-
[show abstract]
[hide abstract]
ABSTRACT: To examine population-based overall and malformation-specific trends in the prenatal diagnosis, pregnancy termination, and perinatal mortality for congenital heart disease (CHD) during a period of rapid progress in prenatal diagnosis and medical management of CHD and to explore the impact of prenatal diagnosis on early neonatal mortality for specific (isolated) cardiac malformations.
A total of 1982 cases of CHD, which were not associated with a known chromosomal anomaly, were obtained from the Paris Registry of Congenital Malformations. Main outcome measures were trends in the proportions diagnosed and terminated prior to birth, stillbirth and early (<1 day, one-week) neonatal mortality for (1) all cases; (2) all cases excluding isolated ventricular septal defects; and (3) malformation-specific trends for transposition of great arteries, hypoplastic left heart syndrome, coarctation of aorta, and tetralogy of Fallot. Analyses included cusum and binomial regression models for analysis of the trends during 1983-2000.
Prenatal diagnosis rates for CHD increased from 23.0% (95%CI: 19.0-27.4) in 1983-1988 to 47.3% (95%CI: 43.8-50.8) in 1995-2000. Termination rates increased between 1983 and 1989 (9.9%; 95%CI: 7.2-13.2) and 1989 and 1994 (14.7%; 95%CI: 12.3-17.4) but seemed to remain stable thereafter. Other than for hypoplastic left heart syndrome, pregnancy termination was exceptional for the other 3 specific malformations examined. Early neonatal mortality decreased to less than a third in the period 1995-2000 as compared with 1983-1989 (risk ratio, first week mortality: 0.31; 95%CI: 0.18-0.53). First week mortality was significantly lower for cases of transposition of great arteries diagnosed before birth (risk difference: 15.4%; 95% CI: 4.0-26.7).
Progress in clinical management, together with policies for increased access to prenatal diagnosis, has resulted in both a substantial increase in the prenatal diagnosis and considerable reductions in early neonatal mortality of CHD in the Parisian population.
Journal de Gynécologie Obstétrique et Biologie de la Reproduction 09/2006; 35(5 Pt 1):455-64. · 0.42 Impact Factor
-
[show abstract]
[hide abstract]
ABSTRACT: To assess overall and malformation-specific trends in the prevalence, prenatal diagnosis and pregnancy termination of congenital malformations in the Parisian population.
We used data from the Paris Registry of Congenital Malformations, which includes all births and pregnancy terminations with structural birth defects or chromosomal anomalies. Data on total and live birth prevalence were available for the period 1981-2000 and for prenatal diagnosis and pregnancy terminations for 1983-2000. Twenty malformations were selected for malformation-specific analyses due to their higher frequency and consistent definitions in different classification systems.
For the period 1981-2000, the overall total prevalence of malformations was 3.2%. Prenatal diagnosis rates consistently increased from 16.2% (95% CI, 13.8-18.6) of cases with malformation in 1983 to 69.1% (95% CI, 66.7-71.5) in 2000. Pregnancy terminations increased from 8.8% (95% CI, 7.0-10.8) of cases with malformation in 1983 to 30.1% (95% CI, 27.7-32.6) in 2000.
Registries of congenital malformations provide population-based data on the prevalence of malformations and their associations with other anomalies. Registry-based data can also be used to evaluate the impact of prenatal testing policies for congenital malformations.
Journal de Gynécologie Obstétrique et Biologie de la Reproduction 03/2005; 34(1 Pt 1):8-16. · 0.42 Impact Factor
-
J Goujard
[show abstract]
[hide abstract]
ABSTRACT: To evaluate the impact of prenatal screening for Down Syndrome in France in relation to the important changes in the French prenatal screening policy during the years 1996-1997: measurement of nuchal translucency and maternal serum screening, for all women.
Data are those published from three congenital anomalies registries implanted in Centre-Est, Paris and Bas-Rhin areas, from 1990 to 2001.
The livebirth prevalence of Down Syndrome decreased from 1/950 in 1990 to 1/1500 in 2000-2001. This decrease was observed from 1994 onwards but has proved stronger since 1996, in spite of the observed increase in the total prevalence partly explained by changes in the maternal age distribution. Extrapolated to all births in France, the calculated annual number of Down Syndrome births would decrease from 800-900 in 1990 to 500-600 in 2001. Consequently, the proportion of induced terminations following prenatal diagnosis increased from 38.6% in 1990 to 75.5% in 2001. In addition, the mean gestational age of the induced terminations is decreasing.
It seems reasonable to assume that the observed trends followed the changes in the French national policy for prenatal screening: a mass screening for all women, whatever the maternal age.
Gynécologie Obstétrique & Fertilité 07/2004; 32(6):496-501. · 0.52 Impact Factor
-
[show abstract]
[hide abstract]
ABSTRACT: To assess mothers' knowledge of screening tests for trisomy 21.
Interview of all women who had recently delivered a healthy child and were present in 15 Paris maternity units during one of the two non-consecutive days in June 1999 (N = 734).
Two-third said that they had access to a nuchal translucency measurement (NTM) and to maternal serum screening (MSS), and 16% to amniocentesis. Thirty-eight percent of the women who had NTMs and 69% of those who had serum screening said that they had been informed of the need for amniocentesis if the results were abnormal. Among the women who had amniocentesis, 20% did not know the risk of miscarriage and 41% had not been informed about the possibility of terminating the pregnancy if trisomy 21 was diagnosed.
Mothers' knowledge about the screening tests for trisomy 21 remains fragmentary. Providing comprehensive information about all these tests should be considered in early pregnancy so that women can make informed choices.
European Journal of Obstetrics & Gynecology and Reproductive Biology 09/2002; 104(1):14-20. · 1.97 Impact Factor
-
[show abstract]
[hide abstract]
ABSTRACT: To study the medical care, staff attitudes and patients' satisfaction from the decision to the post-intervention medical visit for termination of pregnancy for fetal abnormalies.
All patients and their spouses having a termination of pregnancy at the "Unite de Medecine Foetale" in Port-Royal Hospital between November 1996 and July 1997 were contacted for the study. A self-administered questionnaire was mailed six to eight weeks after intervention. Forty seven women and 42 men returned a completed questionnaire, the response rates were respectively 68% and 61%.
The patients and their spouses rated globally very high their satisfaction about the care received. The delay before intervention, the length and pain of labour were rated less positively. The factors associated with satisfaction were the quality of the relationship with the staff, and of information. Positive feelings about delivery were linked with the consideration and relief of pain. Most respondents mentioned that their physical and psychological state has improved at the moment of the survey but the psychological distress subsisted or has increased in one fourth of the cases. On the whole the answers made within the couples were correlated.
The positive results should be moderated by the number of non-respondents. In a context of very high rates of satisfaction, psychological distress is still present for one respondent out of four, six to eight weeks after termination of pregnancy for fetal abnormalies.
Gynécologie Obstétrique & Fertilité 06/2001; 29(5):358-70. · 0.52 Impact Factor
-
[show abstract]
[hide abstract]
ABSTRACT: This study investigated the role of maternal exposures at work during pregnancy in the occurrence of oral clefts.
The occupational exposures of 851 women (100 mothers of babies with oral clefts and 751 mothers of healthy referents) who worked during the first trimester of pregnancy were studied. All the women were part of a multicenter European case-referent study conducted using 6 congenital malformation registers between 1989 and 1992. In each center, the mother's occupational history, obtained from an interview, was reviewed by industrial hygienists who were blinded to the subject's status and who assessed the presence of chemicals and the probability of exposure. Odds ratios (OR) were estimated by a multivariate analysis including maternal occupation or occupational exposures during the first trimester of pregnancy and possible confounding factors such as center of recruitment, maternal age, urbanization, socioeconomic status, and country of origin.
After adjustment for confounding factors, cleft palate only was significantly associated with maternal occupation in services such as hairdressing [OR 5.1, 95% confidence interval (95% CI) 1.0-26.0] and housekeeping (OR 2.8, 95% CI 1.1-7.2). The analysis suggests that the following occupational exposures are associated with orofacial clefts: aliphatic aldehydes (OR 2.1, 95% CI 0.8-5.9) and glycol ethers (OR 1.7, 95% CI 0.9-3.3) for cleft lip with or without cleft palate and lead compounds (OR 4.0, 95% CI 1.3-12.2), biocides (OR 2.5, 95% CI 1.0-6.0), antineoplastic drugs (OR 5.0, 95% CI 0.8-34.0), trichloroethylene (OR 6.7, 95% CI 0.9-49.7), and aliphatic acids (OR 6.0, 95% CI 1.5-22.8) for cleft palate only.
Due to the limited number of subjects, these results must be interpreted with caution. However, they point out some chemicals already known or suspected as reproductive toxins.
Scandinavian journal of work, environment & health 05/2000; 26(2):137-45. · 3.12 Impact Factor
-
[show abstract]
[hide abstract]
ABSTRACT: This study examined the relationship between maternal tobacco and alcohol consumption during the first trimester of pregnancy and oral clefts.
Data were derived from a European multicenter case-control study including 161 infants with oral clefts and 1134 control infants.
Multivariate analyses showed an increased risk of cleft lip with or without cleft palate associated with smoking (odds ratio [OR] = 1.79, 95% confidence interval [CI] = 1.07, 3.04) and an increased risk of cleft palate associated with alcohol consumption (OR = 2.28, 95% CI = 1.02, 5.09). The former risk increased with the number of cigarettes smoked.
This study provides further evidence of the possible role of prevalent environmental exposures such as tobacco and alcohol in the etiology of oral clefts.
American Journal of Public Health 04/2000; 90(3):415-9. · 3.93 Impact Factor
-
[show abstract]
[hide abstract]
ABSTRACT: The results and limitations of current French practices for prenatal diagnosis (PND) of trisomy 21 were examined, using population-based data from the Paris Registry for 1992-97 (219 000 births). Of 670 cases of trisomy 21 reported, 71.0 per cent were terminations of pregnancy (TOP). The PND rate among mothers 38 years and older, all of whom were offered amniocentesis, was 89.9 per cent. Nearly all affected births in this age class followed maternal decisions, either to refuse amniocentesis or continue the affected pregnancy. In younger mothers, the overall French prenatal screening policy (three ultrasound examinations plus serum screening from January 1997) led to an overall PND rate of 67.3 per cent; it reached 78.8 per cent in 1997. Ultrasound accounted for 73.4 per cent of diagnosed cases. Increased detection by nuchal translucency measurement is clearly visible from 1996 onward. The birth prevalence, 8.7 per 10 000 births, diminished only slightly over the study period. The increase observed in the total number of cases in 1996 and 1997, concomitant with PND practice trends, may be due primarily to earlier TOP, which precedes miscarriages that would otherwise have occurred without being recorded. Future trends in prevalence among births must be observed carefully.
Prenatal Diagnosis 01/2000; 19(12):1113-8. · 2.11 Impact Factor
-
[show abstract]
[hide abstract]
ABSTRACT: A drug utilization study was performed using data of the OECM study on Occupational Exposures and Congenital Malformations, which was conducted in six European Registries of Congenital Anomalies (two in France, two in Italy, one in Great Britain, and one in The Netherlands): the mothers were interviewed after delivery for exposures during pregnancy, including use of therapeutic drugs. The analysis of drug use considered only the 1134 control mothers of healthy newborns, and focused on the first trimester of pregnancy: 36.2% of the interviewed mothers used at least one drug (excluding vitamins and minerals) during the first trimester. This rate varied from 22.5% in Glasgow to 50.3% and 44.2% in the French centers. Anti-infectives were the most frequent drugs (12.3% of mothers), then antinauseants (10.6%), and treatments for threatened abortion (5.5%). Important variations between countries were observed, reflecting different medical attitudes towards drug use during pregnancy.
Journal of Clinical Epidemiology 11/1999; 52(10):977-82. · 4.27 Impact Factor
-
J Goujard
[show abstract]
[hide abstract]
ABSTRACT: Published reports of clusters of congenital anomalies, traditionally defined as an aggregation in time and/or space of malformed cases, are interesting to analyse in terms of emergence, management and initiator of a public health decision. Through some examples of clusters for which a suspected source has been suggested, for those having identified important causal relationship or for those where the cluster appeared without any explanation, the paper shows the different steps that were taken after the initial 'alarm' and the time spent between the alarm and a final conclusion. If basic keys such as accurate field investigation, reliable estimation of the expected number and etiological evaluation, are the rule, the handling should remain flexible to take into account the particularity of each cluster. Among the clusters of congenital anomalies published in the last 20 years, very few of them were clearly explained.
European Journal of Epidemiology 10/1999; 15(9):853-62. · 4.71 Impact Factor
-
[show abstract]
[hide abstract]
ABSTRACT: Prenatal events are thought to play an important role in long-term handicap, but the specific role of perinatal factors remains controversial. Our study, conducted in the context of this debate, aimed to break down the various components of perinatal management and to assess the relationship between these components and survival without disability at the age of two years.
A prospective geographically-defined study was conducted in 1985 in the Paris metropolitan area. It covered 53430 births (stillbirths and live births), including 539 that occurred between 25 and 32 weeks gestation. The relationship between perinatal management and survival without disability was studied by a multivariate analysis (logistic regression). The analysis was restricted to a group of 202 infants born at 31 or 32 week's gestation, to avoid indication bias.
An inborn status (delivery in a tertiary care facility) exerted a protective effect on survival without disability at the age of two years (Adjusted Odds Ratio (OR)=7.51 [1.51; 37.4]), even though the area we studied possessed an excellent Medical Neonatal Transport Service. Multiple pregnancies also seemed to have a protective effect (Adjusted OR=2.45 [0.96; 6.27]). No statistically significant association was seen between survival without disability at two years and the presence of a hospital staff paediatrician in the delivery room.
These results lead us to consider what the concept of inborn/outborn represents in the perinatal management of infants at high risk.
European Journal of Obstetrics & Gynecology and Reproductive Biology 08/1998; 79(1):13-8. · 1.97 Impact Factor
-
[show abstract]
[hide abstract]
ABSTRACT: Recently, professionals in France have noticed an increase in newborns with Down syndrome (DS) being placed for adoption. The aim of this study was to investigate DS babies given up at birth for adoption and to consider the possible determinants of this in order to assess social acceptance of DS. A retrospective cohort of all living DS babies was collected from two birth-defect registries (Paris: 1981-1990; Marseilles area: 1984-1990). Follow-up data were collected: characteristics of the baby, biological parents and maternity units, age when given up for adoption, and type of foster care. The results showed that 19.4 per cent of infants with DS (115/593) were rejected by their parents. Multiple regression analysis indicated that foreign origin of the mother, area of residence, no associated major malformation, maternal age (15-24 years), and birth rank (> 2) variables were significantly associated with a lower placement rate. Among the 115 abandoned infants with DS, 88 came from unknown parentage (76.5 per cent). For half of them, adoptive placement (88/115) occurred before the age of 6 months. Socio-cultural attitudes play a great part in these family decisions. Equally important is the manner in which professionals propose adoption as an alternative to these parents of DS babies. They should be encouraged to consider all options before making a decision, so that the best solution can be found for the interest of all.
Prenatal Diagnosis 06/1998; 18(5):437-45. · 2.11 Impact Factor
-
S Cordier,
A Bergeret, J Goujard,
M C Ha,
S Aymé,
F Bianchi,
E Calzolari,
H E De Walle,
R Knill-Jones,
S Candela,
I Dale,
B Dananché,
C de Vigan,
J Fevotte,
G Kiel,
L Mandereau
[show abstract]
[hide abstract]
ABSTRACT: Glycol ethers are found in a wide range of domestic and industrial products, many of which are used in women's work environments. Motivated by concern about their potential reproductive toxicity, we have evaluated the risk of congenital malformations related to glycol ether exposure during pregnancy as part of a multicenter case-control study, conducted in six regions in Europe. The study comprised 984 cases of major congenital malformations and 1,134 controls matched for place and date of birth. Interviews of the mothers provided information about occupation during pregnancy, sociodemographic variables, and other potential risk factors (medical history, tobacco, alcohol, drugs). A chemist specializing in glycol ethers evaluated exposure during pregnancy, using the job description given by the mother, without knowledge of case or control status. We classified malformations into 22 subgroups. The overall odds ratio (OR) of congenital malformation associated with glycol ether exposure was 1.44 [95% confidence interval (CI) = 1.10-1.90], after adjustment for several potential confounders. The association with exposure to glycol ethers appeared particularly strong in three subgroups: neural tube defects (OR = 1.94; 95% CI = 1.16-3.24), multiple anomalies (OR = 2.00; 95% CI = 1.24-3.23), and cleft lip (OR = 2.03; 95% CI = 1.11-3.73). In this last subgroup, risk, especially of an isolated defect, tended to increase with level of exposure.
Epidemiology 08/1997; 8(4):355-63. · 5.57 Impact Factor
-
[show abstract]
[hide abstract]
ABSTRACT: Prenatal diagnosis (PND) is very developed in France, especially in the area of ultrasound (US) screening. The activity is regulated by law, and laboratories have to be authorized to perform any type of prenatal biological test if the purpose is to diagnose fetal defects. There are 70 cytogenetics laboratories and 50 biochemistry laboratories performing serum marker screening, about half of them being private. PND of chromosomal anomalies is offered to women over 37 years of age, to women who already had a child with a chromosomal anomaly, in case of abnormal US findings, if one of the parents has a balanced chromosomal anomaly and if the risk of chromosomal anomaly is higher than 1:250 according to the serum markers. Half of the trisomy 21 cases are now detected prenatally and pregnancies terminated. Fetal cell sampling is performed by amniocentesis in 70% of cases, by chorionic villus sampling in 7% of cases and by fetal blood sampling in 23% of cases. There are no professional guidelines and no quality assessment networks for any of the techniques in use. PND is regulated by two major laws: the Law on Abortion (1975) and the Law on Bioethics (1994).
European Journal of HumanGenetics 02/1997; 5 Suppl 1:26-31. · 4.40 Impact Factor
-
[show abstract]
[hide abstract]
ABSTRACT: Several thousands of compounds with a potential reproductive toxicity have been identified in animals, some of them are teratogens. In humans, only a small number of chemicals, administered as drugs, present in the diet, or in the occupational environment are recognized human teratogens. In parallel, about 60% of congenital anomalies have no identified cause and most probably some compounds present in the environment may contribute to certain anomalies. This paper presents a review of published epidemiological studies on the association between occupational exposures and congenital anomalies, focusing more particularly on some groups of compounds or some occupations such as: anaesthetic gases, laboratory work, solvents, pesticides and lead.
Revue d Épidémiologie et de Santé Publique 02/1994; 42(2):144-59. · 0.78 Impact Factor
-
J Goujard
Revue d Épidémiologie et de Santé Publique 02/1993; 41(6):513-4. · 0.78 Impact Factor
-
[show abstract]
[hide abstract]
ABSTRACT: A case-referent study was conducted to assess the risk of congenital malformations in relation to maternal occupational exposure before and during pregnancy. Three hundred and twenty-five cases of major malformations and 325 normal (at birth) referents identified in 15 maternity hospitals were included in the study. The occupational history obtained from an interview of the mother was blindly reviewed by an industrial hygienist who assessed the presence of chemical exposure and the probability of exposure. The results suggested that mothers of the case children with oral clefts were more often exposed to solvents during pregnancy [odds ratio (OR) 7.9, 90% confidence interval (90% CI) 1.8-44.9] and worked more often as cleaners (four cases, no referents). Digestive anomalies (OR 11.9, 90% CI 2.0-149) and multiple anomalies (OR 4.5, 90% CI 1.4-16.9) were also associated with occupational exposure to solvents at work. These results were not modified when differences in maternal age, area of residence, and socioeconomic status were taken into account.
Scandinavian journal of work, environment & health 03/1992; 18(1):11-7. · 3.12 Impact Factor
-
Revue d Épidémiologie et de Santé Publique 02/1992; 40(5):369-72. · 0.78 Impact Factor
-
[show abstract]
[hide abstract]
ABSTRACT: Because the delivery of growth hormone (GH) was centralized from 1977 in France, it has been possible to conduct, during the second half of 1990, a nationwide survey of the health status of patients treated with GH from the year 1959. A questionnaire regarding the 5,546 patients recorded for the period 1959-1990 was sent to the prescribers or the patients. 5,418 more or less completely documented reports were obtained. The mean age of the patients at the onset of GH treatment was 11.0 +/- 4.1 years. 1,937 of them had at this time some important disease associated with GH deficiency. The mean duration of treatment was 3.99 +/- 3.05 years. 3,446 patients were still under follow-up. Very recent information (1990-1991) was given for 82.7% of patients, less recent data (1985-1989) for 13.4%. For 3.9%, no data beyond 1985 were obtained. 77 patients had died, 38 from neoplastic disease (mainly recurrence of a primary malignancy), 10 from accident, 3 by suicide, 7 with neurological disease [only 1 case of Creutzfeldt-Jakob disease (CJD) was reported at the time of the survey], the others from various causes. No abnormal frequency of posttreatment leukemia, lymphoma, malignancies, hip diseases, glucose intolerance or other disease focusing attention, was found in the survey. From the time when this survey was completed (December 1990) to that of this report (May 1992), other cases of CJD have been reported in France: 3 ascertained, 7 clinically resembling but not yet certain. These 10 patients were treated for complete GH deficiency, 6 of congenital or neonatal cause and 4 after neurosurgery.(ABSTRACT TRUNCATED AT 250 WORDS)
Hormone Research 02/1992; 38 Suppl 1:35-43. · 2.48 Impact Factor
-
[show abstract]
[hide abstract]
ABSTRACT: Improvements in combining obstetrics and neonatology led to a trend to intervene earlier in premature babies born before the 33rd week of gestational age. The enquiry that was carried out in 1985 in the Paris geographical region had as its objective to assess on the one part how many premature deliveries occurred between the 25th and 33rd week of amenorrhea and on the other hand what happened in the short term to the infants born from these pregnancies, i.e., their mortality; and for those who survived, their quality of life. The study was carried out on a representative sample of the deliveries in 1985 in the four departments of the Paris region--Paris and the three departments of the Petite Couronne--where half of all deliveries were assessed. The enquiry covered 53,430 deliveries for which the overall prematurity rate was 4.5%, and those deliveries that occurred before 33 weeks of gestational age constituted 1.0% (539 babies). Twenty children were lost for follow-up after a year. This was 4.9% of the live births and 6.3% of the live children who left the neonatology centres. At 2 years of age, the numbers that were not followed up were eventually 24, which was 5.8% of live births and 7.6% of those that left the centres of neonatology. The results show a very high rate of antepartum mortality but also of mortality during and after labour. Only 379 infants out of the 539 (70%) were transferred into special care baby units. By 1 year of age, the survivors were 57% of the total number of deliveries and 75% of the live births and 82% of those transferred to the special units. As far as concerned those that were live born, the survival rate at the age of 1 year varied considerably according to the duration of the pregnancy. The number of those that survived a pregnancy of less than 27 weeks was low (31%). It was, at 28 weeks, 53%. This is the age where births have to be registered. It reached 87% of the live births that occurred at 32 weeks. One has to point out that there is no statistically significant difference between 27 and 28 weeks of gestation. Whereas there is a significant difference (p less than 0.05) with those delivered at 29 weeks (75%). 80% of those 291 infants that were examined at 1 year of age were considered to be normal as far as psychomotor and sensorial behaviour was concerned.(ABSTRACT TRUNCATED AT 400 WORDS)
Journal de Gynécologie Obstétrique et Biologie de la Reproduction 02/1990; 19(1):25-35. · 0.42 Impact Factor
