Huang M T, Chou C C, Jou S T, Lin K H, Hsieh K H
Department of Pediatrics, College of Medicine, National Taiwan University Hospital, Taipei.
Asian Pac J Allergy Immunol. 1996 Jun;14(1):57-63.
We describe the successful use of HLA-compatible sibling bone marrow transplantation (BMT) in a 17-month-old Chinese boy in whom Wiskott-Aldrich syndrome (WAS) was diagnosed on the basis of eczema, thrombocytopenia, recurrent otitis media and abnormal immunological tests. The conditioning chemotherapy included 2 days' oral busulfan, 40 mg/m2/6 hours, and 2 days' intravenous cyclophosphamide, 60 mg/kg/day (BU2CY2). Complete hematological chimerism was achieved 3 weeks after transplantation. Eight months after his BMT the eczema has resolved, platelet count is normal, and he no longer has frequent infections. BU2CY2 as a preconditioning regimen gave complete lymphohematopoietic engraftment in this WAS patient with no evidence of graft-versus-host disease. The excellent clinical response of this patient and the inevitable fatal outcome of WAS support the opinion that where a histocompatible donor is available, BMT at the earliest opportunity is the best option. We believe this is the first case of successful BMT in a Chinese patient with WAS.
我们描述了一名17个月大的中国男孩成功接受人类白细胞抗原(HLA)相合的同胞骨髓移植(BMT)的案例。该男孩根据湿疹、血小板减少、复发性中耳炎及异常免疫测试被诊断为维斯科特-奥尔德里奇综合征(WAS)。预处理化疗包括连续2天口服白消安,40mg/m²/6小时,以及连续2天静脉注射环磷酰胺,60mg/kg/天(BU2CY2)。移植后3周实现了完全血液学嵌合。骨髓移植8个月后,湿疹消退,血小板计数正常,且他不再频繁感染。作为预处理方案,BU2CY2使该WAS患者实现了完全淋巴细胞造血植入,且无移植物抗宿主病迹象。该患者出色的临床反应以及WAS不可避免的致命结局支持了以下观点:在有组织相容性供者的情况下,尽早进行骨髓移植是最佳选择。我们认为这是中国首例WAS患者成功接受骨髓移植的病例。