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[PMID]: 29492600
[Au] Autor:Vander Meeren S; Heyrman B; Renmans W; Bakkus M; Maes B; De Raeve H; Schots R; Jochmans K
[Ad] Address:Department of Clinical Biology, Hematology Division, Vrije Universiteit Brussel (VUB), Universitair Ziekenhuis Brussel (UZ Brussel), Brussels, Belgium. Sam.vandermeeren@uzbrussel.be.
[Ti] Title:Lymphoma-like monoclonal B cell lymphocytosis in a patient population: biology, natural evolution, and differences from CLL-like clones.
[So] Source:Ann Hematol;, 2018 Feb 28.
[Is] ISSN:1432-0584
[Cp] Country of publication:Germany
[La] Language:eng
[Ab] Abstract:High-count monoclonal B cell lymphocytosis (MBL) with a chronic lymphocytic leukemia (CLL) phenotype is a well-known entity, featuring 1-4% annual risk of progression towards CLL requiring treatment. Lymphoma-like MBL (L-MBL), on the other hand, remains poorly defined and data regarding outcome are lacking. We retrospectively evaluated 33 L-MBL cases within our hospital population and compared them to 95 subjects with CLL-like MBL (C-MBL). Diagnoses of L-MBL were based on asymptomatic B cell clones with Matutes score < 3, B cells < 5.0 × 10 /µl, and negative computerized tomography scans. We found that median B cell counts were considerably lower compared to C-MBL (0.6 vs 2.3 × 10 /µl) and remained stable over time. Based on immunophenotyping and immunogenetic profiling, most L-MBL clones did not correspond to known lymphoma entities. A strikingly high occurrence of paraproteinemia (48%), hypogammaglobulinemia (45%), and biclonality (21%) was seen; these incidences being significantly higher than in C-MBL (17, 21, and 5%, respectively). Unrelated monoclonal gammopathy of undetermined significance was a frequent feature, as the light chain type of 5/12 paraproteins detected was different from the clonal surface immunoglobulin. After 46-month median follow-up, 2/24 patients (8%) had progressed towards indolent lymphoma requiring no treatment. In contrast, 41% of C-MBL cases evolved to CLL and 17% required treatment. We conclude that clinical L-MBL is characterized by pronounced immune dysregulation and very slow or absent progression, clearly separating it from its CLL-like counterpart.
[Pt] Publication type:JOURNAL ARTICLE
[Em] Entry month:1803
[Cu] Class update date: 180308
[Lr] Last revision date:180308
[St] Status:Publisher
[do] DOI:10.1007/s00277-018-3282-0

  2 / 16808 MEDLINE  
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[PMID]: 29408410
[Au] Autor:Holroyd AK; Michie AM
[Ad] Address:Institute of Cancer Sciences, College of Medical, Veterinary and Life Sciences, University of Glasgow, Glasgow, UK. Electronic address: Ailsa.Holroyd@glasgow.ac.uk.
[Ti] Title:The role of mTOR-mediated signaling in the regulation of cellular migration.
[So] Source:Immunol Lett;196:74-79, 2018 Feb 03.
[Is] ISSN:1879-0542
[Cp] Country of publication:Netherlands
[La] Language:eng
[Ab] Abstract:Mechanistic target for rapamycin (mTOR) is a serine/threonine protein kinase that forms two distinct complexes mTORC1 and mTORC2, integrating mitogen and nutrient signals to regulate cell survival and proliferation; processes which are commonly deregulated in human cancers. mTORC1 and mTORC2 have divergent molecular associations and cellular functions: mTORC1 regulates in mRNA translation and protein synthesis, while mTORC2 is involved in the regulation of cellular survival and metabolism. Through AKT phosphorylation/activation, mTORC2 has also been reported to regulate cell migration. Recent attention has focused on the aberrant activation of the PI3K/mTOR pathway in B cell malignancies and there is growing evidence for its involvement in disease pathogenesis, due to its location downstream of other established novel drug targets that intercept B cell receptor (BCR) signals. Shared pharmacological features of BCR signal inhibitors include a striking "lymphocyte redistribution" effect whereby patients experience a sharp increase in lymphocyte count on initiation of therapy followed by a steady decline. Chronic lymphocytic leukemia (CLL) serves as a paradigm for migration studies as lymphocytes are among the most widely travelled cells in the body, a product of their role in immunological surveillance. The subversion of normal lymphocyte movement in CLL is being elucidated; this review aims to describe the migration impairment which occurs as part of the wider context of cancer cell migration defects, with a focus on the role of mTOR in mediating migration effects downstream of BCR ligation and other microenvironmental signals.
[Pt] Publication type:JOURNAL ARTICLE; REVIEW
[Em] Entry month:1802
[Cu] Class update date: 180305
[Lr] Last revision date:180305
[St] Status:Publisher

  3 / 16808 MEDLINE  
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[PMID]: 29270844
[Au] Autor:Wynsberghe MV; Lenain P; Drieux F; Guerrot D; Bertrand D
[Ad] Address:Service de Néphrologie, Centre Hospitalier de Rouen, 76000, Rouen, France.
[Ti] Title:Monoclonal B lymphocytosis and minimal change disease: a new monoclonal B-cell disorder of renal significance?
[So] Source:J Nephrol;31(2):317-320, 2018 Apr.
[Is] ISSN:1724-6059
[Cp] Country of publication:Italy
[La] Language:eng
[Ab] Abstract:Chronic lymphocytic leukemia (CLL) may induce renal complications, which are becoming increasingly common, but in this context the occurrence of minimal change disease (MCD) remains rare. Monoclonal B lymphocytosis (MBL) is a precursor state of CLL and is currently under recognized. Since MBL is seen as a benign disorder that rarely evolves into CLL, screening for MBL is not standardized and does not require any treatment. When reviewing renal disease associated with MBL, there is very scant data in the literature and to date there is no case describing the association between MBL and MCD. Here, we describe the case of a 71-year old woman admitted for nephrotic syndrome (NS). We diagnosed a MBL. Kidney biopsy revealed MCD. Treatment with corticosteroids was introduced but no improvement was observed. Chemotherapy with rituximab and chlorambucil was thus started, leading to complete remission of both MBL and MCD. To our knowledge, this is the first description of the association of MBL and MCD. This case suggests that screening for MBL may have unexpected diagnostic and therapeutic implications in patients presenting with seemingly idiopathic NS.
[Pt] Publication type:JOURNAL ARTICLE
[Em] Entry month:1712
[Cu] Class update date: 180305
[Lr] Last revision date:180305
[St] Status:In-Data-Review
[do] DOI:10.1007/s40620-017-0464-0

  4 / 16808 MEDLINE  
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[PMID]: 29462726
[Au] Autor:Torpiano P; Pace D
[Ad] Address:Department of Paediatrics, Mater Dei Hospital, Tal-Qroqq, Msida, MSD 2090, Malta. Electronic address: paul.torpiano@gov.mt.
[Ti] Title:Clinically-diagnosed Mediterranean Spotted Fever in Malta.
[So] Source:Travel Med Infect Dis;, 2018 Feb 17.
[Is] ISSN:1873-0442
[Cp] Country of publication:Netherlands
[La] Language:eng
[Ab] Abstract:BACKGROUND: Mediterranean Spotted Fever (MSF) is a tick-borne zoonosis caused by Rickettsia conorii which is endemic in Malta, an island in the South Mediterranean that is a popular tourist destination. Diagnosis is frequently based on clinical manifestations as laboratory results are often limited to a retrospective diagnosis. We describe the clinical presentation, diagnosis and treatment of children <16 years who presented with MSF from 2011 to 2016. METHOD: The demographics, clinical findings, laboratory results, management and outcome of all children hospitalised with suspected MSF based on the presence of fever and an eschar, were retrieved from their case notes. RESULTS: Over the five-year study period six children, aged between 17 months and 15 years, were diagnosed with MSF. All children had contact with ticks and the majority presented in summer. Laboratory results were non-specific and included elevated inflammatory markers, lymphocytosis/lymphopenia and hyponatraemia. Serological and molecular techniques were used for diagnosis. Response to clarithromycin or doxycycline was immediate. CONCLUSION: MSF should be included in the differential diagnosis of fever, rash and an eschar in children who travel to Malta. Despite advances in molecular diagnostics, clinical diagnosis remains important in the management of children with suspected MSF.
[Pt] Publication type:JOURNAL ARTICLE
[Em] Entry month:1802
[Cu] Class update date: 180303
[Lr] Last revision date:180303
[St] Status:Publisher

  5 / 16808 MEDLINE  
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[PMID]: 29390386
[Au] Autor:Nusshag C; Morath C; Zeier M; Weigand MA; Merle U; Brenner T
[Ad] Address:Department of Nephrology.
[Ti] Title:Hemophagocytic lymphohistiocytosis in an adult kidney transplant recipient successfully treated by plasmapheresis: A case report and review of the literature.
[So] Source:Medicine (Baltimore);96(50):e9283, 2017 Dec.
[Is] ISSN:1536-5964
[Cp] Country of publication:United States
[La] Language:eng
[Ab] Abstract: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. Most therapy protocols are almost exclusively validated in pediatric cohorts and for primary HLH. Their transferability to adult individuals with mostly secondary HLH is doubtful. Especially the high liver and bone marrow toxicity of applied etoposide-based protocols is discussed controversially and connected to overwhelming infections and death. PATIENT CONCERN: A 51-year old, male, kidney transplant recipient was admitted to our center suffering from diarrhea, fever, nausea, hyponatremia, kidney graft failure, disorientation, progressive hemodynamic instability, and multiorgan failure. DIAGNOSES: Clinical and laboratory findings resembled those of a septic shock. Ferritin and soluble interleukin-2 receptor (sCD25) levels were disproportionally elevated. Only a mild hepatosplenomegaly was diagnosed in a CT scan. A T2-weighted, fluid-attenuated inversion recovery MRI showed marked, bilateral and periventricular white matter hyperintensities. The cerebrospinal fluid (CSF) analysis showed a moderately elevated protein content and cell count. There was no evidence of any bacterial, viral, or parasitic infection. The diagnosis of HLH was made. INTERVENTIONS & OUTCOMES: The patient was successfully treated by a combined approach consisting of plasma exchange (PE), corticosteroids, anakinra, and cyclosporine (CsA). LESSONS: HLH is an important differential diagnosis in critically ill patients. Its unspecific clinical picture complicates an early diagnosis and may be misclassified as sepsis. A combination of plasma exchange (PE), corticosteroids, anakinra, and cyclosporine (CsA) may be a promising and less toxic approach for HLH therapy in adults.
[Mh] MeSH terms primary: Kidney Transplantation
Lymphohistiocytosis, Hemophagocytic/therapy
Plasmapheresis
[Mh] MeSH terms secundary: Adrenal Cortex Hormones/therapeutic use
Antirheumatic Agents/therapeutic use
Combined Modality Therapy
Cyclosporine/therapeutic use
Diagnosis, Differential
Diagnostic Imaging
Humans
Immunosuppressive Agents/therapeutic use
Interleukin 1 Receptor Antagonist Protein/therapeutic use
Lymphohistiocytosis, Hemophagocytic/etiology
Male
Middle Aged
[Pt] Publication type:CASE REPORTS; JOURNAL ARTICLE; REVIEW
[Nm] Name of substance:0 (Adrenal Cortex Hormones); 0 (Antirheumatic Agents); 0 (Immunosuppressive Agents); 0 (Interleukin 1 Receptor Antagonist Protein); 83HN0GTJ6D (Cyclosporine)
[Em] Entry month:1802
[Cu] Class update date: 180301
[Lr] Last revision date:180301
[Js] Journal subset:AIM; IM
[Da] Date of entry for processing:180203
[St] Status:MEDLINE
[do] DOI:10.1097/MD.0000000000009283

  6 / 16808 MEDLINE  
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[PMID]: 29390336
[Au] Autor:Lovisari F; Terzi V; Lippi MG; Brioschi PR; Fumagalli R
[Ad] Address:Department of Anesthesia and Critical Care, ASST Grande Ospedale Metropolitano Niguarda. P.zza Ospedale Maggiore, Milan.
[Ti] Title:Hemophagocytic lymphohistiocytosis complicated by multiorgan failure: A case report.
[So] Source:Medicine (Baltimore);96(50):e9198, 2017 Dec.
[Is] ISSN:1536-5964
[Cp] Country of publication:United States
[La] Language:eng
[Ab] Abstract: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. PATIENT CONCERNS: A 32-year-old woman from the Philippines first diagnosed with upper airway infection, was subsequently hospitalized in infectious disease department and treated for community acquired pneumonia. DIAGNOSES: After clinical picture worsened with a profound respiratory insufficiency, the patient was intubated and transferred to our ICU. During this hospitalization, the clinical picture of fever, cutaneous rashes, lymphadenitis, hepatitis, leukopenia, anemia, hyperferritinemia, hypertriglyceridemia, high level of auto-antibodies, and low NK activity suggested an hemophagocytic lymphohistiocytosis syndrome, even if bone marrow biopsy was negative for hemophagocytosis. INTERVENTIONS: Immunosuppressive therapy with dexamethasone and etoposide was started, and the patient was discharged from ICU 4 months after admission. LESSONS: HLH is a rare disorder of the mononuclear phagocytic system, characterized by systemic proliferation of non- neoplastic histiocytes. The diagnosis is often challenging and not all of the diagnostic criteria may be present at the same time; this case shows how complex the diagnosis could be, how hematic ferritin levels could help in following the course of the disease, and the possibility of severe pulmonary complication either due to the disease itself and to possible sovra infections.
[Mh] MeSH terms primary: Lymphohistiocytosis, Hemophagocytic/complications
Lymphohistiocytosis, Hemophagocytic/diagnosis
Multiple Organ Failure/diagnosis
Multiple Organ Failure/etiology
[Mh] MeSH terms secundary: Adult
Dexamethasone/therapeutic use
Diagnosis, Differential
Drug Therapy, Combination
Etoposide/therapeutic use
Female
Glucocorticoids/therapeutic use
Humans
Lymphohistiocytosis, Hemophagocytic/drug therapy
Multiple Organ Failure/drug therapy
Topoisomerase II Inhibitors/therapeutic use
[Pt] Publication type:CASE REPORTS; JOURNAL ARTICLE
[Nm] Name of substance:0 (Glucocorticoids); 0 (Topoisomerase II Inhibitors); 6PLQ3CP4P3 (Etoposide); 7S5I7G3JQL (Dexamethasone)
[Em] Entry month:1802
[Cu] Class update date: 180301
[Lr] Last revision date:180301
[Js] Journal subset:AIM; IM
[Da] Date of entry for processing:180203
[St] Status:MEDLINE
[do] DOI:10.1097/MD.0000000000009198

  7 / 16808 MEDLINE  
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[PMID]: 29488237
[Au] Autor:Norgan AP; Juskewitch JE; Pritt BS; Winters JL
[Ad] Address:Department of Laboratory Medicine and Pathology, Mayo Clinic, Rochester, Minnesota.
[Ti] Title:The use of cytapheresis in the treatment of infectious diseases.
[So] Source:J Clin Apher;, 2018 Feb 28.
[Is] ISSN:1098-1101
[Cp] Country of publication:United States
[La] Language:eng
[Ab] Abstract:Cytapheresis (removal of cellular blood components) has been employed for treatment of infectious diseases since the 1960s. Techniques have included thrombocytapheresis (buffy coat apheresis) for loiasis, erythrocytapheresis for malaria and babesiosis, and leukocytapheresis for pertussis-associated lymphocytosis. Published data on these applications is largely limited to case level data and small observational studies; as such, recommendations for or against the use of cytapheresis in the treatment of infections have been extrapolated from these limited (and at times flawed) data sets. Consequently, utilization of cytapheresis in many instances is not uniform between institutions, and typically occurs at the discretion of treating medical teams. This review revisits the existing literature on the use of cytapheresis in the treatment of four infections (loasis, malaria, babesiosis, and pertussis) and examines the rationale underlying current treatment recommendations concerning its use.
[Pt] Publication type:JOURNAL ARTICLE; REVIEW
[Em] Entry month:1803
[Cu] Class update date: 180228
[Lr] Last revision date:180228
[St] Status:Publisher
[do] DOI:10.1002/jca.21620

  8 / 16808 MEDLINE  
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[PMID]: 29485673
[Au] Autor:Säflund M; Sjöstrand C; Sveinsson O
[Ad] Address:Karolinska Universitetssjukhuset - Department of neurology Stockholm, Sweden Karolinska Universitetssjukhuset - Department of neurology Stockholm, Sweden.
[Ti] Title:HaNDL ­ viktig men gåtfull differentialdiagnos med svår migränliknande huvudvärk - Förknippat med neurologiska symtom och lymfocytär pleocytos. [

The syndrome of transient headache and neurologic deficits with cerebrospinal fluid lymphocytosis (HaNDL)

].
[So] Source:Lakartidningen;115, 2018 Feb 26.
[Is] ISSN:1652-7518
[Cp] Country of publication:Sweden
[La] Language:swe
[Ab] Abstract:The syndrome of transient headache and neurologic deficits with cerebrospinal fluid lymphocytosis (HaNDL) is a self-limited disorder with an unknown pathogenesis, characterized by one or more transient episodes of severe headache accompanied with neurologic deficits, and lymphocytic pleocytosis in the cerebrospinal fluid (CSF). Despite being uncommon and benign it is important for clinicians to identify and differentiate HaNDL from other potentially fatal neurologic disorders. We present six HaNDL patients from our institution. All had a relatively typical course with repeated migraine-like headaches accompanied by various transient neurologic deficits and a mean CSF lymphocytic pleocytosis of 178 cells/mm3. Most of the patients were disorientated during the attacks, which has been described previously. When neurofilament light in CSF was measured, there was a substantial increase of this marker which normalized after several months, suggesting certain nerve damage.
[Pt] Publication type:ENGLISH ABSTRACT; JOURNAL ARTICLE
[Em] Entry month:1802
[Cu] Class update date: 180227
[Lr] Last revision date:180227
[St] Status:In-Data-Review

  9 / 16808 MEDLINE  
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[PMID]: 29479965
[Au] Autor:Tharwat Abou El-Khier N; Darwish A; El Sayed Zaki M
[Ad] Address:Department of Medical Microbiology and Immunology, Faculty of Medicine, Mansoura University, Egypt. Email: may_s65@hotmail.com
[Ti] Title:Molecular Study of Parvovirus B19 Infection in Children with Acute Myeloid Leukemia
[So] Source:Asian Pac J Cancer Prev;19(2):337-342, 2018 Feb 26.
[Is] ISSN:2476-762X
[Cp] Country of publication:Thailand
[La] Language:eng
[Ab] Abstract:Background: Parvovirus B19 is a common viral infection in children. Nearby evidences are present about its association with acute leukemia, especially acute lymphoblast leukemia. Nevertheless, scanty reports have discussed any role in acute myeloid leukemia (AML). Purpose: To evaluate the frequency of virological markers of B19 infection including its DNA along with specific immunoglobulins G (IgG) and M (IgM) among children with newly diagnosed AML. Besides, describing the clinical importance of Parvovirus B19 infection in those patients. Patients and methods: A case-control retrospective study was conducted on 48 children recently diagnosed with AML before and during chemotherapy induction and 60 healthy control. Specific serum IgM and IgG levels were determined by enzyme linked immunosorbant assay (ELISA) and DNA detection by polymerase chain reaction (PCR). Results: Parvovirus DNA was detected in 20 patients with AML. IgM was found in sera of four patients and one case had positive DNA and IgG (5%). Patients with recent parvovirus B19 infection had a significantly reduced hemoglobin levels, RBCs counts, platelet counts, neutrophil counts and absolute lymphocytosis (p=0.01, p=0.0001, p=0.01, p=0.02, p=0.0003, respectively). There were no clinical findings with statistically significant association to recent infection. Half of the patients with AML had positive PCR and/or IgM for parvovirus B19. Among children with AML under chemotherapy, there were reduced hemoglobin levels (P=0.03), reduced platelet counts (P=0.0001) and absolute neutropenia (mean±SD, 1.200 ±1.00) in those with parvovirus B19 infection. More than half of patients with parvovirus B19 (72.2%) had positive PCR and/or IgM and 36.4% of them had positive IgG. Conclusion: This study highlights that parvovirus B19 is common in children with AML either at diagnosis or under chemotherapy. There are no clinical manifestations that can be used as markers for its presence, but hematological laboratory findings can provide evidence for infection in the presence of anemia and neutropenia. Detection of parvovirus B19 by combined molecular and serological markers is required in such patients for accurate diagnosis.
[Pt] Publication type:JOURNAL ARTICLE
[Em] Entry month:1802
[Cu] Class update date: 180226
[Lr] Last revision date:180226
[St] Status:In-Process

  10 / 16808 MEDLINE  
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[PMID]: 29197090
[Au] Autor:Moavero R; Papetti L; Tarantino S; Battan B; Salfa I; Deodati A; Schiaffini R; Vigevano F; Valeriani M
[Ad] Address:Child Neurology Unit, Neuroscience and Neurorehabilitation Department, Bambino Gesù Children's Hospital, Headache Center, IRCCS, Rome, Italy.
[Ti] Title:Syndrome of Transient Headache and Neurologic Deficits With Cerebrospinal Fluid Lymphocitosis Should Be Considered in Children Presenting With Acute Confusional State.
[So] Source:Headache;58(3):438-442, 2018 Mar.
[Is] ISSN:1526-4610
[Cp] Country of publication:United States
[La] Language:eng
[Ab] Abstract:BACKGROUND: Transient headache and neurologic deficits with cerebrospinal fluid lymphocytosis (HaNDL) is benign and self-limited, with neurologic deficits including sensory disturbance of one body side, aphasia, nausea/vomiting, weakness, decreased vision, homonymous hemianopsia, photophobia. Acute confusional state can rarely occur. Papilledema and intracranial hypertension have also been described. It is a rare entity mainly affecting adults; however, it has been sporadically described in children and adolescents. MAIN FINDINGS: In this clinical observational study, we describe a clinical series of three consecutive pediatric patients being diagnosed with HaNDL after presenting with altered consciousness, papilledema, and increased intracranial pressure. They all recovered without relapses. CONCLUSION: Presentation during childhood and adolescence is rare; the majority of pediatric cases presented with altered consciousness, which is infrequent in HaNDL. This may suggest that in childhood this symptom might be more common than in adults. All three patients presented with increased intracranial pressure and papilledema, thus suggesting that these aspects should be investigated in all patients presenting with this clinical pattern. Finally, all our patients began to suffer from migraine. This feature, together with the benign course of the disease, could favor the hypothesis of a migrainous pathophysiology of this syndrome, although this remains a speculative.
[Pt] Publication type:JOURNAL ARTICLE
[Em] Entry month:1712
[Cu] Class update date: 180226
[Lr] Last revision date:180226
[St] Status:In-Data-Review
[do] DOI:10.1111/head.13238


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