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Lauryn A Benninger, Julia A Ross, Marino E Leon, Raju Reddy
Pleural effusions occur in up to 70% of cases of malignant pleural mesothelioma (MPM). However, MPM rarely presents as a chylous effusion making it a diagnostic challenge. There are only six reported cases to date. Most cases of chylothoraces due to malignancy are due to lymphoma or bronchogenic carcinoma. We report an interesting case of MPM in a 75-year-old man who presented with recurrent chylothorax. He reported a four-month history of dyspnea and chest discomfort. Chest x-ray revealed a pleural effusion...
2018: Respiratory Medicine Case Reports
Ilina Datkhaeva, Valerie A Arboleda, T Niroshi Senaratne, Gelareh Nikpour, Cherise Meyerson, Yipeng Geng, Yalda Afshar, Emily Scibetta, Jeffrey Goldstein, Fabiola Quintero-Rivera, Barbara F Crandall, Wayne W Grody, Joshua Deignan, Carla Janzen
Nonimmune hydrops fetalis (NIHF) is a rare disorder with a high perinatal mortality of at least 50%. One cause of NIHF is generalized lymphatic dysplasia (GLD), a rare form of primary lymphedema of the extremities and systemic involvement including chylothoraces and pericardial effusions. An autosomal recessive form of GLD has been described, caused by variants in the PIEZO1 gene. It has been reported clinically to cause NIHF and childhood onset of facial and limb lymphedema, most of which were diagnosed postnatally...
December 2018: American Journal of Medical Genetics. Part A
Jason M Weissler, Eugenia H Cho, Peter F Koltz, Martin J Carney, Maxim Itkin, Pablo Laje, L Scott Levin, Yoav Dori, Suhail K Kanchwala, Stephen J Kovach
With the expanding horizon of microsurgical techniques, novel treatment strategies for lymphatic abnormalities are increasingly reported. Described in this article is the first reported use of lymphovenous anastomosis surgery to manage recalcitrant chylothoraces in infants. Chylothorax is an increasingly common postoperative complication after pediatric cardiac surgery, with a reported incidence of up to 9.2 percent in infants. Although conservative nutritional therapy has a reported 70 percent success rate in this patient population, failed conservative management leading to persistent chylothorax is associated with a significant risk of multisystem complications and mortality...
June 2018: Plastic and Reconstructive Surgery
Sumedh S Hoskote, Hemang Yadav, Prashant Jagtap, Dennis A Wigle, Craig E Daniels
BACKGROUND: Postoperative chylothorax in children is associated with an increased risk of vascular thrombosis, hypothesized to be from loss of antithrombin into chylous fluid resulting in a hypercoagulable state. In adults, an increased thrombotic risk with chylothorax has not been described. Adults undergoing Ivor-Lewis esophagogastrectomy have two strong thrombotic risk factors-active malignancy and postoperative state-allowing for relative homogeneity in baseline thrombotic risk; therefore, we studied the association of chylothorax with thrombosis in this population...
April 2018: Annals of Thoracic Surgery
Rajiv N Srinivasa, Jeffrey Forris Beecham Chick, Anthony N Hage, Joseph J Gemmete, Douglas C Murrey, Ravi N Srinivasa
BACKGROUND: To report approach, technical success, safety, and short-term outcomes of thoracic duct stent-graft reconstruction for the treatment of chylothorax. METHODS: Two patients, 1 (50%) male and 1 (50%) female, with mean age of 38 years (range: 16-59 years) underwent endolymphatic thoracic duct stent-graft reconstruction between September 2016 and July 2017. Patients had radiographic left-sided chylothoraces (n = 2) from idiopathic causes (n = 1) and heart transplantation (n = 1)...
April 2018: Annals of Vascular Surgery
Alexandra Martirossian, Shiwan Shah, Lola Carrete, Jose Valle, Vincent Valentine
Lymphangioleiomyomatosis (LAM), a rare, multisystem disorder primarily affecting women of reproductive age, is characterized by cystic-appearing lung lesions, progressive loss of lung function, chylous effusions and renal angiomyolipomas. Sirolimus, an mammalian target of rapamycin inhibitor, has been shown to stabilize lung function, reduce symptoms and resolve chylous effusions in the short term for patients with LAM. Herein, we report a premenopausal female with LAM who experienced complete and durable resolution of her chylothoraces with significant and sustained improvement in lung function on sirolimus...
December 2017: American Journal of the Medical Sciences
Gregory Nadolski
Nontraumatic chylothorax is a relatively rare condition in which the intestinal lymph (chyle) leaks into the pleural cavity. Nontraumatic chylothorax is more difficult to treat than the more common traumatic chylothorax because the site of chylous leak may occur in less predictable locations. In the past, patients with nontraumatic chylothoraces were offered traditional fluoroscopically guided lymphangiography and thoracic duct embolization similar to traumatic chylothorax. However, the observation that thoracic duct embolization outcomes for nontraumatic chylothorax differed based on the imaging findings during lymphangiography has led to the development of a treatment algorithm, which incorporates noninvasive diagnostic studies, such as magnetic resonance lymphangiography...
December 2016: Techniques in Vascular and Interventional Radiology
Jessica Hellyer, Hunter Oliver-Allen, Majid Shafiq, Alisha Tolani, Maurice Druzin, Michael Jeng, Stanley Rockson, Robert Lowsky
Introduction Gorham-Stout Disease (GSD) is a rare disorder of bony destruction due to lymphangiomatosis, and is often triggered by hormones. One complication of GSD is the development of chylothorax, which carries a high mortality rate. Very little experience has been published to guide management in GSD during pregnancy to optimize both fetal and maternal health. Case Study A 20-year-old woman with known GSD presented with shortness of breath at 18 weeks of pregnancy, due to bilateral chylothoraces which required daily drainage...
October 2016: American Journal of Perinatology Reports
T G Pillay, B Singh
BACKGROUND: Traumatic chylothorax is an extremely rare complication following thoracic trauma or surgery. The aetiology of traumatic chylothorax is dominated by iatrogenic causes, with a reported incidence of 0.5% to 3% following oesophageal surgery. The mortality from a chylothorax post oesophagectomy can be as high as 50%. Iatrogenic causes in total account for approximately 80% of traumatic causes. Non-iatrogenic traumatic chylothoraces are exceedingly uncommon. The complication rate in blunt thoracic trauma is said to be 0...
March 2016: Injury
Oren W Johnson, Jeffrey Forris Beecham Chick, Nikunj Rashmikant Chauhan, Alexandra Holmsen Fairchild, Chieh-Min Fan, Michael S Stecker, Timothy P Killoran, Alisa Suzuki-Han
UNLABELLED: The thoracic duct is the body's largest lymphatic conduit, draining upwards of 75 % of lymphatic fluid and extending from the cisterna chyli to the left jugulovenous angle. While a typical course has been described, it is estimated that it is present in only 40-60% of patients, often complicating already challenging interventional procedures. The lengthy course predisposes the thoracic duct to injury from a variety of iatrogenic disruptions, as well as spontaneous benign and malignant lymphatic obstructions and idiopathic causes...
August 2016: European Radiology
Vincent Nguyen, Hannah Vassaur, F Paul Buckley
Chylous ascites is an accumulation of milky lipid-rich lymph in the peritoneal cavity. Spontaneous chyle leak is a rare occurrence, often associated with malignancy or cirrhosis. This is a report of spontaneous chylous ascites, chylomediastinum and chylothoraces encountered in the setting of an incarcerated paraesophageal hernia. A 60-year-old female presented to the emergency department with epigastric pain, dysphagia and vomiting. Clinical presentation and imaging were concerning for an incarcerated paraesophageal hernia, and the patient was taken to the operating room...
2015: Journal of Surgical Case Reports
Elisavet Fotiou, Silvia Martin-Almedina, Michael A Simpson, Shin Lin, Kristiana Gordon, Glen Brice, Giles Atton, Iona Jeffery, David C Rees, Cyril Mignot, Julie Vogt, Tessa Homfray, Michael P Snyder, Stanley G Rockson, Steve Jeffery, Peter S Mortimer, Sahar Mansour, Pia Ostergaard
Generalized lymphatic dysplasia (GLD) is a rare form of primary lymphoedema characterized by a uniform, widespread lymphoedema affecting all segments of the body, with systemic involvement such as intestinal and/or pulmonary lymphangiectasia, pleural effusions, chylothoraces and/or pericardial effusions. This may present prenatally as non-immune hydrops. Here we report homozygous and compound heterozygous mutations in PIEZO1, resulting in an autosomal recessive form of GLD with a high incidence of non-immune hydrops fetalis and childhood onset of facial and four limb lymphoedema...
September 3, 2015: Nature Communications
T K Susheel Kumar, Saradha Subramanian, Shyam Sathanandam, John Alexander, Mohammed Ali, Christopher J Knott-Craig
BACKGROUND: Thrombosis and occlusion of the superior vena cava (SVC) can cause massive chylothorax resulting in significant morbidity and mortality among young infants. Medical therapy is often unsuccessful. We report a new surgical technique that entails open thrombectomy and reconstruction of the SVC and innominate vein to treat this condition. METHODS: The charts of 4 consecutive infants with chylothoraces refractory to conservative management were reviewed. The operations were performed on cardiopulmonary bypass without myocardial arrest...
October 2015: Annals of Thoracic Surgery
Margaret E Clark, Russell K Woo, Sidney M Johnson
PURPOSE: Medical management of congenital chylothoraces consists of total parental nutrition and tube thoracostomy. However, these infants are exposed to significant fluid shifts and the related leukopenia carries a high infection risk. The purpose of this review is to describe the technique of parietal pleural clipping as a surgical treatment of congenital chylothorax. METHODS: The medical records of all patients with a chylothorax diagnosis during the study period of January 2002 to April 2014 were retrospectively reviewed...
December 2015: Pediatric Surgery International
Ji Hyun Bang, Sang Hwa Kim, Chun Soo Park, Jeong-Jun Park, Tae-Jin Yun
OBJECTIVE: Thoracic duct mass ligation (TDML) through a right thoracotomy (RT), regardless of the side of the pleural effusion, is a standard procedure for chylothorax that is refractory to medical treatment. This procedure may be unsuccessful in patients with complex congenital heart disease, which necessitates additional left thoracotomy (LT) for left periaortic mass ligation. We hypothesized that failure of the right-sided approach is attributable to the anatomic variation of the path of the thoracic duct...
September 2015: Journal of Thoracic and Cardiovascular Surgery
Sarah Joyce, Kristiana Gordon, Glen Brice, Pia Ostergaard, Rani Nagaraja, John Short, Sandra Moore, Peter Mortimer, Sahar Mansour
The RASopathies, which include Noonan syndrome (NS) and Cardiofaciocutaneous syndrome (CFC), are autosomal dominant disorders with genetic heterogeneity associated with germline mutations of genes in the Ras/mitogen-activated protein kinase (MAPK; RAS-MAP kinase) pathway. The conditions overlap and are characterised by facial dysmorphism, short stature and congenital heart disease. NS and CFC, in particular, are known to be associated with lymphatic problems, but this has not been well characterised to date...
May 2016: European Journal of Human Genetics: EJHG
Malcolm Brodlie, Sara Abdelgalil, Sahar Mansour, David A Spencer
Generalised Lymphatic Dysplasia is a rare condition that may be associated with significant chylothoraces. The management of such effusions is often challenging. We present the case of a 15-year-old girl with bilateral chylothoraces and lymphoedema of her limbs. A clinical diagnosis of Generalised Lymphatic Dysplasia was made and long-term treatment with somatostatin analogues (somatostatin initially followed by monthly octreotide) was initiated. Over 12 months there was symptomatic benefit with some objective improvement in lung function and no adverse effects...
2012: Respiratory Medicine Case Reports
Himanshu Bhardwaj, Bhaskar Bhardwaj, Ahmed Awab
Chylothorax is an unusual type of pleural effusion which results from the accumulation of chyle in the pleural cavity. High triglyceride content and presence of chylomicrons in the chyle give this fluid a characteristic milky appearance. Chylothorax most commonly results from the obstruction of the thoracic duct by a malignant lesion or from its traumatic disruption. Liver cirrhosis is an uncommon and frequently underappreciated cause of chylothorax. Pleural effusion in chylothorax is typically described as a lymphocytic predominant, exudative type and it is exceedingly rare to encounter a transudative type of chylothorax...
July 2015: Heart & Lung: the Journal of Critical Care
T K Pandian, Chad Hamner
The etiologies of pediatric lung injury requiring surgical intervention can be infectious, traumatic, congenital, or iatrogenic. Childhood pneumonia is a significant global health problem affecting 150 million children worldwide. Sequelae of pulmonary infections potentially requiring surgery include bronchiectasis, lung abscess, pneumatocele, and empyema. Trauma, congenital conditions such as cystic fibrosis and iatrogenic injuries can result in pneumothoraces, chylothoraces, or bronchopleural fistulae. Recurrence rates for spontaneous pneumothorax treated non-operatively in pediatric patients approach 50-60%...
February 2015: Seminars in Pediatric Surgery
Jaymin B Morjaria, Imran Aslam, Brian Johnson, Michael A Greenstone, Jack A Kastelik
Bilateral chylothorax is a rare cause of pleural effusions. Here we report an unusual acute presentation of bilateral chylothorax following thoracic outlet surgery. Unique to this case was the disparate characteristics of pleural fluid analyses with an exudate on the left and a transudate on the right. This report describes the recognition and management of bilateral chylothoraces, an uncommon but potentially serious complication of this frequently performed surgical procedure.
January 2015: Therapeutic Advances in Chronic Disease
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