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Медицинская библиотека · Acta medica Iranica · 2016

LPS-Responsive Beige-Like Anchor Gene Mutation Associated With Possible Bronchiolitis Obliterans Organizing Pneumonia Associated With Hypogammaglobulinemia and Normal IgM Phenotype and Low Number of B Cells.

Shokri Sima, Nabavi Mohammad, Hirschmugl Tatjana, Aghamohammadi Asghar, Arshi Saba, Bemanian Mohamad Hassan, Fallahpour Morteza, Molatefi Rasool, Rekabi Mahsa, Eslami Narges, Ahmadian Javad, Darabi Kian, Sedighi Gholam Reza, Monajemzadeh Maryam, Modaresi Mohammadreza, Parvaneh Nima, Boztug Kaan, Rezaei Nima

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Аннотация

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.

Для цитирования: Shokri Sima, Nabavi Mohammad, Hirschmugl Tatjana, Aghamohammadi Asghar, Arshi Saba, Bemanian Mohamad Hassan, Fallahpour Morteza, Molatefi Rasool, Rekabi Mahsa, Eslami Narges, Ahmadian Javad, Darabi Kian, Sedighi Gholam Reza, Monajemzadeh Maryam, Modaresi Mohammadreza, Parvaneh Nima, Boztug Kaan, Rezaei Nima. LPS-Responsive Beige-Like Anchor Gene Mutation Associated With Possible Bronchiolitis Obliterans Organizing Pneumonia Associated With Hypogammaglobulinemia and Normal IgM Phenotype and Low Number of B Cells.. Acta medica Iranica. 2016.

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