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Capillary leakage syndrome

N M Al-Namnam, P Nambiar, P Shanmuhasuntharam, M Harris
Dengue is a mosquito transmitted flaviviral infection which can give rise to severe haemorrhage (dengue haemorrhagic fever/DHF) and with capillary leakage induces hypovolemic shock (dengue shock syndrome/DSS). Although dengue symptoms and complications have been known for many decades there has only been one documented case of osteonecrosis of the maxilla which was treated by excision of the necrotic bone. In this case of dengue infection, extensive maxillary osteonecrosis and minimal root resorption appeared to follow factitious injury with a toothpick but resolved with nonsurgical management...
October 15, 2016: Australian Dental Journal
Radu Campean, Matthias Hasun, Claudia Stöllberger, Johannes Bucher, Josef Finsterer, Christoph Schnack, Franz Weidinger
BACKGROUND: Reversible left ventricular dysfunction, also termed Takotsubo cardiomyopathy, is rarely reported in Addison's disease after initiation of hormone replacement therapy. The pathogenesis of this cardiomyopathy is unknown. CASE PRESENTATION: A 41-year-old white woman with a history of autoimmune Hashimoto thyroiditis diagnosed 3 years earlier and acute adrenal insufficiency diagnosed 3 weeks earlier presented with new onset of heart failure New York Heart Association class IV, which had started shortly after initiation of hormone replacement therapy with hydrocortisone 20 mg/day and fludrocortisone 0...
October 12, 2016: Journal of Medical Case Reports
Muhammad Abdul Mabood Khalil, Muhammad Salman Ghazni, Jackson Tan, Nazish Naseer, Muhammad Ashhad Ullah Khalil
Ovarian hyperstimulation syndrome (OHSS) was first described in 1960. It may occur as a complication of gonadotropin hormone therapy during assisted pregnancy or for primary infertility. A 26-year-old female patient with polycystic ovarian syndrome and primary infertility was treated to conceive. She received intravenous gonadotropin-releasing hormone (GnRH) along with follicle-stimulating hormone in an outside private clinic. She presented to the emergency department with abdominal and chest pain, loose stool, vomiting, shortness of breath and decreasing urine output...
May 2016: Case Reports in Gastroenterology
Kwadwo Kyeremanteng, Gianni D'Egidio, Cynthia Wan, Alan Baxter, Hans Rosenberg
Objective. To describe a single case of Systemic Capillary Leak Syndrome (SCLS) with a rare complication of compartment syndrome. Patient. Our patient is a 57-year-old male, referred to our hospital due to polycythemia (hemoglobin (Hgb) of 220 g/L), hypotension, acute renal failure, and bilateral calf pain. Measurements and Main Results. The patient required bilateral forearm, thigh, and calf fasciotomies during his ICU stay and continuous renal replacement therapy was instituted following onset of acute renal failure and oliguria...
2016: Case Reports in Critical Care
T Ikezoe, J Yang, C Nishioka, K Umezawa, A Yokoyama
Recombinant human soluble thrombomodulin (rTM) counteracted capillary leakage and alleviated edema in individuals with sinusoidal obstruction syndrome and engraftment syndrome after hematopoietic stem cell transplantation. We previously showed that rTM increased levels of antiapoptotic protein Mcl-1 and protected endothelial cells from calcineurin inhibitor cyclosporine A (CsA)-induced apoptosis. However, the molecular mechanisms by which rTM enhances barrier function in vascular endothelial cells remain unknown...
September 19, 2016: Bone Marrow Transplantation
Yuri C Martins, Brandi D Freeman, Oscar B Akide-Ndunge, Louis M Weiss, Herbert B Tanowitz, Mahalia S Desruisseaux
Plasmodium berghei ANKA infection of C57BL/6 mice is a widely used model of experimental cerebral malaria (ECM). By contrast, the nonneurotropic P. berghei NK65 (PbN) causes severe malarial disease in C57BL/6 mice but does not cause ECM. Previous studies suggest that endothelin-1 (ET-1) contributes to the pathogenesis of ECM. In this study, we characterize the role of ET-1 on ECM vascular dysfunction. Mice infected with 10(6) PbN-parasitized red blood cells were treated with either ET-1 or saline from 2 to 8 days postinfection (dpi)...
September 15, 2016: American Journal of Pathology
Aurélien Guffroy, Benjamin Dervieux, Simon Gravier, Camille Martinez, Joelle Deibener-Kaminsky, Eric Hachulla, Marc Michel, Jean-Christophe Weber, Anne-Sophie Korganow, Laurent Arnaud, Jacques-Eric Gottenberg, Jean Sibilia
OBJECTIVES: Systemic capillary leak syndrome (Clarkson's disease) is a rare entity characterized by recurrent and unpredictable attacks of capillary leakage of plasma fluid and proteins throughout the endothelium. Some cases are secondary. We describe the rare association between secondary capillary leak syndrome (SCLS) and autoimmune diseases. METHODS: We conducted a nationwide, retrospective, observational, and collaborative study throughout the hospital units of the Club des Rhumatismes et Inflammations network (CRI) between March and August 2015...
August 12, 2016: Seminars in Arthritis and Rheumatism
Daniel C Moreira, Christopher J Ng, Ralph Quinones, Xiayuan Liang, Dominic W Chung, Jorge Di Paola
Idiopathic systemic capillary leak syndrome (SCLS) is a rare process characterized by acute and recurrent episodes of vascular leakage with severe hypotension, hypoalbuminemia, hemoconcentration and edema. Anemia and thrombocytopenia are not part of this syndrome, but here we present the case of a pediatric patient with a clinical presentation consistent with SCLS who subsequently developed microangiopathic hemolytic anemia at a time when she had significant fluid loss and anasarca. Based on serial ADAMTS-13 levels, we propose that the anemia in this patent developed as a result of ADAMTS-13 loss in the third-space fluid, a novel mechanism for acquired microangiopathic hemolytic anemia...
September 13, 2016: Journal of Thrombosis and Haemostasis: JTH
Tuula K Outinen, Outi K Laine, Satu Mäkelä, Ilkka Pörsti, Heini Huhtala, Antti Vaheri, Jukka Mustonen
BACKGROUND: Hantaviruses cause hemorrhagic fever with renal syndrome (HFRS) and hantavirus cardiopulmonary syndrome (HCPS) in humans. Hantavirus infections are characterized by thrombocytopenia. Our objective was to assess the association of thrombocytopenia with disease severity in HFRS induced by Puumala hantavirus (PUUV). METHODS: Altogether 546 patients treated for acute serologically confirmed PUUV infection during 1982-2013 at Tampere University Hospital, Finland, were examined...
September 2016: Infectious Diseases
V V Kuz'kov, E V Fot, A A Smjotkin, K M Lebedinskij, M Yu Kirov
Current guidelines suggest that an early and aggressive fluid therapy is the best rescue approach to restore and preserve cardiac index, organ function and decrease the risk of multiple organ failure in shock of various origin. However, escala- tion of fluid resuscitation is a double-edged sword often associated with reperfusion, glicocalyx injury, capillary leakage, delayed weight gain and heperhydration. The body of evidences demons trates that an excessive fluid load in ICUpatient with global increased permeability syndrome, and, particularly, in ARDS and acute kidney injury can be devastating, particularly when guided with central venous pressure...
November 2015: Anesteziologiia i Reanimatologiia
J N Gonzales, R Lucas, A D Verin
Acute Respiratory Distress Syndrome (ARDS) is a severe lung inflammatory disorder with a 30-50% mortality. Sepsis and pneumonia are the leading causes of ARDS. On the cellular level there is pulmonary capillary endothelial cell permeability and fluid leakage into the pulmonary parenchyma, followed by neutrophils, cytokines and an acute inflammatory response. When fluid increases in the interstitium then the outward movement continues and protein rich fluid floods the alveolar spaces through the tight junctions of the epithelial cells...
June 4, 2015: Austin Journal of Vascular Medicine
X Lin, M Barravecchia, P Kothari, J L Young, D A Dean
Acute lung injury (ALI) and acute respiratory distress syndrome (ARDS) are associated with diverse disorders and characterized by disruption of the alveolar-capillary barrier, leakage of edema fluid into the lung, and substantial inflammation leading to acute respiratory failure. Gene therapy is a potentially powerful approach to treat ALI/ARDS through repair of alveolar epithelial function. Herein, we show that delivery of a plasmid expressing β1-subunit of the Na(+),K(+)-ATPase (β1-Na(+),K(+)-ATPase) alone or in combination with epithelial sodium channel (ENaC) α1-subunit using electroporation not only protected from subsequent lipopolysaccharide (LPS)-mediated lung injury, but also treated injured lungs...
June 2016: Gene Therapy
Ansgar Reising, Carsten Hafer, Marcus Hiss, Jan T Kielstein, Jan Menne, Faikah Gueler, Jan Beneke, Silvia Linnenweber-Held, Hermann Haller, Gunilla Einecke
PURPOSE: Hemolytic-uremic syndrome (HUS) and acute kidney injury (AKI) after infection with Shiga toxin-producing E. coli (EHEC) are clinically important complications. We present a retrospective analysis of abdominal ultrasound findings in patients with HUS caused by EHEC O104:H4 (n = 41). METHODS: We assessed intrarenal resistance indices and quantitated kidney parenchymal density by the kidney/liver intensity ratio using computer-based image analysis. Findings in EHEC-HUS were compared to those in AKI due to other reasons (n = 60) and 19 healthy volunteers...
April 2016: International Urology and Nephrology
Sancar Serbest, Oktay Belhan, Murat Gürger, Haci Bayram Tosun
Every year, especially in the cooler Fall and Winter months, hundreds of people die because of carbon monoxide poisoning. This occurs usually as an accident. It is a significant cause of poisoning worldwide. We present a case of compartment syndrome in both lower extremities with accompanying acute renal failure and systemic capillary leakage syndrome because of carbon monoxide poisoning.
December 1, 2015: A & A Case Reports
Andrew Ertel, Drew Pratt, Peter Kellman, Steve Leung, Patricia Bandettini, Lauren M Long, Michael Young, Celeste Nelson, Andrew E Arai, Kirk M Druey
BACKGROUND: The Systemic Capillary Leak Syndrome (SCLS) is a rare disorder of unknown etiology presenting as recurrent episodes of shock and peripheral edema due to leakage of fluid into soft tissues. Insights into SCLS pathogenesis are few due to the scarcity of cases, and the etiology of vascular barrier disruption in SCLS is unknown. Recent advances in cardiovascular magnetic resonance (CMR) allow for the quantitative assessment of the myocardial extracellular volume (ECV), which can be increased in conditions causing myocardial edema...
2015: Journal of Cardiovascular Magnetic Resonance
Oscar E Simonson, Dimitrios Mougiakakos, Nina Heldring, Giulio Bassi, Henrik J Johansson, Magnus Dalén, Regina Jitschin, Sergey Rodin, Matthias Corbascio, Samir El Andaloussi, Oscar P B Wiklander, Joel Z Nordin, Johan Skog, Charlotte Romain, Tina Koestler, Laila Hellgren-Johansson, Petter Schiller, Per-Olof Joachimsson, Hans Hägglund, Mattias Mattsson, Janne Lehtiö, Omid R Faridani, Rickard Sandberg, Olle Korsgren, Mauro Krampera, Daniel J Weiss, Karl-Henrik Grinnemo, Katarina Le Blanc
UNLABELLED: Mesenchymal stromal cells (MSCs) have been investigated as a treatment for various inflammatory diseases because of their immunomodulatory and reparative properties. However, many basic questions concerning their mechanisms of action after systemic infusion remain unanswered. We performed a detailed analysis of the immunomodulatory properties and proteomic profile of MSCs systemically administered to two patients with severe refractory acute respiratory distress syndrome (ARDS) on a compassionate use basis and attempted to correlate these with in vivo anti-inflammatory actions...
October 2015: Stem Cells Translational Medicine
Albert C Sek, Zhihui Xie, Kaoru Terai, Lauren M Long, Celeste Nelson, Arkadiusz Z Dudek, Kirk M Druey
Idiopathic systemic capillary leak syndrome (SCLS) is a rare and potentially fatal vascular disorder characterized by reversible bouts of hypotension and edema resulting from fluid and solute escape into soft tissues. Although spikes in permeability-inducing factors have been linked to acute SCLS flares, whether or not they act on an inherently dysfunctional endothelium is unknown. To assess the contribution of endothelial-intrinsic mechanisms in SCLS, we derived blood-outgrowth endothelial cells (BOEC) from patients and healthy controls and examined gene expression patterns...
2015: PloS One
Hui-Ching Wang, Ko Chang, Po-Liang Lu, Kun-Bow Tsai, Huang-Chi Chen
INTRODUCTION: Pulmonary infiltration and pleural effusion caused by permeability syndrome are the hallmark of pulmonary manifestation of dengue cases. OBJECTIVE AND METHODS: We report a 95-year-old chronic obstructive pulmonary disease case having dengue shock syndrome. Chest X-ray examination revealed diffuse lung infiltration. However, bilateral pneumotoceles were unexpectedly found in computed tomography (CT) images. Dengue virus type 2 infection was confirmed by virus culture, serology and reverse transcriptase-polymerase chain reaction...
June 15, 2015: Clinical Respiratory Journal
Yuri Hirosaki, Shunji Hayashidani, Sayako Ouchi, Tukasa Ohshima, Ryuji Nakano, Hideo Yamamoto
INTRODUCTION: Idiopathic systemic capillary leak syndrome is a rare and fatal disease due to the unexplained episodic attacks of capillary leakage of plasma from the intravascular into the interstitial space. The attack consists of three phases, a prodromal phase, peripheral leak phase and recruitment phase. During the peripheral leak phase, generalized edema, mainly in the trunk and extremities, with hemoconcentration and hypoalbuminemia occurs, while usually the visceral organs like lungs, brain, heart and kidneys seem not to be involved...
2015: Journal of Medical Case Reports
Mitrakrishnan Rayno Navinan, Jevon Yudhishdran, Sandamalee Herath, Isurujith Liyanage, Tharshana Kugadas, Damith Kumara, Aruna Kulatunga
BACKGROUND: The spread of Dengue virus infection is reaching pandemic proportions. Dengue is usually dreaded for causing shock due to capillary leakage. However the clinical spectrum of dengue is vast and the newly incorporated expanded dengue syndrome introduces a wide range of presentations that are rarely observed and appreciated but nevertheless have the potential to cause significant morbidity and even mortality. Cardiac involvement in dengue is one such example. CASE PRESENTATION: A 26 year old South-Asian female presented in a state of haemodynamic shock with a history of fever and use of non-steroidal anti inflammatory drugs...
2015: BMC Research Notes
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