Nocardiosis Associated with Primary Immunodeficiencies (Nocar-DIP): an International Retrospective Study and Literature Review.,Journal of Clinical Immunology

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Nocardiosis Associated with Primary Immunodeficiencies (Nocar-DIP): an International Retrospective Study and Literature Review.
Journal of Clinical Immunology ( IF 7.2 ) Pub Date : 2020-09-12 , DOI: 10.1007/s10875-020-00866-8
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}

Affiliation

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.
Laboratory of Clinical Immunology and Microbiology, National Institute of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, MD, USA.
Pediatric Immuno-Haematology and Rheumatology Unit, Assistance Publique-Hôpitaux de Paris (AP-HP), Necker Enfants Malades University Hospital, Paris, France.
French National Reference Center for Primary Immune Deficiencies (CEREDIH), Assistance Publique-Hôpitaux de Paris, (AP-HP), Necker Enfants Malades University Hospital, Paris, France.
Sorbonne Paris Cite, Imagine Institute, INSERM UMR 1163, Université de Paris, Paris, France.
Laboratory of Human Genetics of Infectious Diseases, INSERM U1163, Necker Enfants Malades University Hospital, Paris, France.
Center for the Study of Primary Immunodeficiencies (CEDI), Assistance Publique - Hôpitaux de Paris (AP-HP), Necker Enfants Malades University Hospital, Paris, France.
St. Giles Laboratory of Human Genetics of Infectious Diseases, Rockefeller Branch, The Rockefeller University, New York, NY, USA.
Research group on Bacterial Opportunistic Pathogens and Environment UMR5557 Écologie Microbienne, French Observatory of Nocardiosis, CNRS, VetAgro Sup, Université de Lyon 1, Lyon, France.
Pediatric Allergy and Immunology Unit, Advanced Pediatrics Centre, Department of Pediatrics, Postgraduate Institute of Medical Education and Research, Chandigarh, 160012, India.
Department of Microbiology, Hospital de Basurto, 48013, Bilbao, Basque Country, Spain.
Collège de France, Paris, France.
Université de Paris, 75006, Paris, France.
Service de Microbiologie, Unité Mobile d'Infectiologie, Assistance Publique - Hôpitaux de Paris (AP-HP), Hôpital Européen Georges Pompidou, 20 rue Leblanc, 75015, Paris, France.

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.

中文翻译：

与原发性免疫缺陷相关的诺卡菌病 (Nocar-DIP)：一项国际回顾性研究和文献综述。

目的

诺卡菌病是一种危及生命的传染病。我们旨在描述原发性免疫缺陷病 (PID) 患者的诺卡菌病。

方法

该国际回顾性队列包括 2000 年 1 月 1 日至 2016 年 12 月 31 日诊断和/或发表的 PID 和诺卡菌病患者。为了识别诺卡菌病病例，我们分析了法国国家 PID 参考中心（法国巴黎）的 PID 数据库，美国国立卫生研究院（NIH，美国），我们对 PubMed 进行了文献综述。

结果

包括 49 例与 PID 相关的诺卡菌病：诺卡菌病诊断的中位年龄为 19 (0-56) 岁，大多数病例在慢性肉芽肿病 (CGD) 患者中观察到 (87.8%)。从症状到诊断诺卡菌的中位时间感染时间为 20 (2-257) 天。最常见的临床诺卡菌病表现是肺炎（86.7%）。12 个月死亡率为 4.2%，11.9% 的患者可能出现感染复发。与 CGD 患者相比，非 CGD 患者中诺卡菌病更常导致 PID 的诊断。非 CGD 患者经历更多的脑诺卡菌病和更多的播散性感染，但死亡率和复发率相似。在 PID 队列中，CGD 患者中诺卡菌病的发生率最高（美国和法国分别为 0.0057 和 0.0044 例/患者年），其次是 IL-12p40 缺乏症。