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Congenital pulmonary airway malformation

Hemanshoo Sudhir Thakkar, Jonathan Durell, Subhasis Chakraborty, Bianca-Lea Tingle, Arnwald Choi, Darren J Fowler, Steve J Gould, Lawrence Impey, Kokila Lakhoo
Congenital airway pulmonary malformations are increasingly being diagnosed, but their management continues to remain controversial. Our approach has been to offer surgery to mitigate the risk of infection and possible malignancy. All patients routinely undergo a CT scan of the chest postnatally and once the diagnosis is confirmed, minimal access surgery is offered. Our anesthetists provide single-lung ventilation to enhance the operative view. We conducted a retrospective review over a 10-year period, during which 91 patients were prenatally suspected to have a cystic lung lesion...
October 10, 2016: European Journal of Pediatric Surgery
Mohannad Abu Omar, Emily Tylski, Mouhanna Abu Ghanimeh, Ashraf Gohar
Congenital pulmonary airway malformation (CPAM) is a rare congenital abnormality with unknown exact aetiology or clear genetic association. It is characterised by a failure of bronchial development and localised glandular overgrowth. Typically, it is diagnosed on prenatal ultrasound, only infrequently in children, and even less commonly in adults. We present a case of a 25-year-old man, with no previous lung diseases who presented with right-sided chest pain, fever and cough suggestive of pulmonary infection...
September 26, 2016: BMJ Case Reports
Gopal Singh, Amy Coffey, Robert Neely, Daniel Lambert, Joshua Sonett, Alain C Borczuk, Lyall Gorenstein
Congenital pulmonary airway malformation (CPAM) is a developmental abnormality of the lung, which results from an abnormality of branching during fetal development of the lung. We report the case of an 18 year-old woman who developed Kirsten rat sarcoma virus (KRAS) mutation positive mucinous adenocarcinoma of the lung (AC) in association with mixed CPAM type 1 and 2. This case is unique as KRAS mutation positive AC is present in a setting of both CPAM 1 and 2 in the same lesion.
October 2016: Annals of Thoracic Surgery
Ruben Lamas-Pinheiro, Mafalda David, Tiago Henriques-Coelho
No abstract text is available yet for this article.
August 26, 2016: Neonatology
Louis P Dehner, Yoav H Messinger, Gretchen M Williams, Douglas R Stewart, Laura A Harney, Kris Ann Schultz, D Ashley Hill
No abstract text is available yet for this article.
August 26, 2016: Neonatology
Steven Hardee, Lea Tuzovic, Cicero T Silva, Robert A Cowles, Joshua Copel, Raffaella A Morotti
Congenital cystic lung lesions (CCLL) are a group of rare pathologies that are usually diagnosed in the pre-natal 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...
August 4, 2016: Pediatric and Developmental Pathology
Brenda Hiu Yan Law, Ioana Bratu, Venu Jain, Marc-Antoine Landry
Antenatally, congenital pulmonary airway malformation (CPAM) causing fetal hydrops can be palliated with thoracoamniotic shunts, which may become displaced in utero. We report a case of an infant born at 34 weeks gestational age with an antenatally diagnosed macrocystic lung lesion, fetal hydrops and an internally displaced thoracoamniotic shunt. The infant suffered refractory pneumothoraces despite multiple chest drains, and stabilised only after surgical resection of the lesion. Intraoperatively, the shunt was noted to form a connection between a type I CPAM and the pleural space...
2016: BMJ Case Reports
Sara Costanzo, Claudia Filisetti, Claudio Vella, Mariangela Rustico, Paola Fontana, Gianluca Lista, Salvatore Zirpoli, Marcello Napolitano, Giovanna Riccipetitoni
OBJECTIVES: The objective of our study is to retrospectively analyze a single-centre series of antenatally detected pulmonary malformations (PM) and to evaluate their postnatal outcome. MATERIALS AND METHODS: We retrospectively reviewed all prenatally diagnosed PM patients referred to our Centre in the period between January 1999 and December 2014. All cases were diagnosed by one of our Maternal-Fetal Specialists by US examination. Congenital pulmonary airway malformation (CPAM) volume ratio (CVR), development of fetal complications, need for fetal therapy, need for neonatal resuscitation and timing of surgery were analyzed...
July 2016: Journal of Neonatal Surgery
Christopher W Baird, Sanjay Prabhu, Terry L Buchmiller, Charles Smithers, Russell Jennings
Complex tracheobronchial obstruction and malacia can be associated with conotruncal and aortic arch anomalies. A circumflex aortic arch composed of a left aortic arch and right descending aorta is an extremely rare anomaly that can severely affect the distal trachea and mainstem bronchi, resulting in severe respiratory symptoms. We report the case of a patient with circumflex aortic arch and severe left mainstem bronchial obstruction and malacia in which the external aortic compression and residual bronchomalacia were addressed with direct bronchial and tracheal intervention...
July 2016: Annals of Thoracic Surgery
Amy E Wagenaar, Jun Tashiro, Shevonne S Satahoo, Juan E Sola, Holly L Neville, Anthony R Hogan, Eduardo A Perez
PURPOSE: We sought to determine factors influencing survival and resource utilization in patients undergoing surgical resection of congenital lung malformations (CLM). Additionally, we used propensity score-matched analysis (PSMA) to compare these outcomes for thoracoscopic versus open surgical approaches. METHODS: Kids' Inpatient Database (1997-2009) was used to identify congenital pulmonary airway malformation (CPAM) and pulmonary sequestration (PS) patients undergoing resection...
September 2016: Journal of Pediatric Surgery
Timothy J Barreiro, Lucas Henn, Sisham Ingnam, Michael Sypert
Congenital pulmonary airway malformation (CPAM), previously known as congenital cystic adenomatoid malformation (CCAM), is an inborn abnormality of the lower respiratory system. Most often diagnosed in the perinatal period, these anomalies usually present with tachypnea, cyanosis, and respiratory distress. However, rare cases are asymptomatic and undiagnosed until adulthood.
2016: Respiratory Medicine Case Reports
Mafalda David, Ruben Lamas-Pinheiro, Tiago Henriques-Coelho
Congenital pulmonary airway malformation (CPAM) is one of the most common lung lesions detected prenatally. Despite the research efforts made in the past few years, controversy and lack of clarity in the literature still exist regarding nomenclature, classification, pathogenesis and the management of CPAM. Therefore, it is of greatest importance to delineate the natural history of CPAMs and to create a consensus to guide the management and follow-up of these lesions. This review will focus on classification systems, highlighting the most recent advancements in pathogenesis, and current practice in the prenatal diagnosis of CPAM...
2016: Neonatology
Swapna A Chiluveru, Nandini M Dave, Raylene J Dias, Madhu B Garasia
The association of congenital pulmonary airway malformation (CPAM) with congenital heart disease is rare. We present the case of a 6-month-old child with atrial septal defect and pulmonary hypertension (PH) who presented with severe respiratory distress and hypoxia. The patient underwent right lobectomy for CPAM. With timely management, real-time monitoring, one lung ventilation, and adequate analgesia, we were able to extubate the child in the immediate postoperative period. We conclude that with meticulous planning and multidisciplinary team approach, such complex cases can be managed successfully...
April 2016: Annals of Cardiac Anaesthesia
Astrid Hellmund, Christoph Berg, Annegret Geipel, Meike Bludau, Andreas Heydweiller, Haitham Bachour, Andreas Müller, Annette Müller, Ulrich Gembruch
OBJECTIVE: To describe antenatal findings and evaluate prenatal risk parameters for adverse outcome or need for intervention in fetuses with congenital pulmonary airway malformation (CPAM). METHODS: In our retrospective study all fetuses with a prenatal diagnosis of CPAM detected in our tertiary referral center between 2002 and 2013 were analyzed. Sonographic findings were noted and measurements of mass-to-thorax-ratio (MTR), congenital pulmonary airway malformation volume-ratio (CVR) and observed to expected lung-to head-ratio (o/e LHR) were conducted and correlated to fetal or neonatal morbidity and mortality and/or need for prenatal intervention...
2016: PloS One
Christopher L Sudduth, Sarah J Hill, Mehul V Raval
Two of the most common types of congenital thoracic malformations are congenital pulmonary airway malformations (CPAMs) and bronchopulmonary sequestrations (BPS). Here we present the first known case of a hybrid lesion, with coexisting features of an extralobar sequestration (ELS) and CPAM type 2, with arterial inflow from the celiac trunk and venous outflow to the portal vein. The clinical presentation, investigative imaging and timing of surgery are discussed.
July 2016: Pediatric Surgery International
Ragip Ortac, Gulden Diniz, Hulya Tosun Yildirim, Safiye Aktas, Irfan Karaca
OBJECTIVES: Congenital pulmonary airway malformation (CPAM) is an uncommon congenital abnormality of the lungs that generally presents during prenatal period or early childhood. In this study, we aimed to evaluate clinical and pathologic findings of the children with CPAMs who were referred to our center between 1992 and 2011. MATERIAL AND METHODS: We reviewed 19 children with CPAM, who were diagnosed and treated at the Izmir Dr. Behçet Uz Children's Hospital between 1992 and 2011...
2016: Fetal and Pediatric Pathology
John A Sandoval, Israel Fernandez-Pineda, Alpin D Malkan
OBJECTIVE: A small percentage of pediatric solid cancers arise as a result of clearly identified inherited predisposition syndromes and nongenetic lesions. Evidence supports preemptive surgery for children with genetic [multiple endocrine neoplasia type 2 (MEN2), familial adenomatous polyposis syndrome (FAP), hereditary nonpolyposis colorectal cancer (HNPCC), and hereditary diffuse gastric cancer (HDGC) and nongenetic [thyroglossal duct cysts (TGDC), congenital pulmonary airway malformations (CPAM), alimentary tract duplication cysts (ATDC), and congenital choledochal cysts (CCC)] developmental anomalies...
April 2016: Journal of Pediatric Surgery
Jessica Kapralik, Carolyn Wayne, Emily Chan, Ahmed Nasr
BACKGROUND: The ideal management of infants born with asymptomatic congenital pulmonary airway malformation (CPAM) is controversial. We performed a systematic review and meta-analysis comparing elective resection versus expectant management. METHODS: We searched CENTRAL, MEDLINE, EMBASE, CINAHL, and PubMed for studies describing the management of asymptomatic CPAM and reporting on postoperative morbidity, mortality, and length of hospital stay (LOS). We performed meta-analyses when possible and provide a narrative summary of results...
March 2016: Journal of Pediatric Surgery
Christian L Hermansen, Anand Mahajan
Newborn respiratory distress presents a diagnostic and management challenge. Newborns with respiratory distress commonly exhibit tachypnea with a respiratory rate of more than 60 respirations per minute. They may present with grunting, retractions, nasal flaring, and cyanosis. Common causes include transient tachypnea of the newborn, respiratory distress syndrome, meconium aspiration syndrome, pneumonia, sepsis, pneumothorax, persistent pulmonary hypertension of the newborn, and delayed transition. Congenital heart defects, airway malformations, and inborn errors of metabolism are less common etiologies...
December 1, 2015: American Family Physician
Sophie C Hofferberth, Karen Watters, Reza Rahbar, Francis Fynn-Thompson
BACKGROUND: Congenital tracheal stenosis (CTS) is a serious and rare condition that often presents a complex surgical challenge. We reviewed the evolution of surgical approaches to the management of CTS at a single institution. METHODS: We conducted a retrospective clinical review of all patients who underwent surgical repair of CTS at our institution. From January 1992 to January 2014, a total of 30 patients (median age 92 days; range 3 days-1.7 years) underwent surgery for CTS at our institution...
January 2016: World Journal for Pediatric & Congenital Heart Surgery
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