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Pulmonary alveolar proteinosis

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https://www.readbyqxmd.com/read/27890991/pulmonary-alveolar-proteinosis-experience-from-a-tertiary-care-center-and-systematic-review-of-indian-literature
#1
Vijay Hadda, Pawan Tiwari, Karan Madan, Anant Mohan, Nishkarsh Gupta, Sachidanand Jee Bharti, Vinod Kumar, Rakesh Garg, Anjan Trikha, Deepali Jain, Sudheer Arava, Gopi C Khilnani, Randeep Guleria
BACKGROUND: Pulmonary alveolar proteinosis (PAP) is a rare disorder characterized by deposition of lipoproteinaceous material within alveoli, with a variable clinical course. Here, we report an experience of management of PAP at our center. A systematic review of previously reported cases from India is also included in the article. MATERIALS AND METHODS: This study included patients with primary PAP managed at our center from 2009 to 2015. Diagnosis of primary PAP was based on histopathologic diagnosis on bronchoalveolar lavage or transbronchial lung biopsy and absence of causes of secondary PAP...
November 2016: Lung India: Official Organ of Indian Chest Society
https://www.readbyqxmd.com/read/27879496/death-due-to-pulmonary-alveolar-proteinosis
#2
Emran Altaf, Erik K Mitchel, Chris Berry, Altaf Hossain
Presented are 2 cases of death from pulmonary alveolar proteinosis (PAP). Within the past 2 years, there have been 2 cases of rare nonneoplastic lung disease that consists of the filling of the alveoli of the lung by a periodic acid-Schiff stain-positive lipoproteinaceous material. This condition bears a certain resemblance to interstitial lung disease and/or Pneumocystitis jirovecci infection of the lungs. The presented cases were clinically diagnosed as interstitial lung disease. In the first case presented, the decedent was admitted to hospital with diagnosis of pneumonia but died in hospital despite observation and treatment...
November 23, 2016: American Journal of Forensic Medicine and Pathology
https://www.readbyqxmd.com/read/27851574/1940-pulmonary-alveolar-proteinosis-requiring-ecmo-during-whole-lung-lavage
#3
Douglas Deever, Perry Nystrom
No abstract text is available yet for this article.
December 2016: Critical Care Medicine
https://www.readbyqxmd.com/read/27833966/analysis-of-bronchoalveolar-lavage-fluid-in-a-first-romanian-pulmonary-alveolar-proteinosis-cohort
#4
Oana Claudia Deleanu, Ana Maria Zaharie, Aneta Şerbescu, Floarea Mimi NiŢu, Florin Dumitru MihălŢan, Oana Cristina Arghir
The features of bronchoalveolar lavage fluid (BALF) components and the severity of pulmonary alveolar proteinosis (PAP) were analyzed in the first Romanian cohort of patients admitted to "Marius Nasta" Institute of Pneumophtisiology, Bucharest, Romania. A six-year follow-up study based on bronchoalveolar lavage (BAL) data was performed between January 2007 to December 2012. Study cohort consisted in 20 inpatients diagnosed with PAP, based on BALF cytological findings and÷or on histopathological findings. Demographic, medical history, tobacco use, clinical and radiological features, disease progression with or without whole lung lavage (WLL) therapy were collected...
2016: Romanian Journal of Morphology and Embryology, Revue Roumaine de Morphologie et Embryologie
https://www.readbyqxmd.com/read/27814844/pulmonary-alveolar-proteinosis-crazy-paving-to-whole-lung-lavage
#5
Shahnaz Ul Rehman, Jeffrey R Hammersley
No abstract text is available yet for this article.
November 2016: Mayo Clinic Proceedings
https://www.readbyqxmd.com/read/27783330/inhaled-sargramostim-induces-resolution-of-pulmonary-alveolar-proteinosis-in-lysinuric-protein-intolerance
#6
Laura M Tanner, Johanna Kurko, Maaria Tringham, Heikki Aho, Juha Mykkänen, Kirsti Näntö-Salonen, Harri Niinikoski, Heikki Lukkarinen
Pulmonary alveolar proteinosis (PAP) is a potentially fatal complication of lysinuric protein intolerance (LPI), an inherited disorder of cationic amino acid transport. The patients often present with mild respiratory symptoms, which may rapidly progress to acute respiratory failure responding poorly to conventional treatment with steroids and bronchoalveolar lavations (BALs). The pathogenesis of PAP in LPI is still largely unclear. In previous studies, we have shown disturbances in the function and activity of alveolar macrophages of these patients, suggesting that increasing the activity and the number of macrophages by recombinant human GM-CSF (rhuGM-CSF) might be beneficial in this patient group...
October 26, 2016: JIMD Reports
https://www.readbyqxmd.com/read/27783217/lung-surfactant-metabolism-early-in-life-early-in-disease-and-target-in-cell-therapy
#7
REVIEW
Elena Lopez-Rodriguez, Gemma Gay-Jordi, Adele Mucci, Nico Lachmann, Anna Serrano-Mollar
Lung surfactant is a complex mixture of lipids and proteins lining the alveolar epithelium. At the air-liquid interface, surfactant lowers surface tension, avoiding alveolar collapse and reducing the work of breathing. The essential role of lung surfactant in breathing and therefore in life, is highlighted by surfactant deficiency in premature neonates, which causes neonatal respiratory distress syndrome and results in early death after birth. In addition, defects in surfactant metabolism alter lung homeostasis and lead to disease...
October 25, 2016: Cell and Tissue Research
https://www.readbyqxmd.com/read/27755185/anticytokine-autoantibodies-in-infection-and-inflammation-an-update
#8
Gabriela Barcenas-Morales, Peter Jandus, Rainer Döffinger
PURPOSE OF REVIEW: Concise overview of the field of anticytokine autoantibodies with a focus on recent developments. RECENT FINDINGS: Advances in particular in the analysis of autoantibodies to IFNγ, granulocyte-macrophage colony-stimulating factor (GM-CSF) and type I IFN are presented. The target epitope for anti-IFNγ autoantibodies has been found to have high homology to a protein from Aspergillus suggesting molecular mimicry as a mechanism of breaking self-tolerance...
December 2016: Current Opinion in Allergy and Clinical Immunology
https://www.readbyqxmd.com/read/27743827/-plasmapheresis-failure-in-the-treatment-of-auto-immune-pulmonary-alveolar-proteinosis
#9
A Jézéquel, M Kerjouan, M Lederlin, C Lainé-Caroff, C Camus, P Delaval, S Jouneau
INTRODUCTION: Auto-immune pulmonary alveolar proteinosis is a rare disorder characterized by the accumulation of surfactant proteins in the alveoli. CASE REPORT: We report a case of a 41-year-old smoker, presenting initially with acute respiratory failure. Whole lung lavages were effective initially but only for a few weeks. GM-CSF subcutaneous injections were not effective, and then plasmapheresis were tried. CONCLUSION: This is the fifth report of the use of this treatment in auto-immune pulmonary alveolar proteinosis...
October 13, 2016: Revue des Maladies Respiratoires
https://www.readbyqxmd.com/read/27660154/transbronchial-cryobiopsy-in-diffuse-parenchymal-lung-disease-retrospective-analysis-of-74-cases
#10
Kamonpun Ussavarungsi, Ryan M Kern, Anja C Roden, Jay H Ryu, Eric S Edell
BACKGROUND: Diagnostic evaluation of patients with diffuse parenchymal lung disease (DPLD) is best achieved by a multidisciplinary team correlating clinical, radiologic, and pathologic features. Surgical lung biopsies remain the gold standard for histopathologic diagnosis of idiopathic interstitial pneumonias. Emerging data suggest an increasing role for transbronchial cryobiopsy (TBC) in DPLD evaluation. We describe our experience with TBC in patients with DPLD. METHODS: We retrospectively reviewed medical records of patients with radiographic features of DPLD who underwent TBC at Mayo Clinic, Rochester, Minnesota in June 2013-September 2015...
September 19, 2016: Chest
https://www.readbyqxmd.com/read/27635117/a-new-scale-to-assess-the-severity-and-prognosis-of-pulmonary-alveolar-proteinosis
#11
JiuWu Bai, JinFu Xu, WenLan Yang, Beilan Gao, Weijun Cao, Shuo Liang, Huiping Li
Background. Pulmonary Alveolar Proteinosis (PAP) is a syndrome characterized by pulmonary surfactant accumulation. Small proportion of PAP patients experienced spontaneous remission. Objective. The aim of this study was to assess the severity and prognosis of PAP using various indexes. Methods. Characteristics, PaO2, lung function parameters, and HRCT score of 101 patients with PAP were retrospectively analyzed. Many indexes were explored and integrated into a scale. Results. PaO2 was lower among smokers than among never-smokers...
2016: Canadian Respiratory Journal: Journal of the Canadian Thoracic Society
https://www.readbyqxmd.com/read/27595063/recurrence-of-pulmonary-alveolar-proteinosis-after-bilateral-lung-transplantation-in-a-patient-with-a-nonsense-mutation-in-csf2rb
#12
Masahiro Takaki, Takeshi Tanaka, Yoshihiro Komohara, Yoshiko Tsuchihashi, Daisuke Mori, Kentaro Hayashi, Junya Fukuoka, Naoya Yamasaki, Takeshi Nagayasu, Koya Ariyoshi, Konosuke Morimoto, Koh Nakata
Hereditary pulmonary alveolar proteinosis (PAP) caused by mutations in CSF2RA or CSF2RB, which encode GM-CSF receptor α and β respectively, is a rare disease. Although some experimental therapeutic strategies have been proposed, no clinical evidence has yet been reported. We herein describe the clinical course and recurrence of hereditary PAP after lung transplantation. A 36-year-old woman developed PAP of unknown etiology. She underwent bilateral lung transplantation from living donors at the age of 42 years because of severe respiratory failure complicated by pulmonary fibrosis...
2016: Respiratory Medicine Case Reports
https://www.readbyqxmd.com/read/27592713/successful-treatment-of-autoimmune-pulmonary-alveolar-proteinosis-in-a-pediatric-patient
#13
Mirjana Trukalj, Marija Perica, Željko Ferenčić, Damir Erceg, Marta Navratil, Gzim Redžepi, Boro Nogalo
BACKGROUND Pulmonary alveolar proteinosis (PAP) is a rare condition characterized by the intra-alveolar accumulation of surfactant-derived material, which impairs gas exchange and results in respiratory insufficiency. Two major subtypes of PAP are autoimmune and non-autoimmune PAP. The diagnosis relies on clinical presentation, ground glass opacities on CT scan, bronchoscopy with PAS stain of BAL fluid (BALF), lung biopsy with PAS-positive material in the alveoli, and the presence of anti GM-CSF antibodies in serum or BALF for an autoimmune subtype...
2016: American Journal of Case Reports
https://www.readbyqxmd.com/read/27586802/targeting-gm-csf-in-rheumatoid-arthritis
#14
REVIEW
Ali Berkant Avci, Eugen Feist, Gerd-Rüdiger Burmester
Granulocyte-macrophage colony-stimulating factor (GM-CSF) is well-known as a haemopoietic growth factor. However, it is also essential in regulating functions of mature myeloid cells such as macrophages. Preclinical studies and observations of flares of arthritis in patients following GM-CSF treatment supported its important contribution to the pathogenesis of rheumatoid arthritis (RA). As the most advanced compound, mavrilimumab, a monoclonal antibody against GM-CSF receptor, has already completed phase II trials with a long term of follow-up period of 74 weeks...
July 2016: Clinical and Experimental Rheumatology
https://www.readbyqxmd.com/read/27577926/whole-lung-lavage-therapy-for-pulmonary-alveolar-proteinosis-a-global-survey-of-current-practices-and-procedures
#15
Ilaria Campo, Maurizio Luisetti, Matthias Griese, Bruce C Trapnell, Francesco Bonella, Jan Grutters, Koh Nakata, Coline H M Van Moorsel, Ulrich Costabel, Vincent Cottin, Toshio Ichiwata, Yoshikazu Inoue, Antonio Braschi, Giacomo Bonizzoni, Giorgio A Iotti, Carmine Tinelli, Giuseppe Rodi
BACKGROUND: Whole lung lavage (WLL) is the current standard of care treatment for patients affected by pulmonary alveolar proteinosis (PAP). However, WLL is not standardized and international consensus documents are lacking. Our aim was to obtain a factual portrayal of WLL as currently practiced with respect to the procedure, indications for its use, evaluation of therapeutic benefit and complication rate. METHODS: A clinical practice survey was conducted globally by means of a questionnaire and included 27 centers performing WLL in pediatric and/or adult PAP patients...
2016: Orphanet Journal of Rare Diseases
https://www.readbyqxmd.com/read/27560787/abstract-or001-extracorporeal-membrane-oxygenation-to-support-whole-lung-lavage-in-a-patient-with-pulmonary-alveolar-proteinosis
#16
F N Kaya, A S Bayram, F Kirac, E Basagan-Mogol, S Goren
No abstract text is available yet for this article.
September 2016: Anesthesia and Analgesia
https://www.readbyqxmd.com/read/27560702/pulmonary-pharmacodynamics-of-an-anti-gm-csfr%C3%AE-antibody-enables-therapeutic-dosing-that-limits-exposure-in-the-lung
#17
Jamie Campbell, Josquin Nys, Laura Eghobamien, E Suzanne Cohen, Matthew J Robinson, Matthew A Sleeman
Pulmonary alveolar proteinosis is associated with impaired alveolar macrophage differentiation due to genetic defects in the granulocyte macrophage colony-stimulating factor (GM-CSF) axis or autoantibody blockade of GM-CSF. The anti-GM-CSFRα antibody mavrilimumab has shown clinical benefit in patients with rheumatoid arthritis, but with no accompanying pulmonary pathology observed to date. We aimed to model systemic versus pulmonary pharmacodynamics of an anti-GM-CSFRα antibody to understand the pharmacology that contributes to this therapeutic margin...
October 2016: MAbs
https://www.readbyqxmd.com/read/27548000/pulmonary-fibrosis-on-high-resolution-ct-of-patients-with-pulmonary-alveolar-proteinosis
#18
Masanori Akira, Yoshikazu Inoue, Toru Arai, Chikatoshi Sugimoto, Sayoko Tokura, Koh Nakata, Masanori Kitaichi
OBJECTIVE: The CT findings of pulmonary fibrosis in patients with pulmonary alveolar proteinosis (PAP) are not yet well defined. The objective of this study was to evaluate the CT findings of PAP with a focus on pulmonary fibrosis secondary to PAP. MATERIALS AND METHODS: High-resolution CT (HRCT) scans of 44 patients with PAP were evaluated retrospectively with a focus on pulmonary fibrosis: 33 patients had autoimmune PAP, and 11 patients had secondary PAP. The intervals between the initial and last CT examinations ranged from 1 to 284 months (median, 60 months)...
September 2016: AJR. American Journal of Roentgenology
https://www.readbyqxmd.com/read/27524893/exposure-to-nickel-oxide-nanoparticles-induces-pulmonary-inflammation-through-nlrp3-inflammasome-activation-in-rats
#19
Zhengwang Cao, Yiliang Fang, Yonghui Lu, Fenghua Qian, Qinglong Ma, Mingdi He, Huifeng Pi, Zhengping Yu, Zhou Zhou
With recent advances in the manufacture and application of nickel oxide nanoparticles (NiONPs), concerns about their adverse effects on the respiratory system are increasing. However, the underlying cellular and molecular mechanisms of NiONP-induced pulmonary toxicity remain unclear. In this study, we focused on the impacts of NiONPs on pulmonary inflammation and investigated whether the NLRP3 inflammasome is involved in NiONP-induced pulmonary inflammation and injury. NiONP suspensions were administered by single intratracheal instillation to rats, and inflammatory responses were evaluated at 3 days, 7 days, or 28 days after treatment...
2016: International Journal of Nanomedicine
https://www.readbyqxmd.com/read/27518894/pulmonary-alveolar-proteinosis-in-a-marble-worker
#20
Berna Botan Yildirim, Recep Akgedik, Sukran Akgedik, Hasan Nazaroglu
Pulmonary alveolar proteinosis (PAP) is a rarely seen disease of the alveoli, characterized by accumulation of proteinous material, which stains positive with periodic acid Schiff, in the alveoli. Secondary PAP may develop as a result of occupational exposure to materials such as silica and indium. In the paper, together with a review of the relevant literature, we present an uncommon case of a 47-year old male, marble worker who was diagnosed with PAP associated with a 12-year history of exposure to marble dust...
2016: International Journal of Occupational Medicine and Environmental Health
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