Hassan Fawaz, Mohammad Hassan Hodroj, Nicole Charbel, Sacha El Khoury, Ali Taher
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引用次数: 0
摘要
地中海贫血是一种遗传性血红蛋白病,具有显著的临床变异性,主要受潜在基因突变的影响。我们报告了一位47岁的β-地中海贫血女性患者,其β-珠蛋白基因启动子HBB: C - 136c b> G (-86 C > G)存在罕见的纯合突变。患者对羟基脲治疗表现出明显的临床反应,血红蛋白水平明显升高,脾脏大小明显减小,髓外造血引起的疲劳和骨痛有所改善。本报告强调了基因表征在了解罕见地中海贫血形式中的作用,以及羟基脲作为具有独特遗传决定因素的患者的个性化治疗策略的潜力。
Therapeutic Response to Hydroxyurea in Beta-Thalassemia Intermedia with Rare Mutation: A Case Report.
Beta-thalassemia is a hereditary hemoglobinopathy characterized by significant clinical variability, largely influenced by the underlying genetic mutations. We report a 47-year-old female patient with β-thalassemia intermedia harboring a rare homozygous mutation in the β-globin gene promoter: HBB:c.-136C > G (-86 C > G). The patient showed marked clinical response to hydroxyurea therapy with a notable increase in hemoglobin levels, reduction in spleen size and improvement of fatigue and bone pain due to extramedullary hematopoiesis. This report highlights the role of genetic characterization in understanding rare forms of thalassemia and the potential of hydroxyurea as a personalized treatment strategy for patients with unique genetic determinants.
期刊介绍:
Hemoglobin is a journal in the English language for the communication of research and information concerning hemoglobin in humans and other species. Hemoglobin publishes articles, reviews, points of view
The journal covers topics such as:
structure, function, genetics and evolution of hemoglobins
biochemical and biophysical properties of hemoglobin molecules
characterization of hemoglobin disorders (variants and thalassemias),
consequences and treatment of hemoglobin disorders
epidemiology and prevention of hemoglobin disorders (neo-natal and adult screening)
modulating factors
methodology used for diagnosis of hemoglobin disorders