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

Authors :
Wu XL
Zhao XQ
Zhang BX
Xuan F
Guo HM
Ma FT
Source :
Brazilian journal of medical and biological research = Revista brasileira de pesquisas medicas e biologicas [Braz J Med Biol Res] 2017 Mar 23; Vol. 50 (4), pp. e5727. Date of Electronic Publication: 2017 Mar 23.
Publication Year :
2017

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.

Details

Language :
English
ISSN :
1414-431X
Volume :
50
Issue :
4
Database :
MEDLINE
Journal :
Brazilian journal of medical and biological research = Revista brasileira de pesquisas medicas e biologicas
Publication Type :
Report
Accession number :
28355352
Full Text :
https://doi.org/10.1590/1414-431X20165727