Guilpain P, Montani D, Damaj G, Achouh L, Lefrère F, Le Pavec J, Marfaing-Koka A, Dartevelle P, Simonneau G, Humbert M, Hermine O
Department of Clinical Hematology, Hôpital Necker-Enfants-Malades, Assistance Publique - Hôpitaux de Paris, Université Paris-V, Paris, France.
Respiration. 2008;76(3):295-302. doi: 10.1159/000112822. Epub 2007 Dec 21.
Pulmonary hypertension (PH) is a severe hemodynamic disorder in which the pulmonary artery pressure is persistently elevated, leading to right-sided heart failure. Some studies have suggested an association between PH and myeloproliferative diseases (MPD).
This study describes clinical, hematological and hemodynamic characteristics of PH associated with MPD.
We retrospectively reviewed 10 cases of PH associated with MPD: polycythemia vera (8 patients) and essential thrombocythemia (2 patients), followed between 1993 and 2002. The baseline evaluation was established by right-sided heart catheterization, ventilation/perfusion lung scan and pulmonary angiography if required.
Six patients had confirmed chronic thromboembolic pulmonary hypertension (CTEPH) and 4 had pulmonary arterial hypertension (PAH) associated with MPD without other risk factors for PAH. The hemodynamic characteristics of CTEPH and PAH associated with MPD were similar. The diagnosis of CTEPH was concomitant to that of MPD in all cases (5 polycythemia vera and 1 essential thrombocythemia). The PAH associated with MPD occurred later in the evolution of the MPD (3 polycythemia vera and 1 essential thrombocythemia) with a median of 162 months after the diagnosis of MPD, and it was associated with myeloid metaplasia (p < 0.01).
We describe 2 distinct forms of PH in the context of MPD: CTEPH, which is diagnosed at an early stage of the MPD, and PAH, which occurs later in the course of the MPD and is associated with myeloid metaplasia. Progressively increasing dyspnea in a patient with an MPD warrants further investigation to rule out PAH and CTEPH, while a diagnosis of CTEPH warrants ruling out MPD.
肺动脉高压(PH)是一种严重的血流动力学紊乱疾病,其中肺动脉压力持续升高,导致右心衰竭。一些研究表明PH与骨髓增殖性疾病(MPD)之间存在关联。
本研究描述了与MPD相关的PH的临床、血液学和血流动力学特征。
我们回顾性分析了1993年至2002年间随访的10例与MPD相关的PH病例:真性红细胞增多症(8例患者)和原发性血小板增多症(2例患者)。必要时通过右心导管检查、通气/灌注肺扫描和肺血管造影进行基线评估。
6例患者确诊为慢性血栓栓塞性肺动脉高压(CTEPH),4例患有与MPD相关的肺动脉高压(PAH),且无其他PAH危险因素。与MPD相关的CTEPH和PAH的血流动力学特征相似。所有病例(5例真性红细胞增多症和1例原发性血小板增多症)中CTEPH的诊断与MPD同时出现。与MPD相关的PAH在MPD病程中出现较晚(3例真性红细胞增多症和1例原发性血小板增多症),MPD诊断后中位时间为162个月,且与髓外化生相关(p<0.01)。
我们描述了在MPD背景下的两种不同形式的PH:CTEPH,在MPD早期阶段被诊断;PAH,在MPD病程后期出现且与髓外化生相关。MPD患者进行性加重的呼吸困难需要进一步检查以排除PAH和CTEPH,而CTEPH的诊断需要排除MPD。