Objective: Fetal Alcohol Spectrum Disorders (FASDs) are common, often undiagnosed, lifelong developmental disorders that result from prenatal alcohol exposure. FASD is present at birth and typically identified around ...Objective: Fetal Alcohol Spectrum Disorders (FASDs) are common, often undiagnosed, lifelong developmental disorders that result from prenatal alcohol exposure. FASD is present at birth and typically identified around seven years of age. The most severe outcome in cases of FASD is mortality. The purpose of this scoping review is to 1) use a systematic review to provide an estimated mortality proportion for children with FASD, and 2) update a study published in 2014 by reviewing published reports of mortality in individuals diagnosed with FASD. Method: A search of PubMed, CINAHL, and Google Scholar for reports published between 2013 and 2023 on mortality in individuals with FASD. Results: Three population-based studies have reported on all-cause mortality rates, finding a combined mortality rate of 10.9%, a 2.63 fold (95% CI: 2.61 to 2.65) increase in mortality risk over the general population. Since 2016, this review identified only eight new cases meeting the study inclusion criteria. The reported causes of death were five cases of pneumonia, and one case each of failure to thrive and dehydration, intestinal dilatation and asphyxiation caused by overeating due to pica, and acute gastric volvulus. Discussion: While current research suggests a diagnosis of FASD is associated with a 2.6-fold increase in mortality risk, this is likely an underestimation, as most cases of FASD-related mortality go unreported. Globally, about 1 new case is reported every 15 months. However, in the United States alone, between 1752 to 4400 FASD related deaths occur annually. Our review suggests that FASD is rarely identified as a causal or contributing factor in deaths of children and adolescents, resulting in a substantial undercount of FASD-related deaths. Increased attention to the role of FASD in infant and child mortality case reviews, child death review committee reports, and mortality reviews is needed.展开更多
目的通过成本-效用分析,评价孕早期行胎儿结构畸形超声筛查相比于孕中期行胎儿结构畸形超声筛查的经济性,为临床路径优化提供循证依据。方法从全社会角度出发,构建决策树模型,模拟孕妇于孕早期行胎儿结构畸形超声筛查(干预组)与于孕中...目的通过成本-效用分析,评价孕早期行胎儿结构畸形超声筛查相比于孕中期行胎儿结构畸形超声筛查的经济性,为临床路径优化提供循证依据。方法从全社会角度出发,构建决策树模型,模拟孕妇于孕早期行胎儿结构畸形超声筛查(干预组)与于孕中期行胎儿结构畸形超声筛查(对照组)2种不同产前筛查方案的临床选择及转归路径,评估2组不同路径下的成本-效用情况。模型模拟时长为18周,临床转移概率和成本参数来源于深圳市南山区妇幼保健院数据,效用数据来源于已公开发表的文献。结果指标包括成本、质量调整生命年(qualityadjusted life years,QALYs)和增量成本-效果比(incremental cost-effectiveness ratio,ICER)。通过敏感性分析评估模型结果的稳健性。结果相比对照组,干预组可减少不必要的妊娠发生,以更少的总成本投入获得更多的QALYs,ICER为−161732.68元/QALY,具有绝对成本效果优势。概率敏感性分析表明基础分析结果具有稳健性,干预组具有成本效果的概率为94.60%。结论在全社会角度下,于孕早期进行胎儿结构畸形筛查相比于孕中期具有绝对的经济学优势。展开更多
Background: Women awaiting fetal echocardiography (fECHO) report high anxiety. It is unclear if anxiety decreases after performance of fECHO. Methods: At fECHO, subjects’ current (state) vs baseline (trait) anxiety w...Background: Women awaiting fetal echocardiography (fECHO) report high anxiety. It is unclear if anxiety decreases after performance of fECHO. Methods: At fECHO, subjects’ current (state) vs baseline (trait) anxiety was assessed using the Spielberger State-Trait Anxiety Inventory. Anxiety scores of the pre- and post-fECHO groups were compared. Results: From January 2007 to January 2009, we recruited 84 subjects: 40 pre-fECHO and 44 post-fECHO. Of the post-fECHO group, 30 had normal fetal cardiac structure and function confirmed, 12 were told of an abnormality, and 2 were told to follow up equivocal results. Anxiety scores were compared between the 40 pre-fECHO subjects and the 30 post-fECHO subjects with normal results. The mean state anxiety score of the pre-fECHO group was higher than that of the post-fECHO group (42.1 ± 15.1 vs 30.8 ± 8.5, p < 0.001);there was no difference in trait scores. Neither state nor trait anxiety was associated with maternal age, parity, history of miscarriage or known fetal anomaly. Compared to those with a normal fECHO (N = 30), subjects with an abnormal fECHO result (N = 12) had higher state anxiety (46.8 ± 15.5 vs 30.8 ± 8.5, p = 0.005). There was no difference in anxiety scores between subjects awaiting fECHO and post-fECHO subjects who had an abnormal result. Conclusion: Immediately following normal fECHO, women report low anxiety compared with women awaiting fECHO. Women awaiting fECHO report anxiety levels that are as high as women who are told there is fetal cardiac anomaly.展开更多
Objective: Mortality in FASD has not been well studied. In this paper we review published reports of mortality in FASD. Method: We searched using Pub Med for all years in all languages for reports of all-cause mortali...Objective: Mortality in FASD has not been well studied. In this paper we review published reports of mortality in FASD. Method: We searched using Pub Med for all years in all languages for reports of all-cause mortality associated with any FASD. Results: We located 26 papers reporting on 57 deaths. Cause of death was reported for 49/57 cases (86%). The two most prevalent potential causes of death were malformations of the heart (37 of 49 cases, 75.5%) which varied from atrial septal defect or patent ductus arteriosus to tetralogy of Fallot, hypoplastic left heart, aortic arch interruption, etc. and brain malformations(25 of 49, 51%) including microcephaly, hydrocephalus, porencephaly, agenesis/absence of the corpus callosum and semilobar holoprosencephaly. In several cases potential causal findings overlapped. The three most frequent other causes of death were sepsis (7 cases, 14.3%), kidney malformations (7 cases, 14.3%), and cancer (4 cases, 8.2%). Over half the deaths (30/55, 54.5%) occurred in the first year of life. Discussion: We found that congenital heart disease was the most common cause of death in people with FASD. This may be due to an ascertainment bias since many of the published studies were focused on congenital heart disease in FASD. We conclude that FASD is frequently undetected in mortality events and could be a common finding in infant, child, adolescent and adult mortality.展开更多
文摘Objective: Fetal Alcohol Spectrum Disorders (FASDs) are common, often undiagnosed, lifelong developmental disorders that result from prenatal alcohol exposure. FASD is present at birth and typically identified around seven years of age. The most severe outcome in cases of FASD is mortality. The purpose of this scoping review is to 1) use a systematic review to provide an estimated mortality proportion for children with FASD, and 2) update a study published in 2014 by reviewing published reports of mortality in individuals diagnosed with FASD. Method: A search of PubMed, CINAHL, and Google Scholar for reports published between 2013 and 2023 on mortality in individuals with FASD. Results: Three population-based studies have reported on all-cause mortality rates, finding a combined mortality rate of 10.9%, a 2.63 fold (95% CI: 2.61 to 2.65) increase in mortality risk over the general population. Since 2016, this review identified only eight new cases meeting the study inclusion criteria. The reported causes of death were five cases of pneumonia, and one case each of failure to thrive and dehydration, intestinal dilatation and asphyxiation caused by overeating due to pica, and acute gastric volvulus. Discussion: While current research suggests a diagnosis of FASD is associated with a 2.6-fold increase in mortality risk, this is likely an underestimation, as most cases of FASD-related mortality go unreported. Globally, about 1 new case is reported every 15 months. However, in the United States alone, between 1752 to 4400 FASD related deaths occur annually. Our review suggests that FASD is rarely identified as a causal or contributing factor in deaths of children and adolescents, resulting in a substantial undercount of FASD-related deaths. Increased attention to the role of FASD in infant and child mortality case reviews, child death review committee reports, and mortality reviews is needed.
文摘目的通过成本-效用分析,评价孕早期行胎儿结构畸形超声筛查相比于孕中期行胎儿结构畸形超声筛查的经济性,为临床路径优化提供循证依据。方法从全社会角度出发,构建决策树模型,模拟孕妇于孕早期行胎儿结构畸形超声筛查(干预组)与于孕中期行胎儿结构畸形超声筛查(对照组)2种不同产前筛查方案的临床选择及转归路径,评估2组不同路径下的成本-效用情况。模型模拟时长为18周,临床转移概率和成本参数来源于深圳市南山区妇幼保健院数据,效用数据来源于已公开发表的文献。结果指标包括成本、质量调整生命年(qualityadjusted life years,QALYs)和增量成本-效果比(incremental cost-effectiveness ratio,ICER)。通过敏感性分析评估模型结果的稳健性。结果相比对照组,干预组可减少不必要的妊娠发生,以更少的总成本投入获得更多的QALYs,ICER为−161732.68元/QALY,具有绝对成本效果优势。概率敏感性分析表明基础分析结果具有稳健性,干预组具有成本效果的概率为94.60%。结论在全社会角度下,于孕早期进行胎儿结构畸形筛查相比于孕中期具有绝对的经济学优势。
文摘Background: Women awaiting fetal echocardiography (fECHO) report high anxiety. It is unclear if anxiety decreases after performance of fECHO. Methods: At fECHO, subjects’ current (state) vs baseline (trait) anxiety was assessed using the Spielberger State-Trait Anxiety Inventory. Anxiety scores of the pre- and post-fECHO groups were compared. Results: From January 2007 to January 2009, we recruited 84 subjects: 40 pre-fECHO and 44 post-fECHO. Of the post-fECHO group, 30 had normal fetal cardiac structure and function confirmed, 12 were told of an abnormality, and 2 were told to follow up equivocal results. Anxiety scores were compared between the 40 pre-fECHO subjects and the 30 post-fECHO subjects with normal results. The mean state anxiety score of the pre-fECHO group was higher than that of the post-fECHO group (42.1 ± 15.1 vs 30.8 ± 8.5, p < 0.001);there was no difference in trait scores. Neither state nor trait anxiety was associated with maternal age, parity, history of miscarriage or known fetal anomaly. Compared to those with a normal fECHO (N = 30), subjects with an abnormal fECHO result (N = 12) had higher state anxiety (46.8 ± 15.5 vs 30.8 ± 8.5, p = 0.005). There was no difference in anxiety scores between subjects awaiting fECHO and post-fECHO subjects who had an abnormal result. Conclusion: Immediately following normal fECHO, women report low anxiety compared with women awaiting fECHO. Women awaiting fECHO report anxiety levels that are as high as women who are told there is fetal cardiac anomaly.
文摘Objective: Mortality in FASD has not been well studied. In this paper we review published reports of mortality in FASD. Method: We searched using Pub Med for all years in all languages for reports of all-cause mortality associated with any FASD. Results: We located 26 papers reporting on 57 deaths. Cause of death was reported for 49/57 cases (86%). The two most prevalent potential causes of death were malformations of the heart (37 of 49 cases, 75.5%) which varied from atrial septal defect or patent ductus arteriosus to tetralogy of Fallot, hypoplastic left heart, aortic arch interruption, etc. and brain malformations(25 of 49, 51%) including microcephaly, hydrocephalus, porencephaly, agenesis/absence of the corpus callosum and semilobar holoprosencephaly. In several cases potential causal findings overlapped. The three most frequent other causes of death were sepsis (7 cases, 14.3%), kidney malformations (7 cases, 14.3%), and cancer (4 cases, 8.2%). Over half the deaths (30/55, 54.5%) occurred in the first year of life. Discussion: We found that congenital heart disease was the most common cause of death in people with FASD. This may be due to an ascertainment bias since many of the published studies were focused on congenital heart disease in FASD. We conclude that FASD is frequently undetected in mortality events and could be a common finding in infant, child, adolescent and adult mortality.
