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一种新型的切-东综合征移码突变及其加速期治疗

A novel frameshift mutation of Chediak-Higashi syndrome and treatment in the accelerated phase.

作者信息

Wu X L, Zhao X Q, Zhang B X, Xuan F, Guo H M, Ma F T

机构信息

Department of Pediatric Hematology-Oncology, The Second Hospital of Hebei Medical University, Shijiazhuang, China.

出版信息

Braz J Med Biol Res. 2017 Mar 23;50(4):e5727. doi: 10.1590/1414-431X20165727.

Abstract

Chediak-Higashi syndrome (CHS) is a rare autosomal recessive immunodeficiency disease characterized by frequent infections, hypopigmentation, progressive neurologic deterioration and hemophagocytic lymphohistiocytosis (HLH), known as the accelerated phase. There is little experience in the accelerated phase of CHS treatment worldwide. Here, we present a case of a 9-month-old boy with continuous high fever, hypopigmentation of the skin, enlarged lymph nodes, hepatosplenomegaly and lung infection. He was diagnosed with CHS by gene sequencing, and had entered the accelerated phase. After 8 weeks of therapy, the boy had remission and was prepared for allogenic stem cell transplantation.

摘要

切迪阿克-希加什综合征(CHS)是一种罕见的常染色体隐性免疫缺陷病,其特征为频繁感染、色素减退、进行性神经功能恶化以及噬血细胞性淋巴组织细胞增生症(HLH),即加速期。目前全球范围内在CHS加速期治疗方面经验甚少。在此,我们报告一例9个月大男孩的病例,该男孩持续高热、皮肤色素减退、淋巴结肿大、肝脾肿大并伴有肺部感染。通过基因测序诊断其患有CHS,且已进入加速期。经过8周治疗后,该男孩病情缓解,并准备接受异基因干细胞移植。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c153/5423745/4e85e6f07430/1414-431X-bjmbr-1414-431X20165727-gf01.jpg

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