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诊断戈谢病:血液学家需要不断提高认识。

Diagnosing Gaucher disease: an on-going need for increased awareness amongst haematologists.

机构信息

Lysosomal Storage Disorders Unit, Department of Academic Haematology, Royal Free Hospital and University College Medical School, London, UK.

出版信息

Blood Cells Mol Dis. 2013 Mar;50(3):212-7. doi: 10.1016/j.bcmd.2012.11.004. Epub 2012 Dec 6.

Abstract

Gaucher disease (GD) is an inherited enzyme deficiency characterised by progressive cytopenias, hepatosplenomegaly and destructive bone disease. It is diagnosed by demonstration of beta glucosidase deficiency but may be suspected in presence of abnormal storage cells on tissue biopsy. Specific treatment is available in the form of enzyme replacement (ERT) and is effective in reversing many disease features. Delayed treatment has been associated with increased disease complications. This retrospective review of a single centre cohort of 86 patients was undertaken to ascertain if the diagnostic journey had improved since the introduction of ERT and commissioning of services. Fifty-six percent of patients presented primarily with features related to thrombocytopenia or splenomegaly with a median time from symptom onset to diagnosis of 2years (range 0.5-26years), 19% experiencing delays of 5 or more years. Seventy-five percent of patients were diagnosed by haematologists, 68% following an abnormal bone marrow biopsy. Raised serum ACE levels, low HDL cholesterol and raised ferritin were identified as prevalent laboratory abnormalities at the time of diagnosis. These features, coupled with the relative preservation of haemoglobin and white cell counts compared to the platelet count, help identify patients presenting to haematologists with a possible diagnosis of GD earlier in the diagnostic pathway.

摘要

戈谢病(GD)是一种遗传性酶缺乏症,其特征为进行性细胞减少症、肝脾肿大和破坏性骨病。通过β葡萄糖苷酶缺乏的证明可以诊断,但在组织活检中存在异常储存细胞时可能怀疑。可用酶替代治疗(ERT)进行特异性治疗,并且在逆转许多疾病特征方面有效。延迟治疗与疾病并发症的增加有关。本回顾性研究对 86 例单中心患者队列进行了研究,以确定自 ERT 引入和服务委托以来诊断过程是否有所改善。56%的患者主要表现为与血小板减少症或脾肿大相关的特征,从症状发作到诊断的中位数时间为 2 年(范围 0.5-26 年),19%的患者延迟超过 5 年。75%的患者由血液学家诊断,68%的患者在异常骨髓活检后诊断。在诊断时,发现血清 ACE 水平升高、高密度脂蛋白胆固醇降低和铁蛋白升高是常见的实验室异常。这些特征,加上与血小板计数相比血红蛋白和白细胞计数的相对保留,有助于识别在诊断途径中更早地向血液学家提出可能的 GD 诊断的患者。

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