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1.
Fetal Diagn Ther ; 2024 Feb 06.
Article in English | MEDLINE | ID: mdl-38320542

ABSTRACT

INTRODUCTION: A critical component of an evidence-based reassessment of in-utero intervention for fetal aqueductal stenosis (fetal AS) is determining if the prenatal diagnosis can be accurately made at a gestational age amenable to in-utero intervention. METHODS: A multicenter, prospective, observational study was conducted through the North American Fetal Therapy Network (NAFTNet). Pregnancies complicated by severe CNS ventriculomegaly (lateral ventricle diameter >15 mm) not secondary to a primary diagnosis (myelomeningocele, encephalocele, etc.) were recruited at diagnosis. Imaging and laboratory findings were recorded in an online REDCap database. After evaluation, investigators were asked to render their degree of confidence in the diagnosis of fetal AS. The prenatal diagnosis was compared to the postnatal diagnosis obtained through neonatal neuroimaging. Performance characteristics of ultrasound and MRI were calculated, as was the mean gestational age at diagnosis. RESULTS: Between April 2015 and October 2022, eleven NAFTNet centers contributed 64 subjects with severe fetal CNS ventriculomegaly. Of these, 56 had both prenatal and postnatal diagnoses recorded. Ultrasound revealed 32 fetal AS true positives, 4 false positives, 7 false negatives, and 13 true negatives rendering a sensitivity of 0.82, a specificity of 0.76, a positive predictive value of 0.89, and a negative predictive value of 0.65. The mean gestational age at diagnosis by ultrasound was 25.5 weeks (std +/- 4.7w). The proportion of agreement (true positive + true negative/n) was highest at 24 weeks gestation. For fetal MRI (n=35), the sensitivity for fetal AS was 0.95, specificity was 0.69, positive predictive value was 0.84, and negative predictive value was 0.90. MRI was performed at 25 weeks on average. CONCLUSION: The prenatal diagnosis of fetal aqueductal stenosis can be made with accuracy at a gestational age potentially amenable to in-utero intervention. Only 7% of subjects were incorrectly diagnosed prenatally with fetal AS by ultrasound and 11% by MRI. Diagnostic accuracy of fetal AS will likely improve with increased experience.

2.
Fetal Diagn Ther ; 2024 Feb 07.
Article in English | MEDLINE | ID: mdl-38325342

ABSTRACT

INTRODUCTION: Fetal care centers (FCCs) in the U.S. lack a standardized instrument to measure person-centered care. This study aimed to develop and validate the Person-Centered Care in Fetal Care Centers (PCC-FCC) Scale. METHODS: Initial items were developed based on literature and input from clinicians and former patients. A Delphi study involving 16 experts was conducted to validate the content and construct. Through three rounds of online questionnaires using open-ended questions and Likert scales, consensus on item clarity and relevancy was established. The resulting items were then piloted with former fetal care center patients via a web-based survey. The instrument's reliability and validity were validated using Cronbach's α and exploratory factor analysis, respectively. Concurrent validity was assessed by comparing scores with the Revised Patient Perception of Patient-Centeredness (PPPC-R) Questionnaire. RESULTS: 258 participants completed the 48-item pilot PCC-FCC survey, categorized into six domains. Factor analysis yielded a 2-factor, 28-item scale. Internal consistency of the final scale had good reliability (α=0.969). Data supported content, construct, and concurrent validity. CONCLUSION: The PCC-FCC Scale is a reliable and valid measure of person-centered care in U.S. FCCs. It can be used to enhance services and begin connecting person-centered care to maternal-child health outcomes.

3.
Transfusion ; 64 Suppl 2: S93-S99, 2024 May.
Article in English | MEDLINE | ID: mdl-38404198

ABSTRACT

BACKGROUND: Using low titer group O whole blood (LTOWB) is increasingly popular for resuscitating trauma patients. LTOWB is often RhD-positive, which might cause D-alloimmunization and hemolytic disease of the fetus and newborn (HDFN) if transfused to RhD-negative females of childbearing potential (FCP). This simulation determined the number of life years gained by the FCP and her future children if she was resuscitated with LTOWB compared with conventional component therapy (CCT). METHODS: The model simulated 500,000 injured FCPs of each age between 0 and 49 years with LTOWB mortality relative reductions (MRRs) compared with components between 0.1% and 25%. For each surviving FCP, number of life years gained was calculated using her age at injury and average life expectancy for American women. The number of expected future pregnancies for FCPs that did not survive was also based on her age at injury; each future child was assigned the maximum lifespan unless they suffered perinatal mortality or serious neurological events from HDFN. RESULTS: The LTOWB group with an MRR 25% compared with CCT had the largest total life years gained. The point of equivalence for RhD-positive LTOWB compared to CCT, where life years lost due to severe HDFN was equivalent to life years gained due to FCP survival/future childbearing, occurred at an MRR of approximately 0.1%. CONCLUSION: In this model, RhD-positive LTOWB resulted in substantial gains in maternal and child life years compared with CCT. A >0.1% relative mortality reduction from LTOWB offset the life years lost to HDFN mortality and severe neurological events.


Subject(s)
ABO Blood-Group System , Computer Simulation , Wounds and Injuries , Humans , Female , Infant , Adult , Child , Infant, Newborn , Child, Preschool , Adolescent , Pregnancy , Wounds and Injuries/mortality , Wounds and Injuries/therapy , Middle Aged , Young Adult , Blood Transfusion/methods , Life Expectancy , Male , Rh-Hr Blood-Group System
4.
Trauma Surg Acute Care Open ; 9(Suppl 1): e001252, 2024.
Article in English | MEDLINE | ID: mdl-38196928

ABSTRACT

Background: After the transfusion of RhD-positive red blood cell (RBC)-containing products to an RhD-negative woman of childbearing potential (WCP) during trauma resuscitation, there are several events that must occur for that WCP to have a future pregnancy affected by hemolytic disease of the fetus and newborn (HDFN). This study identified and quantitated the frequency of a novel event in the sequence from RhD-positive transfusion during trauma resuscitation to an HDFN outcome, that is, the development of a high titer anti-D among women who were D-alloimmunized. Methods: The transfusion service records at one maternity hospital were searched to locate all anti-D titers that had been performed on pregnant women between 1996 and 2022. The highest titer score during each pregnancy was recorded for this study. The critical titer threshold at this institution was ≥16. Passive anti-D caused by Rh immunoglobulin were excluded from analysis. Results: There were 97 pregnancies in 85 patients who had an immune-stimulated anti-D; in 60 of 97 (62%) pregnancies, the highest titer score was ≥16. There were 12 patients who had titers performed in two pregnancies during the study period; the correlation between the maximum titer in each pregnancy was not statistically significant (Spearman rank correlation r=0.42, p=0.17). Conclusion: In this single center study, 62% of D-alloimmunized pregnant women had a high titer antibody. When considering all of the events that must occur for HDFN to happen, the rate of perinatal mortality was calculated to be 0.04% and the rate of perinatal death or serious adverse event from HDFN was 0.24%.

5.
Prenat Diagn ; 43(9): 1229-1238, 2023 08.
Article in English | MEDLINE | ID: mdl-37539833

ABSTRACT

To investigate the outcomes of asymptomatic stage I twin-to-twin transfusion syndrome (stage I TTTS) among patients treated with fetoscopic laser photocoagulation (FLP) versus expectant management. Databases such as PubMed, Web of Science and Scopus were systematically searched from inception up to March 1st, 2022. The primary outcome was at least one fetal survival at birth and secondary outcomes included gestational age at delivery, preterm premature rupture of membranes < 32 weeks, preterm birth < 32 weeks, and single and dual fetal survival. Five studies were included in the meta-analysis. There was no significant difference in terms of at least one survival (odds ratio (OR) = 1.40, 95%CI= (0.26, 7.43), P = 0.70), single survival (OR = 0.87, 95%CI= (0.51, 1.48), P = 0.60) and dual survival (OR = 1.63, 95%CI= (0.74, 3.62), P = 0.23) among FLP and expectant groups. Gestational age at delivery (mean difference = 1.19, 95%CI= (-0.25, 2.63), P = 0.10), the risk of PTB<32 weeks (OR = 0.88, 95%CI= (0.50, 1.54), P = 0.65), and pPROM<32 weeks (OR = 1.80, 95% CI= (0.41, 7.98), P = 0.44) were also comparable between the groups. Routine FLP of the placental anastomoses before 26 weeks of gestation is unlikely to be beneficial among asymptomatic stable stage I TTTS patients without cervical shortening as the procedure does not offer a survival advantage compared with expectant management.


Subject(s)
Fetofetal Transfusion , Premature Birth , Pregnancy , Humans , Infant, Newborn , Female , Placenta , Watchful Waiting , Laser Coagulation/methods , Premature Birth/etiology , Fetoscopy/methods , Gestational Age , Retrospective Studies , Lasers , Pregnancy, Twin
7.
J Biomater Appl ; 37(8): 1423-1435, 2023 03.
Article in English | MEDLINE | ID: mdl-36063383

ABSTRACT

Fetal aqueductal stenosis (AS) is one of the most common causes of congenital hydrocephalus, which increases intracranial pressure due to partial or complete obstruction of cerebrospinal fluid (CSF) flow within the ventricular system. Approximately 2-4 infants per 10,000 births develop AS, which leads to progressive hydrocephalus, which enlarges the head often necessitating delivery by cesarean section. Most babies born with AS are severely neurologically impaired and experience a lifetime of disability. Therefore, a new device technology for venticuloamniotic shunting is urgently needed and has been studied to ameliorate or prevent fetal hydrocephalus development, which can provide a significant impact on patients and their family's quality of life and on the decrease of the healthcare dollars spent for the treatment. This study has successfully validated the design of shunt devices and demonstrated the mechanical performance and valve functions. A functional prototype shunt has been fabricated and subsequently used in multiple in vitro tests to demonstrate the performance of this newly developed ventriculoamniotic shunt. The shunt contains a main silicone-nitinol composite tube, a superelastic 90° angled dual dumbbell anchor, and an ePTFE valve encased by a stainless-steel cage. The anchor will change its diameter from 1.15 mm (collapsed state) to 2.75 mm (deployed state) showing up to 1.4-fold diameter change in human body temperature. Flow rates in shunts were quantified to demonstrate the valve function in low flow rates mimicking the fetal hydrocephalus condition showing "no backflow" for the valved shunt while there is up to 15 mL/h flow through the shunt with pressure difference of 20 Pa. In vivo ovine study results show the initial successful device delivery and flow drainage with sheep model.


Subject(s)
Cesarean Section , Hydrocephalus , Humans , Animals , Sheep , Pregnancy , Female , Quality of Life , Cerebrospinal Fluid Shunts/methods , Hydrocephalus/surgery
8.
Obstet Gynecol ; 139(6): 1027-1042, 2022 06 01.
Article in English | MEDLINE | ID: mdl-35675600

ABSTRACT

Fetal therapies undertaken to improve fetal outcome or to optimize transition to neonate life often entail some level of maternal, fetal, or neonatal risk. A fetal therapy center needs access to resources to carry out such therapies and to manage maternal, fetal, and neonatal complications that might arise, either related to the therapy per se or as part of the underlying fetal or maternal condition. Accordingly, a fetal therapy center requires a dedicated operational infrastructure and necessary resources to allow for appropriate oversight and monitoring of clinical performance and to facilitate multidisciplinary collaboration between the relevant specialties. Three care levels for fetal therapy centers are proposed to match the anticipated care complexity, with appropriate resources to achieve an optimal outcome at an institutional and regional level. A level I fetal therapy center should be capable of offering fetal interventions that may be associated with obstetric risks of preterm birth or membrane rupture but that would be very unlikely to require maternal medical subspecialty or intensive care, with neonatal risks not exceeding those of moderate prematurity. A level II center should have the incremental capacity to provide maternal intensive care and to manage extreme neonatal prematurity. A level III therapy center should offer the full range of fetal interventions (including open fetal surgery) and be able manage any of the associated maternal complications and comorbidities, as well as have access to neonatal and pediatric surgical intervention including indicated surgery for neonates with congenital anomalies.


Subject(s)
Fetal Membranes, Premature Rupture , Fetal Therapies , Premature Birth , Child , Female , Humans , Infant, Newborn , Infant, Premature , Pregnancy , Prenatal Care
9.
Fetal Diagn Ther ; 49(3): 117-124, 2022.
Article in English | MEDLINE | ID: mdl-34915495

ABSTRACT

INTRODUCTION: Uterine incision based on the placental location in open maternal-fetal surgery (OMFS) has never been evaluated in regard to maternal or fetal outcomes. OBJECTIVE: The aim of this study was to investigate whether an anterior placenta was associated with increased rates of intraoperative, perioperative, antepartum, obstetric, or neonatal complications in mothers and babies who underwent OMFS for fetal myelomeningocele (fMMC) closure. METHODS: Data from the international multicenter prospective registry of patients who underwent OMFS for fMMC closure (fMMC Consortium Registry, December 15, 2010-June 31, 2019) was used to compare fetal and maternal outcomes between anterior and posterior placental locations. RESULTS: The placental location for 623 patients was evenly distributed between anterior (51%) and posterior (49%) locations. Intraoperative fetal bradycardia (8.3% vs. 3.0%, p = 0.005) and performance of fetal resuscitation (3.6% vs. 1.0%, p = 0.034) occurred more frequently in cases with an anterior placenta when compared to those with a posterior placenta. Obstetric outcomes including membrane separation, placental abruption, and spontaneous rupture of membranes were not different among the 2 groups. However, thinning of the hysterotomy site (27.7% vs. 17.7%, p = 0.008) occurred more frequently in cases of an anterior placenta. Gestational age (GA) at delivery (p = 0.583) and length of stay in the neonatal intensive care unit (p = 0.655) were similar between the 2 groups. Fetal incision dehiscence and wound revision were not significantly different between groups. Critical clinical outcomes including fetal demise, perinatal death, and neonatal death were all infrequent occurrences and not associated with the placental location. CONCLUSIONS: An anterior placental location is associated with increased risk of intraoperative fetal resuscitation and increased thinning at the hysterotomy closure site. Individual institutional experiences may have varied, but the aggregate data from the fMMC Consortium did not show a significant impact on the GA at delivery or maternal or fetal clinical outcomes.


Subject(s)
Fetal Therapies , Meningomyelocele , Female , Fetal Therapies/adverse effects , Gestational Age , Humans , Hysterotomy/adverse effects , Infant, Newborn , Meningomyelocele/etiology , Meningomyelocele/surgery , Placenta/surgery , Pregnancy
10.
J Neurosurg Pediatr ; 28(3): 320-325, 2021 Jun 25.
Article in English | MEDLINE | ID: mdl-34171841

ABSTRACT

OBJECTIVE: Congenital aqueductal stenosis (CAS) is a common etiology of hydrocephalus that occurs in a subset of infants and may be linked to an increased incidence of ophthalmological abnormalities and delayed developmental milestones. Although hydrocephalus is common and widely studied, sparse literature exists on patients with isolated (no identifiable genetic link) CAS along with analysis of ophthalmological manifestations. In this study, the authors sought to describe the ophthalmological abnormalities and delayed developmental milestones of patients with isolated CAS. METHODS: Data of patients with CAS were prospectively entered and monitored in a surgical database maintained by the Department of Neurological Surgery at Children's Hospital of Pittsburgh from January 2005 to October 2016. Patients with a family history of congenital hydrocephalus, positive testing for genetic forms of aqueductal stenosis, other congenital abnormalities suggesting an underlying genetic syndrome, and stenosis/obstruction due to secondary causes were excluded from this study. Prenatal and perinatal history, CSF diversion history, and a variety of outcomes, including ophthalmological deficits and developmental milestones, were collected and analyzed. RESULTS: A total of 41 patients with isolated CAS were identified, with a mean follow-up duration of 6 years. Among that cohort, 26 patients (63.4%) developed neuroophthalmological complications, which were further stratified. Fourteen patients (34.1%) developed strabismus and 11 (26.8%) developed astigmatism, and 1 patient (2.4%) with papilledema was recorded. Among patients with ophthalmological abnormalities, 76.9% had delayed developmental milestones (p = 0.045). CONCLUSIONS: Patients with CAS were found to have increased risk of ophthalmological abnormalities requiring correction, along with an increased risk of delayed developmental milestones. Importantly, there was a significant correlation between the development of ophthalmological abnormalities and delayed developmental milestones that was independent of CSF diversion history. Larger patient cohort studies are required to explore whether earlier development of hydrocephalus, as is the case in CAS, causes elevated rates of neurological and ophthalmological complications, and if earlier CSF diversion correlates with improved outcomes.

11.
Vox Sang ; 116(7): 831-840, 2021 Aug.
Article in English | MEDLINE | ID: mdl-33491789

ABSTRACT

BACKGROUND: A risk assessment model for predicting the risk of haemolytic disease of the fetus and newborn (HDFN) in future pregnancies following the transfusion of Rh(D)-positive red blood cell (RBC)-containing products to females of childbearing potential (FCP) was developed, accounting for the age that the FCP is transfused in various countries. METHODS: The HDFN risk prediction model included the following inputs: risk of FCP death in trauma, Rh(D) alloimmunization rate following Rh(D)-positive RBC transfusion, expected number of live births following resuscitation, probability of carrying an Rh(D)-positive fetus, the probability of HDFN in an Rh(D)-positive fetus carried by an alloimmunized mother. The model was implemented in Microsoft R Open, and one million FCPs of each age between 18 and 49 years old were simulated. Published data from eight countries, including the United States, were utilized to generate country-specific HDFN risk estimates. RESULTS: The risk predictions showed similar characteristics for each country in that the overall risk of having a pregnancy affected by HDFN was higher if the FCP was younger when she received her Rh(D)-positive transfusion than if she was older. In the United States, the overall risk of HDFN if the FCP was transfused at age 18 was 3·4% (mild: 1·20%, moderate: 0·45%; severe: 1·15%; IUFD: 0·57%); the risk was approximately 0% if the FCP was 43 years or older at the time of transfusion. CONCLUSION: This model can be used to predict HDFN outcomes when establishing transfusion policies as it relates to the administration of Rh(D)-positive products for massively bleeding FCPs.


Subject(s)
Erythroblastosis, Fetal , Rh-Hr Blood-Group System , Blood Transfusion , Erythrocytes , Female , Humans , Isoantibodies , Pregnancy
12.
Prenat Diagn ; 40(5): 585-589, 2020 04.
Article in English | MEDLINE | ID: mdl-31994748

ABSTRACT

OBJECTIVE: Thromboelastography (TEG) is a point-of-care device used to evaluate whole blood coagulation function. The TEG is unique as a test of coagulation function in that it measures the interaction of all components of clot formation, which is different than traditional laboratory-based tests that measure isolated components of coagulation. Little is known about fetal coagulation physiology. We sought to evaluate the impact of severe fetal anemia and intrauterine transfusion (IUT) on fetal coagulation physiology by use of the TEG and to compare fetal TEG values to those of healthy neonates and adults. METHOD: One milliliter of fetal blood was collected immediately before (pre) and after (post) IUT of packed red blood cells (PRBCs). Sampling and transfusion were performed for fetal anemia due to hemolytic disease of the fetus and newborn. Samples were run in duplicate. For descriptive summary, duplicate pre-IUT and post-IUT values were averaged. Values for R (initiation of clot in minutes), K (clot firmness in minutes), angle (kinetics of clot development in degrees), and MA (maximum strength in mm) were obtained for each sample and presented using mean ± SE. Pre-IUT values for R, K, angle, and MA were compared with post-IUT values using linear mixed-effect model to account for clustering due to repeated observation from the same fetus. Pre-IUT values are compared with normal healthy term neonates and healthy adults using Wald test. The study was approved by the University of Pittsburgh Institutional Review Board (PRO14050051). RESULTS: Four fetuses underwent nine IUTs rendering 17 pre-IUT and 17 post-IUT specimens. The mean gestational age at IUT was 31 weeks 2 days (25 weeks 4 days to 35 weeks 2 days). The mean IUT volume transfused was 69 mL (30-170 mL). The mean estimated percent intravascular volume transfused was 33.4% (19%-52%). Of the four variables analyzed, only R showed a significant difference, with the initiation of clot formation being modestly delayed after transfusion by an estimated 2.87 minutes (95% CI, 0.82-4.92, P = .0480). Pre-IUT values were compared with 100 normal term neonates and 118 healthy adults. Compared with pre-IUT, only R was affected (shorter) compared with term neonates (mean ± SE = 5.46 ± 0.16 minute, P < .001) and healthy adults (mean ± SE = 6.8 ± 0.13 minute, P < .001). CONCLUSION: OTHER THAN A MODEST PROLONGATION OF CLOT FORMATION TIME, IUT OF PRBCS OF UP TO 52% OF THE ESTIMATED INTRAVASCULAR VOLUME DID NOT AFFECT FETAL COAGULATION FUNCTION BY TEG. OTHER THAN A SHORTER CLOT FORMATION TIME (R), THERE IS NO DIFFERENCE IN TEG VALUES BETWEEN ANEMIC PRETERM FETUSES AND HEALTHY TERM NEONATES AND HEALTHY ADULTS. BEYOND GENERALIZABLE KNOWLEDGE, THIS INFORMATION COULD BE EXPLOITED FOR FUTURE FETAL INTERVENTION TECHNIQUES.


Subject(s)
Blood Coagulation/physiology , Blood Transfusion, Intrauterine , Erythroblastosis, Fetal/blood , Erythroblastosis, Fetal/therapy , Erythrocyte Transfusion , Fetal Blood/physiology , Thrombelastography , Case-Control Studies , Female , Gestational Age , Humans , Pregnancy
13.
Fetal Diagn Ther ; 47(1): 7-14, 2020.
Article in English | MEDLINE | ID: mdl-30974442

ABSTRACT

INTRODUCTION: Documentation of histologic findings associated with congenital hydrocephalus in the fetal lamb model is a critical step in evaluating the efficacy of ventriculoamniotic shunting in the human fetus. METHODS: Four fetal sheep had hydrocephalus induced at approximately 95 days' gestation. Two co-twins remained as controls. The ewes were euthanized at term. The lamb brains were fixed in formalin, paraffin-embedded, stained, and analyzed for markers of neuropathology. Astrocytosis, microgliosis, and axonal loss were assessed with immunocytochemistry for glial fibrillary acidic protein, ionized calcium-binding adapter, and neurofilament/amyloid precursor protein, respectively. Cortical gray matter extracellular matrix was assessed with staining for the lectin Wisteria Floribunda agglutinin. RESULTS: Hydrocephalic lamb brains demonstrated deep white matter damage with loss of projecting axonal tracts in regions physically distorted by hydrocephalus, similar to that seen in hydrocephalic humans. There was no evidence of abnormal neocortical neuronal migration; however, there was evidence for delayed maturation of the neocortical gray matter, possibly from increased intracerebral pressure and subsequent ischemia. Control lamb brains demonstrated none of the above findings. CONCLUSION: This histological approach can be used to further define the mechanism of brain damage associated with hydrocephalus and interpret the efficacy of ventriculoamniotic shunting on fetal lamb brain neuroanatomy.


Subject(s)
Brain/pathology , Hydrocephalus/congenital , Animals , Animals, Newborn , Disease Models, Animal , Female , Hydrocephalus/pathology , Pregnancy , Sheep
14.
Prenat Diagn ; 40(1): 58-65, 2020 01.
Article in English | MEDLINE | ID: mdl-31306500

ABSTRACT

Fetal severe central nervous system ventriculomegaly is associated with poor neurologic outcomes, usually driven by a primary malformation, deformation, or disruption of brain parenchyma. In utero shunting of excess cerebrospinal fluid (CSF) in hopes of improving neurologic outcomes was attempted in the 1980s but was abandoned due to perceived lack of effect, likely due to technological limitations of the time that precluded proper patient selection. Little progress on the antenatal management of severe ventriculomegaly has been made in the intervening decades. A multidisciplinary, evidence-based reassessment of ventriculoamniotic shunting for isolated fetal aqueductal stenosis (FAS), a unique form of severe ventriculomegaly (supratentorial intracranial hypertension), is currently underway. An accurate diagnosis of FAS must precede in utero intervention. Magnetic resonance imaging (MRI) will be an excellent adjunct to high-resolution prenatal ultrasound and next-generation genetic testing to correctly diagnose FAS in a timely fashion while excluding other intracranial and extracranial anomalies. This manuscript will briefly discuss the history, current management, and future directions of the prenatal diagnosis and potential intervention for FAS.


Subject(s)
Cerebrospinal Fluid Shunts/methods , Fetal Diseases/diagnostic imaging , Fetal Therapies/methods , Hydrocephalus/diagnostic imaging , Female , Fetal Diseases/surgery , Humans , Hydrocephalus/surgery , Magnetic Resonance Imaging , Pregnancy , Prenatal Diagnosis , Ultrasonography, Prenatal
16.
Am J Obstet Gynecol ; 220(5): 494.e1-494.e7, 2019 05.
Article in English | MEDLINE | ID: mdl-30885769

ABSTRACT

BACKGROUND: Open maternal-fetal surgery for fetal myelomeningocele results in reduction in neonatal morbidity related to spina bifida but may be associated with fetal, neonatal, and maternal complications in subsequent pregnancies. OBJECTIVE: The objective of this study was to ascertain obstetric risk in subsequent pregnancies after open maternal-fetal surgery for fetal myelomeningocele closure. STUDY DESIGN: An international multicenter prospective observational registry created to track and report maternal, obstetric, fetal/neonatal, and subsequent pregnancy outcomes following open maternal-fetal surgery for fetal myelomeningocele was evaluated for subsequent pregnancy outcome variables. Institutional Review Board approval was obtained for the registry. RESULTS: From 693 cases of open maternal-fetal surgery for fetal myelomeningocele closure entered into the registry, 77 subsequent pregnancies in 60 women were identified. The overall live birth rate was 96.2%, with 52 pregnancies delivering beyond 20 weeks gestational age and median gestational age at delivery of 37 (36.3-37.1) weeks. The uterine rupture rate was 9.6% (n = 5), resulting in 2 fetal deaths. Maternal transfusion was required in 4 patients (7.7%). CONCLUSION: The risk of uterine rupture or dehiscence in subsequent pregnancies with associated fetal morbidity after open maternal-fetal surgery is significant, but is similar to that reported for subsequent pregnancies after classical cesarean deliveries. Future pregnancy considerations should be included in initial counseling for women contemplating open maternal-fetal surgery.


Subject(s)
Fetus/surgery , Meningomyelocele/surgery , Pregnancy Outcome , Abortion, Spontaneous/epidemiology , Adult , Blood Transfusion/statistics & numerical data , Cesarean Section , Female , Fetal Death , Gestational Age , Humans , Live Birth , Pregnancy , Prospective Studies , Registries , Uterine Rupture/epidemiology
17.
Am J Obstet Gynecol MFM ; 1(1): 74-81, 2019 03.
Article in English | MEDLINE | ID: mdl-32832884

ABSTRACT

Background: Despite improvements in fetal survival for pregnancies affected by twin-twin transfusion syndrome since the introduction of laser photocoagulation, prematurity remains a major source of neonatal morbidity and mortality. Objective: To investigate the indications and factors influencing the timing of delivery following laser treatment, we collected delivery information regarding twin-twin transfusion syndrome cases in a large multicenter cohort. Study Design: Eleven North American Fetal Therapy Network (NAFTNet) centers conducted a retrospective review of twin-twin transfusion syndrome patients who underwent laser photocoagulation. Clinical, demographic and ultrasound variables including twin-twin transfusion syndrome stage, and gestational age at treatment and delivery were recorded. Primary and secondary maternal and fetal indications for delivery were identified. Univariate analysis was used to select candidate variables with significant correlation with latency and GA at delivery. Multivariable Cox regression with competing risk analysis was utilized to determine the independent associations. Results: A total of 847 pregnancies were analyzed. After laser, the average latency to delivery was 10.11 ± 4.8 weeks and the mean gestational age at delivery was 30.7 ± 4.5 weeks. Primary maternal indications for delivery comprised 79% of cases. The leading indications included spontaneous labor (46.8%), premature rupture of membranes (17.1%), and placental abruption (8.4%). Primary fetal indications accounted for 21% of cases and the most frequent indications included donor non-reassuring status (20.5%), abnormal donor Dopplers (15.1%), and donor growth restriction (14.5%). The most common secondary indications for delivery were premature rupture of membranes, spontaneous labor and donor growth restriction. Multivariate modeling found gestational age at diagnosis, stage, history of prior amnioreduction, cerclage, interwin membrane disruption, procedure complications and chorioamniotic membrane separation as predictors for both gestational age at delivery and latency. Conclusion: Premature delivery after laser therapy for twin-twin transfusion syndrome is primarily due to spontaneous labor, preterm premature rupture of membranes and non-reassuring status of the donor fetus. Placental abruption was found to be a frequent complication resulting in early delivery. Future research should be directed toward the goal of prolonging gestation after laser photocoagulation to further reduce morbidity and mortality associated with twin-twin transfusion syndrome.


Subject(s)
Fetal Therapies , Fetofetal Transfusion , Laser Therapy , Female , Fetofetal Transfusion/surgery , Fetoscopy , Humans , Infant, Newborn , Laser Therapy/adverse effects , Placenta , Pregnancy , Retrospective Studies , United States
18.
AJP Rep ; 8(4): e230-e233, 2018 Oct.
Article in English | MEDLINE | ID: mdl-30345160

ABSTRACT

Optimal prenatal management of giant placental chorangioma (also known as chorioangioma, angiomyxoma, fibroangiomyxoma, or fibroma) has yet to be determined. Interventions intended to devascularize the tumor such as interstitial laser, bipolar coagulation, fetoscopic laser photocoagulation, and chemical embolization have met mixed results. We report a minimally invasive, extra-amniotic approach, technically similar to cordocentesis, of microcoil embolization of the feeding vessel. These percutaneously placed microcoils initiate clot formation at the site of insertion and are unable to migrate through the tumor, thereby minimizing fetal harm by downstream embolic phenomena. Intervention at 26 and 22 weeks resulted in intraoperative fetal loss in the former and vaginal delivery at term of a healthy neonate in the latter. Preoperative, intraoperative, and placental findings are highlighted. The ease and safety of this procedure may alter the risk-benefit equation toward earlier intervention with potentially better clinical outcomes.

19.
World Neurosurg ; 114: e976-e981, 2018 Jun.
Article in English | MEDLINE | ID: mdl-29588243

ABSTRACT

INTRODUCTION: Hydrocephalus due to congenital aqueductal stenosis (CAS) has significant long-term clinical implications. Previous reports on outcomes after treatment of congenital hydrocephalus are heterogenous and lack specificity for the subgroup of patients with isolated aqueductal stenosis. METHODS: An institutional surgical database was queried for the time period of 2005-2013 for patients with the diagnosis of isolated CAS and >2 years of clinical follow-up. Medical history and neurodevelopmental outcomes were recorded. RESULTS: The institutional cohort consisted of 41 patients with isolated CAS who underwent cerebrospinal fluid diversion. At a mean follow-up of 5.9 years, 48.8% carried a diagnosis of epilepsy and 68% were developmentally delayed. Four patients were diagnosed with cerebral palsy (9.8%). In total, 78% of patients were shunt-dependent, and the remainder had patent third ventriculostomies. Only 32% of patients in our cohort were neurologically normal after long-term follow-up despite contemporary management. CONCLUSIONS: Regardless of the initial treatment strategy, the age at diagnosis, or the timing of cerebrospinal fluid diversion after birth, patients with aqueductal stenosis have high rates of epilepsy, neurodevelopmental delay, and educational difficulties, and few are neurologically normal despite contemporary management. Investigation into in utero identification and correction of hydrocephalus may result in improved outcomes and warrants further investigation.


Subject(s)
Hydrocephalus/diagnostic imaging , Hydrocephalus/surgery , Ventriculoperitoneal Shunt/trends , Ventriculostomy/trends , Child , Child, Preschool , Cohort Studies , Developmental Disabilities/diagnostic imaging , Developmental Disabilities/epidemiology , Epilepsy/diagnostic imaging , Epilepsy/epidemiology , Female , Follow-Up Studies , Humans , Hydrocephalus/epidemiology , Infant , Infant, Newborn , Male , Retrospective Studies , Third Ventricle/diagnostic imaging , Third Ventricle/surgery , Treatment Outcome
20.
J Matern Fetal Neonatal Med ; 31(19): 2599-2604, 2018 Oct.
Article in English | MEDLINE | ID: mdl-28662614

ABSTRACT

BACKGROUND: Thromboelastography (TEG) is utilized as a point-of-care test of coagulation function to improve evidence-based blood product replacement in adults. In contrast to standard indices of coagulation, TEG reflects the dynamic interactions among the elements involved in hemostasis, including fibrinolysis. Although normal adult values and various abnormalities have been characterized, normative values for term neonates have not been described. Studies of neonatal TEG remain limited and have small sample sizes with inconsistent methodology. The aim of this study is to provide normative data on healthy term neonates, and to assess the impact of mode of delivery on TEG parameters at term. METHODS: Venous umbilical blood was obtained from the placenta within 10 min of delivery. TEG analysis of citrated kaolin-activated samples was performed for 50 healthy term vaginal and 50 cesarean deliveries. Samples collected for cesarean sections were from scheduled cases or unscheduled ones due to failure of progression of labor. RESULTS: Healthy neonates with uncomplicated vaginal term deliveries resulted in the following TEG parameters: R: 5.41 ± 1.34 (mean ± SD) min; K: 1.62 ± 0.75 min; α-angle: 65.39 ± 8.77°; MA: 65.86 ± 5.81 mm; and LY30: 1.40 ± 1.18%. Results for the cesarean delivery neonatal TEG assays showed: R: 5.51 ± 1.74 (mean ± SD) min; K: 1.52 ± 0.47 min; α-angle: 64.15 ± 4.61°; MA: 64.15 ± 4.61 mm; and LY30: 2.44 ± 3.51%. Of note, no statistical differences were observed (p < .01) between the groups. CONCLUSION: TEG measurements from term neonates were no different when the neonates were delivered vaginally or by cesarean section. Labor had no effect on neonatal TEG levels. Neonatal TEG values may therefore serve as insight for fetal values at the appropriate postconceptional age.


Subject(s)
Infant, Newborn , Thrombelastography , Delivery, Obstetric , Fetal Blood/physiology , Humans , Reference Values
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