Dipartimento di Scienze Biomediche e Biotecnologie- Università di Cagliari, Ospedale Regionale, Microcitemie ASL Cagliari, Cagliari, Italy.
Orphanet J Rare Dis. 2010 May 21;5:11. doi: 10.1186/1750-1172-5-11.
Beta-thalassemias are a group of hereditary blood disorders characterized by anomalies in the synthesis of the beta chains of hemoglobin resulting in variable phenotypes ranging from severe anemia to clinically asymptomatic individuals. The total annual incidence of symptomatic individuals is estimated at 1 in 100,000 throughout the world and 1 in 10,000 people in the European Union. Three main forms have been described: thalassemia major, thalassemia intermedia and thalassemia minor. Individuals with thalassemia major usually present within the first two years of life with severe anemia, requiring regular red blood cell (RBC) transfusions. Findings in untreated or poorly transfused individuals with thalassemia major, as seen in some developing countries, are growth retardation, pallor, jaundice, poor musculature, hepatosplenomegaly, leg ulcers, development of masses from extramedullary hematopoiesis, and skeletal changes that result from expansion of the bone marrow. Regular transfusion therapy leads to iron overload-related complications including endocrine complication (growth retardation, failure of sexual maturation, diabetes mellitus, and insufficiency of the parathyroid, thyroid, pituitary, and less commonly, adrenal glands), dilated myocardiopathy, liver fibrosis and cirrhosis). Patients with thalassemia intermedia present later in life with moderate anemia and do not require regular transfusions. Main clinical features in these patients are hypertrophy of erythroid marrow with medullary and extramedullary hematopoiesis and its complications (osteoporosis, masses of erythropoietic tissue that primarily affect the spleen, liver, lymph nodes, chest and spine, and bone deformities and typical facial changes), gallstones, painful leg ulcers and increased predisposition to thrombosis. Thalassemia minor is clinically asymptomatic but some subjects may have moderate anemia. Beta-thalassemias are caused by point mutations or, more rarely, deletions in the beta globin gene on chromosome 11, leading to reduced (beta+) or absent (beta0) synthesis of the beta chains of hemoglobin (Hb). Transmission is autosomal recessive; however, dominant mutations have also been reported. Diagnosis of thalassemia is based on hematologic and molecular genetic testing. Differential diagnosis is usually straightforward but may include genetic sideroblastic anemias, congenital dyserythropoietic anemias, and other conditions with high levels of HbF (such as juvenile myelomonocytic leukemia and aplastic anemia). Genetic counseling is recommended and prenatal diagnosis may be offered. Treatment of thalassemia major includes regular RBC transfusions, iron chelation and management of secondary complications of iron overload. In some circumstances, spleen removal may be required. Bone marrow transplantation remains the only definitive cure currently available. Individuals with thalassemia intermedia may require splenectomy, folic acid supplementation, treatment of extramedullary erythropoietic masses and leg ulcers, prevention and therapy of thromboembolic events. Prognosis for individuals with beta-thalassemia has improved substantially in the last 20 years following recent medical advances in transfusion, iron chelation and bone marrow transplantation therapy. However, cardiac disease remains the main cause of death in patients with iron overload.
β-地中海贫血是一组遗传性血液疾病,其特征是血红蛋白β链合成异常,导致表型从严重贫血到临床无症状个体不等。全球每年有症状个体的总发病率估计为每 10 万人中有 1 例,欧盟每 1 万人中有 1 例。已经描述了三种主要形式:重型β-地中海贫血、中间型β-地中海贫血和轻型β-地中海贫血。重型β-地中海贫血患者通常在生命的头两年内出现严重贫血,需要定期输注红细胞(RBC)。在一些发展中国家,未治疗或输血不充分的重型β-地中海贫血患者会出现生长迟缓、苍白、黄疸、肌肉无力、肝脾肿大、腿部溃疡、骨髓外造血引起的肿块形成以及骨骼变化,这些变化是由于骨髓扩张引起的。定期输血治疗会导致铁过载相关并发症,包括内分泌并发症(生长迟缓、性成熟失败、糖尿病、甲状旁腺、甲状腺、垂体功能减退,以及较少见的肾上腺功能减退)、扩张型心肌病、肝纤维化和肝硬化)。中间型β-地中海贫血患者在生命后期出现中度贫血,不需要定期输血。这些患者的主要临床特征是红髓增生伴有骨髓和骨髓外造血及其并发症(骨质疏松症、主要影响脾脏、肝脏、淋巴结、胸部和脊柱的红系组织肿块以及骨骼畸形和典型的面部变化)、胆石症、腿部疼痛性溃疡和增加血栓形成倾向。轻型β-地中海贫血无症状,但部分患者可能有中度贫血。β-地中海贫血是由β珠蛋白基因上的点突变或更罕见的缺失引起的,导致β珠蛋白链(Hb)的合成减少(β+)或缺失(β0)。其遗传方式为常染色体隐性遗传;然而,也有报道称存在显性突变。β-地中海贫血的诊断基于血液学和分子遗传学检测。鉴别诊断通常很直接,但可能包括遗传性铁粒幼细胞贫血、先天性红细胞生成异常性贫血和其他 HbF 水平较高的疾病(如青少年骨髓单核细胞白血病和再生障碍性贫血)。建议进行遗传咨询,可提供产前诊断。重型β-地中海贫血的治疗包括定期输注红细胞、铁螯合和治疗铁过载的继发性并发症。在某些情况下,可能需要切除脾脏。骨髓移植仍然是目前唯一可用的根治方法。中间型β-地中海贫血患者可能需要切除脾脏、补充叶酸、治疗骨髓外红系肿块和腿部溃疡、预防和治疗血栓栓塞事件。近年来,输血、铁螯合和骨髓移植治疗方面的进展极大地改善了β-地中海贫血患者的预后。然而,心脏疾病仍然是铁过载患者死亡的主要原因。