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Congenital pulmonary airway malformation

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https://www.readbyqxmd.com/read/29328793/novel-molecular-and-phenotypic-insights-into-congenital-lung-malformations
#1
Daniel T Swarr, William H Peranteau, Jennifer Pogoriler, David B Frank, N Scott Adzick, Holly L Hedrick, Mike Morley, Su Zhou, Edward E Morrisey
RATIONALE: Disruption of normal pulmonary development is a leading cause of morbidity and mortality in infants. Congenital lung malformations are a unique model to study the molecular pathogenesis of isolated structural birth defects as they are often surgically resected. OBJECTIVES: To provide insight into the molecular pathogenesis of congenital lung malformations through analysis of cell-type and gene expression changes in these lesions. METHODS: Clinical data, and lung tissue for DNA, RNA, and histology were obtained from 58 infants undergoing surgical resection of a congenital lung lesion...
January 12, 2018: American Journal of Respiratory and Critical Care Medicine
https://www.readbyqxmd.com/read/29274836/outcomes-in-children-undergoing-surgery-for-congenital-pulmonary-airway-malformations-in-the-first-year-of-life
#2
Katerina Dukleska, Erin Teeple, Scott Cowan, Charles Vinocur, Loren Berman
BACKGROUND: Treatment of congenital pulmonary airway malformations (CPAMs) is generally surgical resection; however, there is controversy regarding the optimal timing of surgical intervention, especially in asymptomatic patients. STUDY DESIGN: Using the American College of Surgeons National Surgical Quality Improvement Program-Pediatric Participant Use Files from 2012-2015, children who underwent lung resection for CPAM were identified. Outcomes in children who underwent lung resection during the neonatal period were compared with those who underwent resection beyond the neonatal period, but during the first year of life (non-neonates)...
December 21, 2017: Journal of the American College of Surgeons
https://www.readbyqxmd.com/read/29274787/expert-surgical-consensus-for-prenatal-counseling-using-the-delphi-method
#3
Loren Berman, Jordan Jackson, Kristen Miller, Rebecca Kowalski, Paul Kolm, Francois I Luks
BACKGROUND: Pediatric surgeons frequently offer prenatal consultation for congenital pulmonary airway malformation (CPAM) and congenital diaphragmatic hernia (CDH); however, there is no evidence-based consensus to guide prenatal decision making and counseling for these conditions. Eliciting feedback from experts is integral to defining best practice regarding prenatal counseling and intervention. METHODS: A Delphi consensus process was undertaken using a panel of pediatric surgeons identified as experts in fetal therapy to address current limitations...
November 28, 2017: Journal of Pediatric Surgery
https://www.readbyqxmd.com/read/29207383/thoracic-changes-after-full-laser-ablation-of-the-feeding-artery-in-fetuses-with-bronchopulmonary-sequestration
#4
Rogelio Cruz-Martínez, Belén Nieto-Castro, Miguel Martínez-Rodríguez, Alma Gámez-Varela, Edgar Ahumada-Angulo, Jonahtan Luna-García, Maria José Pineda-Pérez, Carlos Rebolledo-Fernández
OBJECTIVE: To assess longitudinal intrathoracic changes after fetal laser surgery in fetuses with bronchopulmonary sequestration (BPS) with hydrops and/or hydrothorax. STUDY DESIGN: The presence of intrafetal fluid effusions, the lung mass volume ratio (congenital pulmonary airway malformation volume ratio [CVR]), and the observed/expected lung-to-head circumference ratio (O/E-LHR) of both lungs were evaluated in a cohort of BPS fetuses with hydrops and/or hydrothorax treated with full laser ablation of the feeding artery (FLAFA)...
December 6, 2017: Fetal Diagnosis and Therapy
https://www.readbyqxmd.com/read/29206544/a-complication-of-percutaneous-sclerotherapy-for-congenital-pulmonary-airway-malformation-intravascular-injection-and-cardiac-necrosis
#5
Andrew H Chon, Moe R Takeda, Juan C Felix, Ramen H Chmait
INTRODUCTION: A congenital pulmonary airway malformation (CPAM) type III may become large enough to cause hydrops fetalis. In such circumstances, the fetus can be treated with open fetal resection, maternal betamethasone administration, or percutaneous sclerotherapy. CASE REPORT: A 24 week gestation fetus with a CPAM type III was treated by percutaneous sclerotherapy using ethanolamine oleate (EO). The EO inadvertently entered the left atrium and ventricle with subsequent fetal bradycardia and demise...
December 2017: Fetal and Pediatric Pathology
https://www.readbyqxmd.com/read/29197847/perforated-neuroenteric-cyst-masquerading-as-congenital-pulmonary-airway-malformation
#6
Sreekar Gundapaneni, Vishesh Jain, Shilpa Sharma, Devendra Kumar Gupta
A 3-month-old child was presented with haemoptysis with respiratory distress. Imaging was suggestive of a cavitary lesion in the lung with surrounding consolidation. Diagnosis of a primary lung pathology like congenital pulmonary airway malformation was considered. Based on clinical suspicion and prior experience, a Tc-99m pertechnetate radionuclide study was performed, which clinched the diagnosis of foregut duplication cyst. Intraoperative findings confirmed the presence of a neuroenteric cyst. The child remains asymptomatic on follow-up awaiting neurosurgical intervention for the intraspinal component of the cyst...
December 2, 2017: BMJ Case Reports
https://www.readbyqxmd.com/read/29196881/gata-6-expression-is-decreased-in-diaphragmatic-and-pulmonary-mesenchyme-of-fetal-rats-with-nitrofen-induced-congenital-diaphragmatic-hernia
#7
Toshiaki Takahashi, Florian Friedmacher, Julia Zimmer, Prem Puri
PURPOSE: Congenital diaphragmatic hernia (CDH) and associated pulmonary hypoplasia are thought to be caused by a malformation of the underlying diaphragmatic and airway mesenchyme. GATA binding protein 6 (Gata-6) is a zinc finger-containing transcription factor that plays a crucial role during diaphragm and lung development. In the primordial diaphragm, Gata-6 expression is restricted to mesenchymal compartments of the pleuroperitoneal folds (PPFs). In addition, Gata-6 is essential for airway branching morphogenesis through upregulation of mesenchymal signaling...
December 2, 2017: Pediatric Surgery International
https://www.readbyqxmd.com/read/29115093/a-case-of-multiple-cardiovascular-and-tracheal-anomalies-presented-with-wolff-parkinson-white-syndrome-in-a-middle-aged-adult
#8
Hyejin Shi, Sungmin Sohn, SungHo Wang, Sungrock Park, SangKi Lee, Song Yi Kim, Sun Young Jeong, Changhwan Kim
Congenital cardiovascular anomalies, such as dextrocardia, persistent left superior vena cava (SVC), and pulmonary artery (PA) sling, are rare disorders. These congenital anomalies can occur alone, or coincide with other congenital malformations. In the majority of cases, congenital anomalies are detected early in life by certain signs and symptoms. A 56-year-old man with no previous medical history was admitted due to recurrent wide QRS complex tachycardia with hemodynamic collapse. A chest radiograph showed dextrocardia...
December 2017: Journal of Korean Medical Science
https://www.readbyqxmd.com/read/29108843/outcomes-following-elective-resection-of-congenital-pulmonary-airway-malformations-are-equivalent-after-3-months-of-age-and-a-weight-of-5-kg
#9
Brian C Gulack, Harold J Leraas, Brian Ezekian, Jina Kim, Christopher Reed, Obinna O Adibe, Henry E Rice, Elisabeth T Tracy
PURPOSE: Resection of congenital pulmonary airway malformations (CPAMs) is often performed to reduce the risk of recurrent infection and malignant transformation. However, there is substantial variation in the timing of resection. This study was performed to determine the association of age and weight on outcomes following elective resection of CPAMs. METHODS: The American College of Surgeons National Surgical Quality Improvement Program-Pediatric database from 2012 to 2014 was queried for infants undergoing elective resection of a CPAM...
October 9, 2017: Journal of Pediatric Surgery
https://www.readbyqxmd.com/read/29099644/medium-term-pulmonary-function-test-after-thoracoscopic-lobectomy-for-congenital-pulmonary-airway-malformation-a-comparative-study-with-normal-control
#10
Chin Tung Lau, Kenneth K Y Wong, Paul Tam
INTRODUCTION: Congenital pulmonary airway malformation (CPAM) is a major indication of lobectomy in children. Early lobectomy had been proposed for the advantage of compensatory lung growth. Despite the increasing use of thoracoscopic lobectomy its effect on postoperative lung function was still not well established in the literature. This study was therefore performed to study the result of postoperative pulmonary function test (PFT) on a medium term basis. MATERIALS AND METHODS: All patients who underwent thoracoscopic lobectomy for CPAM between 2006 and 2010 were recruited into the study...
November 3, 2017: Journal of Laparoendoscopic & Advanced Surgical Techniques. Part A
https://www.readbyqxmd.com/read/29046943/congenital-pulmonary-airway-malformations-state-of-the-art-review-for-pediatrician-s-use
#11
REVIEW
Claire Leblanc, Marguerite Baron, Emilie Desselas, Minh Hanh Phan, Alexis Rybak, Guillaume Thouvenin, Clara Lauby, Sabine Irtan
Congenital pulmonary airway malformations or CPAM are rare developmental lung malformations, leading to cystic and/or adenomatous pulmonary areas. Nowadays, CPAM are diagnosed prenatally, improving the prenatal and immediate postnatal care and ultimately the knowledge on CPAM pathophysiology. CPAM natural evolution can lead to infections or malignancies, whose exact prevalence is still difficult to assess. The aim of this "state-of-the-art" review is to cover the recently published literature on CPAM management whether the pulmonary lesion was detected during pregnancy or after birth, the current indications of surgery or surveillance and finally its potential evolution to pleuro-pulmonary blastoma...
December 2017: European Journal of Pediatrics
https://www.readbyqxmd.com/read/29028157/transthoracic-catheter-drainage-for-large-symptomatic-congenital-pulmonary-airway-malformation
#12
Seong Hee Oh, Chae Young Kim, Byong Sop Lee, Dong Kwan Kim, Ellen Ai-Rhan Kim, Ki-Soo Kim
BACKGROUND: Surgical resection of large symptomatic congenital pulmonary airway malformation (CPAM) in newborns has high risks of mortality and postoperative morbidity. This study aimed to report the clinical outcomes of newborns who underwent percutaneous transthoracic catheter drainage (PTCD) of large symptomatic CPAM before surgical resection. METHODS: This was a retrospective, descriptive study based on review of the medical records of newborn infants who required surgical resection of large symptomatic CPAM at a single tertiary hospital from 2001 to 2017...
December 2017: Pediatric Pulmonology
https://www.readbyqxmd.com/read/28974162/antenatal-assessment-and-postnatal-outcome-of-fetal-echogenic-lung-lesions-a-decade-s-experience-at-a-tertiary-referral-hospital
#13
Stefan C Kane, Fabrício Da Silva Costa, Joseph A Crameri, Karen L Reidy, Helen Kaganov, Ricardo Palma-Dias
INTRODUCTION: Fetal echogenic lung lesions (ELL) are the commonest pulmonary pathology diagnosed on antenatal sonography, and include congenital pulmonary airway malformations (CPAMs) and bronchopulmonary sequestrations. This study aimed to evaluate the predictive utility of the CPAM volume ratio (CVR) at presentation in a series of fetuses with ELLs at a tertiary Australian referral hospital. MATERIAL AND METHODS: Retrospective cohort study of all pregnancies with a prenatal diagnosis of an isolated fetal echogenic lung lesion managed at the Royal Women's Hospital, Victoria, Australia, between 2005 and 2015...
October 16, 2017: Journal of Maternal-fetal & Neonatal Medicine
https://www.readbyqxmd.com/read/28960660/conservative-post-natal-management-of-antenatally-diagnosed-congenital-pulmonary-airway-malformations
#14
Allya V Makhijani, Flora Y Wong
AIM: Management of congenital pulmonary airway malformations (CPAM) is controversial, especially for asymptomatic patients. We aim to describe the clinical manifestations and management of CPAM at a tertiary paediatric hospital using a retrospective audit. METHODS: Infants with CPAM were identified on the Fetal Diagnostic Unit database from 2007 to 2014. Information on antenatal and post-natal management was collected from medical record. RESULTS: Thirty-five infants with antenatally diagnosed CPAM were included...
September 28, 2017: Journal of Paediatrics and Child Health
https://www.readbyqxmd.com/read/28948063/fetal-thoracoamniotic-shunting-in-a-case-of-congenital-pulmonary-airway-malformations-with-hydrops-fetalis
#15
Hayase Nitta, Yusuke Taira, Tadatsugu Kinjo, Yukiko Chinen, Hitoshi Masamoto, Naoya Sanabe, Hideki Goya, Tomohide Yoshida, Rika Sugibayashi, Masahiro Sumie, Seiji Wada, Haruhiko Sago, Yoichi Aoki
Aim  We report a case of congenital pulmonary airway malformation (CPAM) with hydrops in which the fetus underwent thoracoamniotic shunting. Case Report  A 40-year-old (G1P1) woman was diagnosed with a macrocystic CPAM. Thoracoamniotic shunting was performed at 19 weeks of gestation but not well drained and was successfully performed again at 23 weeks. However, the CPAM volume ratio, abdominal circumference, and amniotic fluid index started increasing from 28 weeks and hydrops worsened. The insufficient shunting and the fetal cardiac failure had to be considered...
July 2017: American Journal of Perinatology Reports
https://www.readbyqxmd.com/read/28915526/-exit-a-possible-intervention-for-new-and-earlyborn-babies-with-severe-hydrops-fetalis-and-hydrothoraces-on-both-sides
#16
Sandra Koch, Jochen Essers, Ortraud Beringer, Frank Reister, Helmut Hummler, Anja Moewes
The EXIT (ex utero intrapartum treatment) procedure is an established method of respiratory protection, originally used in the delivery of fetuses with congenital obstructive airway diseases (tumors in the throat area, hygromas, so-called congenital high airway obstruction syndrome (CHAOS)). Meanwhile, the procedure is also carried out in large perinatal centers for pronounced diaphragmatic hernia or other special indications (EXIT to ECMO, congenital lung airway malformations (CCAM), pulmonary atresia). We present our experience with adapted EXIT procedures in 5 preterm infants with secondary generalized hydrops fetalis and pronounced bilateral hydrothoraces...
December 2017: Zeitschrift Für Geburtshilfe und Neonatologie
https://www.readbyqxmd.com/read/28873355/pulmonary-interstitial-glycogenosis-associated-with-a-spectrum-of-neonatal-pulmonary-disorders
#17
Ernest Cutz, Rose Chami, Sharon Dell, Jacob Langer, David Manson
Primary or isolated pulmonary interstitial glycogenosis (PIG) is a rare disease presenting as tachypnea and hypoxemia during the perinatal period. A diffuse interstitial infiltrate with focal hyperinflation is visible on chest imaging. The biopsy findings include diffuse expansion of the interstitium by spindle-shaped cells with pale cytoplasm that, on electron microscopy (EM), are poorly differentiated mesenchymal cells containing abundant monoparticulate glycogen. This glycogenosis appears to be a transient abnormality, usually with a favorable prognosis...
October 2017: Human Pathology
https://www.readbyqxmd.com/read/28812458/congenital-cystic-lung-lesions-evolution-from-in-utero-detection-to-pathology-diagnosis-a-multidisciplinary-approach
#18
Steven Hardee, Lea Tuzovic, Cicero T Silva, Robert A Cowles, Joshua Copel, Raffaella A Morotti
Congenital cystic lung lesions are a group of rare pathologies that are usually diagnosed in the prenatal period. The majority of these lesions are diagnosed at pathology examination as congenital pulmonary airway malformations (CPAM) and bronchopulmonary sequestration (BPS). These lesions are typically managed by surgical intervention within the first year of life and have an excellent prognosis. We examined the evolution of imaging appearances from prenatal diagnosis to postnatal work-up of these lesions and correlate imaging and pathological findings...
September 2017: Pediatric and Developmental Pathology
https://www.readbyqxmd.com/read/28753246/outcome-of-prenatally-diagnosed-congenital-lung-anomalies-in-the-north-of-england-a-review-of-228-cases-to-aid-in-prenatal-counselling
#19
Lesley Walker, Kelly Cohen, Judith Rankin, David Crabbe
OBJECTIVE: To describe data on congenital lung anomalies identified on prenatal ultrasound from two centres in the North of England. METHOD: This retrospective case series includes all cases notified to the Northern Congenital Abnormality Survey from 1990 to 2010 and to Leeds Regional Fetal Medicine Unit 2000-2015. RESULTS: There were a total of 228 cases, 101 from Northern Congenital Abnormality Survey and 127 from Leeds Regional Fetal Medicine Unit...
October 2017: Prenatal Diagnosis
https://www.readbyqxmd.com/read/28737279/exercise-capacity-is-not-decreased-in-children-who-have-undergone-lung-resection-early-in-life-for-congenital-thoracic-malformations-compared-to-healthy-age-matched-children
#20
Ashlee Dunn, Kasey Pearce, Robin Callister, Adam Collison, Matthew Morten, Payal Mandaliya, Lauren Platt, Ben Dascombe, Rajendra Kumar, Hiran Selvadurai, Paul D Robinson, Joerg Mattes
PURPOSE: The purpose of this study was to compare (i) the exercise capacity and (ii) lung function prior to and immediately post cardiopulmonary exercise tests (CPET) of children who underwent early life lung resection for Congenital Pulmonary Airway Malformations (CPAM) to healthy control children. METHOD: Eight children with CPAM (four males, age 9.6 ± 1.8 years) and eight control children without respiratory disease (three males, age 9.4 ± 1.4 years) performed a CPET on a cycle ergometer, during which maximal oxygen consumption (V̇O2max ) and heart rate were measured...
October 2017: Pediatric Pulmonology
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