de Planque M M, Kluin-Nelemans H C, van Krieken H J, Kluin P M, Brand A, Beverstock G C, Willemze R, van Rood J J
Department of Haematology, University Medical Centre, Leiden, The Netherlands.
Br J Haematol. 1988 Sep;70(1):55-62. doi: 10.1111/j.1365-2141.1988.tb02434.x.
Myelodysplasia (MDS) and leukaemia following acquired aplastic anaemia has been reported as a rare event occurring in about 5% of patients. Improved results in survival of patients with severe aplastic anaemia (SAA) and subsequent prolonged follow-up created the possibility of evaluating the occurrence of MDS and leukaemia in 38 adult patients with acquired SAA surviving two or more years without bone marrow transplantation. Five patients, age 22, 35, 47, 56, 72 years, two females, three males, all with idiopathic SAA and normal cytogenetic analysis developed a refractory anaemia (RA) 7, 30, 48, 56, 142 months after diagnosis of SAA. In 3/5 RA evolved into an acute myeloid leukaemia (AML) either via a chronic myelomonocytic leukaemia (CMML) (2/3) or via RA with excess of blasts (RAEB) (1/3). Three patients revealed a monosomy 7 during MDS and/or leukaemic phase. One patient died during RA phase without cytogenetic abnormalities. A pattern of evolution could be identified in these patients revealing well-documented SAA - improvement of bone marrow haematopoiesis - dyshaematopoietic features of one or more cell lines with predominance of dyserythropoiesis - RA - RAEB or CMML - AML. These five patients represent more than 10% of all patients surviving at least 2 years. This implies that the risk of developing MDS and leukaemia in SAA patients surviving with autologous marrow, might increase with longer follow-up.
再生障碍性贫血后发生骨髓增生异常综合征(MDS)和白血病已被报道为一种罕见事件,约5%的患者会出现。严重再生障碍性贫血(SAA)患者生存率的提高以及随后延长的随访,使得评估38例未接受骨髓移植且存活两年或更长时间的获得性SAA成年患者中MDS和白血病的发生情况成为可能。5例患者,年龄分别为22岁、35岁、47岁、56岁、72岁,2例女性,3例男性,均为特发性SAA且细胞遗传学分析正常,在SAA诊断后7、30、48、56、142个月发展为难治性贫血(RA)。在这5例患者中,有3例RA通过慢性粒单核细胞白血病(CMML)(2/3)或伴有过多原始细胞的RA(RAEB)(1/3)演变为急性髓系白血病(AML)。3例患者在MDS和/或白血病期出现7号染色体单体。1例患者在RA期死亡,无细胞遗传学异常。在这些患者中可以确定一种演变模式,即有充分记录的SAA - 骨髓造血改善 - 一种或多种细胞系的造血异常特征,以红细胞生成异常为主 - RA - RAEB或CMML - AML。这5例患者占所有存活至少2年患者的10%以上。这意味着,自体骨髓存活的SAA患者发生MDS和白血病的风险可能会随着随访时间的延长而增加。