| Literature DB >> 27888588 |
Sima Shokri1, Mohammad Nabavi1, Tatjana Hirschmugl2, Asghar Aghamohammadi3, Saba Arshi1, Mohamad Hassan Bemanian1, Morteza Fallahpour1, Rasool Molatefi1, Mahsa Rekabi1, Narges Eslami1, Javad Ahmadian1, Kian Darabi1, Gholam Reza Sedighi1, Maryam Monajemzadeh4, Mohammadreza Modaresi5, Nima Parvaneh3, Kaan Boztug6, Nima Rezaei7.
Abstract
LPS-Responsive Beige-like Anchor (LRBA) deficiency is a disease which has recently been described in a group of patients with common variable immunodeficiency (CVID) in association with autoimmunity and/or inflammatory bowel disease (IBD)-like phenotype. We here describe a 10-year-old boy who experienced recurrent infections, mainly in the respiratory system, associated with thrombocytopenia and anemia. Immunological workup showed low numbers of B cells and low IgG, but normal IgM levels. In spite of therapeutic doses of antibiotics, antivirals, and antifungal agents, in addition to immunoglobulin replacement therapy, he developed disseminated involvement of both lungs with peripheral nodules; transbronchial lung biopsy revealed possible bronchiolitis obliterans organizing pneumonia (BOOP). Combined homozygosity mapping and exome sequencing identified a homozygous LRBA mutation in this patient (p.Asp248Glufs*2). Such clinical and immunological findings have not been described to date and illustrate the broad and variable clinical phenotype of human LRBA deficiency.Entities:
Keywords: Bronchiolitis obliterans organizing pneumonia; Hyper IgM; Hypogammaglobulinemia; LRBA deficiency
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Year: 2016 PMID: 27888588
Source DB: PubMed Journal: Acta Med Iran ISSN: 0044-6025