Mycobacterial disease in patients with chronic granulomatous disease: A retrospective analysis of 71 cases - PubMed (original) (raw)

. 2016 Jul;138(1):241-248.e3.

doi: 10.1016/j.jaci.2015.11.041. Epub 2016 Feb 28.

Saul Oswaldo Lugo-Reyes 2, Lizbeth Blancas Galicia 2, Jianxin He 3, Güzide Aksu 4, Edgar Borges de Oliveira Jr 5, Caroline Deswarte 6, Marjorie Hubeau 6, Neslihan Karaca 4, Maylis de Suremain 6, Antoine Guérin 6, Laila Ait Baba 7, Carolina Prando 8, Gloria G Guerrero 6, Melike Emiroglu 9, Fatma Nur Öz 10, Marco Antonio Yamazaki Nakashimada 11, Edith Gonzalez Serrano 11, Sara Espinosa 11, Isil Barlan 12, Nestor Pérez [ 13](#full-view-affiliation-13 "Immunology Unit, Children's Hospital "Superiora Sor María Ludovica", La Plata, Argentina."), Lorena Regairaz [ 13](#full-view-affiliation-13 "Immunology Unit, Children's Hospital "Superiora Sor María Ludovica", La Plata, Argentina."), Héctor Eduardo Guidos Morales [ 14](#full-view-affiliation-14 "Allergy and Immunology Unit, National Children's Hospital "Benjamín Bloom", San Salvador, El Salvador."), Liliana Bezrodnik [ 15](#full-view-affiliation-15 "Immunology Unit, Children's Hospital "Ricardo Gutierrez", Buenos Aires, Argentina."), Daniela Di Giovanni [ 15](#full-view-affiliation-15 "Immunology Unit, Children's Hospital "Ricardo Gutierrez", Buenos Aires, Argentina."), Ghassan Dbaibo 16, Fatima Ailal 17, Miguel Galicchio [ 18](#full-view-affiliation-18 "Children's Hospital "Victor J. Vilela", Rosario, Argentina."), Matias Oleastro [ 19](#full-view-affiliation-19 "Department of Immunology, "Juan Pedro Garrahan" National Hospital of Pediatrics, Buenos Aires, Argentina."), Jalel Chemli 20, Silvia Danielian [ 19](#full-view-affiliation-19 "Department of Immunology, "Juan Pedro Garrahan" National Hospital of Pediatrics, Buenos Aires, Argentina."), Laura Perez [ 19](#full-view-affiliation-19 "Department of Immunology, "Juan Pedro Garrahan" National Hospital of Pediatrics, Buenos Aires, Argentina."), Maria Claudia Ortega [ 21](#full-view-affiliation-21 "Children's Hospital "San Jose" and Medical School, Bogota, Columbia."), Susana Soto Lavin 22, Joseph Hertecant 23, Ozden Anal 24, Nadia Kechout 25, Eman Al-Idrissi 26, Gehad ElGhazali 26, Anastasia Bondarenko 27, Liudmyla Chernyshova 27, Peter Ciznar 28, Rose-Marie Herbigneaux 29, Aminata Diabate 30, Stéphanie Ndaga 30, Barik Konte 30, Ambre Czarna 30, Mélanie Migaud 6, Sigifredo Pedraza-Sánchez 31, Mussaret Bano Zaidi 32, Guillaume Vogt 6, Stéphane Blanche 33, Imen Benmustapha 34, Davood Mansouri 35, Laurent Abel 36, Stéphanie Boisson-Dupuis 36, Nizar Mahlaoui 37, Ahmed Aziz Bousfiha 17, Capucine Picard 38, Ridha Barbouche 34, Saleh Al-Muhsen 39, Francisco J Espinosa-Rosales 11, Necil Kütükçüler 4, Antonio Condino-Neto 40, Jean-Laurent Casanova 41, Jacinta Bustamante 42

Affiliations

Mycobacterial disease in patients with chronic granulomatous disease: A retrospective analysis of 71 cases

Francesca Conti et al. J Allergy Clin Immunol. 2016 Jul.

Abstract

Background: Chronic granulomatous disease (CGD) is a rare primary immunodeficiency caused by inborn errors of the phagocyte nicotinamide adenine dinucleotide phosphate oxidase complex. From the first year of life onward, most affected patients display multiple, severe, and recurrent infections caused by bacteria and fungi. Mycobacterial infections have also been reported in some patients.

Objective: Our objective was to assess the effect of mycobacterial disease in patients with CGD.

Methods: We analyzed retrospectively the clinical features of mycobacterial disease in 71 patients with CGD. Tuberculosis and BCG disease were diagnosed on the basis of microbiological, pathological, and/or clinical criteria.

Results: Thirty-one (44%) patients had tuberculosis, and 53 (75%) presented with adverse effects of BCG vaccination; 13 (18%) had both tuberculosis and BCG infections. None of these patients displayed clinical disease caused by environmental mycobacteria, Mycobacterium leprae, or Mycobacterium ulcerans. Most patients (76%) also had other pyogenic and fungal infections, but 24% presented solely with mycobacterial disease. Most patients presented a single localized episode of mycobacterial disease (37%), but recurrence (18%), disseminated disease (27%), and even death (18%) were also observed. One common feature in these patients was an early age at presentation for BCG disease. Mycobacterial disease was the first clinical manifestation of CGD in 60% of these patients.

Conclusion: Mycobacterial disease is relatively common in patients with CGD living in countries in which tuberculosis is endemic, BCG vaccine is mandatory, or both. Adverse reactions to BCG and severe forms of tuberculosis should lead to a suspicion of CGD. BCG vaccine is contraindicated in patients with CGD.

Keywords: BCG; Mycobacteria; chronic granulomatous disease; primary immunodeficiency; tuberculosis.

Copyright © 2016 American Academy of Allergy, Asthma & Immunology. Published by Elsevier Inc. All rights reserved.

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