Uderzo C, Locasciulli A, Rajnoldi A C, Mozzana R, Lambertenghi-Deliliers G, Masera G
Clinica Pediatrica, Università di Milano, Ospedale San Gerardo, Monza, Italy.
Med Pediatr Oncol. 1993;21(1):43-8. doi: 10.1002/mpo.2950210109.
Myelodisplastic syndromes (MDS) in childhood deserve a negative prognosis even though disease-free survival has been obtained in 20% of cases by using aggressive chemotherapy. We describe three children with refractory anemia with excess of blasts in transformation (RAEB-T) who underwent bone marrow transplantation (BMT). We also reviewed 21 additional cases (median age was 8 years) with primary MDS recently reported in the literature with the aim of clarifying the role of BMT in treating these patients. Twelve of the 24 children were long-term survivors and free from disease at a median time of 1,320 days (range 302-2,340). There were five relapses, two graft failures, two early deaths (one VOD, one severe GVHD), and three late deaths (two respiratory diseases, one severe GVHD). We didn't find any correlation between karyotype and outcome. In conclusion, so far BMT seems to be the most valid treatment of childhood primary MDS. However, since the major causes of failure were regimen-related toxicity or recurrence of the disease after BMT, it must be pointed out that, when a compatible donor even unrelated is available, BMT for childhood MDS should be given as soon as possible or at any rate prior to blastic crisis.
儿童骨髓增生异常综合征(MDS)预后不良,尽管采用积极化疗后20%的病例实现了无病生存。我们描述了3例转化型原始细胞增多的难治性贫血(RAEB-T)儿童接受了骨髓移植(BMT)。我们还回顾了文献中最近报道的另外21例原发性MDS病例(中位年龄8岁),旨在阐明BMT在治疗这些患者中的作用。24例儿童中有12例为长期存活者,中位1320天(范围302 - 2340天)无病生存。有5例复发、2例移植失败、2例早期死亡(1例肝静脉闭塞病、1例严重移植物抗宿主病)和3例晚期死亡(2例呼吸系统疾病、1例严重移植物抗宿主病)。我们未发现核型与预后之间存在任何相关性。总之,到目前为止,BMT似乎是儿童原发性MDS最有效的治疗方法。然而,由于失败的主要原因是与治疗方案相关的毒性或BMT后疾病复发,必须指出,当有合适供体(即使是无关供体)时,儿童MDS的BMT应尽早进行,无论如何应在原始细胞危象之前进行。