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Article ; Online: Correspondence in reference to the previously published Epub manuscript: immune thrombocytopenic purpura after SARS-CoV-2 vaccine.

Scanvion, Quentin / Lambert, Marc / Hachulla, Eric / Terriou, Louis

2021 Volume 194, Issue 6, Page(s) e93–e95

MeSH term(s)	COVID-19 ; COVID-19 Vaccines ; Humans ; Platelet Factor 4 ; Purpura, Thrombocytopenic, Idiopathic/etiology ; SARS-CoV-2
Chemical Substances	COVID-19 Vaccines ; Platelet Factor 4 (37270-94-3)
Language	English
Publishing date	2021-06-10
Publishing country	England
Document type	Letter ; Comment
ZDB-ID	80077-6
ISSN	1365-2141 ; 0007-1048
ISSN (online)	1365-2141
ISSN	0007-1048
DOI	10.1111/bjh.17628
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Article: Socio-professional impact and quality of life of cryopyrin-associated periodic syndromes in 54 patients in adulthood.

Vasse, Marina / Reumaux, Héloise / Koné-Paut, Isabelle / Quartier, Pierre / Hachulla, Eric

Clinical and experimental rheumatology

2023 Volume 41, Issue 10, Page(s) 2039–2043

Abstract: Objectives: Cryopyrin-associated periodic syndromes (CAPS) belongs to the group of hereditary recurrent fever disorders characterised by interleukin1β-mediated systemic inflammation. Specific treatment by IL-1 targeting drugs has significantly modified ... ...

Abstract	Objectives: Cryopyrin-associated periodic syndromes (CAPS) belongs to the group of hereditary recurrent fever disorders characterised by interleukin1β-mediated systemic inflammation. Specific treatment by IL-1 targeting drugs has significantly modified the disease evolution. We aimed to evaluate the socio-professional impact of CAPS in the long term and the influence of genetic variants in the phenotype. Methods: We made a multicentre, observational and descriptive study and collected retrospective data from childhood to adulthood, and until the last year of follow-up. We assessed the quality of life (QoL) of the patients by phone interviews. We also used the SF36 questionnaire including 8 domains: physical function, physical role, body pain, general health, vitality, social function, emotional role and mental health. A high score means a better QoL. Results: Fifty-four patients were evaluated (14 familial cold autoinflammatory syndrome, 27 Muckle-Wells syndrome, 7 chronic infantileneurological cutaneous and articular syndrome. The study showed improvement in symptoms in adulthood and good QoL in all domains apart from school (87%) and work (61%) absenteeism. The MWS group is intermediate in terms of symptoms but seems to describe a better QoL compared to the other groups. The genetic variant alone does not determine the expression of the disease. Conclusions: Our study shows that CAPS patients have an improvement of symptoms in adulthood and a satisfactory QoL for most of them. Anti-IL1 treatment is the main factor linked to this improvement and therefore early initiation should be encouraged.
MeSH term(s)	Humans ; Child ; Adolescent ; Young Adult ; Cryopyrin-Associated Periodic Syndromes/diagnosis ; Cryopyrin-Associated Periodic Syndromes/drug therapy ; Cryopyrin-Associated Periodic Syndromes/genetics ; Quality of Life ; Retrospective Studies ; Inflammation/genetics ; Skin ; NLR Family, Pyrin Domain-Containing 3 Protein/genetics
Chemical Substances	NLR Family, Pyrin Domain-Containing 3 Protein
Language	English
Publishing date	2023-03-09
Publishing country	Italy
Document type	Journal Article
ZDB-ID	605886-3
ISSN	1593-098X ; 0392-856X
ISSN (online)	1593-098X
ISSN	0392-856X
DOI	10.55563/clinexprheumatol/bfyb6o
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Article ; Online: Osteoporosis is associated with anti-topoisomerase I positivity and glucocorticoids use in patients with systemic sclerosis.

Midol, Charles / Wiebe, Edgar / Siegert, Elise / Huscher, Dörte / Béhal, Hélène / Launay, David / Hachulla, Eric / Matteson, Eric L / Buttgereit, Frank / Sobanski, Vincent

Rheumatology (Oxford, England)

2024

Abstract: Objectives: Patients with systemic sclerosis (SSc) are at increased risk for osteoporosis (OP) and associated fragility fractures. This study aimed to identify underlying risk factors for these conditions in patients with SSc.: Methods: This cross- ... ...

Abstract	Objectives: Patients with systemic sclerosis (SSc) are at increased risk for osteoporosis (OP) and associated fragility fractures. This study aimed to identify underlying risk factors for these conditions in patients with SSc. Methods: This cross-sectional study was based on a large prospective cohort of patients with SSc using retrospectively collected bone health data. OP was defined as the presence of a T-score below -2.5 at the femoral neck or lumbar spine, a previous major osteoporotic fracture, or the prescription of anti-osteoporotic therapy. Results: A total of 485 patients fulfilling the ACR/EULAR 2013 diagnostic criteria for SSc, followed in the Lille University Hospital, were included in the study. The prevalence of OP was 23%; fragility fractures occurred in 18% of patients. OP was associated with higher age, diffuse cutaneous subset, interstitial lung disease (ILD), anti-topoisomerase I positivity, treatment with glucocorticoids (GC) and DMARDs in univariable analysis. Multivariable analysis indicated that higher age (OR 1.06 [95%CI 1.04-1.08]), anti-topoisomerase I antibody positivity (OR 2.22 [1.18-4.16]) and treatment with GC (OR 4.48 [2.42-8.26]) were significantly and independently associated with OP. Conclusion: Our study shows that OP risk in patients with SSc is determined by age, disease-related factors such as diffuse cutaneous subset, ILD and anti-topoisomerase I antibody positivity, but also treatment with GC independently of other factors.
Language	English
Publishing date	2024-03-07
Publishing country	England
Document type	Journal Article
ZDB-ID	1464822-2
ISSN	1462-0332 ; 1462-0324
ISSN (online)	1462-0332
ISSN	1462-0324
DOI	10.1093/rheumatology/keae142
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Article ; Online: Targeting CCR4 with mogamulizumab in refractory CD3-CD4

Ledoult, Emmanuel / Groh, Matthieu / Meresse, Bertrand / Dubois, Romain / Trauet, Jacques / Toussaint, Elise / Delbeke, Marie / Hachulla, Eric / Terriou, Louis / De Masson, Adèle / Vasseur, Michele / Labalette, Myriam / Launay, David / Kahn, Jean-Emmanuel / Lefevre, Guillaume

Haematologica

2024

Abstract: Not available. ...

Abstract	Not available.
Language	English
Publishing date	2024-02-08
Publishing country	Italy
Document type	Journal Article
ZDB-ID	2333-4
ISSN	1592-8721 ; 0017-6567 ; 0390-6078
ISSN (online)	1592-8721
ISSN	0017-6567 ; 0390-6078
DOI	10.3324/haematol.2023.284429
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Article ; Online: Epidemiology of Sjögren Syndrome in Africa: A Scoping Review.

Essouma, Mickael / Noubiap, Jean Jacques / Singwe-Ngandeu, Madeleine / Hachulla, Eric

Journal of clinical rheumatology : practical reports on rheumatic & musculoskeletal diseases

2021 Volume 28, Issue 1, Page(s) e240–e244

Abstract: Background: The epidemiology of Sjögren syndrome (SS) has been extensively studied in America, Europe, and Asia.: Objective: To summarize available data on the epidemiology of SS in Africa.: Methods: MEDLINE, EMBASE, and African Journals Online ... ...

Abstract	Background: The epidemiology of Sjögren syndrome (SS) has been extensively studied in America, Europe, and Asia. Objective: To summarize available data on the epidemiology of SS in Africa. Methods: MEDLINE, EMBASE, and African Journals Online were searched from inception up to May 17, 2020, to identify relevant articles. Data gleaned from these reports have been summarized narratively in this review. Results: Twenty-one hospital-based studies were included. These studies reported 744 cases of SS. The mean age at diagnosis varied between 28 and 73.6 years, and the female proportion ranged from 83.3% to 100%. There was no population-based incidence or prevalence. Among people with autoimmune and other rheumatic conditions, the frequency of primary SS was in the range 1.9% to 47.6%, whereas that of rheumatoid arthritis-associated secondary SS was in the range 4.3% to 100%. Sicca symptoms were the commonest features, with most frequently involved organs being joints, lungs, and neurological structures. Main autoantibodies were anti-Ro/SS antigen A, anti-La/SS antigen B, and antinuclear antibodies. Conclusions: The epidemiology of SS is poorly characterized in Africa. Available data are broadly consistent with those from other populations. Extensive and high-quality research is urgently needed.
MeSH term(s)	Adult ; Aged ; Antibodies, Antinuclear ; Arthritis, Rheumatoid ; Autoantibodies ; Female ; Humans ; Middle Aged ; Prevalence ; Sjogren's Syndrome/diagnosis ; Sjogren's Syndrome/epidemiology
Chemical Substances	Antibodies, Antinuclear ; Autoantibodies
Language	English
Publishing date	2021-12-27
Publishing country	United States
Document type	Journal Article ; Review
ZDB-ID	1283266-2
ISSN	1536-7355 ; 1076-1608
ISSN (online)	1536-7355
ISSN	1076-1608
DOI	10.1097/RHU.0000000000001708
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Article ; Online: Epidemiology of Idiopathic Inflammatory Myopathies in Africa: A Contemporary Systematic Review.

Essouma, Mickael / Noubiap, Jean Jacques / Singwe-Ngandeu, Madeleine / Hachulla, Eric

Journal of clinical rheumatology : practical reports on rheumatic & musculoskeletal diseases

2021 Volume 28, Issue 2, Page(s) e552–e562

Abstract: Background: The epidemiology of idiopathic inflammatory myopathies (IIMs) has been extensively studied in America, Europe, and Asia, but remains unclear in Africa.: Objective: The aim of this review was to summarize available data on the epidemiology ...

Abstract	Background: The epidemiology of idiopathic inflammatory myopathies (IIMs) has been extensively studied in America, Europe, and Asia, but remains unclear in Africa. Objective: The aim of this review was to summarize available data on the epidemiology of IIMs in Africa. Methods: We searched MEDLINE, EMBASE, and African Journals Online for studies published up to December 30, 2020, and reporting epidemiological data on IIMs in Africa. Data were combined through narrative synthesis. The review protocol was registered with PROSPERO, CRD42020186781. Results: We included 39 studies reporting 683 cases (71.7% adults) of IIMs. Incidence rates of ~7.5/1,000,000 person-years and 1.2/1,000,000 person-years were estimated for dermatomyositis (DM), whereas polymyositis (PM) had an incidence rate of 8.8/1,000,000 person-years. Prevalence estimates of 11.49/100,000 and 11/100,000 (95% confidence interval, 0-32) were provided for IIMs and the PM subtype, respectively. Mean age at diagnosis ranged from 7.9 to 57.2 years, and 50% to 100% of the patients were females. Main subtypes of adult-onset IIMs were DM (21%-93%) and PM (12%-79%), whereas the commonest juvenile subtype was juvenile DM (5.8%-9%). Skeletal muscle involvement (56%-100%) was the main disease feature, and esophagus was the most commonly affected internal organ (6%-65.2%). Anti-Jo1/histidyl tRNA synthetase (7%-100%) and anti-Mi2 (17%-45%) antibodies were the most frequent myositis specific antibodies. Early mortality was high (7.8%-45%), and main death causes were infections, cancers and organ damage in respiratory and cardiovascular domains. Conclusions: Apart from a potential younger age at onset of adult IIMs in Africa, current sparse data mostly suggest a similar epidemiology between Africa and other regions. Further high-quality studies are required to validate these findings.
MeSH term(s)	Adolescent ; Adult ; Africa/epidemiology ; Autoantibodies ; Child ; Dermatomyositis/diagnosis ; Female ; Humans ; Middle Aged ; Myositis/diagnosis ; Myositis/epidemiology ; Neoplasms ; Polymyositis/diagnosis ; Young Adult
Chemical Substances	Autoantibodies
Language	English
Publishing date	2021-04-12
Publishing country	United States
Document type	Journal Article ; Review ; Systematic Review
ZDB-ID	1283266-2
ISSN	1536-7355 ; 1076-1608
ISSN (online)	1536-7355
ISSN	1076-1608
DOI	10.1097/RHU.0000000000001736
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Article ; Online: Calcinosis in systemic sclerosis.

Pokeerbux, Mohammad Ryadh / Farhat, Meryem Maud / Merger, Marguerite / Launay, David / Hachulla, Eric

Joint bone spine

2021 Volume 88, Issue 4, Page(s) 105180

MeSH term(s)	Calcinosis/diagnostic imaging ; Calcinosis/etiology ; Humans ; Scleroderma, Systemic/complications ; Scleroderma, Systemic/diagnosis
Language	English
Publishing date	2021-03-31
Publishing country	France
Document type	Journal Article
ZDB-ID	2020487-5
ISSN	1778-7254 ; 1297-319X
ISSN (online)	1778-7254
ISSN	1297-319X
DOI	10.1016/j.jbspin.2021.105180
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Article ; Online: Characterisation of airway disease associated with Sjögren disease.

Meudec, Loïc / Debray, Marie-Pierre / Beurnier, Antoine / Marques, Cindy / Juge, Pierre-Antoine / Dhote, Robin / Larroche, Claire / Fauchais, Anne Laure / Dernis, Emanuelle / Vittecoq, Olivier / Saraux, Alain / Gottenberg, Jacques-Eric / Hachulla, Eric / Le Guern, Véronique / Dieudé, Philippe / Seror, Raphaele / Mariette, Xavier / Nocturne, Gaétane

RMD open

2024 Volume 10, Issue 1

Abstract: Objective: Although airway disease associated with Sjögren's disease (Sjo-AD) is common, it is poorly studied compared with interstitial lung disease (ILD). In this study, we aimed to assess factors associated with Sjo-AD, the characteristics and ... ...

Abstract	Objective: Although airway disease associated with Sjögren's disease (Sjo-AD) is common, it is poorly studied compared with interstitial lung disease (ILD). In this study, we aimed to assess factors associated with Sjo-AD, the characteristics and prognosis of this manifestation. Methods: We performed a retrospective multicentric study involving nine centres. We included Sjo-AD patients confirmed by at least one clinician and one CT scan report. Clinical and biological data, pulmonary function test (PFT), and CT scans were collected. A single radiologist specialist in thoracic diseases reviewed CT scans. Sjo-AD patients were compared with Sjo controls without pulmonary involvement, randomly selected after matching for age and disease duration. Results: We included 31 Sjo-AD and 62 Sjo controls without pulmonary history. Sjo-AD had a higher disease activity (ESSDAI) compared with controls, even when excluding the pulmonary domain of the score (7 vs 3.8, p<0.05), mainly due to the biological activity. Sjo-AD was multilobar (72%) and associated with signs of both bronchiectasis and bronchiolitis (60%). Obstructive lung disease occurred in 32% at the time of Sjo-AD diagnosis. Overall, PFT was stable after 8.7±7 years follow-up but repeated CT scans showed extended lesions in 41% of cases within 6±3.2 years. No patient developed Sjo-ILD. Sjo-AD progression was independent of the global disease activity. Conclusions: Sjo-AD preferentially affects Sjo patients with higher biological activity. It is often characterised as a diffuse disease, affecting both proximal and distal airways, with a slow evolution over time and no progression to Sjo-ILD.
MeSH term(s)	Humans ; Lung/diagnostic imaging ; Lung/pathology ; Lung Diseases, Interstitial/diagnosis ; Lung Diseases, Interstitial/etiology ; Prognosis ; Retrospective Studies ; Sjogren's Syndrome/complications ; Sjogren's Syndrome/diagnosis
Language	English
Publishing date	2024-02-29
Publishing country	England
Document type	Journal Article ; Multicenter Study
ZDB-ID	2812592-7
ISSN	2056-5933 ; 2056-5933
ISSN (online)	2056-5933
ISSN	2056-5933
DOI	10.1136/rmdopen-2023-003866
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Article ; Online: Rituximab resistance at 3months of induction therapy in newly diagnosed or relapsing ANCA-associated vasculitis: A French multicentre retrospective study in 116 patients.

Machet, Thomas / Quémeneur, Thomas / Ledoult, Emmanuel / Mesbah, Rafik / Lebas, Celine / Hachulla, Eric / Pokeerbux, Mohammad Ryadh

Joint bone spine

2023 Volume 90, Issue 5, Page(s) 105591

Abstract: Objective: To evaluate rituximab (RTX) resistance at 3months (M3) of induction therapy in antineutrophil cytoplasmic antibody (ANCA) associated vasculitis (AAV).: Methods: Multicentre French retrospective study conducted between 2010 and 2020 ... ...

Abstract	Objective: To evaluate rituximab (RTX) resistance at 3months (M3) of induction therapy in antineutrophil cytoplasmic antibody (ANCA) associated vasculitis (AAV). Methods: Multicentre French retrospective study conducted between 2010 and 2020 including patients with newly diagnosed or relapsing AAV (granulomatosis with polyangiitis or microscopic polyangiitis) having received induction therapy with RTX. Primary endpoint was the presence of RTX resistance at 3months (M3) defined as uncontrolled disease (worsening feature on the BVAS/WG 1month after RTX induction) or disease flare (increase in BVAS/WG of≥1 point before M3). Results: Out of 121 patients included, we analysed 116 patients. Fourteen patients (12%) had RTX resistance at M3 with no difference in baseline demographics, vasculitis type, ANCA type, disease status or organ involvement. Patients with RTX resistance at M3 had a greater proportion of localized disease (43% vs. 18%, P<0.05) and were less often treated by initial methylprednisolone (MP) pulse (21% vs. 58%, P<0.01). Out of the 14 patients with RTX resistance, seven received additional immunosuppressive therapy. All patients were in remission at 6months. Compared to responders, patients with RTX resistance at M3 were less often treated with prophylactic trimethoprim-sulfamethoxazole (57% vs. 85%, P<0.05). Twenty-four patients died during follow-up, one-third of them from infections and half of them from SARS-CoV-2. Conclusion: Twelve percent of patients had RTX resistance at M3. These patients more often had localized form of the disease and were less treated by initial MP pulse and by prophylactic trimethoprim-sulfamethoxazole.
MeSH term(s)	Humans ; Rituximab ; Retrospective Studies ; Antibodies, Antineutrophil Cytoplasmic ; Induction Chemotherapy ; Trimethoprim, Sulfamethoxazole Drug Combination/therapeutic use ; Treatment Outcome ; COVID-19 ; SARS-CoV-2 ; Anti-Neutrophil Cytoplasmic Antibody-Associated Vasculitis/diagnosis ; Anti-Neutrophil Cytoplasmic Antibody-Associated Vasculitis/drug therapy ; Remission Induction
Chemical Substances	Rituximab (4F4X42SYQ6) ; Antibodies, Antineutrophil Cytoplasmic ; Trimethoprim, Sulfamethoxazole Drug Combination (8064-90-2)
Language	English
Publishing date	2023-05-23
Publishing country	France
Document type	Multicenter Study ; Journal Article
ZDB-ID	2020487-5
ISSN	1778-7254 ; 1297-319X
ISSN (online)	1778-7254
ISSN	1297-319X
DOI	10.1016/j.jbspin.2023.105591
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Article ; Online: Giant cell arteritis-related cerebrovascular ischemic events: a French retrospective study of 271 patients, systematic review of the literature and meta-analysis.

Penet, Thomas / Lambert, Marc / Baillet, Clio / Outteryck, Olivier / Hénon, Hilde / Morell-Dubois, Sandrine / Hachulla, Eric / Launay, David / Pokeerbux, Mohammad Ryadh

Arthritis research & therapy

2023 Volume 25, Issue 1, Page(s) 116

Abstract: Background: Cerebrovascular ischemic events (CIE) are among the most severe complications of giant cell arteritis (GCA). Heterogeneity between different studies in the definition of GCA-related CIE leads to uncertainty regarding their real prevalence. ... ...

Abstract	Background: Cerebrovascular ischemic events (CIE) are among the most severe complications of giant cell arteritis (GCA). Heterogeneity between different studies in the definition of GCA-related CIE leads to uncertainty regarding their real prevalence. The aim of our study was to evaluate the prevalence and describe the characteristics of GCA-related CIE in a well-phenotyped cohort completed by a meta-analysis of the existing literature. Methods: In this retrospective study performed in the Lille University Hospital, all consecutive patients with GCA according to American College of Rheumatology (ACR) diagnostic criteria were included from January 1, 2010, to December 31, 2020. A systematic review of the literature using MEDLINE and EMBASE was performed. Cohort studies of unselected GCA patients reporting CIE were included in the meta-analysis. We calculated the pooled summary estimate of GCA-related CIE prevalence. Results: A total of 271 GCA patients (89 males, mean age 72 ± 9 years) were included in the study. Among them, 14 (5.2%) presented with GCA-related CIE including 8 in the vertebrobasilar territory, 5 in the carotid territory, and 1 patient having multifocal ischemic and hemorrhagic strokes related to intra-cranial vasculitis. Fourteen studies were included in the meta-analysis, representing a total population of 3553 patients. The pooled prevalence of GCA-related CIE was 4% (95% CI 3-6, I Conclusions: The pooled prevalence of GCA-related CIE was 4%. Our cohort identified an association between GCA-related CIE, lower BMI, and vertebral, intracranial, and axillary arteries involvement on various imaging modalities.
MeSH term(s)	Male ; Humans ; Middle Aged ; Aged ; Aged, 80 and over ; Giant Cell Arteritis/complications ; Giant Cell Arteritis/diagnostic imaging ; Giant Cell Arteritis/epidemiology ; Retrospective Studies ; Positron Emission Tomography Computed Tomography ; Carotid Arteries ; Cohort Studies
Language	English
Publishing date	2023-07-07
Publishing country	England
Document type	Systematic Review ; Meta-Analysis ; Journal Article
ZDB-ID	2107602-9
ISSN	1478-6362 ; 1478-6354
ISSN (online)	1478-6362
ISSN	1478-6354
DOI	10.1186/s13075-023-03091-x
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