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PubMed1992

The role of prostaglandins in obstetrical disorders.

All pregnancy-associated tissues are capable of producing prostaglandins including PGI2 and TXA2. In normal pregnancy there is a dominance of PGI2 over TXA2 which may contribute to the maternal circulatory adaptation to pregnancy. Furthermore, both fetoplacental PGI2 and TXA2 production are important regulators of the fetal blood supply. It has been clearly established that in pre-eclampsia PGI2 production decreases in the fetoplacental tissues and quite probably also in the maternal tissues. The effect of this change may be further exaggerated by the simultaneous stimulation in pre-eclampsia of TXA2 production. The reason for PGI2 deficiency is not known. Other vasoactive agents, such as endothelin, may act in concert with prostaglandins. Relative PGI2 deficiency is likely to exist also in IUGR and lupus anticoagulant syndrome of pregnancy. In the latter, lupus anticoagulant may directly inhibit the synthesis of PGI2. One study suggests PGI2 deficiency also in early pregnancies of women with a history of repeated abortions. Prostaglandin production increases during full-term labour, and similar but smaller changes also occur in preterm labour. A silent bacterial infection may trigger the onset of preterm labour through cytokine-stimulated increase of prostaglandin production. No data were found on prostaglandin production in post-term pregnancies. That oligo-polyhydramnios is possibly prostaglandin mediated is suggested by the control of polyhydramnios by indomethacin treatment. Smoking decreases the production of PGI2 and possibly increases that of TXA2, which may lead to decreased blood flow and IUGR. Which constituent of cigarette smoke exerts this effect is not known. Ethanol consumption causes aberrations in prostaglandin metabolism which cannot be directly connected with fetal alcohol effects.

Abortion, Habitual↗
PubMed1998

Risk factors in premature rupture of membranes.

Objective: This retrospective study was undertaken to investigate risk factors in women who have preterm premature rupture of membranes. This information will aid the clinician in targeting at-risk women for intensified obstetric care and entry into prevention programs.Methods: 28,725 deliveries were analyzed over a 16-month time frame (January 1, 1995-April 30, 1996). These data were collected via a 14 county, 23 hospital population based Perinatal Data System. The associations between premature rupture of membranes and risk factors were analyzed using chi(2) and Fisher's Exact test analyses. Odds ratios show the magnitude of these associations.Results: Risk factors included vaginal bleeding, tobacco usage, multiple births, polyhydramnios, maternal age <20 years and >34 years, congenital malformations, parity, race, in vitro fertilization, sexually transmitted disease, prior preterm delivery, and incompetent cervix. Premature rupture of membranes shows a significant association with prior preterm delivery, prior history of a low birth weight infant, incompetent cervix, tobacco use (12 pack per day), multiple births, parity, in vitro fertilization, sexually transmitted diseases, and viral diseases, P <.001 (for all of these risk factors), polyhydramnios and non-white race, P <.01.Conclusions: These associations help to develop a picture of the patient at risk for premature rupture of membranes. Strategies can be better developed to enter high-risk patients into prevention programs to maximize pregnancy outcomes.

Journal Article↗
PubMed2004

Severe neonatal onset of glycogenosis type IV: clinical and laboratory findings leading to diagnosis in two siblings.

Glycogenosis type IV is an autosomal recessive disease, exceptionally diagnosed at birth: only very few reports of the fatal perinatal neuromuscular form have been described. We report on two sibling male newborns who died at 10 and 4 weeks of age with clinical signs of a systemic storage disease. Prenatal history included polyhydramnios, reduced fetal movements and fetal hydrops, and Caesarean section was performed at 36 weeks of gestational age because of fetal distress. At birth, both babies showed severe hypotonia, hyporeflexia and no spontaneous breathing activity. They never showed active movements, sucking and swallowing and were respirator-dependent until death. A muscle biopsy revealed, in both patients, the presence of PAS-positive and partially diastase-resistant cytoplasmic inclusions containing granular and filamentous amylopectin-like material. This suggested that the stored material consisted of abnormal glycogen. At autopsy, ultrastructural examination of cardiac and skeletal muscle, liver, kidney and brain showed PAS-positive diastase-resistant eosinophilic cytoplasmic inclusions. Determination of branching enzyme activity, in cultured fibroblasts from the second patient, showed markedly reduced enzyme activity, confirming diagnosis of glycogenosis type IV. Our patients showed the full spectrum of both prenatal signs (hydrops, polyhydramnios) and postnatal signs (hypotonia, hyporeflexia, absence of active movements, cardiomegaly), which have been reported previously. They suffered from a very severe form of glycogenosis type IV with clinical and histological involvement of many tissues and organs. Diagnosis was accomplished on the second baby and required several biochemical and histological studies, in order to rule out both neuromuscular disorders and the most common storage diseases with neonatal onset. In our experience, the correct interpretation of the histological findings was essential in the search for the diagnosis.

Age of Onset↗
PubMed2002

Poor prenatal detection rate of cardiac anomalies in Noonan syndrome.

BACKGROUND: The wide variation and nonspecific nature of many of the associated ultrasonographic findings complicate prenatal diagnosis of Noonan syndrome. The aim of the present study was to define the rate of prenatal diagnosis of heart malformations in cases diagnosed postnatally with Noonan syndrome. METHODS: English-language literature review of 29 cases of Noonan syndrome examined prenatally with confirmed postnatal diagnosis and four case reports from our center. RESULTS: Cases were evaluated for cervical spine pathologies, cardiac anomalies and other pathological findings, including hydrops fetalis and polyhydramnios. Cardiac anomalies were suspected in only nine of 33 cases; three of these were associated with cystic hygroma. Cardiac anomalies were eventually diagnosed in 31/33 cases postnatally. Polyhydramnios was diagnosed in 19/33 cases in the third trimester, and hydrops fetalis was detected in eight of 33. Cystic hygroma was present in a total of nine cases at mid-trimester. CONCLUSIONS: Noonan syndrome is characterized by late-onset and progressive pathologies, particularly the associated cardiac anomalies, which develop through the course of gestation and postnatal life. This complicates or precludes prenatal diagnosis at mid-trimester or at any time in the prenatal period, and partly explains the low rate of detection of fetal cardiac lesions in this syndrome.

Female↗
PubMed2003

Pregnancy in women with Type 2 diabetes: 12 years outcome data 1990-2002.

AIM: Twelve years' outcome analysis of pregnancies in women with Type 2 diabetes in a multiethnic geographically defined area. METHODS: Information about 182 women delivered between 1990 and 2002 was ascertained from a regional computerized database. The main outcome measures were rates of miscarriage, stillbirth, neonatal/postnatal deaths, congenital malformations, birth weight, mode of delivery, and neonatal unit care as well as maternal morbidities of polyhydramnios, postpartum haemorrhage, pregnancy-induced hypertension/pre-eclampsia. RESULTS: Among 182 singleton pregnancies, 161 (88%) resulted in a live outcome. There were 16 (8.8%) spontaneous miscarriages, two (1.2%) stillbirths, and three (1.6%) terminations. Congenital malformations occurred in 18 pregnancies (99/1000). There were two early and one late neonatal deaths and two further deaths in the postnatal period. Twenty-eight percent of infants were large for gestational age, with 15 (9.3%) greater than 4 kg. Fifty-three percent were delivered by caesarean section and 68 (37%) required admission to neonatal unit (NNU) care. Hypertension/pre-eclampsia was two times, polyhydramnios three times, and postpartum haemorrhage six times more common than in non-diabetic women. CONCLUSIONS: Women with Type 2 diabetes have a less satisfactory pregnancy outcome compared with the general population. Infants have a two-fold greater risk of stillbirth, a 2.5-fold greater risk of a perinatal mortality, a 3.5-fold greater risk of death within the first month and a six-fold greater risk of death up to 1 year compared with regional/national figures. They have an 11 times greater risk of a congenital malformation. We need to develop better educational and screening strategies if we are to improve.

Abortion, Spontaneous↗
PubMed1992

The natural history of isolated fetal hydrothorax.

We reviewed the prenatal course and outcome of 11 fetuses with isolated unilateral or bilateral hydro thoraces identified between 14 and 34 weeks' gestation, in the absence of invasive fetal intervention. Four of these fetuses died, three in utero and one in the newborn period. Six of seven survivors had resolution of hydrothorax before birth. Eight of the 11 fetuses had unilateral and three bilateral hydrothoraces. Six of the eight fetuses with unilateral hydrothorax survived, whereas one of the three with bilateral hydrothoraces lived. Six of the 11 pregnancies were complicated by polyhydramnios; four of these six fetuses died. One fetus was found to have trisomy 21. It is difficult to draw firm conclusions from the small number of patients in this report because of the wide range of outcomes found for the sonographic variables described. This series shows, however, a tendency to complete resolution of primary unilateral fetal hydrothorax, with good outcome, particularly in second-trimester fetuses without polyhydramnios.

Journal Article↗
PubMed1992

Successful outcome after Nd : YAG laser separation of chorioangiopagus-twins under sonoendoscopic control.

Endoscopic laser coagulation of the chorioangiopagus was successfully performed at 25 weeks' gestation in a pregnancy presenting with acute polyhydramnios due to twin transfusion syndrome. Under local anesthesia, a rigid fetoscope housed in a 2.7 mm diameter cannula was introduced transabdominally into the amniotic cavity and the communicating vessels were coagulated by Nd : YAG laser through a fiber that was passed down the side-arm of the cannula. There was rapid normalization of urine output in both fetuses and resolution of the oligo- and polyhydramnios in the donor and recipient twins, respectively. Furthermore, there was normalization in both fetal circulations, as assessed by Doppler velocimetry, and there was gradual resolution of the hydrops in the recipient. Two healthy babies were born 9 weeks after the procedure.

Journal Article↗
PubMedDate not supplied

[7-week-old infant with a hydrolethalus syndrome: case report, differential diagnosis and literature review].

We report on an 7 weeks old male infant with hydrocephalus internus, dysplastic low-set ears, micrognathia, vitium cordis, hypogenitalism, and polyhydramnios in pregnancy. The variability of the Hydrolethalus syndrome will be demonstrated. Our patient shows, that this syndrome is not a lethal in all cases. We report on an 7 weeks old male infant with hydrocephalus internus, dysplastic low-set ears, micrognathia, vitium cordis, hypogenitalism, and polyhydramnios in pregnancy.

Abnormalities, Multiple↗
PubMed1998

Diagnosis and differentiation of congenital diaphragmatic hernia from other noncardiac thoracic fetal masses.

This retrospective study was designed to evaluate individual sonographic parameters that might help differentiate congenital diaphragmatic hernia (CDH) from other noncardiac thoracic masses such as cystic adenomatoid malformation of the lung (CAML) and congenital lobar emphysema (CLE) prenatally. Twenty-four cases of CDH, CAML, and CLE detected during prenatal ultrasound and documented postnatally (with surgical, autopsy, or radiological proof) were identified through extensive chart and record review. The hard copy gray-scale images were retrospectively reviewed for imaging characteristics that may differentiate the three entities. Additionally, the prospective diagnosis during prenatal ultrasound was also compared with the postnatal diagnosis. The most reliable indicators in our retrospective review included confident visualization of a diaphragmatic defect (92.3/100.0 PPV/NPV, p< or =0.002) and/or localization of the stomach within the chest as well as the presence of severe cardiac deviation (both 92.3/62.5 PPV/NPV, p< or =0.01). Other sonographic indicators (including the presence of cystic areas, side and size of the lesion and the presence of polyhydramnios) offered lower levels of sensitivity and specificity. Prospective diagnosis during real-time assessment was also integral, offering >80% sensitivity and specificity (p< or =0.001). Accurate prenatal diagnosis of CDH is difficult despite the relative frequency of this lesion. The classic triad of a thoracic mass accompanying a displaced heart, absence of a normally positioned fluid-filled stomach and polyhydramnios, although seen with CDH, may not adequately differentiate this entity from other noncardiac fetal thoracic masses. Realtime assessment remains integral to the appropriate diagnosis.

Cystic Adenomatoid Malformation of Lung, Congenita↗
PubMed1995

Lack of mandibular movement manifested by absent fetal swallowing: a possible factor in the pathogenesis of micrognathia.

This study was performed to assess the association of lack of mandibular movement as manifested by absent fetal swallowing and micrognathia in a nonrestrictive intrauterine environment. Over a 5-year period, 14 fetuses with sonographic findings of polyhydramnios (amniotic fluid index [AFI] more than 20 cm), absent mandibular movement, and a nonvisualized fetal stomach, all consistent with absent fetal swallowing, were followed. A group of 14 fetuses, each with polyhydramnios (AFI more than 20 cm), yet with sonographic detection of fetal swallowing, served as controls. All gravidas in both groups were normoglycemic throughout gestation. Subsequent mandibular development was assessed at delivery or autopsy. Analysis of the data revealed that in the study group, 12 of these infants were liveborn, and two were stillborn. Eleven of the liveborn infants had an early neonatal death. All 14 infants of the study group demonstrated micrognathia. None of the control infants (all of whom survived) had micrognathia. In conclusion, this study supports the concept that normal mandibular growth may depend on the presence of mandibular movement during intrauterine development.

Adult↗
PubMed1994

Acute maternal obstructive renal failure in a twin gestation despite normal physiological pregnancy-induced urinary tract dilation.

Obstructive renal failure due to an overdistended uterus is a very rare complication of pregnancy. Most previously reported cases have involved twin gestations, the majority occurring in the presence of polyhydramnios. We describe a patient with a twin gestation who developed acute obstructive renal failure despite only very mild physiological pregnancy-induced dilation of the urinary collecting system in the absence of polyhydramnios. This case suggests that acute obstructive uropathy should be considered as a rare cause of acute renal failure even in cases with seemingly normal pregnancy-induced physiological urinary tract dilation.

Acute Kidney Injury↗
PubMed2002

Agenesis of the ductus venosus that is associated with extrahepatic umbilical vein drainage: prenatal features and clinical outcome.

OBJECTIVE: We sought to study the clinical and ultrasonographic findings and outcomes of fetuses with ductus venosus agenesis as the result of extrahepatic umbilical vein drainage. STUDY DESIGN: This was an observational study of 12 fetuses who were diagnosed with these anomalies between 1995 and 2001. RESULTS: The umbilical vein drained either directly into the right atrium (n = 7 fetuses), the inferior vena cava (n = 3 fetuses), or the iliac vein (n = 2 fetuses). Combined cardiac output (n = 8 fetuses) was nearly 2-fold increased to 891 +/- 210 mL/kg per minute. Other relevant findings were (1) considerable umbilical vein enlargement (12/12 cases; range, 8-13 mm) with an accelerated (peak, 0.53 +/- 0.1 m/s), (2) pulsatile Doppler flow pattern within its intra-abdominal course, (3) cardiomegaly (12/12 fetuses), (4) secundum atrial septal defects (5/12 fetuses), (5) extracardiac anomalies (5/12 fetuses), (6) polyhydramnios (4/12 fetuses), and (7) progressive heart failure (3/12 fetuses). Two fetuses died: 1 death was unexplained at 29 weeks of gestation; the other death from hydrops occurred after delivery at 32 weeks of gestation. So far, none of the survivors have had other long-term sequelae that were related to ductus venosus agenesis. CONCLUSION: Careful assessment of the umbilical venous return and the ductus venosus should be part of the routine evaluation of every fetus with heart failure and polyhydramnios.

Abnormalities, Multiple↗
PubMed1998

Congenital chloride diarrhoea in Kuwait: a clinical reappraisal.

Congenital chloride diarrhoea (CCD) is a recessively inherited disorder of chloride transport in the distal ileum and colon. Congenital chloride diarrhoea is a common metabolic disorder in Kuwait with an incidence of 1/3200. Clinical findings in 14 children with CCD are reported over a period of 4 years. Maternal polyhydramnios, abdominal distension, watery diarrhoea, and a high faecal chloride level > 90 mmol/l were the cardinal features in the neonatal period. In spite of the classical features of this disease 75 per cent of our cases were diagnosed beyond the neonatal period and all demonstrated chronic diarrhoea and failure to thrive, with hypochloraemia, hypokalaemia, and metabolic alkalosis. The practice of ultrasonic examination for pregnant women with polyhydramnios and, particularly, for those with previously affected siblings led to early identification of new cases among our population recently. The antenatal ultrasonic examination showed dilated intestinal loops which suggest CCD. The diagnosis was confirmed by a high faecal chloride level.

Child, Preschool↗
PubMed1997

Management of twin pregnancies discordant for anencephaly.

OBJECTIVE: To examine options of management and outcome of twin pregnancies discordant for anencephaly. DESIGN: Retrospective study. SETTING: Research Centre for Fetal Medicine. POPULATION: Twenty-four twin pregnancies discordant for anencephaly. METHODS: A computer search was made of our database for twin pregnancies discordant for anencephaly. The data were reviewed for gestation at presentation, chorionicity, management and pregnancy outcome. MAIN OUTCOME MEASURES: Pregnancy outcome in relation to chorionicity and management. RESULTS: There were 13 dichorionic and 11 monochorionic twin pregnancies discordant for anencephaly. In the dichorionic group five pregnancies had selective fetocide at 17 to 21 weeks; one pregnancy resulted in spontaneous abortion but in the others a healthy infant was born at a median gestation of 37 weeks. The other eight dichorionic pregnancies were managed expectantly, but three developed polyhydramios at 26 to 30 weeks; in one case amniodrainage was performed and in another selective fetocide was carried out. In this group the median gestation at delivery was 35 weeks. All 11 monochorionic pregnancies were managed expectantly and in three there was intrauterine death of both fetuses. In the other eight cases the normal twin was liveborn at a median gestation of 34 weeks; in four of these pregnancies polyhydramnios developed and two were managed by amniodrainage. CONCLUSIONS: In monochorionic pregnancies, expectant management is associated with a high rate of intrauterine lethality of the normal twin. In dichorionic pregnancies selective fetocide in the second trimester prevents the development of polyhydramnios and is associated with a lower risk of preterm delivery but can cause miscarriage.

Anencephaly↗
PubMed1989

Bart's hydrops fetalis--clinical presentation and management--an analysis of 25 cases.

Twenty-nine hydropic infants were born in the Kandang Kerbau Hospital between 1980 and 1985, during which there were 131,658 deliveries, giving an incidence of 1 in 4,540 total births. Twenty-five of these cases were confirmed to be due to homozygous alpha thalassaemia. No case of fetal hydrops due to Rh isoimmunization was detected. The mean age of the mothers was 28.86 +/- 4.05 years (+/- SD). Eight patients had delivered 1 hydropic baby previously while 1 had a history of 2 babies with hydrops fetalis; 92% of the patients had been followed antenatally while 8% were first seen when they were admitted in labour; 25% of the patients had anaemia, 52% had polyhydramnios, 20% developed hypertension and 64% had bilateral lower limb oedema. None of the patients had concomitant hypertension, generalized oedema and proteinuria. In 4 cases of recurrent hydrops, serial ultrasound scans were performed from early pregnancy but ultrasonic features of hydrops fetalis were only seen from 27 weeks' gestation. Spontaneous labour occurred in 75% of patients at a mean of 32.3 +/- 3.3 weeks (+/- SD). All delivered vaginally and only 1 patient required abdominal decompression. Four patients required Caesarean section, 2 for failure to progress after induction of labour, 1 for major placenta praevia and the fourth for fetal distress; in the last case, diagnosis of hydrops fetalis was only made after delivery of the baby. All the babies in the series died within one hour of delivery. Homozygous alpha thalassaemia is the commonest cause of hydrops fetalis in Singapore and is an invariably fatal condition. It is associated with an increased incidence of maternal anaemia, polyhydramnios and prematurity.

Adult↗
PubMed1992

The timing of delivery in diabetic pregnancy: a 10-year review.

The approach of term in the pregnancy of a diabetic woman is a time of anxiety for patient and physician alike. The impact of a conservative approach to the timing of delivery is outlined in this review of the clinical course and outcome of 276 pregnancies of diabetic women, delivered at the National Maternity Hospital, Dublin between 1981 and 1990. The mean gestation at delivery was 39 weeks, 229 patients (83%) delivered at or beyond 38 weeks and 112 patients (41%) delivered at or beyond 40 weeks. The overall induction of labour rate was 27% and the elective Caesarean section rate was 19%. Sixty seven percent of patients achieved a normal delivery, the forceps rate was 5%, and 28% of patients were delivered by Caesarean section. There were 16 perinatal deaths in the series, with 7 due to lethal malformations. There were 5 deaths of normally formed infants occurring at or beyond 38 weeks' gestation. All of these 5 deaths had been preceded by clinically apparent polyhydramnios or macrosomia and recognized poor control. This study stresses the value of strict diabetic control in the management of diabetic pregnancy and highlights the significance of polyhydramnios and macrosomia as indicators of risk approaching term in diabetic pregnancy.

Adolescent↗
PubMed1986

Specific skeletal dysplasias in utero: sonographic diagnosis.

A retrospective study was performed of 13 short-limbed fetuses with lethal skeletal dysplasias that were evaluated with ultrasound (US) from 1981 to 1984. The specific diagnoses were thanatophoric dwarfism, achondrogenesis, osteogenesis imperfecta, and campomelic dwarfism. Death occurred in utero or within 2 weeks after delivery in all cases. US examination showed other associated abnormalities, including polyhydramnios, hydrops, shortened femurs, and CNS abnormalities. Radiographs confirmed these findings and provided more information regarding the shape of the limbs and thorax and the appearance of the spine. The probable diagnosis of lethal short-limbed dwarfism was made antenatally using US in eleven of the fetuses. Spinal appearance, thoracic shape, and associated hydrops and polyhydramnios were most helpful in determining the specific type of dysplasia present. Lethal short-limbed skeletal dysplasia may be diagnosed confidently in utero using US examination; however, obstetric plain film radiography may be required to determine the definitive type of dysplasia. In certain cases, US may be sufficient to make a definitive diagnosis.

Bone Diseases, Developmental↗
PubMed2006

[Characteristics of neonates with and without a single umbilical artery. Analysis of two consecutive series of neonates with and without congenital defects].

BACKGROUND AND OBJECTIVE: The implications of the presence of a single umbilical artery (SUA) are unknown. Although most articles are based on selected samples, they suggest a relationship between SUA and malformations. Consequently, prenatal detection of SUA causes concern, since there are no definitive guidelines that can be followed after identification of this abnormality. The objective of this study was to comparatively analyze SUA in two series of consecutive births, with and without congenital defects. PATIENTS AND METHODS: A total of 19,909 cases and 19,148 controls from the Registry of the Spanish Collaborative Study on Congenital Malformations were studied. The variables analyzed were sex, birth weight, length, occipito-frontal circumference, gestational age, prematurity, delivery by caesarean section, umbilical cord length, placental weight, survival at 72 hours, primiparity, oligohydramnios, and polyhydramnios. Calculations included relative frequency, odds ratios (OR) and 95 % confidence intervals, the chi-square test, Fisher's p-value, and Student's t-test. RESULTS: SUA was found in 2.29 % of cases and in 1.03 % of controls (p = 0.0000001). These figures showed secular variation due to improvements in prenatal diagnosis and interruption of some pregnancies. When cases with and without SUA were compared, those with SUA had lower values of somatometry at birth, umbilical cord length and gestational age and had a higher risk for oligohydramnios, polyhydramnios, caesarean section, and death in the first 72 hours. Among controls, the only differences were a shorter umbilical cord and a higher frequency of oligohydramnios among infants with SUA. CONCLUSIONS: The results suggest that certain malformations associated with SUA could cause some of the differences among cases. Shortening of the umbilical cord and oligohydramnios could be related to SUA, as these abnormalities were found in both cases and controls. Comparison of cases and controls suggests that the etiopathogenesis of SUA could differ in the two groups.

Abnormalities, Multiple↗