Literature DB >> 22639982

Under-recognized complications in patients with paroxysmal nocturnal haemoglobinuria: raised pulmonary pressure and reduced right ventricular function.

Anita Hill1, Robert J Sapsford, Andrew Scally, Richard Kelly, Stephen J Richards, Gus Khurisgara, Mohan U Sivananthan, Peter Hillmen.   

Abstract

Pulmonary hypertension is becoming a recognized complication of the hereditary and acquired haemolytic anaemias, associated with a poor prognosis. Recently we reported that patients with paroxysmal nocturnal haemoglobinuria (PNH) have high levels of N-terminal pro-brain natriuretic peptide (NT-proBNP), a biomarker associated with both right and left ventricular dysfunction and cardiac dysfunction. In the current study we evaluated a cohort of patients (N = 29) with haemolytic PNH for elevated pulmonary artery systolic pressure and cardiac function by Doppler-echocardiography. Of the 29 patients, eight were further studied using cardiac magnetic resonance imaging (MRI), as well as two additional patients (number of patients studied using cardiac MRI = 10). Plasma from the first cohort (N = 29) demonstrated intravascular haemolysis associated with a 12-fold increase in median nitric oxide (NO) consumption when compared with healthy volunteers (P < 0·001). Doppler echocardiography demonstrated normal left ventricular function and elevated pulmonary artery systolic pressure in 41% of patients. Cardiac MRI from the second cohort (N = 10) demonstrated depressed right ventricular function in 80% of PNH patients tested, and 60% had findings suggestive of subclinical small pulmonary emboli. Together, these data suggest a high prevalence of haemolysis-associated NO scavenging, Doppler-estimated systolic pulmonary hypertension, and depressed right ventricular function in patients with PNH.
© 2012 Blackwell Publishing Ltd.

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Year:  2012        PMID: 22639982     DOI: 10.1111/j.1365-2141.2012.09166.x

Source DB:  PubMed          Journal:  Br J Haematol        ISSN: 0007-1048            Impact factor:   6.998


  17 in total

1.  Significant hemolysis is not required for thrombosis in paroxysmal nocturnal hemoglobinuria.

Authors:  Morag Griffin; Peter Hillmen; Talha Munir; Stephen Richards; Louise Arnold; Kathryn Riley; Anita Hill
Journal:  Haematologica       Date:  2018-10-04       Impact factor: 9.941

2.  Paroxysmal nocturnal hemoglobinuria testing in patients with myelodysplastic syndrome in clinical practice-frequency and indications.

Authors:  S A Wong; B I Dalal; H A Leitch
Journal:  Curr Oncol       Date:  2018-10-31       Impact factor: 3.677

Review 3.  Management of thrombosis in paroxysmal nocturnal hemoglobinuria: a clinician's guide.

Authors:  Morag Griffin; Talha Munir
Journal:  Ther Adv Hematol       Date:  2016-12-21

Review 4.  Paroxysmal nocturnal hemoglobinuria.

Authors:  Robert A Brodsky
Journal:  Blood       Date:  2014-09-18       Impact factor: 22.113

5.  Hemolysis-Associated Nitric Oxide Dysregulation during Extracorporeal Membrane Oxygenation.

Authors:  Jason P Sulkowski; Jennifer N Cooper; Erik G Pearson; James T Connelly; Natalie Rintoul; Todd J Kilbaugh; Katherine J Deans; Peter C Minneci
Journal:  J Extra Corpor Technol       Date:  2014-09

Review 6.  Paroxysmal nocturnal haemoglobinuria.

Authors:  Anita Hill; Amy E DeZern; Taroh Kinoshita; Robert A Brodsky
Journal:  Nat Rev Dis Primers       Date:  2017-05-18       Impact factor: 52.329

Review 7.  Pulmonary hypertension associated with chronic hemolytic anemia and other blood disorders.

Authors:  Roberto F Machado; Harrison W Farber
Journal:  Clin Chest Med       Date:  2013-10-17       Impact factor: 2.878

8.  Long-term efficacy and safety of eculizumab in Japanese patients with PNH: AEGIS trial.

Authors:  Yuzuru Kanakura; Kazuma Ohyashiki; Tsutomu Shichishima; Shinichiro Okamoto; Kiyoshi Ando; Haruhiko Ninomiya; Tatsuya Kawaguchi; Shinji Nakao; Hideki Nakakuma; Jun-ichi Nishimura; Taroh Kinoshita; Camille L Bedrosian; Keiya Ozawa; Mitsuhiro Omine
Journal:  Int J Hematol       Date:  2013-08-11       Impact factor: 2.490

9.  Clinical signs and symptoms associated with increased risk for thrombosis in patients with paroxysmal nocturnal hemoglobinuria from a Korean Registry.

Authors:  Jong Wook Lee; Jun Ho Jang; Jin Seok Kim; Sung-Soo Yoon; Je-Hwan Lee; Yeo-Kyeoung Kim; Deog-Yeon Jo; Jooseop Chung; Sang Kyun Sohn
Journal:  Int J Hematol       Date:  2013-05-01       Impact factor: 2.490

10.  Intravascular hemolysis triggers ADP-mediated generation of platelet-rich thrombi in precapillary pulmonary arterioles.

Authors:  Tomasz Brzoska; Ravi Vats; Margaret F Bennewitz; Egemen Tutuncuoglu; Simon C Watkins; Margaret V Ragni; Matthew D Neal; Mark T Gladwin; Prithu Sundd
Journal:  JCI Insight       Date:  2020-07-23
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