Department of Radiology, Academic Medical Center, University of Amsterdam, Amsterdam, The Netherlands.
PLoS One. 2013;8(3):e57507. doi: 10.1371/journal.pone.0057507. Epub 2013 Mar 15.
Long term liver-related complications of type-1 Gaucher disease (GD), a lysosomal storage disorder, include fibrosis and an increased incidence of hepatocellular carcinoma. Splenectomy has been implicated as a risk factor for the development of liver pathology in GD. High ferritin concentrations are a feature of GD and iron storage in Gaucher cells has been described, but iron storage in the liver in relation to liver fibrosis has not been studied. Alternatively, iron storage in GD may be the result of iron supplementation therapy or regular blood transfusions in patients with severe cytopenia. In this pilot study, comprising 14 type-1 GD patients (7 splenectomized, 7 non-splenectomized) and 7 healthy controls, we demonstrate that liver stiffness values, measured by Transient Elastography and MR-Elastography, are significantly higher in splenectomized GD patients when compared with non-splenectomized GD patients (p = 0.03 and p = 0.01, respectively). Liver iron concentration was elevated (>60±30 µmol/g) in 4 GD patients of whom 3 were splenectomized. No relationship was found between liver stiffness and liver iron concentration. HFE gene mutations were more frequent in splenectomized (6/7) than in non-splenectomized (2/7) participants (p = 0.10). Liver disease appeared more advanced in splenectomized than in non-splenectomized patients. We hypothesize a relationship with excessive hepatic iron accumulation in splenectomized patients. We recommend that all splenectomized patients, especially those with evidence of substantial liver fibrosis undergo regular screening for HCC, according to current guidelines.
1 型戈谢病(Gaucher disease,GD)是一种溶酶体贮积症,其长期肝脏相关并发症包括纤维化和肝细胞癌发病率增加。脾切除术已被认为是 GD 发生肝脏病理的一个危险因素。铁蛋白浓度升高是 GD 的一个特征,已描述过戈谢细胞中的铁储存,但与肝纤维化相关的肝内铁储存尚未研究过。或者,GD 中的铁储存可能是由于严重细胞减少症患者的铁补充治疗或定期输血所致。在这项由 14 名 1 型 GD 患者(7 名脾切除术,7 名非脾切除术)和 7 名健康对照组成的初步研究中,我们证明,通过瞬时弹性成像和磁共振弹性成像测量的肝硬度值在脾切除术 GD 患者中明显高于非脾切除术 GD 患者(分别为 p = 0.03 和 p = 0.01)。4 名 GD 患者的肝铁浓度升高(>60±30µmol/g),其中 3 名接受了脾切除术。未发现肝硬度与肝铁浓度之间存在关系。脾切除术组(6/7)的 HFE 基因突变频率高于非脾切除术组(2/7)(p = 0.10)。脾切除术组的肝脏疾病似乎比非脾切除术组更严重。我们假设脾切除术组患者存在过度的肝铁积累。我们建议所有脾切除术患者,尤其是那些有明显肝纤维化证据的患者,根据当前指南,定期筛查 HCC。