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Infections in autoimmune pulmonary alveolar proteinosis: a large retrospective cohort
Axelle Mabo1, Raphael Borie2, Lidwine Wemeau-Stervinou3
1Service de Pneumologie, Centre de Compétence pour les Maladies Pulmonaires Rares, Hôpital Pontchaillou, CHU Rennes, Rennes, France.
Background:
Autoimmune pulmonary alveolar proteinosis (aPAP) is a rare disease, predisposing to an increased risk of infection. A complete picture of these infections is lacking.
Research Question:
Describe the characteristics and clinical outcomes of patients diagnosed with aPAP, and to identify risk factors associated with opportunistic infections.
Methods:
We conducted a retrospective cohort including all patients diagnosed with aPAP between 2008 and 2018 in France and Belgium. Data were collected using a standardised questionnaire including demographics, comorbidities, imaging features, outcomes and microbiological data.
Results:
We included 104 patients, 2/3 were men and median age at diagnosis was 45 years. With a median follow-up of 3.4 years (IQR 1.7-6.6 years), 60 patients (58%), developed at least one infection, including 23 (22%) with opportunistic infections. Nocardia spp was the main pathogen identified (n=10). Thirty-five (34%) patients were hospitalised due to infection. In univariate analysis, male gender was associated with opportunistic infections (p=0.04, OR=3.88; 95% CI (1.02 to 22.06)). Anti-granulocyte macrophage colony-stimulating factor antibody titre at diagnosis was significantly higher among patients who developed nocardiosis (1058 (316-1591) vs 580 (200-1190), p=0.01). Nine patients had died (9%), but only one death was related to infection.
Interpretation:
Patients with aPAP often presented with opportunistic infections, especially nocardiosis, which highlights the importance of systematic search for slow-growing bacteria in bronchoalveolar lavage or whole lung lavage.
Insights
Autoimmune pulmonary alveolar proteinosis (aPAP) patients frequently experience infections, particularly opportunistic ones like nocardiosis. Early detection of slow-growing bacteria is crucial for managing these infections in aPAP patients.
Area of Science:
- Pulmonary Medicine
- Infectious Diseases
- Rare Diseases
Background:
- Autoimmune pulmonary alveolar proteinosis (aPAP) is a rare condition.
- aPAP increases the risk of infections, but a comprehensive understanding is lacking.
Purpose of the Study:
- To characterize infections and clinical outcomes in aPAP patients.
- To identify risk factors for opportunistic infections in aPAP.
Main Methods:
- Retrospective cohort study of 104 aPAP patients (2008-2018) in France and Belgium.
- Data collected via standardized questionnaire: demographics, comorbidities, imaging, outcomes, microbiology.
Main Results:
- 58% of patients developed infections; 22% had opportunistic infections, mainly Nocardia (10 cases).
- Male gender was linked to opportunistic infections (OR=3.88).
- Higher anti-GM-CSF antibody titers correlated with nocardiosis.
Conclusions:
- aPAP patients are prone to opportunistic infections, especially Nocardia.
- Systematic screening for slow-growing bacteria in lavage fluid is recommended for aPAP patients.
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