Kitazume K, Usuki K, Endo M, Osawa M, Iki S, Chiba S, Hirai H, Matsuya S, Urabe A
Division of Hematology, Kanto Teishin Hospital.
Rinsho Ketsueki. 1998 Sep;39(9):686-91.
In October 1992, a 36-year-old man was diagnosed as having mediastinum mixed germ cell tumor (stage II), and was treated with surgical operation and combination chemotherapy including VP16 (total VP16 dose; 1,500 mg/m2). After that, remission had been sustained, but leukocytosis (15,700/microliter) with 37% of peroxidase-negative blasts and thrombocytopenia developed in September, 1995. Bone marrow showed remarkable reticulin fibrosis and increase of atypical immature cells that were immunophenotypically factor VIII+/CD42+/CD61+. Thus, we diagnosed acute megakaryoblastic leukemia (M7). Based on no abnormality of chromosome 11q23 and no rearrangements of MLL gene, we diagnosed the syndrome of mediastinal germ-cell tumors associated with hematologic neoplasia. Furthermore, the neuron-specific enolase level was elevated (95.9 ng/ml). Soon after complete remission was reached by combination chemotherapy, the leukemia was relapsed, and the he died 3 months after the onset of leukemia. To our knowledge, this is the third case report of this syndrome in Japan and the first one of leukemia with high level of serum neuron-specific enolase.
1992年10月,一名36岁男性被诊断为纵隔混合性生殖细胞肿瘤(II期),接受了手术及包括VP16(VP16总剂量;1500mg/m²)在内的联合化疗。此后,病情持续缓解,但1995年9月出现白细胞增多(15700/微升),过氧化物酶阴性原始细胞占37%,并伴有血小板减少。骨髓显示明显的网状纤维组织增生,非典型未成熟细胞增多,免疫表型为因子VIII+/CD42+/CD61+。因此,我们诊断为急性巨核细胞白血病(M7)。基于11q23染色体无异常及MLL基因无重排,我们诊断为纵隔生殖细胞肿瘤合并血液系统肿瘤综合征。此外,神经元特异性烯醇化酶水平升高(95.9ng/ml)。联合化疗达到完全缓解后不久,白血病复发,患者在白血病发病3个月后死亡。据我们所知,这是日本该综合征的第三例报告,也是血清神经元特异性烯醇化酶水平升高的白血病的首例报告。