Database : MEDLINE
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[PMID]: 25114814
[Au] Autor:Khoulani D; Rao B; Khanshour A; Kuriakose P; Yessayan L
[Ad] Address:Department of Internal Medicine, Henry Ford Hospital, 2799 W. Grand Boulevard, Detroit, MI 48202, USA....
[Ti] Title:Failure of Recombinant Activated Factor VII in Treatment of Diffuse Alveolar Hemorrhage due to Cryoglobulinemic Vasculitis.
[So] Source:Case Rep Hematol;2014:283086, 2014.
[Is] ISSN:2090-6560
[Cp] Country of publication:United States
[La] Language:eng
[Ab] Abstract:Diffuse alveolar hemorrhage (DAH) is a serious complication of the small vessel vasculitis syndromes and carries a high mortality. Recombinant activated factor VII (rFVIIa) is used to treat bleeding in patients with hemophilia and antibodies to factor VIII or IX. It is increasingly being used in life-threatening hemorrhage in a variety of other settings in which conventional therapy is unsuccessful. Randomized controlled trials of rFVIIa in DAH are lacking. However, several case reports have described a complete or sustained control of DAH using rFVIIa after patients failed to respond to medical treatment. There are no case reports in the literature describing the use or the failure of rFVIIa in DAH associated with cryoglobulinemic vasculitis. We here report the failure of rFVIIa to control DAH in a patient with CD5+ B-cell non-Hodgkin's lymphoma and cryoglobulinemic vasculitis.
[Pt] Publication type:JOURNAL ARTICLE
[Em] Entry month:1408
[Cu] Class update date: 140815
[Lr] Last revision date:140815
[Da] Date of entry for processing:140812
[St] Status:PubMed-not-MEDLINE
[do] DOI:10.1155/2014/283086

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[PMID]: 24751453
[Au] Autor:Sweiss NJ; Noth I; Mirsaeidi M; Zhang W; Naureckas ET; Hogarth DK; Strek M; Caligiuri P; Machado RF; Niewold TB; Garcia JG; Pangan AL; Baughman RP
[Ad] Address:University of Illinois Hospital and Health Sciences System, and Institute for Personalized Respiratory Medicine. nsweiss@uic.edu.
[Ti] Title:Efficacy Results of a 52-week Trial of Adalimumab in the Treatment of Refractory Sarcoidosis.
[So] Source:Sarcoidosis Vasc Diffuse Lung Dis;31(1):46-54, 2014.
[Is] ISSN:1124-0490
[Cp] Country of publication:Italy
[La] Language:eng
[Ab] Abstract:BACKGROUND: Infliximab, a chimeric, monoclonal, anti-TNF antibody has been shown to be safe and efficacious for refractory sarcoidosis, we investigated whether adalimumab, a fully human, anti-TNF monoclonal antibody, is similarly safe and efficacious in refractory pulmonary sarcoidosis. METHODS: An open-label, single-center study was conducted in 11 patients with refractory pulmonary sarcoidosis. Patients received adalimumab 40 mg weekly for 45 weeks, with a final follow-up at Week 52. The primary endpoint was the percent change in predicted forced vital capacity (FVC) at 24 weeks. Secondary efficacy parameters included the 6-minute walk test (6MWT), Borg dyspnea score, and Physician's (PGA) and Patient's (PaGA) Global Assessments. A successful outcome of the study was defined as reduction in immunosuppressive therapy (prednisone to 10 mg or less), improvement in FVC of 5% or greater, improvement in 6-minute walk test distance (6MWD) of 50 meter or greater at the end of weeks 24 and 52. RESULTS: Eleven patients received adalimumab and had 24-week follow-ups. Only ten patients had a Week 52 evaluation. FVC stabilized in seven patients, and four patients showed improvement in FVC. Five patients had improved 6MWD, and nine had lower Borg dyspnea scores. PGA and PaGA improved at weeks 24 and 52 for all patients (P<0.008 for all comparisons). Among 11 patients who underwent adalimumab treatment, 9 (82%) and 8(80%) had a successful outcome at the end of 24 and 52 weeks respectively. No severe adverse incidents were reported. CONCLUSIONS: In this small, open-label study, adalimumab improved refractory pulmonary sarcoidosis and was well tolerated (ClinicalTrials.gov identifier NCT00311246).
[Pt] Publication type:JOURNAL ARTICLE
[Em] Entry month:1404
[Js] Journal subset:IM
[St] Status:In-Data-Review

  3 / 32102 MEDLINE  
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[PMID]: 24831429
[Au] Autor:Birnbaum AD; Fawzi AA; Rademaker A; Goldstein DA
[Ad] Address:Department of Ophthalmology, Northwestern University Feinberg School of Medicine, Chicago, Illinois....
[Ti] Title:Correlation between clinical signs and optical coherence tomography with enhanced depth imaging findings in patients with birdshot chorioretinopathy.
[So] Source:JAMA Ophthalmol;132(8):929-35, 2014 Aug 1.
[Is] ISSN:2168-6173
[Cp] Country of publication:United States
[La] Language:eng
[Ab] Abstract:IMPORTANCE: Birdshot chorioretinopathy (BCR) is a bilateral posterior uveitis that typically requires aggressive therapy to prevent loss of vision. Clinical signs of disease activity may be subtle and visual acuity is often preserved despite significant loss of visual function. Optical coherence tomography with enhanced depth imaging (OCT-EDI), a new technology that allows visualization of structures posterior to the retinal pigment epithelium, may be a useful tool to monitor disease activity in these patients. OBJECTIVE: To determine the correlation between symptoms and signs of disease activity in BCR and specific findings on OCT-EDI. DESIGN, SETTING, AND PARTICIPANTS: Retrospective medical record review of 14 patients treated for BCR in the uveitis clinic at Northwestern University. All patients underwent OCT-EDI (58 scans). Clinical symptoms of photopsias/vibrating vision and signs of macular edema, vitreous haze, and retinal vasculitis were graded; a second grading scale was developed for the evaluation of OCT-EDI. Individual scans of each eye of each patient at each point were graded in a masked fashion. EXPOSURE: Optical coherence tomography with EDI in BCR. MAIN OUTCOMES AND MEASURES: Spearman rank correlation of clinical measures to OCT-EDI measures. RESULTS: The most frequent score in each clinical category was 0 (inactive). In those BCR patients with symptoms (21 eye examinations), the subjective complaint of photopsias/vibrating vision was associated with the objective finding of suprachoroidal fluid on OCT-EDI (P = .003), and the frequency and severity of photopsias correlated with the thickness of the fluid band (Pearson product moment correlation, 0.39). Two of the clinical markers of disease activity measured in this study (vasculitis and vitreous haze) also showed a significant Spearman rank correlation with the presence and amount of suprachoroidal fluid on OCT-EDI (vasculitis, 0.45 [P < .001]; vitreous haze, 0.59 [P < .001]). CONCLUSIONS AND RELEVANCE: The presence of suprachoroidal fluid on OCT-EDI appears to correlate with the subjective complaints of photopsias in patients with BCR and other more easily assessed clinical features such as vasculitis and vitreous haze. Optical coherence tomography with EDI may be a useful tool for objective monitoring of BCR.
[Pt] Publication type:JOURNAL ARTICLE
[Em] Entry month:1408
[Js] Journal subset:AIM; IM
[St] Status:In-Data-Review
[do] DOI:10.1001/jamaophthalmol.2014.877

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[PMID]: 25051945
[Au] Autor:Varma N; Hinojar R; D'Cruz D; Arroyo Ucar E; Indermuehle A; Peel S; Greil G; Gaddum N; Chowienczyk P; Nagel E; Botnar RM; Puntmann VO
[Ad] Address:Cardiovascular Imaging Department, Division of Imaging Sciences and Biomedical Engineering, King's College London, London, United Kingdom....
[Ti] Title:Coronary Vessel Wall Contrast Enhancement Imaging as a Potential Direct Marker of Coronary Involvement: Integration of Findings From CAD and SLE Patients.
[So] Source:JACC Cardiovasc Imaging;7(8):762-70, 2014 Aug.
[Is] ISSN:1876-7591
[Cp] Country of publication:United States
[La] Language:eng
[Ab] Abstract:OBJECTIVES: This study investigated the feasibility of visual and quantitative assessment of coronary vessel wall contrast enhancement (CE) for detection of symptomatic atherosclerotic coronary artery disease (CAD) and subclinical coronary vasculitis in autoimmune inflammatory disease (systemic lupus erythematosus [SLE]), as well as the association with aortic stiffness, an established marker of risk. BACKGROUND: Coronary CE by cardiac magnetic resonance (CMR) is a novel noninvasive approach to visualize gadolinium contrast uptake within the coronary artery vessel wall. METHODS: A total of 75 subjects (CAD: n = 25; SLE: n = 27; control: n = 23) underwent CMR imaging using a 3-T clinical scanner. Coronary arteries were visualized by a T2-prepared steady state free precession technique. Coronary wall CE was visualized using inversion-recovery T1 weighted gradient echo sequence 40 min after administration of 0.2 mmol/kg gadobutrol. Proximal coronary segments were visually examined for distribution of CE and quantified for contrast-to-noise ratio (CNR) and total CE area. RESULTS: Coronary CE was prevalent in patients (93%, n = 42) with a diffuse pattern for SLE and a patchy/regional distribution in CAD patients. Compared with control subjects, CNR values and total CE area in patients with CAD and SLE were significantly higher (mean CNR: 3.9 ± 2.5 vs. 6.9 ± 2.5 vs. 6.8 ± 2.0, respectively; p < 0.001; total CE area: median 0.8 [interquartile range (IQR): 0.6 to 1.2] vs. 3.2 [IQR: 2.6 to 4.0] vs. 3.3 [IQR: 1.9 to 4.5], respectively; p < 0.001). Both measures were positively associated with aortic stiffness (CNR: r = 0.61, p < 0.01; total CE area: 0.36, p = 0.03), hypercholesterolemia (r = 0.68, p < 0.001; r = 0.61, p < 0.001) and hypertension (r = 0.40, p < 0.01; r = 0.32, p < 0.05). CONCLUSIONS: We demonstrate that quantification of coronary CE by CNR and total CE area is feasible for detection of subclinical and clinical uptake of gadolinium within the coronary vessel wall. Coronary vessel wall CE may become an instrumental novel direct marker of vessel wall injury and remodeling in subpopulations at risk.
[Pt] Publication type:JOURNAL ARTICLE
[Em] Entry month:1408
[Js] Journal subset:IM
[St] Status:In-Data-Review

  5 / 32102 MEDLINE  
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[PMID]: 24809802
[Au] Autor:Chirico V; Lacquaniti A; Salpietro V; Munafò C; Calabrò MP; Buemi M; Arrigo T; Salpietro C
[Ad] Address:Department of Pediatric Sciences, University of Messina, 98100, Messina, Italy, valeriachirico@hotmail.it.
[Ti] Title:High-mobility group box 1 (HMGB1) in childhood: from bench to bedside.
[So] Source:Eur J Pediatr;173(9):1123-36, 2014 Sep.
[Is] ISSN:1432-1076
[Cp] Country of publication:Germany
[La] Language:eng
[Ab] Abstract:UNLABELLED: High-mobility group box protein 1 (HMGB1) is a nonhistone nuclear protein that has a dual function. Inside the cell, HMGB1 binds DNA, regulating transcription and determining chromosomal architecture. Outside the cell, HMGB1 activates the innate system and mediates a wide range of physiological and pathological responses. HMGB1 exerts these actions through differential engagement of multiple surface receptors, including Toll-like receptor (TLR)2, TLR4, and receptor for advanced glycation end products (RAGE). HMGB1 is implicated as a late mediator of sepsis and is also involved in inflammatory and autoimmune diseases, such as rheumatoid arthritis and systemic lupus erythematosus. Interestingly, HMGB1 was associated with tumor progression, becoming a potential therapeutic target, due to its involvement in the resistance to chemotherapy. Its implication on the pathogenesis of systemic vasculitis and inflammatory bowel diseases has also been evaluated. Moreover, it regulates neuroinflammation after traumatic brain injuries or cerebral infectious diseases. The aim of this review is to analyze these different roles of HMGB1, both in physiological and pathological conditions, discussing clinical and scientific implications in the field of pediatrics. CONCLUSION: HMGB1 plays a key role in several pediatric diseases, opening new scenarios for diagnostic biomarkers and therapeutic strategies development.
[Pt] Publication type:JOURNAL ARTICLE
[Em] Entry month:1408
[Js] Journal subset:IM
[St] Status:In-Data-Review
[do] DOI:10.1007/s00431-014-2327-1

  6 / 32102 MEDLINE  
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[PMID]: 25125628
[Au] Autor:Larson AR; Granter SR
[Ad] Address:From the Department of Pathology, Brigham and Women's Hospital, and Harvard Medical School, Boston, MA.
[Ti] Title:Utility of immunofluorescence testing for vascular IgA in adult patients with leukocytoclastic vasculitis.
[So] Source:Am J Clin Pathol;142(3):370-4, 2014 Sep.
[Is] ISSN:1943-7722
[Cp] Country of publication:United States
[La] Language:eng
[Ab] Abstract:OBJECTIVES: The purpose of this study was to examine the utility of immunofluorescence (IF) testing in patients with leukocytoclastic vasculitis (LCV), particularly with regard to usefulness in the diagnosis of Henoch-Schönlein purpura (HSP). METHODS: We retrospectively analyzed the results of IF testing in 96 patients with LCV and compared results with clinical criteria and clinical impression at the time of biopsy by review of the medical record. RESULTS: Sensitivity and specificity of vascular immunoglobulin A (IgA) for the diagnosis of HSP were 0.86 and 0.84, respectively. Positive predictive value was 0.48 and negative predictive value was 0.97. Of the 53 patients with LCV who did not meet clinical criteria for HSP and carried a low clinical suspicion for the disease at the time of biopsy, seven had moderate to strong staining for vascular IgA. Only one of these patients was determined to have HSP. CONCLUSIONS: Our data confirm that vascular IgA is nonspecific and also demonstrate that the utility of IF studies for vasculitis is influenced by the clinical presentation and the clinician's level of suspicion for HSP. Our data show that the clinical features and the overall clinical impression are helpful in selecting which patients are most likely to benefit from IF testing.
[Pt] Publication type:JOURNAL ARTICLE
[Em] Entry month:1408
[Js] Journal subset:AIM; IM
[St] Status:In-Data-Review
[do] DOI:10.1309/AJCPMB1A9QSWUJDY

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[PMID]: 25125622
[Au] Autor:Lin MW; Silvestrini RA; Culican S; Campbell D; Fulcher DA
[Ad] Address:From the Department of Immunopathology, Pathology West, ICPMR, Westmead Hospital, Westmead, Australia, and Discipline of Medicine, Sydney Medical School, University of Sydney, Sydney, Australia. ming-wei.lin@sydney.edu.au....
[Ti] Title:A dual-fixed neutrophil substrate improves interpretation of antineutrophil cytoplasmic antibodies by indirect immunofluorescence.
[So] Source:Am J Clin Pathol;142(3):325-30, 2014 Sep.
[Is] ISSN:1943-7722
[Cp] Country of publication:United States
[La] Language:eng
[Ab] Abstract:OBJECTIVES: To determine whether the addition of a formalin-fixed neutrophil substrate could improve interpretation and prediction of autoantigenic specificity in antineutrophil cytoplasmic antibody (ANCA) testing. METHODS: Routine diagnostic samples sent for ANCA testing were analyzed prospectively on a dual substrate of both ethanol- and formalin-fixed neutrophils. Positive samples on ethanol-fixed neutrophils were deemed "typical" if formalin-fixed neutrophils also stained, and "atypical" if not. Indirect immunofluorescence (IIF) results were correlated with antimyeloperoxidase (MPO) and anti-proteinase 3 (PR3) results with an enzyme-linked immunosorbent assay (ELISA). RESULTS: Of 1,426 samples, 201 from unique patients were ANCA-positive (200 on IIF, 1 on ELISA alone). Thirty-two (45%) of 71 typical ANCA staining patterns were positive for either an anti-MPO or anti-PR3 antibodies, whereas only one (0.8%) of 129 atypical patterns was ELISA-positive, in a patient without systemic vasculitis. Only one (3%) of 34 ELISA-positive samples had a negative IIF-ANCA (1/1,426 patients, 0.07%), and this patient did not have vasculitis. CONCLUSIONS: Concomitant staining on formalin fixation of IIF-positive ethanol-fixed ANCA samples improves the interpretation of ANCA testing and is predictive of vasculitis autoantigens MPO and PR3.
[Pt] Publication type:JOURNAL ARTICLE
[Em] Entry month:1408
[Js] Journal subset:AIM; IM
[St] Status:In-Data-Review
[do] DOI:10.1309/AJCPG02FGQVAUSIU

  8 / 32102 MEDLINE  
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[PMID]: 24722305
[Au] Autor:Obusez EC; Hui F; Hajj-Ali RA; Cerejo R; Calabrese LH; Hammad T; Jones SE
[Ad] Address:From the Department of Diagnostic Radiology (E.C.O., S.E.J.), Imaging Institute....
[Ti] Title:High-resolution MRI vessel wall imaging: spatial and temporal patterns of reversible cerebral vasoconstriction syndrome and central nervous system vasculitis.
[So] Source:AJNR Am J Neuroradiol;35(8):1527-32, 2014 Aug.
[Is] ISSN:1936-959X
[Cp] Country of publication:United States
[La] Language:eng
[Ab] Abstract:BACKGROUND AND PURPOSE: High-resolution MR imaging is an emerging tool for evaluating intracranial artery disease. It has an advantage of defining vessel wall characteristics of intracranial vascular diseases. We investigated high-resolution MR imaging arterial wall characteristics of CNS vasculitis and reversible cerebral vasoconstriction syndrome to determine wall pattern changes during a follow-up period. MATERIALS AND METHODS: We retrospectively reviewed 3T-high-resolution MR imaging vessel wall studies performed on 26 patients with a confirmed diagnosis of CNS vasculitis and reversible cerebral vasoconstriction syndrome during a follow-up period. Vessel wall imaging protocol included black-blood contrast-enhanced T1-weighted sequences with fat suppression and a saturation band, and time-of-flight MRA of the circle of Willis. Vessel wall characteristics including enhancement, wall thickening, and lumen narrowing were collected. RESULTS: Thirteen patients with CNS vasculitis and 13 patients with reversible cerebral vasoconstriction syndrome were included. In the CNS vasculitis group, 9 patients showed smooth, concentric wall enhancement and thickening; 3 patients had smooth, eccentric wall enhancement and thickening; and 1 patient was without wall enhancement and thickening. Six of 13 patients had follow-up imaging; 4 patients showed stable smooth, concentric enhancement and thickening; and 2 patients had resoluton of initial imaging findings. In the reversible cerebral vasoconstriction syndrome group, 10 patients showed diffuse, uniform wall thickening with negligible-to-mild enhancement. Nine patients had follow-up imaging, with 8 patients showing complete resolution of the initial findings. CONCLUSIONS: Postgadolinium 3T-high-resolution MR imaging appears to be a feasible tool in differentiating vessel wall patterns of CNS vasculitis and reversible cerebral vasoconstriction syndrome changes during a follow-up period.
[Pt] Publication type:JOURNAL ARTICLE
[Em] Entry month:1408
[Js] Journal subset:IM
[St] Status:In-Data-Review
[do] DOI:10.3174/ajnr.A3909

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[PMID]: 25033936
[Au] Autor:Ferlazzo E; Gambardella A; Bellavia M; Gasparini S; Mumoli L; Labate A; Cianci V; Russo C; Aguglia U
[Ti] Title:Positivity to p-ANCA in patients with status epilepticus.
[So] Source:BMC Neurol;14:148, 2014.
[Is] ISSN:1471-2377
[Cp] Country of publication:England
[La] Language:eng
[Ab] Abstract:BACKGROUND: Status epilepticus (SE) may occur in the setting of several internal or neurologic diseases. Anti-neutrophilic cytoplasmic antibodies (ANCA) are a group of Ig that may be observed in patients with different autoimmune disorders but are particularly associated with systemic vasculitis named ANCA-associated-vasculities (AAV). We herein report 3 patients with SE and positivity to p-ANCA. CASE PRESENTATION: One patient had a catastrophic evolution and died 5 months after disease onset. The other two patients had a good outcome and remained seizure-free at 30 months and 5 years of follow-up respectively. CONCLUSION: This report highlights the importance of considering ANCA dosage in patients with SE of unclear origin.
[Pt] Publication type:JOURNAL ARTICLE
[Em] Entry month:1407
[Js] Journal subset:IM
[St] Status:In-Process
[do] DOI:10.1186/1471-2377-14-148

  10 / 32102 MEDLINE  
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[PMID]: 25061613
[Au] Autor:Park UC; Kim TW; Yu HG
[Ad] Address:Department of Ophthalmology, National Medical Center, 245 Euljiro, Jung-gu, Seoul 100-799, Republic of Korea.
[Ti] Title:Immunopathogenesis of ocular Behçet's disease.
[So] Source:J Immunol Res;2014:653539, 2014.
[Is] ISSN:2314-7156
[Cp] Country of publication:United States
[La] Language:eng
[Ab] Abstract:Behçet's disease (BD) is a chronic recurrent systemic inflammatory disorder of unknown etiology characterized by oral and genital ulcerations, skin lesions, and uveitis. The ocular involvement of BD, or Behçet's uveitis (BU), is characterized by panuveitis or posterior uveitis with occlusive retinal vasculitis and tends to be more recurrent and sight threatening than other endogenous autoimmune uveitides, despite aggressive immunosuppression. Although pathogenesis of BD is unclear, researches have revealed that immunological aberrations may be the cornerstone of BD development. General hypothesis of BD pathogenesis is that inflammatory response is initiated by infectious agents or autoantigens in patients with predisposing genetic factors and perpetuated by both innate and acquired immunity. In addition, a network of immune mediators plays a substantial role in the inflammatory cascade. Recently, we found that the immunopathogenesis of BU is distinct from other autoimmune uveitides regarding intraocular effector cell profiles, maturation markers of dendritic cells, and the cytokine/chemokine environment. In addition, accumulating evidence indicates the involvement of Th17 cells in BD and BU. Recent studies on genetics and biologics therapies in refractory BU also support the immunological association with the pathogenesis of BU. In this review, we provide an overview of novel findings regarding the immunopathogenesis of BU.
[Pt] Publication type:JOURNAL ARTICLE
[Em] Entry month:1407
[Js] Journal subset:IM
[St] Status:In-Process
[do] DOI:10.1155/2014/653539


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