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Artikel ; Online: Susceptibility to mycobacterial infection in VEXAS syndrome.

Riescher, Stanislas / Lecomte, Raphael / Danic, Gwenvael / Graveleau, Julie / Le Bris, Yannick / Hello, Muriel / Guillouzouic, Aurélie / Guardiolle, Vianney / Garnier, Alice / Grossi, Olivier / Gaborit, Benjamin / Néel, Antoine

Rheumatology (Oxford, England)

2024

Abstract: Objectives: VEXAS is a recently described acquired auto-inflammatory and hematologic syndrome caused by somatic mutations in UBA1. To date, VEXAS is not a recognized cause of acquired immunodeficiency.: Patients and methods: Two of our 10 VEXAS ... ...

Abstract	Objectives: VEXAS is a recently described acquired auto-inflammatory and hematologic syndrome caused by somatic mutations in UBA1. To date, VEXAS is not a recognized cause of acquired immunodeficiency. Patients and methods: Two of our 10 VEXAS patients developed a disseminated Mycobacterium avium infection. To shed light on this observation, we retrospectively studied all patients with disseminated non-tuberculous mycobacterial infections (NTMi) seen at our institution over 13 years. Inclusion criteria were a positive blood/bone marrow culture, or 2 positive cultures from distinct sites, or one positive culture with 2 involved sites. Results: patient 1 presented with fever, rash, orbital cellulitis and lung infiltrates. Patient 2 presented with fever and purpura. In both cases, Mycobacterium avium was identified on bone marrow culture. Twenty cases of disseminated NTMi were reviewed. Among 11 HIV-negative patients, three had chronic immune-mediated disease; three had untreated myeloid neoplasm; two had VEXAS; one had undergone kidney transplantation; one had GATA-2 deficiency; and one had no identified aetiology. None had lymphoid neoplasia or had undergone bone marrow transplantation. HIV-negative cases had higher CD4 counts than HIV-positive patients (median CD4: 515/mm3 vs 38/mm3, p< 0.001). Monocytopenia was present in seven cases. At 2 years, six patients had died, including both VEXAS patients. Discussion: VEXAS patients have an intrinsic susceptibility to disseminated NTMi, which may result from monocytic dysfunction. NTMi can mimic VEXAS flare. Clinicians should maintain a high suspicion for opportunistic infections before escalating immunosuppressive therapy. Further studies are needed to confirm and better decipher the herein reported observations.
Sprache	Englisch
Erscheinungsdatum	2024-02-05
Erscheinungsland	England
Dokumenttyp	Journal Article
ZDB-ID	1464822-2
ISSN	1462-0332 ; 1462-0324
ISSN (online)	1462-0332
ISSN	1462-0324
DOI	10.1093/rheumatology/keae087
Datenquelle	MEDical Literature Analysis and Retrieval System OnLINE

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Artikel ; Online: Clinical, Radiologic, and Immunologic Features of Patients With CTLA4 Deficiency With Neurologic Involvement.

Neurology

2023 Band 101, Heft 15, Seite(n) e1560–e1566

Abstract: Objectives: CTLA4 deficiency (CTLA4d) is a disease with multisystem autoimmune features, including neurologic manifestations. We aimed to describe neurologic involvement in these patients.: Methods: We performed a cross-sectional observational study ... ...

Abstract	Objectives: CTLA4 deficiency (CTLA4d) is a disease with multisystem autoimmune features, including neurologic manifestations. We aimed to describe neurologic involvement in these patients. Methods: We performed a cross-sectional observational study using the French Reference Centre for Primary Immunodeficiencies (CEREDIH) registry plus a surveillance in national society networks. Participants with confirmed CTLA4d and neurologic involvement were included. Clinical, laboratory, and radiologic features were collected, as well as treatments. Available MRI was double-reviewed. Results: Among 70 patients with CTLA4d, 13 patients (21%) had neurologic involvement. Neurologic symptoms began at a median age of 18 [15-45] years, mostly occurring after systemic manifestations (median delay: 8.5 [4.5-10.5] years). Main symptoms included headaches, focal deficit (54% each), and seizures (38%). MRI detected at least 1 large contrast-enhancing lesion in 8 patients. Lesions reminiscent of multiple sclerosis lesions were found in 6 patients. Cerebellar (6 patients) and large spinal cord lesions (3 patients) were common. Ten patients were treated with abatacept, of whom 9 (90%) showed good clinical and radiologic response. Discussion: Neurologic involvement is common among patients with CTLA4d. Despite its rarity, and considering the suspected efficacy of abatacept, neurologists should be aware of the characteristics of CTLA4d neurologic involvement.
Mesh-Begriff(e)	Humans ; Adolescent ; Young Adult ; Adult ; Middle Aged ; CTLA-4 Antigen/genetics ; Abatacept/therapeutic use ; Cross-Sectional Studies ; Spinal Cord Diseases ; Multiple Sclerosis
Chemische Substanzen	CTLA-4 Antigen ; Abatacept (7D0YB67S97) ; CTLA4 protein, human
Sprache	Englisch
Erscheinungsdatum	2023-07-24
Erscheinungsland	United States
Dokumenttyp	Observational Study ; Journal Article
ZDB-ID	207147-2
ISSN	1526-632X ; 0028-3878
ISSN (online)	1526-632X
ISSN	0028-3878
DOI	10.1212/WNL.0000000000207609
Datenquelle	MEDical Literature Analysis and Retrieval System OnLINE

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Artikel ; Online: Characteristics and outcome of adults with severe autoimmune hemolytic anemia admitted to the intensive care unit: Results from a large French observational study.

Pouchelon, Clara / Lafont, Charlotte / Lafarge, Antoine / Comont, Thibault / Riviere, Etienne / Boutboul, David / Fieschi, Claire / Dossier, Antoine / Audia, Sylvain / Vaidie, Julien / Ruivard, Marc / Gobert, Delphine / Bonnard, Guillaume / Graveleau, Julie / Mahevas, Matthieu / Godeau, Bertrand / Michel, Marc

American journal of hematology

2022 Band 97, Heft 10, Seite(n) E371–E373

Mesh-Begriff(e)	Adult ; Anemia, Hemolytic ; Anemia, Hemolytic, Autoimmune/therapy ; Hospitalization ; Humans ; Intensive Care Units
Sprache	Englisch
Erscheinungsdatum	2022-08-02
Erscheinungsland	United States
Dokumenttyp	Letter ; Observational Study
ZDB-ID	196767-8
ISSN	1096-8652 ; 0361-8609
ISSN (online)	1096-8652
ISSN	0361-8609
DOI	10.1002/ajh.26665
Datenquelle	MEDical Literature Analysis and Retrieval System OnLINE

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Artikel ; Online: Deleterious neurological impact of diagnostic delay in immune-mediated thrombotic thrombocytopenic purpura.

Renaud, Arthur / Caristan, Aurélie / Seguin, Amélie / Agard, Christian / Blonz, Gauthier / Canet, Emmanuel / Eveillard, Marion / Godmer, Pascal / Graveleau, Julie / Lecouffe-Desprets, Marie / Maisonneuve, Hervé / Perrin, François / Hamidou, Mohamed / Néel, Antoine

PloS one

2021 Band 16, Heft 11, Seite(n) e0260196

Abstract: Background: Immune-mediated thrombotic thrombocytopenic purpura (iTTP) is a rare life-threatening thrombotic microangiopathy requiring urgent therapeutic plasma exchange (TPE). However, the exact impact of a slight delay in TPE initiation on the ... ...

Abstract	Background: Immune-mediated thrombotic thrombocytopenic purpura (iTTP) is a rare life-threatening thrombotic microangiopathy requiring urgent therapeutic plasma exchange (TPE). However, the exact impact of a slight delay in TPE initiation on the subsequent patients' outcome is still controversial. Aim: We aimed to study the frequency, short-term neurological consequences, and determinants of diagnostic delay in iTTP. Methods: We conducted a retrospective monocentric study including patients with a first acute episode of iTTP (2005-2020) classified into 2 groups: delayed (>24h from first hospital visit, group 1) and immediate diagnosis (≤24h, group 2). Results: Among 42 evaluated patients, 38 were included. Eighteen cases (47%) had a delayed diagnosis (median: 5 days). The main misdiagnosis was immune thrombocytopenia (67%). The mortality rate was 5% (1 death in each group). Neurological events (stroke/TIA, seizure, altered mental status) occurred in 67% vs 30% patients in group 1 and 2, respectively (p = 0.04). Two patients in group 1 exhibited neurological sequelae. The hospital length of stay was longer in group 1 (p = 0.02). At the first hospital evaluation, potential alternative causes of thrombocytopenia were more prevalent in group 1 (33% vs 5%, p = 0.04). Anemia was less frequent in group 1 (67% vs 95%, p = 0.04). All patients had undetectable haptoglobin levels. By contrast, 26% of schistocytes counts were <1%, mostly in group 1 (62% vs 11%, p = 0.01). Conclusion: Diagnostic delay is highly prevalent in iTTP, with a significant impact on short-term neurological outcome. In patients with profound thrombocytopenia, the thorough search for signs of incipient organ dysfunction, systematic hemolysis workup, and proper interpretation of schistocytes count are the key elements of early diagnosis of TTP.
Mesh-Begriff(e)	Adolescent ; Adult ; Aged ; Anemia, Sickle Cell/diagnosis ; Delayed Diagnosis ; Female ; Humans ; Length of Stay ; Male ; Middle Aged ; Nervous System Diseases/diagnosis ; Purpura, Thrombocytopenic, Idiopathic/diagnosis ; Purpura, Thrombotic Thrombocytopenic/diagnosis ; Retrospective Studies ; Young Adult
Sprache	Englisch
Erscheinungsdatum	2021-11-19
Erscheinungsland	United States
Dokumenttyp	Journal Article
ISSN	1932-6203
ISSN (online)	1932-6203
DOI	10.1371/journal.pone.0260196
Datenquelle	MEDical Literature Analysis and Retrieval System OnLINE

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Artikel ; Online: Disease patterns and specific trajectories of anti-MDA5-related disease: a multicentre retrospective study of 70 adult patients.

de Boysson, Hubert / Cuchet, Marie / Cassius, Charles / Cuchet, Pierre / Agard, Christian / Audemard-Verger, Alexandra / Marchand-Adam, Sylvain / Cohen-Sors, Raphaëlla / Gallay, Laure / Graveleau, Julie / Lesort, Cécile / Ly, Kim / Meyer, Alain / Monseau, Grégoire / Néel, Antoine / Bonnotte, Bernard / Pérard, Laurent / Schleinitz, Nicolas / Mariotte, Delphine /

Le Mauff, Brigitte / Bourdenet, Gwladys / Masmoudi, Wafa / Deshayes, Samuel / Dumont, Anaël / Dompmartin, Anne / Kottler, Diane / Aouba, Achille

Frontiers in immunology

2024 Band 14, Seite(n) 1319957

Abstract: Introduction: This study aimed to provide an updated analysis of the different prognostic trajectories of patients with anti-melanoma differentiation-associated gene 5 (MDA5) antibodies.: Methods: Among a cohort of 70 patients, baseline ... ...

Abstract	Introduction: This study aimed to provide an updated analysis of the different prognostic trajectories of patients with anti-melanoma differentiation-associated gene 5 (MDA5) antibodies. Methods: Among a cohort of 70 patients, baseline characteristics and phenotypes, treatments and outcomes were analyzed. A Cox proportional hazards model was used to identify factors associated with poor outcomes, i.e., death or progressive disease at the last follow-up. Results: Among the 70 patients, 45 were women, and 54 were Caucasian. A dermatologic involvement was observed in 58 (83%) patients, including 40 with MDA5 vasculopathy-related skin lesions. Muscular involvement was observed in 39 (56%) patients. Interstitial lung disease (ILD) was observed at baseline in 52 (74%) patients, including 23 (44%) who developed rapidly progressive (RP) ILD. Seven (10%) patients showed thromboembolic complications within the first weeks of diagnosis, and eight (11%) other patients developed a malignancy (4 before the diagnosis of anti-MDA5 disease). Poor outcomes were observed in 28 (40%) patients, including 13 (19%) deaths. Among the 23 patients with RP-ILD, 19 (79%) showed poor outcomes, including 12 (63%) who died. In multivariate analyses, RP-ILD (hazard ratio (HR), 95% CI: 8.24 [3.21-22], p<0.0001), the occurrence of thromboembolic events (HR: 5.22 [1.61-14.77], p=0.008) and the presence of any malignancy (HR: 19.73 [6.67-60], p<0.0001) were the three factors independently associated with poor outcomes. Discussion: This new independent cohort confirms the presence of different clinical phenotypes of anti-MDA5 diseases at baseline and the poor prognosis associated with RP-ILD. Thromboembolic events and malignancies were also identified as prognostic factors.
Mesh-Begriff(e)	Adult ; Humans ; Female ; Male ; Retrospective Studies ; Multivariate Analysis ; Thromboembolism ; Lung Diseases, Interstitial/etiology ; Neoplasms
Sprache	Englisch
Erscheinungsdatum	2024-01-08
Erscheinungsland	Switzerland
Dokumenttyp	Multicenter Study ; Journal Article ; Comment
ZDB-ID	2606827-8
ISSN	1664-3224 ; 1664-3224
ISSN (online)	1664-3224
ISSN	1664-3224
DOI	10.3389/fimmu.2023.1319957
Datenquelle	MEDical Literature Analysis and Retrieval System OnLINE

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Artikel ; Online: Gamma heavy chain disease associated with large granular lymphocytic leukemia: A report of two cases and review of the literature.

Wahbi, Anais / Neel, Antoine / Perrin, Francois / Graveleau, Julie / Mahe, Beatrice / Dejoie, Thomas / Hamidou, Mohamed

Hematology (Amsterdam, Netherlands)

2016 Band 21, Heft 2, Seite(n) 92–94

Abstract: Objective and importance: Gamma heavy chain diseases (γHCD) and large granular lymphocyte (LGL) leukemia are two rare lymphoproliferative diseases, respectively with B and T phenotype. Both γHCD and LGL leukemia share some similar clinical features, ... ...

Abstract	Objective and importance: Gamma heavy chain diseases (γHCD) and large granular lymphocyte (LGL) leukemia are two rare lymphoproliferative diseases, respectively with B and T phenotype. Both γHCD and LGL leukemia share some similar clinical features, such as cytopenias, splenomegaly, and recurrent infections. Association of these two diseases is exceptional and suggest pathogenic link. We report two cases of γHCD associated with T-LGL leukemia. Clinical presentation: Patient 1 was a 70-year-old woman, with lymphoplasmacytic lymphoma, refractory to chlorambucil-rituximab treatment. She developed during the follow up a γHCD with T-LGL leukemia, unresponsive to melphalan, thalidomide, and steroids, requiring supportive care. Patient 2 was a 40-year-old man with chronic severe asymptomatic neutropenia, revealing both γHCD and T-LGL leukemia. He is still well without any treatment nor complications, with 7 years follow up. Conclusion: Several types of B lymphoproliferative disease are associated with LGL leukemia. Although exceptional, this association of two rare lymphoproliferative disorders, with a different phenotype, does not seem fortuitous.
Mesh-Begriff(e)	Adult ; Aged ; Female ; Heavy Chain Disease/complications ; Heavy Chain Disease/drug therapy ; Humans ; Leukemia, Lymphoid/complications ; Leukemia, Lymphoid/drug therapy ; Male
Sprache	Englisch
Erscheinungsdatum	2016-03
Erscheinungsland	England
Dokumenttyp	Case Reports ; Journal Article ; Review
ZDB-ID	1341428-8
ISSN	1607-8454 ; 1024-5332 ; 1024-5340
ISSN (online)	1607-8454
ISSN	1024-5332 ; 1024-5340
DOI	10.1179/1607845415Y.0000000037
Datenquelle	MEDical Literature Analysis and Retrieval System OnLINE

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Artikel ; Online: Muscle biopsy in anti-neutrophil cytoplasmic antibody-associated vasculitis: diagnostic yield depends on anti-neutrophil cytoplasmic antibody type, sex and neutrophil count.

Lacou, Mathieu / Leroy, Maxime / Le Lan, Nowenn / Toquet, Claire / Espitia-Thibault, Alexandra / Graveleau, Julie / Masseau, Agathe / Agard, Christian / Volteau, Christelle / Mussini, Jean-Marie / Hamidou, Mohamed / Néel, Antoine

Rheumatology (Oxford, England)

2020 Band 60, Heft 2, Seite(n) 699–707

Abstract: Objectives: This study aimed to examine the sensitivity of muscle biopsy (MB) in ANCA-associated vasculitis (AAV), identify factors predicting MB positivity and assess the prognostic value of a positive MB.: Methods: We conducted a single-centre ... ...

Abstract	Objectives: This study aimed to examine the sensitivity of muscle biopsy (MB) in ANCA-associated vasculitis (AAV), identify factors predicting MB positivity and assess the prognostic value of a positive MB. Methods: We conducted a single-centre retrospective study of AAV with an MB performed at diagnosis. AAV classification [granulomatosis with polyangiitis (GPA), microscopic polyangiitis (MPA), eosinophilic granulomatosis with polyangiitis (EGPA)] followed the European Medicines Agency algorithm. A logistic regression model was used to identify the factors associated with MB positivity. Survival curves were generated using the Kaplan-Meier method. Results: Among 276 AAV patients (1995-2018), 101 had an MB. Seventy-eight patients were included: 33 with GPA, 25 with MPA and 20 with EGPA. MB samples were positive in 45 cases (58%): 17 GPA, 16 MPA and 12 EGPA. Univariate analysis focussed on GPA and MPA, revealed that the MB yield was higher in females [22/31 (71%) vs 11/27 (41%); P = 0.02] and in anti-MPO patients [25/37 (68%) vs 6/19 (32%) for anti-PR3; P = 0.01]. By multivariate analysis, three factors predicted MB positivity: anti-MPO ANCA [odds ratio (OR) 10.67 (CI 2.09, 81.68)], female sex [OR 5.3 (CI 1.16, 32.35)] and neutrophil count [OR 1.33 (CI 1.07, 1.8)]. MB positivity had no impact on relapse, death or end-stage renal disease-free survival. Conclusions: MB is a safe and efficient diagnostic tool for AAV. Predictors of MB yield include ANCA type, sex and neutrophil count. MB cannot substitute for kidney biopsy when indicated, but should be considered in other cases.
Mesh-Begriff(e)	Aged ; Algorithms ; Anti-Neutrophil Cytoplasmic Antibody-Associated Vasculitis/diagnosis ; Anti-Neutrophil Cytoplasmic Antibody-Associated Vasculitis/epidemiology ; Anti-Neutrophil Cytoplasmic Antibody-Associated Vasculitis/immunology ; Antibodies, Antineutrophil Cytoplasmic/immunology ; Biopsy/methods ; Female ; France/epidemiology ; Humans ; Incidence ; Leukocyte Count ; Male ; Middle Aged ; Muscle, Skeletal/immunology ; Muscle, Skeletal/metabolism ; Muscle, Skeletal/pathology ; Neutrophils/immunology ; Neutrophils/pathology ; Prognosis ; Recurrence ; Retrospective Studies ; Sex Factors
Chemische Substanzen	Antibodies, Antineutrophil Cytoplasmic
Sprache	Englisch
Erscheinungsdatum	2020-08-12
Erscheinungsland	England
Dokumenttyp	Journal Article
ZDB-ID	1464822-2
ISSN	1462-0332 ; 1462-0324
ISSN (online)	1462-0332
ISSN	1462-0324
DOI	10.1093/rheumatology/keaa233
Datenquelle	MEDical Literature Analysis and Retrieval System OnLINE

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Artikel: Large-vessel involvement is predictive of multiple relapses in giant cell arteritis.

de Mornac, Donatienne / Espitia, Olivier / Néel, Antoine / Connault, Jérôme / Masseau, Agathe / Espitia-Thibault, Alexandra / Artifoni, Mathieu / Achille, Aurélie / Wahbi, Anaïs / Lacou, Mathieu / Durant, Cécile / Pottier, Pierre / Perrin, François / Graveleau, Julie / Hamidou, Mohamed / Hardouin, Jean-Benoit / Agard, Christian

Therapeutic advances in musculoskeletal disease

2021 Band 13, Seite(n) 1759720X211009029

Abstract: Background: Giant cell arteritis (GCA) is the most common systemic vasculitis. Relapses are frequent. The aim of this study was to identify relapse risk factors in patients with GCA with complete large-vessel imaging at diagnosis.: Methods: Patients ... ...

Abstract	Background: Giant cell arteritis (GCA) is the most common systemic vasculitis. Relapses are frequent. The aim of this study was to identify relapse risk factors in patients with GCA with complete large-vessel imaging at diagnosis. Methods: Patients with GCA followed in our institution between April 1998 and April 2018 were included retrospectively. We included only patients who had undergone large vascular imaging investigations at diagnosis by computed tomography (CT)-scan and/or positron emission tomography (PET)-scan and/or angio-magnetic resonance imaging (MRI). Clinical, biological, and radiological data were collected. Relapse was defined as the reappearance of GCA symptoms, with concomitant increase in inflammatory markers, requiring treatment adjustment. Relapsing patients (R) and non-relapsing patients (NR) were compared. Relapse and multiple relapses (>2) risk factors were identified in multivariable Cox analyses. Results: This study included 254 patients (73.2% women), with a median age of 72 years at diagnosis and a median follow up of 32.5 months. At diagnosis, 160 patients (63%) had an inflammatory large-vessel involvement on imaging, 46.1% (117 patients) relapsed at least once, and 21.3% (54 patients) had multiple relapses. The median delay of first relapse after diagnosis was 9 months. The second relapse delay was 21.5 months. NR patients had more stroke at diagnosis than R ( Conclusion: Male gender was a protective factor for GCA relapse and peripheral musculoskeletal manifestations appeared as a relapsing risk factor. Moreover, this study identified a particular clinical phenotype of multi-relapsing patients with GCA, characterized by peripheral musculoskeletal manifestations, negative temporal artery biopsy, and large-vessel involvement with upper limb ischemia or inflammation of arm arteries. Plain language summary: At giant cell arteritis diagnosis, large-vessel inflammatory involvement is predictive of multiple relapses
Sprache	Englisch
Erscheinungsdatum	2021-05-18
Erscheinungsland	England
Dokumenttyp	Journal Article
ZDB-ID	2516075-8
ISSN	1759-7218 ; 1759-720X
ISSN (online)	1759-7218
ISSN	1759-720X
DOI	10.1177/1759720X211009029
Datenquelle	MEDical Literature Analysis and Retrieval System OnLINE

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Artikel: Risk factors for symptomatic vascular events in giant cell arteritis: a study of 254 patients with large-vessel imaging at diagnosis.

de Mornac, Donatienne / Agard, Christian / Hardouin, Jean-Benoit / Hamidou, Mohamed / Connault, Jérôme / Masseau, Agathe / Espitia-Thibault, Alexandra / Artifoni, Mathieu / Ngohou, Chan / Perrin, François / Graveleau, Julie / Durant, Cécile / Pottier, Pierre / Néel, Antoine / Espitia, Olivier

Therapeutic advances in musculoskeletal disease

2021 Band 13, Seite(n) 1759720X211006967

Abstract: Aims: To identify factors associated with vascular events in patients with giant cell arteritis (GCA).: Methods: We performed a retrospective study of GCA patients diagnosed over a 20-year-period, who all underwent vascular imaging evaluation at ... ...

Abstract	Aims: To identify factors associated with vascular events in patients with giant cell arteritis (GCA). Methods: We performed a retrospective study of GCA patients diagnosed over a 20-year-period, who all underwent vascular imaging evaluation at diagnosis. Symptomatic vascular events were defined as the occurrence of any aortic event (aortic dissection or symptomatic aortic aneurysm), stroke, myocardial infarction, limb or mesenteric ischemia and Results: Thirty-nine (15.4%) of the 254 included patients experienced at least one symptomatic vascular event during follow-up, with a median time of 21.5 months. Arterial hypertension, diabetes, lower limbs arteritis or vascular complication at diagnosis were more frequent in VE+ patients ( Conclusion: In this study on GCA cases with large vessels imaging at diagnosis, aortic surgery, stroke, upper or lower limb ischemia, aortic atheroma and aortitis of the descending thoracic aorta on CT-scan, at GCA diagnosis, were independent predictive factors of a vascular event. Plain language summary: Risk factors for symptomatic vascular events in giant cell arteritisThis study was performed to identify the risk factors for developing symptomatic vascular event during giant cell arteritis (GCA) because these are poorly known.We performed a retrospective study of GCA patients diagnosed over a 20-year-period, who all underwent vascular imaging evaluation at diagnosis.Patients with symptomatic vascular event (VE+) and without (VE-) were compared, and risk factors were identified in a multivariable analysis.Thirty-nine patients experienced at least one symptomatic vascular event during follow-up, with a median time of 21.5 months.Arterial hypertension, diabetes, lower limbs arteritis or vascular complication at diagnosis were significantly more frequent in VE+ patients, as an abnormal CT-scan at diagnosis, aortitis, particularly of the descending thoracic aorta and atheroma. Deaths were more frequent in the VE+ group.Among 254 GCA patients, 39 experienced at least one vascular event during follow-up.Aortic surgery, stroke, upper and lower limb ischemia were vascular event risk factors.Aortic atheroma and descending thoracic aorta aortitis on CT-scan were vascular event risk factors.This study on GCA cases with large vessels imaging at diagnosis, showed that aortic surgery, stroke, upper or lower limb ischemia, aortic atheroma and aortitis of the descending thoracic aorta on CT-scan, at GCA diagnosis, were independent predictive factors of a vascular event.
Sprache	Englisch
Erscheinungsdatum	2021-06-27
Erscheinungsland	England
Dokumenttyp	Journal Article
ZDB-ID	2516075-8
ISSN	1759-7218 ; 1759-720X
ISSN (online)	1759-7218
ISSN	1759-720X
DOI	10.1177/1759720X211006967
Datenquelle	MEDical Literature Analysis and Retrieval System OnLINE

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Artikel ; Online: Thoracic involvement and imaging patterns in IgG4-related disease.

Muller, Romain / Habert, Paul / Ebbo, Mikael / Graveleau, Julie / Groh, Mathieu / Launay, David / Audia, Sylvain / Pugnet, Gregory / Cohen, Fleur / Perlat, Antoinette / Benyamine, Audrey / Bienvenu, Boris / Gaigne, Lea / Chanez, Pascal / Gaubert, Jean Yves / Schleinitz, Nicolas

European respiratory review : an official journal of the European Respiratory Society

2021 Band 30, Heft 162

Abstract: Objective: Immunoglobulin G4-related disease (IgG4-RD) is a rare orphan disease. Lung, pleura, pericardium, mediastinum, aorta and lymph node involvement has been reported with variable frequency and mostly in Asian studies. The objective of this study ... ...

Abstract	Objective: Immunoglobulin G4-related disease (IgG4-RD) is a rare orphan disease. Lung, pleura, pericardium, mediastinum, aorta and lymph node involvement has been reported with variable frequency and mostly in Asian studies. The objective of this study was to describe thoracic involvement assessed by high-resolution thoracic computed tomography (CT) in Caucasian patients with IgG4-RD. Methods: Thoracic CT scans before treatment were retrospectively collected through the French case registry of IgG4-RD and a single tertiary referral centre. CT scans were reviewed by two experts in thoracic imagery blinded from clinical data. Results: 48 IgG4-RD patients with thoracic involvement were analysed. All had American College of Rheumatology/European League Against Rheumatism classification scores ≥20 and comprehensive diagnostic criteria for IgG4-RD. CT scan findings showed heterogeneous lesions. Seven patterns were observed: peribronchovascular involvement (56%), lymph node enlargement (31%), nodular disease (25%), interstitial disease (25%), ground-glass opacities (10%), pleural disease (8%) and retromediastinal fibrosis (4%). In 37% of cases two or more patterns were associated. Asthma was significantly associated with peribronchovascular involvement (p=0.04). Among eight patients evaluated by CT scan before and after treatments, only two patients with interstitial disease displayed no improvement. Conclusion: Thoracic involvement of IgG4-RD is heterogeneous and likely underestimated. The main thoracic CT scan patterns are peribronchovascular thickening and thoracic lymph nodes.
Mesh-Begriff(e)	Humans ; Immunoglobulin G4-Related Disease ; Lung/diagnostic imaging ; Retrospective Studies ; Thorax ; Tomography, X-Ray Computed
Sprache	Englisch
Erscheinungsdatum	2021-10-05
Erscheinungsland	England
Dokumenttyp	Journal Article ; Review
ZDB-ID	1077620-5
ISSN	1600-0617 ; 0905-9180
ISSN (online)	1600-0617
ISSN	0905-9180
DOI	10.1183/16000617.0078-2021
Datenquelle	MEDical Literature Analysis and Retrieval System OnLINE

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