Meloni Antonella, Pistoia Laura, Ricchi Paolo, Putti Maria Caterina, Gamberini Maria Rita, Cuccia Liana, Messina Giuseppe, Massei Francesco, Facchini Elena, Righi Riccardo, Renne Stefania, Peritore Giuseppe, Positano Vincenzo, Cademartiri Filippo
Department of Radiology, Fondazione G. Monasterio CNR-Regione Toscana, 56124 Pisa, Italy.
U.O.C. Bioingegneria, Fondazione G. Monasterio CNR-Regione Toscana, 56124 Pisa, Italy.
J Pers Med. 2022 Mar 4;12(3):400. doi: 10.3390/jpm12030400.
We evaluated the impact of the genotype on hepatic, pancreatic and myocardial iron content, and on hepatic, cardiac and endocrine complications in children with transfusion-dependent β-thalassemia (β-TDT). We considered 68 β-TDT patients (11.98 ± 3.67 years, 51.5% females) consecutively enrolled in the Extension-Myocardial Iron Overload in Thalassemia network. Iron overload was quantified by T2* technique and biventricular function by cine images. Replacement myocardial fibrosis was evaluated by late gadolinium enhancement technique. Three groups of patients were identified: homozygous β+ (N = 19), compound heterozygous β0β+ (N = 24), and homozygous β0 (N = 25). The homozygous β0 group showed significantly lower global heart and pancreas T2* values than the homozygous β+ group. Compared to patients with homozygous β+ genotype, β0β+ as well as β0β0 patients were more likely to have pancreatic iron overload (odds ratio = 6.53 and 10.08, respectively). No difference was detected in biventricular function parameters and frequency of replacement fibrosis. No patient had cirrhosis/fibrosis, diabetes or heart failure, and the frequency of endocrinopathies was comparable among the groups. In pediatric β-TDT patients, there is an association between genotype and cardiac and pancreatic iron overload. The knowledge of patients' genotype can be valuable in predicting some patients' phenotypic features and in helping the clinical management of β-TDT patients.
我们评估了基因型对输血依赖型β地中海贫血(β-TDT)患儿肝脏、胰腺和心肌铁含量以及肝脏、心脏和内分泌并发症的影响。我们纳入了连续入选地中海贫血网络中“心肌铁过载扩展研究”的68例β-TDT患者(年龄11.98±3.67岁,女性占51.5%)。采用T2技术定量铁过载,通过电影图像评估双心室功能。采用延迟钆增强技术评估替代性心肌纤维化。确定了三组患者:纯合子β+(N = 19)、复合杂合子β0β+(N = 24)和纯合子β0(N = 25)。纯合子β0组的整体心脏和胰腺T2值显著低于纯合子β+组。与纯合子β+基因型患者相比,β0β+以及β0β0患者更易出现胰腺铁过载(比值比分别为6.53和10.08)。双心室功能参数和替代性纤维化频率未检测到差异。没有患者出现肝硬化/纤维化、糖尿病或心力衰竭,各亚组内分泌疾病的发生率相当。在儿童β-TDT患者中,基因型与心脏和胰腺铁过载之间存在关联。了解患者的基因型对于预测某些患者的表型特征以及帮助β-TDT患者的临床管理可能具有重要价值。