收稿日期: 2024-02-24
修回日期: 2024-05-10
网络出版日期: 2024-10-25
A case of secondary myelofibrosis in visceral leishmaniasis
Received date: 2024-02-24
Revised date: 2024-05-10
Online published: 2024-10-25
患者,男,15岁,学生,河南林州人。2021年3月3日因“间断发热半月余”至当地市级医院就诊,血常规示白细胞2.60 × 109/L、血小板65 × 109/L、血红蛋白113 g/L,诊断为感染性发热并白细胞血小板减少、脾大,予抗感染治疗后好转出院。后患者病情时有反复,于2022年4月19日以“反复发热、乏力,发现全血细胞少1年余”入住省级医院血液科,入院血常规示白细胞1.79 × 109/L,血小板54 × 109/L,血红蛋白73 g/L;彩超示双侧颈部、腋窝肿大淋巴结,肝大脾大并脾静脉增宽;骨髓涂片示增生活跃,粒系减低,红系增生明显活跃,成熟红细胞轻度缗钱状排列,巨核细胞0个/片,血小板少见;骨髓活检示骨髓广泛纤维化,红系比例增高,巨核细胞无明显减少,T细胞较易见,散在个别大细胞;正电子发射计算机断层显像(PET-CT)示脾大代谢弥漫性较活跃,肝大代谢未见异常。综合考虑脾淋巴瘤转外科行脾切除 + 腹腔淋巴结清扫手术,脾脏病理示脾脏组织内见中等量组织细胞增生,胞质内见病原体感染,基因测序提示杜氏利什曼原虫,伴髓外造血样改变,确诊内脏利什曼病。患者有犬只接触史,居住城市有内脏利什曼病病例报告。2022年6月27日转入安阳市第五人民医院开始葡萄糖酸锑钠治疗。入院查体:消瘦,营养不良,腹部饱满,肝脏触诊右锁骨中线肋下10 cm,质中等,无触痛。血常规示白细胞11.17 × 109/L,血红蛋白104 g/L,血小板245 × 109/L;rK39黑热病抗体快速检测阳性;腹部CT示肝脏明显增大。予葡萄糖酸锑钠静脉输注1疗程(0.6 g/d,9 d),患者体温恢复正常,一般情况好转,复查骨髓涂片,可见利杜体,继续输注葡萄糖酸锑钠1疗程,复查骨髓未见利杜体,痊愈出院。约10个月后随访,患者无不适,骨髓涂片细胞学检查大致正常,未见利杜体,骨髓活检纤维化消失,腹部CT肝脏有所缩小。
杨梅 , 向保云 , 魏涛 , 池永娥 , 李娟 . 内脏利什曼病继发骨髓纤维化1例[J]. 中国寄生虫学与寄生虫病杂志, 2024 , 42(5) : 684 -687 . DOI: 10.12140/j.issn.1000-7423.2024.05.021
The case was a 15-year-old male patient from Linzhou, Henan province and he was a student. Due to “intermittent fever for more than half a month”, he went to a local municipal hospital on March 3, 2021. The blood routine test showed leukocyte was 2.60 × 109/L, platelet was 65 × 109/L and hemoglobin was 113 g/L. He was diagnosed with infectious fever with leukothrombopenia and splenomegaly. After anti-infection treatment, the symptoms were improved and he was discharged home. But the condition sometimes recurred and he was admitted to the hematology department of a provincial hospital because of “repeated fever with fatigue and pancytopenia for more than 1 year” on April 19, 2022. The blood routine test showed white blood cell was 1.79 × 109/L, hemoglobin was 73 g/L and platelet was 54 × 109/L. Ultrasound showed enlarged lymphnodes in the neck and axilla, large liver and spleen with widened splenic veins. Bone marrow smear showed hyperplasia was active, granulocyte series reduced, erythrocyte series was significantly active while mature erythrocytes clustered and arranged in row, and no megakaryocyte was found in this smear. Bone marrow biopsy showed extensive fibrosis, increasing proportion of erythrocyte series, many megakaryocyte cells and also showed T cells were easy to be seen with scattered individual large cells. Positron emission tomography-computed tomography (PET-CT) showed diffuse active in splenic metabolism and no abnormalities in hepatic metabolism. Then the patient was suspected to have a splenic lymphoma and was transferred to surgery for splenectomy + abdominal lymph node dissection. Pathology of the spleen showed moderate histiocytic hyperplasia and pathogen in the cytoplasm of the histocyte which was verified Leishmania donovani by gene sequencing. Pathology of the spleen also showed extramedullary hematopoietic. The patient was diagnosed with visceral leishmaniasis. The patient had a history of contact with dogs and the county where he lived had visceral leishmaniasis case report. He was admitted to the Fifth People’s Hospital of Anyang in June 27, 2022 and was began to be treated with sodium stibogluconate injection from that time. The admission examination showed emaciation, malnutrition and hepatomegaly that liver could be palpated 10cm below costal margin on right midclavicular line, moderate quality, no tenderness. Blood routine test showed that the white blood cell was 11.17 × 109/L, hemogloubin was 104 g/L and platelet was 245 × 109/L. The result of rK39 kala-azar detect rapid test was positive. Abdominal CT showed a significantly enlarged liver. After treated with sodium stibogluconate injection for 1 course (0.6 g/d, 9 d), The temperature returned to normal and the general condition improved, but Leishmania amastigote was visible in his bone marrow smear. So another course sodium stibogluconate injection (0.6 g/d, 9 d) was given to him. After the second course’s treatment, the bone marrow smear was reexamined and no Leishmania amastigote could be found. The paitent was recovered and discharged home. He felt well and the abdominal CT showed liver was somewhat reduced and bone marrow trephine biopsy performed had returned to normal and bone marrow smear was not found Leishmania amastigote about 10 months after initiation of the therapy.
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