Olivier Lortholary1,2, Steven M Holland3, Emmanuel Lafont1, Beatriz E Marciano3, Nizar Mahlaoui4,2, Bénédicte Neven4,2,5, Jacinta Bustamante5,6,7,8, Veronica Rodriguez-Nava9, Amit Rawat10, Miren Josebe Unzaga11, Alain Fischer4,2,5,12, Stéphane Blanche4,2,5, David Lebeaux13,14,15. 1. Department of Infectious Diseases and Tropical Medicine, Centre d'Infectiologie Necker -Pasteur, Assistance Publique-Hôpitaux de Paris (AP-HP), Necker-Enfants Malades University Hospital, Université de Paris, Paris, France. 2. French National Reference Center for Primary Immune Deficiencies (CEREDIH), Assistance Publique-Hôpitaux de Paris, (AP-HP), Necker Enfants Malades University Hospital, Paris, France. 3. Laboratory of Clinical Immunology and Microbiology, National Institute of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, MD, USA. 4. Pediatric Immuno-Haematology and Rheumatology Unit, Assistance Publique-Hôpitaux de Paris (AP-HP), Necker Enfants Malades University Hospital, Paris, France. 5. Sorbonne Paris Cite, Imagine Institute, INSERM UMR 1163, Université de Paris, Paris, France. 6. Laboratory of Human Genetics of Infectious Diseases, INSERM U1163, Necker Enfants Malades University Hospital, Paris, France. 7. Center for the Study of Primary Immunodeficiencies (CEDI), Assistance Publique - Hôpitaux de Paris (AP-HP), Necker Enfants Malades University Hospital, Paris, France. 8. St. Giles Laboratory of Human Genetics of Infectious Diseases, Rockefeller Branch, The Rockefeller University, New York, NY, USA. 9. Research group on Bacterial Opportunistic Pathogens and Environment UMR5557 Écologie Microbienne, French Observatory of Nocardiosis, CNRS, VetAgro Sup, Université de Lyon 1, Lyon, France. 10. Pediatric Allergy and Immunology Unit, Advanced Pediatrics Centre, Department of Pediatrics, Postgraduate Institute of Medical Education and Research, Chandigarh, 160012, India. 11. Department of Microbiology, Hospital de Basurto, 48013, Bilbao, Basque Country, Spain. 12. Collège de France, Paris, France. 13. Department of Infectious Diseases and Tropical Medicine, Centre d'Infectiologie Necker -Pasteur, Assistance Publique-Hôpitaux de Paris (AP-HP), Necker-Enfants Malades University Hospital, Université de Paris, Paris, France. david.lebeaux@aphp.fr. 14. Université de Paris, 75006, Paris, France. david.lebeaux@aphp.fr. 15. Service de Microbiologie, Unité Mobile d'Infectiologie, Assistance Publique - Hôpitaux de Paris (AP-HP), Hôpital Européen Georges Pompidou, 20 rue Leblanc, 75015, Paris, France. david.lebeaux@aphp.fr.
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.
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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