Kawahara Shimpei, Kato Atushi, Oshimi Kazuo, Ida Hiroyuki
Department of Hematology, Juntendo University School of Medicine.
Rinsho Ketsueki. 2008 May;49(5):335-9.
A 63-year-old man who was incidentally found to have thrombocytopenia at a periodic physical examination visited our hospital. The spleen was palpable 3 finger-breadths below the navel level, and the liver was palpable 1 finger-breadth below the right costal margin. Peripheral blood examination showed WBC 2,900/microl, Hb 13.4 g/dl, and platelets 54 X 10(3)/ microl. Gaucher cells were recognized in the bone marrow by aspiration, and serum levels of total acid phosphatase and angiotensin converting enzyme were increased. Glucocerebrosidase activity was lower than the control level in bone marrow stroma cells, and modification of glucocerebrosidase genotype N188S was shown, which had been identified in the past. Furthermore, neurological examination was normal. Based on these results, we diagnosed the patient with Gaucher disease type I, and started enzyme replacement therapy. Gaucher disease is rare in Japanese, approximately 100 cases having been reported; diagnosis at older age is also relatively rare and, as far as we know, the oldest age reported in Japanese was 57 years old. Gaucher disease should be considered a differential diagnosis when thrombocytopenia and splenomegaly are found in elderly patients, although it is relatively rare.
一名63岁男性在定期体检时偶然发现血小板减少,遂来我院就诊。脾脏在脐水平以下3指宽处可触及,肝脏在右肋缘下1指宽处可触及。外周血检查显示白细胞2900/微升,血红蛋白13.4克/分升,血小板54×10³/微升。骨髓穿刺可见戈谢细胞,血清总酸性磷酸酶和血管紧张素转换酶水平升高。骨髓基质细胞中葡萄糖脑苷脂酶活性低于对照水平,且显示出过去已鉴定出的葡萄糖脑苷脂酶基因型N188S的改变。此外,神经学检查正常。基于这些结果,我们诊断该患者为Ⅰ型戈谢病,并开始酶替代治疗。戈谢病在日本人中较为罕见,据报道约有100例;老年患者的诊断也相对少见,据我们所知,日本人中报道的最高年龄为57岁。尽管戈谢病相对罕见,但在老年患者中发现血小板减少和脾肿大时,应考虑将其作为鉴别诊断。