Database : MEDLINE
Search on : Behcet and Syndrome [Words]
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[PMID]: 29516432
[Au] Autor:Voiriot G; Parrot A; Antoine M; Gibelin A; Haddad S; Carette MF; Fartoukh M; Khalil A
[Ad] Address:Service de Réanimation Médico-chirurgicale, Hôpital Tenon, Hôpitaux Universitaires Est Parisien, Assistance Publique-Hôpitaux de Paris, 4, rue de la Chine, 75020, Paris, France. guillaume.voiriot@aphp.fr.
[Ti] Title:Transcatheter embolotherapy of pulmonary artery aneurysms as emergency treatment of hemoptysis in Behcet patients: experience of a referral center and a review of the literature.
[So] Source:Intern Emerg Med;, 2018 Mar 07.
[Is] ISSN:1970-9366
[Cp] Country of publication:Italy
[La] Language:eng
[Ab] Abstract:Hemoptysis is a life-threatening complication of Behcet's disease that is likely related to pulmonary artery aneurysm (PAA). Vascular interventional radiology may offer effective emergency therapeutic option, but has not been thoroughly investigated in this setting. A case series of a French referral center for hemoptysis combined with a literature review of case reports was conducted. Between 1995 and 2016, 12 patients were referred to our center for hemoptysis revealing or complicating the course of Behcet's disease. Pulmonary artery aneurysm (PAA) was the mechanism of hemoptysis in ten patients, nine of whom were treated by a transcatheter embolotherapy. Combining an additional 8 case reports from the literature, 17 patients treated by transcatheter embolotherapy for PAA were analyzed. The duration of the course of Behcet's disease was 22 months [IQR 3-45] at the time of PAA diagnosis. Transcatheter embolotherapy of PAA was successful for immediately controlling hemoptysis in all patients, without major complication except for one. Hemoptysis recurred in seven patients (41%) within 5 months [IQR 1-12]. The use of coils for transcatheter embolotherapy was associated with hemoptysis recurrence. A bronchosystemic hypervascularization related to the previously occluded PAA was the main mechanism of bleeding recurrence, leading to bronchosystemic artery embolization in four patients and surgery in two patients. Behcet's disease-related hemoptysis is mainly due to PAA. Transcatheter embolotherapy should be considered as the first-line emergency treatment for PAA-related hemoptysis, in association with the immunosuppressive regimen. Hemoptysis may recur in half of the cases, involving preferentially a bronchosystemic arterial mechanism.
[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/s11739-018-1817-y

  2 / 8833 MEDLINE  
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[PMID]: 29490685
[Au] Autor:Zerkaoui M; Laarabi FZ; Ajhoun Y; Chkirate B; Sefiani A
[Ad] Address:Human Genomic Centre, Faculty of Medicine and Pharmacy, Mohammed V University, Rabat, Morocco. maria.zerkaoui@gmail.com.
[Ti] Title:A novel single variant in the MEFV gene causing Mediterranean fever and Behçet's disease: a case report.
[So] Source:J Med Case Rep;12(1):53, 2018 Mar 01.
[Is] ISSN:1752-1947
[Cp] Country of publication:England
[La] Language:eng
[Ab] Abstract:BACKGROUND: Familial Mediterranean fever is an autoinflammatory disease of unknown etiology, characterized clinically by recurrent attacks of sudden-onset fever with arthralgia and/or thoracoabdominal pain and pathogenetically by autosomal recessive inheritance due to a mutation in the MEFV gene. Behçet's disease is an inflammatory disease characterized by recurrent oral and genital aphthous ulcerations, uveitis, and skin lesions. Preliminarily, our literature review suggested that patients with familial Mediterranean fever who also have Behçet's disease have only a single mutated familial Mediterranean fever gene. The MEFV gene mutation responsible for familial Mediterranean fever is probably a susceptibility factor for Behçet's disease, particularly for patients with vascular involvement, and both disorders can occur concurrently in a patient, as in the present case. CASE PRESENTATION: A 10-year-old girl of Moroccan origin presented to our institution for genetic consultation for genetic testing of the MEFV gene. She had fever associated with abdominal and diffuse joint pain in addition to headache. These symptoms have oriented pediatricians to familial Mediterranean fever. The evolution was marked by Behçet's syndrome symptoms. Sanger sequencing followed by complete exome sequencing analysis of the MEFV gene for the proband mutation revealed a novel variant. We conclude that the novel single variant c.2078 T > A (p.Met693Lys) could be responsible for the association of familial Mediterranean fever and Behçet's disease. CONCLUSION: To the best of our knowledge, this is the first report of a new variant in exon 10 of the MEFV gene in a Moroccan family. This novel variant should be listed in the MEFV sequence variant databases.
[Pt] Publication type:JOURNAL ARTICLE
[Em] Entry month:1803
[Cu] Class update date: 180307
[Lr] Last revision date:180307
[St] Status:In-Process
[do] DOI:10.1186/s13256-017-1552-4

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[PMID]: 28468639
[Au] Autor:Sasajima H; Yagi S; Osada H; Zako M
[Ad] Address:Department of Ophthalmology, Aichi Medical University, Nagakute, Aichi, Japan.
[Ti] Title:Botulinum toxin-induced acute anterior uveitis in a patient with Behçet's disease under infliximab treatment: a case report.
[So] Source:J Med Case Rep;11(1):124, 2017 May 04.
[Is] ISSN:1752-1947
[Cp] Country of publication:England
[La] Language:eng
[Ab] Abstract:BACKGROUND: Injections of lipopolysaccharide in animal models generate acute anterior uveitis (also known as endotoxin-induced uveitis), but the effects of lipopolysaccharide injection are unknown in humans. We describe an unusual case in which acute anterior uveitis was dramatically activated subsequent to botulinum toxin injection in a patient with Behçet's disease but the acute anterior uveitis was satisfactorily attenuated by infliximab. CASE PRESENTATION: A 53-year-old Japanese man had normal ocular findings at his regularly scheduled appointment. He had been diagnosed as having incomplete-type Behçet's disease 11 years before. Three years after the diagnosis he was given systemic infusions of 5 mg/kg infliximab every 8 weeks and he had not experienced a uveitis attack for 8 years with no treatment other than infliximab. Two days after the eye examination, he received intracutaneous botulinum toxin injections to treat axillary hyperhidrosis on both sides. Three hours after the injections, he noted rapidly increasing floaters in his right eye. Four days after the injections, his right eye showed severe acute anterior uveitis with deteriorated aqueous flare and anterior vitreous opacity. He received his scheduled infliximab injection, and the right acute anterior uveitis immediately attenuated. CONCLUSIONS: Botulinum toxin may have clinical effects similar to those of lipopolysaccharide in endotoxin-induced uveitis models. To the best of our knowledge, this is the first report to suggest that botulinum toxin may trigger acute anterior uveitis, although the precise mechanism is still unclear.
[Mh] MeSH terms primary: Behcet Syndrome/drug therapy
Botulinum Toxins/adverse effects
Hyperhidrosis/drug therapy
Infliximab/administration & dosage
Neurotoxins/administration & dosage
Uveitis/chemically induced
[Mh] MeSH terms secundary: Humans
Injections, Subcutaneous
Male
Middle Aged
Tomography, Optical Coherence
Uveitis/diagnostic imaging
Uveitis/drug therapy
[Pt] Publication type:CASE REPORTS; JOURNAL ARTICLE
[Nm] Name of substance:0 (Neurotoxins); B72HH48FLU (Infliximab); EC 3.4.24.69 (Botulinum Toxins)
[Em] Entry month:1803
[Cu] Class update date: 180305
[Lr] Last revision date:180305
[Js] Journal subset:IM
[Da] Date of entry for processing:170505
[St] Status:MEDLINE
[do] DOI:10.1186/s13256-017-1288-1

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[PMID]: 29390339
[Au] Autor:Chen Y; Shen Y; Ma HF; Cai JF; Hua YQ; Zou J; Guan JL
[Ad] Address:Rheumatology and Immunology Department.
[Ti] Title:Infliximab associated with life-threatening lung infection in a patient with Behcet disease with intestinal and hematopoietic system involvement: A case report.
[So] Source:Medicine (Baltimore);96(50):e9202, 2017 Dec.
[Is] ISSN:1536-5964
[Cp] Country of publication:United States
[La] Language:eng
[Ab] Abstract:RATIONALE: Tumor necrosis factor (TNF-α) participates in the pathophysiology of Behcet's disease (BD) and myelodysplastic syndrome (MDS). Infliximab is recommaned for the most severe type of BD, however, there is little evidence for its effectiveness in BD associated MDS. PATIENT CONCERNS: A 46-year-old female, initially diagnosed with intestinal BD and leukopenia was later diagnosed as MDS. Treatement with infliximab and other immunoregulators lead to life-threatening pneumonia. DIAGNOSIS: Intestinal BD associated with MDS involving trisomy 8. INTERVENTIONS: The patient initially treated with methylprednisolone, thalidomide, cyclosporine A, and infliximab, which lead to severe lung infection. Therefore, the patient was transferred to Intensive Care Unit for life supportive, anti-infection and immune improving therapy. OUTCOMES: The patient survived from the lung infection. With combination of methylprednisolone, thalidomide and cyclosporine A, the patient recovered from her intestinal ulceration and MDS manifestations. LESSONS: Infliximab treatment may not benefit a patient with BD associated with MDS but place the patient at risk of infection.
[Mh] MeSH terms primary: Antirheumatic Agents/adverse effects
Antirheumatic Agents/therapeutic use
Behcet Syndrome/complications
Behcet Syndrome/drug therapy
Infliximab/adverse effects
Infliximab/therapeutic use
Myelodysplastic Syndromes/drug therapy
Myelodysplastic Syndromes/etiology
Pneumonia/chemically induced
[Mh] MeSH terms secundary: Cyclosporine/therapeutic use
Female
Glucocorticoids/therapeutic use
Humans
Immunosuppressive Agents/therapeutic use
Methylprednisolone/therapeutic use
Middle Aged
Thalidomide/therapeutic use
[Pt] Publication type:CASE REPORTS; JOURNAL ARTICLE
[Nm] Name of substance:0 (Antirheumatic Agents); 0 (Glucocorticoids); 0 (Immunosuppressive Agents); 4Z8R6ORS6L (Thalidomide); 83HN0GTJ6D (Cyclosporine); B72HH48FLU (Infliximab); X4W7ZR7023 (Methylprednisolone)
[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.0000000000009202

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[PMID]: 29269695
[Au] Autor:Hamada K; Takei R; Sakiyama Y; Moriyama H; Hashiguchi A; Takashima H
[Ad] Address:Department of Neurology and Geriatrics, Kagoshima University Graduate School of Medical and Dental Sciences.
[Ti] Title:[A case of chronic progressive neuro-Behçet disease with extensive cerebral atrophy and elevated CSF IL-6 activity treated with infliximab].
[So] Source:Rinsho Shinkeigaku;58(1):30-34, 2018 Jan 26.
[Is] ISSN:1882-0654
[Cp] Country of publication:Japan
[La] Language:jpn
[Ab] Abstract:A 43-year-old man without a previous episode of uveitis presented with slowly progressive neurological symptoms that appeared within the past year such as dysarthria, ataxic gait, and behavioral changes. Brain MRI findings showed atrophic lesions in the brainstem and cerebellum. Because these clinical symptoms and abnormal MRI findings indicated spinocerebellar degeneration as the initial diagnosis, he was admitted to our hospital. On admission, we noticed that he had non-neurological manifestations of Behçet disease, such as stomatitis, genital ulcers, and folliculitis. HLA-B51 was positive. He also showed pleocytosis (29 cells/mm , predominantly mononuclear cells) and elevated cerebrospinal fluid (CSF) IL-6 levels (213 pg/ml), hence he was diagnosed with chronic progressive neuro-Behçet disease (CPNBD). The therapeutic effect of a high-dose intravenous methylprednisolone pulse (1,000 mg/day for 3 days) and methotrexate (maximum dosage, 16 mg/week) was poor against both neurological symptoms and CSF findings. Intravenous infliximab therapy (5 mg/kg, 2 weeks) dramatically decreased CSF IL-6 levels (13 pg/ml) but clinical symptoms remained unchanged. MRI findings of extensive cerebral atrophy and increased CSF IL-6 levels at the pretreatment time point reflected irreversible neurological involvement in CPNBD. For cases with progressive psychiatric symptoms and cerebellar ataxia in the early stage of the disease, skin manifestations should be examined immediately, CSF IL-6 levels measured, and immunosuppressive therapy initiated before CPNBD progresses to brainstem atrophy.
[Mh] MeSH terms primary: Behcet Syndrome/diagnosis
Behcet Syndrome/drug therapy
Brain/pathology
Infliximab/administration & dosage
Interleukin-6/cerebrospinal fluid
[Mh] MeSH terms secundary: Adult
Atrophy
Behcet Syndrome/cerebrospinal fluid
Behcet Syndrome/pathology
Biomarkers/cerebrospinal fluid
Chronic Disease
Disease Progression
Humans
Male
Treatment Outcome
[Pt] Publication type:CASE REPORTS; JOURNAL ARTICLE
[Nm] Name of substance:0 (Biomarkers); 0 (Interleukin-6); B72HH48FLU (Infliximab)
[Em] Entry month:1802
[Cu] Class update date: 180227
[Lr] Last revision date:180227
[Js] Journal subset:IM
[Da] Date of entry for processing:171223
[St] Status:MEDLINE
[do] DOI:10.5692/clinicalneurol.cn-001086

  6 / 8833 MEDLINE  
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[PMID]: 29481922
[Au] Autor:Lakha S; Png CYM; Chun K; Ting W
[Ad] Address:Icahn School of Medicine at Mount Sinai, New York, NY 10029.
[Ti] Title:Recurrent Iliofemoral Venous Thrombosis in the Setting of May-Thurner Syndrome as the Presenting Symptom of Behcet's Disease.
[So] Source:Ann Vasc Surg;, 2018 Feb 23.
[Is] ISSN:1615-5947
[Cp] Country of publication:Netherlands
[La] Language:eng
[Ab] Abstract:OBJECTIVE: Vascular manifestations including pulmonary artery aneurysms and venous thrombosis are seen in up to 14% of patients with Behcet's Disease. We report a patient who had recurrent deep vein thrombosis (DVT) as the presenting symptom of Behcet's Disease. METHODS: A 19-year-old male who presented with acute iliofemoral DVT, confirmed by intravascular ultrasound (IVUS) and venogram. May-Thurner syndrome was also observed. Repeated catheter-based pharmaco-mechanical thrombolysis, thrombectomy and subsequent iliac vein stenting were performed. The patient was then discharged on rivaroxaban and aspirin. RESULTS: Five months later, the patient experienced left calf pain. In the interim, he had been diagnosed with Behcet's Disease by a rheumatologist who was consulted due to oral ulcers and skin lesions, and accordingly started on prednisone, colchicine and azathioprine. At this time, IVUS and venogram revealed thrombotic occlusion of the previously placed stent. tPA was infused into the stent and pharmaco-mechanical thrombectomy restored flow through the left iliac veins. Follow-up laboratory workup revealed that subtherapeutic azathioprine dosing, and after appropriate adjustment, the patient has been asymptomatic for 12 months. CONCLUSIONS: Acute refractory DVT is a possible presenting symptom of Behcet's Disease, which may be complicated by May-Thurner syndrome. Such patients should receive therapeutic immunosuppression in addition to anticoagulation.
[Pt] Publication type:JOURNAL ARTICLE
[Em] Entry month:1802
[Cu] Class update date: 180226
[Lr] Last revision date:180226
[St] Status:Publisher

  7 / 8833 MEDLINE  
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[PMID]: 29463220
[Au] Autor:Lee JH; Lee CS; Lee SC
[Ad] Address:Department of Ophthalmology, The Institute of Vision Research, Yonsei University College of Medicine, Yonsei-ro 50-1, Seodaemun-gu, Seoul, Republic of Korea.
[Ti] Title:Interferon alpha-2a treatment for refractory Behcet uveitis in Korean patients.
[So] Source:BMC Ophthalmol;18(1):52, 2018 Feb 20.
[Is] ISSN:1471-2415
[Cp] Country of publication:England
[La] Language:eng
[Ab] Abstract:BACKGROUND: To evaluate therapeutic outcomes of interferon alpha-2a (IFNα2a) treatment in patients with Behcet's disease who were refractory to immunosuppressive agents. METHODS: This retrospective case series reviewed the medical records of 5 patients with refractory Behcet uveitis from January 2011 to February 2017. IFNα2a was administered at a dose of 3 million IU 3 times per week. Clinical response, relapse rate, and change of visual acuity were evaluated. RESULTS: The mean age of patients was 39.60 ± 9.21 years, and the median treatment duration was 6 months. Four of the 5 patients (80%) presented with responses to IFNα2a without any uveitis attack during the treatment period. The mean number of uveitis attacks/year per patient during the treatment was 0.40 ± 0.89. The mean log of the Minimum Angle of Resolution visual acuity improved from 1.44 ± 0.38 at baseline to 1.02 ± 0.58 at the final follow up. CONCLUSIONS: IFNα2a is an effective therapy for Behcet uveitis refractory to conventional immunosuppressants in Korean patients.
[Pt] Publication type:JOURNAL ARTICLE
[Em] Entry month:1802
[Cu] Class update date: 180223
[Lr] Last revision date:180223
[St] Status:In-Process
[do] DOI:10.1186/s12886-018-0719-0

  8 / 8833 MEDLINE  
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[PMID]: 29443756
[Au] Autor:Zhu Z; Shu X; Long S; Jiang X; Lu N; Zhu X; Liao W
[Ad] Address:Department of Gastroenterology, The First Affiliated Hospital, Nanchang University, Nanchang, Jiangxi, China.
[Ti] Title:Ulcerative colitis followed by the development of typical intestinal Behçet disease: A case report.
[So] Source:Medicine (Baltimore);97(7):e9882, 2018 Feb.
[Is] ISSN:1536-5964
[Cp] Country of publication:United States
[La] Language:eng
[Ab] Abstract:RATIONALE: Intestinal Behçet disease (intestinal BD) and inflammatory bowel disease (IBD) share a lot of characteristics, including genetic background, clinical manifestations, and therapeutic strategies, especially the extraintestinal manifestations, such as oral ulcers, arthralgia, eye lesions, skin lesions, etc, but the coexistence of these 2 diseases are uncommon. Behçet disease with gastrointestinal involvement in ulcerative colitis (UC) patient has been reported in just 1 previous case report, but, which can not be diagnosed as definite intestinal BD based on Korean novel diagnositic criteria due to lacking the typical ileocecal ulcer. PATIENT CONCERNS: We present a 23-year-old woman with ulcerative disease who developed typical intestinal BD, which is the first case report of patient with coexisting UC and typical intestinal BD. DIAGNOSES: This patient was diagnosed as coexistence of intestinal BD and UC base on the clinical manifestations, extra intestinal manifestations and typical colonoscopic findings. INTERVENTIONS: Steroid and methotrexate were administered. OUTCOMES: This patient achieved clinical remission and mucosal healing. LESSONS: Coexistence of intestinal BD and UC is uncommon, and the combination with steroid, methotrexate, and 5-aminosalicylic acids is an effective therapy.
[Mh] MeSH terms primary: Behcet Syndrome
Colitis, Ulcerative
Colonoscopy/methods
Gastrointestinal Tract
Glucocorticoids/administration & dosage
Mesalamine/administration & dosage
Methotrexate/administration & dosage
[Mh] MeSH terms secundary: Adult
Anti-Inflammatory Agents, Non-Steroidal/administration & dosage
Antirheumatic Agents/administration & dosage
Behcet Syndrome/complications
Behcet Syndrome/diagnosis
Behcet Syndrome/physiopathology
Colitis, Ulcerative/complications
Colitis, Ulcerative/diagnosis
Colitis, Ulcerative/physiopathology
Gastrointestinal Tract/diagnostic imaging
Gastrointestinal Tract/pathology
Humans
Male
Remission Induction
Treatment Outcome
[Pt] Publication type:CASE REPORTS; JOURNAL ARTICLE
[Nm] Name of substance:0 (Anti-Inflammatory Agents, Non-Steroidal); 0 (Antirheumatic Agents); 0 (Glucocorticoids); 4Q81I59GXC (Mesalamine); YL5FZ2Y5U1 (Methotrexate)
[Em] Entry month:1802
[Cu] Class update date: 180222
[Lr] Last revision date:180222
[Js] Journal subset:AIM; IM
[Da] Date of entry for processing:180215
[St] Status:MEDLINE
[do] DOI:10.1097/MD.0000000000009882

  9 / 8833 MEDLINE  
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[PMID]: 29429506
[Au] Autor:Jachiet M; Rybojad M; Bouaziz JD
[Ad] Address:Service de dermatologie, hôpital Saint-Louis, INSERM U976, université Paris-Diderot-Paris VII, Sorbonne-Paris-Cité, Paris, France.
[Ti] Title:Quoi de neuf en médecine interne ? [What's new in internal medecine?]
[So] Source:Ann Dermatol Venereol;143 Suppl 3:S23-S28, 2016 Dec.
[Is] ISSN:0151-9638
[Cp] Country of publication:France
[La] Language:fre
[Ab] Abstract:Answering the question « what's new in internal medecine in 2016? ¼ is very challenging. We used 3 methods of article selection to reduce the selection bias: 3 authors, a systematic review of the articles discussed in the weekly bibliographic meeting of our unit (Dermatology department, Saint-Louis Hospital, Paris, France) and a selection of the best articles by several internal medecine practitioners in Paris. Eleven « hot topics ¼ were analyzed: i/lowering cholesterol level but not blood blessure has a significant impact on cardiovascular morbi-mortality in cardiovascular intermediate risk patients; ii/the « treat to treat target ¼ is efficient in psoriatic arthritis; iii/ a genotype/ phenotype correlation favors the separation of ileal Crohn's disease, colonic Crohn's disease and ulcerative colitis; iv/ tocilizumab treatment (anti-IL-6 monoclonal antibody ) is very efficient in giant cell arteritis and slightly efficient in systemic sclerosis; v/ combination therapy using methotrexate plus steroids compared with steroids alone becomes the « gold standard ¼ treatment for juvenile dermatomyositis; vi/ dupilumab treatment (antibody blocking IL-4 and IL-13 receptors) is not only efficient in atopic dermatitis but also in asthma; vii/ think of eosinophilic oesophagitis in a patient with atopic dermatitis and dypshagia or food impaction; viii/ genetic A2 protein dysfunction induces NF-kB hyperactivation and an autoinflammatory disorder with features similar to Behcet's disease; ix/ no new biotherapies have shown high efficacy in systemic lupus erythematosus; x/ nanoparticles loaded with autoantigens induce Tregs and Bregs and may be a promising therapeutic option to treat auto-immune disease in the future; xi/ ipilimumab treatment (anti-CTLA4 antibody, immune checkpoint inhibitor) may induce complete remission in acute myeloid leukemia patients relapsing after haematological stem cell transplantation. Year 2016 is full of great discoveries in internal medicine keeping the dermatologist brain fully open minded.
[Mh] MeSH terms primary: Internal Medicine
[Mh] MeSH terms secundary: Antibodies, Monoclonal/therapeutic use
Antihypertensive Agents/therapeutic use
Antineoplastic Agents, Immunological/therapeutic use
Arthritis, Rheumatoid/drug therapy
Asthma/drug therapy
Autoimmune Diseases/drug therapy
Behcet Syndrome/genetics
Cardiovascular Diseases/prevention & control
Humans
Hydroxymethylglutaryl-CoA Reductase Inhibitors/therapeutic use
Inflammatory Bowel Diseases/genetics
Ipilimumab/therapeutic use
Skin Diseases/therapy
[Pt] Publication type:JOURNAL ARTICLE; REVIEW
[Nm] Name of substance:0 (Antibodies, Monoclonal); 0 (Antihypertensive Agents); 0 (Antineoplastic Agents, Immunological); 0 (Hydroxymethylglutaryl-CoA Reductase Inhibitors); 0 (Ipilimumab); 0 (SAR231893)
[Em] Entry month:1802
[Cu] Class update date: 180221
[Lr] Last revision date:180221
[Js] Journal subset:IM
[Da] Date of entry for processing:180213
[St] Status:MEDLINE

  10 / 8833 MEDLINE  
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[PMID]: 29328521
[Au] Autor:Maggi P; Absinta M; Grammatico M; Vuolo L; Emmi G; Carlucci G; Spagni G; Barilaro A; Repice AM; Emmi L; Prisco D; Martinelli V; Scotti R; Sadeghi N; Perrotta G; Sati P; Dachy B; Reich DS; Filippi M; Massacesi L
[Ad] Address:Department of Neurology, Université Libre de Bruxelles, Brussels, Belgium.
[Ti] Title:Central vein sign differentiates Multiple Sclerosis from central nervous system inflammatory vasculopathies.
[So] Source:Ann Neurol;83(2):283-294, 2018 Feb.
[Is] ISSN:1531-8249
[Cp] Country of publication:United States
[La] Language:eng
[Ab] Abstract:OBJECTIVES: In multiple sclerosis (MS), magnetic resonance imaging (MRI) is a sensitive tool for detecting white matter lesions, but its diagnostic specificity is still suboptimal; ambiguous cases are frequent in clinical practice. Detection of perivenular lesions in the brain (the "central vein sign") improves the pathological specificity of MS diagnosis, but comprehensive evaluation of this MRI biomarker in MS-mimicking inflammatory and/or autoimmune diseases, such as central nervous system (CNS) inflammatory vasculopathies, is lacking. In a multicenter study, we assessed the frequency of perivenular lesions in MS versus systemic autoimmune diseases with CNS involvement and primary angiitis of the CNS (PACNS). METHODS: In 31 patients with inflammatory CNS vasculopathies and 52 with relapsing-remitting MS, 3-dimensional T2*-weighted and T2-fluid-attenuated inversion recovery images were obtained during a single MRI acquisition after gadolinium injection. For each lesion, the central vein sign was evaluated according to consensus guidelines. For each patient, lesion count, volume, and brain location, as well as fulfillment of dissemination in space MRI criteria, were assessed. RESULTS: MS showed higher frequency of perivenular lesions (median = 88%) than did inflammatory CNS vasculopathies (14%), without overlap between groups or differences between 3T and 1.5T MRI. Among inflammatory vasculopathies, Behçet disease showed the highest median frequency of perivenular lesions (34%), followed by PACNS (14%), antiphospholipid syndromes (12%), Sjögren syndrome (11%), and systemic lupus erythematosus (0%). When a threshold of 50% perivenular lesions was applied, central vein sign discriminated MS from inflammatory vasculopathies with a diagnostic accuracy of 100%. INTERPRETATION: The central vein sign differentiates inflammatory CNS vasculopathies from MS at standard clinical magnetic field strengths. Ann Neurol 2018;83:283-294.
[Pt] Publication type:JOURNAL ARTICLE
[Em] Entry month:1801
[Cu] Class update date: 180221
[Lr] Last revision date:180221
[St] Status:In-Data-Review
[do] DOI:10.1002/ana.25146


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