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Congenital Lung Malformations

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A case report of an unusual non-mucinous papillary variant of CPAM type 1 with KRAS mutations.

Timco Koopman, Bart L Rottier, Arja Ter Elst, Wim Timens,

BACKGROUND:congenital pulmonary airway malformation (CPAM) is the most frequent congenital lung disorder. CPAM type 1 is the most common subtype, typically having a cystic radiological and histological appearance. Mucinous clusters in CPAM type 1 have been identified as premalignant precursors for mucinous adenocarcinoma. These mucinous adenocarcinomas and the mucinous clusters ... Read more >>

BMC Pulm Med (BMC pulmonary medicine)
[2020, 20(1):52]

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Neonatal outcome of children born after ICSI with epididymal or testicular sperm: A 10-year study in China.

Lei Jin, Zhou Li, Longjie Gu, Bo Huang,

Some studies show that children born after ICSI with non-ejaculated sperm are at increased risk of birth defects, other studies hold the opposite view. Does neonatal outcome including congenital malformations in children born after ICSI with percutaneous epididymal sperm aspiration (PESA) and testicular sperm aspiration (TESA) differ from neonatal outcome ... Read more >>

Sci Rep (Scientific reports)
[2020, 10(1):5145]

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Obstructive lung disease secondary to compression of the bronchus by an enlarged pulmonary artery.

Justin Panthappattu, Vidhu Sharma, Sameer Verma, Arunabh Talwar,

In clinical practice, the presence of wheezing generally indicates an airway disease. In rare circumstances, adjacent mediastinal structures may compress the tracheobronchial tree leading to obstructive physiology. Compression of the tracheobronchial region by an enlarged pulmonary artery (PA) is exceedingly rare. We present here a case of pulmonary hypertension, where ... Read more >>

Lung India (Lung India : official organ of Indian Chest Society)
[2020, 37(2):158-160]

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[Clinical analysis of 96 cases of congenital cystic lung lesions in children].

R Guo, Y X Zou, J Zhai, L S Zhao,

Objective: To summarize the diagnosis, clinical manifestations, treatment and prognosis of congenital cystic lung lesions. Methods: A retrospective study described the clinical course of 96 patients (46 female and 50 male) diagnosed with congenital cystic lung lesions treated at the Tianjin Children's Hospital from January 2010 to March 2019. The ... Read more >>

Zhonghua Er Ke Za Zhi (Zhonghua er ke za zhi = Chinese journal of pediatrics)
[2020, 58(1):19-24]

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Pulmonary arteriovenous fistula diagnosed by contrast echocardiography: a case report of a daughter and a mother

Gaoyun Pan, Xinxin Dong, Jianguo Xu,

<title>Abstract</title> <p> Background Pulmonary arteriovenous fistula is a rare disease with a direct connection between the pulmonary artery and the vein, and in most cases is congenital. In a proportion of patients, it can cause hypoxemia, cyanosis and dyspnea. The golden standard for the diagnosis of PAVF is ... Read more >>

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Congenital cystic adenomatoid malformations of the lung: an epithelial transcriptomic approach.

Guillaume Lezmi, Shamila Vibhushan, Claudia Bevilaqua, Nicolas Crapart, Nicolas Cagnard, Naziha Khen-Dunlop, Christine Boyle-Freyssaut, Alice Hadchouel, Christophe Delacourt,

BACKGROUND:The pathophysiology of congenital cystic adenomatoid malformations (CCAM) of the lung remains poorly understood. AIM:This study aimed to identify more precisely the molecular mechanisms limited to a compartment of lung tissue, through a transcriptomic analysis of the epithelium of macrocystic forms. METHODS:Tissue fragments displaying CCAM were obtained during planned surgical ... Read more >>

Respir. Res. (Respiratory research)
[2020, 21(1):43]

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Thoracoscopic Lobectomy for Congenital Pulmonary Airway Malformation: Where Are We in 2019?

Arnaud Bonnard,

Thoracoscopic surgery for congenital pulmonary airway malformation (CPAM) is still a matter of debate and used by approximately 50% of the surgeons in Europe. Several questions need to be addressed about CPAM. The adequate treatment, the surgical approach, and the follow-up are few of them. A review of recent articles ... Read more >>

Eur J Pediatr Surg (European journal of pediatric surgery : official journal of Austrian Association of Pediatric Surgery ... [et al] = Zeitschrift fur Kinderchirurgie)
[2020, :]

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Preoperative delineation of pulmonary fissural anatomy at multi-detector computed tomography in children with congenital pulmonary malformations and impact on surgical complications and postoperative course.

María Navallas, Priscilla Chiu, Afsaneh Amirabadi, David E Manson,

BACKGROUND:Delineation of the anatomy and integrity of the pulmonary fissures at CT is important because anomalous or incomplete fissures might increase the risk of surgery and of postoperative complications. OBJECTIVE:To preoperatively evaluate the integrity of the pleural fissures in children with congenital lung malformations and determine whether anomalous fissural anatomy ... Read more >>

Pediatr Radiol (Pediatric radiology)
[2020, :]

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Fetal Malformations and the Pediatric Surgeon.

Subhasis Roy Choudhury,

J Indian Assoc Pediatr Surg (Journal of Indian Association of Pediatric Surgeons)
[2020, 25(2):65-67]

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4q27 deletion and 7q36.1 microduplication in a patient with multiple malformations and hearing loss: a case report.

Maolan Wu, Xiangrong Zheng, Xia Wang, Guoyuan Zhang, Jian Kuang,

BACKGROUND:Chromosome deletions of the long arm of chromosome 4 in 4q syndrome are characterized by mild facial and digital dysmorphism, developmental delay, growth retardation, and skeletal and cardiac anomalies, which is regarded as an autism spectrum disorder. Moreover, some scarce reports indicate that patients with 4q interstitial deletion and 7p ... Read more >>

BMC Med Genomics (BMC medical genomics)
[2020, 13(1):31]

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Vascular lesions of the head and neck: an update on classification and imaging review.

Akshaar N Brahmbhatt, Kamila A Skalski, Alok A Bhatt,

Vascular lesions have a varied appearance and can commonly occur in the head and neck. A majority of these lesions are cutaneous and congenital; however, some may be acquired and malignant. The presentation and clinical history of patients presenting with head and neck lesions can be used to guide further ... Read more >>

Insights Imaging (Insights into imaging)
[2020, 11(1):19]

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Oral management of a patient with down syndrome and agammaglobulinemia: a case report.

Yasuka Kusumoto, Kohsuke Imai, Yoshio Ohyama, Haruhisa Fukayama, Osamu Shinozuka,

BACKGROUND:Down syndrome is characterized by a variety of dysmorphic features and congenital malformations, such as congenital heart disease, gastrointestinal disease, and other conditions like leukemia and autoimmune disorders. Patients with Down syndrome are highly prone to respiratory tract infections, which might be fatal to them. However, there are only few ... Read more >>

BMC Oral Health (BMC oral health)
[2020, 20(1):71]

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A clinical guideline for structured assessment of CT-imaging in congenital lung abnormalities.

Sergei M Hermelijn, Bernadette B L J Elders, Pierluigi Ciet, René M H Wijnen, Harm A W M Tiddens, J Marco Schnater,

OBJECTIVES:To develop a clinical guideline for structured assessment and uniform reporting of congenital lung abnormalities (CLA) on Computed Tomography (CT)-scans. MATERIALS AND METHODS:A systematic literature search was conducted for articles describing CT-scan abnormalities of congenital pulmonary airway malformation (CPAM), bronchopulmonary sequestration (BPS), congenital lobar emphysema (CLE) and bronchogenic cyst (BC). ... Read more >>

Paediatr Respir Rev (Paediatric respiratory reviews)
[2020, :]

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Further delineation of the phenotypic spectrum of nevus comedonicus syndrome to include congenital pulmonary airway malformation of the lung and aneurysm.

Sarah E Sheppard, Anna Smith, Katheryn Grand, Jennifer Pogoriler, Adam I Rubin, Erica Schindewolf, Mark P Fitzgerald, Julie Moldenhauer, Pablo Laje, William Peranteau, Elizabeth Bhoj, Patrick McMahon, Leslie Castelo-Soccio,

Nevus comedonicus syndrome (NCS) is a rare epidermal nevus syndrome characterized by ocular, skeletal, and central nervous system anomalies. We present a 23-month-old boy with a history of a congenital pulmonary airway malformation (CPAM) of the lung and a congenital cataract who developed progressive linear and curvilinear plaques of dilated ... Read more >>

Am. J. Med. Genet. A (American journal of medical genetics. Part A)
[2020, 182(4):746-754]

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Asymptomatic Congenital Lung Malformations: Timing of Resection Does Not Affect Adverse Surgical Outcomes.

Vincent Duron, Ariela Zenilman, Cornelia Griggs, Jennifer DeFazio, Jessica C Price, Weijia Fan, Matthew Vivero, Juliana Castrillon, Maggie Schmaedick, Emaad Iqbal, Steven Rothenberg,

Background: Optimal timing for resection of asymptomatic congenital lung malformations (CLMs) remains controversial. The aim of this study is to define optimal timing for surgical intervention of patients with CLMs and define clinical variables that affect surgical outcomes. Methods: An IRB-approved retrospective analysis was conducted for patients undergoing surgery for ... Read more >>

Front Pediatr (Frontiers in pediatrics)
[2020, 8:35]

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Early Diagnosis of Left Pulmonary Artery Sling During First Week of Life in a Term Baby Boy: A Case Report.

Dima Kayal, Samer Minkara, Faissal Tleiss,

Pulmonary artery sling is a rare cause of neonatal respiratory distress. Most patients with pulmonary artery sling present in early infancy with stridor and signs of respiratory distress. Diagnosis of pulmonary artery sling, like other vascular ring anomalies, can be made using various imaging modalities, and management encompasses urgent surgical ... Read more >>

Cureus (Cureus)
[2020, 12(2):e6889]

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Kaposiform hemangioendothelioma: current knowledge and future perspectives.

Yi Ji, Siyuan Chen, Kaiying Yang, Chunchao Xia, Li Li,

Kaposiform hemangioendothelioma (KHE) is a rare vascular neoplasm with high morbidity and mortality. The initiating mechanism during the pathogenesis of KHE has yet to be discovered. The main pathological features of KHE are abnormal angiogenesis and lymphangiogenesis. KHEs are clinically heterogeneous and may develop into a life-threatening thrombocytopenia and consumptive ... Read more >>

Orphanet J Rare Dis (Orphanet journal of rare diseases)
[2020, 15(1):39]

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Accuracy of prenatal and postnatal imaging for management of congenital lung malformations.

Candace C Style, Amy R Mehollin-Ray, Mariatu A Verla, Oluyinka O Olutoye, Patricio E Lau, Brittany L Johnson, Alice King, Sundeep G Keswani, Timothy C Lee,

PURPOSE:The purpose of this study was to compare the accuracy of prenatal and postnatal imaging modalities for evaluation and management of congenital lung malformations (CLMs). METHODS:A retrospective review was performed of all fetuses evaluated for a CLM between December 2001 and January 2018. Pre and postnatal imaging findings, operative treatment, ... Read more >>

J. Pediatr. Surg. (Journal of pediatric surgery)
[2020, :]

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Elevated Systemic IL-10 Levels Indicate Immunodepression Leading to Nosocomial Infections after Aneurysmal Subarachnoid Hemorrhage (SAH) in Patients.

Shafqat Rasul Chaudhry, Ulf Dietrich Kahlert, Thomas Mehari Kinfe, Alf Lamprecht, Mika Niemelä, Daniel Hänggi, Sajjad Muhammad,

BACKGROUND:Aneurysmal subarachnoid hemorrhage (SAH) is a highly complex disease with very high mortality and morbidity. About one-third of SAH patients suffer from systemic infections, predominantly pneumonia, that can contribute to excess mortality after SAH. Immunodepression is probably the most important mechanism leading to infections. Interleukin-10 (IL-10) is a master regulator ... Read more >>

Int J Mol Sci (International journal of molecular sciences)
[2020, 21(5):]

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Serum osteocalcin level is associated with the mortality in Chinese patients with Fibrodysplasia ossificans progressiva aged ≤18 years at diagnosis.

Dunmin She, Ran Li, Ping Fang, Guannan Zong, Ying Xue, Keqin Zhang,

BACKGROUND:Fibrodysplasia ossificans progressiva (FOP) is an ultra-rare genetic disorder characterized by extraskeletal heterotopic ossification. It is well recognized that FOP can lead to a devastating condition of disability. However, the mortality rate of FOP patients in China and risk factors for mortality are still largely unclear. METHODS:We conducted a retrospective ... Read more >>

BMC Musculoskelet Disord (BMC musculoskeletal disorders)
[2020, 21(1):152]

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Thoracic Magnetic Resonance Imaging Applications in Children.

Adem Karaman,

Lung pathologies in the pediatric population can usually be detected using chest radiography. Multidetector computed tomography (MDCT) imaging is often used as a supplementary method in the evaluation of lung diseases. Recently, magnetic resonance imaging (MRI) techniques were found to be reliable in the evaluation of pulmonary diseases in the ... Read more >>

Eurasian J Med (The Eurasian journal of medicine)
[2020, 52(1):94-97]

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Insights to correlations and discrepancies between impaired lung function and heart failure in Eisenmenger patients.

Lina Gumbiene, Lina Kapleriene, Dovile Jancauskaite, Monika Laukyte-Sleniene, Elena Jureviciene, Virginija Rudiene, Egle Paleviciute, Mindaugas Mataciunas, Virginija Sileikiene,

Impaired lung function and spirometric signs of airway obstruction without common risk factors for chronic obstructive pulmonary disease could be found in patients with Eisenmenger syndrome. This study aimed to analyse the association between lung function parameters and disease severity (including heart failure markers, associated congenital heart defect) as well ... Read more >>

Pulm Circ (Pulmonary circulation)
[2020, 10(1):1350650120909729]

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Asthma in Pregnancy: Pathophysiology, Diagnosis, Whole-Course Management, and Medication Safety.

Huijie Wang, Na Li, Huaqiong Huang,

Asthma in pregnancy is a health issue of great concern. Physiological changes and drug compliance during pregnancy can affect asthma control in varying degrees, and the control level of asthma and the side effects of asthma medications are closely related to the adverse perinatal outcomes of mother and fetus. This ... Read more >>

Can. Respir. J. (Canadian respiratory journal)
[2020, 2020:9046842]

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Analysis of Wnt7B and BMP4 expression patterns in congenital pulmonary airway malformation.

Hao Zhu, Dan Liu, Huimin Jia,

BACKGROUND:Congenital pulmonary airway malformation (CPAM) is a rare disorder characterized by aberrant overgrowth of terminal bronchioles. The objective of this study was to describe wingless-type MMTV integration site family 7B (Wnt7B) and bone morphogenetic protein 4 (BMP4) expression patterns in human CPAM lesions and to explore the possible roles of ... Read more >>

Pediatr. Pulmonol. (Pediatric pulmonology)
[2020, 55(3):765-770]

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Congenital pulmonary airway malformation: A case report of a rare cause of neonatal respiratory distress and review of the literature.

E A Disu, O A Kehinde, A L Anga, P O Ubuane, A Itiola, I J Akinola, B Falase,

Congenital pulmonary airway malformation (CPAM), formerly known as congenital cystic adenomatoid malformation (CCAM), is a rare developmental dysplastic lesion of the fetal tracheobronchial tree. CPAM is a rare cause of neonatal respiratory distress; however, its presence may span fetal to adult period. In two previous case-reports from Nigeria, CPAM was ... Read more >>

Niger J Clin Pract (Nigerian journal of clinical practice)
[2019, 22(11):1621-1625]

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