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12.09.2020 | Original Article

Nocardiosis Associated with Primary Immunodeficiencies (Nocar-DIP): an International Retrospective Study and Literature Review

verfasst von: Emmanuel Lafont, Beatriz E. Marciano, Nizar Mahlaoui, Bénédicte Neven, Jacinta Bustamante, Veronica Rodriguez-Nava, Amit Rawat, Miren Josebe Unzaga, Alain Fischer, Stéphane Blanche, Olivier Lortholary, Steven M. Holland, David Lebeaux

Erschienen in: Journal of Clinical Immunology | Ausgabe 8/2020

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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 non-CGD 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.

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Titel: Nocardiosis Associated with Primary Immunodeficiencies (Nocar-DIP): an International Retrospective Study and Literature Review
verfasst von: Emmanuel Lafont
Beatriz E. Marciano
Nizar Mahlaoui
Bénédicte Neven
Jacinta Bustamante
Veronica Rodriguez-Nava
Amit Rawat
Miren Josebe Unzaga
Alain Fischer
Stéphane Blanche
Olivier Lortholary
Steven M. Holland
David Lebeaux
Publikationsdatum: 12.09.2020
Verlag: Springer US
Erschienen in: Journal of Clinical Immunology / Ausgabe 8/2020
Print ISSN: 0271-9142
Elektronische ISSN: 1573-2592
DOI: https://doi.org/10.1007/s10875-020-00866-8

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Nocardiosis Associated with Primary Immunodeficiencies (Nocar-DIP): an International Retrospective Study and Literature Review