The autoimmune manifestations associated with myelodysplastic syndrome respond to 5‐azacytidine: a report on three cases

Pilorge, Sylvain; Doleris, L Moulonguet; Dreyfus, F.; Park, Sophie

doi:10.1111/j.1365-2141.2010.08557.x

Cited by 35 publications

(19 citation statements)

References 9 publications

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“…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). The effect of MDS treatment on SAIDs outcome is not established, and few casereports described SAIDs remission with hypomethylating agents (18)(19)(20). Among our 6 cases treated with 5-azacytidine or decitabine, 4 showed SAIDs remission associated with hematological response.…”

Section: Discussionmentioning

confidence: 99%

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: Discussionmentioning

confidence: 99%

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

et al. 2016

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“…Furthermore, there is strong evidence that a history of AID might predict a better clinical response to DNA methyltransferase inhibitors [17] or other immunomodulants (i.e. 5-azacytidine) [53,54] in MDS. However, one study has reported that steroids or azathioprine could only temporarily control the autoimmune process but not ultimately cure the hematopoietic neoplasm [55].…”

Section: Discussionmentioning

confidence: 99%

A Close Association of Autoimmune-Mediated Processes and Autoimmune Disorders with Chronic Myelomonocytic Leukemia: Observation from a Single Institution

Peker¹,

Padron²,

Bennett

et al. 2014

Acta Haematol

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Chronic myelomonocytic leukemia (CMML), a clonal hematopoietic stem cell disease, may be linked to immune-mediated processes and/or autoimmune disorders (AID), although the exact pathogens are still elusive. We retrospectively analyzed 123 CMML patients in our institution. Twenty-four CMML patients (19.5%) had at least one immune-mediated disorder, most commonly idiopathic thrombocytopenic purpura, gout and psoriasis. Four of these 24 patients (15%) had more than one AID. We found that, in contrast to the general population with a prevalence rate of 3.2-5.2%, newly diagnosed CMML patients demonstrated a high prevalence and variety of immune-mediated processes and/or AID. When we compared the results with those of myelodysplastic syndromes published in the literature, the prevalence of AID in these two groups of patients is similar. Our results also showed that the presence of cytogenetic abnormalities was less in CMML patients with AID (6 of 21; 28.6%) than in those without AID (37 of 94; 39.4%), although there was no statistical significance (p = 0.334). A multicenter large cohort study of CMML with AID is recommended to illustrate the molecular relationship between the two distinct groups. © 2014 S. Karger AG, Basel

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“…Despite a lack of consensus on how to treat the underlying MDS complicated with DM or other autoimmune diseases, three case reports mentioned that cord blood stem cell transplantation resolved the symptoms of Behçet's disease complicated with MDS (15)(16)(17). Furthermore, three MDS case reports showed that the administration of 5-azacytidine for the treatment of MDS also ameliorated the accompanying autoimmune symptoms (18). Another case report of SLE complicated with MDS showed a clinical improvement in SLE with negative results for ANAs and a significant decrease in the CD4+FoxP3+ regulatory T cell count after the administration of 5-azacytidine for the treatment of MDS (19).…”

Section: Discussionmentioning

confidence: 99%

Refractory Dermatomyositis Complicated with Myelodysplastic Syndrome

et al. 2015

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We herein describe a case of refractory dermatomyositis (DM) complicated with myelodysplastic syndrome (MDS). Despite intensive immunosuppressive therapies, the activity of myositis, skin ulcers, and interstitial pneumonia did not improve. The patient ultimately died following the progression of interstitial pneumonia. There are few reports of DM accompanying MDS to date, and any association in the pathogenesis between the two is still unclear. However, underlying MDS may have the potential to influence the therapeutic response of DM.

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The autoimmune manifestations associated with myelodysplastic syndrome respond to 5‐azacytidine: a report on three cases

Cited by 35 publications

References 9 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

A Close Association of Autoimmune-Mediated Processes and Autoimmune Disorders with Chronic Myelomonocytic Leukemia: Observation from a Single Institution

Refractory Dermatomyositis Complicated with Myelodysplastic Syndrome

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