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A novel frameshift mutation of Chediak-Higashi syndrome and treatment in the accelerated phase.
- 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.
- Subjects :
- Chediak-Higashi Syndrome pathology
Delayed Diagnosis
Hair pathology
Humans
Hypopigmentation genetics
Hypopigmentation pathology
Infant
Lymphohistiocytosis, Hemophagocytic genetics
Male
Pneumonia diagnostic imaging
Pneumonia genetics
Skin pathology
Treatment Outcome
Chediak-Higashi Syndrome drug therapy
Chediak-Higashi Syndrome genetics
Frameshift Mutation
Subjects
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