Introduction: Twin pregnancy is associated with higher rates of maternal morbidities including gestational diabetes and hypertension. Dichorionic twins are believed to have greater placental mass. Our objective was to study the incidence of two placenta-related disorders: gestational diabetes and hypertension, in dichorionic versus monochorionic twin pregnancies.
Methods: Patients' data of all consecutive twin pregnancies over a period of 12 years were collected from medical records. Data on chorionicity were retrieved from 1st trimester ultrasound reports. Maternal complications including gestational hypertension and diabetes were collected, and incidence was compared between dichorionic and monochorionic twin pregnancies. Records lacking chorionicity data and cases with pregestational diabetes were excluded.
Results: A total of 960 twin pregnancies, 121 monochorionic and 839 dichorionic, were included. Average maternal age did not differ significantly between the groups. The median gestational age at delivery was 36.0 weeks in monochorionic and 36.7 in dichorionic twins. Primiparity (40.4% vs. 23.1%, p < 0.001) and the rate of infertility treatments (51.5% vs. 7.4%, p < 0.001) were both more common in the dichorionic group. The incidence of gestational hypertension disorders was 14% in monochorionic versus 11% in dichorionic twins (p = 0.36). Gestational diabetes was more common in dichorionic compared to monochorionic twins (9.4% and 2.5%, respectively); however, logistic multivariate analysis showed that gestational diabetes was highly correlated with maternal age (p < 0.001) and infertility treatments (p < 0.001) but not with chorionicity (p = 0.136).
Conclusion: Our results may imply that greater placental mass does not increase the risk for gestational hypertension and diabetes. This might support the role of additional multiple maternal factors associated with these complications.
{"title":"The Role of Chorionicity in Placenta-Related Disorders.","authors":"Yana Brudner, Arwa Issa, Hila Sharabi, Livna Shafat, Boaz Weisz, Alon Shrim","doi":"10.1159/000541457","DOIUrl":"10.1159/000541457","url":null,"abstract":"<p><strong>Introduction: </strong>Twin pregnancy is associated with higher rates of maternal morbidities including gestational diabetes and hypertension. Dichorionic twins are believed to have greater placental mass. Our objective was to study the incidence of two placenta-related disorders: gestational diabetes and hypertension, in dichorionic versus monochorionic twin pregnancies.</p><p><strong>Methods: </strong>Patients' data of all consecutive twin pregnancies over a period of 12 years were collected from medical records. Data on chorionicity were retrieved from 1st trimester ultrasound reports. Maternal complications including gestational hypertension and diabetes were collected, and incidence was compared between dichorionic and monochorionic twin pregnancies. Records lacking chorionicity data and cases with pregestational diabetes were excluded.</p><p><strong>Results: </strong>A total of 960 twin pregnancies, 121 monochorionic and 839 dichorionic, were included. Average maternal age did not differ significantly between the groups. The median gestational age at delivery was 36.0 weeks in monochorionic and 36.7 in dichorionic twins. Primiparity (40.4% vs. 23.1%, p < 0.001) and the rate of infertility treatments (51.5% vs. 7.4%, p < 0.001) were both more common in the dichorionic group. The incidence of gestational hypertension disorders was 14% in monochorionic versus 11% in dichorionic twins (p = 0.36). Gestational diabetes was more common in dichorionic compared to monochorionic twins (9.4% and 2.5%, respectively); however, logistic multivariate analysis showed that gestational diabetes was highly correlated with maternal age (p < 0.001) and infertility treatments (p < 0.001) but not with chorionicity (p = 0.136).</p><p><strong>Conclusion: </strong>Our results may imply that greater placental mass does not increase the risk for gestational hypertension and diabetes. This might support the role of additional multiple maternal factors associated with these complications.</p>","PeriodicalId":12189,"journal":{"name":"Fetal Diagnosis and Therapy","volume":null,"pages":null},"PeriodicalIF":1.6,"publicationDate":"2024-09-23","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142307416","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Catherine Hamzeh, Jessica Green, Grace Hamadeh, Lisa M Korst, Ramen H Chmait
Introduction: To compare the blood volume transfused for fetal anemia in cases of placental chorioangioma versus red blood cell (RBC) alloimmunization in patients matched for gestational age (GA) and hydrops.
Methods: Study patients had intrauterine transfusions and were obtained from 3 sources: group (1) placental chorioangioma patients treated at our center (2016-2023); group (2) placental chorioangioma patients reported in the medical literature; and group (3) RBC alloimmunization patients treated for fetal anemia at our center (2016-2023) matched (2:1) to patients in groups 1 and 2 by GA at procedure and presence of hydrops. The expected volume (cc) of transfusion was calculated for all patients based on a formula commonly used for fetal anemia in cases of RBC alloimmunization that includes the GA at procedure, pre-transfusion hemoglobin, donor hemoglobin, and target hemoglobin. The ratio of the volume actually transfused to the expected volume was calculated for each group and for groups 1 and 2 combined; comparisons were made using nonparametric testing.
Results: By group, the patients studied included: (1) 7 treated chorioangioma patients, (2) 8 chorioangioma patients described in the literature, and (3) 30 matched RBC alloimmunization patients. The median (range) GA at procedure for groups 1, 2, and 3 was 29.6 (22.7-32.7), 27.0 (24.0-30.0), 28.4 (22.7-34.3) weeks. The median pre-procedure hemoglobin for each group was 8.5 (5.8-12.5), 6.7 (5.6-12.0), and 5.3 (2.2-10.7) g/dL, and the median post-procedure hemoglobin for each group was 12.9 (11.5-14.0), 12.7 (9.6-14.7), and 13.6 (8.0-15.7) g/dL. The median (range) ratio of the actual to the expected volume transfused for each group was 2.50 (1.79-8.33), 1.64 (1.11-3.85), and 1.10 (0.69-1.86) (p < 0.001). When groups 1 and 2 were combined, the median ratio was 1.89 (1.11-8.33), which remained statistically significant when compared to group 3 (p < 0.001).
Conclusion: Intrauterine transfusion for fetal anemia in cases of large placental chorioangiomas appeared to require nearly twice the blood volume that was normally anticipated for cases of RBC alloimmunization, although the actual amount transfused varied widely.
{"title":"Increased Intrauterine Transfusion Blood Volume Needed to Correct Fetal Anemia due to Placental Chorioangioma.","authors":"Catherine Hamzeh, Jessica Green, Grace Hamadeh, Lisa M Korst, Ramen H Chmait","doi":"10.1159/000541560","DOIUrl":"10.1159/000541560","url":null,"abstract":"<p><strong>Introduction: </strong>To compare the blood volume transfused for fetal anemia in cases of placental chorioangioma versus red blood cell (RBC) alloimmunization in patients matched for gestational age (GA) and hydrops.</p><p><strong>Methods: </strong>Study patients had intrauterine transfusions and were obtained from 3 sources: group (1) placental chorioangioma patients treated at our center (2016-2023); group (2) placental chorioangioma patients reported in the medical literature; and group (3) RBC alloimmunization patients treated for fetal anemia at our center (2016-2023) matched (2:1) to patients in groups 1 and 2 by GA at procedure and presence of hydrops. The expected volume (cc) of transfusion was calculated for all patients based on a formula commonly used for fetal anemia in cases of RBC alloimmunization that includes the GA at procedure, pre-transfusion hemoglobin, donor hemoglobin, and target hemoglobin. The ratio of the volume actually transfused to the expected volume was calculated for each group and for groups 1 and 2 combined; comparisons were made using nonparametric testing.</p><p><strong>Results: </strong>By group, the patients studied included: (1) 7 treated chorioangioma patients, (2) 8 chorioangioma patients described in the literature, and (3) 30 matched RBC alloimmunization patients. The median (range) GA at procedure for groups 1, 2, and 3 was 29.6 (22.7-32.7), 27.0 (24.0-30.0), 28.4 (22.7-34.3) weeks. The median pre-procedure hemoglobin for each group was 8.5 (5.8-12.5), 6.7 (5.6-12.0), and 5.3 (2.2-10.7) g/dL, and the median post-procedure hemoglobin for each group was 12.9 (11.5-14.0), 12.7 (9.6-14.7), and 13.6 (8.0-15.7) g/dL. The median (range) ratio of the actual to the expected volume transfused for each group was 2.50 (1.79-8.33), 1.64 (1.11-3.85), and 1.10 (0.69-1.86) (p < 0.001). When groups 1 and 2 were combined, the median ratio was 1.89 (1.11-8.33), which remained statistically significant when compared to group 3 (p < 0.001).</p><p><strong>Conclusion: </strong>Intrauterine transfusion for fetal anemia in cases of large placental chorioangiomas appeared to require nearly twice the blood volume that was normally anticipated for cases of RBC alloimmunization, although the actual amount transfused varied widely.</p>","PeriodicalId":12189,"journal":{"name":"Fetal Diagnosis and Therapy","volume":null,"pages":null},"PeriodicalIF":1.6,"publicationDate":"2024-09-20","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142282695","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Laura Szlang,Rabih Chaoui,Bernd Opgen-Rhein,Mi-Young Cho,Wolfgang Henrich
Introduction Anomalous origin of the left pulmonary artery (AOLPA) is an exceptionally rare congenital malformation, requiring particular care to be detected during fetal echocardiography. Case presentation A 30-year-old woman, gravida 1, para 0, was referred for a mid-trimester anomaly scan. The three-vessel tracheal view in fetal echocardiographic examination led to the prenatal detection of an anomalous origin of the left pulmonary artery in the presence of a right-sided aortic arch. Additionally, a bilateral arterial duct and the ductal origin of the left pulmonary artery (LPA) were detected postnatally. Prenatal diagnosis enabled the scheduling of the delivery in a tertiary perinatal center, immediate postnatal treatment with prostaglandin E1 to avoid obstruction of the isolated LPA as well as surgical repair of the anomaly. Conclusion The rareness of the disease led to only sporadically published cases of prenatal diagnosis of AOLPA. However, early detection makes prenatal diagnosis crucial regarding the infants' outcome. This case report underlines the importance of a meticulous examination of the bifurcation of the pulmonary trunk during fetal echocardiography.
{"title":"Prenatal Diagnosis of Anomalous Origin of the Left Pulmonary Artery in Fetus with a Right-Sided Aortic Arch - Case Report and Review of the Literature.","authors":"Laura Szlang,Rabih Chaoui,Bernd Opgen-Rhein,Mi-Young Cho,Wolfgang Henrich","doi":"10.1159/000541432","DOIUrl":"https://doi.org/10.1159/000541432","url":null,"abstract":"Introduction Anomalous origin of the left pulmonary artery (AOLPA) is an exceptionally rare congenital malformation, requiring particular care to be detected during fetal echocardiography. Case presentation A 30-year-old woman, gravida 1, para 0, was referred for a mid-trimester anomaly scan. The three-vessel tracheal view in fetal echocardiographic examination led to the prenatal detection of an anomalous origin of the left pulmonary artery in the presence of a right-sided aortic arch. Additionally, a bilateral arterial duct and the ductal origin of the left pulmonary artery (LPA) were detected postnatally. Prenatal diagnosis enabled the scheduling of the delivery in a tertiary perinatal center, immediate postnatal treatment with prostaglandin E1 to avoid obstruction of the isolated LPA as well as surgical repair of the anomaly. Conclusion The rareness of the disease led to only sporadically published cases of prenatal diagnosis of AOLPA. However, early detection makes prenatal diagnosis crucial regarding the infants' outcome. This case report underlines the importance of a meticulous examination of the bifurcation of the pulmonary trunk during fetal echocardiography.","PeriodicalId":12189,"journal":{"name":"Fetal Diagnosis and Therapy","volume":null,"pages":null},"PeriodicalIF":2.2,"publicationDate":"2024-09-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142262424","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Ashlyn E Whitlock,Kamila Moskowitzova,Ina Kycia,Jeffrey Nelson,David Zurakowski,Dario O Fauza
INTRODUCTIONSecretory immunoglobulin-A (SIgA), which is not produced perinatally, binds bacteria enhancing mucosal immunity. Higher levels of intestinal bacteria bound by SIgA are protective against necrotizing enterocolitis. Transamniotic fetal immunotherapy (TRAFIT) has previously been used to deliver SIgA to the fetal digestive tract, however with unclear functional impact. We sought to determine whether SIgA administered via TRAFIT could functionally bind intestinal bacteria postnatally.METHODSFetuses (n=38) from four dams underwent intra-amniotic injections of human SIgA on gestational-day 19 (E19; term=E22-E23). After spontaneous delivery, pups were survived for 1-2 days postnatally before intestinal contents were procured and submitted to flow cytometry. Specimens were stained for bacteria (Syto-GFP) and human-SIgA (PE) to prevent cross-reactivity with maternal rat SIgA.RESULTSOverall survival was 94.7% (36/38). SIgA-bacterial complexes were identified in all samples at all time points showing significantly higher positive PE events than unstained controls (p=0.03-0.05). The proportion of bacteria bound by IgA decreased daily, from 45.6% to 29.9% bound at 4 to 6 days post-TRAFIT, respectively (overall p=0.05).CONCLUSIONSTransamniotic fetal immunotherapy with secretory-IgA leads to functionally IgA-bound bacteria into the postnatal period and may be a novel strategy for enhancing early mucosal immunity, potentially protecting the neonate against necrotizing enterocolitis.
简介分泌性免疫球蛋白-A(SIgA)不会在围产期产生,它能与细菌结合,增强粘膜免疫能力。SIgA 结合的肠道细菌水平越高,对坏死性小肠结肠炎越有保护作用。经羊膜胎儿免疫疗法(TRAFIT)曾被用于向胎儿消化道输送 SIgA,但其功能影响尚不明确。我们试图确定通过 TRAFIT 给药的 SIgA 是否能在出生后与肠道细菌发生功能性结合。方法四个母体的胎儿(38 个)在妊娠第 19 天(E19;足月=E22-E23)接受了人类 SIgA 的羊膜腔内注射。自然分娩后,幼崽在产后存活 1-2 天,然后采集肠道内容物并进行流式细胞术检测。标本经细菌(Syto-GFP)和人SIgA(PE)染色,以防止与母体大鼠SIgA发生交叉反应。结果总存活率为94.7%(36/38)。在所有时间点的所有样本中都发现了 SIgA-细菌复合物,显示 PE 阳性事件明显高于未染色对照组(p=0.03-0.05)。结论用分泌型 IgA 进行经羊膜胎儿免疫治疗可使 IgA 结合细菌进入出生后阶段,这可能是增强早期粘膜免疫的一种新策略,有可能保护新生儿免受坏死性小肠结肠炎的侵害。
{"title":"Transamniotic Fetal Immunotherapy (TRAFIT) with Secretory IgA: A Potential Novel Ancillary Strategy for the Prevention of Necrotizing Enterocolitis.","authors":"Ashlyn E Whitlock,Kamila Moskowitzova,Ina Kycia,Jeffrey Nelson,David Zurakowski,Dario O Fauza","doi":"10.1159/000541434","DOIUrl":"https://doi.org/10.1159/000541434","url":null,"abstract":"INTRODUCTIONSecretory immunoglobulin-A (SIgA), which is not produced perinatally, binds bacteria enhancing mucosal immunity. Higher levels of intestinal bacteria bound by SIgA are protective against necrotizing enterocolitis. Transamniotic fetal immunotherapy (TRAFIT) has previously been used to deliver SIgA to the fetal digestive tract, however with unclear functional impact. We sought to determine whether SIgA administered via TRAFIT could functionally bind intestinal bacteria postnatally.METHODSFetuses (n=38) from four dams underwent intra-amniotic injections of human SIgA on gestational-day 19 (E19; term=E22-E23). After spontaneous delivery, pups were survived for 1-2 days postnatally before intestinal contents were procured and submitted to flow cytometry. Specimens were stained for bacteria (Syto-GFP) and human-SIgA (PE) to prevent cross-reactivity with maternal rat SIgA.RESULTSOverall survival was 94.7% (36/38). SIgA-bacterial complexes were identified in all samples at all time points showing significantly higher positive PE events than unstained controls (p=0.03-0.05). The proportion of bacteria bound by IgA decreased daily, from 45.6% to 29.9% bound at 4 to 6 days post-TRAFIT, respectively (overall p=0.05).CONCLUSIONSTransamniotic fetal immunotherapy with secretory-IgA leads to functionally IgA-bound bacteria into the postnatal period and may be a novel strategy for enhancing early mucosal immunity, potentially protecting the neonate against necrotizing enterocolitis.","PeriodicalId":12189,"journal":{"name":"Fetal Diagnosis and Therapy","volume":null,"pages":null},"PeriodicalIF":2.2,"publicationDate":"2024-09-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142262425","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Kamila Moskowitzova,Abbie E Naus,Tanya T Dang,David Zurakowski,Dario O Fauza
INTRODUCTIONWe sought to determine whether exogenous surfactant protein B (SPB) mRNA could be incorporated and translated by the fetal lung after simple transamniotic administration.METHODSFetuses (n=149) of twelve time-dated dams underwent intra-amniotic injections of either human SPB (hSPB) mRNA encapsulated into lipopolyplex (mRNA, n=99), or of lipopolyplex without mRNA (control; n=50) on gestational day 17 (E17, term=E21-22). Lungs were screened for hSPB by ELISA daily until term. Phosphatidylcholine (a surrogate for surfactant production) was measured in the amniotic fluid by fluorometric assay. Statistical analysis included nonparametric Wilcoxon rank sum test.RESULTSSignificantly improved survival in the mRNA group compared to controls was observed at E18 (100% vs. 85.7%) and E20 (100% vs. 83.3%) (both p<0.001). When controlled by mRNA-free injections, hSPB protein was detected in the mRNA group's lungs at E18, 19, and term (p=0.002 to <0.001). Amniotic fluid phosphatidylcholine levels were increased compared to control at term [285.9 (251.1, 363.9)μM vs. 263.1 (222.8, 309.1)μM], however this did not reach significance (p=0.33).CONCLUSIONSEncapsulated exogenous SPB mRNA can be incorporated and translated by fetal lung cells following intra-amniotic injection in a healthy rat model. Transamniotic mRNA delivery could become a novel strategy for perinatal surfactant protein replacement.
{"title":"Transamniotic Delivery of Surfactant Protein B mRNA in a Healthy Model.","authors":"Kamila Moskowitzova,Abbie E Naus,Tanya T Dang,David Zurakowski,Dario O Fauza","doi":"10.1159/000541429","DOIUrl":"https://doi.org/10.1159/000541429","url":null,"abstract":"INTRODUCTIONWe sought to determine whether exogenous surfactant protein B (SPB) mRNA could be incorporated and translated by the fetal lung after simple transamniotic administration.METHODSFetuses (n=149) of twelve time-dated dams underwent intra-amniotic injections of either human SPB (hSPB) mRNA encapsulated into lipopolyplex (mRNA, n=99), or of lipopolyplex without mRNA (control; n=50) on gestational day 17 (E17, term=E21-22). Lungs were screened for hSPB by ELISA daily until term. Phosphatidylcholine (a surrogate for surfactant production) was measured in the amniotic fluid by fluorometric assay. Statistical analysis included nonparametric Wilcoxon rank sum test.RESULTSSignificantly improved survival in the mRNA group compared to controls was observed at E18 (100% vs. 85.7%) and E20 (100% vs. 83.3%) (both p<0.001). When controlled by mRNA-free injections, hSPB protein was detected in the mRNA group's lungs at E18, 19, and term (p=0.002 to <0.001). Amniotic fluid phosphatidylcholine levels were increased compared to control at term [285.9 (251.1, 363.9)μM vs. 263.1 (222.8, 309.1)μM], however this did not reach significance (p=0.33).CONCLUSIONSEncapsulated exogenous SPB mRNA can be incorporated and translated by fetal lung cells following intra-amniotic injection in a healthy rat model. Transamniotic mRNA delivery could become a novel strategy for perinatal surfactant protein replacement.","PeriodicalId":12189,"journal":{"name":"Fetal Diagnosis and Therapy","volume":null,"pages":null},"PeriodicalIF":2.2,"publicationDate":"2024-09-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142262426","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Anthony di Natale, Sabrina Flohr, Leny Mathew, Cara Katterman, Colby Gallagher, Thomas A Reynolds, Juliana S Gebb, Howard B Panitch, Edward R Oliver, Natalie E Rintoul, William H Peranteau, Alan W Flake, N Scott Adzick, Holly L Hedrick
Introduction: Congenital pulmonary airway malformations (CPAM), intra- and extralobar bronchopulmonary sequestrations (iBPS/eBPS), CPAM-BPS hybrid lesions (HL), congenital lobar emphysema (CLE), bronchial atresia (BA), and foregut duplication cysts (FDC), collectively referred to as congenital thoracic lesions (CTL), are mostly solitary. Patients with multiple CTL are rare, and reports on such cases are scarce. To address this dearth, we analyzed a large multifocal CTL patient cohort.
Methods: Retrospective chart review of patients born between September 1, 2013, and March 31, 2023, who underwent surgery for a CTL at our tertiary center. Patients with radiological and surgical diagnosis of multifocal CTL, defined as ≥2 CTL present in more than one lobe were included to record pre-, peri-, and postnatal patient characteristics.
Results: Among 701 CTL patients, 74 (10.5%) had multiple CTL. CTL multifocality was prenatally recognized correctly in 8 (12.9%) patients. Most multiple CTL were right-sided, unilateral multilobar lesions (n = 33, 44%). Bilateral CTL were found in 9 (12.1%) patients. CPAM-CPAM lesions were the most prevalent CTL types (n = 36, 49%). Genetic syndromes were confirmed in 3 (4%) and additional congenital anomalies in 9 (12.9%) patients, 5 of those had multiple congenital anomalies. Of 49 (65%) patients with multilobar CTL, 25 (51%) underwent bilobectomy and 24 (49%) lung-sparing surgery. Length of stay was similar. Mortality was 5.4%.
Conclusion: We report on the largest patient cohort with multiple CTL to date. Multiple CTL occurred in 1/10 patients with CTL, and only 12.9% were recognized prenatally. Lung-sparing surgery can be considered. Multiple additional congenital anomalies and genetic syndromes may be more common and genetic testing should be considered. Overall, outcomes in this patient population are favorable.
{"title":"Neonatal Outcomes of Multiple Congenital Thoracic Lesions.","authors":"Anthony di Natale, Sabrina Flohr, Leny Mathew, Cara Katterman, Colby Gallagher, Thomas A Reynolds, Juliana S Gebb, Howard B Panitch, Edward R Oliver, Natalie E Rintoul, William H Peranteau, Alan W Flake, N Scott Adzick, Holly L Hedrick","doi":"10.1159/000541319","DOIUrl":"10.1159/000541319","url":null,"abstract":"<p><strong>Introduction: </strong>Congenital pulmonary airway malformations (CPAM), intra- and extralobar bronchopulmonary sequestrations (iBPS/eBPS), CPAM-BPS hybrid lesions (HL), congenital lobar emphysema (CLE), bronchial atresia (BA), and foregut duplication cysts (FDC), collectively referred to as congenital thoracic lesions (CTL), are mostly solitary. Patients with multiple CTL are rare, and reports on such cases are scarce. To address this dearth, we analyzed a large multifocal CTL patient cohort.</p><p><strong>Methods: </strong>Retrospective chart review of patients born between September 1, 2013, and March 31, 2023, who underwent surgery for a CTL at our tertiary center. Patients with radiological and surgical diagnosis of multifocal CTL, defined as ≥2 CTL present in more than one lobe were included to record pre-, peri-, and postnatal patient characteristics.</p><p><strong>Results: </strong>Among 701 CTL patients, 74 (10.5%) had multiple CTL. CTL multifocality was prenatally recognized correctly in 8 (12.9%) patients. Most multiple CTL were right-sided, unilateral multilobar lesions (n = 33, 44%). Bilateral CTL were found in 9 (12.1%) patients. CPAM-CPAM lesions were the most prevalent CTL types (n = 36, 49%). Genetic syndromes were confirmed in 3 (4%) and additional congenital anomalies in 9 (12.9%) patients, 5 of those had multiple congenital anomalies. Of 49 (65%) patients with multilobar CTL, 25 (51%) underwent bilobectomy and 24 (49%) lung-sparing surgery. Length of stay was similar. Mortality was 5.4%.</p><p><strong>Conclusion: </strong>We report on the largest patient cohort with multiple CTL to date. Multiple CTL occurred in 1/10 patients with CTL, and only 12.9% were recognized prenatally. Lung-sparing surgery can be considered. Multiple additional congenital anomalies and genetic syndromes may be more common and genetic testing should be considered. Overall, outcomes in this patient population are favorable.</p>","PeriodicalId":12189,"journal":{"name":"Fetal Diagnosis and Therapy","volume":null,"pages":null},"PeriodicalIF":1.6,"publicationDate":"2024-09-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142153521","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Background: The International Fetal Medicine and Surgery Society (IFMSS) was founded a little over 40 years ago, to offer a collegial, interdisciplinary forum for fetal specialists worldwide. Its inclusive culture has allowed innovations, successes, and failures to be reported freely. The present report examines progress in the field of fetal medicine, as reflected by the diversity and frequency of IFMSS presentations. We also test the hypothesis that throughout the decades, IFMSS has remained at the forefront of new developments in fetal diagnosis and therapy.
Summary: The scientific programs of annual conferences were reviewed for title and subject and assigned one or more keywords, and a single category (resulting in absolute, rather than relative incidences of topics). Select procedures covered at IFMSS meetings were plotted over time against comparable queries in PubMed. 5,467 presentations were reviewed and categorized. Keywords were plotted as a heatmap, showing a gradual shift from mostly observational to increasingly invasive studies. The relative interest of particular topics varied widely over the years, from bladder obstruction and renal pathology in early meetings, to complicated twin gestations, to fetal surgery for diaphragmatic hernia and myelomeningocele. Reports on the treatment of twin-to-twin transfusion syndrome, fetal surgery for myelomeningocele and stem cell therapy preceded similar publications in the world literature by several years.
Key messages: In its 40-year history, IFMSS has continued to offer an interdisciplinary forum for fetal medicine specialists, even as the topics of interests have evolved with the pace of technology, long-term follow-up, and new scientific discoveries.
简介:国际胎儿医学与外科学会(International Fetal Medicine and Surgery Society,IFMSS)成立于1984年,旨在为全世界的胎儿专家提供一个跨学科的学术论坛,让创新、成功和失败的案例得以自由报道。在此,我们将从 IFMSS 发言的多样性和频率来考察胎儿医学的进展。我们还检验了 IFMSS 始终站在该领域新发展前沿的假设:我们对年度会议的科学计划进行了标题和主题审查,并指定了一个或多个关键词和一个类别。将 IFMSS 会议涉及的部分程序与 PubMed 中的可比查询进行对比:结果:共审查并分类了 5,467 篇演讲。关键词被绘制成一张热图,从大部分观察性研究到越来越多的侵入性研究。关注的主题差异很大,从早期会议的肾脏病理学,到双胞胎输血综合征(TTTS)、膈疝和脊髓膜膨出的胎儿手术。关于TTTS、骨髓膜缺失修复和干细胞疗法的报告比世界文献中类似文章的发表时间要早数年:在其 40 年的历史中,IFMSS 为胎儿专家提供了一个跨学科的论坛,即使感兴趣的话题随着技术、长期随访和新科学发现的步伐而不断发展。
{"title":"IFMSS at Forty: You Heard It Here First.","authors":"Karen Mei Xian Lim, Francois I Luks","doi":"10.1159/000541136","DOIUrl":"10.1159/000541136","url":null,"abstract":"<p><strong>Background: </strong>The International Fetal Medicine and Surgery Society (IFMSS) was founded a little over 40 years ago, to offer a collegial, interdisciplinary forum for fetal specialists worldwide. Its inclusive culture has allowed innovations, successes, and failures to be reported freely. The present report examines progress in the field of fetal medicine, as reflected by the diversity and frequency of IFMSS presentations. We also test the hypothesis that throughout the decades, IFMSS has remained at the forefront of new developments in fetal diagnosis and therapy.</p><p><strong>Summary: </strong>The scientific programs of annual conferences were reviewed for title and subject and assigned one or more keywords, and a single category (resulting in absolute, rather than relative incidences of topics). Select procedures covered at IFMSS meetings were plotted over time against comparable queries in PubMed. 5,467 presentations were reviewed and categorized. Keywords were plotted as a heatmap, showing a gradual shift from mostly observational to increasingly invasive studies. The relative interest of particular topics varied widely over the years, from bladder obstruction and renal pathology in early meetings, to complicated twin gestations, to fetal surgery for diaphragmatic hernia and myelomeningocele. Reports on the treatment of twin-to-twin transfusion syndrome, fetal surgery for myelomeningocele and stem cell therapy preceded similar publications in the world literature by several years.</p><p><strong>Key messages: </strong>In its 40-year history, IFMSS has continued to offer an interdisciplinary forum for fetal medicine specialists, even as the topics of interests have evolved with the pace of technology, long-term follow-up, and new scientific discoveries.</p>","PeriodicalId":12189,"journal":{"name":"Fetal Diagnosis and Therapy","volume":null,"pages":null},"PeriodicalIF":1.6,"publicationDate":"2024-08-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142105874","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Introduction: The objective of this study was to determine the impact of the stage-based classification of fetal growth restriction (FGR) on the magnitude of FGR, preterm births (PTBs), and birthweight (BW) in a rural population of Madhya Pradesh in Central India.
Methods: The program covered 168 public sector centers for pregnant women and infants that provided services to nearly 220,000 people. The third-trimester assessments included fetal biometry, growth and environment assessments, and Doppler assessments. Fetal growth was staged using the Barcelona protocol as stages 1-4 FGR, small for gestational age, and no FGR. The data from the last ultrasound assessment before childbirth were considered. Regular training programs covering preconception care, antenatal and postnatal care were organized in the local language for the public sector community health workers of the program district. Childbirth outcomes were collected from the obstetric service of the local public sector hospital.
Results: The analysis included 1,229 pregnancies from 2019 to 2023. The overall magnitude of FGR using estimated fetal weight <10th centile was 19.61% and reduced to 13.34% with the stage-based classification. The magnitude of FGR using the stage-based classification reduced from 27.59% in 2019 to 8.95% in 2023. The PTB in the stage-based FGR subgroup declined from 35.0% in 2019 to 3.45% in 2023 and 96.55% of the stage 1 FGR babies in 2023 were delivered at term. The overall mean BW in the program area improved from 2,772.41 (357.11) g in 2019 to 2,819.68 (377.31) g in 2023. The perinatal mortality rate (8.95 per 1,000 pregnancies) in the program area for 2019-2023 was much lower than the 31.9 per 1,000 pregnancies reported for Madhya Pradesh.
Conclusion: The change to a stage-based classification of FGR integrated with low-dose aspirin and fetal Doppler studies reduced the incidence of FGR and PTB and perinatal mortality and increased BW in this rural community.
{"title":"Impact of a Stage-Based Classification on the Incidence of Fetal Growth Restriction, Preterm Birth Rates, and Birthweight in a Rural Community of Central India.","authors":"Lalit Kishore Sharma, Rijo Mathew Choorakuttil, Praveen Kumar Nirmalan","doi":"10.1159/000540199","DOIUrl":"10.1159/000540199","url":null,"abstract":"<p><strong>Introduction: </strong>The objective of this study was to determine the impact of the stage-based classification of fetal growth restriction (FGR) on the magnitude of FGR, preterm births (PTBs), and birthweight (BW) in a rural population of Madhya Pradesh in Central India.</p><p><strong>Methods: </strong>The program covered 168 public sector centers for pregnant women and infants that provided services to nearly 220,000 people. The third-trimester assessments included fetal biometry, growth and environment assessments, and Doppler assessments. Fetal growth was staged using the Barcelona protocol as stages 1-4 FGR, small for gestational age, and no FGR. The data from the last ultrasound assessment before childbirth were considered. Regular training programs covering preconception care, antenatal and postnatal care were organized in the local language for the public sector community health workers of the program district. Childbirth outcomes were collected from the obstetric service of the local public sector hospital.</p><p><strong>Results: </strong>The analysis included 1,229 pregnancies from 2019 to 2023. The overall magnitude of FGR using estimated fetal weight <10th centile was 19.61% and reduced to 13.34% with the stage-based classification. The magnitude of FGR using the stage-based classification reduced from 27.59% in 2019 to 8.95% in 2023. The PTB in the stage-based FGR subgroup declined from 35.0% in 2019 to 3.45% in 2023 and 96.55% of the stage 1 FGR babies in 2023 were delivered at term. The overall mean BW in the program area improved from 2,772.41 (357.11) g in 2019 to 2,819.68 (377.31) g in 2023. The perinatal mortality rate (8.95 per 1,000 pregnancies) in the program area for 2019-2023 was much lower than the 31.9 per 1,000 pregnancies reported for Madhya Pradesh.</p><p><strong>Conclusion: </strong>The change to a stage-based classification of FGR integrated with low-dose aspirin and fetal Doppler studies reduced the incidence of FGR and PTB and perinatal mortality and increased BW in this rural community.</p>","PeriodicalId":12189,"journal":{"name":"Fetal Diagnosis and Therapy","volume":null,"pages":null},"PeriodicalIF":1.6,"publicationDate":"2024-08-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142105875","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Inshirah Sgayer, Mohamed Elafawi, Olga Braude, Sarah Abramov, Lior Lowenstein, Marwan Odeh
Introduction: Limited evidence exists on borderline oligohydramnios. Our objective was to determine perinatal outcomes in uncomplicated term pregnancies with borderline oligohydramnios.
Methods: This retrospective analysis compared adverse perinatal outcomes among pregnancies during 2018-2022, between those with borderline oligohydramnios defined as amniotic fluid index (AFI) of 5.1-8.0 cm, those with oligohydramnios (AFI ≤5 cm), and those with normal AFI (8.1-25 cm). The latter matched one-to-one to the borderline oligohydramnios group and served as the control group. The outcomes compared included birthweight, cesarean delivery due to fetal distress, the presence of meconium-stained amniotic fluid, Apgar scores, neonatal intensive care unit admission, and the occurrence of small-for-gestational-age (SGA) neonates.
Results: During the study period, 140 women had borderline oligohydramnios and 345 had oligohydramnios; the control group included 140 women. Borderline oligohydramnios was associated with increased rates of delivering SGA neonates (adjusted odds ratio [aOR] = 3.6, 95% confidence interval [CI] 1.1-11.6, p = 0.034) and cesarean delivery due to fetal distress (aOR = 3.0, 95% CI 1.1-8.3, p = 0.032). Rates of composite neonatal outcome (including at least one of the following: cesarean delivery due to fetal distress, meconium-stained amniotic fluid, 5-min Apgar score <7, umbilical artery pH <7.15, or neonatal intensive care unit admission) were higher in both the borderline oligohydramnios (20.7%) and oligohydramnios (18.6%) groups compared to the control group (9.3%) (p = 0.019).
Conclusions: Pregnancies with borderline oligohydramnios were associated with increased risks of delivering SGA neonates and requiring cesarean delivery due to fetal distress. These findings support considering closer antepartum surveillance for these pregnancies, especially for identifying abnormal fetal growth.
{"title":"Perinatal Outcomes of Pregnancies with Borderline Oligohydramnios at Term.","authors":"Inshirah Sgayer, Mohamed Elafawi, Olga Braude, Sarah Abramov, Lior Lowenstein, Marwan Odeh","doi":"10.1159/000541008","DOIUrl":"10.1159/000541008","url":null,"abstract":"<p><strong>Introduction: </strong>Limited evidence exists on borderline oligohydramnios. Our objective was to determine perinatal outcomes in uncomplicated term pregnancies with borderline oligohydramnios.</p><p><strong>Methods: </strong>This retrospective analysis compared adverse perinatal outcomes among pregnancies during 2018-2022, between those with borderline oligohydramnios defined as amniotic fluid index (AFI) of 5.1-8.0 cm, those with oligohydramnios (AFI ≤5 cm), and those with normal AFI (8.1-25 cm). The latter matched one-to-one to the borderline oligohydramnios group and served as the control group. The outcomes compared included birthweight, cesarean delivery due to fetal distress, the presence of meconium-stained amniotic fluid, Apgar scores, neonatal intensive care unit admission, and the occurrence of small-for-gestational-age (SGA) neonates.</p><p><strong>Results: </strong>During the study period, 140 women had borderline oligohydramnios and 345 had oligohydramnios; the control group included 140 women. Borderline oligohydramnios was associated with increased rates of delivering SGA neonates (adjusted odds ratio [aOR] = 3.6, 95% confidence interval [CI] 1.1-11.6, p = 0.034) and cesarean delivery due to fetal distress (aOR = 3.0, 95% CI 1.1-8.3, p = 0.032). Rates of composite neonatal outcome (including at least one of the following: cesarean delivery due to fetal distress, meconium-stained amniotic fluid, 5-min Apgar score <7, umbilical artery pH <7.15, or neonatal intensive care unit admission) were higher in both the borderline oligohydramnios (20.7%) and oligohydramnios (18.6%) groups compared to the control group (9.3%) (p = 0.019).</p><p><strong>Conclusions: </strong>Pregnancies with borderline oligohydramnios were associated with increased risks of delivering SGA neonates and requiring cesarean delivery due to fetal distress. These findings support considering closer antepartum surveillance for these pregnancies, especially for identifying abnormal fetal growth.</p>","PeriodicalId":12189,"journal":{"name":"Fetal Diagnosis and Therapy","volume":null,"pages":null},"PeriodicalIF":1.6,"publicationDate":"2024-08-22","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142035564","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Andrea Di Tonto, Laura Nogue, Beatrice Valentini, Mar Bennasar, Chiara Melito, Sara Sorrentino, Olga Gómez, Enrico Corno, Maria Teresa Baffa, Tullio Ghi, Fàtima Crispi, Andrea Dall'Asta
Introduction: Speckle tracking echocardiography is a non-Doppler modality allowing the semiautomated evaluation of the fetal cardiac function by tracking the speckles of the endocardial borders. Little evidence is available on the evaluation and comparison of different software for the functional assessment of the fetal heart by means of speckle tracking echocardiography. The aim of this study was to evaluate the reproducibility and agreement of two different proprietary speckle tracking software for the prenatal semiautomated assessment of the fetal cardiac function.
Methods: The prospective study including non-anomalous fetuses was referred for different indications at two tertiary academic units in Italy (University of Parma) and Spain (University of Barcelona). Two-dimensional clips of the four-chamber view of the fetal heart were acquired by two dedicated operators using high-end ultrasound machines with a frame rate higher than 60 Hz. The stored clips were pseudo-anonymized and shared between the collaborating units. Functional echocardiographic analyses were independently performed using the two proprietary software (TomTec GmbH and FetalHQ®) by the same operators. Inter-software reproducibility of the endocardial global longitudinal strain (EndoGLS) and fractional area change (FAC) of the left (LV) and the right ventricles (RV) and ejection fraction (EF) of the LV were evaluated by the intraclass correlation coefficient (ICC).
Results: Forty-eight fetuses were included at a median of 31+2 (21+6-40+3) gestational weeks. Moderate reproducibility was found for the functional parameters of the LV: EndoGLS (Pearson's correlation 0.456, p < 0.01; ICC 0.446, 95% CI: 0.189-0.647, p < 0.01); EF (Pearson's correlation 0.435, p < 0.01; ICC 0.419, 95% CI: 0.156-0.627, p < 0.01); FAC (Person's correlation 0.484, p < 0.01; ICC 0.475, 95% CI: 0.223-0.667, p < 0.01). On the contrary, RV functional parameters showed poor reproducibility between the two software: EndoGLS (Pearson's correlation 0.383, p = 0.01; ICC 0.377, 95% CI: 0.107-0.596, p < 0.01) and FAC (ICC 0.284, 95% CI: 0.003-0.524, p = 0.02).
Conclusion: Our results demonstrate a moderate reproducibility of the speckle tracking analysis of the LV using TomTec GmbH and FetalHQ®, with poor reproducibility for RV analysis.
{"title":"Reproducibility Analysis of Two Speckle Tracking Software for the Antenatal Semiautomated Assessment of the Fetal Cardiac Function.","authors":"Andrea Di Tonto, Laura Nogue, Beatrice Valentini, Mar Bennasar, Chiara Melito, Sara Sorrentino, Olga Gómez, Enrico Corno, Maria Teresa Baffa, Tullio Ghi, Fàtima Crispi, Andrea Dall'Asta","doi":"10.1159/000541007","DOIUrl":"10.1159/000541007","url":null,"abstract":"<p><strong>Introduction: </strong>Speckle tracking echocardiography is a non-Doppler modality allowing the semiautomated evaluation of the fetal cardiac function by tracking the speckles of the endocardial borders. Little evidence is available on the evaluation and comparison of different software for the functional assessment of the fetal heart by means of speckle tracking echocardiography. The aim of this study was to evaluate the reproducibility and agreement of two different proprietary speckle tracking software for the prenatal semiautomated assessment of the fetal cardiac function.</p><p><strong>Methods: </strong>The prospective study including non-anomalous fetuses was referred for different indications at two tertiary academic units in Italy (University of Parma) and Spain (University of Barcelona). Two-dimensional clips of the four-chamber view of the fetal heart were acquired by two dedicated operators using high-end ultrasound machines with a frame rate higher than 60 Hz. The stored clips were pseudo-anonymized and shared between the collaborating units. Functional echocardiographic analyses were independently performed using the two proprietary software (TomTec GmbH and FetalHQ®) by the same operators. Inter-software reproducibility of the endocardial global longitudinal strain (EndoGLS) and fractional area change (FAC) of the left (LV) and the right ventricles (RV) and ejection fraction (EF) of the LV were evaluated by the intraclass correlation coefficient (ICC).</p><p><strong>Results: </strong>Forty-eight fetuses were included at a median of 31+2 (21+6-40+3) gestational weeks. Moderate reproducibility was found for the functional parameters of the LV: EndoGLS (Pearson's correlation 0.456, p < 0.01; ICC 0.446, 95% CI: 0.189-0.647, p < 0.01); EF (Pearson's correlation 0.435, p < 0.01; ICC 0.419, 95% CI: 0.156-0.627, p < 0.01); FAC (Person's correlation 0.484, p < 0.01; ICC 0.475, 95% CI: 0.223-0.667, p < 0.01). On the contrary, RV functional parameters showed poor reproducibility between the two software: EndoGLS (Pearson's correlation 0.383, p = 0.01; ICC 0.377, 95% CI: 0.107-0.596, p < 0.01) and FAC (ICC 0.284, 95% CI: 0.003-0.524, p = 0.02).</p><p><strong>Conclusion: </strong>Our results demonstrate a moderate reproducibility of the speckle tracking analysis of the LV using TomTec GmbH and FetalHQ®, with poor reproducibility for RV analysis.</p>","PeriodicalId":12189,"journal":{"name":"Fetal Diagnosis and Therapy","volume":null,"pages":null},"PeriodicalIF":1.6,"publicationDate":"2024-08-19","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142003989","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}