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https://www.readbyqxmd.com/read/29440240/atypical-presentation-of-atypical-haemolytic-uraemic-syndrome
#1
Ratna Basak, Xiaotong Wang, Caitlin Keane, Robert Woroniecki
A 17-year-old girl presented with fever, myalgia, vomiting for 1 month and oliguria and dyspnoea for 4 days. She was tachycardic,hypertensive, with pedal oedema and decreased breath sounds. She had high serum creatinine (3 mg/dL), anaemia, thrombocytopenia, leucocytosis and eosinophilia with schistocytes. Lactate dehydrogenase, transaminases were high , with low haptoglobin and high ferritin (5269 ng/mL). Complement C3/C4 and fibrinogen were normal. Urinalysis showed large blood and protein and stool studies were negative...
February 11, 2018: BMJ Case Reports
https://www.readbyqxmd.com/read/29434707/leuconostoc-pseudomesenteroides-associated-hemophagocytic-syndrome-a-case-report
#2
Xinfeng Lin, Qilong Jiang, Jiduo Liu, Fu Zhao, Weitao Chen
Hemophagocytic lymphohistiocytosis (HLH) is a rare hyperinflammatory syndrome characterized by fever, pancytopenia and splenomegaly. The underlying hemophagocytosis occurs primarily in the bone marrow, liver and lymph nodes. Multiple microbiological agents, including cytomegalovirus, Epstein-Barr virus and Mycobacterium tuberculosis, have been implicated in the pathogenesis of HLH. The present study presents a case of HLH associated with Leuconostoc pseudomesenteroides infection treated successfully with clindamycin...
February 2018: Experimental and Therapeutic Medicine
https://www.readbyqxmd.com/read/29433397/hemophagocytic-lymphohistiocytosis-in-an-hiv-positive-patient-with-concomitant-disseminated-histoplasmosis
#3
Nisha Loganantharaj, Brianna Oliver, Taylor Smith, Rachna Jetly, Lee Engel, Shane Sanne
A 46-year-old Dominican man, known to have HIV, presented with constitutional symptoms of two week's duration. The patient was found to have cytopenias, significantly elevated ferritin level and lymphadenopathy. Biopsies and laboratory studies met the criteria for hemophagocytic lymphohistiocytosis (HLH). A concomitant diagnosis of histoplasmosis was confirmed as the trigger for HLH and treatment resulted in clinical improvement and resolution of symptoms.
January 1, 2018: International Journal of STD & AIDS
https://www.readbyqxmd.com/read/29432306/parvovirus-b19-associated-hemophagocytic-lymphohistiocytosis-in-a-patient-with-glucose-6-phosphate-dehydrogenase-deficiency
#4
Aida Zečkanović, Matej Perovnik, Janez Jazbec, Miroslav Petrovec, Marko Pokorn, Marko Kavčič
We report a case of a 12-year-old male with glucose-6-phosphate dehydrogenase deficiency presenting with clinical signs of sepsis and pancytopenia. Investigations revealed parvovirus B19 (PVB19)-associated hemophagocytic lymphohistiocytosis (HLH). The patient recovered fully and quickly with symptomatic treatment. Current evidence suggests that PVB19-associated HLH has a favorable prognosis. Mild undiagnosed cases of HLH may be the cause of pancytopenia in PVB19 infections.
February 9, 2018: Journal of Pediatric Hematology/oncology
https://www.readbyqxmd.com/read/29431300/epstein-barr-virus-associated-hemophagocytic-lymphohistiocytosis-in-adults-a-retrospective-analysis-of-23-patients-in-china
#5
Xiaoyan Shao, Ye Xu, Xihui Xu, Yong Xu, Hu Chen, Ming Hong, Lingxiang Liu
BACKGROUND: Epstein-Barr virus (EBV)-associated hemophagocytic lymphohistiocytosis (HLH) is a rare, life-threatening disease with poor prognosis despite intensive therapy. OBJECTIVES: To discuss the ideal therapy of EBV-associated HLH for adults. METHODS: We retrospectively studied 23 adult patients with EBV-associated HLH at our institution between January 2000 and June 2015. The clinical characteristics, treatment, and prognosis of adult EBV-associated HLH were analyzed...
February 2018: Israel Medical Association Journal: IMAJ
https://www.readbyqxmd.com/read/29420734/convergent-pathways-of-the-hyperferritinemic-syndromes
#6
Grant S Schulert, Scott W Canna
Hyperferritinemia and pronounced hemophagocytosis help distinguish a subset of patients with a particularly inflammatory and deadly systemic inflammatory response syndrome. Two clinically similar disorders typify these hyperferritinemic syndromes: hemophagocytic lymphohistiocytosis (HLH) and macrophage activation syndrome (MAS). HLH is canonically associated with a complete disturbance of perforin/granzyme-mediated cytotoxicity, whereas MAS occurs in the context of the related rheumatic diseases systemic juvenile idiopathic arthritis (SJIA) and adult-onset Still's disease (AOSD), with associated IL-1 family cytokine activation...
February 6, 2018: International Immunology
https://www.readbyqxmd.com/read/29411124/malignancy-and-chemotherapy-induced-haemophagocytic-lymphohistiocytosis-in-children-and-adolescents-a-single-centre-experience-of-20%C3%A2-years
#7
Volker Strenger, Gerald Merth, Herwig Lackner, Stephan W Aberle, Harald H Kessler, Markus G Seidel, Wolfgang Schwinger, Daniela Sperl, Petra Sovinz, Anna Karastaneva, Martin Benesch, Christian Urban
Haemophagocytic lymphohistiocytosis (HLH) is a possibly life-threatening syndrome of immune dysregulation and can be divided into primary (hereditary) and secondary forms (including malignancy-associated HLH (M-HLH)). We retrospectively analysed epidemiological, clinical, virological and laboratory data from patients with M-HLH treated at our department between 1995 and 2014. Out of 1.706 haemato-/oncologic patients treated at our department between 1995 and 2014, we identified 22 (1.29%) patients with secondary HLH (1...
February 6, 2018: Annals of Hematology
https://www.readbyqxmd.com/read/29410181/outcomes-of-children-with-hemophagocytic-lymphohistiocytosis-given-allogeneic-hematopoietic-stem-cell-transplantation-in-italy
#8
Chiara Messina, Marco Zecca, Franca Fagioli, Attilio Rovelli, Stefano Giardino, Pietro Merli, Fulvio Porta, Maurizio Aricò, Elena Sieni, Giuseppe Basso, Mimmo Ripaldi, Claudio Favre, Marta Pillon, Antonio Marzollo, Marco Rabusin, Simone Cesaro, Mattia Algeri, Maurizio Caniglia, Paolo Di Bartolomeo, Ottavio Ziino, Francesco Saglio, Arcangelo Prete, Franco Locatelli
We report on 109 patients with hemophagocytic lymphohistiocytosis (HLH) undergoing 126 procedures of allogeneic hematopoietic stem cell transplantation (HSCT) between 2000 and 2014 in centers associated with the Italian Pediatric Hematology Oncology Association. Genetic diagnosis was FHL2 (32%), FHL3 (33%) or other defined disorders known to cause HLH (15%); in the remaining patients, no genetic abnormality was found. Donor for first transplant was an HLA-matched sibling for 25 patients (23%), an unrelated donor for 73 (67%) and an HLA-partially matched family donor for 11 children (10%)...
February 1, 2018: Biology of Blood and Marrow Transplantation
https://www.readbyqxmd.com/read/29409136/novel-unc13d-intronic-variant-disrupting-a-nf%C3%AE%C2%BAb-enhancer-in-a-patient-with-recurrent-macrophage-activation-syndrome-and-systemic-juvenile-idiopathic-arthritis
#9
Grant S Schulert, Mingce Zhang, Ammar Husami, Ndate Fall, Hermine Brunner, Kejian Zhang, Randy Q Cron, Alexei A Grom
OBJECTIVE: Macrophage activation syndrome (MAS) is a life-threatening complication of systemic juvenile idiopathic arthritis (SJIA), and has pathologic similarity to hemophagocytic lymphohistiocytosis (HLH). Intronic variants in UNC13D are found in patients with familial HLH-3 (FHL3), but the role of non-coding variants in MAS is unknown. The objective of this study was to identify deep intronic UNC13D variants in patients with MAS. METHODS: A custom enrichment library was constructed to sequence approximately 1 MB flanking UNC13D in 24 patients with SJIA, recurrent MAS, and negative prior genetic (exon/coding) testing...
February 6, 2018: Arthritis & Rheumatology
https://www.readbyqxmd.com/read/29408742/role-of-lh-polymorphisms-and-r-hlh-supplementation-in-gnrh-agonist-treated-art-cycles-a-cross-sectional-study
#10
Ramaraju G A, Ravikrishna Cheemakurthi, Kavitha Prathigudupu, Kavitha Lakshmi Balabomma, Madan Kalagara, Sivanarayana Thota, Muralikrishna Kota
STUDY OBJECTIVES: To investigate the effect of N312S polymorphism in the LHCGR gene as a predictive pharmacogenetic marker on clinical and embryological parameters and determining the need of r-hLH supplementation combine with r-hFSH in patients undergoing ART treatment. STUDY DESIGN: In a cross-sectional study, a retrospective analysis of women (n = 553), who underwent controlled ovarian stimulation treatment protocol was conducted during the years 2012-2014...
February 2, 2018: European Journal of Obstetrics, Gynecology, and Reproductive Biology
https://www.readbyqxmd.com/read/29405821/modulatory-upregulation-of-an-insulin-peptide-gene-by-different-pathogens-in-c-%C3%A2-elegans
#11
Song-Hua Lee, Shizue Omi, Nishant Thakur, Clara Taffoni, Jérôme Belougne, Ilka Engelmann, Jonathan J Ewbank, Nathalie Pujol
When an animal is infected, its innate immune response needs to be tightly regulated across tissues and coordinated with other aspects of organismal physiology. Previous studies with Caenorhabditis elegans have demonstrated that insulin-like peptide genes are differentially expressed in response to different pathogens. They represent prime candidates for conveying signals between tissues upon infection. Here, we focused on one such gene, ins-11 and its potential role in mediating cross-tissue regulation of innate immune genes...
February 6, 2018: Virulence
https://www.readbyqxmd.com/read/29402541/synthesis-and-characterization-of-ciprofloxacin-containing-divinyl-oligomers-and-assessment-of-their-biodegradation-in-simulated-salivary-esterase
#12
Yasaman Delaviz, Mitchell A Nascimento, Michael W Laschuk, Timothy W Liu, Meilin Yang, J Paul Santerre
OBJECTIVE: Two leading causes contributing to dental restoration replacement are the marginal breakdown at the composite/dentin interface and secondary caries mediated by bacteria. The objective of the present study was to synthesize oligomers which incorporated enhanced bio-stability but would also be able to generate antimicrobial function if they underwent degradation. METHODS: Stability was incorporated into the oligomers by generating structural features that would physically hinder the availability of hydrolytically sensitive groups in the oligomers...
February 2, 2018: Dental Materials: Official Publication of the Academy of Dental Materials
https://www.readbyqxmd.com/read/29390580/successful-management-of-a-complicated-clinical-crisis-a-patient-with-left-sided-endocarditis-and-secondary-hemophagocytic-lymphohistiocytosis-a-rare-case-report-and-literature-review
#13
Peipei Xu, Hui Zeng, Min Zhou, Jian Ouyang, Bing Chen, Qiguo Zhang
RATIONALE: Hemophagocytic lymphohistiocytosis (HLH) secondary to methicillin-resistant Staphylococcus epidermidis (MRSE)-related left-sided infectious endocarditis had never been reported before. In the last decade, daptomycin, a novel lipopeptide antibiotic, showed its excellent role in anti-Gram-positive bacteria, including soft tissue infection, bloodstream and deep tissueinfection. PATIENT CONCERNS: An Asian women under sever condition due to the cooccurrence of HLH and MRSE-related endocarditis while also be allergic to vancomycin...
December 2017: Medicine (Baltimore)
https://www.readbyqxmd.com/read/29390386/hemophagocytic-lymphohistiocytosis-in-an-adult-kidney-transplant-recipient-successfully-treated-by-plasmapheresis-a-case-report-and-review-of-the-literature
#14
Christian Nusshag, Christian Morath, Martin Zeier, Markus A Weigand, Uta Merle, Thorsten Brenner
RATIONALE: Hemophagocytic lymphohistiocytosis (HLH) is a life-threatening disease entity primarily described in children, but not less relevant in adults. It is characterized by a misdirected activation of the immune system, resulting in an uncontrolled cytokine release from macrophages and cytotoxic T-cells (CTLs). Primary HLH relies on a genetic predisposition, whereas secondary HLH develops in the context of infections, malignancies or autoimmune diseases. However, the awareness and therapeutic knowledge for HLH in adulthood is limited...
December 2017: Medicine (Baltimore)
https://www.readbyqxmd.com/read/29390336/hemophagocytic-lymphohistiocytosis-complicated-by-multiorgan-failure-a-case-report
#15
Federica Lovisari, Valeria Terzi, Monica G Lippi, Paolo R Brioschi, Roberto Fumagalli
RATIONALE: We present a case of hemophagocytic lymphohistiocytosis (HLH) with severe pulmonary complication and acute respiratory distress syndrome (ARDS) hospitalized in our intensive care unit (ICU) in 2014; distinctive trait of this case has been the challenging diagnosis, with a bone marrow biopsy always negative, the severe pulmonary complication with ARDS and severe pulmonary hypertension, and the ferritin temporal kinetics that precisely followed the clinical course of disease...
December 2017: Medicine (Baltimore)
https://www.readbyqxmd.com/read/29384877/acute-appendicitis-complicated-with-necrotizing-fasciitis-in-a-patient-with-adult-onset-still-s-disease-a-case-report
#16
Zheng-Hao Huang, Yu-Chen Chiu, Li-Lu Ho, Hsiu-Lung Fan, Chun-Chi Lu
RATIONALE: Adult-onset Still disease (AOSD) is a rare systemic inflammatory disease of unknown etiology characterized by evanescent salmon-pink rash, spiking fever, arthralgia/ arthritis, and lymphadenopathy. AOSD sometimes was fatal when it is complicated by macrophage activation syndrome (MAS) or hemophagocytic lymphohistiocytosis (HLH). Nonetheless, the literature provides no recommendations for treatment of AOSD patients with severe sepsis. PATIENT CONCERNS: A previously healthy 65-year-old man with history of AOSD was referred to our hospital for persistent right lower quadrant abdominal pain for 2 days...
February 2018: Medicine (Baltimore)
https://www.readbyqxmd.com/read/29381934/pregnancy-associated-hemophagocytic-lymphohistiocytosis-secondary-to-nk-t-cells-lymphoma-a-case-report-and-literature-review
#17
Mengzhou He, Jing Jia, Jingyi Zhang, Rajluxmee Beejadhursing, Lali Mwamaka Sharifu, Jun Yu, Shaoshuai Wang, Ling Feng
RATIONALE: Hemophagocytic lymphohistiocytosis (HLH) occurs primarily in pediatric population, or secondary to malignancy, infection, or autoimmune disease. This disease is rare and prognosis is generally poor. Only a small number of cases during pregnancy have been reported in literature. PATIENT CONCERNS: We report a case of pregnancy-associated HLH secondary to natural killer (NK)/T cells lymphoma. She was admitted at 30 weeks and 3 days of pregnancy with complaints of abdominal pain and fever as high as 39...
November 2017: Medicine (Baltimore)
https://www.readbyqxmd.com/read/29378820/genetic-loci-controlling-carotenoid-biosynthesis-in-diverse-tropical-maize-lines
#18
Girum Azmach, Abebe Menkir, Charles Spillane, Melaku Gedil
The discovery and use of genetic markers associated with carotenoid levels can help to more effectively exploit the genetic potential of maize for provitamin A accumulation. Provitamin A carotenoids are classes of carotenoids that are precursors of vitamin A, an essential micronutrient in humans. Vitamin A deficiency is a global public health problem affecting millions of people, especially in developing countries. Maize is one of the most important staple crops targeted for provitamin A biofortification to help alleviate vitamin A deficiency in developing countries...
January 29, 2018: G3: Genes—Genomes—Genetics
https://www.readbyqxmd.com/read/29371779/hyperferritinemia-in-hemophagocytic-lymphohistiocytosis-a-single-institution-experience-in-pediatric-patients
#19
Surupa Basu, Biplab Maji, Santanu Barman, Apurba Ghosh
Hemophagocytic lymphohistiocytosis (HLH) is an inflammatory condition that may run a rapid fatal course and calls for prompt diagnosis. Early intervention with steroids and other immunosuppressive drugs can contain the disease process and favours positive outcome. Ferritin ≥500 ng/ml is a HLH diagnostic criterion. We evaluated the diagnostic potential of admission ferritin, in children with HLH. Pediatric patients of a referral teaching hospital from Feb 2010-Oct 2013 having been investigated for ferritin on admission were included...
January 2018: Indian Journal of Clinical Biochemistry: IJCB
https://www.readbyqxmd.com/read/29371734/haemophagocytic-lymphohistiocytosis-in-a-preterm-infant-a-case-report
#20
Na Mi Lee, Dae Yong Yi, Shin Weon Yoon, Soo Ahn Chae, In Seok Lim, Yong Sung Choi
Haemophagocytic lymphohistiocytosis (HLH) is a rare disease with a sepsis-like progression that leads to multiple organ dysfunction syndrome, especially in preterm infants. We present herein a case of HLH in a premature infant presenting with disseminated intravascular coagulopathy (DIC) and liver failure. A male infant, with weight 810g and delivered at the gestational age of 25 weeks and 2 days, was misdiagnosed with tyrosinaemia for several weeks. He presented with anaemia, thrombocytopaenia, persistent DIC, and elevated liver enzymes despite continuous transfusion and broad-spectrum antibiotics...
January 2018: JPMA. the Journal of the Pakistan Medical Association
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