Cetingül N, Yener E, Oztop S, Nisli G, Soydan S
Ege University, Faculty of Medicine, Department of Pediatrics, Izmir, Turkey.
Acta Paediatr Jpn. 1994 Dec;36(6):697-700. doi: 10.1111/j.1442-200x.1994.tb03273.x.
Myelofibrosis with myeloid metaplasia, or agnogenic myeloid metaplasia (AMM) is a chronic myeloproliferative disorder characterized by fibrosis of the bone marrow accompanied by aniso- and poikilocytosis, leukoerythroblastosis and hepatosplenomegaly with extramedullary hematopoiesis. Agnogenic myeloid metaplasia is very rare in children. In this report, two cases of AMM in whom the onset of the illness were at 3 and 12 months of age, are presented. Both had severe anemia, hepatosplenomegaly and bone marrow fibrosis. Lymph node biopsy of the first patient and liver biopsy of the second revealed extramedullary hematopoiesis. They were treated with an intravenous high dose of methylprednisolone (daily 30 mg/kg for 3 days, 20 mg/kg for 4 days, 10 mg/kg for 1 week, 5 mg/kg for 1 week). A complete improvement of hematological and clinical findings was observed.
骨髓纤维化伴髓样化生,或原发性髓样化生(AMM)是一种慢性骨髓增殖性疾病,其特征为骨髓纤维化,伴有红细胞大小不均和异形红细胞症、幼粒 - 幼红细胞血象以及肝脾肿大并伴有髓外造血。原发性髓样化生在儿童中非常罕见。在本报告中,呈现了两例发病年龄分别为3个月和12个月的AMM病例。两例均有严重贫血、肝脾肿大和骨髓纤维化。第一例患者的淋巴结活检以及第二例患者的肝脏活检均显示有髓外造血。他们接受了静脉注射大剂量甲泼尼龙治疗(每日30mg/kg,共3天;20mg/kg,共4天;10mg/kg,共1周;5mg/kg,共1周)。观察到血液学和临床症状完全改善。