Nakao Kensuke, Oka Satoshi, Utsumi Takahiko, Yamada Seiko, Kondo Toshinori, Tohyama Kaoru, Asagoe Kohsuke
Department of Hematology/Oncology, Shiga General Hospital.
Department of Laboratory Medicine/Laboratory Hematology, Kawasaki Medical School.
Rinsho Ketsueki. 2019;60(8):915-919. doi: 10.11406/rinketsu.60.915.
A 83-year-old female patient was admitted to our hospital due to hematological manifestation of juvenile granulocytes and macrocytic anemia. Bone marrow (BM) examination revealed erythroid dysplasia and cytoplasmic blasts, and hence the patient was diagnosed with myelodysplastic syndrome with ring sideroblasts and with single lineage dysplasia (MDS-RS-SLD). Erythrocyte transfusion was performed as a supportive therapy, and there was a gradual increase in the number of blood cells. Therefore, BM re-examination was performed and it was confirmed that the number of megakaryocytes increased, so the patient's condition was determined as myelodysplastic/myeloproliferative neoplasms with ring sideroblasts and thrombocytosis (MDS/MPN with RS-T). Incidentally, gene mutation analysis showed CALR gene mutation. Thereafter, administration of hydroxycarbamide and anagrelide did not show adverse events and complications, and a good blood count control was obtained. Furthermore, it was also confirmed that an SF3B1 gene mutation is highly positive in MDS-RS. There was no report on CALR-mutant MDS/MPN in Japan, and it is a rare disease overseas.
一名83岁女性患者因出现幼稚粒细胞和大细胞性贫血的血液学表现而入住我院。骨髓(BM)检查显示红系发育异常和细胞质母细胞,因此该患者被诊断为伴有环形铁粒幼细胞和单系发育异常的骨髓增生异常综合征(MDS-RS-SLD)。作为支持治疗进行了红细胞输血,血细胞数量逐渐增加。因此,进行了骨髓复查,证实巨核细胞数量增加,所以患者的病情被确定为伴有环形铁粒幼细胞和血小板增多症的骨髓增生异常/骨髓增殖性肿瘤(MDS/MPN伴RS-T)。偶然发现,基因突变分析显示存在CALR基因突变。此后,给予羟基脲和阿那格雷未出现不良事件和并发症,血细胞计数得到良好控制。此外,还证实SF3B1基因突变在MDS-RS中呈高度阳性。日本尚无关于CALR突变型MDS/MPN的报道,该病在海外也较为罕见。