Patton W N, Bunce C M, Larkins S, Brown G
Department of Haematology, Medical School, University of Birmingham, UK.
Br J Cancer. 1991 Jul;64(1):128-31. doi: 10.1038/bjc.1991.255.
A case of primary myelofibrosis was identified with a previously unreported complex karyotype with two abnormal clones in addition to a proportion of normal cells: 46,XY,-2,-11, + der(2)t(2;11) (q24/31;q13), + mar and 45,XY,-2,-11, + der(2)t(2;11)(q24/31;q13), + mar, -17, del(7q). Study of circulating committed progenitors from this patient consistently showed (1) an absence of erythroid progenitors which is uncommon and (2) greatly increased granulocyte-monocyte progenitors (CFU-GM) which is generally observed in myelofibrosis. Further study showed that peripheral blood mononuclear cells co-cultured with irradiated normal bone marrow stroma generated increased numbers of CFU-GM compared with controls but failed to generate erythroid progenitors, providing evidence for an intrinsic defect in erythropoiesis. Only once previously has the absence of erythroid progenitors in primary myelofibrosis been studied in relation to cytogenetic abnormalities. This case also revealed a complex karyotype which, however, shared with our case a defect on chromosome 11. The identification of two cases of primary myelofibrosis which lack committed erythroid progenitor cells and which show in common a chromosomal defect on chromosome 11 point to the existence of genes on this chromosome which play a key role during erythropoiesis.
确诊1例原发性骨髓纤维化患者,其核型复杂,此前未见报道,除一部分正常细胞外,还有两个异常克隆:46,XY,-2,-11,+der(2)t(2;11)(q24/31;q13),+mar和45,XY,-2,-11,+der(2)t(2;11)(q24/31;q13),+mar,-17,del(7q)。对该患者循环定向祖细胞的研究持续显示:(1) 红系祖细胞缺失,这并不常见;(2) 粒-单核祖细胞(CFU-GM)大幅增加,这在骨髓纤维化中通常可见。进一步研究表明,与经辐照的正常骨髓基质共培养的外周血单个核细胞与对照组相比,产生的CFU-GM数量增加,但未能产生红系祖细胞,这为红细胞生成的内在缺陷提供了证据。原发性骨髓纤维化中红系祖细胞缺失与细胞遗传学异常相关的情况此前仅研究过一次。该病例也显示了复杂的核型,不过与我们的病例一样,11号染色体存在缺陷。两例原发性骨髓纤维化患者均缺乏定向红系祖细胞且11号染色体存在共同的染色体缺陷,这表明该染色体上存在在红细胞生成过程中起关键作用的基因。