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The application of aortopulmonary shunt in cyanotic congenital heart disease with severe pulmonary hypoplasia in adolescents and adults

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South China Journal of Cardiology 2020年第2期21卷 104-111页

作者： HE Biao-chuan ZHAO Jun-fei TENGYun CHEN Ji-mei Department of Cardiovascular Surgery Guangdong Provincial People's HospitalGuangdong Cardiovascular InstituteGuangdong Provincial Key Laboratory of South China Structural Heart DiseaseGuangdong Academy of Medical SciencesGuangzhou 510080China

Background pulmonary arterial hypoplasia is a common complication in complex cyanotic congenital heart disease. For extreme stenosis of the branch pulmonary arteries,which is not feasible for complete repair,palliative systemic-pulmonary shunt is needed to increase pulmonary blood flow and pulmonary tree growth as early as beyond 2 years old. Unfortunately,due to poor medical setting in local hospitals,there are still a small number of patient with severe hypoplastic pulmonary arteries in developing area who have failed to undergo appropriate surgical intervention till to teenagers even adults. In order to explore the resolution of the dilemma for these notable cyanotic patients,hence we began to utilize three-dimensional computed tomography(CT)to reconstruct pulmonary artery anatomy and to simulate virtual palliative systemic pulmonary shunt conduit module,to facilitate and improve the intraoperative aortopulmonary shunt performance. Methods FromApril 2011 to August 2018,13 consecutive patients undergoing aortopulmonary shunt with older age(13-35 years old)who missed the optimal timing for surgery were identified from 196 cases involving palliative systemic pulmonary shunt. An individually pre-designed prosthetic expanded poly tetra fluoroethylene(ePTFE)conduit was utilized based on the threedimensional enhanced computed tomography reconstruction and simulation. The post-operative recovery courses and complications were documented. Blood gas analysis,electrocardiogram,echocardiography were performed routinely prior to discharge and compared with the preoperative data. Re-evaluation of finger pulse saturation(SpO2),echocardiography and electrocardiogram was performed in clinical follow-up in 1 st month,6 th month,12 th month and every year postoperatively. A retrospective analysis of operative data,postoperative outcomes and complications were performed. Results All the 13 consecutive patients underwent successfully non-cardiopulmonary bypass systemic-pulmonary shun

关键词： pulmonary hypoplasia cyanosis aortopulmonary shunt palliation complex congenital heart disease adolescents and adults

Antenatal use of bosentan and/or sildenafil attenuates pulmonary features in rats with congenital diaphragmatic hernia

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World Journal of Pediatrics 2014年第4期10卷 354-359页

作者： María de Lourdes Lemus-Varela Amed Soliz Belinda Claudia Gómez-Meda Ana Lourdes Zamora-Perez José Manuel Ornelas-Aguirre Valery Melnikov Blanca Miriam Torres-Mendoza Guillermo Moisés Zúñiga-González Division of Neonatology High Specialty Medical UnitPediatric HospitalWestern National Medical CenterMexican Social Security InstituteGuadalajaraJaliscoMéxico Neonatal Intensive Care Unit Division of NeonatologyMiami Children's HospitalMiami FLDUSA Molecular Biology in Medicine Institute Molecular Biology and Genomic DepartmentHealth Sciences University CenterUniversity of GuadalajaraGuadalajaraJaliscoMéxico Odontology Research Institute Integrated Odontology Clinic DepartmentHealth Sciences University CenterUniversity of GuadalajaraGuadalajaraJaliscoMéxico Pathology Department High Specialty Medical UnitNorth-western National Medical CenterMexican Social Security InstituteCiudad ObregónSonoraMéxico School of Medicine University of ColimaColimaMéxico Division of Neurosciences Western Biomedical Research CenterMexican Social Security Institute and Department of ClinicsHealth Sciences University CenterUniversity of GuadalajaraGuadalajaraJaliscoMéxico Mutagenesis Laboratory Western Biomedical Research CenterMexican Social Security InstituteGuadalajaraJaliscoMéxico

Background:Lung hypoplasia,pulmonary persistent hypertension of the newborn and its morphological changes are the main features in congenital diaphragmatic hernia(CDH).This study was undertaken to investigate if antenatal use of sildenafil and/or bosentan attenuates vascular remodeling,promotes branching,and improves alveolarization in experimental nitrofeninduced ***:Nitrofen(100 mg)was gavage-fed to pregnant rats at post conception day(PCD)9 to induce *** rats were randomized to 5 groups:1)control;2)nitrofen;3)nitrofen+sildenafil 100 mg/kg per day at PCD 16-20;4)nitrofen+bosentan 30 mg/kg per day,at PCD 16-20,and 5)nitrofen+bosentan+sildenafil,same doses and administration *** cesarean delivery,the offsprings were sacrifi *** diaphragmatic defect and pulmonary hypoplasia were identified,and the lungs were *** wall thickness,bronchiolar density and alveolarization were ***:The offsprings with CDH were characterized by severe pulmonary hypoplasia(lung weight-to-body weight ratio:0.0263[95%confidence interval(CI)0.0242-0.0278)]in the nitrofen group versus 0.0385(95%CI 0.0355-0.0424)in the control group(P=0.0001).pulmonary arterial wall thickness was decreased to 3.0(95%CI 2.8-3.7)μm in the nitrofen+sildenafil group versus 5.0(95%CI 4.1-4.9)μm in the nitrofen group(P=0.02).Terminal bronchioles increased to 13.7(95%CI 10.7-15.2)μm in the nitrofen+bosentan group in contrast to 8.7(95%CI 7.2-9.4)μm in the nitrofen group(P=0.002).More significant differences(P=0.0001)were seen in terminal bronchioles in the nitrofen+sildenafil+bosentan group than in the nitrofen group[14.0(95%CI 12.5-15.4)μm versus 8.5(95%CI 7.1-9.3)μm].pulmonary arterial wall thickness was also decreased in the former ***:In this rat model,antenatal treatment with sildenafil attenuates vascular *** promotes the development of terminal bronchioles in nitrofen-induced CDH.

关键词： antenatal treatment bosentan congenital diaphragmatic hernia pulmonary hypoplasia sildenafil

Nitrofen suppresses cell proliferation and promotes mitochondria-mediated apoptosis in type Ⅱ pneumocytes

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Acta Pharmacologica Sinica 2007年第5期28卷 672-684页

作者： Qiang-song TONG~(2,5),Li-duan ZHENG~3,Shao-tao TANG~2,Guo-song JIANG~2,Qing-lan RUAN~2,Fu-qing ZENG~2,Ji-hua DONG~4~2 Department of Surgery,~3 Department of Pathology,and ~4 Department of Central Laboratory,Union Hospital of Tongji Medical College,Huazhong University of Science and Technology,Wuhan 430022,China Department of Surgery Union Hospital of Tongji Medical College Huazhong University of Science and Technology Wuhan China Department of Pathology Union Hospital of Tongji Medical College Huazhong University of Science and Technology Wuhan China Department of Central Laboratory Union Hospital of Tongji Medical College Huazhong University of Science and Technology Wuhan China

Aim:To characterize the molecular mechanisms of nitrofen-induced ***:After administration of nitrofen to cultured type Ⅱ A549pneumocytes,cell proliferation and DNA synthesis were investigated by 3-(4,5-dimethylthiazol-2-yl)-2,5-diphenyl tetrazolium bromide colorimetry,colony forma-tion assay,flow cytometry and[~3H]-thymidine incorporation *** measured by terminal transferase-mediated dUTP nick-end-labeling,acridineorange-ethidium bromide staining and flow *** of proliferatingcell nuclear antigen (PCNA) and apoptosis-related genes was assayed byimmunofluorescence,RT-PCR and Western ***:Nitrofen inhibited thecell proliferation of A549 cells in a dose-and time-dependent manner,accompa-nied by downregulation of *** a result,the DNA synthesis of nitrofen-treated A549 cells decreased,while cell cycle was arrested at G0/***,nitrofen induced apoptosis of A549 cells,which was not abolished by Z-Val-Ala-Asp(OCH3)-*** addition,nitrofen decreased the expressionof Bcl-xL,but not of Bcl-2,Bax,and Bak,resulting in a loss of mitochondrialmembrane potential and the nuclear translocation of apoptosis-inducing factor(AIF).Meanwhile,nitrofen strongly activated the p38 mitogen-activated proteinkinase (p38-MAPK).Pretreatment of cells with SB203580 (5 μmol/L) blockednitrofen-induced phosphorylation of p38-MAPK and abolished nitrofen-inducedAIF translocation and apoptosis in A549 ***:Nitrofen suppressesthe proliferation of cultured type Ⅱ pneumocytes accompanied by thedownregulation of PCNA,and induces mitochondria-mediated apoptosis involv-ing the activation of p38-MAPK.

关键词： nitrofen pulmonary hypoplasia proliferating cell nuclear antigen apoptosis-inducing factor

Congenital Hernia of the Diaphragmatic Dome with Neonatal Revelation, Experience from the Neonatology and Neonatal Intensive Care Unit at the University Hospital Center of Oujda

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Open Journal of Pediatrics 2024年第5期14卷 790-799页

作者： Addou Bebana Anass Ayyad Mohammed Ec-Chebab Sahar Messaoudi Rim Amrani Pediatrics/Residency Mohammed VI University Hospital Oujda Morocco Department of Neonatology and Neonatal Intensive Care Unit Mohammed VI University Hospital Medical School Mohammed First University Oujda Morocco The Maternal-Child & Mental Health Research Laboratory Oujda Morocco

Congenital hernia of the diaphragmatic dome (CHDD) is an embryonic malformation in which all or part of the diaphragmatic dome fails to develop properly. In the majority of cases (80% to 90%), this malformation affects the left posterolateral part of the diaphragm, while in 10% to 15% of cases it affects the right. Bilateral cases are extremely rare, accounting for less than 1% of cases. This malformation is estimated to occur at a frequency of around 1 in 3500 births, with a male predominance. The diaphragmatic defect causes the abdominal organs to rise into the thoracic cavity during critical phases of lung development. These anomalies result in bilateral pulmonary hypoplasia, a reduced number of pulmonary vessels, and pulmonary arterial hypertension (PAH). The combination of these anatomical and functional anomalies, in varying degrees, explains the wide variability of symptoms at birth. Diagnosis is usually made prenatally by ultrasound, which enables severe forms of the disease to be detected and appropriate management initiated. The prognosis remains generally grave, with a neonatal mortality rate of between 30% and 60% depending on the study, and around half of all children will have long-term sequelae.

关键词： Congenital Diaphragmatic Hernia Prenatal Diagnosis pulmonary hypoplasia pulmonary Hypertension Medical-Surgical Treatment

A Typical Case of Classic Potter’s Syndrome: A Case Report

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Open Journal of Pediatrics 2024年第3期14卷 482-487页

作者： Mohamed El Kerim Mahy Mohammed Ech-Chebab Anass Ayyad Sahar Messaoudi Rim Amrani Pediatrics/Residency Mohammed VI University Hospital Oujda Morocco Department of Neonatology and Neonatalintensive Care Unit Mohammed VI University Hospital Medical School University Mohammed 1st Oujda Morocco The Maternal-Child & Mental Health Research Laboratory Oujda Morocco

Potter syndrome is a rare congenital malformation that primarily affects male fetuses;it is characterized by pulmonary hypoplasia, skeletal malformation, and kidney abnormalities. The pressure of the uterine wall due to oligohydramnios leads to an unusual facial appearance, abnormal limbs in abnormal positions, or contractures. The fetus generally dies soon after birth due to respiratory insufficiency. The baby was a live preterm male, born to a 30-year-old multigravida, out of a non-consanguineous marriage via cesarean section. There was no liquor at the time of delivery. The baby did not cry immediately after birth and required resuscitation, followed by mechanical ventilation. Multiple congenital anomalies suggestive of Potter’s syndrome were noted including facial features, flattened nose, low protruding ear, retrognathism, and epicanthal folds with unilateral atresia of the choana. Chest X-ray showed small volume lung fields suggestive of pulmonary hypoplasia, and we had on ultrasonography bilateral polycystic kidney disease on ultrasonography. At 42 hours of life, the baby developed tachypnea and severe chest retractions and died due to respiratory insufficiency. Our case highlights the importance of regular prenatal checks and examinations in each pregnancy, which helps to collect suspected cases and improve knowledge of this syndrome for better management.

关键词： Potter Syndrome pulmonary hypoplasia Potter’s Facies Polycystic Kidney Oligohydramnios

MR ASSESSMENT OF LUNG DEVELOPMENT IN FETAL ANOMALIES USING RATIO OF LUNG TO LIVER SIGNAL INTENSITIES

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Journal of Shanghai Second Medical University(Foreign Language Edition) 2007年第2期19卷 151-153页

作者：刘明张玉珍冯赟李玉华 Deparment of Radiology Xinhua Hospital School of Medicine Shanghai Jiaotong University

Objective To evaluate the monitoring and diagnostic potential of MR in fetal lung development using lung signal intensity changes in the fetus. Methods The lung-to-liver intensity ratio was calculated by means of region-of-interest analysis in all 35 fetuses including 7 bilateral urinary anomalies, 9 unilateral urinary anomalies, 5 pulmonary anomalies, and 14 others. Results All fetuses with urinary anomalies and pulmonary anomalies showed low ratio of lung-to-liver signal intensity. Conclusion Low ratio of lung-to-liver signal intensity on MR imaging indicates pulmonary hypoplasia after 22 weeks’ gestation.

关键词： urinary malformation pulmonary hypoplasia MR fetus