1,225 results match your criteria Congenital Cystic Adenomatoid Malformation


A Missed Late Presentation of a Congenital Pulmonary Airway Malformation as a Large Infected .

Sultan Qaboos Univ Med J 2018 Nov 28;18(4):e541-e544. Epub 2019 Mar 28.

Department of Radiology, Hospital Tengku Ampuan Rahimah, Klang, Malaysia.

A congenital pulmonary airway malformation (CPAM) is a rare cystic anomaly that may occur during development of the fetal airways. The vast majority of CPAMs are detected in neonates; as such, it is unusual for diagnosis to occur in adulthood. We report a 21-year-old male patient who presented to the emergency department of the Hospital Ampang, Kuala Lumpur, Malaysia, in 2015 with chest pain, breathlessness and tachypnoea. Read More

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https://journals.squ.edu.om/index.php/squmj/article/view/300
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http://dx.doi.org/10.18295/squmj.2018.18.04.020DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6443284PMC
November 2018
2 Reads

Lung protective ventilation during pulmonary resection in children: a prospective, single-centre, randomised controlled trial.

Br J Anaesth 2019 May 8;122(5):692-701. Epub 2019 Mar 8.

Department of Anaesthesiology and Pain Medicine, Seoul National University Hospital, Seoul National University College of Medicine, Seoul, Republic of Korea. Electronic address:

Background: Perioperative ventilatory strategies for lung protection in children are underexplored. This study evaluated the effects of lung protective ventilation (LPV) on postoperative clinical outcomes in children requiring one-lung ventilation (OLV) for pulmonary resection.

Methods: Children age ≤5 yr scheduled for video-assisted thoracoscopic lung lobectomy or segmentectomy were randomly assigned to LPV or control ventilation. Read More

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http://dx.doi.org/10.1016/j.bja.2019.02.013DOI Listing
May 2019
1 Read

Pulmonary Infantile Hemangioma Mimicking a Congenital Cystic Adenomatoid Malformation.

Pediatr Dev Pathol 2019 Mar 26:1093526619838743. Epub 2019 Mar 26.

2 Department of Pathology, Hôpital Femme Mère Enfant, CHU de Lyon, Bron, France.

Infantile hemangioma (IH) is the most common benign vascular tumor of infancy, occurring predominantly in the head and neck. It is characterized by specific endothelial expression of glucose transporter-1 (GLUT-1) and involution with time, spontaneous or on beta-blockers treatment. Although some predisposing factors are known, the exact pathogenesis remains unclear. Read More

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http://dx.doi.org/10.1177/1093526619838743DOI Listing
March 2019
1 Read

Development of a multi-institutional registry for children with operative congenital lung malformations.

J Pediatr Surg 2019 Feb 28. Epub 2019 Feb 28.

Section of Pediatric Surgery, Department of Surgery, University of Michigan and Michigan Medicine, C.S. Mott Children's and Von Voigtlander Women's Hospital, Ann Arbor, MI, USA.

Introduction: The purpose of this study was to develop a multi-institutional registry to characterize the demographics, management, and outcomes of a contemporary cohort of children undergoing congenital lung malformation (CLM) resection.

Methods: After central reliance IRB approval, a web-based, secure database was created to capture retrospective cohort data on pathologically-confirmed CLMs performed between 2009 and 2015 within a multi-institutional research collaborative.

Results: Eleven children's hospitals contributed 506 patients. Read More

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http://dx.doi.org/10.1016/j.jpedsurg.2019.01.058DOI Listing
February 2019
2 Reads

Short-term respiratory outcomes of neonates with symptomatic congenital lung malformations.

J Pediatr Surg 2019 Feb 24. Epub 2019 Feb 24.

Section of Pediatric Surgery, Department of Surgery, Michigan Medicine, C.S. Mott, Children's and Von Voigtlander Women's Hospital, Ann Arbor, MI, USA 48109. Electronic address:

Introduction: The purpose of this study was to evaluate short-term respiratory outcomes in neonates with symptomatic congenital lung malformations (CLM).

Methods: Consecutive newborns who underwent surgical resection of a CLM were retrospectively reviewed. Demographic, prenatal, and outcomes data were analyzed as appropriate (p < 0. Read More

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http://dx.doi.org/10.1016/j.jpedsurg.2019.01.056DOI Listing
February 2019
1 Read

Cystic and pseudocystic pulmonary malformations in children: Clinico-pathological correlation.

Ann Diagn Pathol 2019 Feb 16;39:78-85. Epub 2019 Feb 16.

Unidad de Investigación Médica en Enfermedades Respiratorias, Unidad Médica de Alta Especialidad, Hospital de Pediatría Silvestre Frenk Freund, Centro Médico Nacional Siglo XXI, Instituto Mexicano del Seguro Social, Ciudad de México, Mexico.

Pulmonary malformations are rare disorders, with cystic and pseudocystic pulmonary malformations (CPPM) the most frequent, and constitute the first cause of lobectomy in children <1 year of age. Morphological overlap of congenital cystic pulmonary lesions might correspond to a spectrum of lesions in which bronchial atresia is a common etiopathogenetic mechanism. We aimed to report the frequency of CPPM resected in a tertiary-level hospital and to evaluate the degree of agreement between presurgical and anatomopathological diagnoses. Read More

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http://dx.doi.org/10.1016/j.anndiagpath.2019.02.005DOI Listing
February 2019
2 Reads

Agreement between magnetic resonance imaging and computed tomography in the postnatal evaluation of congenital lung malformations: a pilot study.

Eur Radiol 2019 Feb 22. Epub 2019 Feb 22.

Pediatric Radiology and Neuroradiology, ASST Fatebenefratelli-Sacco Milano, Children's Hospital V. Buzzi, Via Castelvetro 32, 20154, Milan, Italy.

Objectives: To compare postnatal magnetic resonance imaging (MRI) with the reference standard computed tomography (CT) in the identification of the key features for diagnosing different types of congenital lung malformation (CLM).

Methods: Respiratory-triggered T2-weighted single-shot turbo spin echo (ss-TSE), respiratory-triggered T1-weighted turbo field echo (TFE), balanced fast field echo (BFFE), and T2-weighted MultiVane sequences were performed at 1.5 T on 20 patients prospectively enrolled. Read More

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http://dx.doi.org/10.1007/s00330-019-06042-wDOI Listing
February 2019
1 Read

Prenatal diagnosis of fetal intraabdominal extralobar pulmonary sequestration: a 12-year 3-center experience in China.

Sci Rep 2019 Jan 30;9(1):943. Epub 2019 Jan 30.

Department of Ultrasound Diagnosis, The Second Xiangya Hospital, Central South University, Changsha, Hunan, 410011, China.

To provide useful information for diagnosing and predicting fetal intraabdominal extralobar pulmonary sequestration (IEPS), a retrospective review of diagnostic approaches was conducted. Ultrasonography was performed serially in 21 fetuses with IEPS from 2005 to 2017. Prenatal sonographic features, treatment, and outcomes of each case were evaluated and collected. Read More

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http://dx.doi.org/10.1038/s41598-018-37268-1DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6353922PMC
January 2019
2 Reads

Nuss procedure for patients with pectus excavatum with a history of intrathoracic surgery.

J Plast Reconstr Aesthet Surg 2018 Dec 15. Epub 2018 Dec 15.

Department of Plastic and Reconstructive Surgery, Nagoya University Graduate School of Medicine, 65 Tsurumai-cho Showa-ku, Nagoya, Aichi 464-8560, Japan.

Background: The aim of this study was to demonstrate the feasibility and safety of the Nuss procedure for patients with pectus excavatum (PE) with a history of intrathoracic surgery.

Patients: From April 2010 to December 2013, we performed 6 cases of PE repair in patients with a history of intrathoracic surgery. The causes of previous operations were congenital cystic adenomatoid malformation in 4 patients and congenital diaphragmatic hernia in 2. Read More

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http://dx.doi.org/10.1016/j.bjps.2018.12.027DOI Listing
December 2018
3 Reads

Gene expression profiling reveals differential patterns between microcystic congenital cystic adenomatoid malformation and congenital lobar emphysema.

Early Hum Dev 2019 01 22;128:77-80. Epub 2018 Dec 22.

Translational Medicine Center, Guangdong Women and Children Hospital, Guangzhou, China. Electronic address:

Introduction: Congenital cystic adenomatoid malformation (CCAM), especially type-III, shares similar sonographic features with congenital lobar emphysema (CLE) in routine ultrasound scan. Thus, prenatal differentiation of CLE from a microcystic CCAM is challenging and difficult in practice. Discovery of molecular biomarkers has important clinical significance. Read More

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http://dx.doi.org/10.1016/j.earlhumdev.2018.12.014DOI Listing
January 2019
4 Reads

[Pulmonary glial heterotopia: Unique lesion in an infant with anencephalic twin].

Ann Pathol 2019 Feb 12;39(1):24-28. Epub 2018 Dec 12.

Service de pathologie, hôpital universitaire Robert-Debré, AP-HP, 48, boulevard Sérurier, 75019 Paris, France; Université Diderot Paris 7 - SPC, 16, rue Huchard, 75018 Paris, France.

Pulmonary glial heterotopia is an extremely rare entity whose pathophysiology remains unclear. We report for the first time one case of pulmonary glial heterotopia occured in a one-month baby free from any malformation. She has the particularity of being born from monozygotic monochorionic twin pregnancy where her anencephalic exencephalic sister suffered a medical termination of pregnancy. Read More

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http://dx.doi.org/10.1016/j.annpat.2018.08.006DOI Listing
February 2019
3 Reads

Early vs late resection of asymptomatic congenital lung malformations.

J Pediatr Surg 2019 Jan 5;54(1):70-74. Epub 2018 Oct 5.

Texas Children's Fetal Center, Baylor College of Medicine, Houston, TX; Michael E. DeBakey Department of Surgery, Baylor College of Medicine, Houston, TX; Department of Obstetrics and Gynecology, Baylor College of Medicine, Houston, TX. Electronic address:

Purpose: To examine postsurgical outcomes of a consecutive series of children treated with elective operations for congenital lung malformations (CLM).

Methods: A retrospective review was performed on a prospectively collected dataset of all fetuses evaluated for a CLM between July 2001 and June 2016. Prenatal findings, operative treatment and postnatal outcomes were collected. Read More

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http://dx.doi.org/10.1016/j.jpedsurg.2018.10.035DOI Listing
January 2019
2 Reads

Fetal Congenital Peripheral Bronchial Atresia Diagnosed by Magnetic Resonance Imaging: Two Case Reports.

AJP Rep 2018 Oct 9;8(4):e201-e205. Epub 2018 Oct 9.

Department of Obstetrics and Gynecology, Graduate School of Medicine, University of the Ryukyus, Okinawa, Japan.

Two types of congenital bronchial atresia (proximal and peripheral) have been classified. We report two cases of peripheral bronchial atresia diagnosed by prenatal ultrasonography (US) and magnetic resonance imaging (MRI). Evaluating an enlarged lung mass that is homogeneously hyperechoic on US and hyperintense on T2-weighted MRI can help in determining whether bronchial atresia is present. Read More

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http://www.thieme-connect.de/DOI/DOI?10.1055/s-0038-1673620
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http://dx.doi.org/10.1055/s-0038-1673620DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6177339PMC
October 2018
4 Reads

An autopsy based descriptive study of the spectrum of pulmonary lesions encountered in fetal deaths at a tertiary care center.

Indian J Pathol Microbiol 2018 Oct-Dec;61(4):495-499

Department of Pathology, Mahadevappa Rampure Medical College, Kalaburagi, Karnataka, India.

Background: : Fetal autopsy has been an indispensable tool in evaluating the cause of death. The most common cause for fetal deaths is associated with respiratory disorders accounting for around 60%. This study was undertaken to evaluate the gross and microscopic features of lungs in fetal autopsies and estimate the incidence of deaths attributable to pulmonary causes. Read More

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http://www.ijpmonline.org/text.asp?2018/61/4/495/242983
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http://dx.doi.org/10.4103/IJPM.IJPM_461_17DOI Listing
February 2019
22 Reads

Natural prenatal history of congenital pulmonary malformations: the MALFPULM population-based cohort study.

Ultrasound Obstet Gynecol 2018 Sep 27. Epub 2018 Sep 27.

Inserm UMR 1153, Maternité Port-Royal, Paris, France.

Background: Congenital pulmonary malformations (CPM) are rare diseases that are for the most part identified prenatally and can consist of cystic and/or hyperechoic intrathoracic lesions. No prospective, population-based data are available on the pattern of changes in the volume of CPM during pregnancy according to their ultrasound appearance, nor on the association between the size of the CPM and risk of compression.

Objectives: Our objectives were to: i) assess prenatal changes in the volume of CPM and to examine whether these changes were different for cystic vs. Read More

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http://dx.doi.org/10.1002/uog.20130DOI Listing
September 2018
19 Reads

Long-term pulmonary function after lobectomy for congenital pulmonary airway malformation: is thoracoscopic approach really better than open?

J Pediatr Surg 2018 Dec 5;53(12):2383-2385. Epub 2018 Sep 5.

Department of Surgery, The University of Hong Kong, Queen Mary Hospital, Hong Kong. Electronic address:

Introduction: Congenital pulmonary airway malformation (CPAM) is the most common lung pathology diagnosed antenatally. Thoracoscopic lobectomy has shown increasing popularity, but the long-term result is still lacking. In this study we compared long -term pulmonary function after thoracoscopic and open lobectomy. Read More

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https://linkinghub.elsevier.com/retrieve/pii/S00223468183053
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http://dx.doi.org/10.1016/j.jpedsurg.2018.08.029DOI Listing
December 2018
22 Reads
1.310 Impact Factor

Predictors of early lobectomy after birth in prenatally diagnosed congenital pulmonary airway malformation.

J Pediatr Surg 2018 Dec 5;53(12):2386-2389. Epub 2018 Sep 5.

Department of Pathology National Center for Child Health and Development, Tokyo, Japan.

Purpose: The purpose of this study was to clarify the relationship between congenital pulmonary airway malformation volume ratio (CVR) of bronchial atresia (BA), CVR of congenital cystic adenomatoid malformations (CCAM), and time of surgery after birth.

Method: We retrospectively analyzed data of 36 BA and CCAM cases, prenatally diagnosed as CPAM from 2009 through 2014.

Results: Within 2 h after birth, 12 neonatal patients underwent emergent (EMG) lobectomy. Read More

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http://dx.doi.org/10.1016/j.jpedsurg.2018.08.025DOI Listing
December 2018
1 Read

Congenital cystic adenomatoid malformation in adults detected after infection.

Respirol Case Rep 2018 Nov 14;6(8):e00364. Epub 2018 Sep 14.

Department of Respiratory Medicine National Hospital Organization Toneyama National Hospital Toyonaka Japan.

Congenital cystic adenomatoid malformation (CCAM) is a benign congenital tumour in which a part of the lung becomes polycystic. Case 1 was a 64-year-old male who was diagnosed with pneumonia, with multiple cysts in the right lower lung lobe, using chest computed tomography (CT). After treatment of the pneumonia, including , a right lower lobectomy was performed. Read More

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http://dx.doi.org/10.1002/rcr2.364DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6138539PMC
November 2018
2 Reads

An investigation on clinical differences between congenital pulmonary airway malformation and bronchial atresia.

J Pediatr Surg 2018 Dec 1;53(12):2390-2393. Epub 2018 Sep 1.

Division of Surgery, National Center for Child Health and Development, Tokyo, Japan.

Background/purpose: Differences in clinical features between congenital pulmonary airway malformation (CPAM) and bronchial atresia (BA) have not yet been clearly described.

Methods: We retrospectively reviewed 112 patients with a pathological diagnosis of CPAM or BA. The clinical parameters were statistically analyzed between these diseases. Read More

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http://dx.doi.org/10.1016/j.jpedsurg.2018.08.031DOI Listing
December 2018
13 Reads

Congenital cystic adenomatous malformation: a case report and a literature review.

Acta Med Litu 2018 ;25(2):95-100

Clinic of Obstetrics and Gynaecology, Institute of Clinical Medicine, Faculty of Medicine, Vilnius University, Vilnius, Lithuania.

Background: A congenital cystic adenomatoid malformation (CCAM) is a foetal pulmonary development abnormality caused by airway dysgenesis that is characterized by cystic or adenomatous lesions in the terminal bronchioles. The size of the mass, the degree of the mediastinal shift, and the presence of hydrops and polyhydramnios can all affect the severity of a case. Treatment can be initiated at early stages by applying prenatal and postnatal methods. Read More

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http://dx.doi.org/10.6001/actamedica.v25i2.3762DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6130925PMC
January 2018
3 Reads

Dichorionic-diamniotic twins: One case each of esophageal atresia and congenital pulmonary airway malformation.

Pediatr Int 2018 Sep 5;60(9):901-903. Epub 2018 Sep 5.

Department of Pediatrics, Takamatsu Red Cross Hospital, Takamatsu, Kagawa, Japan.

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http://dx.doi.org/10.1111/ped.13659DOI Listing
September 2018
2 Reads

Hybrid Lesion: Extralobar Sequestration with Cystic Adenomatoid Malformation - Misdiagnosed as Pulmonary Tuberculosis.

J Coll Physicians Surg Pak 2018 Sep;28(9):S204-S206

Department of Pediatric Surgery, King Edward Medical University, Lahore.

We report a rare case of bronchopulmonary extralobar sequestration in a 9-year girl mimicking pulmonary tuberculosis. Patient had recurrent chest infections and two episodes of massive hemoptysis. Preoperative x-ray chest showed an opacity in left lower lobe and computed tomography-aortogram showed that the lesion had systemic arterial supply from celiac trunk. Read More

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http://dx.doi.org/10.29271/jcpsp.2018.09.S204DOI Listing
September 2018
2 Reads

Hybrid Lesion of Congenital Cystic Adenomatoid Malformation and Bronchopulmonary Sequestration.

J Coll Physicians Surg Pak 2018 Sep;28(9):S201-S203

Department of Pediatrics Surgery, Military Hospital, Rawalpindi.

Hybrid congenital cystic lesions of lung comprising of both congenital cystic adenomatoid malformation (CCAM) and broncho-pulmonary sequestration (BPS) are very rare congenital malformations of the lung. We present a case of a newborn who presented to us with severe respiratory distress and later on found to have a hybrid lesion of the lung. It is a very rare occurrence and no such case has been reported from Pakistan so far. Read More

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http://dx.doi.org/10.29271/jcpsp.2018.09.S201DOI Listing
September 2018
4 Reads

Proximity to the Diaphragm Predicts the Presence of Rhabdomyomatous Dysplasia in Congenital Pulmonary Airway Malformations.

Eur J Pediatr Surg 2019 Feb 13;29(1):49-52. Epub 2018 Aug 13.

Division of Pediatric Surgery, Brown University Warren Alpert Medical School, Providence, Rhode Island, United States.

Introduction:  Rhabdomyomatous dysplasia (RD) is a pathologic finding in CPAMs that was incorrectly attributed to their malignant potential. The increasing recognition of extrathoracic (intradiaphragmatic and intraabdominal) congenital pulmonary airway malformations (CPAMs) offers a clue to the origin of RD. We hypothesize that the presence of RD is related to the CPAM's anatomic location. Read More

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http://dx.doi.org/10.1055/s-0038-1668140DOI Listing
February 2019
4 Reads

[Clinical Report of 128 Cases of Meticulous Thoracoscopic Lobectomy in Children].

Sichuan Da Xue Xue Bao Yi Xue Ban 2018 May;49(3):474-477

Department of Pediatric Surgery,People's Hospital of Guizhou Province,Guizhou 550002,China.

Objective: To investigate the feasibility and effect of precise thoracoscopic lobectomy in children.

Methods: The clinical data of precise thoracoscopic lobectomy in infants and children were analyzed retrospectively in West China Hospital of Sichuan University.

Results: There were total 128 cases (male 69,female 59) of precise thoracoscopic lobectomy in infants and children from Oct,2013 to March,2017,which including 62 cases of congenital cyst of lungs,47 cases of cystic adenomatoid malformations,17 cases of pulmonary sequestrations,and 2 cases of lung tumors. Read More

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May 2018
2 Reads

General Anesthesia With Dexmedetomidine and Remifentanil in a Neonate During Oracotomy and Resection of a Congenital Cystic Adenomatoid Malformation.

J Pediatr Pharmacol Ther 2018 May-Jun;23(3):215-218

Based on animal data, concern has been expressed regarding the potential deleterious neurocognitive effects of general anesthesia during infancy and early life. Although there are no definitive data to prove this effect, the neonatal period has been suggested to be the most vulnerable period. While various inhaled and intravenous anesthetic agents have been implicated, dexmedetomidine and the opioids may be devoid of such effects. Read More

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http://dx.doi.org/10.5863/1551-6776-23.3.215DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6027977PMC
July 2018
12 Reads

Thoracoscopic Resection of Congenital Cystic Adenomatoid Malformation in a Patient With Fused Lung Fissure Using Hookwire.

Innovations (Phila) 2018 May/Jun;13(3):226-229

From the Department of Surgery, The University of Hong Kong, Queen Mary Hospital, Hong Kong, China.

Thoracoscopic resection is being used more commonly for the treatment of congenital cystic adenomatoid malformation in neonates and infants. However, in the rare case of congenital cystic adenomatoid malformation with congenital absence of lung fissure, thoracoscopic lobectomy cannot be performed safely. Moreover, if the lung lesion is deep-seated and cannot be visualized on the pleural surface, wedge resection may result in residual lesion. Read More

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http://dx.doi.org/10.1097/IMI.0000000000000501DOI Listing
December 2018

Elevated Krüppel-like factor 5 expression in spatiotemporal mouse lungs is similar to human congenital cystic adenomatoid malformation of the lungs.

J Int Med Res 2018 Jul 13;46(7):2856-2865. Epub 2018 Jun 13.

1 Department of Prenatal Diagnosis, Women and Children's Hospital of Sichuan Province, Chengdu, China.

Objective The study aimed to investigate the role of high Krüppel-like factor 5 (KLF5) expression on the pathogenesis of congenital cystic adenomatoid malformation of the lungs (CCAML) in mice. Methods A mouse model of high KLF5 expression in the lungs was established. KLF5 expression and the pulmonary lumen diameter were examined by immunohistochemistry to determine a successful model. Read More

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http://dx.doi.org/10.1177/0300060518774998DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6124288PMC
July 2018
4 Reads

[Antenatal ultrasound diagnosis and neonatal results of the congenital cystic adenomatoid malformation of the lung].

Rev Chil Pediatr 2018 Apr;89(2):224-230

Hospital Universitario de Canarias, Santa Cruz de Tenerife, España.

Introduction: Congenital cystic adenomatoid malformation (CCAM) is a rare congenital lung di sease, and in the most of cases, prenatal diagnosis is feasible. There are discrepancies regarding pre natal management and postpartum treatment.

Objective: To analyze prenatally diagnosed CCAM in our hospitals, in order to evaluate ultrasound findings with fetal and postnatal evolution. Read More

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http://dx.doi.org/10.4067/S0370-41062018000200224DOI Listing
April 2018
23 Reads

Congenital pulmonary airway malformation.

Autops Case Rep 2018 Apr-Jun;8(2):e2018022. Epub 2018 May 2.

Hospital Guilherme Alvaro Santos, Department of Pathology . Santos, SP , Brazil.

Congenital pulmonary airway malformation (CPAM) is a developmental malformation of the lower respiratory tract. We report the case of a male newborn diagnosed with cystic lung disease during prenatal ultrasound. A cesarean section was performed at the 32nd gestational week because of premature rupture of the membranes, and soon after the delivery the newborn developed respiratory failure and died. Read More

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http://dx.doi.org/10.4322/acr.2018.022DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5953188PMC
May 2018
2 Reads

Long-Term Neurodevelopmental and Respiratory Outcome after Intrauterine Therapy for Fetal Thoracic Abnormalities.

Fetal Diagn Ther 2019 7;45(3):162-167. Epub 2018 May 7.

Division of Neonatology, Department of Pediatrics, Leiden University Medical Center, Leiden, The

Introduction: The aim of this study is to evaluate long-term neurodevelopmental and respiratory outcome after fetal therapy for fetal pleural effusion, congenital cystic adenomatoid malformation, and bronchopulmonary sequestration.

Methods: Children ≥18 months of age underwent an assessment of neurologic, motor, and cognitive development. Medical records were reviewed to determine respiratory outcome. Read More

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http://dx.doi.org/10.1159/000488486DOI Listing
May 2018
8 Reads

Congenital cystic adenomatoid malformation in adults, presenting as a single cyst.

Asian Cardiovasc Thorac Ann 2018 Jun 2;26(5):407-409. Epub 2018 May 2.

1 Thoracic Surgery Unit, University of Modena and Reggio Emilia, Modena, Italy.

Congenital cystic adenomatoid malformations are lung anomalies usually detected prenatally or in newborns and infants. Type 1 congenital cystic adenomatoid malformations appears as a multicystic lesion, with cysts up to 2 cm in diameter, or as a single large cyst. In the latter case, when detected in adults, the preoperative diagnosis is challenging because congenital cystic adenomatoid malformations can be confused with other more common lesions. Read More

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http://dx.doi.org/10.1177/0218492318776572DOI Listing
June 2018
2 Reads

Congenital cystic adenomatoid malformation: Antenatal ultrasonography features.

Med J Armed Forces India 2018 Apr 7;74(2):177-179. Epub 2017 Apr 7.

Medical Officer (Radiodiagnosis), Military Hospital Allahabad, UP 211001, India.

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http://dx.doi.org/10.1016/j.mjafi.2017.03.006DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5912119PMC
April 2018
1 Read

Congenital cystic adenomatoid malformation - diagnostic and therapeutic procedure: 8-year experience of one medical centre.

Kardiochir Torakochirurgia Pol 2018 Mar 28;15(1):10-17. Epub 2018 Mar 28.

Department of Surgery, Urology and Children Transplantology, Institute of Mother's Polish Health Centre, Lodz, Poland.

Introduction: Congenital cystic adenomatoid malformation (CCAM) is a rare anomaly. The mechanisms and the time at which the abnormality develops are still unclear. The malformation is characterized by the presence of single large or multiple but smaller cysts. Read More

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http://dx.doi.org/10.5114/kitp.2018.74669DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5907611PMC
March 2018
9 Reads

Types II and III congenital pulmonary airway malformation with hydrops treated in utero with percutaneous sclerotherapy.

Prenat Diagn 2018 06 7;38(7):493-498. Epub 2018 May 7.

Department of Obstetrics and Gynecology, Division of Maternal-Fetal Medicine, Keck School of Medicine, University of Southern California, Los Angeles, CA, USA.

Objective: To present outcomes of fetuses with congenital pulmonary airway malformation (CPAM) treated with sclerotherapy.

Methods: Retrospective study of 8 patients with a prenatal diagnosis of CPAM type II or III with secondary hydrops treated with percutaneous sclerotherapy using 5% ethanolamine oleate (EO). All patients underwent ultrasonic measurement of the CPAM volume ratio. Read More

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http://dx.doi.org/10.1002/pd.5266DOI Listing
June 2018
10 Reads

Symptom development in originally asymptomatic CPAM diagnosed prenatally: a systematic review.

Pediatr Surg Int 2018 Jun 9;34(6):613-620. Epub 2018 Apr 9.

Department of Surgery, Children's Hospital of Eastern Ontario, 401 Smyth Rd, Ottawa, ON, K1H 8L1, Canada.

Purpose: For the approximately three quarters of patients with a prenatal diagnosis of congenital pulmonary airway malformation (CPAM) who are asymptomatic at birth, the chance of eventually developing symptoms is unknown. We sought to explore the natural history of asymptomatic CPAM.

Methods: We searched EMBASE, MEDLINE, and the first 50 results from Google Scholar. Read More

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http://dx.doi.org/10.1007/s00383-018-4264-yDOI Listing
June 2018
4 Reads

Congenital pulmonary airway malformation mimicking as pulmonary tuberculosis in five paediatric patients: a diagnostic dilemma.

Trop Doct 2018 Jul 6;48(3):247-250. Epub 2018 Apr 6.

3 Senior Professor and Head, Department of Paediatric Surgery, PGIMS, Rohtak, Haryana, India.

A series of five children with delayed presentation of congenital pulmonary airway malformation (CPAM), all initially misdiagnosed as pulmonary tuberculosis, is described. All five children presented with recurrent respiratory infections and failure to thrive, having shown poor response to multiple antibiotics course, including anti-tubercular therapy for variable periods. Successful surgical intervention was belatedly instigated. Read More

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http://dx.doi.org/10.1177/0049475518765305DOI Listing
July 2018
17 Reads

Asymptomatic congenital lung malformations: Is nonoperative management a viable alternative?

J Pediatr Surg 2018 Jun 5;53(6):1092-1097. Epub 2018 Mar 5.

Section of Pediatric Surgery, Department of Surgery, Michigan Medicine, C.S. Mott Children's and Von Voigtlander Women's Hospital, Ann Arbor, MI.

Introduction: The purpose of this study was to evaluate clinical outcomes in children with asymptomatic congenital lung malformations (CLM) who were initially managed nonoperatively.

Methods: An IRB-approved retrospective review was performed on all CLMs at a single tertiary care referral center (Jan 2006-Dec 2016, n=140). Asymptomatic cases that did not undergo elective resection were evaluated for subsequent CLM-related complications based on clinical records and a telephone quality of life survey. Read More

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http://dx.doi.org/10.1016/j.jpedsurg.2018.02.065DOI Listing
June 2018
5 Reads

Usefulness of fetal magnetic resonance imaging for postnatal management of congenital lung cysts: prediction of probability for emergency surgery.

BMC Pediatr 2018 03 8;18(1):105. Epub 2018 Mar 8.

Department of Pediatric Surgery, Nagoya University Graduate School of Medicine, 65 Tsurumai-cho, Showa-ku, Nagoya, 466-8550, Japan.

Background: Emergency rescue lung resection is rarely performed to treat congenital lung cysts (CLCs) in neonates. Many reports have described fetal CLC treatment; however, prenatal predictors for postnatal respiratory failure have not been characterized. We hypothesized that fetal imaging findings are useful predictors of emergency surgery. Read More

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http://dx.doi.org/10.1186/s12887-018-1085-zDOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5842628PMC
March 2018
10 Reads

Congenital cystic adenomatoid malformation in adults: Report of a case presenting with a recurrent pneumothorax and a literature review of 60 cases.

Respir Med Case Rep 2019 25;26:328-332. Epub 2018 Feb 25.

Division of General Thoracic Surgery, Department of Surgery, Tokai University School of Medicine, Japan.

Congenital cystic adenomatoid malformation (CCAM) is a congenital pulmonary cystic disease that is mostly detected and diagnosed prenatally or during the neonatal period, while rarely being observed in adults. Here, we report an adult case of CCAM that was diagnosed following surgery for a recurrent pneumothorax. We further review 60 case reports on adult CCAM that have been previously published. Read More

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http://dx.doi.org/10.1016/j.rmcr.2018.02.002DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6424061PMC
February 2018
1 Read

Case of mucinous adenocarcinoma of the lung associated with congenital pulmonary airway malformation in a neonate.

Korean J Pediatr 2018 Jan 22;61(1):30-34. Epub 2018 Jan 22.

Department of Pediatrics, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea.

Congenital pulmonary airway malformation (CPAM), previously known as congenital cystic adenomatoid malformation, is a rare developmental lung abnormality associated with rhabdomyosarcoma, pleuropulmonary blastoma, and mucinous adenocarcinoma of the lung. We report an unusual case of a 10-day-old male newborn with a left lower lobe pulmonary cyst who underwent lobectomy, which revealed type II CPAM complicated by multifocal mucinous adenocarcinoma. KRAS sequencing revealed a somatic mutation in Codon12 (GGT → GAT), suggesting the development of a mucinous adenocarcinoma in the background of mucinous metaplasia. Read More

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http://kjp.or.kr/journal/view.php?doi=10.3345/kjp.2018.61.1.
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http://dx.doi.org/10.3345/kjp.2018.61.1.30DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5807988PMC
January 2018
11 Reads

A "blind" vascular ring in association with congenital cystic adenomatoid malformation: A case report.

Medicine (Baltimore) 2017 Dec;96(51):e8915

Department of Maternal-Fetal Medicine, Guangdong Women and Children Hospital, Guangzhou, China.

Rationale: The occurrence of congenital cystic adenomatoid malformation (CCAM) and vascular ring (VR) is extremely rare.

Patient Concerns: We present a case of left CCAM with VR consisting of a left aortic arch and right descending aorta with left tracheal compression causing atelectasis.

Diagnoses: A high-risk male neonate with the diagnosis of left CCAM was diagnosed at 20 weeks gestational age by antenatal ultrasound. Read More

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http://dx.doi.org/10.1097/MD.0000000000008915DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5758127PMC
December 2017
5 Reads

Foregut duplication cyst: a novel computed tomography finding mimicking a small bowel hernia: A case report.

Medicine (Baltimore) 2017 Dec;96(50):e9184

Department of Radiology.

Rationale: A foregut duplication cyst (FDC) is an uncommon congenital disease. This report presents a case of mediastinal foregut duplication cyst that mimicked a diaphragmatic small bowel hernia.

Patient Concern: A 27-month-old girl was first referred for a mediastinal lesion found incidentally on a chest radiograph. Read More

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http://dx.doi.org/10.1097/MD.0000000000009184DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5815740PMC
December 2017
10 Reads

Novel Molecular and Phenotypic Insights into Congenital Lung Malformations.

Am J Respir Crit Care Med 2018 May;197(10):1328-1339

6 Penn Center for Pulmonary Biology, and.

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. Read More

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http://dx.doi.org/10.1164/rccm.201706-1243OCDOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5955056PMC
May 2018
14 Reads

[Cystic adenomatoid malformation of fetus].

Ceska Gynekol Winter 2017;82(6):474-477

Objective: Presentation of prenataly diagnosed fetus with congenital cystic adenomatoid malformation (CCAM). Summary of clinical and histological findings in fetuses with CCAM, in utero ultrasound diagnosis, prognosis, in utero dispensarisation, timing of delivery and postanatal management.

Design: Case report. Read More

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April 2019
7 Reads

Congenital Cystic Adenomatoid Malformation Diagnosed During First-Trimester Ultrasound Scan.

Am J Case Rep 2018 Jan 1;19:1-4. Epub 2018 Jan 1.

Department of Obstetrics and Gynecology, Rene-Dubos Hospital, Cergy-Pontoise, France.

BACKGROUND Congenital cystic adenomatoid malformation (CCAM) is mostly reported from the second trimester of pregnancy. We report a case of a microcystic type of CCAM that was suggested by routine ultrasound examination at a gestational age of 12 weeks. CASE REPORT First-trimester ultrasound screening revealed the presence of a hyperechoic image that occupied the whole of the right lung, without no other any associated complications. Read More

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http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5757404PMC
January 2018
6 Reads

Congenital Pulmonary Malformations Diagnosed Over a Period of 10 Years.

Arch Bronconeumol 2018 Mar 13;54(3):165-167. Epub 2017 Dec 13.

Unidad de Neumología y Alergia Pediátrica, Hospital Universitario Parc Tauli, Barcelona, España.

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http://dx.doi.org/10.1016/j.arbres.2017.08.009DOI Listing
March 2018
9 Reads

A Complication of Percutaneous Sclerotherapy for Congenital Pulmonary Airway Malformation: Intravascular Injection and Cardiac Necrosis.

Fetal Pediatr Pathol 2017 Dec 5;36(6):437-444. Epub 2017 Dec 5.

a Department of Obstetrics and Gynecology, Division of Maternal Fetal Medicine , University of Southern California , Los Angeles , CA , USA.

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). Read More

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https://www.tandfonline.com/doi/full/10.1080/15513815.2017.1
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http://dx.doi.org/10.1080/15513815.2017.1346017DOI Listing
December 2017
21 Reads

Congenital cystic adenomatoid malformation.

Int J Pediatr Adolesc Med 2017 Dec 30;4(4):159-160. Epub 2017 Dec 30.

Pediatric Radiology Fellows, King Faisal Specialist Hospital and Research Centre, Riyadh, Saudi Arabia.

Congenital pulmonary airway malformation of the lung is a rare lesion that commonly affects the lower respiratory tract, which manifests as progressive respiratory distress in the neonatal period. This case is highlighted to raise awareness of the variable evolving morphological and radiological appearance of this entity. Read More

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http://dx.doi.org/10.1016/j.ijpam.2017.12.001DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6372638PMC
December 2017