Literature DB >> 27888588

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.

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

Mesh:

Substances:

Year:  2016        PMID: 27888588

Source DB:  PubMed          Journal:  Acta Med Iran        ISSN: 0044-6025


  4 in total

1.  LncRNA SNHG16 is Downregulated in Pneumonia and Downregulates miR-210 to Promote LPS-Induced Lung Cell Apoptosis.

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Journal:  Mol Biotechnol       Date:  2022-08-22       Impact factor: 2.860

Review 2.  Bacille Calmette-Guerin Complications in Newly Described Primary Immunodeficiency Diseases: 2010-2017.

Authors:  Cristiane de Jesus Nunes-Santos; Sergio D Rosenzweig
Journal:  Front Immunol       Date:  2018-06-22       Impact factor: 7.561

3.  Multifocal gastric adenocarcinoma in a patient with LRBA deficiency.

Authors:  Nina Bratanič; Jernej Kovač; Katka Pohar; Katarina Trebušak Podkrajšek; Alojz Ihan; Tadej Battelino; Magdalena Avbelj Stefanija
Journal:  Orphanet J Rare Dis       Date:  2017-07-18       Impact factor: 4.123

4.  Clinical, Immunological, and Functional Characterization of Six Patients with Very High IgM Levels.

Authors:  Vera Gallo; Emilia Cirillo; Rosaria Prencipe; Alessio Lepore; Luigi Del Vecchio; Giulia Scalia; Vincenzo Martinelli; Gigliola Di Matteo; Carol Saunders; Anne Durandy; Viviana Moschese; Antonio Leonardi; Giuliana Giardino; Claudio Pignata
Journal:  J Clin Med       Date:  2020-03-17       Impact factor: 4.241

  4 in total

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