Relationship between immunological abnormalities and paroxysmal nocturnal haemoglobinuria‐associated clones in myelodysplastic syndromes

Okamoto, Takahiro; Okada, Masaya; Takatsuka, Hiroyuki; Wada, Hiroshi; Fujimori, Yoshihiro; Kakishita, Eizo

doi:10.1046/j.1365-2141.2001.02924.x

Cited by 14 publications

(18 citation statements)

References 10 publications

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“…A correlation between immunologic abnormalities and prognosis in patients with MDS has been observed by some authors, and data have been contradicted by other reports (22,24). Moreover, our results revealed a lack of difference in the incidence of clonal alterations between the 2 groups (43% and 39%, respectively).…”

Section: Discussioncontrasting

confidence: 99%

“…However, a higher prevalence of 18.5% has been reported in a 4-year prospective study (22). Moreover, in a large Asian study, Okamoto et al observed an incidence of 12% of systemic manifestations associated with MDS and immune biologic abnormalities in 63% of cases (24). We report a high incidence of systemic and autoimmune manifestations in our series of MDS patients (19%), and we further suggest that this incidence might even be underestimated, since we performed a retrospective study with subsequent difficulties in collecting data.…”

Section: Discussionmentioning

confidence: 94%

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Systemic and immune manifestations in myelodysplasia: A multicenter retrospective study

Hollanda¹,

Beucher²,

Henrion³

et al. 2011

Arthritis Care & Research

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Objective. The presence of systemic and/or immune manifestations in myelodysplasia has been currently reported. The influence of these manifestations on the natural outcome of myelodysplastic syndrome has to be considered. We present a multicenter retrospective study (2002-2009) of patients with myelodysplastic syndrome disclosing systemic and/or immune manifestations. Methods. Forty-six patients with myelodysplasia presenting with systemic and/or immune manifestations were compared in terms of survival with 189 patients with myelodysplasia lacking these features. Results. The clinical picture in these cases consisted of fever (13%), arthralgia or arthritis (13%), and cutaneous manifestations (67%). Four cases of systemic vasculitis have been reported in our series, and they have a worse prognosis. Immune anomalies were recorded in 29% of the cases, and the presence of cryoglobulins was also associated with a worse prognosis. Conclusion. A difference in survival between patients with myelodysplastic syndrome with systemic manifestations and patients lacking these manifestations has been observed in the presence of systemic vasculitis and/or cryoglobulins.

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Section: Discussioncontrasting

confidence: 99%

Section: Discussionmentioning

confidence: 94%

Systemic and immune manifestations in myelodysplasia: A multicenter retrospective study

Hollanda¹,

Beucher²,

Henrion³

et al. 2011

Arthritis Care & Research

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“…As compared with CMML without SAIDs, our CMML patients with SAIDs showed unfavorable CMML features: more frequently CMML-2, poor karyotype and greater proportion of medullar blasts. No previous study has focused on survival of patients with CMML-associated SAIDs, and the impact of the SAIDs on survival remains controversial (16,17). Several reports showed that in patients with various SAIDs types, survival was similar in comparison with MDS without SAIDs.…”

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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“…However, sole PIG-A mutation is not sufficient for expansion of a PNH clone and clinical symptoms. Precise mechanisms of the clonal expansion are not yet known, probably, being coupled to bone marrow failure [12,21]. Such physiological mechanism is confirmed by higher incidence of PNH clone in patients with aplastic anemia (AA) and myelodysplastic syndrome (MDS) [11,18,27,26,29,30].…”

Section: Introductionmentioning

confidence: 94%

A mini-review on paroxysmal nocturnal hemoglobinuria and a case of Eculizumab treatment of PNH in elderly MDS patient

Дудина¹

2017

CTT

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SummaryParoxysmal nocturnal hemoglobinuria (PNH) is a systemic, acquired clonal hematological disorder which results from loss of complement-inhibiting proteins (CD55 and CD 59) from the blood cell surface. Therefore, such patients develop chronic complement-mediated intravascular hemolysis, with thromboses manifesting as most severe clinical complications. Recommendations of International PNH groups discern certain risk groups based on detection of a pathological cell clone, including patients with myelodysplastic syndrome (MDS). Here we describe detection of a clinically sound PNH clone in an elderly patient with MDS, as well as positive results of patho-genetic treatment with Eculizumab.

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Relationship between immunological abnormalities and paroxysmal nocturnal haemoglobinuria‐associated clones in myelodysplastic syndromes

Abstract: ACKNOWLEDGMENTThe authors are grateful to Mrs Aki Nomura for invaluable assistance with cell preparations and flow cytometric analysis.

Cited by 14 publications

References 10 publications

Systemic and immune manifestations in myelodysplasia: A multicenter retrospective study

Systemic and immune manifestations in myelodysplasia: A multicenter retrospective study

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

A mini-review on paroxysmal nocturnal hemoglobinuria and a case of Eculizumab treatment of PNH in elderly MDS patient

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