Nocardiosis Associated with Primary Immunodeficiencies (Nocar-DIP): an International Retrospective Study and Literature
- PDF / 531,996 Bytes
- 12 Pages / 595.276 x 790.866 pts Page_size
- 1 Downloads / 172 Views
ORIGINAL ARTICLE
Nocardiosis Associated with Primary Immunodeficiencies (Nocar-DIP) : an International Retrospective Study and Literature Review Emmanuel Lafont 1 & Beatriz E. Marciano 2 & Nizar Mahlaoui 3,4 & Bénédicte Neven 3,4,5 & Jacinta Bustamante 5,6,7,8 & Veronica Rodriguez-Nava 9 & Amit Rawat 10 & Miren Josebe Unzaga 11 & Alain Fischer 3,4,5,12 & Stéphane Blanche 3,4,5 & Olivier Lortholary 1,4 & Steven M. Holland 2 & David Lebeaux 1,13,14 Received: 9 June 2020 / Accepted: 7 September 2020 # Springer Science+Business Media, LLC, part of Springer Nature 2020
Abstract Purpose Nocardiosis is a life-threatening infectious disease. We aimed at describing nocardiosis in patients with primary immunodeficiency diseases (PID). Methods This international retrospective cohort included patients with PID and nocardiosis diagnosed and/or published from Jan 1, 2000, to Dec 31, 2016. To identify nocardiosis cases, we analyzed PID databases from the French National Reference Center for PID (Paris, France) and the National Institute of Health (NIH, United States of America) and we performed a literature review on PubMed. Results Forty-nine cases of nocardiosis associated with PID were included: median age at diagnosis of nocardiosis was 19 (0–56) years and most cases were observed among chronic granulomatous disease (CGD) patients (87.8%). Median time from symptoms to diagnosis of Nocardia infection was 20 (2–257) days. Most frequent clinical nocardiosis presentation was pneumonia (86.7%). Twelve-month mortality rate was 4.2%, and 11.9% of patients experienced a possible recurrence of infection. Nocardiosis more frequently led to the diagnosis of PID among nonCGD patients than in CGD patients. Non-CGD patients experienced more cerebral nocardiosis and more disseminated infections, but mortality and recurrence rates were similar. Highest incidences of nocardiosis among PID cohorts were observed among CGD patients (0.0057 and 0.0044 cases/patient-year in the USA and in France, respectively), followed by IL-12p40 deficiency. Conclusions Among 49 cases of nocardiosis associated with PID, most patients had CGD and lung involvement. Both mortality and recurrence rates were low. Keywords Nocardiosis . Nocardia farcinica . primary immunodeficiency diseases . chronic granulomatous disease
Abbreviations ABT AST ADA BAL
Antibiotic therapy Antibiotic susceptibility testing Adenosine deaminase Bronchoalveolar lavage
Olivier Lortholary and Steven M. Holland contributed equally to this work. Electronic supplementary material The online version of this article (https://doi.org/10.1007/s10875-020-00866-8) contains supplementary material, which is available to authorized users. * David Lebeaux [email protected]
CGD CEREDIH CNS COPD CS CVID HIV HCST ICL IL12RB1 IL-12p40 deficiency
Extended author information available on the last page of the article
Chronic Granulomatous Disease Centre de Référence des Déficits Immunitaires Héréditaires Central nervous system Chronic obstructive pulmonary disease Corticosteroids Common variable immu
Data Loading...
