Development of clinical paroxysmal nocturnal haemoglobinuria in children with aplastic anaemia

被引：12

作者：

Narita, Atsushi ^{[1
]}

Muramatsu, Hideki ^{[1
]}

Okuno, Yusuke ^{[1
]}

Sekiya, Yuko ^{[1
]}

Suzuki, Kyogo ^{[1
]}

Hamada, Motoharu ^{[1
]}

Kataoka, Shinsuke ^{[1
]}

Ichikawa, Daisuke ^{[1
]}

Taniguchi, Rieko ^{[1
]}

Murakami, Norihiro ^{[1
]}

Kojima, Daiei ^{[1
]}

Nishikawa, Eri ^{[1
]}

Kawashima, Nozomu ^{[1
]}

Nishio, Nobuhiro ^{[1
]}

Hama, Asahito ^{[1
]}

Takahashi, Yoshiyuki ^{[1
]}

Kojima, Seiji ^{[1
]}

机构：

[1] Nagoya Univ, Grad Sch Med, Dept Paediat, Nagoya, Aichi, Japan

来源：

BRITISH JOURNAL OF HAEMATOLOGY | 2017年 / 178卷 / 06期

关键词：

paroxysmal nocturnal haemoglobinuria; aplastic anaemia; PIGA; Children; BONE-MARROW FAILURE; GLYCOSYLPHOSPHATIDYLINOSITOL; EVOLUTION; GLOBULIN; MUTATION; DISEASE; CLONES; CELLS; GENE;

D O I：

10.1111/bjh.14790

中图分类号：

R5 [内科学];

学科分类号：

1002 ; 100201 ;

摘要：

The clinical significance of paroxysmal nocturnal haemoglobinuria (PNH) in children with aplastic anaemia (AA) remains unclear. We retrospectively studied 57 children with AA between 1992 and 2010. During the follow-up, five patients developed clinical PNH, in whom somatic PIGA mutations were detected by targeted sequencing. The 10-year probability of clinical PNH development was 10.2% (95% confidence interval, 3.6-20.7%). Furthermore, the detection of minor PNH clones by flow cytometry at AA diagnosis was a risk factor for the subsequent development of clinical PNH. These patients with PNH clones at AA diagnosis should undergo periodic monitoring for potential clinical PNH development.

引用

页码：954 / 958

页数：5

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