Tokgoz Huseyin, Caliskan Umran, Yüksekkaya Hasan Ali, Kucukkaya Reyhan
a Department of Pediatric Haematology, Meram Medical Faculty , Necmettin Erbakan University , Konya , Turkey .
b Department of Pediatric Gastroenterology, Meram Medical Faculty , Necmettin Erbakan University , Konya , Turkey , and.
Platelets. 2015;26(8):805-8. doi: 10.3109/09537104.2015.1041900. Epub 2015 May 13.
Essential thrombocythemia (ET) is an extremely rare childhood disorder characterised by clonal expansion of megakaryocytic lineage in bone marrow, leading to a persistent increase in the number of circulating thrombocytes and thus increased risk for thrombotic and haemorrhagic events. The molecular mechanisms of ET are not fully understood. Most children with ET have the JAK2 V617F somatic mutation; however, another mutation, involving a W to L or K substitution at Mpl codon 515, was reported in a small proportion of adult ET patients that is extremely rare in children. Herein, we describe a Mpl W515K somatic mutation in a paediatric case of ET who presented with Budd-Chiari syndrome. No paediatric patient harbouring a Mpl W515K mutation has been previously reported.
原发性血小板增多症(ET)是一种极其罕见的儿童疾病,其特征是骨髓中巨核细胞系的克隆性扩增,导致循环血小板数量持续增加,从而增加血栓形成和出血事件的风险。ET的分子机制尚未完全明确。大多数ET患儿存在JAK2 V617F体细胞突变;然而,在一小部分成年ET患者中报道了另一种突变,即Mpl密码子515处的W到L或K替代,这种情况在儿童中极为罕见。在此,我们描述了1例患有布加综合征的ET患儿中的Mpl W515K体细胞突变。此前尚无携带Mpl W515K突变的儿科患者的报道。