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https://www.readbyqxmd.com/read/29030294/type-i-interferon-pathway-activation-in-copa-syndrome
#1
Stefano Volpi, Jessica Tsui, Marcello Mariani, Claudia Pastorino, Roberta Caorsi, Oliviero Sacco, Angelo Ravelli, Anthony K Shum, Marco Gattorno, Paolo Picco
Mutations of the COPA gene cause an immune dysregulatory disease characterised by polyarticular arthritis and progressive interstitial lung disease with pulmonary haemorrhages. We report the case of a young girl that presented at age 3 with polyarticular arthritis, chronic cough and high titer rheumatoid factor. Radiologic imaging showed interstitial lung disease with tree-in-a-bud nodules and air-filled cysts. Targeted genetic analysis of COPA gene showed the reported c.698G>A mutation. The patient was lost to follow up for 3years during which therapy was discontinued with the development of joint damage and deformities...
October 10, 2017: Clinical Immunology: the Official Journal of the Clinical Immunology Society
https://www.readbyqxmd.com/read/29028157/transthoracic-catheter-drainage-for-large-symptomatic-congenital-pulmonary-airway-malformation
#2
Seong Hee Oh, Chae Young Kim, Byong Sop Lee, Dong Kwan Kim, Ellen Ai-Rhan Kim, Ki-Soo Kim
BACKGROUND: Surgical resection of large symptomatic congenital pulmonary airway malformation (CPAM) in newborns has high risks of mortality and postoperative morbidity. This study aimed to report the clinical outcomes of newborns who underwent percutaneous transthoracic catheter drainage (PTCD) of large symptomatic CPAM before surgical resection. METHODS: This was a retrospective, descriptive study based on review of the medical records of newborn infants who required surgical resection of large symptomatic CPAM at a single tertiary hospital from 2001 to 2017...
October 13, 2017: Pediatric Pulmonology
https://www.readbyqxmd.com/read/29026365/incidental-finding-of-bronchopulmonary-sequestration-in-a-64-year-old-female
#3
Pichapong Tunsupon, Ayesha Arshad, Sumit Patel, M Jeffery Mador
BACKGROUND: Bronchopulmonary sequestration is a congenital abnormality of the primitive foregut. In adults, the typical age at presentation is 20-25 years. CASE REPORT: A 64-year-old female was referred for evaluation of an 8 × 6-cm right lower lobe cystic lesion. Her medical history was significant for recurrent right lower lobe pneumonia requiring multiple hospitalizations. Her physical examination was significant for crackles at the right lung base. Computed tomography (CT) of the chest with contrast showed cystic changes with thickened septation of the medial segment of the right lower lobe lacking distinct visceral pleura and with arterial supply from the anomalous branch of the thoracic aorta arising near the celiac trunk...
2017: Ochsner Journal
https://www.readbyqxmd.com/read/28990897/chronic-necrotizing-pulmonary-aspergillosis-in-an-immunocompetent-patient-after-the-surgery-of-hydatid-cyst
#4
Nagehan Emiralioğlu, Hatice Nursun Özcan, Diclehan Orhan, Mithat Haliloğlu, Ali Bülent Cengiz, Elmas Ebru Yalçın, Deniz Doğru Ersöz, Nural Kiper, Hayriye Uğur Özçelik
Chronic necrotizing pulmonary aspergillosis (CNPA) is a condition caused by the ubiquitous fungus Aspergillus fumigatus in non-immunocompromised individuals. Numerous underlying conditions have been associated with CNPA. Tuberculosis, non-tuberculous mycobacterial infection and allergic bronchopulmonary aspergillosis (ABPA) remain the predominant risk factors for development of CNPA. Development of CNPA in echinococcal cyst cavities is very rare and the optimal therapeutic regimen and treatment duration have not been established...
June 2017: Tüberküloz Ve Toraks
https://www.readbyqxmd.com/read/28981362/the-abcs-of-bhd-an-in-depth-review-of-birt-hogg-dub%C3%A3-syndrome
#5
Shiva Gupta, Hyunseon C Kang, Dhakshinamoorthy Ganeshan, Ajaykumar Morani, Rabindra Gautam, Peter L Choyke, Vikas Kundra
OBJECTIVE: Birt-Hogg-Dubé (BHD) syndrome is an autosomal dominant inherited syndrome involving multiple organs. In young patients, renal neoplasms that are multiple, bilateral, or both, such as oncocytomas, chromophobe renal cell carcinoma (RCC), hybrid chromophobe RCC-oncocytomas, clear cell RCC, and papillary RCC, can suggest BHD syndrome. Extrarenal findings, including dermal lesions, pulmonary cysts, and spontaneous pneumothoraces, also aid in diagnosis. CONCLUSION: Radiologists may be one of the first medical specialists to suggest the diagnosis of BHD syndrome...
October 5, 2017: AJR. American Journal of Roentgenology
https://www.readbyqxmd.com/read/28977397/pulmonary-cyst-embolism-a-rare-complication-of-hydatidosis
#6
Eleonore Ravis, Alexis Theron, Bernard Lecomte, Vlad Gariboldi
Hydatid disease is an endemic parasitosis that results from the ingestion of echinococcosis tapeworm eggs. This condition leads to the formation of cysts, mainly in the liver and lungs, and causes life-threatening complications. Cardiac involvement represents only 0.5-2% of the localizations. We report a rare case of a pulmonary cyst embolism that required emergency surgical intervention.
August 1, 2017: European Journal of Cardio-thoracic Surgery
https://www.readbyqxmd.com/read/28971108/hydatid-cyst-of-the-interventricular-septum
#7
Endale Tefera, Joseph Knapp, Michael Teodori
While cardiac involvement is not a common presentation in human echinococcosis, it may lead to life-threatening complications including cyst rupture; anaphylactic shock; tamponade; pulmonary, cerebral or peripheral arterial embolism; acute coronary syndrome; dysrhythmias; infection; ventricular or valvular dysfunction, as well as sudden death. Here we report a 9-year old girl who was diagnosed to have hydatid cyst of the interventricular septum four years after diagnosis and medical treatment of pulmonary hydatidosis...
March 31, 2017: Global Cardiology Science & Practice
https://www.readbyqxmd.com/read/28958014/intracardiac-and-pulmonary-artery-hydatidosis-causing-thromboembolic-pulmonary-hypertension
#8
Gökçen Orhan, Murat Bastopcu, Bülent Aydemir, Mehmet Sait Ersoz
Hydatidosis is a serious parasitic infection in endemic areas. A rare presentation is pulmonary arterial cysts causing thromboembolic pulmonary hypertension. We report the case of a young man who presented with clinical and radiological findings of thromboembolic pulmonary hypertension. The patient was found to have hydatid cysts in both pulmonary arteries and in the right ventricular outflow tract. To remove all cysts without causing rupture, anaphylactic shock or systemic emboli, placing the patient under cardiopulmonary arrest was necessary, and in the case of pulmonary arterial involvement, total circulatory arrest was necessary...
September 7, 2017: European Journal of Cardio-thoracic Surgery
https://www.readbyqxmd.com/read/28953671/castleman-disease-associated-diffuse-parenchymal-lung-disease-a-strobe-compliant-retrospective-observational-analysis-of-22-cases-in-a-tertiary-chinese-hospital
#9
Hui Huang, Ruie Feng, Jian Li, Xinyu Song, Shan Li, Kai Xu, Jian Cao, Lu Zhang, Yalan Bi, Zuojun Xu
Intrathoracic involvement is common in Castleman disease (CD), but CD-associated diffuse parenchymal lung disease (DPLD) is rare and not well-reported.We conducted a retrospective analysis of 262 CD patients with a definite pathological diagnosis who were hospitalized between 1999 and 2015.Twenty-two CD patients had DPLD based on chest computed tomography (CT) scans. Among them, 9 were male and 13 were female, with a mean age of 45.3 years. Coughing (72.7%), fever (68.2%), and dyspnea (59.1%) were the common clinical manifestations...
September 2017: Medicine (Baltimore)
https://www.readbyqxmd.com/read/28924092/extralobar-pulmonary-sequestration-in-two-pinniped-species
#10
Shotaro Nakagun, Hirotaka Watanabe, Kenji Ochiai, Kaoru Kohyama, Wataru Goshima, Yoko Goto, Yumi Kobayashi, Kennichi Watanabe, Noriyuki Horiuchi, Jun Sasaki, Masanobu Goryo, Yoshiyasu Kobayashi
Two cases of extralobar pulmonary sequestrations from a walrus (Odobenus rosmarus) and a Steller sea lion (Eumetopias jubatus) are described in the present study. Grossly, an independent, soft unilocular cystic mass was found within the abdominal cavities of both animals, adherent to the diaphragm in O. rosmarus and attached to the cardia of the stomach in E. jubatus. Histopathologically, the cysts were lined by pseudostratified ciliated columnar epithelium with abundant goblet cells, while the wall comprised of glands, hyaline cartilage, bronchiole- and alveolus-like structures, smooth muscles, and large, well-developed elastic and muscular arteries...
September 17, 2017: Journal of Veterinary Medical Science
https://www.readbyqxmd.com/read/28899426/benign-metastasizing-leiomyoma-presenting-as-multiple-cystic-pulmonary-nodules-a-case-report
#11
Yeong Hun Choe, So Yeon Jeon, Yoon Chae Lee, Myung Ja Chung, Seung Yong Park, Yong Chul Lee, So Ri Kim
BACKGROUND: Benign metastatic leiomyoma (BML) is an extremely rare disease. Although uterine leiomyomas are benign histologically, they can metastasize to distant sites. While the incidence is very low, the lung is the organ most frequently affected by BML. Pulmonary BML usually presents as numerous well-defined nodules of various sizes, while the cavitary or cystic features in the nodules are rarely observed on radiologic images. CASE PRESENTATION: A 52-year-old woman complained of cough and dyspnea for one month...
September 12, 2017: BMC Women's Health
https://www.readbyqxmd.com/read/28893673/extracorporeal-membrane-oxygenation-ecmo-as-salvage-treatment-for-pulmonary-echinococcus-granulosus-infection-with-acute-cyst-rupture
#12
Sören L Becker, Sebastian Fähndrich, Franziska C Trudzinski, Barbara Gärtner, Frank Langer, Torben K Becker, Robert Bals, Philipp M Lepper, Christian Lensch
Extracorporeal membrane oxygenation (ECMO) has been used successfully for the treatment of patients with respiratory failure due to severe infections. Although rare, parasites can also cause severe pulmonary disease. Tapeworms of the genus Echinococcus give rise to the development of cystic structures in the liver, lungs, and other organs. Acute cyst rupture leads to potentially life-threatening infection, and affected patients may deteriorate rapidly. The case of a young woman from Bulgaria who was admitted to hospital with severe dyspnoea, progressive chest pain, and haemoptysis is described...
September 8, 2017: International Journal of Infectious Diseases: IJID
https://www.readbyqxmd.com/read/28883209/is-anatomic-lung-resection-necessary-in-surgical-treatment-of-giant-lung-hydatid-cysts-in-childhood
#13
Omer Onal, Omer Faruk Demir
PURPOSE: The aim of this study is to evaluate the results of parenchymal saving methods for giant lung hydatid cysts and to discuss the necessity of anatomic lung resection in childhood. METHODS: The patients under the age of 16 years who were operated between January 2000 and January 2017 due to pulmonary hydatid cyst were evaluated retrospectively (n = 200). In all, 32 patients who had giant hydatid cyst were included in this study. Parenchymal saving methods (cystotomy-capitonnage) were preferred and decortication was also performed for pleural thickening if needed...
September 5, 2017: Annals of Thoracic and Cardiovascular Surgery
https://www.readbyqxmd.com/read/28878832/concomitant-lung-and-liver-hydatid-cyst-managed-as-one-stage-surgery
#14
Usha Dalal, Ashwani Kumar Dalal, Rikki Singal
OBJECTIVE: Over the world, hydatidosis is endemic in many countries. It is more prevalent in Turkey. We came across with concomitant hydatidosis of the lung and liver and reviewed the management. MATERIAL AND METHODS: This is a prospective study that was carried out in the Government Medical College and Hospital, sector-32, Chandigarh, India, between 2004 and 2010, in the Department of Surgery. A total of five patients diagnosed with concomitant liver and pulmonary hydatid disease underwent surgery...
January 2017: Mædica
https://www.readbyqxmd.com/read/28877576/unusual-location-of-hydatid-cysts-report-of-two-cases-in-the-heart-and-hip-joint-of-romanian-patients
#15
Simona Gurzu, Marius Alexandru Beleaua, Emeric Egyed-Zsigmond, Ioan Jung
Hydatid cyst is usually located in the liver and lungs, rare cases showing localization in other organs or tissues. In the unusual location, echinococcosis is an excluding diagnosis that is established only after microscopic evaluation. Our first case occurred in a 67-year-old female previously diagnosed with pulmonary tuberculosis and hospitalized with persistent pain in the hip joint. The clinical diagnosis was tuberculosis of the joint, but the presence of the specific acellular membrane indicated a hydatid cyst of the synovial membrane, without bone involvement...
August 2017: Korean Journal of Parasitology
https://www.readbyqxmd.com/read/28872098/is-a-diagnostic-video-assisted-thoracoscopic-thymectomy-an-acceptable-first-line-approach-to-the-suspicious-anterior-mediastinal-mass
#16
Ricky Vaja, Vijay Joshi, Alan G Dawson, David A Waller
INTRODUCTION: The incidental early-stage thymic mass presents a diagnostic challenge. Video-assisted thoracoscopic (VAT) thymectomy is an attractive but potentially morbid solution. The aim was to show it can be safely applied as a first-line modality in those with undiagnosed thymic enlargement with acceptable long-term results. METHODS: A total of 45 patients were identified (24 male, median age 52 interquartile range [IQR]: 41-66 years) in a 14-year experience who had CT evidence of an enlarged, possibly malignant thymic mass, but no tissue diagnosis before undertaking VAT thymectomy...
October 2017: Journal of Minimal Access Surgery
https://www.readbyqxmd.com/read/28862061/pulmonary-metastasis-of-undifferentiated-uterine-sarcoma-forming-fluid-containing-cyst
#17
Genki Usui, Hirotsugu Hashimoto, Jun Matsumoto, Masaaki Akahane, Hajime Tsunoda, Yuko Sasajima, Hajime Horiuchi
Undifferentiated uterine sarcoma (UUS) is a high-grade sarcoma with no specific differentiation; however, it exhibits variable positivity for CD10 as an immunohistochemical marker of endometrial stroma, suggesting immunohistochemical differentiation into endometrial stroma. It has been reported that some low-grade endometrial stromal sarcoma pulmonary metastatic foci show cystic changes; however, whether pulmonary metastatic UUS foci are solid or cystic remains undescribed. A 63-year-old woman underwent a hysterectomy...
August 1, 2017: International Journal of Surgical Pathology
https://www.readbyqxmd.com/read/28840031/bronchopulmonary-foregut-malformation-presenting-as-extralobar-pulmonary-sequestration-associated-with-a-bronchogenic-cyst-an-unusual-clinical-and-radiological-feature-in-an-adolescent-patient
#18
Ji Yung Choo, Jinwook Hwang, Joo Han Lee, Ki Yeol Lee
We present a case of symptomatic complex bronchopulmonary foregut malformation (BPFM), including extralobar pulmonary sequestration and a bronchogenic cyst, in the left anterior mediastinum of a 15-year-old boy. Preoperative computed tomography showed a cystic mass with heterogeneous enhancement of adjacent soft tissue components and pleural effusion. We suggested the infected bronchogenic cyst as the first impression. However, pathological examination after surgical resection revealed extralobar pulmonary sequestration and a bronchogenic cyst with unusual manifestation, which was located in the left upper hemithorax and supplied by the pulmonary artery...
July 2017: Journal of Thoracic Disease
https://www.readbyqxmd.com/read/28839995/clinical-and-genetic-study-of-a-large-chinese-family-presented-with-familial-spontaneous-pneumothorax
#19
Huajie Xing, Yanguo Liu, Guanchao Jiang, Xiao Li, Yanyan Hou, Fan Yang, Jun Wang
BACKGROUND: Familial spontaneous pneumothorax (FSP) is an inherited disease, and Birt-Hogg-Dubé (BHD) syndrome is its leading cause. BHD syndrome is an autosomal dominant disorder characterized by pulmonary cysts, spontaneous pneumothorax, renal cancer, and skin fibrofolliculomas. It is caused by germline mutations in the FLCN gene. Thus far a variety of mutations have been reported; however, the unique characteristics of BHD syndrome-related FSP are still unclear. METHOD: We reviewed the family history of a large Chinese family that presented with FSP...
July 2017: Journal of Thoracic Disease
https://www.readbyqxmd.com/read/28835429/a-rare-case-of-pulmonary-toxoplasmosis-in-a-patient-with-undifferentiated-inflammatory-arthritis-on-chronic-methotrexate-and-corticosteroid-therapy
#20
Abdullateef Abdulkareem, Ryan Steven D'Souza, Nitin Patel, Anthony A Donato
Pulmonary toxoplasmosis is a serious pulmonary condition caused by the protozoan Toxoplasma gondii It typically affects immunocompromised patients presenting acutely with cough, fever, myalgias, arthralgias and lymphadenopathy, and chronically with persistent cough and dyspnoea. Because of its protean features, it can mimic many more common lung conditions in the immunocompromised patient, including atypical pneumonia, Pneumocystis pneumonia and interstitial lung disease. In this article, we present the case of a 55-year-old woman who presented to our hospital with persistent dyspnoea and cough, initially suspected to have an arthritis-related interstitial lung disease...
August 23, 2017: BMJ Case Reports
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