The Experts below are selected from a list of 35193 Experts worldwide ranked by ideXlab platform
Olivier Benveniste - One of the best experts on this subject based on the ideXlab platform.
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granulomatosis associated Myositis high prevalence of sporadic inclusion body Myositis
Neurology, 2020Co-Authors: Yannick Dieudonne, Olivier Benveniste, Yves Allenbach, Kuberaka Mariampillai, Benoit Nespola, Sarah Leonardlouis, B Hervier, Beatrice Lannes, Andoni Echanizlaguna, Daniel WendlingAbstract:Objective To refine the predictive significance of muscle granuloma in patients with Myositis. Methods A group of 23 patients with Myositis and granuloma on muscle biopsy (granuloma-Myositis) from 8 French and Belgian centers was analyzed and compared with (1) a group of 23 patients with Myositis without identified granuloma (control-Myositis) randomly sampled in each center and (2) a group of 20 patients with sporadic inclusion body Myositis (sIBM) without identified granuloma (control-sIBM). Results All but 2 patients with granuloma-Myositis had extramuscular involvement, including signs common in sarcoidosis that were systematically absent in the control-Myositis and the control-sIBM groups. Almost half of patients with granuloma-Myositis matched the diagnostic criteria for sIBM. In these patients, other than the granuloma, the characteristics of the myopathy and its nonresponse to treatment were similar to the control-sIBM patients. Aside from 1 patient with Myositis overlapping with systemic sclerosis, the remaining patients with granuloma-Myositis did not match the criteria for a well-defined Myositis subtype, suggesting pure sarcoidosis. Matching criteria for sIBM was the sole feature independently associated with nonresponse to myopathy treatment in patients with granuloma-Myositis. Conclusion Patients with granuloma-Myositis should be carefully screened for sIBM associated with sarcoidosis in order to best tailor their care.
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CD8+T-bet+ cells as a predominant biomarker for inclusion body Myositis
Autoimmunity Reviews, 2019Co-Authors: Gaëlle Dzangué-tchoupou, Yves Allenbach, Kuberaka Mariampillai, Loïs Bolko, Damien Amelin, Wladimir Mauhin, Aurélien Corneau, Catherine Blanc, Olivier BenvenisteAbstract:Background: Myositis is a heterogeneous group of muscular auto-immune diseases with clinical and pathological criteria that allow the classification of patients into different sub-groups. Inclusion body Myositis is the most frequent Myositis above fifty years of age. Diagnosing inclusion body Myositis requires expertise and is challenging. Little is known concerning the pathogenic mechanisms of this disease in which conventional suppressive-immune therapies are inefficacious. Objectives: Our aim was to deepen our understanding of the immune mechanisms involved in inclusion body Myositis and identify specific biomarkers. Methods: Using a panel of thirty-six markers and mass cytometry, we performed deep immune profiling of peripheral blood cells from inclusion body Myositis patients and healthy donors, divided into two cohorts: test and validation cohorts. Potential biomarkers were compared to Myositis controls (anti-Jo1-, anti-3-hydroxyl-3-methylglutaryl CoA reductase-, and anti-signal recognition particle-positive patients). Results: Unsupervised analyses revealed substantial changes only within CD8+ cells. We observed an increase in the frequency of CD8+ cells that expressed high levels of T-bet, and containing mainly both effector and terminally differentiated memory cells. The senescent marker CD57 was overexpressed in CD8+T-bet+ cells of inclusion body Myositis patients. As expected, senescent CD8+T-bet+ CD57+ cells of both patients and healthy donors were CD28nullCD27nullCD127null. Surprisingly, non-senescent CD8+T-bet+ CD57- cells in inclusion body Myositis patients expressed lower levels of CD28, CD27, and CD127, and expressed higher levels of CD38 and HLA-DR compared to healthy donors. Using classification and regression trees alongside receiver operating characteristics curves, we identified and validated a frequency of CD8+T-bet+ cells >51.5% as a diagnostic biomarker specific to inclusion body Myositis, compared to Myositis control patients, with a sensitivity of 94.4%, a specificity of 88.5%, and an area under the curve of 0.97. Conclusion: Using a panel of thirty-six markers by mass cytometry, we identify an activated cell population (CD8+T-bet+ CD57- CD28lowCD27lowCD127low CD38+ HLA-DR+) which could play a role in the physiopathology of inclusion body Myositis, and identify CD8+T-bet+ cells as a predominant biomarker of this disease.
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DISTAL MUSCLE INVOLVEMENT IN GRANULOMATOUS Myositis CAN MIMIC INCLUSION BODY Myositis
Journal of Neurology Neurosurgery and Psychiatry, 2010Co-Authors: Sandrine Larue, Olivier Benveniste, Thierry Maisonobe, Catherine Chapelon-abric, Olivier Lidove, Thomas Papo, Bruno Eymard, Odile DubourgAbstract:We report on four aged patients with chronic myopathy suggestive of sporadic inclusion body Myositis. They presented progressive and selective weakness of the quadriceps femoris muscles. Asymmetric and selective atrophy of the forearm muscles was noted in all, with more severe involvement of the flexors than the extensors. Biopsy revealed granulomatous Myositis. Histological features of s-IBM were lacking. Evidence for systemic sarcoidosis was found in one patient. Corticosteroids treatment was associated with partial but significant improvement in two patients. Granulomatous Myositis may mimick inclusion body Myositis and may be steroids responsive.
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Distal muscle involvement in granulomatous Myositis can mimic inclusion body Myositis
Journal of neurology neurosurgery and psychiatry, 2010Co-Authors: Sandrine Larue, Olivier Benveniste, Thierry Maisonobe, Catherine Chapelon-abric, Olivier Lidove, Thomas Papo, Bruno Eymard, Odile DubourgAbstract:The authors report on four patients aged over 50 with chronic myopathy suggestive of sporadic inclusion body Myositis. They present progressive and selective weakness of the quadriceps femoris muscles. Asymmetrical and selective atrophy of the forearm muscles were noted in all, with more severe involvement of the flexors than the extensors. Biopsy revealed granulomatous Myositis. Histological features of sporadic inclusion body Myositis were lacking. Evidence for systemic sarcoidosis was found in one patient. Corticosteroid treatment was associated with a partial but significant improvement in two patients. Granulomatous Myositis may mimic inclusion body Myositis and may be steroid-responsive.
Kerstin Pullmann - One of the best experts on this subject based on the ideXlab platform.
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human invasive muscular sarcocystosis induces th2 cytokine polarization and biphasic cytokine changes based on an investigation among travelers returning from tioman island malaysia
Clinical and Vaccine Immunology, 2015Co-Authors: Dennis Tappe, Gunther Slesak, Jose Vicente Perezgiron, Johannes Schafer, Andreas Langeheinecke, Gudrun Justnubling, Cesar Munozfontela, Kerstin PullmannAbstract:Sarcocystis nesbitti is a parasite responsible for a biphasic eosinophilic febrile Myositis syndrome in two recent outbreaks in Malaysia. We demonstrate Th2 cytokine polarization in infected travelers, an overall cytokine production decrease in the early phase of the disease suggestive of initial immunosuppression, and elevated levels of proinflammatory and chemotactic cytokines in the later myositic phase.
Cesar Munozfontela - One of the best experts on this subject based on the ideXlab platform.
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human invasive muscular sarcocystosis induces th2 cytokine polarization and biphasic cytokine changes based on an investigation among travelers returning from tioman island malaysia
Clinical and Vaccine Immunology, 2015Co-Authors: Dennis Tappe, Gunther Slesak, Jose Vicente Perezgiron, Johannes Schafer, Andreas Langeheinecke, Gudrun Justnubling, Cesar Munozfontela, Kerstin PullmannAbstract:Sarcocystis nesbitti is a parasite responsible for a biphasic eosinophilic febrile Myositis syndrome in two recent outbreaks in Malaysia. We demonstrate Th2 cytokine polarization in infected travelers, an overall cytokine production decrease in the early phase of the disease suggestive of initial immunosuppression, and elevated levels of proinflammatory and chemotactic cytokines in the later myositic phase.
Kuberaka Mariampillai - One of the best experts on this subject based on the ideXlab platform.
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granulomatosis associated Myositis high prevalence of sporadic inclusion body Myositis
Neurology, 2020Co-Authors: Yannick Dieudonne, Olivier Benveniste, Yves Allenbach, Kuberaka Mariampillai, Benoit Nespola, Sarah Leonardlouis, B Hervier, Beatrice Lannes, Andoni Echanizlaguna, Daniel WendlingAbstract:Objective To refine the predictive significance of muscle granuloma in patients with Myositis. Methods A group of 23 patients with Myositis and granuloma on muscle biopsy (granuloma-Myositis) from 8 French and Belgian centers was analyzed and compared with (1) a group of 23 patients with Myositis without identified granuloma (control-Myositis) randomly sampled in each center and (2) a group of 20 patients with sporadic inclusion body Myositis (sIBM) without identified granuloma (control-sIBM). Results All but 2 patients with granuloma-Myositis had extramuscular involvement, including signs common in sarcoidosis that were systematically absent in the control-Myositis and the control-sIBM groups. Almost half of patients with granuloma-Myositis matched the diagnostic criteria for sIBM. In these patients, other than the granuloma, the characteristics of the myopathy and its nonresponse to treatment were similar to the control-sIBM patients. Aside from 1 patient with Myositis overlapping with systemic sclerosis, the remaining patients with granuloma-Myositis did not match the criteria for a well-defined Myositis subtype, suggesting pure sarcoidosis. Matching criteria for sIBM was the sole feature independently associated with nonresponse to myopathy treatment in patients with granuloma-Myositis. Conclusion Patients with granuloma-Myositis should be carefully screened for sIBM associated with sarcoidosis in order to best tailor their care.
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CD8+T-bet+ cells as a predominant biomarker for inclusion body Myositis
Autoimmunity Reviews, 2019Co-Authors: Gaëlle Dzangué-tchoupou, Yves Allenbach, Kuberaka Mariampillai, Loïs Bolko, Damien Amelin, Wladimir Mauhin, Aurélien Corneau, Catherine Blanc, Olivier BenvenisteAbstract:Background: Myositis is a heterogeneous group of muscular auto-immune diseases with clinical and pathological criteria that allow the classification of patients into different sub-groups. Inclusion body Myositis is the most frequent Myositis above fifty years of age. Diagnosing inclusion body Myositis requires expertise and is challenging. Little is known concerning the pathogenic mechanisms of this disease in which conventional suppressive-immune therapies are inefficacious. Objectives: Our aim was to deepen our understanding of the immune mechanisms involved in inclusion body Myositis and identify specific biomarkers. Methods: Using a panel of thirty-six markers and mass cytometry, we performed deep immune profiling of peripheral blood cells from inclusion body Myositis patients and healthy donors, divided into two cohorts: test and validation cohorts. Potential biomarkers were compared to Myositis controls (anti-Jo1-, anti-3-hydroxyl-3-methylglutaryl CoA reductase-, and anti-signal recognition particle-positive patients). Results: Unsupervised analyses revealed substantial changes only within CD8+ cells. We observed an increase in the frequency of CD8+ cells that expressed high levels of T-bet, and containing mainly both effector and terminally differentiated memory cells. The senescent marker CD57 was overexpressed in CD8+T-bet+ cells of inclusion body Myositis patients. As expected, senescent CD8+T-bet+ CD57+ cells of both patients and healthy donors were CD28nullCD27nullCD127null. Surprisingly, non-senescent CD8+T-bet+ CD57- cells in inclusion body Myositis patients expressed lower levels of CD28, CD27, and CD127, and expressed higher levels of CD38 and HLA-DR compared to healthy donors. Using classification and regression trees alongside receiver operating characteristics curves, we identified and validated a frequency of CD8+T-bet+ cells >51.5% as a diagnostic biomarker specific to inclusion body Myositis, compared to Myositis control patients, with a sensitivity of 94.4%, a specificity of 88.5%, and an area under the curve of 0.97. Conclusion: Using a panel of thirty-six markers by mass cytometry, we identify an activated cell population (CD8+T-bet+ CD57- CD28lowCD27lowCD127low CD38+ HLA-DR+) which could play a role in the physiopathology of inclusion body Myositis, and identify CD8+T-bet+ cells as a predominant biomarker of this disease.
Yves Allenbach - One of the best experts on this subject based on the ideXlab platform.
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granulomatosis associated Myositis high prevalence of sporadic inclusion body Myositis
Neurology, 2020Co-Authors: Yannick Dieudonne, Olivier Benveniste, Yves Allenbach, Kuberaka Mariampillai, Benoit Nespola, Sarah Leonardlouis, B Hervier, Beatrice Lannes, Andoni Echanizlaguna, Daniel WendlingAbstract:Objective To refine the predictive significance of muscle granuloma in patients with Myositis. Methods A group of 23 patients with Myositis and granuloma on muscle biopsy (granuloma-Myositis) from 8 French and Belgian centers was analyzed and compared with (1) a group of 23 patients with Myositis without identified granuloma (control-Myositis) randomly sampled in each center and (2) a group of 20 patients with sporadic inclusion body Myositis (sIBM) without identified granuloma (control-sIBM). Results All but 2 patients with granuloma-Myositis had extramuscular involvement, including signs common in sarcoidosis that were systematically absent in the control-Myositis and the control-sIBM groups. Almost half of patients with granuloma-Myositis matched the diagnostic criteria for sIBM. In these patients, other than the granuloma, the characteristics of the myopathy and its nonresponse to treatment were similar to the control-sIBM patients. Aside from 1 patient with Myositis overlapping with systemic sclerosis, the remaining patients with granuloma-Myositis did not match the criteria for a well-defined Myositis subtype, suggesting pure sarcoidosis. Matching criteria for sIBM was the sole feature independently associated with nonresponse to myopathy treatment in patients with granuloma-Myositis. Conclusion Patients with granuloma-Myositis should be carefully screened for sIBM associated with sarcoidosis in order to best tailor their care.
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CD8+T-bet+ cells as a predominant biomarker for inclusion body Myositis
Autoimmunity Reviews, 2019Co-Authors: Gaëlle Dzangué-tchoupou, Yves Allenbach, Kuberaka Mariampillai, Loïs Bolko, Damien Amelin, Wladimir Mauhin, Aurélien Corneau, Catherine Blanc, Olivier BenvenisteAbstract:Background: Myositis is a heterogeneous group of muscular auto-immune diseases with clinical and pathological criteria that allow the classification of patients into different sub-groups. Inclusion body Myositis is the most frequent Myositis above fifty years of age. Diagnosing inclusion body Myositis requires expertise and is challenging. Little is known concerning the pathogenic mechanisms of this disease in which conventional suppressive-immune therapies are inefficacious. Objectives: Our aim was to deepen our understanding of the immune mechanisms involved in inclusion body Myositis and identify specific biomarkers. Methods: Using a panel of thirty-six markers and mass cytometry, we performed deep immune profiling of peripheral blood cells from inclusion body Myositis patients and healthy donors, divided into two cohorts: test and validation cohorts. Potential biomarkers were compared to Myositis controls (anti-Jo1-, anti-3-hydroxyl-3-methylglutaryl CoA reductase-, and anti-signal recognition particle-positive patients). Results: Unsupervised analyses revealed substantial changes only within CD8+ cells. We observed an increase in the frequency of CD8+ cells that expressed high levels of T-bet, and containing mainly both effector and terminally differentiated memory cells. The senescent marker CD57 was overexpressed in CD8+T-bet+ cells of inclusion body Myositis patients. As expected, senescent CD8+T-bet+ CD57+ cells of both patients and healthy donors were CD28nullCD27nullCD127null. Surprisingly, non-senescent CD8+T-bet+ CD57- cells in inclusion body Myositis patients expressed lower levels of CD28, CD27, and CD127, and expressed higher levels of CD38 and HLA-DR compared to healthy donors. Using classification and regression trees alongside receiver operating characteristics curves, we identified and validated a frequency of CD8+T-bet+ cells >51.5% as a diagnostic biomarker specific to inclusion body Myositis, compared to Myositis control patients, with a sensitivity of 94.4%, a specificity of 88.5%, and an area under the curve of 0.97. Conclusion: Using a panel of thirty-six markers by mass cytometry, we identify an activated cell population (CD8+T-bet+ CD57- CD28lowCD27lowCD127low CD38+ HLA-DR+) which could play a role in the physiopathology of inclusion body Myositis, and identify CD8+T-bet+ cells as a predominant biomarker of this disease.
