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PubMed1996

Preterm delivery and perinatal death among black and white infants in a Chicago-area perinatal registry.

OBJECTIVE: To explore associations between race, preterm delivery, etiologic classification of preterm delivery, and perinatal mortality. METHODS: The study population consisted of 13,010 black and 19,007 white mother-infant pairs delivered at Chicago-area hospitals in 1988-1989 categorized as term or preterm births. Preterm births were further divided by severity and etiology. Black-white differences in perinatal mortality within groups were calculated and adjusted for birth weight and other potential confounding variables. RESULTS: Black women were nearly twice as likely as whites to experience preterm (before 37 weeks' gestation) and very preterm (before 32 weeks' gestation) delivery associated with premature rupture of membranes (PROM) or classified as idiopathic. Although black infants were also found to have twice the perinatal mortality risk of white infants (relative risk [RR] 2.1, 95% confidence interval [CI] 1.7-2.5), the overall preterm perinatal mortality rates did not differ between black and white women (RR 1.0, 95% CI 0.8-1.2). However, among preterm births, perinatal mortality was not uniform within categories of medical etiology. The mortality risk was the same for black and white infants born preterm following polyhydramnios or placental complications (RR 1.1, 95% CI 0.6-1.9), the same for black and white infants born preterm after labor induction (RR 1.1, 95% CI 0.6-1.9), and higher for black infants classified as idiopathic preterm deliveries (RR 1.6, 95% CI 1.1-2.3). In contrast, mortality rates tended to be lower for black infants born preterm following PROM-amnionitis (RR 0.8, 95% CI 0.5-1.2). The idiopathic disparity was explained by a differential birth weight distribution (adjusted RR 1.1, 95% CI 0.7-1.9); however, the apparent survival benefit among black infants born preterm following PROM increased even further after adjustment for birth weight (adjusted RR 0.4, 95% CI 0.2-0.7). CONCLUSION: Black infants born preterm after PROM appear to have a survival advantage compared with their white counterparts, an effect not observed within other etiologic categories of preterm delivery.

Adult↗
PubMed1983

Foeto-maternal morbidity in the presence of antibodies to Trypanosoma cruzi.

Pregnancy outcome and maternal morbidity were analysed in 302 pregnant women with positive Machado-Guerreiro Complement Fixation Test (MGCFT) for antibodies to Trypanosoma cruzi and 302 women with a negative MGCFT resident of Córdoba, Argentina. A comparison of positive and negative cases was employed to investigate the association between MGCFT reaction and pregnancy outcome assessed from abortion and perinatal infant mortality rates; similarly, the data were analysed for demographic and socio-economic factors, numbers of previous pregnancies (birth order) and maternal complications. A twofold increase in the risk of pregnancy loss associated with a positive MGCFT was found. The incidence of polyhydramnios and varicosities of the leg was significantly higher in the serologically reactive group.

Abortion, Spontaneous↗
PubMed1984

Direct prenatal chromosome diagnosis of a malignancy.

A fetal tumor was suspected at 31 weeks of gestation. The occurrence of polyhydramnios led to an ultrasound examination, which revealed deformation of the fetal head, face, eye, and neck. This was confirmed by computerized tomography. Amniocentesis yielded cells with an inverted duplication of chromosome #1. This abnormality of chromosome #1 marked the malignant teratoma cells in the amniotic fluid. Cytogenetic analysis of tumor tissue and of normal tissue obtained postnatally confirmed that the abnormality of chromosome #1 observed in amniotic fluid cells was confined to the tumor. The constitutional karyotype was normal. To our knowledge, this is the first report of the direct chromosomal detection of malignancy before birth.

Adult↗
PubMed1995

The grand multipara.

Grandmultiparity (GMP) has long been considered an obstetric complication for both mother and fetus, although recent studies indicate that, with proper perinatal care, women with high-parity rates are no longer at high risk. The current study examines the outcome of delivery in 1700 women in their fifth or more delivery, as compared with two control groups: 622 primiparas and 735 multiparas (two to three previous deliveries). Excellent prenatal care was available free of charge to all parturients. Our objectives were to evaluate the management of GMP in contemporary obstetrics and to assess whether grand multiparas are still high-risk patients. The age of the grandmultiparas was significantly higher compared with with the control groups, which may explain the higher incidence among them of antenatal medical disorders, such as diabetes mellitus and hypertensive disease. No significant differences were found among the three groups for preterm or post-term births, small-for-gestational-age infants, polyhydramnios, oligohydramnios, perinatal death, fetal distress, multiple births, placenta previa, abruptio placentae or cord prolapse. Macrosomia was markedly higher in the grandmultiparas and multiparas than in nulliparas. Thus, our results indicate that good perinatal care can ensure better results in grandmultiparas, and that grandmultiparity no longer needs to be considered a high-risk obstetric category in our population.

Adult↗
PubMedDate not supplied

The spectrum of congenital facial diplegia (Moebius syndrome).

The Moebius syndrome consists of congenital facial diplegia with associated anomalies. No single pathophysiologic hypothesis accounts for all aspects of the syndrome. We present six cases which manifest a very broad spectrum of associated neurologic anomalies. Postmortem examination was performed on two cases. Midline brainstem necrosis was evident in one while the other had subtle brainstem hypoplasia. The four other patients have demonstrated fixed deficits on follow-up examinations. Improvement in brainstem evoked potential waveforms has occurred in three cases, but the significance is uncertain. Many of the cases were delivered by caesarean section because of failure of labor to progress, and had associated polyhydramnios and arthrogryposis. These features suggest that a defect is established in utero. Only one infant, born prematurely, may have suffered some perinatal brainstem insult. Congenital facial diplegia is a heterogenetic entity which can affect the nervous system in many different ways.

Abnormalities, Multiple↗
PubMedDate not supplied

Clinical features for prediction of survival in neonatal muscle disease.

Review of 17 newborns with muscle disease demonstrated that clinical features associated with survival beyond 1 year of age included gestational age of at least 35 weeks and requirement of mechanical ventilation for less than 21 days. In contrast, poor outcome was associated with Apgar scores below 5 at 5 min, pulmonary complications, arthrogryposis, or other congenital anomalies. The incidence of decreased fetal movements, polyhydramnios, hypotonia, and assisted delivery was not statistically different between infants who died early and those who survived beyond 1 year of age. Clinical features of newborns with muscle disease may be useful for prediction of outcome, especially when muscle biopsy abnormalities are nonspecific.

Biopsy↗
PubMed1994

Neuropathology of infant with Pena-Shokeir I syndrome.

In an infant with clinical features of Pena-Shokeir I syndrome, who survived for 182 days, neuropathologic examination revealed little myelination in peripheral nerves with group atrophy of muscle fibers, dysplasia of inferior olivary and dentate nuclei, and leptomeningeal heterotopia. Congenital peripheral neuropathy associated with minor brain anomalies is characteristic in this patient, and may cause absence of fetal movements leading to ankylosis of multiple joints, absence of breathing in association with pulmonary hypoplasia, absence of swallowing causing polyhydramnios, and absence of movements of facial muscles causing craniofacial anomalies.

Abnormalities, Multiple↗
PubMedDate not supplied

Sodium valproate augments spontaneous neural tube defects and axial skeletal malformations in TO mouse fetuses [corrected].

The TO mouse exhibits a low incidence (3.65%) of spontaneous exencephaly at birth. The objectives of this study were to determine if sodium valproate (VPA) would augment this background frequency of exencephaly and to characterize its gross and histologic bases. Single doses of 200, 400, or 600 mg/kg of VPA were administered on one of gestation days (GD) 7 to 10 and fetuses were collected on GD 18. Significant augmentation of the background incidence of exencephaly was observed in the GD 7 and 8 treatment groups. Absence of the skull vault, hemorrhage, and degeneration of the exposed brain, polyhydramnios, and a female excess characterized the abnormality. Exencephalic embryos were markedly growth retarded. In addition to craniofacial and urogenital anomalies, severe axial skeletal malformations were found to be consistently associated with exencephaly. Morphometric evaluation of the alizarin red-stained skeleton confirmed significant skeletal growth inhibition. Histologic sections of GD 10 embryos revealed early onset of treatment-related growth retardation. Arrest of closure appeared to affect intermittent segments of the neural tube. The closure defect sometimes only involved the surface ectoderm of the dorsal midline. The unclosed neural tube was at times covered by a continuous layer of surface ectoderm. Cell death per se was not pronounced in the neuroepithelium. The mesenchyme was generally sparse and edema was obvious in embryos with partial closure. Growth inhibition of the optic and otic primordia was marked by pronounced cell death in these structures as well as in the otic and trigeminal ganglia and in the pharyngeal arch mesenchyme. Evidence for neural crest cell migration was also recorded. These data indicate that VPA interacts with genetic susceptibility, augments the frequency of exencephaly, and also induces other malformations in the TO mouse. The widespread malformations of the cranifacial structures are suggestive of the preferential effect of VPA on the neural crest or its derivatives.

Analysis of Variance↗
PubMed1998

X-linked myotubular myopathy--a long-term follow-up study.

X-Linked myotubular myopathy is a well delineated congenital myopathy, with a high neonatal and early childhood mortality. Only a single gene, mapping to Xq28 has been implicated and has recently been characterized. Phenotypic variability, both inter- and intrafamilial, has been recorded. Its severest expression is a uniform disease with polyhydramnios due to prenatal (neuromuscular) swallowing disorder, and partial inability to expand the lungs postnatally leading to early postnatal death in all. The mildest expression appears to be represented by the first family reported in the literature in which intrafamilial phenotypic variability was marked. There was neonatal asphyxia, but recovery took place in most affected patients and very mild expression permitting normal life into adulthood has been found in two patients. A long-term follow-up is given on both these families. Results emphasize the importance of the family history when trying to prognosticate in an individual case.

Adult↗
PubMed2005

Cloning and cellular expression of aquaporin 9 in ovine fetal membranes.

OBJECTIVE: Amniotic fluid (AF) absorption across fetal membranes is essential for AF volume homeostasis, balancing fetal swallowing, urine flow, and lung liquid production. In sheep, AF is absorbed primarily across the amniotic membrane into fetal vasculature situated between the amnion and chorion. Aquaporins (AQPs) are cell membrane proteins that serve as water channels. Recent studies have demonstrated the expression of AQP 1, 3, 8, and 9 in human chorioamniotic membranes and placenta. As AF dynamics continued to be explored primarily in the ovine model, we sought to clone and characterize the expression of ovine AQP9 in fetal membranes. METHODS: Ovine AQP9 gene was cloned with the use of homology reverse transcriptase-polymerase chain reaction (RT-PCR). RT-PCR and Northern analysis were used to determine AQP9 gene expression, and immunohistochemistry (IHC) used to localize AQP9 protein expression in ovine fetal membranes. RESULTS: A 2085-base pair (bp) full-length complementary DNA (cDNA) sequence of ovine AQP9 was cloned. The ovine AQP9 cDNA is 86%, 82%, and 82%, and the predicted amino acid sequence (295 amino acids) is 77%, 71%, and 69% identical to human, rat, and mouse AQP9, respectively. RT-PCR and Northern analysis detected AQP9 messenger RNA expression in ovine amnion and allantois, but not in placenta, chorion, or umbilical cord. Immunohistochemistry localized AQP9 protein in epithelia of amnion and allantois. CONCLUSION: The presence of significant AQP9 messenger RNA and protein expression in ovine fetal membranes suggests that AQP9 may be a major water channel for intramembranous AF resorption in sheep. The cloning of ovine AQP9 and the demonstration of AQP9 expression in amnion and allantois significantly enhances our understanding of ovine AF regulation and offers the potential for therapeutic approaches for the treatment of oligohydramnios and polyhydramnios.

Allantois↗
PubMed2006

Accuracy of obstetric diagnoses and procedures in hospital discharge data.

OBJECTIVE: The objective of the study was to estimate the validity of obstetric procedures and diagnoses in California patient discharge data. STUDY DESIGN: We randomly sampled 1611 deliveries from 52 of 267 California hospitals that performed more than 678 eligible deliveries in 1992 to 1993. We compared hospital-reported procedures and diagnoses against our recoding of the same records. RESULTS: Cesarean, forceps, and vacuum delivery were accurately reported, with sensitivities and positive predictive values exceeding 90%. Episiotomy was underreported (70% sensitivity). Cesarean indications were reported with at least 60% sensitivity, except uterine inertia, herpes, and long labor. Among comorbidities, sensitivity exceeded 60% for chorioamnionitis, diabetes, premature labor, preeclampsia, and intrauterine death. Sensitivity was poor (less than 60%) for anemia, asthma, thyroid disorders, mental disorders, drug abuse, genitourinary infections, obesity, fibroids, excessive fetal growth, hypertension, premature rupture, polyhydramnios, and postdates. CONCLUSION: The validity of hospital-reported obstetric procedures and diagnoses varies, with moderate to high accuracy for some codes but poor accuracy for others.

Female↗
PubMed2006

Prevalence and management of late fetal complications following successful selective laser coagulation of chorionic plate anastomoses in twin-to-twin transfusion syndrome.

OBJECTIVE: This study was undertaken to report on the prevalence and management of late complications in twin-to-twin transfusion syndrome (TTTS) treated by laser therapy when both twins are alive 1 week after surgery. STUDY DESIGN: A total of 151 consecutive TTTS cases were treated by selective fetoscopic laser therapy. Cases in which both twins were alive 1 week after surgery were followed up with ultrasound and Doppler examination, including middle-cerebral artery peak systolic velocity measurement (MCA-PSV). RESULTS: In the 151 cases treated with laser, both twins were still alive 7 days after the procedure in 101 cases. Intrauterine death of 1 and both twins occurred in 7 and 1 cases, respectively. Recurrence of TTTS with the polyhydramnios-oligohydramnios sequence occurred in 14 (14%) cases. In another 13 (13%) cases, amniotic fluid remained normal in both sacs, but MCA-PSV increased above 1.5 MoM in 1 twin and decreased concomitantly below 0.8 MoM in the co-twin. This was related to anemia and polycythemia, respectively, and resulted from unidirectional feto-fetal blood transfusion, mainly from former recipients into former donors. Late complications were managed accordingly by repeat laser, amnioreduction, cord coagulation, intrauterine blood transfusion, or elective delivery. CONCLUSION: MCA-PSV Doppler measurements are useful in the follow-up of double survivors to detect and manage late complications after selective laser therapy.

Anemia↗
PubMed2006

Surgical management of twin reversed arterial perfusion sequence.

OBJECTIVE: The purpose of this study was to review our experience in the management of twin reversed arterial perfusion sequence to derive management recommendations. STUDY DESIGN: All patients with twin reversed arterial perfusion sequence who were seen for consultation between 1993 and 2004 were studied. Criteria for umbilical cord occlusion included abdominal circumference of the twin reversed arterial perfusion fetus that was more than or equal to the pump twin, polyhydramnios (maximum vertical pocket > or = 8 cm), abnormal Doppler studies or hydrops of the pump twin, or monoamniotic twins. Various surgical and access techniques for umbilical cord occlusion were used as they were developed. RESULTS: Seventy-four patients with twin reversed arterial perfusion sequence were studied. Sixty-five patients were considered surgical candidates, of which 51 patients underwent umbilical cord occlusion attempt. The overall perinatal survival for surgical candidates who had umbilical cord occlusion was 65% (33/51 patients) versus 42.9% (6/14 patients) for the surgical candidates who did not undergo umbilical cord occlusion (P = .1). However, perinatal outcomes in surgical patients were significantly better than expectantly treated surgical candidates if the dividing membrane was not disrupted purposely (22/28 patients; 78.5%; P = .02). Surgery within the sac of the twin reversed arterial perfusion fetus was feasible in 23.5% of patients and was associated with no incidence of premature rupture of membranes, with 83% perinatal survival, and with a significantly greater gestational age at delivery (36 weeks). There were no significant differences in perinatal outcome relative to the specific surgical technique that was used. CONCLUSION: Surgical management of twin reversed arterial perfusion sequence is indicated in high-risk patients. The surgical approach and the surgical technique should be tailored to the specific clinical presentation, preferably by performing the surgery within the sac of the twin reversed arterial perfusion sequence fetus and avoiding disruption of the dividing membrane.

Arteries↗
PubMed2006

Clinical evidence of intrauterine disturbance in Prader-Willi syndrome, a genetically imprinted neurodevelopmental disorder.

BACKGROUND: Imprinted genes are considered to play an important role in growth and early development but much of the research is based on animal studies. AIM: This study reports clinical data from a French population concerning prenatal, perinatal and postnatal complications in Prader-Willi syndrome (PWS), a genetically imprinted neurodevelopmental disorder associated with growth retardation, intellectual impairment and obesity. STUDY DESIGN: Data from family health records concerning prenatal, perinatal and postnatal complications were collected from 52 French people with the deletion form (DEL), and 34 French people with the maternal disomy form of PWS (UPD) and compared against national norms and between groups. RESULTS: Significant findings include: a history of miscarriage, high rate of polyhydramnios (12/34 UPD, 11/52 DEL), a high rate of induced labour, a high rate of Caesarian section (20/34 UPD, 26/52 DEL), small gestational age (10/34 UPD, 22/52 DEL), hypotonia (34/34 UPD, 49/52 DEL), and suckling deficit (25/34 UPD, 46/52 DEL). Significant differences between genetic subtypes include a higher rate of induced labour in UPD (27/34 UPD, 25/52 DEL), an increased risk of premature term in UPD (9/34 UPD vs. 4/52 DEL), raised maternal age in UPD (36.4 years vs. 29.3 years), low birth weight for newborns with a deletion form of PWS (girls 2.8 kg, boys 2.7 kg), a positive correlation between parental weight and offspring birth weight only for patients with UPD (UPD maternal: r=0.62, paternal: r=0.51). CONCLUSION: The results indicate significant intrauterine disturbance in PWS, particularly in PWS due to UPD, but a more significant weight disturbance for PWS due to deletion.

Adolescent↗
PubMed2005

Relationships between perinatal and maternal characteristics and hepatoblastoma: a report from the UKCCS.

Within the context of a national population-based case-control study--the United Kingdom Childhood Cancer Study (UKCCS)--we aimed to explore relationships between perinatal and maternal factors and childhood hepatic tumours, for participants with data available from medical records. 26/28 children with hepatic tumours (22/24 hepatoblastomas, 4/4 hepatocellular carcinomas (HCC)) and 4753 age- and sex-matched controls were included. Polyhydramnios was associated with 0.9% of control pregnancies and 13.6% of case pregnancies (Odds Ratio (OR)=28.64, 95% Confidence Interval (CI)=6.94-118.21, P<0.0001); eclampsia or severe pre-eclampsia complicated the pregnancies of 16.7% of mothers whose children developed hepatoblastoma compared with 0.5% of control pregnancies (OR=52.50, 95% CI=10.75-257.05, P<0.0001). Three children with hepatoblastoma weighed <1500 g at birth, two of whom weighed <1000 g (OR for birthweight <1500 g=69.00, 95% CI=11.98-397.17, P<0.0001). Of children with hepatoblastoma, 50% (11/22) had records of congenital anomalies, as did two of their mothers. Three mothers of children with hepatoblastoma had diagnoses of cancer--two of papillary carcinoma of the thyroid and one of acute lymphoblastic leukaemia (ALL). Paediatricians and others should be alert to the possibility of familial or genetic syndromes in children with hepatoblastomas. Potential links between maternal pre-eclampsia, low birthweight and subsequent malignancy merit further investigation. Hepatoblastoma is an extremely rare childhood tumour, but understanding the mechanism(s) underlying severe pre-eclampsia and eclampsia may also shed light on factors that contribute to the development of hepatoblastoma.

Carcinoma, Hepatocellular↗
PubMed2005

Perinatal and reproductive factors: a report on haematological malignancies from the UKCCS.

The United Kingdom Childhood Cancer Study was designed to examine the potential aetiological role of a range of perinatal and reproductive factors. Our use of clinical records permitted a more exact characterisation of reproductive events than is possible in investigations that rely on self-reporting; and the increased specificity with which antecedent events were measured produced more precise risk estimates, albeit ones based on progressively smaller numbers. Information on the conduct of this component of the study and results for 1485 children with haematological malignancies and 4864 controls are presented. The 'find' rate for obstetric records was high at 86% for cases, with 81% having information on both matched controls. Associations were seen for severe hyperemesis (Odds Ratio=3.6, 95%Confidence Interval=1.3-10.1, for all leukaemias), polyhydramnios (OR=4.0, 95%CI=1.5-10.3, for acute myeloid leukaemia (AML)), anaemia (haemoglobin <10 g, OR=2.6, 95%CI=1.7-4.1, for AML), and pre-eclampsia (OR=1.7, 95%CI=1.1-2.7, for non-Hodgkin's lymphoma). Babies who developed leukaemia were heavier at birth (>4000 g, OR=1.2, 95%CI=1.0-1.4), as were their older siblings (>4000 g, OR=1.4, 95%1.0-1.9). Mothers' whose children developed common B-cell precursor acute lymphoblastic leukaemia (ALL) were more likely to have had a previous molar pregnancy (OR=5.2, 95%CI=1.9-14.7). Gender-specific analysis revealed that findings often differed markedly for boys and girls; and, in common with other reports, strong associations with Down's syndrome were seen for both ALL and AML.

Acute Disease↗
PubMed2003

Longitudinal observation of antenatally detected congenital lung malformations (CLM): natural history, clinical outcome and long-term follow-up.

OBJECTIVE: The objective of the study is to present longitudinal observations in antenatally detected congenital lung malformations (CLM), particularly pulmonary sequestration (PS) and cystic adenomatoid malformation (CAM). METHODS: Fetuses found to have a CLM on prenatal ultrasound (US) were included in this study and followed up until delivery. In all newborns radiographs and computerized tomography (CT) studies of the thorax were performed. Surgical procedures included sequesterectomy, lobectomy, segmentectomy, and non-anatomic resection. Based on prenatal US findings, intrauterine course, postpartum chest radiographs and CT scans, as well as clinical signs and surgical findings patients were divided into six groups. RESULTS: Over a period of 6 years, routine prenatal US revealed suggestion of CLM in a series of 35 consecutive fetuses. In six cases pregnancy was terminated or the fetuses suffered fetal demise. Another four fetuses became symptomatic in utero when sequential scanning revealed hydrops, hydrothorax, and enlargement of cysts or polyhydramnios. Three cases in this group received serial therapeutic amniocentesis and serial puncture of either the hydrothorax or intrapulmonary cysts. After postpartum treatment in the intensive care unit surgical procedures were performed uneventfully and confirmed the diagnosis of CAM, PS or hybrid type lesions. In 11 patients US findings were considered to demonstrate spontaneous resolution of the lesion, but disappearance without sequelae could be confirmed only in six infants. Five infants were shown to have persistent CLM on postpartum CT scans. These infants underwent resection of the lesion within the first year of life. In 11 fetuses CLM were continuously demonstrated during pregnancy with only slight changes in size and structure. Postpartum the infants were asymptomatic and were subjected to a systematic plan of diagnostic work-up and treatment. Surgery in these infants revealed a large number of hybrid type lesions (n=5). In three infants, the primary diagnosis of PS or CAM had to be corrected during the diagnostic and therapeutic work-up. CONCLUSION: CLM are diagnosed antenatally with an increasing frequency and are shown to be quite different from previously applied concepts. The expected clinical outcome is far better than thought to be possible.

Abortion, Legal↗