Systemic inflammatory and autoimmune manifestations associated with myelodysplastic syndromes and chronic myelomonocytic leukaemia: a French multicentre retrospective study

Mékinian, A.; Grignano, Éric; Braun, Thorsten; Decaux, Olivier; Liozon, É.; Costédoat-Chalumeau, Nathalie; Kahn, Jean‐Emmanuel; Hamidou, M.; Park, Sophie; Puéchal, Xavier; Toussirot, Éric; Falgarone, Géraldine; Launay, David; Morel, Nathalie; Trouiller, Sébastien; Mathian, Alexis; Gombert, Bruno; Schoindre, Y.; Lioger, Bertrand; Wazières, B. de; Amoura, Zahir; Buchdaul, Anne-Laure; Georgin‐Lavialle, Sophie; Dion, Jérémie; Madaule, S.; Raffray, L.; Cathébras, P.; Piette, Jean Charles; Rose, Christian; Ziza, Jean Marc; Lortholary, Olivier; Montestruc, François; Omouri, Mohammed; Denis, Guillaume; Rossignol, Julien; Nimubona, Stanislas; Adès, Lionel; Gardin, Claude; Fenaux, Pierre; Fain, Olivier

doi:10.1093/rheumatology/kev294

Cited by 200 publications

(203 citation statements)

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“…No previous studies focused on CMML-associated SAIDs treatment and outcomes, and response rates to steroids alone was similar comparing to our larger cohort of MDS-associated SAIDs (80%) (3). Despite this initial good response, 40% of patients needed a second-line treatment for relapse or steroid dependence, comparing to 48% in other MDS-associated SAIDs (3).…”

Section: Discussionsupporting

confidence: 70%

“…CPSS score was 0 (n=6, 37.5%), 1 (n= 6, 37.5%), 2 or 3 (n=3, 19%), and 4 (n=1, 6%) ( Table 1). According to the French-American-British classification of CMML, 55% of patients had a myelodysplastic form (WBC <13000 /mm 3 ) and 45% a myeloproliferative form (WBC >13000 /mm 3 ).…”

Section: Cmml and Saids Characteristicsmentioning

confidence: 99%

“…Systemic autoimmune and/or inflammatory diseases (SSAIDs) have been reported in 15% to 25% of MDS/CMML patients, in series that included only few CMML cases, without focus on this disorder (2). In a recent report, we described 123 patients with MDS/CMMLassociated SAIDs, mainly systemic vasculitis (32%), connective tissue diseases (25%) and inflammatory arthritis (23%) (3). The type of MDS/CMML was refractory anemia in 25% of cases, refractory anemia with blasts excess-1 in 15% and CMML in 15%.…”

Section: Introductionmentioning

confidence: 99%

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Autoimmune and inflammatory diseases associated with chronic myelomonocytic leukemia: A series of 26 cases and literature review

et al. 2016

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We wanted to describe the characteristics, treatment and outcome of autoimmune and inflammatory diseases (SAIDs) associated with chronic myelomonocytic leukemia (CMML), and conducted a French multicenter retrospective study and a literature review.We included 26 cases of CMML (median age 75 years, 54% female), 80% with CMML-1. CPSS score was low (0 or 1) in 75% of cases. SAIDS was systemic vasculitis in 54%. Diagnosis of the 2 diseases was concomitant in 31% cases, and CMML was diagnosed before SAIDs in 12 cases (46%). First line treatment for SAIDs consisted mostly of steroid, with 85% of response. Second-line treatment was needed in 40% cases. Six patients received hypomethylating agents, with 66% response on SAIDs. A literature review found 49 cases of CMML-associated SAIDs, in whom SAIDs was systemic vasculitis in 29% cases. Hence, vasculitis is the most frequent SAIDs associated with CMML. After initial response to steroids, recurrence and steroid-dependence were frequent. Hypomethylating agents may be interesting in this context.

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Section: Discussionsupporting

confidence: 70%

Section: Cmml and Saids Characteristicsmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Autoimmune and inflammatory diseases associated with chronic myelomonocytic leukemia: A series of 26 cases and literature review

et al. 2016

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“…Association with inflammatory myopathies has been very rarely described (8)(9)(10)(11)(12)(13)(14)(15). In particular, in the case report published by Tsuji et al, antinuclear antigens were found, but the assay for anti-Jo1, RNP and Scl70 resulted negative.…”

Section: Case Reportmentioning

confidence: 99%

First report of anti-TIF1γ dermatomyositis in a patient with myelodysplastic syndrome

2017

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SUMMARYInflammatory myopathies as para-neoplastic phenomena were first described by Sterz in 1916. Recently, myositis specific autoantibodies were described in cancer-associated myositis. Anti-transcription intermediary factor 1 gamma (anti-TIF1γ) antibodies have been found in both young adults affected by juvenile dermatomyositis and in elderly patients with cancer-associated myositis. In this regard, we report herein the first case of anti-TIF1γ dermatomyositis secondary to a myelodysplastic syndrome.

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“…Bei anderen Non-Hodgkin-Lymphomen (NHL) und auch beim Morbus Hodgkin wurde in Einzelberichten neben Arthralgien und Myalgien auch eine eosinophile Fasziitis, eine Arthritis oder Gelenkinfiltration mit Tumorzellen beschrieben [9,31]. Etwa ein Drittel der Arthritiden lag dabei als Polyarthritis vor, und bei ca.…”

Section: Introductionunclassified

Rheumatische Krankheitserscheinungen bei hämatologischen Neoplasien

Kleinert¹,

Rosée

Krüger

2017

Z Rheumatol

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Systemic inflammatory and autoimmune manifestations associated with myelodysplastic syndromes and chronic myelomonocytic leukaemia: a French multicentre retrospective study

Abstract: The spectrum of SIADs associated to MDS is heterogeneous, steroid sensitive, but often steroid dependent.

Cited by 200 publications

References 23 publications

Autoimmune and inflammatory diseases associated with chronic myelomonocytic leukemia: A series of 26 cases and literature review

Autoimmune and inflammatory diseases associated with chronic myelomonocytic leukemia: A series of 26 cases and literature review

First report of anti-TIF1γ dermatomyositis in a patient with myelodysplastic syndrome

Rheumatische Krankheitserscheinungen bei hämatologischen Neoplasien

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