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PubMed1990

Hypoplastic left heart syndrome: natural history in a geographically defined population.

Advances in surgical treatment of hypoplastic left heart syndrome with the Norwood procedure and cardiac transplantation have made essential the understanding of the natural history of hypoplastic left heart syndrome. In a geographically defined population, we ascertained the prevalence of hypoplastic left heart syndrome in children born in Oregon from 1971 through 1986. Clinical and anatomic data were extracted from the charts of the 98 affected children and the survival rate was calculated. Hypoplastic left heart syndrome occurred in 0.162 per 1000 live births in Oregon during this period. No syndrome complex was prevalent and 84% were free of other congenital malformations. However, there was an increased occurrence of congenital heart defects in first-degree relatives of probands with hypoplastic left heart syndrome. Of the affected children 15 +/- 4% died on the first day of life, 70 +/- 5% died within the first week, and 91 +/- 3% died within 30 days. No secular change in survival occurred during the study. Palliation with the Norwood procedure was performed in 20 children. Although survival was significantly improved with this surgery (P = .01), the effect was observed principally through 30 days of life and only one of these children remains alive. Hypoplastic left heart syndrome is a lethal congenital heart defect in children and poses management and ethical dilemmas.

Abnormalities, Multiple↗
PubMed1992

Frontal plagiocephaly: synostotic, compensational, or deformational.

Plagiocephaly is a term commonly used to describe congenital forehead asymmetry. Sixty patients with frontal plagiocephaly were evaluated retrospectively and separated into three types: synostotic (N = 24), compensational (N = 3), and deformational (N = 33). Categorization of frontal plagiocephaly as synostotic or deformational was reliably made by physical examination, focusing on the supraorbital rims, nasal root, ears, and malar eminences. Other anatomic parameters useful in the differential diagnosis included chin point, palpebral fissures, and facial height. This study documented that birth histories were similar for synostotic and deformational frontal plagiocephalic infants. However, other deformational anomalies were more common in deformational frontal plagiocephalic infants, whereas malformations had an equal incidence in deformational and synostotic frontal plagiocephalic infants. Torticollis was an associated finding in 64 percent of infants with deformational frontal plagiocephaly; almost all were ipsilateral. In contrast, head tilt, usually to the contralateral side, was noted in 14 percent of patients with synostotic frontal plagiocephaly. Female preponderance was noted in both synostotic (79 percent) and deformational (76 percent) frontal plagiocephaly. Left-sided involvement was seen in 73 percent of patients with deformational frontal plagiocephaly and in 46 percent of patients with synostotic frontal plagiocephaly. Premature pelvic descent, in the left occipital anterior position, may account for the high incidence of left-sided deformational plagiocephaly and ipsilateral torticollis.

Abnormalities, Multiple↗
PubMed1987

Trends in tracheoesophageal fistula.

The changing trends in the management of patients with esophageal atresia, with or without tracheoesophageal fistula during 1974 to 1983, and its effect on survival and morbidity rates are analyzed herein. Of 139 patients in this series, 44 per cent were in Waterston group C. The over-all mortality rate was 25.5 per cent but this decreased consistently after 1980 to 11.0 per cent in 1983. The incidence of infants in the series who weighed less than 1.8 kilograms at birth increased from 7 per cent before 1979 to 18 per cent after 1979 and the survival rate increased from zero to 50 per cent. All six infants who were 30 to 34 weeks gestation and referred after 1979 survived. Severe pneumonia at admission decreased from 24 to 11 per cent in two five year periods. Delayed primary anastomosis and Livaditis circular myotomy overcame many problems of the "long gap" associated with pure esophageal atresia, resulting in successful anastomosis in all eight patients treated in the latter years. Of six patients who presented before 1979, three patients died and three required interposition grafts. Significant anastomotic leaks decreased from 14.7 to 3.2 per cent and the mortality rate decreased from 70 to zero per cent, reflecting many factors, such as extrapleural repair, use of total parenteral nutrition and improved neonatal, ventilatory and intensive care.

Abnormalities, Multiple↗
PubMed1992

Prediction of low birth weight outcome in Thailand: multiple regression analysis.

In the present study the risk approach using maternal risk factors for LBW newborns in Thailand is applied. The study may be considered as a managerial tool for developing local strategies and is particularly useful in maternal and child health care. The main objective is to develop a model to predict LBW outcome with an aim to screen those at risk at the level of health centres, district and provincial hospitals. Using MLR analysis, it is concluded that Model I and Model II (Table 3 and 4) can be used to predict which pregnant women are likely to deliver LBW newborns. The prediction can be made as early as the first trimester. If the prediction is shown to be accurate, the two models will be very helpful for use in Thai people. For general use, the investigators believe that Model I will be appropriate for application at the level of provincial and district hospitals, while Model II should be used at the health centre level peripheral to the health delivery system.

Case-Control Studies↗
PubMed1976

Diaphragmatic hernia in the south-west of England.

A retrospective anatomical, family, and epidemiological study was made of 143 patients (81 female and 62 male) with diaphragmatic hernia who were born in the south-west of England between 1943 and 1974. Thirty-nine cases were stillborn. Seventy-five per cent of patients had a left-sided diaphragmatic defect, 22% had a right-sided defect, and 3% had a bilateral defect. Fifty per cent of the patients had other congenital malformations, most frequently of the nervous system. No maternal age or birth order effect was noted. Cases of diaphragmatic hernia without other malformations had in general a normal fetal growth rate. Eight per cent of the cases were illegitimate. There were two pairs of twins discordant for diaphragmatic hernia, one pair being dizygotic and the other monozygotic. In no case of diaphragmatic hernia was there a relative affected with a diaphragmatic hernia. The most common type of diaphragmatic defect was a posterolateral hernia (92%), followed in frequency by an eventration of the diaphragm (5%), the least common defect being a retrocostosternal hernia (2%). Diaphragmatic hernia appears to be aetiologically as well as anatomically heterogeneous. In this series there were two cases of trisomy 18, one case of trisomy 21, one case trisomic for a small part of chromosome 20, and two cases with the Pierre Robin syndrome. It seems likely that diaphragmatic hernia is a non-specific consequence of several teratological processes.

Abnormalities, Multiple↗
PubMed1999

[Esophageal atresia and associated malformations].

BACKGROUND: Survival of newborns with esophageal atresia and tracheoesophageal fistula has increased in the last years. AIM: To assess the prevalence of esophageal atresia and describe associated malformations in Chilean newborns. MATERIAL AND METHODS: All births occurring between January 1983 and June 1998 were studied. All malformed children were registered and the next non malformed born child was considered as control. RESULTS: During the study period, 50,965 births occurred and 3,336 malformed children were born. Eighteen (3 stillborn) had esophageal atresia with a rate of 3,53 per 10,000 born alive. Overall survival was 73%. Survival among children classified in Waterson groups A and B was 100% and 50% among those classified in group C. Seventy two percent had associated malformations, being congenital cardiopathies and skeletal malformations the most frequent. VACTERL association was found in 44% of children. All stillborn children had other severe malformations. When compared to controls, malformed children had a lower weight, a lower gestational age, their mothers had a higher age, a higher frequency of relatives with malformations and a higher frequency of maternal diseases during the first trimester of pregnancy. CONCLUSIONS: The rate of esophageal atresia found in this study is similar to that reported in other Chilean obstetrical units as part of the Latin American Study of Congenital Malformations (ECLAMC).

Abnormalities, Multiple↗
PubMed2003

The epidemiology of cardiovascular defects, part I: a study based on data from three large registries of congenital malformations.

To analyze complex and noncomplex cardiac malformations regarding prevalence and in relation to demographic variables, we pooled data on infants (age 1 year or younger) with congenital cardiovascular defects from three large birth defect registries in California, Sweden, and France. Altogether, 12,932 infants had one or more congenital heart defects out of 4.4 million live births and stillbirths. The registries in Sweden and France obtained data through reporting from various sources; in California, medical records were reviewed. As expected, definitions and ascertained conditions differed among each of the registries. The total rates for severe defects were similar (1.43 per 1,000), but differed for specific defects. Clear differences in epidemiological characteristics existed for specific defects; for example, severe cardiac defects sex ratios were significantly high for hypoplastic left heart syndrome, d-transposition of great vessels, double outlet right ventricle, total anoralous pulmonary venous return, tetralogy of Fallot, and significantly low for pulmonary atresia without ventricular septal defect and endocardial cushion defect. Few defects were similar for several epidemiological characteristics, but, for example, the combination of ventricular and atrial septal defects appeared equivalent with endocardial cushion defect under some circumstances, yet behaved differently with regard to associated noncardiovascular defects.

Age Factors↗
PubMed2002

Feasibility of thoracoscopic repair of esophageal atresia with distal fistula.

BACKGROUND/PURPOSE: Evaluation of the feasibility of thoracoscopic correction of esophageal atresia with distal fistula. METHODS: Eight consecutive neonates with esophageal atresia and distal fistula were treated thoracoscopically. Mean birth weight was 3,048 g (range, 2,140 to 3,770). The patients were intubated endotracheally and placed in a 3/4 left prone position. Three cannulae were inserted along the inferior tip of the scapula. CO(2) was insufflated at a pressure of 5mm Hg and a flow of 0.5 L/min. The fistula was either clipped or ligated. The proximal esophagus was opened and an anastomosis was made over a 6F or 8F nasogastric tube with interrupted 5-0 Vicryl. RESULTS: All procedures were completed thoracoscopically without major peroperative complications. The mean operating time was 198 minutes (range, 138 to 250). One patient had a major leak, resulting in a stormy postoperative course, but the leak healed on conservative treatment. This patient and 3 others had stenosis requiring dilatation, respectively, 3, 6, 12, and 1 times. The babies were fed after a median period of 8 days. The median hospital stay was 13 days. CONCLUSIONS: Thoracoscopic repair of esophageal atresia with distal fistula is feasible. Larger series are needed to determine the exact place of the thoracoscopic approach.

Abnormalities, Multiple↗
PubMed1987

Tracheoesophageal anomalies in Waterston C neonates: a 30-year perspective.

Two hundred and seventy-eight infants with congenital tracheoesophageal anomalies have been managed at the Columbus Children's Hospital since 1955. Fifty-four (20%) have been classified as high risk using Waterston's criteria of birth weight, associated anomalies, and pneumonitis. The infants have been divided into group I (n = 27, 1955 to 1969) and group II (n = 27, 1970 to 1984) to reflect the onset of mechanical ventilation and effective neonatal intensive care. The mean birth weight of infants in group II was significantly less than in group I (1,753 +/- 390 g v 1,950 +/- 505 g, P less than .05). The incidence of prematurity, measured by gestational age, has significantly increased with 9 of 27 (33%) infants in group II and 2 of 27 (7%) infants in group I less than 32 weeks gestation (P less than .05). The presence or severity of associated anomalies was not significantly different in groups I and II. Twenty patients in group I and 22 patients in group II underwent definitive management of their tracheoesophageal anomaly. Operative survival was 30% in group I and 74% in group II (P less than .05). Long-term survival was 15% in group I and 64% in group II (P less than .05). Early postoperative complications included aspiration pneumonitis, anastomotic leak, or stricture. The most frequent complications in long-term follow-up were recurrent pneumonias, which were observed in 9 of 16 children. Esophageal dysmotility or gastroesophageal reflux was documented in five of these children; four were managed successfully with positional or pharmacologic manipulations while one required an antireflux procedure. Thirty percent (5/16) are asymptomatic.(ABSTRACT TRUNCATED AT 250 WORDS)

Abnormalities, Multiple↗
PubMed2002

Gastroschisis: a 15-year, single-center experience.

70 cases of gastroschisis (GS) were surgically treated at the Pediatric Surgical University Clinic, Münster, from 1984 through 1998. The defect occurred more frequently in males (44) than females (26). The average birth weight was 2,383 g and mean gestational age 36.8 weeks. 9 infants (12.9%) were delivered vaginally and the rest (87.1%) by cesarean section; 34 of the 61 (55.7%) cesarean sections were done solely for prenatal ultrasonic identification of the abdominal-wall defect. 10 infants (14.3%) underwent primary closure; in 19 (27.1%) primary closure of the skin was possible, however, a single solvent-dried dura (SDD) graft was required for fascial enlargement. The remaining 41 infants (58.6%) had extensive defects and required two grafts for optimal closure. 22 patients (31.4%) had associated anomalies, the most common being bowel atresias and undescended testis. 14 (20%) required secondary laparotomies because of bowel-associated complications and 1 (1.4%) for a urinary-bladder perforation. 11 patients (15.7%) had non-bowel-associated complications. The average postoperative tracheal intubation time was 3.9 days and the average hospital stay was 75.6 days. The overall mortality was 2.8%. No major complications associated with SDD implants were encountered; only 4 patients (5.7%) had minor complications such as local inflamation and infection and were managed conservatively. The present data support the employment of SDD implants as acceptable biomaterial for the repair of large GS defects.

Abnormalities, Multiple↗
PubMed1995

Twin study using mortality data: a new sampling method.

BACKGROUND: To evaluate the success of a novel approach to twin studies using death discordant twin pairs in a disease of low prevalence. METHOD: A population study based on all registered deaths at the Office of Population Censuses and Surveys of England and Wales, classified under the ICD code 335.2 (motor neuron disease [MND]) for the period 1979-1989 inclusive. From the above database of 10,872 people, individuals born after 31 December 1899 were traced in the Birth Indices for England & Wales to enable identification of possible twins. In all 131 twin pairs were found and the co-twin details were sent to the National Health Service Central Register (NHS-CR) to enable location of the relevant Family Health Services Authority and thence the co-twin's general practitioner (GP). A letter requesting access was sent to the family practitioners. If given, the co-twin can be approached and interviewed. RESULTS: The search produced: 54 living co-twins; 31 who died as adults; 29 infant deaths; 5 emigrated; 3 incorrectly diagnosed index twins; and 9 untraceable co-twins. Among the adult deaths two concordant pairs were identified. This has created the largest twin population sample worldwide for MND. CONCLUSION: This new twin study method is clearly viable, and has produced a large unbiased sample compared to that possible using traditional methods. It relies heavily on the accuracy of death certificates and zygosity reporting by living co-twins, but is possibly the only way of collecting twins in rare conditions.

Adult↗
PubMed1990

Predicting death from initial disease severity in very low birthweight infants: a method for comparing the performance of neonatal units.

OBJECTIVES: To investigate (a) which clinical variables and physiological measures of disease severity best predict death in very low birthweight infants and (b) their use in comparing mortality between two neonatal units. DESIGN: Retrospective study of two cohorts of very low birthweight infants from overlapping time periods who received mechanical ventilation. SETTING: Two neonatal intensive care units (hospitals A and B). SUBJECTS: 262 Very low birthweight infants, 130 in hospital A, 132 in hospital B. MAIN OUTCOME MEASURE: Death in hospital. RESULTS: In hospital A the mean level of oxygenation in the first 12 hours of life, whether measured as inspired oxygen requirement (FIO2), arterial/alveolar oxygen (a/AO2) ratio, or alveolar-arterial oxygen difference (A-aDO2), was more closely associated with death than any of four "traditional" risk factors: low birth weight, short gestation, the diagnosis of respiratory distress syndrome, and male sex. Mean pH in the first 12 hours was as strongly associated with death as birth weight. Multiple logistic regression models were derived in infants from hospital A using the four traditional risk factors with measures of oxygenation and pH. The validity of each model was then tested in infants from hospital B. The model based on the four traditional risk factors alone predicted death in hospital B with only 31% sensitivity. Adding mean a/AO2 ratio and mean pH increased its sensitivity to 75%, and when mean a/AO2 ratio was replaced by mean FIO2 its sensitivity increased further to 81%. Based on crude mortality rates alone, the odds of death in hospital A versus hospital B were 0.67 (95% confidence interval 0.37 to 1.23). After correcting for traditional risk factors and mean FIO2 and mean pH, however, the odds of death in hospital A increased to 3.27 (1.35 to 7.92; p less than 0.01). This increased risk persisted after adjusting for the time difference between each cohort. CONCLUSIONS: Crude comparisons of hospital mortality can be highly misleading. Reliable assessment of neonatal outcome is impossible without correcting for major risk factors, particularly initial disease severity. International agreement on a minimum core dataset of clinical and physiological information could improve neonatal audit and help to identify effective treatments and policies.

Gestational Age↗
PubMed2001

[Delayed birth of the second child in multiple gestation].

Five case histories illustrate the issue of delayed interval deliveries. In the first two cases, the first child was born at a gestational age of 20 and 18 weeks, respectively. The first woman (40 years old) gave birth to the second child after successful prolongation of pregnancy to a gestational age of 38 weeks. In the second case (28 years old), the attempt to delay delivery failed and the second child was born at 19 weeks of gestation. The third case (32 years old), illustrates the enormous differences in neonatal course between a child born at 26 weeks of gestation, who had to be treated at length for respiratory distress syndrome, hypotension and patent ductus arteriosus, and his twin brother born two weeks later and who recovered more quickly. The fourth case (24 years old) describes delayed delivery to allow administration of antenatal glucocorticoids. The last case (32 years old) deals with a serious maternal complication of placental abruption during an attempt to delay the birth of the second twin. Early tocolytic and antibiotic therapy may delay delivery and, in combination with antenatal glucocorticoids to stimulate lung maturation, may thereby improve the condition of the second twin. The role of cervical cerclage remains controversial. There is an important publication bias in the literature due to under-reporting of the failed attempts of delayed deliveries. In multiple gestation with imminent very preterm birth, delayed delivery of the second child is a feasible management option.

Adult↗
PubMed1968

Intestinal atresia.

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Abnormalities, Multiple↗
PubMed1979

Esophageal atresia. A 41-year experience.

Since the first attempted repair in 1935, a total of 365 infants have undergone treatment for esophageal atresia with or without tracheoesophageal fistula. To evaluate the improvement in our results, we have divided our patients into four, approximately equal, time periods. The overall late survival has indeed improved during each of the four decades from between 36% and 53% to 68% in the last ten years. During this past decade, the group A risk infants (35 patients) had a 3% mortality, the group B (20 patients) had a 5% mortality, but the group C babies (22 patients) had a 41% mortality due to associated anomalies or severe prematurity. Anastomotic leaks and recurrent fistulas were each encountered in approximately 5% of the cases in all decades. Postoperative stricture has been encountered in 20% of the entire series and no esophageal replacements have been required.

Abnormalities, Multiple↗