Myelodysplastic syndromes and autoimmune diseases—Case series and review of literature

Ustwani, Omar Al; Ford, Laurie; Sait, Sheila; Block, Anne Marie W.; Barcos, Maurice; Vigil, Carlos Enrique; Griffiths, Elizabeth A.; Thompson, James E.; Wang, Eunice S.; Ambrus, Julian L.; Wetzler, Meir

doi:10.1016/j.leukres.2013.04.007

Cited by 68 publications

(53 citation statements)

References 53 publications

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“…A Swedish population-based study demonstrated a strong association between chronic immune stimulation and MDS predisposition, where a history of autoimmune disease was associated with a two-fold significantly increased risk of MDS (OR 2.1 [95% CI 1.7-2.6]) [6]. Small case series have reported distinct clinical features and outcomes for MDS patients with autoimmune diseases [7]. We report here, in a large cohort, the prevalence of autoimmune diseases among MDS patients and compare characteristics and outcomes of MDS patients with and without autoimmune diseases.…”

Section: Introductionmentioning

confidence: 99%

Autoimmune diseases and myelodysplastic syndromes

et al. 2016

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Immune dysregulation and altered T-cell hemostasis play important roles in the pathogenesis of myelodysplastic syndromes (MDS). Recent studies suggest an increased risk of MDS among patients with autoimmune diseases. Here, we investigated the prevalence of autoimmune diseases among MDS patients, comparing characteristics and outcomes in those with and without autoimmune diseases. From our study group of 1408 MDS patients, 391 (28%) had autoimmune disease, with hypothyroidism being the most common type, accounting for 44% (n 5 171) of patients (12% among all MDS patients analyzed). Other autoimmune diseases with 5% prevalence included idiopathic thrombocytopenic purpura in 12% (n 5 46), rheumatoid arthritis in 10% (n 5 41), and psoriasis in 7% (n 5 28) of patients. Autoimmune diseases were more common in female MDS patients, those with RA or RCMD WHO subtype, and those who were less dependent on red blood cell transfusion. Median overall survival (OS) was 60 months (95% CI, 50-70) for patients with autoimmune diseases versus 45 months (95% CI, 40-49) for those without (log-rank test, P 5 0.006). By multivariate analysis adjusting for revised IPSS and age >60 years, autoimmune diseases were a statistically significant independent factor for OS (HR 0.78; 95% CI, 0.66-0.92; P 5 0.004). The rate of acute myeloid leukemia (AML) transformation was 23% (n 5 89) in MDS patients with autoimmune disease versus 30% (n 5 301) in those without (P 5 0.011). Patient groups did not differ in response to azacitidine or lenalidomide treatment. Autoimmune diseases are prevalent among MDS patients. MDS patients with autoimmune diseases have better OS and less AML transformation.Am. J. Hematol. 91:E280-E283,

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Section: Introductionmentioning

confidence: 99%

Autoimmune diseases and myelodysplastic syndromes

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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“…Collectively, the incidence of this association has been reported in several retrospective analyses to be in the range of 7-25% of MDS patients [10]. However, these studies differ in their definition of MDS and vary in their definition for MDS-associated AICs (table 1).…”

Section: The Spectrum Of Systemic Aics In Mdsmentioning

confidence: 99%

“…One systematic review summarized data from 10 retrospective studies dating from the early 1990s to 2009 and found vasculitides (isolated or systemic) to be the most frequent diagnosis in patients with MDS and an associated AIC (40%), followed by seronegative arthritis (27%), neurologic disease (24%), bronchiolitis obliterans with organizing pneumonia/interstitial pulmonary fibrosis (15%), glomerulonephritis (12%), rheumatoid arthritis (12%), and Sweet's syndrome (10%) [10]. …”

Section: The Spectrum Of Systemic Aics In Mdsmentioning

confidence: 99%

Autoimmunity and Inflammation in Myelodysplastic Syndromes

Wolach

Stone²

2016

Acta Haematol

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Autoimmune and inflammatory conditions (AICs) are encountered in up to 25% of patients with myelodysplastic syndromes (MDS). A wide range of AICs have been reported in association with MDS and can range from limited clinical manifestations to systemic diseases affecting multiple organs. Vasculitides, connective tissue diseases, and inflammatory arthritis are frequently reported in different studies; noninfectious fever and constitutional symptoms at presentation are common. Associations between AICs and specific MDS characteristics vary by study, but the available data suggest that AICs cluster more often in younger patients with higher-risk MDS. In general, AICs do not seem to confer worse survival, although certain AICs may be associated with adverse outcome (e.g. vasculitis) or progression of MDS (Sweet's syndrome). Nonetheless, these complications may have a significant impact on quality of life and affect the timing and type of MDS-directed therapy. The mainstay of management of these complications in the short term relies on immunosuppressive drugs. Increasing evidence suggests that hypomethylating agents may be effective in treating these complications and reduce steroid dependence. While the pathogenesis of AICs is incompletely understood, growing appreciation of cellular immune deregulation, cytokine hypersecretion, and the genetic heterogeneity underlying MDS may improve our understanding of common pathways linking MDS, inflammation, and autoimmunity.

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Myelodysplastic syndromes and autoimmune diseases—Case series and review of literature

Cited by 68 publications

References 53 publications

Autoimmune diseases and myelodysplastic syndromes

Autoimmune diseases and myelodysplastic syndromes

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

Autoimmunity and Inflammation in Myelodysplastic Syndromes

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