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Nishant Gupta, Dale Langenderfer, Francis X McCormack, Daniel P Schauer, Mark H Eckman
RATIONALE: Patients without a known history of lung disease presenting with a spontaneous pneumothorax are generally diagnosed as primary spontaneous pneumothorax. However, occult diffuse cystic lung diseases such as Birt-Hogg-Dubé syndrome (BHD), lymphangioleiomyomatosis (LAM), and pulmonary Langerhans cell histiocytosis (PLCH) can also first present with a spontaneous pneumothorax, and their early identification by high-resolution computed tomographic (HRCT) imaging of chest has implications for subsequent management...
October 13, 2016: Annals of the American Thoracic Society
Marek Lommatzsch, Kai Bratke, Paul Stoll, Norbert Mülleneisen, Friedrich Prall, Andrea Bier, J Christian Virchow
BACKGROUND: The histologic diagnosis of Pulmonary Langerhans cell histiocytosis (PLCH) is invasive and can cause complications. To confirm the diagnosis of PLCH, guidelines therefore recommend measuring CD1a-positive bronchoalveolar lavage fluid (BALF) cells despite its poor sensitivity and specificity. Thus, an improved diagnostic accuracy of BALF cell analysis would be desirable. METHODS: Using four-colour flow cytometry, plasmacytoid and myeloid dendritic cells (DCs) were analysed in BALF of 10 newly diagnosed, untreated, smoking patients with PLCH, and compared with BALF DCs from 40 asymptomatic smokers and 21 never-smokers...
October 2016: Respiratory Medicine
Fatemeh Faraji, Mohammadreza Mahzounieh, Azizollah Ebrahimi, Fatemeh Fallah, Omid Teymournejad, Behnaz Lajevardi
Pseudomonas aeruginosa possesses various virulence factors which contribute to the bacterial invasion and toxicity. Moreover, children suffered from Cystic Fibrosis (CF) and burn wounds are at a high risk of various bacterial infections. The aim of this study was to determine the prevalence of virulent genes in P. aeruginosa isolated from children with CF and burn wounds and comparing their virulence genes to figure out the role of every virulent factor in the infections. P. aeruginosa were isolated from sputum, oropharyngeal swabs, and broncho-alveolar lavage (BAL) specimens from CF and burn wounds between June 2013 and June 2014 in Tehran's hospitals...
October 2016: Microbial Pathogenesis
Kenji Nemoto, Shuji Oh-Ishi, Toshihide Inui, Mariko Nakazawa, Kentaro Hyodo, Masayuki Nakajima, Jun Kanazawa, Yukiko Miura, Takio Takaku, Yuko Minami, Kenji Hayashihara, Takefumi Saito, Yoshinori Kawabata
Pulmonary arterial hypertension (PAH) secondary to pulmonary Langerhans cell histiocytosis (PLCH) is known to be a relatively common complication and is associated with a poor prognosis. However, the optimal therapeutic approach for these cases remains to be established. A 57-year-old man visited our hospital because of a progressive dry cough. A thoracic computed tomography examination showed a combination of diffuse thick-walled cysts and reticulonodular shadows that were predominant in bilateral upper lobes of the lungs...
2016: Respiratory Medicine Case Reports
Cheng-Wei Li, Man-Hui Li, Jiang-Xiong Li, Ru-Jia Tao, Jin-Fu Xu, Wei-Jun Cao
BACKGROUND: Pulmonary Langerhans cell histiocytosis (PLCH) is an orphan disease in respiratory medicine, which most affects adult smokers. The purpose of this article was to discuss the clinical features, especially the radiologic features of PLCH patients during their hospitalization through a retrospective analysis on clinical data. Furthermore, the current literature was also reviewed. METHODS: Between December 2008 and June 2012, 14 patients with PLCH were assessed at Shanghai Pulmonary Hospital, Tongji University School of Medicine, Shanghai, China...
June 2016: Journal of Thoracic Disease
Mariano Fielli, Fabio Avila, Agustina Saino, Deborah Seimah, Marcelo Fernández Casares
The diffuse cystic lung diseases (DCLDs) are a pathophysiologically heterogeneous processes characterized by the presence of multiple thin-walled, air-filled spaces within the pulmonary parenchyma. The most common causes of DCLD are lymphangioleiomyomatosis (LAM) and pulmonary Langerhans cell histiocytosis (PLCH). DCLD develops rarely as a result of malignancy, typically secondary to metastases from peripheral sarcomas and mesenchymal tumors. DCLD have also been reported in a variety of other metastatic disease such as adenocarcinoma...
2016: Respiratory Medicine Case Reports
Mihaela Georgescu, Irina Gheorghe, Carmen Curutiu, Veronica Lazar, Coralia Bleotu, Mariana-Carmen Chifiriuc
BACKGROUND: The purpose of this study was to evaluate the virulence profiles of Pseudomonas aeruginosa clinical strains recently isolated from patients hospitalized for chronic leg ulcers in the Dermatology Department of Central Military Emergency University Hospital "Carol Davila", Bucharest, Romania. METHODS: The phenotypic screening evaluated eight soluble virulence factors (haemolysins, lecithinase, lipase, caseinase, gelatinase, amylase, DNase, aesculin hydrolysis), as well as adherence ability (Cravioto adapted method) and invasion capacity on HeLa cells (gentamicin protection assay)...
2016: BMC Infectious Diseases
Zohreh Heidary, Eshagh Bandani, Mohamad Eftekhary, Ali Akbar Jafari
Virulent and resistant strains Pseudomonas aeruginosa (P. aeruginosa) is one of the most important cause of UTIs in pediatrics. The present study was carried to investigate the frequency of virulence factors in the multi-drug resistant strains of P. aeruginosa isolated from pediatrics hospitalized due to the UTIs. One-hundred and forty three urine samples were collected from pediatric patients suffered from UTIs. Samples were cultured and those that were P. aeruginosa positive were analyzed for the presence of putative virulence genes...
March 2016: Acta Medica Iranica
Yoshiaki Kinoshita, Kentaro Watanabe, Atsuhiko Sakamoto, Kouko Hidaka
Pulmonary Langerhans cell histiocytosis (PLCH) is a rare, smoking-related, interstitial lung disease, and pulmonary hypertension (PH) is associated with mortality. We herein report a case of PLCH complicated by severe PH and respiratory impairment. After developing PH, the patient displayed a cystic pattern on chest high-resolution computed tomography (HRCT). This, in turn, corresponded with the scarring stage of PLCH. However, the patient's PH and respiratory impairment improve dramatically following smoking cessation...
2016: Internal Medicine
Anja C Roden, Eunhee S Yi
CONTEXT: Pulmonary Langerhans cell histiocytosis (PLCH) is a rare histiocytic disorder that almost exclusively affects the lungs of smokers. PLCH is characterized by bronchiolocentric nodules and/or cysts in an upper and mid lung distribution with sparing of the costophrenic angles. The diagnosis can be challenging and often requires transbronchial biopsy or surgical lung biopsy. Pulmonary hypertension is a relatively common and sometimes severe complication of PLCH. The pathogenesis of PLCH is still debated...
March 2016: Archives of Pathology & Laboratory Medicine
Michal Kamionek, Parnian Ahmadi Moghaddam, Ali Sakhdari, Alexandra E Kovach, Matthew Welch, Xiuling Meng, Karen Dresser, Keith Tomaszewicz, Ediz F Cosar, Eugene J Mark, Armando E Fraire, Lloyd Hutchinson
AIMS: Pulmonary Langerhans cell histiocytosis (PLCH) is an idiopathic cigarette smoking-related disorder of the lung. Molecular changes in cellular or fibrotic stages of PLCH have not been investigated. We studied the prevalence of extracellular signal-regulated kinase (ERK) pathway mutations in different PLCH stages and other non-PLCH smoking-related lung diseases. METHODS AND RESULTS: The cohort included 28 PLCH with cellular (n = 10), mixed cellular/fibrotic (n = 4) and fibrotic histology (n = 14)...
September 2016: Histopathology
Abdellatif Tazi, Constance de Margerie-Mellon, Laetitia Vercellino, Jean Marc Naccache, Stéphanie Fry, Stéphane Dominique, Stéphane Jouneau, Gwenaël Lorillon, Emmanuelle Bugnet, Raphael Chiron, Benoit Wallaert, Dominique Valeyre, Sylvie Chevret
BACKGROUND: An important objective on diagnosis of patients with Langerhans cell histiocytosis (LCH) is to determine the extent of disease. However, whether systematic extrathoracic investigation is needed in adult patients with clinically isolated pulmonary LCH (PLCH) has not been evaluated. METHODS: In this prospective, multicentre study, 54 consecutive patients with newly diagnosed clinically isolated PLCH were systematically evaluated at inclusion by bone imaging and blood laboratory testing to search for subclinical extrapulmonary LCH involvement...
2016: Orphanet Journal of Rare Diseases
J Temporal, L Armstrong, N Bhatt, A R L Medford
No abstract text is available yet for this article.
March 2016: QJM: Monthly Journal of the Association of Physicians
Fatma Ibrahim Sonbol, Maha Abd El Fattah Khalil, Abdelfattah Badr Mohamed, Sameh Samir Ali
BACKGROUND/AIM: Virulent Pseudomonas aeruginosa. is frequently life-threatening and often challenging to treat, and the emergence of multidrug-resistant isolates presents a critical problem for patients. The aim of the study was concerned with molecular analysis of the virulence factors and antimicrobial resistance profile of multidrug-resistant P. aeruginosa (MDRPA). MATERIALS AND METHODS: Out of 44 MDRPA isolates, 12 isolates representing different resistance profiles and sources of samples were selected for further molecular studies...
2015: Turkish Journal of Medical Sciences
Adam May, Garvan Kane, Eunhee Yi, Robert Frantz, Robert Vassallo
Pulmonary Langerhans cell histiocytosis (PLCH) is an uncommon diffuse lung disease characterized by the abnormal accumulation of Langerhans' cells around small airways and other distal lung compartments. Although pulmonary hypertension (PH) is a frequent complication of PLCH, the role of advanced PH therapies for PLCH-related PH is not well-established. We describe a PLCH patient with severe, disease-related PH that responded unexpectedly well to advanced PH therapy with sustained improvement over a 10 year follow-up period...
2015: Respiratory Medicine Case Reports
Tatyana E Fontenot, Neal Viradia, Elizabeth Pollard, Harold R Neitzschman
PURPOSE: Obtaining a tissue sample diagnostic of pulmonary Langerhans cell histiocytosis (PLCH) by transbronchial biopsy is notoriously difficult. The condition's appearance on computed tomography is well described and singularly characteristic, perhaps adequate for definitive diagnosis. We propose an approach to diagnosis of these patients. METHODS: Radiology case report of PLCH in a middle-aged female smoker with two week history of nonproductive cough, low grade fevers, and fatigue...
January 2015: Journal of the Louisiana State Medical Society: Official Organ of the Louisiana State Medical Society
Limin Gao, Huifang Li, Gandi Li, Weiping Liu, Jinnan Li, Wenyan Zhang
We report an uncommon 22-year-old male Pulmonary Langerhans Cell Histiocytosis (PLCH) case which co-existed with pulmonary tuberculosis (TB). Unlike the common PLCH cases, this PLCH case has cervical lymph node involvement and right pneumothorax. The diagnosis was established by the imaging of lung and the biopsies of the lung and left neck lymph node. Imaging of the chest showed characteristic small nodules and thin-walled cysts and right pneumothorax. The LCH cells in the lung and left neck lymph node were characterized by large convoluted nuclei with cerebriform indentations of the nuclear envelope and longitudinal grooves...
2015: International Journal of Clinical and Experimental Pathology
Davide Elia, Olga Torre, Roberto Cassandro, Antonella Caminati, Sergio Harari
BACKGROUND: Pulmonary Langerhans cell histiocytosis (PLCH) is a rare interstitial disease affecting primarily young adult smokers. In order to highlight the clinical features of the disease, we conducted a retrospective analysis on clinical data of PLCH patients followed at our center; moreover, we reviewed the current literature on PLCH. METHOD AND RESULTS: Between January 2004 and July 2014, 40 patients with PLCH were evaluated at our Division. The average patients' age was 40 (± 14) years, and 22 of them were females...
June 2015: European Journal of Internal Medicine
Abdellatif Tazi, Constance de Margerie, Jean Marc Naccache, Stéphanie Fry, Stéphane Dominique, Stéphane Jouneau, Gwenaël Lorillon, Emmanuelle Bugnet, Raphael Chiron, Benoit Wallaert, Dominique Valeyre, Sylvie Chevret
BACKGROUND: The natural history of pulmonary Langerhans cell histiocytosis (PLCH) has been unclear due to the absence of prospective studies. The rate of patients who experience an early progression of their disease is unknown. Additionally, conflicting effects of smoking cessation on the outcome of PLCH have been reported. METHODS: In this prospective, multicentre study, 58 consecutive patients with newly diagnosed PLCH were comprehensively evaluated over a two-year period...
2015: Orphanet Journal of Rare Diseases
Lalita Fernandes, Rohit Vadala, Anthony Menezes Mesquita, Pradeep Vaideeswar
Adult Pulmonary Langerhans Cell Histiocytosis (PLCH) is a rare interstitial lung disease which occurs almost exclusively in smokers. A marked male predominance was initially reported, but recent studies show both men and women are equally affected due to the increasing smoking habits in women. The natural history is variable with 25% of patients having asymptomatic disease while 10-20% progress rapidly to respiratory insufficiency and death. The diagnosis is not easily recognized by clinicians or pathologists...
January 2015: Indian Journal of Tuberculosis
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