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Pulmonary Alveolar Proteinosis

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Inhaled granulocyte-macrophage colony stimulating factor for mild-to-moderate autoimmune pulmonary alveolar proteinosis - a phase II randomized study for 6 months and follow-up to 24 months

Kai-Feng Xu, Xinlun Tian, Yanli Yang, Lulu Chen, Xin Sui, Wenshuai Xu, Xue Li, Xiaobei Guo, Lingshan Liu, Yusen Situ, Jun Wang, Yang Zhao, Shuzhen Meng, Wei Song, Yonglong Xiao,

<title>Abstract</title> <p> Background: Treatment of autoimmune pulmonary alveolar proteinosis (aPAP) by inhaled granulocyte-macrophage colony stimulating factor (GM-CSF) is considered safe and effective. Evidences of benefit from GM-CSG inhalation for mild to moderate aPAP patients are limited. Methods: In this multicenter, randomized, open-labeled clinical trial, 36 aPAP patients with ... Read more >>

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How to do whole lung lavage for treatment of pulmonary alveolar proteinosis.

Tovi Vo, Justin C Y Chan, Michael Worthington,

Pulmonary alveolar proteinosis is a rare condition characterized by progressive accumulation of surfactant lipoproteins in the alveolar space, leading to poor gas exchange. We describe our technique of whole lung lavage for management of pulmonary alveolar proteinosis. ... Read more >>

ANZ J Surg (ANZ journal of surgery)
[2020, :]

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Proteogenomic analysis of granulocyte macrophage colony- stimulating factor autoantibodies in the blood of a patient with autoimmune pulmonary alveolar proteinosis.

Atsushi Hashimoto, Shiho Takeuchi, Ryo Kajita, Akira Yamagata, Ryota Kakui, Takahiro Tanaka, Koh Nakata,

Recently, attempts to reveal the structures of autoantibodies comprehensively using improved proteogenomics technology, have become popular. This technology identifies peptides in highly purified antibodies by using an Orbitrap device to compare spectra from liquid chromatography-tandem mass spectrometry against a cDNA database obtained through next-generation sequencing. In this study, we first ... Read more >>

Sci Rep (Scientific reports)
[2020, 10(1):4923]

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Pulmonary alveolar proteinosis in Korea: analysis of prevalence and incidence via a nationwide population-based study.

Hee-Young Yoon, Ji Hyeon Kim, Ye-Jee Kim, Jin Woo Song,

BACKGROUND:Pulmonary alveolar proteinosis (PAP) is a very rare lung disease and its prevalence and incidence remain unclear. The prevalence and incidence of PAP were investigated by using nationwide claims data from the Korean Health Insurance Review and Assessment service. METHODS:Data were extracted for adults who visited any secondary or tertiary ... Read more >>

BMC Pulm Med (BMC pulmonary medicine)
[2020, 20(1):34]

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Improvement of native pulmonary alveolar proteinosis after contralateral single living-donor lobar lung transplantation: A case report.

Kazuma Kobayashi, Shinya Ohkouchi, Yoji Sasahara, Masahito Ebina, Koh Nakata, Ryoko Saito, Miki Akiba, Tetsu Sado, Hisashi Oishi, Tatsuaki Watanabe, Hajime Kurosawa, Yoshinori Okada,

PAP is a rare disease characterized by the accumulation of surfactant materials in the alveolar spaces due to the imbalance of surfactant homeostasis (production and clearance). We herein report a case of an 8-year-old girl who developed PAP after BMT from her mother for the treatment of DBA. The anemia ... Read more >>

Pediatr Transplant (Pediatric transplantation)
[2020, 24(2):e13659]

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Validation of a new serum granulocyte-macrophage colony-stimulating factor autoantibody testing kit.

Koh Nakata, Tatsuki Sugi, Keiko Kuroda, Kazutaka Yoshizawa, Toshinori Takada, Ryushi Tazawa, Takahiro Ueda, Ami Aoki, Mitsuhiro Abe, Koichiro Tatsumi, Ryosuke Eda, Shotaro Kondoh, Konosuke Morimoto, Takeshi Tanaka, Etsuro Yamaguchi, Ayumu Takahashi, Miku Oda, Haruyuki Ishii, Shinyu Izumi, Haruhito Sugiyama, Atsushi Nakagawa, Keisuke Tomii, Masaru Suzuki, Satoshi Konno, Shinya Ohkouchi, Taizou Hirano, Tomohiro Handa, Toyohiro Hirai, Yoshikazu Inoue, Toru Arai, Katsuaki Asakawa, Takuro Sakagami, Takahiro Tanaka, Ayako Mikami, Nobutaka Kitamura,

Very recently, a modest but significant efficacy of granulocyte-macrophage colony-stimulating factor (GM-CSF) inhalation therapy for the treatment of mild to moderate autoimmune pulmonary alveolar proteinosis (aPAP) has been reported. As the ability to measure the level of GM-CSF autoantibody (GMAb) in the serum is required to decide the indication for ... Read more >>

ERJ Open Res (ERJ open research)
[2020, 6(1):]

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A novel approach to conducting clinical trials in the community setting: utilizing patient-driven platforms and social media to drive web-based patient recruitment.

Janelle Applequist, Cristina Burroughs, Artemio Ramirez, Peter A Merkel, Marc E Rothenberg, Bruce Trapnell, Robert J Desnick, Mustafa Sahin, Jeffrey P Krischer,

BACKGROUND:Participant recruitment for clinical research studies remains a significant challenge for researchers. Novel approaches to recruitment are necessary to ensure that populations are easier to reach. In the context of rare diseases, social media provides a unique opportunity for connecting with patient groups that have representatively lower diagnosis rates when ... Read more >>

BMC Med Res Methodol (BMC Medical Research Methodology)
[2020, 20(1):58]

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[Whole lung lavage in an infant with pulmonary alveolar proteinosis and literature review].

K An, J Bai, H B Gu, H L Chen, B R Li, B T Ning, Y Wang, J Qian,

Objective: To investigate the safety, feasibility and operation key points of whole lung lavage in infants with pulmonary alveolar proteinosis. Methods: The clinical manifestations, genetic screening, therapeutic interventions and outcome of an infant with pulmonary alveolar proteinosis complicated with respiratory failure who received whole lung lavage in November 2018 in ... Read more >>

Zhonghua Er Ke Za Zhi (Zhonghua er ke za zhi = Chinese journal of pediatrics)
[2020, 58(1):46-50]

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Natural Autoantibodies in Chronic Pulmonary Diseases.

Kiyoharu Fukushima, Kazuyuki Tsujino, Shinji Futami, Hiroshi Kida,

In autoantibody-mediated autoimmune diseases, pathogenic autoantibodies generated by a failure of central or peripheral tolerance, have different effects mediated by a variety of mechanisms. Interestingly, even non-autoimmune chronic diseases have a set of disease-specific natural autoantibodies that are maintained for a long time. Because most of these natural autoantibodies target ... Read more >>

Int J Mol Sci (International journal of molecular sciences)
[2020, 21(3):]

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Therapeutic effect of subcutaneous injection of low dose recombinant human granulocyte-macrophage colony-stimulating factor on pulmonary alveolar proteinosis.

Fen Zhang, Dong Weng, Yiliang Su, Chengsheng Yin, Li Shen, Yuan Zhang, Ying Zhou, Qiuhong Li, Yang Hu, Huiping Li,

OBJECTIVE:To observe the efficacy of recombinant human granulocyte-macrophage colony-stimulating factor (rhGM-CSF) for pulmonary alveolar proteinosis (PAP). MATERIALS AND METHODS:A total of 55 patients with PAP were screened at Shanghai Pulmonary Hospital between May 2014 and May 2018. Among these, 42 were diagnosed with idiopathic PAP, 24 were included in this ... Read more >>

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

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Secondary Pulmonary Alveolar Proteinosis Following Treatment With Azacitidine for Myelodysplastic Syndrome.

Miki Hashimoto, Hidehiro Itonaga, Yasuhito Nannya, Hirokazu Taniguchi, Yuichi Fukuda, Takafumi Furumoto, Machiko Fujioka, Sachie Kasai, Masataka Taguchi, Hiroaki Taniguchi, Shinya Sato, Yasushi Sawayama, Sunao Atogami, Keisuke Iwasaki, Tomoko Hata, Hiroshi Soda, Yukiyoshi Moriuchi, Koh Nakata, Seishi Ogawa, Yasushi Miyazaki,

Secondary pulmonary alveolar proteinosis (sPAP) is a complication of myelodysplastic syndrome (MDS). A 60-year-old woman was diagnosed with MDS with excess blasts-1. Fifty-four months after the initial diagnosis, treatment with azacitidine was initiated. Seventy-three months after the diagnosis, a bone marrow examination revealed increased myeloblasts, at which time computed tomography ... Read more >>

Intern. Med. (Internal medicine (Tokyo, Japan))
[2019, :]

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Crazy Paving in Pulmonary Alveolar Proteinosis.

Brian W Allwood, Sami Bennji,

N. Engl. J. Med. (The New England journal of medicine)
[2020, 382(3):275]

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Inhaled GM-CSF for Pulmonary Alveolar Proteinosis. Reply.

Ryushi Tazawa, Nobutaka Kitamura, Koh Nakata,

N. Engl. J. Med. (The New England journal of medicine)
[2020, 382(2):198]

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Inhaled GM-CSF for Pulmonary Alveolar Proteinosis.

Spyros A Papiris, Matthias Griese, Effrosyni D Manali,

N. Engl. J. Med. (The New England journal of medicine)
[2020, 382(2):197]

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Distinct interferon signatures and cytokine patterns define additional systemic autoinflammatory diseases.

Adriana A de Jesus, Yangfeng Hou, Stephen Brooks, Louise Malle, Angelique Biancotto, Yan Huang, Katherine R Calvo, Bernadette Marrero, Susan Moir, Andrew J Oler, Zuoming Deng, Gina A Montealegre Sanchez, Amina Ahmed, Eric Allenspach, Bita Arabshahi, Edward Behrens, Susanne Benseler, Liliana Bezrodnik, Sharon Bout-Tabaku, AnneMarie C Brescia, Diane Brown, Jon M Burnham, Maria Soledad Caldirola, Ruy Carrasco, Alice Y Chan, Rolando Cimaz, Paul Dancey, Jason Dare, Marietta DeGuzman, Victoria Dimitriades, Ian Ferguson, Polly Ferguson, Laura Finn, Marco Gattorno, Alexei A Grom, Eric P Hanson, Philip J Hashkes, Christian M Hedrich, Ronit Herzog, Gerd Horneff, Rita Jerath, Elizabeth Kessler, Hanna Kim, Daniel J Kingsbury, Ronald M Laxer, Pui Y Lee, Min Ae Lee-Kirsch, Laura Lewandowski, Suzanne Li, Vibke Lilleby, Vafa Mammadova, Lakshmi N Moorthy, Gulnara Nasrullayeva, Kathleen M O'Neill, Karen Onel, Seza Ozen, Nancy Pan, Pascal Pillet, Daniela Gp Piotto, Marilynn G Punaro, Andreas Reiff, Adam Reinhardt, Lisa G Rider, Rafael Rivas-Chacon, Tova Ronis, Angela Rösen-Wolff, Johannes Roth, Natasha Mckerran Ruth, Marite Rygg, Heinrike Schmeling, Grant Schulert, Christiaan Scott, Gisella Seminario, Andrew Shulman, Vidya Sivaraman, Mary Beth Son, Yuriy Stepanovskiy, Elizabeth Stringer, Sara Taber, Maria Teresa Terreri, Cynthia Tifft, Troy Torgerson, Laura Tosi, Annet Van Royen-Kerkhof, Theresa Wampler Muskardin, Scott W Canna, Raphaela Goldbach-Mansky,

BACKGROUNDUndifferentiated systemic autoinflammatory diseases (USAIDs) present diagnostic and therapeutic challenges. Chronic interferon (IFN) signaling and cytokine dysregulation may identify diseases with available targeted treatments.METHODSSixty-six consecutively referred USAID patients underwent underwent screening for the presence of an interferon signature using a standardized type-I IFN-response-gene score (IRG-S), cytokine profiling, and genetic evaluation ... Read more >>

J. Clin. Invest. (The Journal of clinical investigation)
[2020, :]

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Knowledge of Secondary Pulmonary Alveolar Proteinosis Complicated With Myelodysplastic Syndrome.

Ishii Haruyuki,

Intern. Med. (Internal medicine (Tokyo, Japan))
[2019, :]

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ABC Transporters: An Overlooked Mechanism of Drug Failure in Our Preclinical Models?

Elizabeth F Redente,

Am. J. Respir. Cell Mol. Biol. (American journal of respiratory cell and molecular biology)
[2020, 62(2):130-131]

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Aggravation of atherosclerosis by pulmonary exposure to indium oxide nanoparticles.

Dong-Keun Lee, Hyung Seok Jang, Hyunji Chung, Soyeon Jeon, Jiyoung Jeong, Jae-Hoon Choi, Wan-Seob Cho,

The use of indium oxide (In2O3) and indium-metal hybrids for various applications, including the manufacture of batteries and liquid crystal displays, increases the chances of human exposure to In2O3 via inhalation, especially in occupational settings. However, there is little information available on the toxic effects of In2O3 nanoparticles (NPs) on ... Read more >>

Nanotoxicology (Nanotoxicology)
[2020, 14(3):355-371]

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Spectrum of 18F-FDG Uptake in Bilateral Lung Parenchymal Diseases on PET/CT.

Meghana Prabhu, Shobhana Raju, Dhritiman Chakraborty, Saurabh Arora, Rakesh Kumar,

Bilateral lung parenchymal involvement is seen in infective as well as noninfective conditions, appearing as focal or diffuse lung disease. PET/CT with FDG helps in characterization (increased glucose utilization is seen by both inflammatory and neoplastic cells). In this article, we describe the spectrum of patterns of FDG uptake and ... Read more >>

Clin Nucl Med (Clinical nuclear medicine)
[2020, 45(1):e15-e19]

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Pneumocystis jirovecii colonization and its association with pulmonary diseases: a multicenter study based on a modified loop-mediated isothermal amplification assay.

Ting Xue, Zhuang Ma, Fan Liu, Weiqin Du, Li He, Jinyan Wang, Chunli An,

BACKGROUND:Pneumocystis jirovecii (P. jirovecii) is an opportunistic fungal pathogen and the role of its colonization in pulmonary diseases has become a popular focus in recent years. The aim of this study was to develop a modified loop-mediated isothermal amplification (LAMP) assay for detection of Pneumocystis jirovecii (P. jirovecii) DNA amongst ... Read more >>

BMC Pulm Med (BMC pulmonary medicine)
[2020, 20(1):70]

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The early onset and persistent worsening pulmonary alveolar proteinosis in rats by indium oxide nanoparticles.

Sung-Hyun Kim, Soyeon Jeon, Dong-Keun Lee, Seonghan Lee, Jiyoung Jeong, Jong Sung Kim, Wan-Seob Cho,

Workplace inhalation exposure to indium compounds has been reported to produce 'indium lung disease' characterized by pulmonary alveolar proteinosis (PAP), granulomas, and pulmonary fibrosis. However, there is little information about the pulmonary toxicity of nano-sized indium oxide (In2O3), which is widely used in various applications such as liquid crystal displays. ... Read more >>

Nanotoxicology (Nanotoxicology)
[2019, :1-11]

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[Rare occupational respiratory diseases].

Anna Witkowska, Marta Wiszniewska, Jolanta Walusiak-Skorupa,

This paper reviews rare occupational respiratory diseases with uncommon causes. Among others, it refers to the Ardystil syndrome characterized by the occurrence of organizing pneumonia in the textile printing sprayers, as a result of inhalation of substances used in aerographic textile printing. Furthermore, secondary pulmonary alveolar proteinosis due to exposure ... Read more >>

Med Pr (Medycyna pracy)
[2020, 71(1):89-104]

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Pulmonary alveolar proteinosis (PAP) in idiopathic hypoparathyroidism.

Soma Saha, Karan Madan, Deepali Jain, Ravinder Goswami,

Idiopathic hypoparathyroidism (IH) and autoimmune pulmonary alveolar proteinosis (PAP) are rare disorders. A patient with IH and optimal calcaemic control on calcium and alfacalcidol was detected to have PAP after 8 years of follow-up. Patient had no respiratory complaints. Routine abdominal imaging for renal calcification showed patchy ground glass opacities ... Read more >>

BMJ Case Rep (BMJ case reports)
[2019, 12(11):]

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Valuable Serum Markers in Pulmonary Alveolar Proteinosis.

Shenyun Shi, Lulu Chen, Xiaohua Qiu, Qi Zhao, Yonglong Xiao, Xin Yan,

Objective:Several serum markers were reported to reflect the severity of pulmonary alveolar proteinosis (PAP). The aim of this study is to investigate a reliable and facile marker to access and monitor the clinical course of PAP in a large cohort. Methods:PAP patients from January 2010 to June 2018 were enrolled. ... Read more >>

Dis. Markers (Disease markers)
[2019, 2019:9709531]

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Pulmonary alveolar proteinosis and pulmonary Kaposi sarcoma occurring simultaneously in a perinatally infected HIV patient: A case report.

Katelyn Klimowich, Richard Rutstein, Joseph Piccione,

Pediatr. Pulmonol. (Pediatric pulmonology)
[2020, 55(2):285-287]

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