myelomeningocele Study.Study designEligible women were randomly assigned to undergo standard postnatal repair or prenatal repair <26 weeks gestation. We evaluated a composite of mental development and motor function outcome at 30 months for all enrolled patients as well as independent ambulation and the Bayley Scales of Infant Development, Second Edition. We assessed whether there was a differential effect of prenatal surgery in subgroups defined by: fetal leg movements, ventricle size, presence of hindbrain herniation, gender, and location of the myelomeningocele lesion. Within the prenatal surgery group only, we evaluated these and other baseline parameters as predictors of 30-month motor and cognitive outcomes. We evaluated whether presence or absence of a shunt at 1 year was associated with 30-month motor outcomes.ResultsThe data for the full cohort of 183 patients corroborate the original findings of Management of Myelomeningocele Study, confirming that prenatal repair improves the primary outcome composite score of mental development and motor function (199.4 ± 80.5 vs 166.7 ± 76.7, P = .004). Prenatal surgery also resulted in improvement in the secondary outcomes of independent ambulation (44.8% vs 23.9%, P = .004), WeeFIM self-care score (20.8 vs 19.0, P = .006), functional level at least 2 better than anatomic level (26.4% vs 11.4%, P = .02), and mean Bayley Scales of Infant Development, Second Edition, psychomotor development index (17.3% vs 15.1%, P = .03), but does not affect cognitive development at 30 months. On subgroup analysis, there was a nominally significant interaction between gender and surgery, with boys demonstrating better improvement in functional level and psychomotor development index. For patients receiving prenatal surgery, the presence of in utero ankle, knee, and hip movement, absence of a sac over the lesion and a myelomeningocele lesion of ≤L3 were significantly associated with independent ambulation. Postnatal motor function showed no correlation with either prenatal ventricular size or postnatal shunt placement.ConclusionThe full cohort data of 30-month cognitive development and motor function outcomes validate in utero surgical repair as an effective treatment for fetuses with myelomeningocele. Current data suggest that outcomes related to the need for shunting should be counseled separately from the outcomes related to distal neurologic functioning.
| S-EPMC7737375 | biostudies-literature
Project description:ObjectiveTo compare associations between individual antidepressants and newborn outcomes.DesignRetrospective cohort study.SettingDeliveries in a large, US medical system.PopulationWomen who received at least one antidepressant prescription 3 months prior to conception through delivery.MethodsEligible women had maternal characteristics and newborn outcomes extracted from medical record data. Exposure was defined by the timing of the prescription during pregnancy.Main outcome measuresNewborn outcomes (any adaptation syndrome, neonatal intensive care unit (NICU) admission) were analyzed for each antidepressant and compared using standard statistics and multivariable regression compared to exposure to bupropion. Odds of outcomes based on timing of exposure were also explored.ResultsA total of 3,694 women were analyzed. Rates of any adaptation syndrome (p < 0.001), NICU admission (p < 0.001), and transient tachypnea of newborn (TTN) (p = 0.006) were significantly different between drugs. Infants exposed to duloxetine had the highest rates of NICU admissions (39.6%) and adaptation syndromes (15.1%). Venlafaxine-exposed infants had the highest rates of TTN (18.2%). Controlling for maternal age, race, insurance, and gestational age at delivery, early pregnancy antidepressant exposure was associated with adaptation syndrome and NICU admission for both duloxetine (adjusted odds ratio (aOR) 2.31 [95% Confidence Interval (CI) 1.11-4.80] and aOR 2.47 [95% CI 1.40-4.34], respectively) and escitalopram (aOR 1.72 [95% CI 1.09-2.70] and aOR 1.64 [95% CI 1.21-2.22], respectively). Exposure in the third trimester was associated with any adaptation syndrome for citalopram, duloxetine, escitalopram, fluoxetine, sertraline, and venlafaxine and NICU admission for bupropion, citalopram, duloxetine, escitalopram, and fluoxetine.ConclusionDuloxetine and escitalopram appear to have the strongest associations with any adaptation syndrome and NICU admission whereas bupropion and sertraline tended to have among the lowest risks of these outcomes. These results can help providers and patients discuss choice of individual antidepressant drugs during pregnancy.
| S-EPMC8608752 | biostudies-literature
Project description:BackgroundPrenatal repair of myelomeningocele, the most common form of spina bifida, may result in better neurologic function than repair deferred until after delivery. We compared outcomes of in utero repair with standard postnatal repair.MethodsWe randomly assigned eligible women to undergo either prenatal surgery before 26 weeks of gestation or standard postnatal repair. One primary outcome was a composite of fetal or neonatal death or the need for placement of a cerebrospinal fluid shunt by the age of 12 months. Another primary outcome at 30 months was a composite of mental development and motor function.ResultsThe trial was stopped for efficacy of prenatal surgery after the recruitment of 183 of a planned 200 patients. This report is based on results in 158 patients whose children were evaluated at 12 months. The first primary outcome occurred in 68% of the infants in the prenatal-surgery group and in 98% of those in the postnatal-surgery group (relative risk, 0.70; 97.7% confidence interval [CI], 0.58 to 0.84; P<0.001). Actual rates of shunt placement were 40% in the prenatal-surgery group and 82% in the postnatal-surgery group (relative risk, 0.48; 97.7% CI, 0.36 to 0.64; P<0.001). Prenatal surgery also resulted in improvement in the composite score for mental development and motor function at 30 months (P=0.007) and in improvement in several secondary outcomes, including hindbrain herniation by 12 months and ambulation by 30 months. However, prenatal surgery was associated with an increased risk of preterm delivery and uterine dehiscence at delivery.ConclusionsPrenatal surgery for myelomeningocele reduced the need for shunting and improved motor outcomes at 30 months but was associated with maternal and fetal risks. (Funded by the National Institutes of Health; ClinicalTrials.gov number, NCT00060606.).
| S-EPMC3770179 | biostudies-literature
Project description:IntroductionSeveral studies have reported increasing prevalence of prescription opioid use among pregnant women. However, little is known regarding the effects of maternal opioid use on neurodevelopmental disorders in early childhood in pregnant women with no evidence of opioid use disorders or drug dependence.ObjectiveThe aim of this study was to quantify the association between prenatal opioid exposure from maternal prescription use and neurodevelopmental outcomes in early childhood.MethodsThis retrospective study included pregnant women aged 12-55 years and their live-birth infants born from 2010 to 2012 present in Optum's deidentified Clinformatics® Data Mart database. Eligible infants born to mothers without opioid use disorders or drug dependence were followed till occurrence of neurodevelopmental disorders, loss to follow-up, or study end (December 31, 2017), whichever came first. Propensity score by fine stratification was applied to adjust for confounding by demographic characteristics, obstetric characteristics, maternal comorbid mental and pain conditions, and measures of burden of illnesses and to obtain adjusted hazard ratios (HR) and 95% confidence intervals (CI). Exposed and unexposed infants were compared on the incidence of neurodevelopmental disorders.ResultsOf 24,910 newborns, 7.6% (1899) were prenatally exposed to prescription opioids. Overall, 1562 children were diagnosed with neurodevelopmental disorders, with crude incidence rates of 2.9 per 100 person-years in exposed children versus 2.5 per 100 person-years in unexposed children. After adjustment, we observed no association between fetal opioid exposure and the risk of neurodevelopmental disorders (HR 1.10; 95% CI 0.92-1.32). However, increased risk of neurodevelopmental disorders were observed in children with longer cumulative exposure duration (HR 1.70; 95% CI 1.05-2.96) or high cumulative opioid doses (HR 1.22; 95% CI 1.01-1.54).Conclusion and relevanceIn pregnant women without opioid use disorders or drug dependence, maternal opioid use was not associated with increased risk of neurodevelopmental disorders in early childhood. However, increased risks of early neurodevelopmental disorders were observed in children born to women receiving prescription opioids for longer duration and at higher doses during pregnancy.
| S-EPMC8830246 | biostudies-literature
Project description:BackgroundThe severity of myelomeningocele (MMC) stems both from a loss of neurons due to neural tube defect and a loss of function in viable neurons due to reduced movement experience during the first year after birth. In young infants with MMC, the challenge is to reinforce excitability and voluntary control of all available neurons. Muscle vibration paired with voluntary movement may increase motoneuron excitability and contribute to improvements in neural organization, responsiveness, and control.ObjectivesThis study examined whether infants with or without MMC respond to vibration by altering their step or stance behavior when supported upright on a treadmill.DesignThis was a cross-sectional study.MethodsTwenty-four 2- to 10-month-old infants, 12 with typical development (TD) and 12 with MMC (lumbar and sacral lesions), were tested. Infants were supported upright with their feet in contact with a stationary or moving treadmill during 30-second trials. Rhythmic alternating vibrations were applied to the right and left rectus femoris muscles, the lateral gastrocnemius muscle, or the sole of the foot. Two cameras and behavior coding were used to determine step count, step type, and motor response to vibration onset.ResultsStep count decreased and swing duration increased in infants with TD during vibration of the sole of the foot on a moving treadmill (FT-M trials). Across all groups the percentage of single steps increased during vibration of the lateral gastrocnemius muscle on a moving treadmill. Infants with MMC and younger infants with TD responded to onset of vibration with leg straightening during rectus femoris muscle stimulation and by stepping during FT-M trials more often than older infants with TD.ConclusionsVibration seems a viable option for increasing motor responsiveness in infants with MMC. Follow-up studies are needed to identify optimal methods of administering vibration to maximize step and stance behavior in infants.
| S-EPMC3317481 | biostudies-literature
Project description:BackgroundMyelomeningocele (MMC) is a neural tube defect disease. Antenatal repair of fetal MMC is an alternative to postnatal repair. Many agents can be used as tocolytics during the in utero fetal repair such as β2-agonists and oxytocin receptor antagonists, with possible maternal and fetal repercussions. This study aims to compare maternal arterial blood gas analysis between terbutaline or atosiban, as tocolytic agents, during intrauterine MMC repair.MethodsRetrospective cohort study. Patients were divided into two groups depending on the main tocolytic agent used during intrauterine MMC repair: atosiban (16) or terbutaline (9). Maternal arterial blood gas samples were analyzed on three moments: post induction (baseline, before the start of tocolysis), before extubation, and two hours after the end of the surgery.ResultsTwenty-five patients were included and assessed. Before extubation, the terbutaline group showed lower arterial pH (7.347 ± 0.05 vs. 7.396 ± 0.02 for atosiban, p = 0.006) and higher arterial lactate (28.33 ± 12.76 mg.dL-1 vs. 13.06 ± 6.35 mg.dL-1, for atosiban, p = 0.001) levels.ConclusionsPatients who received terbutaline had more acidosis and higher levels of lactate, compared to those who received atosiban, during intrauterine fetal MMC repair.
| S-EPMC10992278 | biostudies-literature