A Case of Macrophage Activation Syndrome Developing in a Patient With Chronic Granulomatous Disease-associated Colitis

被引：21

作者：

Akagi, Kazuko ^{[1
]}

Kawai, Toshinao ^{[2
]}

Watanabe, Nobuyuki ^{[2
]}

Yokoyama, Midori ^{[2
]}

Arai, Katsuhiro ^{[3
]}

Harayama, Shizuko ^{[2
,4
]}

Oana, Shinji ^{[1
]}

Onodera, Masafumi ^{[2
]}

机构：

[1] Natl Ctr Child Hlth & Dev, Dept Interdisciplinary Med, Tokyo 1578535, Japan

[2] Natl Ctr Child Hlth & Dev, Dept Human Genet, Tokyo 1578535, Japan

[3] Natl Ctr Child Hlth & Dev, Div Gastroenterol, Tokyo 1578535, Japan

[4] Jikei Univ, Sch Med, Dept Pediat, Tokyo, Japan

来源：

JOURNAL OF PEDIATRIC HEMATOLOGY ONCOLOGY | 2014年 / 36卷 / 03期

关键词：

chronic granulomatous disease; macrophage activation syndrome; CGD-associated colitis; inflammation; immunodeficiency; INFLAMMATORY-BOWEL-DISEASE; HEMOPHAGOCYTIC SYNDROME; THERAPY; CGD;

D O I：

10.1097/MPH.0b013e31828e5dae

中图分类号：

R73 [肿瘤学];

学科分类号：

100214 [肿瘤学];

摘要：

Although macrophage activation syndrome (MAS) develops in some patients with chronic granulomatous disease (CGD), all of the reported cases have been associated with pathogenic microbial infections. We report a 2-year-old boy with CGD-associated colitis who suffered from MAS without any clinical signs of a microbial infection. He was treated with 1 course of methylprednisolone pulse therapy and the clinical symptoms improved; however, the colitis was difficult to control even with immunosuppressive drugs, and he eventually required hematopoietic stem cell transplantation 1 year after the onset of MAS. It is likely that MAS develops in patients with CGD colitis independent of microbial infections.

引用

页码：E169 / E172

页数：4

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