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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.

Authors :
Esmaeilzadeh H
Bemanian MH
Nabavi M
Arshi S
Fallahpour M
Fuchs I
zur Stadt U
Warnatz K
Ammann S
Ehl S
Lehmberg K
Rezaei N
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.

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