Obliterative bronchiolitis in a patient with myelodysplastic syndrome before bone marrow transplantation

Mobeireek, Abdullah; Almohareb, Fahad; Al‐Dayel, Fouad; Al-Gazlan, Suliman; Al-Abdely, Hail M.; Khalid, Mohammad

doi:10.1016/j.rmed.2006.04.025

Cited by 3 publications

(1 citation statement)

References 12 publications

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“…There are several reports in the literature correlating various autoimmune manifestations such as leukocytoclastic vasculitis [7], relapsing polychondritis [8], polyarteritis nodosa, Grave's disease, Sjögren's syndrome [9], Wegener's granulomatosis [10], bronchiolitis obliterans [11], autoimmune hemolytic anemia [12], and thrombocytopenia [13,14], red cell aplasia [15], dermatomyositis [16], peripheral polyneuropathy and inflammatory bowel disease [17], giant cell arteritis/polymyalgia rheumatica [18,19], and Addison's disease [20]. …”

Section: Discussionmentioning

confidence: 99%

Antiretroviral activity of 5-azacytidine during treatment of a HTLV-1 positive myelodysplastic syndrome with autoimmune manifestations

Diamantopoulos

Michael

Benopoulou

et al. 2012

Virol J

135

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Myelodysplastic syndromes (MDS) are often accompanied by autoimmune phenomena. The underlying mechanisms for these associations remain uncertain, although T cell activation seems to be important. Human T-lymphotropic virus (HTLV-1) has been detected in patients with myelodysplastic syndromes, mostly in regions of the world which are endemic for the virus, and where association of HTLV-1 with rheumatological manifestation is not rare. We present here the case of a 58 year old man who presented with cytopenias, leukocytoclastic vasculitis of the skin and glomerulopathy, and was diagnosed as MDS (refractory anemia with excess blasts - RAEB 1). The patient also tested positive for HTLV-1 by PCR. After 8 monthly cycles of 5-azacytidine he achieved a complete hematologic remission. Following treatment, a second PCR for HTLV-1 was carried out and found to be negative. This is the first report in the literature of a HTLV-1-positive MDS with severe autoimmune manifestations, which was treated with the hypomethylating factor 5-azacitidine, achieving cytogenetic remission with concomitant resolution of the autoimmune manifestations, as well as HTLV-1-PCR negativity. HTLV-1-PCR negativity may be due to either immune mediated clearance of the virus, or a potential antiretroviral effect of 5-azacytidine. 5-azacytidine is known for its antiretroviral effects, although there is no proof of its activity against HTLV-1 infection in vivo.

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Section: Discussionmentioning

confidence: 99%

Antiretroviral activity of 5-azacytidine during treatment of a HTLV-1 positive myelodysplastic syndrome with autoimmune manifestations

Diamantopoulos

Michael

Benopoulou

et al. 2012

Virol J

135

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

et al. 2013

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Our objective was to recognize the association of autoimmune disease (AD) in patients with myelodysplastic syndromes (MDS) and understand how this association could affect prognosis and management of both diseases. We describe our cohort of 10 patients and 34 patients reported in the English literature in addition to ten cohort studies. Interestingly, four cases showed improvement in AD after 5-azacitidine treatment. The mechanism(s) of the association between AD and MDS are discussed. Treatment could be targeted against AD, MDS or both, though based on recent reports, treating MDS with hypomethylating agents alone could improve the associated AD.

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Systemic Pulmonary Events Associated with Myelodysplastic Syndromes: A Retrospective Multicentre Study

Scanvion

Pascal

Sy³

et al. 2021

JCM

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Although pulmonary events are considered to be frequently associated with malignant haemopathies, they have been sparsely studied in the specific context of myelodysplastic syndromes (MDS). We aimed to describe their different types, their relative proportions and their relative effects on overall survival (OS). We conducted a multicentre retrospective cohort study. Patients with MDS (diagnosed according to the 2016 WHO classification) and pulmonary events were included. The inclusion period was 1 January 2007 to 31 December 2017 and patients were monitored until August 2019. Fifty-five hospitalized patients were included in the analysis. They had 113 separate pulmonary events. Thirteen patients (23.6%) had a systemic autoimmune disease associated with MDS. Median age at diagnosis of MDS was 77 years. Median time to onset of pulmonary events was 13 months. Pulmonary events comprised: 70 infectious diseases (62%); 27 interstitial lung diseases (23.9%), including 13 non-specific interstitial pneumonias and seven secondary organizing pneumonias or respiratory bronchiolitis–interstitial lung diseases; 10 pleural effusions (8.8%), including four cases of chronic organizing pleuritis with exudative effusion; and six pulmonary hypertensions (5.3%). The median OS of the cohort was 29 months after MDS diagnosis but OS was only 10 months after a pulmonary event. The OS was similar to that of the general myelodysplastic population. However, the occurrence of a pulmonary event appeared to be either an accelerating factor of death or an indicator for the worsening of the underlying MDS in our study. More than a third of pulmonary events were non-infectious and could be systemic manifestations of MDS.

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Obliterative bronchiolitis in a patient with myelodysplastic syndrome before bone marrow transplantation

Cited by 3 publications

References 12 publications

Antiretroviral activity of 5-azacytidine during treatment of a HTLV-1 positive myelodysplastic syndrome with autoimmune manifestations

Antiretroviral activity of 5-azacytidine during treatment of a HTLV-1 positive myelodysplastic syndrome with autoimmune manifestations

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

Systemic Pulmonary Events Associated with Myelodysplastic Syndromes: A Retrospective Multicentre Study

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