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- Bell, Jane C., et al.
(författare)
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Survival of infants born with esophageal atresia among 24 international birth defects surveillance programs
- 2021
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Ingår i: Birth Defects Research. - : Wiley. - 2472-1727. ; 113:12, s. 945-957
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Tidskriftsartikel (refereegranskat)abstract
- Background: Esophageal atresia (EA) affects around 2.3–2.6 per 10,000 births world-wide. Infants born with this condition require surgical correction soon after birth. Most survival studies of infants with EA are locally or regionally based. We aimed to describe survival across multiple world regions. Methods: We included infants diagnosed with EA between 1980 and 2015 from 24 birth defects surveillance programs that are members of the International Clearinghouse for Birth Defects Surveillance and Research. We calculated survival as the proportion of liveborn infants alive at 1 month, 1- and 5-years, among all infants with EA, those with isolated EA, those with EA and additional anomalies or EA and a chromosomal anomaly or genetic syndrome. We also investigated trends in survival over the decades, 1980s–2010s. Results: We included 6,466 liveborn infants with EA. Survival was 89.4% (95% CI 88.1–90.5) at 1-month, 84.5% (95% CI 83.0–85.9) at 1-year and 82.7% (95% CI 81.2–84.2) at 5-years. One-month survival for infants with isolated EA (97.1%) was higher than for infants with additional anomalies (89.7%) or infants with chromosomal or genetic syndrome diagnoses (57.3%) with little change at 1- and 5-years. Survival at 1 month improved from the 1980s to the 2010s, by 6.5% for infants with isolated EA and by 21.5% for infants with EA and additional anomalies. Conclusions: Almost all infants with isolated EA survived to 5 years. Mortality was higher for infants with EA and an additional anomaly, including chromosomal or genetic syndromes. Survival improved from the 1980s, particularly for those with additional anomalies.
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- Li, Xinjun, et al.
(författare)
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Family and neighborhood socioeconomic inequality in cryptorchidism and hypospadias : A nationwide study from Sweden
- 2019
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Ingår i: Birth Defects Research. - : Wiley. - 2472-1727. ; 111:2, s. 78-87
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Tidskriftsartikel (refereegranskat)abstract
- Objectives: To examine whether there is an association between neighborhood deprivation and incidence of cryptorchidism and hypospadias, after accounting for family-level and individual-level sociodemographic characteristics. Methods: All boys born in Sweden between January 1, 2001 and December 31, 2010 were followed. Data were analyzed by multilevel logistic regression, with family-level and individual-level characteristics at the first level and level of neighborhood deprivation at the second level. Results: During the study period, among a total of 497,584 boys, 8,584 (1.7%) and 3,704 (0.7%) were diagnosed with cryptorchidism and hypospadias, respectively. Cumulative rates for cryptorchidism and hypospadias increased with increasing levels of neighborhood deprivation. In the study population, 1.5 per 100 and 2.0 per 100 boys, in the least and most deprived neighborhoods were diagnosed with cryptorchidism and 0.7 per 100 and 0.9 per 100 boys were diagnosed with hypospadias. Incidence of hospitalization for cryptorchidism and hypospadias increased with increasing neighborhood-level deprivation across all family-level and individual-level sociodemographic categories. The odds ratio (OR) for cryptorchidism and hypospadias for those living in high-deprivation neighborhoods versus those living in low-deprivation neighborhoods was 1.13 (95% confidence interval [CI] = 1.05–1.21) and 1.24 (95% CI = 1.12–1.37). High neighborhood deprivation remained significantly associated with higher odds of hypospadias after adjustment for family-level and individual-level sociodemographic characteristics (OR = 1.20, 95% CI = 1.08–1.35). Conclusions: This study is the largest so far on neighborhood influences on cryptorchidism and hypospadias. Our results suggest that neighborhood deprivation is associated with a moderate incidence of hypospadias independent of family-level and individual-level sociodemographic characteristics.
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- Mc Goldrick, Niall, et al.
(författare)
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A multi-program analysis of cleft lip with cleft palate prevalence and mortality using data from 22 International Clearinghouse for Birth Defects Surveillance and Research programs, 1974–2014
- 2023
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Ingår i: Birth Defects Research. - 2472-1727. ; 115:10, s. 980-997
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Tidskriftsartikel (refereegranskat)abstract
- Background: Cleft lip with cleft palate (CLP) is a congenital condition that affects both the oral cavity and the lips. This study estimated the prevalence and mortality of CLP using surveillance data collected from birth defect registries around the world. Methods: Data from 22 population- and hospital-based surveillance programs affiliated with the International Clearinghouse for Birth Defects Surveillance and Research (ICBDSR) in 18 countries on live births (LB), stillbirths (SB), and elective terminations of pregnancy for fetal anomaly (ETOPFA) for CLP from 1974 to 2014 were analyzed. Prevalence and survival (survival for LB only) estimates were calculated for total and subclassifications of CLP and by pregnancy outcome. Results: The pooled prevalence of total CLP cases was 6.4 CLP per 10,000 births. The prevalence of CLP and all of the pregnancy outcomes varied across programs. Higher ETOPFA rates were recorded in most European programs compared to programs in other continents. In programs reporting low ETOPFA rates or where there was no ascertainment of ETOPFA, the rate of CLP among LB and SB was higher compared to those where ETOPFA rates were ascertained. Overall survival for total CLP was 91%. For isolated CLP, the survival was 97.7%. CLP associated with multiple congenital anomalies had an overall survival of 77.1%, and for CLP associated with genetic/chromosomal syndromes, overall survival was 40.9%. Conclusions: Total CLP prevalence reported in this study is lower than estimates from prior studies, with variation by pregnancy outcomes between programs. Survival was lower when CLP was associated with other congenital anomalies or syndromes compared to isolated CLP.
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- Sabeti Rad, Zahra, et al.
(författare)
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Congenital malformations in offspring of women with a history of malignancy
- 2017
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Ingår i: Birth Defects Research. - : Wiley. - 2472-1727. ; 109:3, s. 224-233
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Tidskriftsartikel (refereegranskat)abstract
- BACKGROUND: Survival after malignancy has increased and the question of risks, including risk for congenital malformations for the offspring of these women has become important. Data on congenital malformations in such offspring are limited.METHODS: We compared congenital malformation in offspring, born 1994 to 2011 of women with a history of malignancy (at least 1 year before delivery) with all other offspring. Adjustment for confounders was mainly made by Mantel-Haenszel methodology. Data were obtained by linkage between Swedish national health registers.RESULTS: We identified 71,954 (4.1%) infants with congenital malformation, of which 47,081 (2.7%) were relatively severe (roughly corresponding to major malformation). Among 7284 infants to women with a history of malignancy 204 relatively severe malformations were found (2.8%; odds ratio [OR] = 1.04; 95% confidence interval [CI], 0.91-1.20). After in vitro fertilization, the risk of a relatively severe malformation was significantly increased in women without a history of malignancy (OR = 1.31; 95% CI, 1.24-1.38) and still more in women with such a history (risk ratio = 1.85; 95% CI, 1.08-2.97). However, there were no significant differences neither, for any malformations (OR = 1.04; 95% CI, 0.92-1.16) nor for relatively severe malformations (OR = 1.04; 95% CI, 0.91-1.20), when comparing offspring only after maternal history of malignancy.CONCLUSION: No general increase in malformation rate was found in infants born to women with a history of malignancy. A previously known increased risk after in vitro fertilization was verified and it is possible that this risk is further augmented among infants born of women with a history of malignancy. Birth Defects Research 109:224-233, 2017. © 2016 Wiley Periodicals, Inc.
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