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Novel Patient with Late-Onset Familial Hemophagocytic Lymphohistiocytosis with STXBP2 Mutations Presenting with Autoimmune Hepatitis, Neurological Manifestations and Infections Associated with Hypogammaglobulinemia.
- Source :
-
Journal of clinical immunology [J Clin Immunol] 2015 Jan; Vol. 35 (1), pp. 22-5. Date of Electronic Publication: 2014 Dec 10. - Publication Year :
- 2015
-
Abstract
- Familial hemophagocytic lymphohistiocytosis (FHL) is a genetically heterogeneous hyperinflammatory syndrome, caused by an uncontrolled and ineffective proliferation and activation of T-lymphocytes, NK-cells, and macrophages that infiltrate multiple organs. Herein, a patient is presented who suffered from hepatitis and atypical brain lesions. Genetic studies revealed a homozygous mutation in the STXP2 gene; and thus, the diagnosis of FHL5 was confirmed.
- Subjects :
- Agammaglobulinemia genetics
Agammaglobulinemia immunology
Age of Onset
Brain pathology
Child
Fatal Outcome
Hepatitis, Autoimmune genetics
Hepatitis, Autoimmune immunology
Homozygote
Humans
Killer Cells, Natural immunology
Lymphohistiocytosis, Hemophagocytic diagnosis
Lymphohistiocytosis, Hemophagocytic immunology
Magnetic Resonance Imaging
Male
Mutation
T-Lymphocytes immunology
Lymphohistiocytosis, Hemophagocytic genetics
Munc18 Proteins genetics
Subjects
Details
- Language :
- English
- ISSN :
- 1573-2592
- Volume :
- 35
- Issue :
- 1
- Database :
- MEDLINE
- Journal :
- Journal of clinical immunology
- Publication Type :
- Academic Journal
- Accession number :
- 25491289
- Full Text :
- https://doi.org/10.1007/s10875-014-0119-z