Changing prognosis in paroxysmal nocturnal haemoglobinuria disease subcategories: an analysis of the International PNH Registry.
Identifieur interne : 001981 ( PubMed/Corpus ); précédent : 001980; suivant : 001982Changing prognosis in paroxysmal nocturnal haemoglobinuria disease subcategories: an analysis of the International PNH Registry.
Auteurs : G. Socié ; H. Schrezenmeier ; P. Muus ; I. Lisukov ; A. Röth ; A. Kulasekararaj ; J W Lee ; D. Araten ; A. Hill ; R. Brodsky ; A. Urbano-Ispizua ; J. Szer ; A. Wilson ; P. HillmenSource :
- Internal medicine journal [ 1445-5994 ] ; 2016.
English descriptors
- KwdEn :
- Adult, Anemia, Aplastic (epidemiology), Antibodies, Monoclonal, Humanized (therapeutic use), Cause of Death, Erythrocyte Transfusion, Female, France, Hemoglobinuria, Paroxysmal (classification), Hemoglobinuria, Paroxysmal (mortality), Hemoglobinuria, Paroxysmal (therapy), Humans, Incidence, Male, Middle Aged, Multivariate Analysis, Prognosis, Registries, Retrospective Studies, Risk Factors, Survival Analysis, Thrombosis (epidemiology).
- MESH :
- chemical , therapeutic use : Antibodies, Monoclonal, Humanized.
- classification : Hemoglobinuria, Paroxysmal.
- epidemiology : Anemia, Aplastic, Thrombosis.
- mortality : Hemoglobinuria, Paroxysmal.
- therapy : Hemoglobinuria, Paroxysmal.
- Adult, Cause of Death, Erythrocyte Transfusion, Female, France, Humans, Incidence, Male, Middle Aged, Multivariate Analysis, Prognosis, Registries, Retrospective Studies, Risk Factors, Survival Analysis.
Abstract
Paroxysmal nocturnal haemoglobinuria (PNH) is a rare disease. Although much progress has been made in the understanding of the pathophysiology of the disease, far less is known with respect to the clinical outcomes of patients with PNH. Few retrospective studies provide survival estimates, and even fewer have explored the clinical heterogeneity of the disease. Haemolytic and aplastic anaemia (AA) forms of the disease have been recognised as main disease categories, with the haemolytic form being associated with the worst prognosis by the largest studied cohort some years ago.
DOI: 10.1111/imj.13160
PubMed: 27305361
Links to Exploration step
pubmed:27305361Le document en format XML
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<term>France</term>
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<front><div type="abstract" xml:lang="en">Paroxysmal nocturnal haemoglobinuria (PNH) is a rare disease. Although much progress has been made in the understanding of the pathophysiology of the disease, far less is known with respect to the clinical outcomes of patients with PNH. Few retrospective studies provide survival estimates, and even fewer have explored the clinical heterogeneity of the disease. Haemolytic and aplastic anaemia (AA) forms of the disease have been recognised as main disease categories, with the haemolytic form being associated with the worst prognosis by the largest studied cohort some years ago.</div>
</front>
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<pubmed><MedlineCitation Status="MEDLINE" Owner="NLM"><PMID Version="1">27305361</PMID>
<DateCreated><Year>2016</Year>
<Month>09</Month>
<Day>16</Day>
</DateCreated>
<DateCompleted><Year>2017</Year>
<Month>08</Month>
<Day>24</Day>
</DateCompleted>
<DateRevised><Year>2017</Year>
<Month>08</Month>
<Day>24</Day>
</DateRevised>
<Article PubModel="Print"><Journal><ISSN IssnType="Electronic">1445-5994</ISSN>
<JournalIssue CitedMedium="Internet"><Volume>46</Volume>
<Issue>9</Issue>
<PubDate><Year>2016</Year>
<Month>Sep</Month>
</PubDate>
</JournalIssue>
<Title>Internal medicine journal</Title>
<ISOAbbreviation>Intern Med J</ISOAbbreviation>
</Journal>
<ArticleTitle>Changing prognosis in paroxysmal nocturnal haemoglobinuria disease subcategories: an analysis of the International PNH Registry.</ArticleTitle>
<Pagination><MedlinePgn>1044-53</MedlinePgn>
</Pagination>
<ELocationID EIdType="doi" ValidYN="Y">10.1111/imj.13160</ELocationID>
<Abstract><AbstractText Label="BACKGROUND" NlmCategory="BACKGROUND">Paroxysmal nocturnal haemoglobinuria (PNH) is a rare disease. Although much progress has been made in the understanding of the pathophysiology of the disease, far less is known with respect to the clinical outcomes of patients with PNH. Few retrospective studies provide survival estimates, and even fewer have explored the clinical heterogeneity of the disease. Haemolytic and aplastic anaemia (AA) forms of the disease have been recognised as main disease categories, with the haemolytic form being associated with the worst prognosis by the largest studied cohort some years ago.</AbstractText>
<AbstractText Label="AIMS" NlmCategory="OBJECTIVE">To describe mortality and causes of death in PNH overall and by PNH classification and to evaluate risk factors associated with mortality.</AbstractText>
<AbstractText Label="METHODS" NlmCategory="METHODS">We analysed data of 2356 patients enrolled in the International PNH Registry with multivariate analyses, using time-dependent covariates. Patients were classified into haemolytic, AA/PNH syndrome or intermediate PNH.</AbstractText>
<AbstractText Label="RESULTS" NlmCategory="RESULTS">Overall, 122 (5.2%) patients died after enrolment, the incidence according to subcategories being 5.1, 11.7, 2.0 and 4.8% for patients with haemolytic PNH, AA-PNH, intermediate and insufficient data respectively. Older age and decreased performance status also affected survival in multivariate analysis. Improved outcome of patients with haemolytic PNH suggests that eculizumab treatment in PNH may be associated with improved survival.</AbstractText>
<AbstractText Label="CONCLUSION" NlmCategory="CONCLUSIONS">A detailed analysis of clinical presentations and causes of death in patients with PNH, overall and by disease subcategories, provide evidence that in the current era, patients with haemolytic PNH are no longer those who harbour the worst prognosis. This finding differs sharply from what has been previously reported.</AbstractText>
<CopyrightInformation>© 2016 Royal Australasian College of Physicians.</CopyrightInformation>
</Abstract>
<AuthorList CompleteYN="Y"><Author ValidYN="Y"><LastName>Socié</LastName>
<ForeName>G</ForeName>
<Initials>G</Initials>
<AffiliationInfo><Affiliation>Hematology Transplantation, AP-HP, Hospital Saint Louis, University Paris VII Denis Diderot and Inserm UMR 1160, Paris, France. gerard.socie@sls.aphp.fr.</Affiliation>
</AffiliationInfo>
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<Initials>H</Initials>
<AffiliationInfo><Affiliation>Institute of Transfusion Medicine, University of Ulm Ulm and Institute of Clinical Transfusion Medicine and Immunogenetics, German Red Cross Blood Transfusion Service Baden-Württemberg-Hessen and University Hospital Ulm, Ulm, Germany.</Affiliation>
</AffiliationInfo>
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<Author ValidYN="Y"><LastName>Muus</LastName>
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<ForeName>J W</ForeName>
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<AffiliationInfo><Affiliation>Seoul St. Mary's Hospital, Seoul, Korea.</Affiliation>
</AffiliationInfo>
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<Initials>J</Initials>
<AffiliationInfo><Affiliation>Royal Melbourne Hospital, Melbourne, Victoria, Australia.</Affiliation>
</AffiliationInfo>
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<Author ValidYN="Y"><LastName>Wilson</LastName>
<ForeName>A</ForeName>
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<AffiliationInfo><Affiliation>Alexion Pharmaceuticals, New Haven, Connecticut, USA.</Affiliation>
</AffiliationInfo>
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