Fetal Diagnosis and Therapy最新文献

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Transamniotic Delivery of Surfactant Protein B mRNA in a Healthy Model. 在健康模型中经羊膜输送表面活性蛋白 B mRNA。
IF 2.2 3区 医学
Fetal Diagnosis and Therapy Pub Date : 2024-09-12 DOI: 10.1159/000541429
Kamila Moskowitzova,Abbie E Naus,Tanya T Dang,David Zurakowski,Dario O Fauza
{"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":"6 1","pages":"1-14"},"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}
引用次数: 0
Neonatal Outcomes of Multiple Congenital Thoracic Lesions. 多发性先天性胸廓病变的新生儿结局。
IF 1.6 3区 医学
Fetal Diagnosis and Therapy Pub Date : 2024-09-07 DOI: 10.1159/000541319
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
{"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":" ","pages":"1-13"},"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}
引用次数: 0
IFMSS at Forty: You Heard It Here First. 四十岁的 IFMSS:你在这里先听到的。
IF 1.6 3区 医学
Fetal Diagnosis and Therapy Pub Date : 2024-08-30 DOI: 10.1159/000541136
Karen Mei Xian Lim, Francois I Luks
{"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":" ","pages":"1-8"},"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}
引用次数: 0
Middle Cerebral Artery Doppler before and after Fetal Spina Bifida Repair: An Indirect Sign of Hindbrain Compression and Decompression? 胎儿脊柱裂修复前后的大脑中动脉多普勒:后脑受压和减压的间接标志?
IF 1.6 3区 医学
Fetal Diagnosis and Therapy Pub Date : 2024-06-14 DOI: 10.1159/000539773
Ladina Vonzun, Ladina Ruegg, Julia Zepf, Nele Strübing, Patrice Grehten, Martin Meuli, Luca Mazzone, Ueli Moehrlen, Nicole Ochsenbein-Koelble
{"title":"Middle Cerebral Artery Doppler before and after Fetal Spina Bifida Repair: An Indirect Sign of Hindbrain Compression and Decompression?","authors":"Ladina Vonzun, Ladina Ruegg, Julia Zepf, Nele Strübing, Patrice Grehten, Martin Meuli, Luca Mazzone, Ueli Moehrlen, Nicole Ochsenbein-Koelble","doi":"10.1159/000539773","DOIUrl":"10.1159/000539773","url":null,"abstract":"<p><strong>Introduction: </strong>Reduced middle cerebral artery resistance indices (MCA-RI) in fetuses with spina bifida (fSB) are commonly observed. Compression of neuronal pathways in the brainstem due to hindbrain herniation (HH) and disturbed cerebrospinal fluid circulation likely cause an imbalance of the autonomic nervous system. This may increase systemic vasoconstriction and compensatory increase cerebral vasodilation (like brain sparing). The aim of this study was to systematically analyze all fetal MCA-RI before and after fSB repair and to compare their correlation with the presence and postsurgical resolution of HH.</p><p><strong>Methods: </strong>173 patients were included. Standardized ultrasound examinations including MCA and umbilical artery (UA) Doppler as well as assessment of HH presence and regression were performed. Fetuses with MCA-RI &lt;5th percentile (P) before fetal surgery were compared to the group with normal MCA-RI and correlated to the presence of HH before and its regression after fSB repair.</p><p><strong>Results: </strong>30% (49/161) fetuses showed RI's &lt;5th P before fSB repair. All fetuses had normal UA-RI. 99.4% of fetuses (160/161) showed normal of MCA-RI before delivery. Normalization occurred within a mean of 1.3 ± 1.2 weeks. HH regression was observed in 97% in the group with normal MCA-RI and in 96% in the group with MCA-RI &lt;5th P before surgery (p = 0.59). Time lapse to HH regression after fSB repair was 1.8 ± 1.7 and 1.9 ± 1.6 weeks, respectively.</p><p><strong>Conclusion: </strong>In fetuses with MCA-RIs &lt;5 P before fSB repair, a parallel timely course of MCA-RI normalization and HH regression was noted. To suggest common pathogenic factor(s), more studies are needed. However, normalization of the fetal cerebral circulation could be a further benefit of fSB repair.</p>","PeriodicalId":12189,"journal":{"name":"Fetal Diagnosis and Therapy","volume":" ","pages":"1-9"},"PeriodicalIF":1.6,"publicationDate":"2024-06-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141330699","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}
引用次数: 0
Comparison of Serial Amnioinfusion Strategies for Isolated Early-Onset Fetal Renal Anhydramnios. 针对孤立性早发性胎儿肾性无羊水症的连续无羊水灌注策略比较。
IF 1.6 3区 医学
Fetal Diagnosis and Therapy Pub Date : 2024-06-10 DOI: 10.1159/000539732
Joyce M Cheng, Ahmet A Baschat, Meredith A Atkinson, Mara Rosner, Michelle L Kush, Denise Wolfson, Sarah Olson, Kristin Voegtline, Lindsey Goodman, Angie C Jelin, Jena L Miller
{"title":"Comparison of Serial Amnioinfusion Strategies for Isolated Early-Onset Fetal Renal Anhydramnios.","authors":"Joyce M Cheng, Ahmet A Baschat, Meredith A Atkinson, Mara Rosner, Michelle L Kush, Denise Wolfson, Sarah Olson, Kristin Voegtline, Lindsey Goodman, Angie C Jelin, Jena L Miller","doi":"10.1159/000539732","DOIUrl":"10.1159/000539732","url":null,"abstract":"<p><strong>Introduction: </strong>The optimal protocol for serial amnioinfusions to maintain amniotic fluid in pregnancies with early-onset fetal renal anhydramnios before 22 weeks is not known. We compared the performance of two different approaches.</p><p><strong>Methods: </strong>A secondary analysis was conducted of serial amnioinfusions performed by a single center during the external pilot and feasibility phases of the Renal Anhydramnios Fetal Therapy (RAFT) trial. During the external pilot, higher amnioinfusion volumes were given less frequently; in the feasibility study, smaller volume amnioinfusions were administered more frequently. Procedural details, complications, and obstetric outcomes were compared between the two groups using Pearson's χ2 or Fisher's exact tests for categorical variables and Student's t tests or Wilcoxon rank-sum tests for continuous variables. The adjusted association between procedural details and chorioamniotic separation was obtained through a multivariate repeated measure logistic regression model.</p><p><strong>Results: </strong>Eleven participants underwent 159 amnioinfusions (external pilot: 3 patients, 21 amnioinfusions; feasibility: 8 patients, 138 amnioinfusions). External pilot participants had fewer amnioinfusions (7 vs. 19.5 in the feasibility group, p = 0.04), larger amnioinfusion volume (750 vs. 500 mL, p &lt; 0.01), and longer interval between amnioinfusions (6 [4-7] vs. 4 [3-5] days, p &lt; 0.01). In the external pilot, chorioamniotic separation was more common (28.6% vs. 5.8%, p &lt; 0.01), preterm prelabor rupture of membranes (PPROM) occurred sooner after amnioinfusion initiation (28 ± 21.5 vs. 75.6 ± 24.1 days, p = 0.03), and duration of maintained amniotic fluid between first and last amnioinfusion was shorter (38 ± 17.3 vs. 71 ± 19 days, p = 0.03), compared to the feasibility group. While delivery gestational age was similar (35.1 ± 1.7 vs. 33.8 ± 1.5 weeks, p = 0.21), feasibility participants maintained amniotic fluid longer.</p><p><strong>Conclusion: </strong>Small volume serial amnioinfusions performed more frequently maintain normal amniotic fluid volume longer because of delayed occurrence of PPROM.</p>","PeriodicalId":12189,"journal":{"name":"Fetal Diagnosis and Therapy","volume":" ","pages":"1"},"PeriodicalIF":1.6,"publicationDate":"2024-06-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11628636/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141300472","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Needle Fetal Thoracoscopy: A Technique to Assist with Ultrasound-Guided Placement of Challenging Thoracoamniotic Shunts. 针式胎儿胸腔镜检查:一种在超声引导下辅助进行高难度胸羊膜分流术的技术。
IF 2.2 3区 医学
Fetal Diagnosis and Therapy Pub Date : 2024-05-15 DOI: 10.1159/000539274
Steven T Papastefan, Daniel R Liesman, Katherine C Ott, Federico Scorletti, Xavier F Pombar, Aimen F Shaaban, Amir M Alhajjat
{"title":"Needle Fetal Thoracoscopy: A Technique to Assist with Ultrasound-Guided Placement of Challenging Thoracoamniotic Shunts.","authors":"Steven T Papastefan, Daniel R Liesman, Katherine C Ott, Federico Scorletti, Xavier F Pombar, Aimen F Shaaban, Amir M Alhajjat","doi":"10.1159/000539274","DOIUrl":"10.1159/000539274","url":null,"abstract":"<p><strong>Introduction: </strong>Fetal thoracoamniotic shunts are common lifesaving interventions but frequently require replacement. Needle fetal thoracoscopy is a technique that uses standard thoracoamniotic shunt introducer sheaths to permit direct visualization and even instrument manipulation during shunt deployment to facilitate optimal positioning and primary shunt function in the most challenging cases.</p><p><strong>Case presentation: </strong>In this study, 5 patients who underwent needle fetal thoracoscopy-assisted thoracoamniotic shunt placement were reviewed. Three patients with large, macrocystic congenital pulmonary airway malformations (CPAMs) with evidence of worsening mediastinal shift and/or hydrops and 2 patients with large chylothorax with fetal hydrops were treated. Four cases had previous shunts that failed due to poor sonographic visualization during initial placement, cyst septations, shunt obstruction, or dislodgment. Needle fetal thoracoscopy was used to disrupt cyst walls and septations, clear hematoma, and confirm the optimal initial position of the shunt. In this series, 1 severe CPAM patient with a short cervix developed preterm labor postoperatively resulting in neonatal demise. The remaining 4 patients experienced resolution of hydrops and progressed to successful delivery with excellent neonatal outcomes.</p><p><strong>Conclusion: </strong>Needle fetal thoracoscopy is a procedure that may be selectively deployed in challenging thoracoamniotic shunt cases impacted by recurrent failure, poor sonographic windows, and challenging fetal positioning.</p>","PeriodicalId":12189,"journal":{"name":"Fetal Diagnosis and Therapy","volume":" ","pages":"1-7"},"PeriodicalIF":2.2,"publicationDate":"2024-05-15","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140916026","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}
引用次数: 0
Delivery Room Resuscitation of Infants with Congenital Diaphragmatic Hernia: Lessons Learned through Video Review. 产房抢救先天性膈疝婴儿:通过视频回顾吸取经验教训。
IF 2.2 3区 医学
Fetal Diagnosis and Therapy Pub Date : 2024-03-26 DOI: 10.1159/000538536
K Taylor Wild, Natalie Rintoul, Holly L Hedrick, Lauren Heimall, Leane Soorikian, Elizabeth E Foglia, Anne M Ades, Heidi M Herrick
{"title":"Delivery Room Resuscitation of Infants with Congenital Diaphragmatic Hernia: Lessons Learned through Video Review.","authors":"K Taylor Wild, Natalie Rintoul, Holly L Hedrick, Lauren Heimall, Leane Soorikian, Elizabeth E Foglia, Anne M Ades, Heidi M Herrick","doi":"10.1159/000538536","DOIUrl":"10.1159/000538536","url":null,"abstract":"<p><strong>Introduction: </strong>Delivery room (DR) interventions for infants with congenital diaphragmatic hernia (CDH) are not well described. This study sought to describe timing and order of DR interventions and identify system factors impacting CDH DR resuscitations using a human factors framework.</p><p><strong>Methods: </strong>Single center observational study of video recorded CDH DR resuscitations documenting timing and order of interventions. The team used the Systems Engineering Initiative for Patient Safety (SEIPS) model to identify system factors impacting DR resuscitations and time to invasive ventilation.</p><p><strong>Results: </strong>We analyzed 31 video recorded CDH resuscitations. We observed variability in timing and order of resuscitation tasks. The 'Internal Environment' and 'Tasks' components of the SEIPS model were prominent factors affecting resuscitation efficiency; significant room and bed spatial constraints exist, and nurses have a significant task burden. Additionally, endotracheal tube preparation was a prominent barrier to timely invasive ventilation.</p><p><strong>Conclusion: </strong>Video review revealed variation in event timing and order during CDH resuscitations. Standardization of room set-up, equipment, and event order and reallocation of tasks facilitate more efficient intubation and ventilation, representing targets for CDH DR improvement initiatives. This work emphasizes the utility of rigorous human factors review to identify areas for improvement during DR resuscitation.</p>","PeriodicalId":12189,"journal":{"name":"Fetal Diagnosis and Therapy","volume":" ","pages":""},"PeriodicalIF":2.2,"publicationDate":"2024-03-26","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140293193","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}
引用次数: 0
Differences between Myeloschisis and Myelomeningocele in patients undergoing prenatal repair of Open Spina Bifida. 接受开放性脊柱裂产前修复术的患者中脊髓脊膜膨出症和脊髓脑膜膨出症的区别。
IF 2.2 3区 医学
Fetal Diagnosis and Therapy Pub Date : 2024-03-12 DOI: 10.1159/000538099
José Miguel Müller, Edgardo Corral Sereño, Aura Jimenez, Rodrigo Zapata, Silvana Echeverria, Juan Pablo Jara, Alvaro Santibanez, Carolina Lindsay, Renatto Anfossi
{"title":"Differences between Myeloschisis and Myelomeningocele in patients undergoing prenatal repair of Open Spina Bifida.","authors":"José Miguel Müller, Edgardo Corral Sereño, Aura Jimenez, Rodrigo Zapata, Silvana Echeverria, Juan Pablo Jara, Alvaro Santibanez, Carolina Lindsay, Renatto Anfossi","doi":"10.1159/000538099","DOIUrl":"https://doi.org/10.1159/000538099","url":null,"abstract":"<p><strong>Introduction: </strong>Open spina bifida (OSB) manifests as myelomeningocele (MMC) or myeloschisis (MS). Both lesions theoretically leak cerebrospinal fluid (CSF) and produce different degrees of Chiari II malformation (CHMII). However, it is not entirely clear whether these forms of OSB have different clinical manifestations. This study aimed to evaluate the clinical and/or radiological differences between myeloschisis and myelomeningocele in patients who underwent prenatal OSB repair.</p><p><strong>Methods: </strong>A total of 71 prenatal repairs were performed with the open technique at the Public Hospital of Rancagua, Chile, between 2012 and 2022. We performed follow-up MRI imaging of fetuses that qualified for prenatal OSB repair surgery. We examined the correlations between various anthropomorphic measurements and clinical and imaging variables, such as the type of lesion and dimensions such as ventricle atrium diameter, degree of severity of CHMII, need for CSF shunt at 12 months, and walking at 30 months.</p><p><strong>Results: </strong>This study included 71 fetuses with OSB for which 38 MRI examinations were analyzed; 61% (43/71) of lesions were MMC and 39% (28/71) were MS. Grade 3 (severe) CHMII were found in 80% (12/15) of MS and 43% (10/23) of MMC (p<0.05). Fetuses with an atrial diameter less than 13.48 mm had a lower probability of requiring a CSF shunt at 12 months (p<0.05). MMC was associated with a higher frequency of clubfoot at birth (p<0.05), whereas MS was significantly associated with more severe CHMII (p<0.05). Although the correlations were not significant, we observed clear trends that more children with MS required shunts at 12 months and could walk at 30 months compared to children with MMC.</p><p><strong>Conclusions: </strong>MS and MMC are distinct subtypes of OSB. Further studies of larger cohorts that include biomolecular and histological analysis are required to better understand differences between these lesions. This study may enable healthcare providers to better advise parents and prepare healthcare teams earlier for the management of patients undergoing prenatal repair of OSB.</p>","PeriodicalId":12189,"journal":{"name":"Fetal Diagnosis and Therapy","volume":" ","pages":""},"PeriodicalIF":2.2,"publicationDate":"2024-03-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140109780","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}
引用次数: 0
Open Fetal Surgery for Ventricular-Amniotic Valve Implantation in Aqueductal Stenosis-Dependent Severe Fetal Hydrocephalus: A Case Report with 7-Year Follow-Up. 在导水管狭窄依赖性重度胎儿脑积水中进行开放胎儿手术以植入心室-羊膜瓣--一例随访 7 年的病例报告。
IF 2.2 3区 医学
Fetal Diagnosis and Therapy Pub Date : 2024-01-01 Epub Date: 2024-02-17 DOI: 10.1159/000536121
Mateusz Zamłyński, Anita Olejek, Tomasz Koszutski, Janusz Bohosiewicz, Marek Mandera, Jacek Zamłyński, Iwona Maruniak-Chudek, Izabela Herman-Sucharska, Agnieszka Pastuszka
{"title":"Open Fetal Surgery for Ventricular-Amniotic Valve Implantation in Aqueductal Stenosis-Dependent Severe Fetal Hydrocephalus: A Case Report with 7-Year Follow-Up.","authors":"Mateusz Zamłyński, Anita Olejek, Tomasz Koszutski, Janusz Bohosiewicz, Marek Mandera, Jacek Zamłyński, Iwona Maruniak-Chudek, Izabela Herman-Sucharska, Agnieszka Pastuszka","doi":"10.1159/000536121","DOIUrl":"10.1159/000536121","url":null,"abstract":"<p><strong>Introduction: </strong>Fetal ventriculomegaly is one of the most commonly diagnosed central nervous system pathologies of the second trimester, occurring with a frequency of 0.3-0.5/1,000 births. Severe fetal ventriculomegaly (SVM) may necessitate intrauterine intervention. Most such interventions have been made percutaneously with ultrasound guidance insertion of a pigtail catheter, which sadly often became obstructed or migrated.</p><p><strong>Case presentation: </strong>Our case report presents the possibility of ventriculo-amniotic valve implantation (VAVI) by classic hysterotomy in isolated severe fetal hydrocephalus (IVSM) due to aqueductal stenosis. The patient was operated on similarly to open fetal surgery MOMS criteria at 24+4/7 GA, with an initial lateral ventricular dimension of 22.5 mm. A female newborn was delivered by elective cesarean section at 31+1/7 GA due to PPROM (Apgar 10' 8 points, birth weight 1,600 g), required CPAP, and removal of the drainage system due to infection and narrow lateral ventricles. Evans index (EI) gradual increase and clinical symptoms of high-pressure hydrocephalus after 10 days required a ventricle-peritoneal shunt (VPS) implantation. The newborn was discharged home after 28 days with stabile hydrocephalus (EI: 0.59-0.6), in good clinical condition. The 7-year follow-up was complicated by epilepsy, VPS shunt infections, delay in motor and intellectual functions (mild to moderate), and symptoms of atypical autism, the phenotype possibly related to a variant in ZEB2 gene.</p><p><strong>Conclusion: </strong>Intrauterine VAVI is a one-step procedure that is effective in draining CFS. The limitations of the method remain complications due to preterm labor and infection of the drainage system.</p>","PeriodicalId":12189,"journal":{"name":"Fetal Diagnosis and Therapy","volume":" ","pages":"278-284"},"PeriodicalIF":2.2,"publicationDate":"2024-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"139899604","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}
引用次数: 0
Predictors of Poor Outcomes in Monochorionic Diamniotic Twin Pregnancies Complicated by Selective Fetal Growth Restriction. 单绒毛膜双羊膜妊娠并发选择性胎儿生长受限不良预后的预测因素。
IF 2.2 3区 医学
Fetal Diagnosis and Therapy Pub Date : 2024-01-01 Epub Date: 2024-02-17 DOI: 10.1159/000537861
Shelly Soni, Juliana Gebb, Kendra Miller, Edward R Oliver, Christina Paidas Teefey, Julie S Moldenhauer, Nahla Khalek
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