地中海贫血

Thalassemia.

作者信息

Cohen Alan R, Galanello Renzo, Pennell Dudley J, Cunningham Melody J, Vichinsky Elliott

出版信息

Hematology Am Soc Hematol Educ Program. 2004:14-34. doi: 10.1182/asheducation-2004.1.14.

Abstract

New developments in the epidemiology, treatment and prognosis of thalassemia have dramatically altered the approach to the care of affected patients, and these developments are likely to have an even greater impact in the next few years. Demographic changes have required an awareness and understanding of the unique features of thalassemia disorders that were previously uncommon in North America but are now seen more frequently in children and recognized more consistently in adults. New methods for measuring tissue iron accumulation and new drugs to remove excessive iron are advancing two of the most challenging areas in the management of thalassemia as well as other transfusion-dependent disorders. Improved survival of patients with thalassemia has given new importance to adult complications such as endocrinopathies and hepatitis that have a major impact on the quality of life. This chapter describes how these changes are redefining the clinical management of thalassemia. In Section I, Dr. Renzo Galanello describes recent advances in iron chelation therapy. Several new chelators are either licensed in some countries, are in clinical trials or are in the late stages of preclinical development. Some of these iron chelators, such as deferiprone (DFP) and ICL670, are orally active. Others, such as hydroxybenzyl-ethylenediamine-diacetic acid (HBED) and starch deferoxamine, require parenteral administration but may be effective with less frequent administration than is currently required for deferoxamine. Chelation therapy employing two chelators offers the possibility of more effective removal of iron without compromising safety or compliance. Other strategies for chelation therapy may take advantage of the ability of particular chelators to remove iron from specific target organs such as the heart and the liver. In Section II, Dr. Dudley Pennell addresses cardiac iron overload, the most frequent cause of death from chronic transfusion therapy. The cardiac complications related to excessive iron may result from long-term iron deposition in vulnerable areas or may be due to the more immediate effects of nontransferrin-bound iron. Cardiac disease is reversible in some patients with intensive iron chelation therapy, but identification of cardiac problems prior to the onset of serious arrhythmias or congestive heart failure has proven difficult. New methods using magnetic resonance imaging (MRI) have recently been developed to assess cardiac iron loading, and studies suggest a clinically useful relationship between the results using these techniques and critical measures of cardiac function. Measurements such as T2* may help guide chelation therapy in individual patients and may also enhance the assessment of new chelators in clinical trials. The use of MRI-based technology also holds promise for wider application of non-invasive assessment of cardiac iron in the management of patients with thalassemia. In Section III, Dr. Melody Cunningham describes some of the important complications of thalassemia that are emerging as patients survive into adulthood. Hepatitis C infection is present in the majority of patients older than 25 years. However, antiviral therapy in patients with thalassemia has been held back by the absence of large clinical trials and concern about ribavirin-induced hemolysis. More aggressive approaches to the treatment of hepatitis C may be particularly valuable because of the additive risks for cirrhosis and hepatocellular carcinoma that are posed by infection and iron overload. Thrombosis is recognized with increasing frequency as a significant complication of thalassemia major and thalassemia intermedia, and pulmonary hypertension is now the focus of intense study. Risk factors for thrombosis such as splenectomy are being identified and new approaches to anticoagulation are being initiated. Pregnancies in women with thalassemia are increasingly common with and without hormonal therapy, and require a better understanding of the risks of iron overload and cardiac disease in the mother and exposure of the fetus to iron chelators. In Section IV, Dr. Elliott Vichinsky describes the dramatic changes in the epidemiology of thalassemia in North America. Hemoglobin E-beta thalassemia is seen with increasing frequency and poses a particular challenge because of the wide variability in clinical severity. Some affected patients may require little or no intervention, while others need chronic transfusion therapy and may be appropriate candidates for hematopoietic stem cell transplantation. Enhancers of fetal hemoglobin production may have a unique role in Hb E-beta thalassemia since a modest increase in hemoglobin level may confer substantial clinical benefits. Alpha thalassemia is also being recognized with increasing frequency in North America, and newborn screening for Hemoglobin Barts in some states is leading to early detection of Hb H disease and Hb H Constant Spring. New data clarify the importance of distinguishing these two disorders because of the increased severity associated with Hb H Constant Spring. The use of intrauterine transfusions to sustain the viability of fetuses with homozygous alpha thalassemia has created a new population of patients with severe thalassemia and has raised new and complex issues in genetic counseling for parents with alpha thalassemia trait.

摘要

地中海贫血在流行病学、治疗和预后方面的新进展显著改变了对患者的护理方式,并且这些进展在未来几年可能会产生更大的影响。人口结构的变化要求人们认识和理解地中海贫血症的独特特征,这些特征以前在北美并不常见,但现在在儿童中更为常见,在成人中也得到了更一致的认可。测量组织铁蓄积的新方法和去除过量铁的新药正在推动地中海贫血以及其他依赖输血疾病管理中两个最具挑战性的领域的发展。地中海贫血患者生存率的提高使成人并发症(如内分泌疾病和肝炎)具有了新的重要性,这些并发症对生活质量有重大影响。本章描述了这些变化如何重新定义地中海贫血的临床管理。在第一部分,伦佐·加拉内洛博士描述了铁螯合疗法的最新进展。几种新的螯合剂在一些国家已获许可,正在进行临床试验或处于临床前开发的后期阶段。其中一些铁螯合剂,如去铁酮(DFP)和ICL670,具有口服活性。其他的,如羟基苄基乙二胺二乙酸(HBED)和淀粉去铁胺,需要肠胃外给药,但可能比目前去铁胺所需的给药频率更低也有效。采用两种螯合剂的螯合疗法提供了在不影响安全性或依从性的情况下更有效去除铁的可能性。螯合疗法的其他策略可能利用特定螯合剂从心脏和肝脏等特定靶器官去除铁的能力。在第二部分,达德利·彭内尔博士论述了心脏铁过载,这是慢性输血治疗最常见的死亡原因。与过量铁相关的心脏并发症可能是由于长期铁沉积在易损区域,或者可能是由于非转铁蛋白结合铁的更直接影响。在一些接受强化铁螯合疗法的患者中,心脏疾病是可逆的,但在严重心律失常或充血性心力衰竭发作之前识别心脏问题已被证明很困难。最近开发了使用磁共振成像(MRI)的新方法来评估心脏铁负荷,研究表明使用这些技术的结果与心脏功能的关键指标之间存在临床上有用的关系。诸如T2*等测量可能有助于指导个体患者的螯合疗法,也可能加强对临床试验中新螯合剂的评估。基于MRI的技术的应用也有望在更广泛地应用于地中海贫血患者管理中对心脏铁进行无创评估。在第三部分,梅洛迪·坎宁安博士描述了随着患者存活至成年而出现的地中海贫血的一些重要并发症。丙型肝炎感染在大多数25岁以上的患者中存在。然而,由于缺乏大型临床试验以及对利巴韦林诱导的溶血的担忧,地中海贫血患者的抗病毒治疗受到了阻碍。由于感染和铁过载对肝硬化和肝细胞癌构成的附加风险,更积极的丙型肝炎治疗方法可能特别有价值。血栓形成作为重型地中海贫血和中间型地中海贫血的一种重要并发症越来越频繁地被认识到,肺动脉高压现在是深入研究的焦点。正在确定诸如脾切除术等血栓形成的危险因素,并正在启动新的抗凝方法。无论有无激素治疗,地中海贫血女性的怀孕越来越普遍,这需要更好地了解母亲中铁过载和心脏疾病的风险以及胎儿接触铁螯合剂的情况。在第四部分,埃利奥特·维钦斯基博士描述了北美地中海贫血流行病学的巨大变化。血红蛋白E-β地中海贫血的发病率越来越高,由于临床严重程度差异很大,这带来了特殊的挑战。一些受影响的患者可能几乎不需要或不需要干预,而另一些患者需要慢性输血治疗,可能是造血干细胞移植的合适候选者。胎儿血红蛋白生成增强剂在血红蛋白E-β地中海贫血中可能具有独特作用,因为血红蛋白水平的适度增加可能带来实质性的临床益处。α地中海贫血在北美也越来越频繁地被认识到,一些州对血红蛋白巴氏的新生儿筛查正在导致Hb H病和Hb H恒河猴型的早期检测。新的数据阐明了区分这两种疾病的重要性,因为与Hb H恒河猴型相关的严重程度增加。使用宫内输血来维持纯合子α地中海贫血胎儿的生存能力产生了一群患有严重地中海贫血的新患者,并在为具有α地中海贫血特征的父母进行遗传咨询时提出了新的复杂问题。

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